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A systematic review of injectable corticosteroid for osteoarthritis of the first metatarsophalangeal joint

by Ian Reilly BSc, MSc, MCPod, FCPodS, FFPM RCPS(Glasg); Gillian Bromley BSc(Hons), MCPod; George Flanagan BSc(Hons), MCPod, FCPodS

The Foot and Ankle Online Journal 13 (3): 12

Intra-articular steroid injection is a common treatment modality for relief of pain and inflammation associated with degenerative joint disease. Use of injectable steroid preparations is widely accepted as safe and effective for the treatment of osteoarthritis of the 1st metatarsophalangeal joint. Despite the frequency of use, literature specific to pathology of the 1st metatarsophalangeal joint is sparse. The aim of this systematic review was to determine if good quality research exists to enable clinicians to adopt an evidenced based approach to corticosteroid injection of the 1st metatarsophalangeal joint. Despite the frequency of use, this review found no high quality studies that support the use of intra-articular corticosteroid injection of the 1st metatarsophalangeal joint in osteoarthritis.

Keywords: steroid injection, first metatarsophalangeal joint, osteoarthritis, hallux rigidus, systematic review

ISSN 1941-6806
doi: 10.3827/faoj.2020.1303.0012

1 – Department of Podiatric Surgery, Northamptonshire Healthcare Foundation NHS Trust, Danetre Hospital, Daventry, Northamptonshire, NN11 4DY. UK
* – Corresponding author: ianreilly@nhs.net


The use of injectable corticosteroid as part of a treatment strategy for painful joints is a common treatment modality. In degenerative disease the intended aim is to reduce the pain and inflammation associated with osteoarthritis (OA) as well as improve joint function [1]. The use of intra-articular (IA) corticosteroid injections (CSIs) for the treatment of OA is supported by guidelines provided by the United Kingdom (UK) National Institute for Health and Care Excellence (NICE) in patients who experience joint pain that is not adequately controlled by oral and/or topical options or where such treatment is contraindicated [2]. The basis for this guidance is largely derived from conclusions drawn from research into the efficacy of IA CSI’s at the knee and shoulder [3,4]: data from these studies has been extrapolated and applied to other synovial joints such as the first metatarsophalangeal joint (1st MPJ).

Osteoarthritis is the leading cause of disability in adults worldwide and results in significant morbidity [5]. Joints in the foot are often affected by this condition with the 1st MPJ being most commonly affected pedal joint [6]. Symptomatic 1st MPJ OA affects approximately 10% of the adult population and the prevalence increases with age – as do comorbidities amongst sufferers – with the result that reduced pharmacological treatment options available for pain relief in these patients [7]. Symptoms arising from OA are notoriously difficult to manage with oral analgesics alone: this ultimately results in a significant burden on primary care services [8]. This provides the niche for IA CSI, i.e. where other conservative treatment has failed, is contraindicated or where there is a desire or requirement to postpone the need for surgical intervention. Unmanaged foot pain is an independent risk factor for depression and falls in adults [9,10,11].

The authors are experienced injectors and are active in teaching CSI techniques to under- and postgraduate students. Anecdotally, we find that 80-90% of patients experience improvement following IA CSI for 1st MPJ OA but the extent and duration of that improvement varies. The variability in outcomes following CSI for 1st MPJ OA raises numerous questions: to what extent is pain reduced? Is joint function improved? Which patients are most likely to benefit from this treatment? What is the frequency with which corticosteroid should be administered and whether the use of ultrasound guided injections improves treatment outcomes [12,13,14]. Furthermore, there has been debate surrounding whether a steroid based solution, when combined with local analgesia, may even be chondrotoxic [15]. A Cochrane Review from 2010 [16] concerned with identifying optimal treatment modalities for 1st MPJ OA found low level evidence for physical therapy only. A systematic literature review was therefore undertaken (as part of a larger body of work being undertaken by the lead author) in order to identify randomized trials that had used IA CSI for OA of the 1st MPJ.

Methods

The research question is: is the use of corticosteroid injections for osteoarthritis of the first metatarsophalangeal joint in adults a safe and effective method of reducing pain and improving joint function?

In order to ensure a systematic review, minimize the risk of bias and provide transparency for replication of the process, a predetermined research methodology protocol was used, based on the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) checklist [17]. This was registered with PROSPERO. (Trial registration number: CRD42019135950. Available from: http://www.crd.york.ac.uk/PROSPERO/display_record.php?ID=CRD42019135950).

Selection criteria

Inclusion

Predetermined inclusion and exclusion criteria were used. Only systematic reviews, randomized controlled trials (RCTs), quasi randomized trials and controlled clinical trials were considered for inclusion as they form the hierarchy of evidence and are most likely to provide a robust evidence base suitable for informing clinical practice [18]. Those papers found were then screened for the following criteria:

  • Trials in which an IA CSI into the 1st MPJ used for the treatment of OA in adults,
  • Diagnosis and grading of OA in participants could be achieved via clinical examination and/ or via radiological means [19],
  • Any gender or ethnicity was considered.

In order to be able to determine the efficacy of treatment, trials were required to have provided quantitative or qualitative measures both pre- and post-intervention in order to be able to ascertain the mean differences relating to pain and/or joint function outcomes.

Exclusion

Trials in which intradermal, subcutaneous, intramuscular or extracapsular corticosteroid injections were performed were excluded, as were not trials that tested the efficacy of IA CSIs for conditions other than for OA, or tested CSIs at joints other than the 1st MPJ. Due to the high risk of bias, cohort and case studies, articles based on expert opinion, retrospective studies and narrative-based literature reviews were excluded [18].

Search strategy and data sources

To answer the research question a keyword search of six electronic databases (AMED, CINAHL, EMBASE, MEDLINE, PUBMED, and COCHRANE) up to February 2020 was undertaken by graduate research podiatrist (GB) to identify clinical trials that had tested the efficacy of IA CSI for the treatment of 1st MPJ OA.

AMED (1985 to 05.02.2020)

CINAHL (1982 to 05.02.2020)

EMBASE (1974 to 05.02.2020)

MEDLINE (1950 to 05.02.2020)

PUBMED (1966 to 05.02.2020)

COCHRANE (1966 to 05.02.2020)

No date or language restrictions were applied. Reference lists were reviewed, and key author searches were made to reduce the risk of any pertinent literature being missed. A list of keywords and results yielded are provided in Table 1.

# Database Search term Results
1 AMED (osteoarthritis).ti,ab 2945
2 AMED (hallux).ti,ab 1252
3 AMED (metatarsophalangeal).ti,ab 771
4 AMED (injection).ti,ab 2035
5 AMED (steroid).ti,ab 454
6 AMED (hallux limitus).ti,ab 62
7 AMED (hallux rigidus).ti,ab 178
8 AMED (1 AND 2) 35
9 AMED (1 AND 3) 37
10 AMED (6 OR 7 OR 8 OR 9) 272
11 AMED (4 AND 10) 5
23 CINAHL (osteoarthritis).ti,ab 21838
24 CINAHL (hallux).ti,ab 2033
25 CINAHL (metatarsophalangeal).ti,ab 1197
26 CINAHL (injection).ti,ab 43132
27 CINAHL (steroid).ti,ab 15241
28 CINAHL (hallux limitus).ti,ab 100
29 CINAHL (hallux rigidus).ti,ab 319
30 CINAHL (23 AND 24) 63
31 CINAHL (23 AND 25) 82
32 CINAHL (28 OR 29 OR 30 OR 31) 472
33 CINAHL (26 AND 32) 13
34 EMBASE (osteoarthritis).ti,ab 79498
35 EMBASE (hallux).ti,ab 5812
36 EMBASE (metatarsophalangeal).ti,ab 3924
37 EMBASE (injection).ti,ab 581417
38 EMBASE (steroid).ti,ab 163137
39 EMBASE (hallux limitus).ti,ab 153
40 EMBASE (hallux rigidus).ti,ab 664
41 EMBASE (34 AND 35) 183
42 EMBASE (34 AND 36) 258
43 EMBASE (39 OR 40 OR 41 OR 42) 1068
44 EMBASE (37 AND 43) 21
45 EMBASE (38 AND 43) 12
46 CINAHL (27 AND 32) 5
48 AMED (5 AND 10) 4
49 Medline (osteoarthritis).ti,ab 54837
50 Medline (hallux).ti,ab 4904
51 Medline (metatarsophalangeal).ti,ab 3209
52 Medline (injection).ti,ab 449653
53 Medline (steroid).ti,ab 125109
54 Medline (hallux limitus).ti,ab 139
55 Medline (hallux rigidus).ti,ab 586
56 Medline (49 AND 50) 137
57 Medline (49 AND 51) 189
58 Medline (54 OR 55 OR 56 OR 57) 858
59 Medline (52 AND 58) 13
60 Medline (53 AND 58) 5
61 PubMed (osteoarthritis).ti,ab 80277
62 PubMed (hallux).ti,ab 6554
63 PubMed (metatarsophalangeal).ti,ab 4096
64 PubMed (injection).ti,ab 708493
65 PubMed (steroid).ti,ab 936715
66 PubMed (hallux limitus).ti,ab 167
67 PubMed (hallux rigidus).ti,ab 656
68 PubMed (61 AND 62) 251
69 PubMed (61 AND 63) 298
70 PubMed (66 OR 67 OR 68 OR 69) 1054
71 PubMed (64 AND 70) 26
72 PubMed (65 AND 70) 10

Table 1 Search terminology and results yielded by database.

Risk of bias

In order to assess their validity, RCTs were reviewed using the Critical Appraisal Skills Programme (CASP) checklist [20], which uses six quality assessments of studies and considers the risk of (selection, performance, detection, attrition and reporting) bias. Systematic reviews were appraised using a Centre for Evidence-Based Medicine (CEBM) appraisal tool for systematic reviews [21] which uses six quality assessments to determine validity of reviews based on methodological design. Each quality assessment for data was awarded a ‘low’, ‘high’ or ‘unclear’ risk of bias. Two reviewers independently (GB, GF) appraised the studies and results were collated. If there was disparity between results, a discussion was to be raised. If consensus could not be achieved the senior author (INR – a consultant podiatric surgeon with a special interest in injection therapy) was appointed to make the final decision. Evidence from the identified literature was considered and an appropriate weighting awarded based on the quality of evidence they provided.

Initial inter-rater results following an appraisal of studies was 84% consistent between two reviewers. Following a discussion regarding the variation in quality assessment, 100% consensus between reviewers was achieved. Evidence from the identified literature was considered and an appropriate weighting awarded based on the quality of evidence they provided. Themes regarding joint pain, function and the safety of CSIs are discussed. Due to only one RCT being identified for inclusion, no meta-analysis was possible.

Data extraction

Data was extracted from research that fulfilled the inclusion criteria by using a predetermined list of parameters to determine the efficacy of the intervention and validity of methods used for testing.

Figure 1 PRISMA flow chart for trials selected for review [17].

These parameters considered: the design of study, sample size, demographics, diagnostic criteria used, disease severity, intervention tested (type, dosage, method of administration), outcomes, follow up and results. Reported adverse effects (type, duration and severity) were recorded to determine the safety of the intervention. Data from these themes was entered into a spreadsheet to be used for discussion.

Results

A search of electronic databases identified 111 studies for possible inclusion. Sixty-four duplicates were excluded and 47 titles and abstracts were assessed. Titles and abstracts were assessed independently (GB and GF) and evaluated against the aims of this study and its predetermined selection criteria. Full-text articles believed to be appropriate were accessed and further assessed for relevance against the predetermined inclusion criteria. If there was a difference in opinion as to whether an article should be included for review, a discussion was raised between the two main authors and if it was not possible to reach a consensus then the senior author was given the final vote on selection. 36 articles were rejected and 11 full-text articles were retrieved for assessment against the selection criteria (Figure 1). One RCT and one systematic review were identified for inclusion in this review.

Randomized controlled trials

One single blinded randomized trial that compared the efficacy of a single dose of intra-articular triamcinolone acetonide (TA) with sodium hyaluronate (SH) delivered without image guidance for mild symptomatic hallux rigidus in thirty-seven adults was identified for inclusion [22] – see Table 2. The title of the paper was misleading (sodium hyaluronate in the treatment of hallux rigidus. A single blind randomized study) in that its use of CSI was not mentioned.

Pons et al. 2007 [22]
Quality Assessment: Result: Bias Risk: Quality score:
Did the trial ask a clearly focused question? Yes Screening question 2/2
Was the assignment of patients randomized? Unclear Selection bias 1/2
Were all the patients who entered the trial properly accounted for at its conclusion? Yes Attrition bias, reporting bias 2/2
Were patients, health care workers and study personnel ‘blind’ to treatment? No Performance bias, detection bias 0/2
Were the groups similar at the start of the trial? Unclear Selection bias 1/2
Aside from the experimental intervention, were the groups treated equally? Yes Performance bias 2/2

Table 2 Quality assessment of randomised controlled trials (CASP checklist).

Zammit et al. 2010 [16]
Quality Assessment: Result: Quality Score:
What question did the systematic review address? Which interventions are optimal for treating osteoarthritis of the big toe? 2/2
Is it unlikely that important, relevant studies were missed? Yes 2/2
Were the criteria used to select articles for inclusion appropriate? Yes 2/2
Were the included studies sufficiently valid for the type of question asked? No, identified a lack of available evidence and high risk of bias. 0/2
Were the results similar from study to study? One study identified for inclusion only. 0/2

Table 3 Quality assessment of systematic reviews (CEBM framework).

Changes in joint pain and function

A reduction in mean visual analogue scale (VAS) pain scores at rest or on palpation was observed in both treatment groups. Mean VAS scores (n/100 mm) reduced at baseline from 58.7 mm to 34.1 mm in the TA group. A significant decrease in dorsiflexion or plantarflexion VAS pain scores was also observed in both groups: mean VAS scores decreased from 64.2 mm to 41.6 mm in the TA group. TH demonstrated reduced improvement in VAS pain scores on walking 20 metres compared to SH. Recipients of TA were reported to have a mean improvement in hallux function of 4.1 on the American Orthopaedic Foot and Ankle Society Score (AOFAS) for hallux evaluation. Overall, TA was found to be inferior in terms of the number positive responders to treatment, pain reduction and improvement in hallux function when compared to those treated with SH. Benefits were reported as relatively short lasting in both arms of the trial: 52.9% in the TA group and 46.6 % in the SH group progressed to surgery within 12 months.

The mean quality score for the RCT reviewed was 66% demonstrating limited methodological quality and potential bias. In this trial there was no attempt to blind investigators involved in data collection and evaluation of outcome measures. The trial had a small sample size with a significant female gender bias and all participants had mild joint disease potentially limiting the application of conclusions drawn from this to other patient populations. However, the most significant limitation with this trial was that interventions were administered to participants with 1st MPJ OA and hallux valgus with no sub-group analysis provided according to condition. This caused the paper to be rejected from the 2015 Cochrane review [16]. Given that the underlying pathophysiology of these distinct conditions differs, it is reasonable to expect that treatment outcomes relating to joint pain and function following an IA SCI may vary between recipients with different conditions. Furthermore, the proportion of recipients reported to have progressed to surgery may have been skewed given that the usual treatment for hallux valgus is surgical correction of the deformity. From this trial it was not possible to determine the efficacy of corticosteroids as an intervention to treat osteoarthritis at the 1st MPJ.

Adverse effects

Similarly, the lack of blinding in data collection and evaluation of adverse effects associated with the interventions administered poses a significant bias risk. Due to the lack of sub group analysis it was not possible to determine whether the frequency or type of adverse effects differed by condition. Data relating to adverse effects was collected by non-blinded investigators post intervention, were mild and arose in just 5% of recipients; no serious adverse effects were reported.

Systematic reviews

A recent review [14] that set out to provide a comprehensive list of evidence-based recommendations regarding conservative treatment modalities for 1st MPJ OA included a review of injection therapy. Authors of the review found ‘fair evidence’ to support the use of IA CSIs to treat 1st MPJ OA. However, the methodology was neither systematic nor comprehensive: only a single database was searched for clinical trials and the risk of pertinent literature having been missed was high. The author’s recommendations were made based on an appraisal system [23] that allocates a level of evidence for an intervention based solely on the design of studies identified; it does not consider the methodological quality of trials or risk of bias. Rama [24] pointed out that this system is a derivative of the levels of evidence system [25] and cautioned regarding the limitations of this style of review. He highlighted the need to not generalise evidence in order to avoid misleading conclusions being drawn.

The injection therapy trials identified in this review lacked heterogeneity in terms of solutions tested and design of trials. In spite of this, the authors grouped six trials relating to injection therapy together for data analysis and a collective level of evidence was allocated to injection therapy as a whole. Since this review did not consider the risk of bias and validity or clinical significance of outcomes from trials it identified, and failed to use a systematic methodology the study was excluded from this review as it was deemed to provide a summary of interventions for healthcare professionals only [24].

This review identified one systematic review that considered the efficacy of any treatment modality, including but not limited to injection therapy, for 1st MPJ OA [16]. The 2010 systematic review (see table 3) was a comprehensive piece of research with high quality methodology and low risk of bias. It identified one low quality study with a high risk of bias to support the use of physical therapy to reduce the pain of osteoarthritis at the big toe joint. It found no evidence to support the efficacy of corticosteroid injections for hallux rigidus (see note above re Pons et al, 2007).

Discussion

Originally suggested by Cotterill in 1887 [26], hallux rigidus/limitus (1st MPJ OA) are terms used to describe arthritic changes at the 1st MPJ. Many theories regarding the etiology of 1st MPJ OA have been postulated. Traditionally, osteoarthritis was viewed simply as a degenerative condition characterized by the degeneration of joint cartilage over time that resulted in progressive pain, stiffness and loss of joint function. However, a greater understanding of the pathophysiology of osteoarthritis indicates that symptoms arising from the disease are caused by the body’s attempt to repair damaged cartilage and that it is this process of repair and remodelling that results in abnormal bone growth and inflammation that involves the entire joint [16].

In a review of 114 patients it was found that irrespective of age, females are twice as likely to develop 1st MPJ OA [27]. A positive family history is strongly associated with bilateral joint disease, whereas unilateral joint involvement is often precipitated by trauma and does not routinely progress to involve both feet. Little consensus exists between studies regarding other possible causes although Coughlin and Shurnas [27] discuss pes planus, Achilles tendon contracture, hallux valgus, hallux valgus interphalangeus, a flat metatarsal head, metatarsus adductus, a long first metatarsal, metatarsus primus elevatus, and first ray hypermobility in the development of this condition. Furthermore, a number of recent retrospective studies that have considered the natural course of 1st MPJ OA suggest that progression of the disease is far more variable than previously thought and that for many it may follow a more benign course with symptoms that can be adequately managed with conservative treatment methods such as physical, mechanical or pharmacological therapy [28]. It is therefore increasingly important for clinicians to understand when to administer IA CSIs and which patients would derive the greatest benefit from treatment.

Corticosteroid is a synthetic version of the endogenous hormone glucocorticoid found in vertebrates that is produced in the adrenal gland cortex. Amongst its other functions in the cardiovascular, metabolic and nervous systems; glucocorticoids provide a feedback mechanism within the immune system to reduce inflammation. Synthetic corticosteroids administered orally or via injection can be exploited to mimic this action and can be used to suppress unwanted, immune mediated inflammatory responses caused by many disease processes including osteoarthritis. Corticosteroids act to reduce inflammation and suppress the immune response at various levels:

  • Leukocytes and monocytes transform into macrophages, a larger and more bactericidal cell that releases lysosomal enzymes that ushers in further inflammatory processes. By suppressing the adhesion of leukocytes, the formation of macrophages is reduced which inhibits the release of lysosomal enzyme and leads to a reduction in further inflammation [29].
  • Lymphocytes aid in activation of T cells and macrophages that have been produced causing rapid division and cytokine secretion. Cytokines are associated with both the initial activation and ongoing sensitization of the nociceptive receptors on sensory neurons perceived as chronic pain mediators. By reducing the effect of lymphocytes by depleting the amount of T cells and secretion of cytokines pain is reduced [30].
  • Cytokines are also responsible for releasing eicosanoid, a signalling molecule that stimulates other inflammatory mediators including histamine and prostaglandins. Both histamine and prostaglandins cause vasodilation of the surrounding blood vessels. This vasodilation leads to increased swelling and also contributes to the sensitisation of nerves resulting in pain perception. By reducing vasodilation and stimulation of pain receptors swelling and pain are reduced [31].

This systematic review was conducted in order to assess the effectiveness and safety of intra-articular corticosteroid injection as a treatment modality for 1st MPJ OA. A thorough and systematic literature search was completed in order to identify pertinent literature on the subject area and forty-seven studies were identified for possible inclusion. After exclusions were applied from the selection criteria to ensure that the correct condition, joint and treatment were being considered 11 pieces of literature remained of which two have been considered in detail. The remaining literature was mainly comprised of studies that provide low level evidence such as narrative reviews, retrospective case studies or non-controlled clinical trials.

One single blind randomized trial that compared the efficacy of a single corticosteroid injection with hyaluronate was identified [22]. A critical appraisal of this trial found it to have a high risk of bias. Furthermore, the solutions administered to participants were for two distinct conditions, hallux valgus and hallux rigidus and no details for sub group analysis were provided. It was therefore not possible to determine what influence this may have had on the outcome measures relating to pain reduction and improved joint function for hallux rigidus. From this trial it was not possible to determine with any level of certainty or specificity the efficacy of corticosteroids as an intervention to treat osteoarthritis at the hallux.

CSIs are generally considered safe drugs with steroid flare being the most commonly reported adverse event, though rare complications that may arise following administration of intra-articular steroid including anaphylaxis, disturbance of menstrual pattern and avascular necrosis [32]. Data relating to adverse effects was collected by Pons, et al., post intervention were mild, and arose in just 5% of recipients. It was not possible to determine the quality of reporting of adverse effects in this trial or whether adverse effects arose in hallux valgus and/or hallux rigidus joints. However, the reported rate of adverse effects is homogenous with the 6% rate of mild adverse effects reported by following 1,708 steroid injections into both soft tissue and joints of the foot and ankle [33]. The most common side effect reported was a steroid ‘flare’, an acute inflammatory reaction to the steroid solution which made up 75% of the reported side effects. Vasovagal episodes, facial flushing, local skin reactions, short term paraesthesia and a temporary increase in blood glucose levels were also reported but were rare. No infections were reported by the study, a result consistent with the view that joint infection is a very rare complication resulting in septic arthritis. No adverse effects following the administration of 22 CSIs for hallux rigidus were noted by Grice, et al., [34] although they do report that the positive results (seen in 20 of the 22 patients) only lasted longer than three months in three of that cohort. At two years, two patients (9%) remained asymptomatic, but 12 patients (55%) had undergone surgery. Peterson and Hodler [35] and Kilmartin [36] also note that most adverse effects experienced following an intra-articular joint injection of steroid are mild and transient and can be managed by the patient with self-care advice. These papers support the anecdotal view that in general, CSIs are safe and that adverse effects tend to be moderate and time-limited.

Numerous narrative reviews exist regarding treatments for hallux rigidus and include CSIs but provide no evidence-based recommendations for treatment. An exception to this was a comprehensive review [14], the aim of which was to provide evidence-based recommendations regarding conservative treatment modalities for hallux rigidus and included a review of injection therapy. Authors of the review based their recommendations on an established appraisal system [23] that allocates a level of evidence for an intervention based on the design of studies identified. Rama [24] pointed out that this system is a derivative of the widely established levels of evidence system [25] and cautioned regarding the limitations of this style of review. He highlighted the need to not generalize evidence in order to avoid misleading conclusions being drawn. King, et al., grouped six trials relating to injection therapy together for data analysis regardless of the fact that interventions and trial designs differed. A ‘collective’ level of evidence was allocated to injection therapy in general rather than by individual solutions. This led to skewed results given that the quality of trial design that had tested hyaluronate was superior to other interventions such as corticosteroid. Given that this review did not use a methodology that considered the risk of bias, validity or clinical significance of results of trials this study was excluded from this review as it was deemed to provide a narrative review.

One systematic literature review that included an appraisal of the efficacy of corticosteroid injections for osteoarthritis at the big toe joint [16] was included in this review. The Cochrane review was well designed, well executed and found to have a low risk of bias. Zammit, et al., [16] did not identify any robust evidence to support the efficacy of corticosteroid injections for the treatment of hallux rigidus and made no recommendations regarding its safety due to the high risk of bias. This view is consistent with the findings of this review that found it was only possible to make generalizations relating to the safety of intra-articular corticosteroid injections.

This review did not find evidence of sufficient quality to confirm whether intra-articular corticosteroid injections are an effective intervention for the management of symptomatic osteoarthritis at the 1st MPJ. The current literature that exists was found to be of poor methodological design. In the only randomized controlled clinical trial that tested corticosteroid, it was found to be mildly inferior to hyaluronate in terms of pain reduction for patients with mild osteoarthritis [22]. However, in a robust randomized placebo controlled [38] trial of intra-articular injections for osteoarthritis no benefit was derived from sodium hyaluronate vs saline placebo.

Conclusion

There are a number of narrative reviews concerned with the conservative and surgical treatment modalities that can be used to inform the management of symptomatic hallux rigidus. A number of cases and retrospective [26,27] studies have evaluated the use of injectable corticosteroids in the foot or ankle but controlled clinical trials in this area are few.

Many interventions exist that are intended to reduce the symptoms associated with OA of the 1st MPJ. In spite of the lack of evidence to support their use, IA CSI remains popular amongst health care professionals and patients alike because they are quick and inexpensive to administer with the perception of rapid relief, minimal recovery time and few side effects [32]. In cases of mild osteoarthritis, some retrospective studies indicate that CSIs may provide months and occasionally, years of relief for hallux rigidus [28]; a retrospective study by Smith, et al., in 2000 [37] found 75% of patients that had previously declined surgical treatment for symptomatic hallux rigidus were happy with this decision, had not experienced an increase in pain undergone despite degeneration of the joint, and were able to manage symptoms with stiff soled shoes and accommodative footwear. It is unclear whether progression to surgery has any association with the administration of intra-articular corticosteroid but given the risk of chondrotoxicity [15] this warrants further investigation.

This review found no high quality evidence to support the use of IA CSI as an effective treatment modality for symptomatic 1st MPJ OA. Uncertainty regarding variables that may influence treatment outcomes such as concomitant footwear use [39] remains. Existing research that tested intra-articular corticosteroid was found to be of poor methodological design with a high risk of bias. High quality, randomized, controlled clinical trials that test the efficacy of IA CSI are required. The severity of 1st MPJ OA amongst recipients in trials should be classified prior to intervention by clinical and radiological examination [19] and a sub group analysis of outcome measures provided according to disease severity. Further research to determine whether treatment outcomes are improved by the use of image guidance, extrapolation of side effects [40] and whether the use of IA CSI in 1st MPJ reduces surgical burden would be beneficial.

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Expansive unicameral bone cyst occupying the distal tibia: A case report

by Andrew Robitaille, DPM1*; Lawrence M. Fallat, DPM, FACFAS 2

The Foot and Ankle Online Journal 13 (3): 11

Unicameral bone cysts (UBC) of the distal tibia are usually incidental findings. We present a case of a 50-year-old female who initially presented with chronic bilateral heel pain. Initial radiographs revealed plantar heel spurs, but also a large intraosseous cyst in the distal right tibia. Computed tomography was obtained which showed a large, multiseptated, lucent, expansile bone lesion in the central medullary canal the distal metaphyseal-diaphyseal junction of the distal tibia. To prevent fracture of the thin cortex and stop expansion of the cyst, surgical intervention was chosen. This case report serves to show how standard x-ray revealed a large UBC that could result in fracture of the distal tibia.

Keywords: bone tumor, cyst, unicameral, tibia, benign

ISSN 1941-6806
doi: 10.3827/faoj.2020.1303.0011

1 – Resident, Submitted during Postgraduate Year 1, Beaumont Hospital, Wayne, MI, Podiatric Foot and Ankle Surgical Residency
2 – Director, Beaumont Health Wayne Podiatric Foot and Ankle Surgical Residency, Beaumont Hospital, Wayne, MI
* – Corresponding author: andrew.robitaille@beaumont.org


Unicameral bone cysts (UBC) are relatively uncommon benign bone tumors found mostly in the metaphysis of long bones, such as the humerus or femur with a male to female ratio of 3:1 [1]. UBC represents about 3% of primary tumors seen within the first two decades of life. In a meta-analysis by Kadhim, et al., in 2014, the distal tibia was only affected by UBC in 0.07% of reported cases [1]. Most of these lesions go unnoticed as they are usually asymptomatic in the absence of pathologic fracture [2]. In 1876, Virchow first described these lesions as cystic structures caused by abnormalities in local circulation [3].

Treatment goals for UBC include reestablishing bone strength, cortical thickness and elimination of the cyst [1,2]. There are various treatment modalities for UBC, which include conservative and surgical treatment [4]. Before surgical intervention, a thorough examination and clinical history must be obtained. Plain film radiography is usually sufficient for visualization and diagnosis of bone tumor [1]. Computed tomography (CT) or magnetic resonance imaging (MRI) should be obtained to evaluate the extent, size, and character of the tumor for treatment planning [2].

In this report, we present an unusual case of an adult female patient who was diagnosed on standard radiographs with an incidental finding of a large cystic lesion occupying the metaphyseal-diaphyseal region of the distal tibia. With the aid of CT, the lesion was further evaluated and then surgically treated with curettage and filled with allogenic bone chips and demineralized bone matrix (DBM).

Case Report

A 50-year-old female presented to the clinic with the chief complaint of bilateral plantar heel pain. Plain films were obtained and a large cystic lesion was noted to the patient’s right distal tibia (Figure 1). The patient denied any inciting event. Upon physical exam, there was mild dull pain at the end range of dorsiflexion to the right ankle, but was otherwise unremarkable. Computed tomography (CT) of her right ankle was obtained to further evaluate the cystic lesion.

Figure 1 Lateral and anteroposterior radiographs showing initial clinical presentation of a large expansive cystic lesion of the distal tibia.

Figure 2 Computed tomography imaging revealing a large multiseptated cyst in the metaphyseal-diaphyseal junction of the tibia with noted thinning of the medial cortex.

CT results showed a large, multiseptated, lucent, expansile bone lesion with thin medial cortex in the central medullary canal in the metaphyseal-diaphyseal junction of the distal tibia. The lesion measured 3.9 x 4.1 x 6.8 cm (Figure 2). The patient was booked for surgical excision and curettage of the right distal tibia bone cyst with insertion of allogenic bone graft, DBM, and included a biopsy.

The patient was brought into the operating room and placed on the operating room table in the supine position. General anesthesia was administered. The right lower extremity was prepped, marked and draped in the usual aseptic manner and a pneumatic thigh tourniquet was then inflated to 325mmHg.

Figure 3 A) Cortical window on the anterior distal tibia B) Cystic contents which included fatty tissue with hard and soft bone.

Figure 4 Intraoperative anteroposterior and lateral views after filling of cyst cavity with allograft and demineralized bone matrix. Also shown is the fixation of the bone window with one third tubular plate with two 3.5mm nonlocking cortical screws placed proximally and one distally.

An anterior incision 8 cm in length, just medial to the tibialis anterior tendon, was made overlying the anterior ankle which allowed direct visualization of the distal tibia. Four drill holes were made in the anterior tibia outlining the planned cortical window which measured 4 cm x 2cm. The window was cut and removed in one piece revealing the contents of the cyst which contained multiple osseous septa with both hard and soft bone (Figure 3). The entire area of the bone cyst was curetted and excised. Complete cyst excision was confirmed with fluoroscopy and direct visualization. It was noted that there was a large posterior portion of the cyst tunneling 2 cm proximally and inferiorly from the cortical window. Following intramedullary debridement, the cortex remained intact on all sides with no evidence of fracture.  All the material was removed from the cyst and was sent to pathology. The cavity of the cyst was irrigated and 89% phenol was applied to the entire bone cyst area.

Figure 5 Histological slides displaying fragments of sclerotic trabecular bone, consistent with unicameral bone cyst (original magnification 10x and 40x, hematoxylin and eosin).

Due to the large defect post-curettage, the cavity was filled with a combination of crushed allogenic bone chips and DBM. The cystic cavity was completely packed and the bone window was replaced and tamped into position. To prevent displacement of the cortical window, a 7-hole 1/3 tubular plate was placed anteriorly with a distal bend to fit the contour of the tibia. Alignment of the plate was confirmed both visually and with fluoroscopy. Following this, two 3.5 non-locking cortical screws were placed proximal, and one 3.5 non-locking cortical screw was placed distally (Figure 4). The surgical site was flushed with copious amounts of antibiotic solution and closure was completed. Following the procedure, the patient was placed in a well-padded, bivalved, below-the-knee cast and instructed to remain non-weightbearing with the use of crutches. She was prescribed hydrocodone for pain and aspirin 325 mg twice daily to be taken for deep vein thrombosis prophylaxis. The pathology specimen revealed fragments of sclerotic trabecular bone with spindle cells, most consistent with unicameral bone cyst (Figure 5).

Figure 6 Anteroposterior and lateral foot view of patient at twelve months postoperatively highlighting complete consolidation of cortical window.

The patient continued to present to the clinic on a regular basis for postoperative evaluation and serial radiographs. On the first two postoperative appointments (week 1 and 3), the patient’s visual analog pain score (VAS) was 2 out of 10. Radiographs showed incorporation of bone graft material and the patient was allowed to partial weight bear as tolerated. By the patient’s third postoperative appointment (8 weeks), the patient’s VAS score was 0 out of 10. Radiographs were taken, revealing consolidation of the cortical window in the right tibia with no recurrence of bone cyst. At this time, the patient was transitioned into normal shoe gear and sent to physical therapy with goals of decreasing edema, increasing range of motion, and increasing strength. At the patient’s one-year follow-up, radiographs were taken revealing no recurrence of bone cyst and the patient remained asymptomatic and had no limitations on full activity (Figure 6).

Discussion

Multiple theories have been postulated for the pathogenesis of unicameral bone cysts. Blockage in the venous drainage is the most favored mechanism which occurs in rapidly growing and remodeling cancellous bone [2, 3, 4]. This increased pressure may lead to the resorption of bone. Others have postulated that there could be a disturbance in bone growth, intramedullary hemorrhages secondary to trauma that do not completely resolve, degenerative phase of benign tumor, and osteomyelitis [2,10]. Cyst fluid analysis has shown increased levels of prostaglandin e2, IL1 beta, and proteolytic enzymes which could lead to bone resorption and cyst formation [5].

Treatment for UBCs is either observation or surgical intervention [2]. Reported surgical treatment includes medullary decompression with cannulated screws or intramedullary nails, steroid injections, or curettage with autograft or allograft [1, 2, 5-8]. Scaglietti, et al., were first to describe percutaneous injection of methylprednisolone acetate for UBC treatment in 1974 with only 24% healing rate after one injection [8]. Several authors have found satisfactory results with healing rates between 50-90% with steroid injections, but most reported that several procedures were necessary for cyst consolidation [1,6]. It has been reported that steroid injection may prevent the pro-inflammatory cytokine activity that leads to cyst formation as well as to relieve cyst pressure due to trepanation [1, 8].

Other injectable materials including bone graft and demineralized bone matrix (DBM) have been evaluated. Lokiec, et al., in 1996 was the first to report the use of autologous bone marrow injections for UBC treatment in children with 100% success rate [9]. Other studies have used bone marrow graft in combination with DBM [7, 11]. These studies attribute the high success rate due to bone marrow’s osteoprogenitor cells in combination with the osteoinduction and osteoconduction properties of DBM [7, 11]. Multiple authors have evaluated the effectiveness of DMB alone as an injection with high success rates [6,7]. Cho, et al., in 2012 evaluated twenty-five patients with a unicameral bone cyst who were treated with intramedullary decompression followed by grafting of demineralized bone matrix [2]. They used a small incision to create a cortical window to allow for curettage and decompression of the cyst and subsequently injection a mixture of allograft bone and DMB. Their success rate was 100% with a mean healing time of 6.6 months. Two patients required a second procedure, which they determined the initial amount of bone void filler was not enough to fill the entire space. The authors concluded that mixture of bone graft material, DBM, and completely filling the cyst was successful with satisfactory results [2].

Due to the size of our patient’s cyst, we used a longer incision and created a cortical window over the anterior distal tibia. This approach made it possible for visualization and use of curettes to completely remove all cyst material and to obtain a biopsy. This approach may appear more aggressive than other reported procedures, but because of the large size of the cyst and age of the patient, it was necessary for visualization and complete curettage, biopsy, and filling of the cyst with allograft and DBM. Similar techniques have been reported from multiple authors with healing rates greater than 90% [1, 6, 7]. The surgical curettage is necessary to resolve the cyst, but also allows for biopsy, which is necessary to rule out malignancy such as Ewing’s sarcoma and osteosarcoma because they also present as cystic lesions radiographically [5].

Unicameral bone cysts are usually incidental finding with many factors that could contribute to their formation. Although there is no standardized treatment for UBC, the goal of treatment is to prevent pathological fracture and, in children and adolescents, to prevent skeletal deformities during growth. Surgical procedures such as curettage with allogenic bone graft have been shown to be successful treatment with low rates of recurrence [1, 6, 7]. The surgical curettage is necessary to obtain tissue for biopsy to determine pathology including Ewing’s sarcoma and osteosarcoma [5]. In this report, we present a patient who was treated successfully with surgical curettage and allogenic bone graft for a distal tibia UBC with no cyst recurrence after one year. Long-term clinical follow-up is necessary for observation of potential cyst recurrence.

References

  1. Kadhim, M, Thacker, M, Kadhim, A, & Holmes, L Treatment of unicameral bone cyst: systematic review and meta-analysis. Journal of Childrens Orthopaedics 2014;8(2):171–191.
  2. Cho, HS, Seo, SH, Park, SH, Park, JH, Shin, DS, & Park, IH. Minimal invasive surgery for unicameral bone cyst using demineralized bone matrix: a case series. BMC Musculoskelet Disord 2012;13:124.
  3. Virchow R. On the formation of bony cysts, in Uber die Bildung von Knochencysten. In SB Akad Wiss, 1876; pp 369–381, Berlin.
  4. Noordin, S, Allana, S, Umer, M, Jamil, M, Hilal, K, & Uddin, N. Unicameral bone cysts: Current concepts. Annals of Medicine and Surgery, 2018;34: 43–49.
  5. Cohen J. Unicameral bone cysts. A current synthesis of reported cases. Orthop Clin North Am 1977;8:715–736.
  6. Kanellopoulos AD, Mavrogenis AF, Papagelopoulos PJ, Soucacos PN. Elastic intramedullary nailing and DBM-bone marrow injection for the treatment of simple bone cysts. World Journal of Surgical Oncology 2007;5, 1:111.
  7. Rougraff BT, Kling TJ. Treatment of active unicameral bone cysts with percutaneous injection of demineralized bone matrix and autogenous bone marrow. J Bone Jt Surg Am 2002;84-A 6:921–929.
  8. Scaglietti O, Marchetti PG, Bartolozzi P. Final results obtained in the treatment of bone cysts with methylprednisolone acetate (depo-medrol) and a discussion of results achieved in other bone lesions. Clin Orthop Relat 1982;165:33‐42.
  9. Lokiec F, Ezra E, Khermosh O, Wientroub S: Simple bone cysts treated by percutaneous autologous marrow grafting. A preliminary report. J Bone Joint Surg 1996;78: 934-937.
  10. Rosario, MS, Yamamoto, N, Hayashi, K, Takeuchi, A, Kimura, H, Miwa, S, Tsuchiya, H. An unusual case of proximal humeral simple bone cyst in an adult from secondary cystic change. World Journal of Surgical Oncology 2017;15:102.
  11. Di Bella C, Dozza B, Frisoni T, Cevolani L, Donati D. Injection of demineralized bone matrix with bone marrow concentrate improves healing in unicameral bone cyst. Clin Orthop Relat 2010;468: 3047-3055.

Treatment criteria for madura foot: Case report and literature review

by Wathmi Wijesinghe, MS21; Andrew Lee, MS21; Adrienne Estes, DPM2; David Shofler, DPM, MSHS2; Laura O’Connell, DPM3

The Foot and Ankle Online Journal 13 (3): 10

Mycetoma is a chronic granulomatous infection of the skin and underlying tissues, which affects remote populations in tropical and subtropical countries. We report the case of a 35-year-old male with an over 10 year history of left foot mycetoma, who initially presented with diffuse non-draining papules, edema, and dull chronic pain of the left foot. Radiographic imaging depicted erosive changes throughout the left midfoot and forefoot, while bone biopsy of the left navicular and first metatarsal confirmed actinomycetes. After being prescribed amoxicillin-clavulanate and trimethoprim-sulfamethoxazole and undergoing surgical debridement, the patient had marked improvements with less frequent pain and fewer blisters. One year later, the patient’s ankle joint remained untouched by mycetoma, yet his condition began to deteriorate with the reemergence of draining granules and chronic pain. As of now, the patient has been scheduled for below-the-knee limb amputation. The treatment of mycetoma aims to preserve limb function and prevent recurrences, but further research and investigations are necessary.

Keywords: Actinomycetoma, eumycetoma

ISSN 1941-6806
doi: 10.3827/faoj.2020.1303.0010

1- Podiatric Medical Student, Western University of Health Sciences
2- Assistant Professor of Podiatric Medicine, Surgery & Biomechanics, Western University of Health Sciences
3- Podiatric Medicine and Surgery Resident, Chino Valley Medical Center
* – Corresponding author: andrew.lee@westernu.edu


Mycetoma, also known as Madura foot, is a neglected tropical disease that induces a granulomatous inflammatory response in the subcutaneous tissue or deep dermis. The etiology of mycetoma can be fungal or bacterial, respectively termed as eumycetoma or actinomycetoma. Even though mycetoma is dispersed throughout the world, it was found to be indigenous to tropical and subtropical areas between latitudes 15° South and 30° North, an area commonly known as the “Mycetoma Belt”. Male populations living in these rural areas earn for their families by engaging in manual labor without proper footwear. Thus, transmission most commonly occurs through lacerations from cactus thorns, corn husks, or acacia trees. Manifestation includes foot pain with swelling and firm lesions to the site of laceration; raised ulceration sites; draining sinus tracts; and induration [1]. Diagnosis is best approached with biopsy, organism granule evaluation, serological and molecular methods, and imaging [1,2]. Magnetic resonance imaging (MRI) is generally more sensitive than radiographs, especially at earlier stages [1]. An antifungal or antibiotic regimen in conjunction with surgical debridement is considered to be the gold standard for treatment [1,2]. In its advanced stages, mycetoma may invade deeper structures including muscle, fascia, and bone and increase the risk of mortality or severe disability via amputations [1].

Treatment and management of mycetoma have been difficult in rural regions due to factors such as low health education, poor access to health care, social stigma, and socioeconomic burden; thus resulting in late presentation to clinical care and poor compliance to treatment [3]. It has been difficult to correct these factors as there is a limited bank of research and knowledge about mycetoma around the world. There is an increased need for more epidemiological studies to obtain a clear understanding and accurate data on transmission, prevalence, and incidence of this condition [4]. Though advanced diagnostic techniques are available in resourceful regions, health care professionals in endemic regions mostly rely on visual inspection for diagnosis thus increasing the need for more accessible diagnostic modalities [5]. Research on more aggressive treatments for mycetoma and the implementation of prevention methods may be required to improve the quality of life of the patient. However, when medication and surgical debridement are not the solution, health care professionals may resort to amputation to reduce the risk of mortality in these patients [6].

In the present report, we describe a 35-year-old male presenting with an advanced manifestation of this uncommon clinical condition. A review of the condition, including presentation, conservative treatment options, and surgical treatment options, is then discussed.

Case Report

A 35-year-old male construction worker with a history of a heart murmur and streptococcal pharyngitis presented for podiatry consultation for left foot pain and swelling in December, 2017. He was unclear on how the pain occurred. However, there was a high suspicion for Madura foot on this patient by his primary care physician (PCP). Further discussion in a follow-up visit revealed injuring himself from a corn husk while working on a corn field in Mexico. The symptoms had been slowly progressing over the preceding 10+ years before he presented to our care in 2017. The patient reported the pain to be chronic and dull. He had no known allergies, pertinent surgical history and pertinent family history. His medications included 875mg-125 mg per tablet of oral amoxicillin-clavulanate (Augmentin) which needs to be taken 2x day and 800mg-160mg per tablet of oral trimethoprim-sulfamethoxazole which needs to be taken 2x day. His physical exam of the extremities revealed hyperpigmentation and rigidity on the entire left foot with pain on the medial aspect (Figures 1 and 2). Papules were evident diffusely with no obvious purulence or drainage. This consultation ended with discussions and informed consent for his left foot bone biopsy as requested by his PCP.

Figure 1 Clinical image at presentation, Left dorsal foot. Hyperpigmentation noted diffusely from digits, extending proximally to the ankle with patches of fibrotic hypopigmentation.

Figure 2 Clinical image at presentation, Left plantar foot. Roughened, plantar bumps and papules of mycetoma with contour irregularity and hyperpigmentation.

A bone biopsy was performed on the left first metatarsal and the navicular. The first metatarsal biopsy was 0.5 cm in length and 2 mm in diameter for a dumbbell shaped portion of brown-colored osseous tissue. The pathology report of the first metatarsal identified benign-appearing fibro-osseous tissue however it was in part non-viable. The navicular bone biopsy was several fragments of light tan-colored osseous tissue measuring in aggregate of 0.5×0.2×0.2 cm. The report identified osseous tissue with numerous plasma cells, acute inflammation, periosteal fibrosis, and aggregates of filamentous microorganisms indicative of actinomycetes.  Wound cultures were positive for Staphylococcus aureus.

Radiographic images demonstrated destructive and erosive changes throughout the left midfoot and forefoot (Figure 3). An MRI revealed rounded lesions of intermediate signal with low signal central foci in the talar neck and body, calcaneus, cuboid, navicular, tarsal bones, cuneiforms, and metatarsals. Nodules were present in the dorsal soft tissue, medial soft tissue adjacent to the first metatarsal, and the plantar soft tissue adjacent to first and second metatarsals. Soft tissue edema was also noted extending to sinus tarsi. Bone marrow edema and intraosseous/extraosseous lesions surrounded by subcutaneous edema were identified with enhancement. Increased signal intensity on T2 views was evident of the intrinsic musculature and of the distal aspect of flexor hallucis longus muscle.

Three months later, he demonstrated marked improvements with only 1-2 blisters resurfacing with intermittent throbbing pain for 1-2 times per month. At this point, his surgical history included left foot biopsy, irrigation, and debridement of his left foot. He was still on the same medication plan however has not been consistent with his management of Madura foot. His physical exam revealed improved edema on his left foot but mild serous drainage from the dorsal aspect were present. And, indurated skin was present from digits to rear foot without any pain. Given the slow progression of the infection at that point, it was decided to continue his medications and closely monitor his conditions to see whether it reached the ankle joint.

After one year has passed, he presented with worsening pain over the past 4 months with granules which open and close. His current medications included a 500mg capsule of oral cephalexin (Keflex) which needed to be taken 1 capsule 2x daily. Next medication was 5-325mg per tablet of oral hydrocodone-acetaminophen (NORCO) which needed to be taken 1 tablet every 6 hours prn. Next medication included an 800 mg tablet of oral ibuprofen which needed to be taken 1 tablet every 8 hours prn. His physical exam of the lower extremity revealed improved edema on the left foot however with an appreciable amount of drainage. Indurated skin was present from digits to rear foot. Pain was noted on midtarsal joint range of motion.

Figure 3 Radiographic images taken in June, 2017. Left foot dorsoplantar view. Degeneration and erosions throughout the midfoot and forefoot with superimposed sclerosis and significantly narrowed joint spaces.

His recent x-ray images taken in December of 2019 have revealed the extent of erosions from phalanges to calcaneus however erosions still have not penetrated the ankle joint.

He continued to cycle between improvements and deterioration of symptoms. This may be attributable to his non-compliance to the given recommendations such as taking the appropriate medications in a timely manner and frequent visits to the health care providers. At this time, he has been experiencing deterioration of his symptoms with worsening pain and granule drainage. Therefore, he consented to a below the knee amputation (BKA) in hopes of obtaining permanent relief of his symptoms. As of now, the amputation was scheduled to take place sometime in 2020.

Figure 4 MRI image, sagittal view of the left foot. Rounded lesions of intermediate signal with low signal central foci were present in the talar neck and body, calcaneus, cuboid, navicular, tarsal bones, cuneiforms, and metatarsals. Soft tissue nodules were present in the dorsal soft tissue, medial soft tissue adjacent to the first metatarsal, and the plantar soft tissue adjacent to first and second metatarsals.

Figure 5 MRI axial view of the calcaneus, depicting rounded lesions of low to intermediate signal intensity with low signal central foci, characteristic of mycetoma.

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Figure 6 X ray images taken December, 2019. Degeneration and erosions have increased in number throughout midfoot and forefoot with superimposed sclerosis and significantly narrowed joint spaces. The erosions had extended to involve the rear foot with involvement of talus and calcaneus. However, his erosions still have not reached the ankle joint.

Discussion

Mycetoma is a chronic granulomatous infection of the subcutaneous tissue caused by true fungi (eumycetoma) or filamentous bacteria (actinomycetoma) residing in various habitats, including soil and residing organisms such as earthworms. Thus, mycetoma poses an occupational hazard to cultivators, farm laborers, shepherds, or agricultural workers. Offending bacterial or fungal pathogens penetrate via an abrasion site to permeate subcutaneous tissues. The infection remains dormant until spreading to deeper tissues and skeletal systems. Early diagnosis may be essential for better prognosis of these conditions however patients seeking medical treatment are usually at late stages making management extremely difficult [1]. Some of the contributing factors may include misdiagnoses and the lack of resources, trained health care professionals, and familiarity of the condition [4].

Clinical manifestations of mycetomas are similar despite the causative microorganism. Current research does not define a precise incubation period but suggests that it may range from three months to 50 years. During early stages, pain is not an essential component of the clinical picture. Overall the initial appearance of mycetoma can be characterized by papules, nodules, abscesses or indurated tissue without clear boundaries [3]. With time, draining nodules expel grains from interconnected sinuses tracking from the innermost abscesses [7]. Fungal organisms tend to generate different colors but mostly white or black granules, whereas bacterial granules generate a range of colors except black. Even though these granules help the offending organisms to evade immune detection, granule colorations are not pathognomonic for diagnostic purposes [8].

The advanced stage is characterized by a triad of symptoms including swollen, indurated, and deformed tissues; numerous communicating sinus tracts; and discharging aggregates of granules [9,10]. The affected skin retains the ability to heal draining sinus tracts, however, new tracts continuously form with discharge from various deep abscesses [1]. As the granuloma enlarges, the affected skin continuously deforms as a result of stretching of skin, pigmentation, and hyperhidrosis. Sweat gland hyperplasia and hypertrophy may be a consequence of overactivation of sympathetic nerves and/or rise in temperature due to extensive vascularization of the enlarging lesion [1,7]. During the period preceding highly advanced stages, this vascularization is apparent on angiography as dilated and convoluted arterial branches and veins proximal to the affected skin [1]. Blood supply is sufficient to preserve nerves and tendons until advanced stages of mycetoma [3,7]. During the advanced stages, pain may result from subsequent bacterial infections, metastasis to bone, and nerve damage. Despite the shared similarities in clinical presentation of mycetoma, it is notable that actinomycetoma is rapidly destructive and tends to extend to bone faster than eumycetoma [3]. Actinomycetoma was reported to invade the lymphatic system in rapid fashion leading to enlarged region lymph nodes [7]. Ultimately, delayed care can limit options and lead to poor outcomes, which may include severe disability, limb amputation or the need for multiple staged surgical excisions [4].

According to Hay et al., many health care professionals in endemic regions mostly rely on dermatological manifestations of the affected region for diagnosis due to lack of a required training and availability and economic burdens of other diagnostic tools [5]. This may contribute to numerous false positive outcomes and delays in accurate treatment for patients [11]. Since patients present in advanced stages of mycetoma, other diagnostic modalities are imperative to determine the extent of disease, staging of the disease, rule out differential diagnoses, such as Yaws and elephantitis, and confirm the exact causative treatment before starting treatment [12]. Cultures can be isolated via swab of sinus drainage or fine needle aspiration to distinguish the etiological agent, but cultures tend to be unreliable due to species variation, time needed for specimen growth, and high risk of contamination. Serological diagnosis via immunoelectrophoresis and enzyme-linked immunosorbent assay (ELISA) is another diagnostic option but is costly and unreliable with the need for purified antigens and the cross-reactivity between organisms. Yet, these disadvantages may be overcomed via histopathology [7].

Histopathology is a required diagnostic procedure with the potential to distinguish between eumycetoma and actinomycetoma before starting treatment [3,7]. Specimens need to be obtained from a deep surgical biopsy from a deep sinus tract or abscess to avoid contamination [7]. Grains can be visualized via Hematein-eosin-safran (HES), Periodic acid Schiff, and Gomori silver staining. More specifically, H&E staining illustrates eumycetoma as branched hyphae that arrange in groups and form vacuoles. Actinomycetoma with H&E staining demonstrates granules that are surrounded by eosinophilic fringe. Despite these benefits of histopathology, a major drawback includes the ability to only identify the mycetoma type not the causative agent [6].

In Western countries, molecular systematic protocols have been adapted to identify particular species and genera, allowing accurate selection of appropriate treatment options [11]. For actinomycetoma, 16S rRNA gene sequencing, PCR coupled with restriction endonuclease analyses, DNA fingerprinting with PCR amplification, or mass spectrometry can be employed to ascertain etiological species. For eumycetoma, fungal species can be recognized through PCR on an internal transcribed spacer sequence on fungal ribosomal DNA regions, PCR-restriction fragment length polymorphism analysis, and molecular typing via restriction endonuclease analyses and amplified fragment length polymorphism analyses [1,11] Despite these advantages, molecular diagnostics should not be a substitute for traditional methods, as molecular techniques are typically unavailable and costly in endemic regions [3]. Thus, traditional methods in conjunction with diagnostic imaging should be ordered initially if possible.

Diagnostic imaging is essential for confirmation of appropriate pathology and disease severity. Radiographic evaluation of mycetoma can be characterized by soft tissue edema, periosteal reactions, thickened and eroded cortex, destroyed joints, moth eaten appearance, osteopenia, and osteolysis [13]. Radiographic details suggestive of eumycetoma include a small number of bone lesions which are typically ≥ 1 cm in diameter while actinomycetoma contains more numerous, but smaller bone cavities [8]. Radiographic stages are defined in order to determine the severity and direction of metastasis. Radiographic stages are defined in order to determine the severity and direction of metastasis. Stage 1 features an enlarging granuloma with external pressure on bone. Stage 2 demonstrates periosteal reaction or reactive sclerosis. Stage 3 depicts the first indication of osseous involvement with erosion or cavitation on a single bone. Stage 4 demonstrates vertical progression into a single bone. Stage 5 is characterized by metastasis in horizontal direction to affect neighboring structures. Stage 6 is the most advanced stage of progression with metastasis of multiple planes [13].

Ultrasound is preferred for an accurate diagnosis of mycetoma even though it might not be readily available in endemic regions [1]. Granuloma containing granules adapt unique appearances which may aid in ruling out differential diagnoses and distinguishing actinomycetoma from eumycetoma [12]. The most distinguishing feature to confirm mycetoma diagnosis from ultrasound images is the “dot in the circle sign”. This sign can be defined by multiple round hypoechoic lesions with a hyperechoic focus in the center [4]. Doppler ultrasound can demonstrate the effect on mycetoma on vasculature of the affected region. Also, bacterial and fungal grains with a capsule and the accompanying granuloma have distinguishing ultrasound features which may be helpful in ruling out differential diagnoses [12]. Ultrasound can be helpful to determine the severity of mycetoma which may be essential for surgical planning [3].

In addition to ultrasound, MRI images are accepted to be highly essential in examining the extent of lesions and metastasis to adjacent structures [1]. Dot in the circle sign is not solely seen on ultrasound as it may be seen on MRI as oval hyperechoic lesions with a hypoechoic center [14]. On MRI images, high signal regions represent inflammatory granuloma while low signal regions within represent the granules. This is highly pathognomonic of mycetoma. Despite these advantages, MRI is expensive in rural regions, requires a high level of expertise to interpret the results and cannot be used to discriminate between actinomycetoma and eumycetoma [3,12].

Medical Management

The treatment and outcome of mycetoma depend on the causative agent and stage of disease progression [7]. Generally, patients with actinomycetoma have more favorable prognoses to medical treatment than those with eumycetoma [13]. Greater drug penetrance, due to the smaller granules (~1µm diameter) in actinomycetoma, effectively supports a wider array of antibiotic regimen options [15,16]. Since the 1960s, trimethoprim-sulfamethoxazole (TMP-SMX) has been the gold standard for first-line treatment of actinomycetoma, either as monotherapy or in conjunction with dapsone, a penicillin, or an aminoglycoside for more resistant organisms [17]. Currently, combination drug therapy is recommended to avoid resistance and enhance efficacy [7,18]. Antibiotic sensitivity testing should be performed to select the most optimal combination, treatment duration, and number of cycles, all of which vary from case-to-case and depend on soft-tissue or osseous involvement [6,16].

The Welsh regimen is a well-acknowledged combination therapy that has achieved cure rates of more than 90% in previous studies [16,19]. Interestingly, most of the cases from Welsh’s studies were from Mexico, where 98% of mycetoma is due to actinomycetes (86% is further classified as Nocardia brasiliensis) [20]. During the intensive phase, intramuscular amikacin (15 mg/kg/day) is administered in two divided doses, combined with oral TMP-SMX (7+35 mg/kg/day) in three divided doses for 21 days. One to three cycles are performed at 15-day intervals. While in the maintenance phase, oral TMP-SMX (7+35 mg/kg/day) is administered at the same dose for 15 days after the last cycle [3,16,21]. Using the Welsh regimen as a template, other case studies have published modifications to optimize drug efficacy [16].

Damle et al. modified the Welsh regimen by incorporating rifampicin, which resulted in successful remission of all 16 patients included in the study who had all previously had unsatisfactory responses to therapy [16,22]. Rifampicin was specifically selected for its potency as a second-line drug and its medical familiarity within developing regions in the treatment of leprosy and tuberculosis. Agarwal et al. then modified the procedure presented by Damle et al. by increasing the number of cycles to three cycles in cases with soft-tissue involvement and up to five cycles in those with osseous involvement.

Another well-established treatment schedule is the modified two-step Ramam regimen, which consists of intravenous gentamicin (80 mg) twice daily and oral cotrimoxazole (320/1600 mg) twice daily for 4 weeks in the intensive phase. The successive step includes oral doxycycline (100 mg) twice daily and oral cotrimoxazole (320/1600 mg) twice daily in the maintenance phase until five-six months after the complete healing of all sinus tracts is noted [3,16,23]. Although amikacin is the preferred aminoglycoside due to less nephrotoxicity and its characteristic resistance to bacterial aminoglycoside-inactivating enzymes, the Ramam regimen instead utilizes gentamicin primarily due to cost [16,23].

In patients with allergy or noted drug resistance, TMP-SMX can be substituted with amoxicillin-clavulanate, and amikacin can be replaced by netilmicin [1,6]. Additionally, amoxicillin-clavulanate can be used as monotherapy during pregnancy. Amikacin can also be combined with a carbapenem in cases refractory to sulfonamides [1,24]. Importantly, patients must be routinely monitored during and in between treatment cycles for any adverse effects from antimicrobial usage [6]. Especially for amikacin or other aminoglycosides, testing for renal, liver, and auditory function should be performed every three to five weeks to detect potential toxicity [25]. Surgery is rarely indicated in actinomycetoma, but may be advantageous for reducing disease load in larger lesions to enhance drug response or to control secondary bacterial infections in unresponsive patients [19,26].

Eumycetoma is associated with larger granules and extensive fibrosis, thus requiring more prolonged treatment durations ranging from one-to-three years for eumycetoma, compared with three months to one year for actinomycetoma due to lower drug penetrance [8,15]. Itraconazole (200-400 mg daily) for 9 to 12 months followed by surgical intervention has demonstrated favorable clinical responses in many studies and is currently the gold standard for treating eumycetoma [8,27]. In prior decades, Ketoconazole (400-800 mg daily) was a popular antifungal regimen, but has more recently fallen out of favor due to the risk for liver and adrenal toxicity [1,6]. It should be noted that disappointing and inconsistent results are common in pharmacotherapy of eumycetoma with recurrence rates between 20% and 90% [11,13]. Itraconazole and ketoconazole may not necessarily be curative, but their extended usage has exhibited the formation of thick fibrous capsules around the lesions, thus promoting the localization of disease for an easier and more complete surgical excision [7,26]. In a 20-patient open-label study, high-dose terbinafine (1000 mg daily) was administered for 24-48 months and demonstrated moderate efficacy. Results stated that 25% of patients were cured, while 55% had notable clinical improvement by the completion of the study [27,28]. Newer antifungal drugs from the azole class are currently being investigated for their promising potential, featuring broad spectra, low toxicity and favorable bioavailability [6,11].

Treatment therapies for both actinomycetoma and eumycetoma must be continued until the conditions for complete cure are met. Those criteria include clinical healing of sinus tracts, complete resolution of soft tissue masses both clinically and radiologically, restoration of normal osseous appearance with radiological evidence of remodeling, cytological absence of granules, and ultrasonographic absence of cavities [7,26].

Surgical Intervention

Generally, mycetoma of fungal origin requires more extensive surgical management than that of bacterial etiology [19]. In cases with no osseous involvement, wide surgical excision is indicated for localized early lesions to reduce disease burden and to facilitate therapeutic response [7,19,29]. If osseous involvement is confirmed, surgical debridement can be carefully performed to remove granules and damaged tissue from the cavities [6].

Amputation may be considered in advanced stages of mycetoma, when massive osseous destruction, secondary bacterial infection, or sepsis occur [6]. Still, is it important to note that amputation will not necessarily result in a complete cure, as noted by high postoperative recurrence rates of 25% to 50% [2]. In a 1013 patient retrospective study by Wadal, et al., several indicators were correlated to predicting post-operative recurrences, including lesion size greater than 10 cm at initial presentation, positive family history of mycetoma, previous surgeries, and disease duration of more than five years [30]. In addition, surgeons must meticulously excise with wide margins to ensure adequate excision of infected tissues or there may be a risk of new satellite lesions forming from lymphatic spread [2,6,26].

Limb amputation is accompanied by significant socioeconomic, functional and psychological consequences. Mycetoma most commonly affects young men between 20 and 40 years of age, who are generally the highest producers and earners in endemic communities [1]. Disabilities and deformities resulting from mycetoma can compromise employment and relationship opportunities in adults, while children are susceptible to becoming socially rejected and discontinuing education [3]. In a 2015 case study of two drug-resistant patients, Maiti, et al., examined the outcomes between an actinomycetoma patient who had undergone amputation after 9 years of treatment and an eumycetoma patient who continued to manage the slow progression of disease for 16 years after refusing amputation. The actinomycetoma patient was diagnosed with moderate depression post-amputation and also developed disease recurrence on her amputated stump 3 years later. The eumycetoma patient, however, maintained sufficient functionality for daily activities and was spared from anxiety or depression associated with amputation [31]. Thus, Maiti, et al., proposed that symptomatic management of drug-resistant mycetoma may perhaps be more beneficial to a patient’s quality of life than amputation and the corresponding morbidities [31].

Conclusion

Mycetoma is an exceedingly rare condition that is most commonly encountered in underdeveloped regions. Experimental studies and randomized clinical trials may help determine the efficacy of various treatment regimens to prevent the progression of Mycetoma to more advanced stages [11]. Further, there exists a need for further research and development of a standardized treatment regimen available at low cost in endemic countries.

References

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Distraction osteogenesis for treatment of a shortened first metatarsal after failed first metatarsophalangeal joint arthroplasty

by Tyler Reber DPM1*; Lindsey Hjelm , DPM2; Mallory Schweitzer, DPM, AACFAS3; Craig E Clifford, DPM, MHA, FACFAS, FACPM4

The Foot and Ankle Online Journal 13 (3): 9

Distraction osteogenesis is well documented in the literature as a viable treatment option for large bony defects and brachymetatarsia. Few studies have examined the use of this technique after failed arthrodesis or arthroplasty of the 1st metatarsophalangeal joint (MPJ). Acute correction with autograft is typically the procedure of choice for treatment of defects of the 1st MPJ. However, the amount of length that can be achieved is limited in acute correction and distraction osteogenesis should be considered for larger defects. This case study presents our treatment of a failed first MPJ arthroplasty that resulted in a defect at the 1st MPJ of greater than 2 centimeters and our technique of using external fixation followed by internal fixation to regain desired length and minimize time in the external fixator.

Keywords: Distraction osteogenesis, external fixation, monolateral rail, mini rail, failed 1st metatarsal arthroplasty, infected implant

ISSN 1941-6806
doi: 10.3827/faoj.2020.1303.0009

1 – Resident Physician, PGY-2, Franciscan Foot & Ankle Institute, Federal Way, WA
2 – Resident Physician, PGY-2, Franciscan Foot & Ankle Institute, Federal Way, WA
3 – Fellow Physician, CHI Franciscan Health Advanced Foot & Ankle Reconstructive Surgery Fellowship, Burien, WA,
4 – Residency Director, Franciscan Foot & Ankle Institute, Federal Way, WA
* – Corresponding author: rebertyler@gmail.com


Failed arthrodesis or arthroplasty of the first MPJ can often leave the revision surgeon with limited surgical options. The literature recommends, when possible, acute correction of bony defect over distraction osteogenesis due to decreased complication rates, faster time to healing, and reduced psychological strain that an external fixator can potentially cause a patient [1-3]. However, acute correction is limited by the soft tissues with potential neurovascular compromise. In these instances, distraction osteogenesis should be considered as it allows for the soft tissues to adapt as the bone lengthens [4].

Distraction osteogenesis by external fixation has been widely used and studied since Dr. Ilizarov first described his technique in the 1950’s. Dr. Illizarov described two main phases: the first being distraction where he determined the ideal rate was 1 mm per day and the second being consolidation wherein the newly formed regenerate gains strength [5-7]. In the standard technique, the patient spends the entire consolidation period in the external fixator and this can more than double the amount of time the patient spends in the fixator. A new technique that is gaining popularity is the conversion of the external fixation to internal fixation to limit the duration the patient spends in the fixator, and this technique was used in this case [8-9].

This case study presents our treatment of a failed first MPJ arthroplasty from deep infection after a failed first MPJ arthrodesis due to a non-union that left a defect at the 1st MPJ of 22 millimeters.

https://lh4.googleusercontent.com/5bnygpI1Ghf8MjT7-gUvUB51hSJklyjVtp3kD_klZeNma3XlUigLNCJSNpkFjTZQe75PeiawZxcX-1lbeD2IO3t3DYMM5GhWMe5dVKm0c5637bkcXZFhcohASHvyglR8BTx2A4pei_E https://lh4.googleusercontent.com/D8lVo-bSk6WO4ECc1wxueQVKAzC-KW2LfQlwVtjNdT7PWy-0fDxWppfX9tU4rKRiKnViAZ13HLkZVkqDwo-47JbUojLFpHSUXAO6Sn7zt0ceOi9CimViepjrNS9GjUOcGZF0337XQYI

Figure 1 (A) Preoperative clinical photo (B) Preoperative X-ray with antibiotic spacer.

The objective of this case study is to present our surgical technique for lengthening of the first metatarsal by distraction osteogenesis with external fixation to show its use as a viable treatment option for a shortened first metatarsal.

Case Study

A case is presented of a 52-year-old female who failed all conservative care for 1st MPJ arthritis of the left foot and underwent a 1st MPJ fusion. The 1st MPJ fusion failed due to a non-union and the decision was made to convert to a first MPJ arthroplasty. Infection of the implant with osteomyelitis was subsequently diagnosed with MRI at 3 months post-arthroplasty. The implant was removed at 3 months postoperative and was replaced by an antibiotic spacer and the patient was able to tolerate the spacer for two years (Figure 1). The area eventually became painful and removal of the antibiotic spacer was deemed necessary. Labs were obtained for vitamin D, prealbumin, comprehensive metabolic panel, parathyroid, and thyroid panel which were all within normal range. The length able to be achieved with acute correction was not felt to be adequate to restore proper function of the first ray, so distraction osteogenesis was selected as the treatment option. After removal of the antibiotic spacer, a mono-lateral external fixator was applied and used to lengthen the first metatarsal 22 millimeters of length, which was achieved after 26 days of distraction.

https://lh3.googleusercontent.com/zMuv_t3pOvBlUJL3IzVE1cTiKX4sePxiRFFbC4lJ4jUdvvrclQDKMQjcV18zBzxs9jjXfnZXipfmV389-yr9fJYbEBsvDSAZa2bKOKSdzgpAYImtQryXOKFz3b9X5IUO6yBCIZldEyc https://lh6.googleusercontent.com/lk9Ksc7-dsMJSDjdU1oCNCL3HRCyfqNGFpeP8fZHQ1rtWPGdxEV9x691XdWQu3gaGMpDHy7YcQ9_eSDu_5tQd_FnFhJCHQHNycR-Hn50qKXdyib66RxZI4gpR-t3_vQTYhkDtedF3tQ https://lh4.googleusercontent.com/dM9ngSpoCOSE6huTfVoEMMl4w0PvbLGxThXoInM52wJhFToe-pJ2oImKEhPj8Bu1u1_e3VupJxCt939m6plP4IOhVQfeNVW9hTT5qJKLEUDw48DM6TcX-_CmVrKnZdqTuQOJ4UH4JP4 https://lh6.googleusercontent.com/wOqg2On9WrwutLiv8LtYiX8hWOblFdNMAddGvkfMBndOPusZaLqOxMWs_stK6ISV11bsmTNJIov49hpOD6ZxDp-48h8TgBRN99PVaEBzZVxNzN8-FdZQgWAzr1okq3fhkhv0RLg_Z9A

Figure 2 (A)Intraoperative, Closure with External fixator application (B) Immediate postoperative with compression at corticotomy site (C) X-ray after 26 days distraction, 22mm of lengthening (D) Consolidation period post external fixator removal and internal fixation.

The external fixator remained on for an additional month to allow consolidation of the regenerate, but was removed at the patients request and was replaced with a locking plate. After five months, the locking plate was removed due to hardware irritation. A CT confirmed partial union of the first MPJ prior to hardware removal and the fusion site was stressed intraoperatively and found to be stable. The locking plate and screws were then removed. The patient was able to ambulate in tennis shoes without pain and was able to perform activities of daily living 1 year postoperatively.

Surgical Technique

A longitudinal incision was made medially over the 1st metatarsocuneiform joint extending distally to the medial hallux interphalangeal joint. Layered dissection was carried along the length of the incision down to the level of the antibiotic spacer within the 1st metatarsophalangeal joint site.

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Figure 3 (A) CT scan 14 weeks status post 1st MTPJ fusion showing osseous consolidation at the first MPJ (B) Immediate post-op hardware removal.

The spacer was removed and the nonunion margins were curetted and fenestrated down to healthy bleeding bone. Orthobiologics were used to augment the fusion site. A 0.062” K-wire was inserted through the tip of the distal hallux, crossing the hallux interphalangeal joint, and into the 1st metatarsophalangeal nonunion site. Correct placement of this K-wire is key to help guide the axis of regenerate formation as the bone is lengthened.

An osteotomy was made 1 centimeter distal to the base of the first metatarsal by perforating the bone with a k-wire and then completing the osteotomy with an osteotome. Intraoperative fluoroscopy was used to confirm the completion of the osteotomy.

Thereafter, a monolateral external fixator was applied to the dorsal aspect of the foot with 3 proximal pins within the medial cuneiform, 1 pin in the base of the first metatarsal, and 2 distal pins in the midshaft of the metatarsal. The osteotomy site was then compressed utilizing the external fixator and the 0.062” K-wire was driven across the osteotomy and into the base of the first metatarsal.

The patient began lengthening at POD #12 at a distraction rate of 0.25 millimeters four times per day. After 26 days of distraction, there was 22 millimeters of length achieved and distraction was discontinued. One month into the consolidation phase the external fixator and K-wire were removed at the patients request. The metatarsal was docked to the proximal phalanx at this time after fenestrating the opposing bony surfaces and a locking plate was used to span the regenerate and provide stability (Figure 3).

Discussion

This case study supports the current literature that distraction osteogenesis is a viable option for treatment of a large bony defect at the 1st MPJ. The patient was able to gain 22 millimeters of length through distraction osteogenesis and was able to undergo a successful fusion at the 1st MPJ with a stable fusion site and regenerate bone at the time of hardware removal. Placement of the K-wire so that it parallels the first metatarsal in both the sagittal and transverse planes is key in order to avoid malalignment of the newly formed regenerate. Although the patient did require removal of her hardware, she was able to return to normal shoe gear without pain and was able to perform her activities of daily living that had previously been limited at her one-year follow-up.

The literature currently supports single-stage procedures over distraction osteogenesis when possible. Jones, et al., in 2015 performed a systematic review of the literature comparing single stage vs distraction osteogenesis in the treatment of brachymetatarsia. They found that the overall major complication rate of distraction osteogenesis was 12.62% compared to 3.72% with single stage procedures and the minor complications rate was 39.18% for distraction osteogenesis compared to 15.76% for single stage. They also found that distraction osteogenesis is associated with a greater time to heal with an average of 2.31 months for every centimeter gained compared to 1.9 month per centimeter with a single stage procedure [1].

Even with these disadvantages, distraction osteogenesis does have a major advantage over single stage bone grafting in that much greater length can be achieved and donor site complications can be avoided. Recommendations have been made that no more than 15 millimeters or 25% of the original length of the bone be attempted with a bone graft. With callus distraction, the recommendation has been that the metatarsal can be increased in length up to 40% of its original length [11-12].

This case study also supports the conversion to internal fixation at the time of external fixation removal during the consolidation period. This allows for the benefits of external fixation in being able to gain greater lengths of correction while also limiting the time the patient spends in the external fixator. This technique is well supported in the literature and can help make external fixation more tolerable for the patient [8-9].

In any patient with a history of a non-union it is important to rule out any underlying conditions that may have caused the non-union. There are a variety of different causes for a non-union from improper joint preparation, improper/loss of fixation, smoking, low vitamin D levels, underlying metabolic disorders, among others [14-15]. It is important to order the appropriate labs pre-operatively and medically optimize the patient before considering return to the OR for surgical intervention. The protocol for ruling out underlying metabolic bone conditions for the senior author in our study (C.C.) is to order labs for vitamin D, prealbumin, a comprehensive metabolic panel, parathyroid, and a thyroid panel. In our patient these labs all came back normal indicating they most likely did not contribute to the patient’s original non-union.

In conclusion, bony defects too large for acute correction in the 1st MPJ provide difficult challenges to the surgeon but distraction osteogenesis is a viable option. Converting the external fixation to internal fixation can also help make distraction osteogenesis more tolerable to patients that are hesitant to spend extended periods of time in an external fixator.

References

  1. Jones MD, Pinegar DM, Rincker SA. Callus Distraction Versus Single-Stage Lengthening With Bone Graft for Treatment of Brachymetatarsia: A Systematic Review. J Foot Ankle Surg. 2015 Sep-Oct;54(5):927-31. doi:10.1053/j.jfas.2015.02.013. Epub 2015 May 19. Review. PubMed PMID: 25998479.
  2. Oh CW, Satish BR, Lee ST, Song HR. Complications of distraction osteogenesis in short first metatarsals. J Pediatr Orthop. 2004 Nov-Dec;24(6):711-5. PubMed PMID: 15502575.
  3. Mather R, Hurst J, Easley M, Nunley J. (2008). First Metatarsal Lengthening. Techniques in Foot & Ankle Surgery. 7. 25-30. 10.1097/BTF.0b013e318165c21c.
  4. Choi IH, Chung MS, Baek GH, Cho TJ, Chung CY. Metatarsal lengthening in congenital brachymetatarsia: one-stage lengthening versus lengthening by callotasis. J Pediatr Orthop. 1999 Sep-Oct;19(5):660-4. PubMed PMID: 10488871.
  5. Ilizarov GA. The tension-stress effect on the genesis and growth of tissues. Part I. The influence of stability of fixation and soft-tissue preservation. Clin Orthop Relat Res. 1989 Jan;(238):249-81. PubMed PMID: 2910611.
  6. Ilizarov GA. The tension-stress effect on the genesis and growth of tissues:Part II. The influence of the rate and frequency of distraction. Clin OrthopRelat Res. 1989 Feb;(239):263-85. PubMed PMID: 2912628.3. Skirving AP, Newman JH. Elongation of the first metatarsal. J Pediatr Orthop.3:508-510. 1983.
  7. Skirving AP, Newman JH. Elongation of the first metatarsal. J Pediatr Orthop. 1983 Sep;3(4):508-10. PubMed PMID: 6630498.
  8. Oh CW, et al. Submuscular plating after distraction osteogenesis in children. J Pediatr Orthop B. 2008;17(5):265–269. doi:10.1097/BPB.0b013e32830688d8
  9. Harbacheuski R, Fragomen AT, Rozbruch SR. Does lengthening and then plating (LAP) shorten duration of external fixation?. Clin Orthop Relat Res. 2012;470(6):1771–1781. doi:10.1007/s11999-011-2178-2
  10. Bartolomei FJ. Surgical correction of brachymetatarsia. J Am Podiatr Med Assoc. 1990;80(2):76–82. doi:10.7547/87507315-80-2-76
  11. Kim HT, Lee SH, Yoo CI, Kang JH, Suh JT. The management of brachymetatarsia. J Bone Joint Surg Br. 2003;85(5):683–690.
  12. Lee WC, Suh JS, Moon JS, Kim JY. Treatment of brachymetatarsia of the first and fourth ray in adults. Foot Ankle Int. 2009;30(10):981–985. doi:10.3113/FAI.2009.0981
  13. Harbacheuski R, Fragomen AT, Rozbruch SR. Does lengthening and then plating (LAP) shorten duration of external fixation?. Clin Orthop Relat Res. 2012;470(6):1771–1781. doi:10.1007/s11999-011-2178-2
  14. Moore KR, Howell MA, Saltrick KR, Catanzariti AR. Risk Factors Associated With Nonunion After Elective Foot and Ankle Reconstruction: A Case-Control Study. J Foot Ankle Surg. 2017;56(3):457–462. doi:10.1053/j.jfas.2017.01.011
  15. DeFontes K, Smith JT. Surgical Considerations for Vitamin D Deficiency in Foot and Ankle Surgery. Orthop Clin North Am. 2019;50(2):259–267. doi:10.1016/j.ocl.2018.10.008

 

Subchondroplasty in the lower extremity: A literature review

by Steven Cooperman, DPM1*; Thomas Yates, DPM1; David Shofler, DPM, MSHS1

The Foot and Ankle Online Journal 13 (3): 8

Osteoarthritis is one of the most common and debilitating conditions encountered by foot and ankle surgeons. This osteoarthritis is often accompanied by a coinciding bone marrow lesion (BML) which has been shown to result in poorer patient outcomes. The subchondroplasty procedure was developed with the aim of targeting these painful BMLs in order to slow the progression of osteoarthritic changes. There has been a trend in both orthopedic and podiatric literature towards the use of this procedure in the lower extremity. This review is meant to bring forward the information most pertinent to the procedure to help inform the foot and ankle surgeon of its uses and potential, as well as to encourage future research into the procedure.

Keywords: subchondroplasty, bone marrow lesion, osteoarthritis, calcium phosphate, bone substitute material

ISSN 1941-6806
doi: 10.3827/faoj.2020.1303.0008

1 – Department of Podiatric Medicine, Surgery, and Biomechanics, Western University College of Podiatric Medicine, 309 E 2nd Street, Pomona, CA 91766
* – Corresponding author: Scooperman@westernU.edu


Osteoarthritis (OA) remains one of most common and debilitating conditions encountered by foot and ankle surgeons. Whether the result of trauma or degenerative overuse, orthopedic and podiatric surgeons alike can agree that the sequelae of OA can be challenging to manage. The natural history of OA involves persistent joint pain, lack of normal function, and can include a vicious cycle which may progress to osteonecrosis of the affected bones. While the current body of evidence of in vitro cartilage repair and regenerative medicine is rapidly growing, there are perhaps other more readily available methods of treating OA which may ultimately demonstrate equal benefit to patients. Subchondroplasty® (SCP) (Zimmer Knee Creations, West Chester, PA) is a surgical system, developed in 2007, in which flowable bone substitute material (BSM) is injected into subchondral bone in order to fill a defect. The procedure acts to support the subchondral bone layer by providing a scaffold over which new, healthier osteochondral elements may be produced [1]. Although this technique has primarily been described in literature to treat bone marrow lesions (BMLs) in the knee joint, this technique has recently been applied to the foot and ankle with comparably successful outcomes.

This paper is not meant to serve as a technique guide, but a review of available relevant literature. As such, the use of the term subchondroplasty throughout the paper will be in reference to the procedure itself, not the proprietary system. The goal of this review is to benefit the foot and ankle surgeon by: first, providing a general understanding of the procedure and its expanding applications; second, by presenting the largely positive patient outcomes in both the orthopedic and podiatric literature in an attempt to encourage further study into a relatively new – yet promising – tool in the foot and ankle surgeon’s array of treatments.

Methods

An extensive search of available literature related to: 1) subchondral bone and the osteochondral unit; 2) lower extremity osteoarthritis; 3) bone substitute materials; 4) the subchondroplasty procedure, including its related radiographic findings and clinical outcomes in the lower extremity.

Background

Within joints, the subchondral bone layer is a supporting structure for the overlying articular cartilage. Subchondral bone is an underappreciated, yet vital component to the function of each osteochondral unit and overall joint health [2]. Bone metabolism is dynamic, in concert with Wolff’s law, and a normal subchondral bone plate displays the same capacity to increase in thickness according to physiologic loading [3].

In osteoarthritis, this typically dynamic nature of the subchondral bone plate is disrupted. Increased and imbalanced dispersion of joint forces, combined with a concentration of stresses and synovial fluid infiltration into the subchondral bone, can lead to reduced healing capacity and abnormalities within the underlying cancellous bone. These abnormalities can be identified both histologically and on magnetic resonance imaging (MRI) as bone marrow lesions (BMLs) [4-7].

The mechanism of coinciding pain associated with these BMLs is currently under debate, but has been attributed to the healing response secondary to trauma and trabecular injury and/or impaired venous drainage [8-10]. Histologically, BMLs have been shown to be focal areas of demineralization, increased fibrosis, and vascular abnormalities. These abnormalities can mimic chronic stress fractures, which may then progress to areas of focal necrosis [6,11-15]. Clinically, it is of great importance that BMLs be identified and treated, as they have been linked to increased arthritic pain and may hasten the progression of joint deterioration [5,16-18].

These potential consequences have been attributed to both improper load transmission across the affected joints and an underlying imbalance in bone metabolism––favoring bone resorption when a BML is present [19]. A direct correlation between increasing size of BMLs and increased pain in the knee was identified in a study by Felson, et al., in 2007. Patients experiencing pain were found to have a 3.31-fold greater likelihood of significant findings on MRI compared to non-painful patients with the same radiologic degree of arthrosis [20]. Additionally, Saltzman and Kijowski found that BML prevalence, depth, and cross-sectional area under arthroscopy were each directly correlated with worsening grades of corresponding articular cartilage defects [2,21].

Osteoarthritis occurring in the hip and knee joints primarily occurs as a degenerative process. However, due to histological, anatomic, and biomechanical differences in the cartilage of the ankle joint, arthritis in the ankle most commonly occurs after significant trauma [22-24]. Due to the post-traumatic presentation of ankle joint arthritis, there exists a propensity for a wider range of ages at which osteoarthritis may present in the affected ankle, which has important implications with how these patients are definitively treated. Younger and more active patients with ankle joint arthritis are less tolerant of arthrodesis or arthroplasty procedures than are their elderly and less active counterparts. As such, it stands to reason that there should be a great deal of interest in the potential for joint sparing procedures in these patients.

The Procedure

In this procedure, BMLs are triangulated using fluoroscopy, and subsequently injected and filled with flowable, biologically-compatible ceramic materials. The injected bone substitute material (BSM) then undergoes an endothermic reaction, resulting in crystallization of the BSM which affords properties similar to that of cancellous bone. This is believed to assist in supporting the trabecular structure of the bone, and to slow or even halt the pathologic processes at work. Typically, this procedure has been performed with calcium phosphate (CaP), calcium sulfate (CaS) or hydroxyapatite (HA), with CaP being the more commonly used of the three [25]. However, in terms of osteobiology, these products only offer one component of the osteobiology triad: osteoconduction. As such, these products only function as a scaffolding upon which healing may take place.

In 2016, Hood et al proposed that the two remaining osteobiologic properties, osteogenesis and osteoinduction, could be imparted via the addition of bone marrow aspirate concentrate (BMAC) to the osteoconductive materials used during the procedure [25]. It had previously been shown that osseous regeneration occurs at a faster rate with the use of a combination of BMA and osteoconductive ceramic materials, as opposed to either alone [26]. The premise behind this is that replacing the 0.9% normal saline solution (NSS)––which is typically used for rehydration of the bone substitute material––with autogenous BMAC, bone healing potential can be improved.

The addition of BMACs, the osteoconductive CaP would have the theoretical benefit of mesenchymal stem cells (MSCs), osteoprogenitor cells (OPCs), hematopoietic stem cells (HSCs), platelets, vascular endothelial growth factor (VEGF) and transforming growth factor beta (TGF-β) to assist in the reparative process [26-29]. In patients with concomitant cartilaginous defects, particulated juvenile allograft cartilage (PJAC) can be used to address the overlying cartilaginous defect after hardening of the CaP scaffold [8].

Hood et al. presented a case report for a 26 year old female with two years of recalcitrant left ankle pain after a motor vehicle accident. This patient eventually underwent the modified SCP® technique with rehydration using BMAC for a talar dome BML [25]. It was reported that the patient’s pain decreased from a preoperative VAS score of 9 to occasional 1-2/10 discomfort at 6 weeks postoperatively.

CaP with BMAC has since become a popular choice among bone and joint surgeons, though other orthobiologic combinations have also been reported with promising results: CaS with platelet-rich plasma (PRP), HA with BMAC, and HA-tri-CaP with MSC [30-32]. Subsequent studies have aimed to clarify the following: the ideal osteoinductive/osteogenic adjunct, the proper amount and consistency of adjunct, the effect on curing time and handling, and the adjunct’s effect on the scaffolding material.

In 2015, Colon et al. evaluated in vitro injectability of common commercially available bone substitute materials (BSMs). Histologically, bone marrow lesions (BMLs) demonstrate micro-trabecular damage characteristic of stress fractures [15]. For injection of materials into these microtrabeculae to be considered possible, the materials must have the ability to be injected into a highly pressurized space. Eight of the most common commercially available BSMs were tested (AccuFill® (Zimmer, Inc.), Beta-BSM™ (Zimmer, Inc.), Cerament™ (Biomet, Inc.), HydroSet™ (Styker®), Norian™ SRS (DePuy Synthes®), Pro-Dense® (Wright Medical Inc.), StrucSure™ CP (Smith & Nephew plc), Simplex™ (Stryker®)) using the polyurethane block material, while three were additionally tested in femoral condyle cadaveric bone blocks from healthy donors (AccuFill®, Beta-BSM™ and StrucSure™). The results found that although these materials are all considered injectable BSMs, only three were able to flow into the closed structure of the polyurethane block (AccuFill®, Simplex™ and StrucSure™). Additionally, AccuFill® was shown to outperform the other BSMs in several areas: the ability to flow within micro-architecture without damage from the applied force, the lowest injection force, the highest volume injected, the greatest area covered by material injected, and the ability to set without an exothermic reaction. The knowledge that these commercial calcium phosphate (CaP) products have differing properties, and understanding how this may affect different aspects of the procedure, can help inform the decision making of the surgeon.

Imaging

In 2016, Agten, et al., and Nevalainen, et al., both published papers describing diagnostic imaging related to the subchondroplasty procedure in the knee. The goal was to educate radiologists and familiarize them with expected post-procedure findings. Agten, et al., reviewed the pre- and postoperative imaging for nine patients, with the first postoperative imaging at three months post-operatively. Nevalainen, et al., discussed two knee subchondroplasty case studies. Preoperative imaging revealed that insufficiency fracture was associated with a greater amount of bone marrow edema than osteoarthritis [33].

Following the procedure, postoperative radiographs should display an increased radiodensity at the site of calcium phosphate injection, which should correlate with the locations of bone marrow edema (BME) on preoperative imaging [33,34]. CaP extravasation into soft tissues may occur along the track of the injection, which predictably mimics the appearance of heterotopic ossification. Extra-articular extravasation of calcium phosphate may resolve over time, while intra-articular leakage is a complication that should be addressed intraoperatively.

When evaluating patients, it is important to identify the cause of bone marrow edema, as this is a relatively non-specific finding, particularly on MRI. Trauma, including bone contusions, is the most common cause of positive BME findings on MRI [35]. The remaining causes of BME on MRI are transient BME syndromes (including transient osteoporosis, regional migratory osteoporosis, and complex regional pain syndrome), repetitive microtrauma and stress fractures, and non-traumatic causes such as avascular necrosis, spontaneous osteonecrosis, reactive polyarthritis, and neoplasms [2].

Classic findings of BMLs include a focal area of BME appearing as high signal intensity on T2-weighted, fat-saturated images and low signal intensity on T1-weighted, fat sensitive images. The increased signal intensity of BMLs on T2-weighted, fat-saturated MRI sequences has been suggested to be a result of increased subchondral vascularity [1]. Additionally, a low-signal-intensity line in the subchondral region of T2-weighted, fat-saturated images may be present, corresponding to impaction of the trabecular bone [35]. If present, it has been shown that a length and thickness of this line greater than 14mm and 4mm, respectively, are risk factors for lesion progression and subchondral collapse [36]. This signal should change to a decreased signal intensity on both T1-weighted fat-sensitive and T2-weighted fat-saturated images following injection of the CaP [33,34]. On fat-saturated, fluid-sensitive images there may also be a fine rim of increased signal intensity surrounding the CaP, representing surrounding edema [33,34]. It should be noted that a direct correlation between increasing BME signal intensity and more advanced cartilage degradation on MRI has also been identified [37].

Preoperative CT scan may be useful in conjunction with MRI, especially in the case of concurrent cartilage injury, as this can be difficult to assess on MRI [38,39]. Concurrent evaluation of the cartilage portion of the osteochondral unit should be considered of utmost importance, as 60% of patients with surgically confirmed chondral degeneration in the knee have been shown to have associated BMLs [21]. Additionally, both cartilage thinning and bony edema can lead to over- or underestimation of cartilage and bone damage on MRI [40]. Postoperatively on CT scan, any drill holes will be seen as a hypodense track with the surrounding hyperdense CaP [33].

Notably, the changes described correlating to post-procedure imaging have been shown to regress over time. Still, the specific time-frame is currently unclear and likely variable. In canine models, the majority of BSM has been found to be absorbed by two years postoperatively [41].

Use for OA/BML in the knee

Subchondroplasty was originally described for use in the treatment of moderate to severe osteoarthritic knee pain present for more than 2-6 months, with concomitant presence of a BML localized to the area of pain [42]. The presence of a BML in these patients is particularly concerning, as patients with knee OA compounded with a BML have a highly predictable progression to total knee arthroplasty (TKA). In fact, this occurs approximately nine times more frequently over a three year period when compared to OA in patients without a coinciding BML [4,43-45]. Previous treatment of cartilaginous defects in the knee by arthroscopic debridement alone has not been shown to yield success for patients suffering from moderate to end stage osteoarthritis, with several studies showing either no improvement or minor improvement at six months, and no improvement at two years. [4,45-48].

In 2016, a study by Cohen, et al., evaluated the combined treatment of subchondroplasty and arthroscopy in the knee in 66 patients who initially presented for TKA consultation [4]. Pain was significantly decreased and function significantly improved in all groups, including at both 6 and 24 months post-op. Notably, there was a 70% 2-year joint preservation survivorship. Patients who ultimately received TKA were significantly older and were more likely to have had a history of prior meniscectomy. A follow-up study from Brazil also noted positive results, with improvement on both VAS and knee injury and osteoarthritis outcome scores (KOOS) at 24 weeks postoperatively [14]. Longer-term outcomes of treatment with CaP in post-traumatic, impact-induced BMLs in a medial femoral condyle canine model have also shown symptomatic and functional benefits for up to two years [41].

The effect on TKA

Logically, the next question to address is whether the technique of treating BML using CaP bone substitutes affects outcomes in patients who fail this joint preserving technique and require knee replacement. It has previously been reported that the complexity of knee arthroplasty increases in patients who have had previous knee surgery, resulting in the potential for more complications and poorer outcomes [49-52]. In 2016, Yoo, et al., evaluated the effect of prior BML treatment on the complexity and outcomes of future knee arthroplasty procedures [53]. A total of 22 patients who had undergone prior arthroscopic repair of BMLs were demographically matched in a 1:2 ratio to a group of controls undergoing knee arthroplasty, either unicompartmental knee arthroplasty (UKA) or total knee arthroplasty (TKA). Patients were followed up for an average of 23.5 months (ranging from 12-52 months), with no significant differences identified between the groups. There were no cases of intra-operative UKA conversion to TKA, no differences in surgical complications or technical challenges between groups, and no cases of non-standard primary components required. Additionally, on intraoperative inspection of the CaP bone substitute, it was reported to be consistently well incorporated without signs of compromise or inconsistencies from the subchondral bone. Based on their findings, Yoo, et al., concluded that previous treatment of BMLs using CaP bone substitute did not compromise knee arthroplasty outcomes or surgical performance.

Functional/Subjective outcomes in the knee

Functional and subjective outcomes have been generally favorable following subchondroplasty. In 2018, a literature review of 8 articles and 164 total patients treated with CaP injection for BMLs in the femoral condyles or tibial plateau noted significant improvement in symptoms, few complications, and return to activity at an average of three months [42]. Of the articles reviewed, only a single paper reported a subgroup of patients who experienced poor outcomes from the procedure. Chatterjee, et al., identified an inverse relationship between the subjective postoperative Tegner-Lysholm knee scoring scale and preoperative Kellgren-Lawrence osteoarthritis grade [54]. In other words, a correlation was identified between poorer subjective outcomes and more severe preoperative osteoarthritis. Despite this, other studies have failed to report similar correlation between OA grade and outcomes. As such, future prospective studies would be valuable in confirming this finding.

Described use in the Foot and Ankle

At this time, the literature regarding treatment of BMLs using flowable calcium phosphate (CaP) has been primarily directed to cases in the knee. However, due to the need for joint-sparing procedures for ankle osteoarthritis and for the treatment of symptomatic BMLs, there has been growing interest in its application in the foot and ankle. Since subchondroplasty was first introduced into the field of foot and ankle surgery in 2015, more than six thousand foot and ankle subchondroplasty procedures have been performed [55]. The first reported subchondroplasty procedures performed in the foot and ankle were from Miller, et al., [56]. Two cases were reported, the first in a 48 year old male with complaints of chronic left ankle pain and instability, and the second in a 28 year old male with chronic ankle pain following a fibular non-union. In both cases, the patients exhibited talar BMLs on MRI that were recalcitrant to conservative treatments. Each patient underwent a subchondroplasty procedure, combined with other indicated procedures. The first patient was able to return to full activity at 12 weeks post-operatively, while the second sustained a tibial fracture due to a syncopal event at 13 weeks post-op. Miller, et al., reported minimal subjective pain in both cases at 10-month follow-up with no activity restrictions.

Shortly thereafter in 2018, Chan, et al., reported an 11-patient retrospective cohort study of symptomatic talar osteochondral defects (OCDs) treated with subchondroplasty with bone marrow aspirate concentrate (BMAC) injection [57]. In this cohort, the mean talar OCD size was 1.3 cm x 1.4 cm. All subjective outcomes improved from preoperative baseline to final one year follow-up, including visual analog pain scale and Foot and Ankle Outcome Score, with 10 out of the 11 patients reporting they would undergo the procedure again. There was a single reported complication in the cohort, with a talar neck stress fracture at bone-BSM interface after the patient had previously experienced full resolution of symptoms. All patients, except for the aforementioned complication, returned to full activity between three and nine weeks postoperatively.

Barp, et al., published two case reports, including a 25 year-old male tennis player and a 53 year-old female, treated with percutaneous injection of CaP into the 2nd metatarsal head (Frieberg’s infraction) and cuboid (stress fracture), respectively. Both patients were allowed protected weightbearing as tolerated at one week postoperatively, returned to full activity without pain at four weeks, and remained free of related complaints at final follow-up at one and three years, respectively [58].

BMLs in the foot have also been found to be associated with plantar fasciitis, specifically patients requiring surgical intervention [59]. This may have significant clinical implications. In a report by Bernhard, et al., a single case of recalcitrant plantar fasciitis was shown to have a concomitant calcaneal BML on MRI [60]. This patient was treated with repeat plantar fasciotomy and CaP injection of the BML, successfully resulting in full return to activity and pain-free follow-up at 3 and 10 months.

Complications

Perhaps due to the minimally invasive nature of the procedure, few complications of subchondroplasty have been reported in the literature. While rare, the surgeon should be aware of the following potential complications: pain secondary to overfilling with CaP, intra- or extra-articular extravasation of CaP, deep vein thrombosis of the operative limb, subsequent soft tissue or bone infection, stress fracture at the bone-BSM interface, and avascular necrosis [8,57,61].

Pain secondary to overfilling with CaP has been identified as the most common complication of the subchondroplasty procedure, and has been described clinically as a disproportionate pain which often resolves completely within 72 hours postoperatively [42]. Over-pressurization and failure to completely fill a BML have both been associated with poorer outcomes in the orthopedic knee literature and are highly preventable with increased surgeon experience [62]. A single case of osteomyelitis secondary to subchondroplasty in the medial femoral condyle was reported by Dold, et al. In their report, the authors considered that this procedure may have a predisposition for infectious complications due to direct seeding and the hydrophilic nature of CaP, which can result in prolonged wound drainage, poor healing, and eventual sinus tract formation [61]. In a series of 11 patients receiving CaP injection in the talus for painful osteochondral defects, Chan, et al., reported a single complication in a patient with a BMI of 34 kg/m² who experienced a talar neck stress fracture at the bone-BSM interface [57].

Conclusion

Overall, subchondroplasty for the treatment of BMLs has led to promising outcomes and infrequent complications. The range of potential applications of the technique is constantly expanding, with increasing use in the treatment of foot and ankle pathology. Additional studies may help clarify the potential benefits in the setting of osteoarthritis of the foot and ankle, including the procedures potential in delaying and/or preventing total ankle arthroplasty.

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Rapid learning curve with telehealth; a clinical audit at the time of ‘flattening the infection curve’ during the coronavirus (SARS Cov-2) pandemic

by Angela M Evans, PhD, FFPM RCPS (Glasg)

The Foot and Ankle Online Journal 13 (3): 7

The current coronavirus pandemic has necessitated rapid and substantial changes to clinical practice in all areas of healthcare to protect patients, healthcare professionals, and administrative staff. Whilst telehealth per se, is not new, it is new for podiatry. Consulting online is still consulting, and it is important that in the flurry to connect a platform, adapt camera angles, audio and lighting, that the ‘basics’, i.e. consent, privacy, clinical records, and sound diagnoses-based plans, remain intact.

Keywords: telehealth, coronavirus, Covid-19

ISSN 1941-6806
doi: 10.3827/faoj.2020.1303.0007

1 – Discipline of Podiatry, College of Science, Health and Engineering, La Trobe University, Victoria 3086, Australia
* – Corresponding author: angela.evans@latrobe.edu.au


It is known that telehealth services may be as effective as face-to-face interventions [1], and that telehealth has the potential to address many of the key challenges to providing healthcare in a country like Australia, with its substantial land area and widely dispersed population [2], and yet e-health to address allied health needs of people living in rural and remote Australia, appears unrealised [3]. The internet has also opened new opportunities in health care, from remote diabetes glycaemia monitoring, to smart sensors for diabetic foot ulcers, and medical device integration [4].

History-taking is (almost) everything with telehealth

Thorough history taking is always the primary information source of consultations, with an estimated 80% of information relevant to the clinical encounter may be derived from gaining a good history (5). Telehealth may further enhance the importance of history taking, given that some physical cues and the opportunity for direct clinical examination are diminished. This is fundamental to all clinical consulting, and the message here, is to spend time on an initial history, with a purposeful, methodical and reasoned manner, active listening, and simultaneously build online rapport with patients, who will be variably familiar with screen-based communication. Using a basic SOAP structured history method, it is helpful to summarise the consult verbally with patients, and ensure that all SOA-Plans are complete, understood, and shared decisions.

Whilst instrumentation treatment is not availed using telehealth, do not underestimate a patient’s capacity to imitate your on-screen methods, e.g. apply a basic taping for heel pain. Similarly, coached self-examining and provocation tests are very feasible, e.g. percussion of posterior tibial nerve at medial ankle, palpation of lateral ankle ligaments, range of motion. It is useful to demonstrate, as well as verbally guide. Foot posture, lower limb stance positions, gait and footwear, are all easily visible. In my paediatric patients, there is always a parent to assist with balance and strength tests, as well as gait-based neuromotor assessments. Exercises can be demonstrated, and then watched to fine-tune the imitation as needed. More specific details can be accessed from the listed resources.

Results

Patients of all ages to be very appreciative of availing telehealth as safe access to clinical care, and everyone has been immensely cooperative, punctual, and engaged with this novel consultation experience. Postage of items to patients reduces physical contact. If sending any form of insole or foot orthoses, an emailed/SMS photo/tracing of the sock-liner (and shoe size) enables trim-to-fit, prior to post/delivery.

Given the necessary physical distancing, it is even more important to provide clear guidelines as to what is expected, what is acceptable, and what is not (e.g. ingrown nail resection will be sore initially, should subside over 24 hours, should not have increased pain, redness, or purulence). It is also important for communication access (email, phone, follow up telehealth appointments) to be provided, as people are often anxious amid this pandemic. As always, if there are doubts or suspicions, arrange to check-in after a few days, this can be very important as the following case illustrates.

Case synopsis

Day 2 of telehealth, and 12-month review of a 14.5 year-old girl with an intellectual disability, with her mother. A patient since age seven years, with very flatfeet, which were painful before use of prefabricated foot orthoses and better shoes. She had been schooling at home, barefoot consistently, and complaining of heel pain for the last month, limiting walking. Generally, an active girl, participating in Special Olympics soccer, and swimming.

Listen: the girl described heel pain most of the time, not liking to walk at all, and finding shoes and orthoses uncomfortable (previously relieving foot pain).

Think: what is probable here? Too old for apophysitis, less active with school at home, unusual location for juvenile arthritis symptoms, possibly plantar strain given flatfeet and increased barefoot time, verruca unlikely given pain when non-weight-bearing (although worse with weight-bearing).

Look: location is plantar-mid heel pad, nil to see re: redness/swelling, the mother was asked to compare left/right for temperature (assessed as same); mother said skin ‘drier/harder’ – mother was asked to squeeze skin area – not provoked, tender adjacent), asked for close up photo re: skin striations.

Listen further: the child was asked more about the pain and responses included: it hurts at night, is mild when wakes up in the morning, it hurts to touch, and it hurts to wear shoes

List diagnostic probabilities for heel pain presentation – the main suspicion was infection, given constant pain and location, with the main concerns being osteomyelitis, sepsis, and bone tumor. Whilst one of the least frequent etiologies of pediatric heel pain, it is crucial that abnormal and unusual clinical patterns are recognized. The following ‘diary’ outlines

  • Day 1: advised to see the GP regarding pain, suggest blood tests, imaging, medication
  • Day 2: the GP prescribed antibiotics (five-day course, bd); x-rays nil, sonography detected ‘fluid area adjacent to, and larger than, the painful site’
  • Day 10: the mother called as the child was no better; advised to return to GP regarding blood tests, medication review, further imaging (e.g. MRI)
  • Day 12: GP prescribed another antibiotic (7-day course, qd)
  • Day 17: follow-up via phone, and the mother reported the child as ‘grumpy’, the heel less red, pain static; advised to return to GP (offered to liaise with GP; acknowledging that GPs are also adapting to pandemic circumstances)
  • Day 20: follow-up via phone as agreed, had seen GP who ordered another sonographic scan, with review in three days. Mother reported that the child’s foot-leg appeared more ‘swollen’ and pain remained, child ‘out of sorts’; mother concerned (and me). Discussed and advised mother to take child to children’s hospital outpatient department; provided a letter summarizing presentation and main clinical concerns, i.e., cellulitis, osteomyelitis, sepsis.
  • Day 21: Mother sent a text, thanking for referral and letter. Blood tests normal, awaiting MRI. Significantly, L foot/leg now cool to touch, hence complex pain also now considered.
  • Day 23 (today): phoned to follow-up with mother; awaiting results of MRI, and specialist appointment. Foot infection, and complex pain are current medical considerations.
  • This is clearly an unusual presentation, and one not to dismiss. The main diagnostic information was provided with the history taking via telemedicine, reducing likelihood of more usual conditions, and increasing clinical suspicion of more serious factors. The circumstances amid the novel coronavirus pandemic, coupled with a stoic child with communication limitations, required diligent follow up.

Discussion

This audit has shown that whilst patient numbers have diminished, the usual range of presentations has continued. Of the 49 patients seen in the three weeks of reduced consulting hours, 32 were telehealth consultations, and 17 were direct clinical consultations. Using telehealth, effectively enabled 29/49 patients to stay at home, and observe the Australian Government’s physical distancing advice. The 17 telehealth consultations which then required direct clinical consultations, were mostly adult patients with foot pain (12/17). Telehealth consultations were dominated by painful presentations (18/32), followed by pediatric reviews and follow up (11/32). The pediatric list (age range 2 – 16 years) included: musculoskeletal and elite sports, hypermobility (hypotonia), intellectual disability, developmental delay, Marfan syndrome, LHONs (mitochondrial disorder), CRPS (complex pain), ingrown nails, heel infection. Adult presentations (age range 16 to 71 years) have been mostly musculoskeletal, neurological, diabetes, foot pain (see Table 1).

Whilst telehealth has been enabling, it has been far from ‘business as usual’ (see Table). Overall, my case-load has dropped to less than 50%, admittedly, facilitated by encouraging non-essential visits to be deferred. My decision has been to physically see patients, only if they will be worse off without doing so, e.g. infections, pain. In this extraordinary time of a novel virus, it is important to be aware for Covid-19 foot signs; it may not be a common chilblain: https://metro.co.uk/2020/04/15/bruises-feet-sign-coronavirus-12557291/.

Given this novel experience, and that of my patients, telehealth has been invaluable, and enabled good consultation with patients of all ages, presenting with a wide range of conditions, and requiring podiatry care during this globally unprecedented time. I suspect that telehealth will remain part of clinical practice post this coronavirus pandemic.

References

  1. Speyer R, Denman D, Wilkes-Gillan S, Chen Y, Bogaardt H, Kim J, Heckathorn D, Cordier R: Effects of telehealth by allied health professionals and nurses in rural and remote areas: A systematic review and meta-analysis. J Rehabil Med 2018, 50:225–235.
  2. Bradford NK, Caffery LJ, Smith AC: Telehealth services in rural and remote Australia: a systematic review of models of care and factors influencing success and sustainability. Rural Remote Health 2016, 16:4268.
  3. Iacono T, Stagg K, Pearce N, Chambers AH: A scoping review of Australian allied health research in ehealth. BMC Health Services Research 2016:1–8.
  4. Basatneh R, Najafi B, Armstrong DG: Health Sensors, Smart Home Devices, and the Internet of Medical Things: An Opportunity for Dramatic Improvement in Care for the Lower Extremity Complications of Diabetes. J Diabetes Sci Technol 2018, 12:577–586.
  5. Paley L, Zornitski T, Cohen J, Friedman J, Kozak N, Schattner A: Utility of clinical examination in the diagnosis of emergency department patients admitted to the department of medicine of an academic hospital. Arch Intern Med., 2011, 171:1394–1396.

Further Resources

  1. Blog link: https://angelaevanspodiatrists.com.au/telehealth-a-few-tips-for-consulting-during-the-coronavirus-pandemic-april-2020/
  2. Australian Podiatry Association telehealth-podiatry: https://www.podiatry.org.au/about/news/telehealth-for-podiatrists
  3. Royal Australian College of General Practitioner telehealth guidelines: https://www1.racgp.org.au/newsgp/professional/new-guidelines-for-telehealth-consultations
 ID no. Age Gender With parent Old/New Visit purpose Assessment Diagnosis Action Attend clinic Comments
  years M/F/O Y/N O/N Review Foot Ok Review Y/N  (specific to ID)
          complaint Shoes Pain Refer  
            Gait condition postage  
            Fos      
            Lesion      
            imaging      
AGE  
ave 19.4 12 – M 22 – Y 4 – New Paed review – 8 Msk – 16 (* 13) Review 1m – 14 Y – 3
min 2 20 – F 28 – Old Pain – 18 JH/S – 9 Review 6-12m – 12 N – 29
max 71 Referred – 3 NDIS – 4 Footwear – 9
Follow up – 3 Dev delay – 5 Exercises – 19
0 – 5 6 Marfan – 1  Refer – 1
6 – 10 7 LHONs – 1  Postage – 6
11 – 15 4  17 paed CVA – 1 Orthoses (9):
16 – 30 6   CRPS – 1 prefabricated – 7 Patient total – 49
31 – 50 5   *RA – 1 bespoke – 2 Telehealth consults – 32
51 – 70 3   *Rural – 1 Clinical (2 paed) – 17
>70 1  15 adult elite sport – 3 . Diabetes risk – 1
Infection – 1 . Msk/orthotic/pain – 12
. Ingrown nails – 4
* adults

Table 1 Summary of 49 consultations, 1-24, April 2020. Telehealth consultations comprised 32/49 patient cases. Abbreviations: Msk – musculoskeletal, JH/S – joint hypermobility/syndrome (painful), NDIS – National Disability Insurance Scheme funded, Dev delay – developmental delay, Marfan – Marfan syndrome, LHONs – Leber’s Hereditary Optical Neuropathy, CVA – cerebrovascular accident or ‘stroke’, CRPS – complex regional pain syndrome, RA – rheumatoid arthritis.

 

Limb salvage for calcaneal osteomyelitis with pin to bar external fixation 

by Aaron Chokan, DPM, FACFAS1; Les P. Niehaus, DPM, FACFAS2; Joseph Albright, DPM, AACFAS3; David Bishop, DPM, AACFAS3; Frederick Garland, DPM3

The Foot and Ankle Online Journal 13 (3): 6

The prevalence of heel ulcers is as high as 18% in hospitalized patients. Due to lack of underlying muscle, protective fat pad, and constant pressure, poor tissue perfusion to the area inhibits healing. Concomitant comorbidities such as diabetes, neuropathy, and peripheral arterial disease provide added challenges to limb salvage. The incidence of surgical intervention in a diabetic patient with foot ulcers is 97%, with 71% going on to some form of amputation. Our study includes 10 patients with underlying calcaneal osteomyelitis who were treated with partial calcanectomy with primary flap closure and offloading pin to bar external fixation. Primary closure was achieved in 100% of patients with an average time of 106 days (ranging from 43 to 205 days), with no pin tract infections, revisional bone debridement, or subsequent BKA/AKA. Average follow-up time was 20.9 months (ranging 12 to 45 months).  Partial calcanectomy with offloading pin to bar fixation allows for cost-effective fixation, accelerated healing, and a satisfying functional result in true limb salvage cases.

Keywords: Limb salvage, calcaneal osteomyelitis, external fixation, infection

ISSN 1941-6806
doi: 10.3827/faoj.2020.1303.0006

1- Ohio Foot & Ankle Center, Stow, OH
2- Alliance Foot and Ankle Center, Alliance, OH
3- Aultman Alliance Community Hospital, PGY-3, Alliance, OH


Pressure ulcers to the heel are recognizably difficult to treat due to their anatomic location, and the prevalence of heel ulcers is as high as 18% in hospitalized patients [1]. The plantar and posterior aspects of the calcaneus are constant areas of pressure in both the sedentary or standing position. The lack of underlying muscle and common atrophy of its protective fat pad hinders tissue perfusion to the area. The associated diagnoses including diabetes, neuropathy, and arterial disease inhibits normal healing. Calcaneal ulcers are also accompanied by higher costs and have proven to be two to three times less likely to heal in comparison to forefoot ulcers [2]. Many of these patients are quickly consulted for a below knee amputation as a definitive treatment. Patients are able to use a prosthesis for a quick return to function, however, a BKA amputation increases energy expenditure by 25% and 33% of BKA amputees do not survive beyond two years [3,4].

Calcaneal osteomyelitis can be classified based on route of infection. The Waldvogel  classification includes hematogenous, direct or contiguous, and chronic osteomyelitis [5-7].  Hematogenous osteomyelitis results in bacteria disseminated into the bloodstream emanating from an identifiable focus of infection or developing during transient bacteremia unrelated to infection. Direct or contiguous osteomyelitis is caused by spread from adjacent sources or contact between bacteria and tissue and may be traumatically or surgically induced. Chronic osteomyelitis is the result of the coexistence of infected, nonviable tissues and an ineffective host response [8]. The attempt of preserving the calcaneus is beneficial for functionality but is much more difficult to fully eradicate the infection. The utilization of a static external fixator frame enables both stabilization and immobilization to achieve complete offloading through the final maturation stage of wound healing. The SALSAstand has been introduced for this purpose and its construct prevents any unwanted tension on skin edges as well as pressure-induced ischemia due to weight bearing [9].

Excluding case studies, there is lack of literature evaluating the combination of partial calcanectomy with primary closure and external fixation. Our study aims to provide a reproducible surgical approach to the treatment of heel ulcers with underlying calcaneal osteomyelitis. Partial calcanectomy with primary flap closure and offloading pin to bar external fixation allows for cost-effective fixation, accelerated healing, and a satisfying functional result in true limb salvage cases.

Patients and Methods

Patients diagnosed with osteomyelitis of the calcaneus were treated with radical resection of the calcaneus with primarily closure and with utilization of SALSAstand pin to bar external fixation. All patients were treated by a single surgical attending from January 2016 to May 2019.  The inclusion criteria included patients with type 1 or 2 diabetes mellitus, those with at least a Wagner stage 3 ulceration to the heel, patients who had been diagnosed with osteomyelitis of the calcaneus with MRI advanced imaging or white blood labelled indium scans if patient was unable to have MRI, over 20% involvement of the calcaneus, and a minimum follow up of 6 months after achievement of primarily closure.

In our experience these patients had multiple co-morbidities requiring a multi-specialty medical approach. Consults for infectious disease, cardiology, vascular, endocrinology, anesthesiology, physical therapy and internal medicine were used for safety and to increase efficacy of the operative procedure.  Additionally, patient demographics were examined.

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Figure 1 A – Plantar lateral wound probing directly to calcaneus. B – Posterosuperior Flap from achilles area rotated plantarly. C – Sutured flap over deficit, knots tied outside flap.

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Figure 2 Planned resection of calcaneus with section taken. 0.5 cm margin using MRI guided resection.

Risk factors included obesity, hemoglobin A1C, peripheral vascular disease, history of tobacco use, and end stage renal disease. Other significant findings evaluated included history of attempted surgical treatment, number of operations required, and number of re-hospitalizations following the initial procedure.

Surgical Technique

The patients were placed under general anesthesia and were initially placed in the prone position. Thigh tourniquets were used unless a patient had recently undergone vascular intervention. A combination of two incisional approaches were utilized based on the location of the heel ulcer. Straight elliptical excisions for plantar wounds and a posterosuperior flap for posterior calcaneal ulcers (Figure 1). Full-thickness incisions were created with meticulous dissection to not harm the skin flaps. Once the flap was freed from attachments and the primary wound excised, the Achilles tendon was completely resected at its insertion. Utilizing a large saw, the calcaneus was resected from proximal superior to distal inferior in an oblique fashion (Figure 2).

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Figure 3 SALSAstand method for offloading. Two half pins into tibia and two half pins into midfoot.

A margin of 0.5 cm of bone was resected from the involved bone via diagnostic advanced imaging. All the rough edges of bone were then smoothed down. Portions of the bone were sent to both microbiology and pathology. A combination of 2-0 Prolene vertical mattress technique and staples were utilized to ensure closure.

After proper closure of the flap, tourniquets were deflated and then the patient was flipped to supine position with care to prevent shearing forces or pressure on the flap. A pin to bar external fixation frame was then applied to the leg for offloading of the posterior flap. In safe zone 4, just distal to the midshaft of the tibia, using a parallel guide and clamp, two 5-0 half pins were placed into the tibial crest [10]. Two 45 degree elbows were placed in the tibial clamp and 2 bar frames were then extended toward the level of the forefoot and the heel.  Two more 5-0 half pins were then inserted medially and laterally separately into the navicular and the cuboid to help construct the offloading frame. Fluoroscopy was employed to ensure placement. Pin to bar mechanism was then utilized to connect the two bars from the elbow to the midfoot pins as well as a large offloading “U” frame that went posterior around the heel (Figure 3). The “U” frame kept the patient from externally rotating the leg and forcing any pressure on the calcaneal flap. All the pin sites were covered with xeroform and dry dressing was applied to the leg.

All patients were still placed on intravenous antibiotics for 6 week depending on microbiology results. All patients were kept non-weight bearing to the operative leg until closure of the surgical wound. After complete healing, the external fixation device was removed and the patient was casted for custom solid AFO.

Results

A total of 12 patients were identified. Two patients were excluded due to one inadequate follow-up and one patient who was deceased before adequate follow-up, leaving 10 patients that met the inclusion criteria. Of those who met the inclusion criteria, 30% (3/10) were active tobacco smokers, 50% (5/10) were diagnosed with ESRD, 70% (7/10) had a history of PVD, previous surgical intervention occurred in 90% (9/10),  average BMI among the 10 patients was 31 and average hemoglobin A1C was 7.5%.  Demographic and medical history is seen in Table 1.

Patient Characteristics Median or no. (percentage)
Patient Age 64
Gender
Male 7
Female 3
BMI 31.3
HbA1C 7.6
Diabetes Mellitus 10 (100%)
ESRD 5 (50%)
PVD 7 (70%)
Current Tobacco Use 3 (30%)
Previous Surgical Intervention 9 (90%)
Follow up (months)
Mean 15.9
Range 7 to 42

Table 1 Patient Demographics (N=10).

Complication n
Dehiscence 3 (30%)
Flap necrosis 2 (20%)
Recurrent ulcer 2 (20%)

Table 2 Complications.

Variable
Wound Size 6.4 x 5.6 cm (35.8 cm2)
Average duration of wound 38 weeks (4 to 204)
Calcaneus resection size 122 cm3
Time to healing 106 days (43 to 205)
Time in external fixation 41 days (15 to 77)

Table 3 Pre and post operative results.

Mean wound size preoperatively was 6.4 cm x 5.6 cm (35.8 cm2), mean size of calcaneal bone resected was 6.6 cm x 4.9 cm x 3.6 cm (116.4 cm3). Average time to primary closure was 106 days (ranging 43 to 205 days), average days in external fixation devices was 41 days (ranging 15 to 77 days), and number of operating room visits following initial procedure was 1.5 visits (ranging from 1 to 3 visits). Complications encountered included partial wound dehiscence in 3/10 patients, flap necrosis in 2/10 patients, and re-ulceration in 2/10 patients.

Re-ulceration occurred at an average of 5 weeks post op (ranging 4 weeks to 6 weeks). Due to complications, subsequent adjunctive grafting occurred in 6 patients to aid in healing and 2 patients required rehospitalization. No pin tract infection, revisional bone debridement, or subsequent BKA/AKA was observed. Average follow up time was 20.9 months (ranging 12 to 45 months).

Discussion

A similar study by Akkurt, et al, utilized MRI guided debridement with application of Ilizarov external fixation for patients with pedal ulcers and concomitant calcaneal osteomyelitis.[11] The mean size of calcaneal osteomyelitis was 8.73 cm3 (range 3–18 cm3) and the authors advocated for a preoperative MRI-guided resection plus a maximum 0.5 cm of resection in depth as far as healthy osseous tissue was sufficient in all patients.  The authors recommendations is the same guideline we utilized for our resection. The wounds healed in 18 of the 23 patients (78%), partial recovery occurred and subsequent flap operation was performed in three patients (13%), and below-the-knee amputation was performed in two patients (9%). Pin tract infections were the most common complication seen in 16 patients (69.5%).[11]  Our study showed complete healing in 100% of patients with no below-knee-amputations or pin tract infection as a result. Pin tract infection was a common complication possibly due to the complexity of the frames in the study by Akkurt, et al.[11] We hypothesize utilizing a four half-pin fixation construct decreases the chance for pin tract infection and subsequent amputation. There is less chance of loosening and pistoning without smooth wire fixation. Bollinger, et al., performed partial calcanectomies and evaluated the functional status of their patients. Thirteen of the 22 patients had confirmed osteomyelitis. Eighteen patients were available for follow-up. Twelve had delayed wound healing that required either a split thickness skin graft or serial debridements.  Nine patients had diabetes and all had delayed wound healing with an average follow up time of 27 months. They found that ulcers larger than 7 cm would not allow for a tension-free closure. They also recommended casting in plantar flexion for a minimum of 4 weeks post-operative. This study resulted in 100%  satisfaction rates of its subjects. However over 50% had delayed wound healing with the need of additional surgical treatment [12]. Our experience saw similar results in delayed healing with subsequent grafting at 60%. A combination of biologics and split thickness skin grafts were utilized depending on size of surgical wound. With our average wound size of 35.8 cm2, we found that even with larger deficits, utilizing a rotational flap allowed for tension free initial closure of skin.

Vac therapy is also a conservative option to attempt and close these long standing ulcers. However, the frequency of dressing changes, time needed, and prolonged non-weight bearing make the negative pressure therapy a very involved task. Nather et al., looked at wound vac therapy for diabetic foot wounds in 11 patients and administered VAC therapy for an average of 23.3 ± 10.3 days.  Initial wound sizes ranged from 6.9 to 124.0 cm2 and post therapy had an average reduction of 10.1 cm2 with an average reduction of 24.9%, which was not statistically significant [13]. The use of wound vac therapy alone in diabetics cost an average of $13,262 for a 12 week therapy course [14]. Conservative treatment through vac therapy, debridements, and serial grafting increases both cost to patient and chance of infection. Our patient population only required 1.5 visits to the OR after the initial procedure where at least one of the visits involved was to remove the external fixation device. The average healing time after calcanectomy and primary closure was about 15 weeks where the average duration of the wound being present was 38 weeks. This procedure allows for complete eradication of infected bone and tissue, properly offloading, and primary tissue healing for practical and functional results.

Dalla Paola, et al., used a combination of the treatments discussed. They enrolled 18 consecutive patients with large heel ulcers complicated by osteomyelitis. Treatment was performed in a two-step manner, first including MRI guided resection of  the infected calcaneus, application of circular external fixator, and negative pressure wound therapy  with dermal substitute. The second stage included application of split thickness skin graft over the wound. Complete healing was achieved in all patients with mean time of 69+/- 64 days. Total time for maintenance of the circular frame was 78.2 +/- 31.5 days [15]. Another surgical alternative is the use of myofascial flaps to cover soft tissue deficits in the heel. The robust nature of the muscle belly aids in bone healing and increased antibiotic deliverance to the site of infection. Abductor hallucis, reverse sural artery, and saphenous flaps are all viable options depending on the size of muscle needed for coverage. However, these surgical procedures are technically demanding and require attentive wound care. Increased risk of flap breakdown may be attributed to the high pressure area they weren’t designed for.  Flap rejection is cited from 5% to 25% while diabetics have an increased rate of necrosis at 32% [16].

References

  1. Cuddigan J, Berlowitz DR, Ayello EA. Pressure ulcers in America: prevalence, incidence, and implica- tions for the future. Adv Skin Wound Care 2001; 14:208-15.
  2. Jacobs TS, Kerstein MD. Is there a difference in outcome of heel ulcers in diabetic patients and non-diabetic patients? Wounds 2000; 12(4):96-101.
  3. Cuccurullo, Sara J. Physical Medicine and Rehabilitation Board Review. 2nd ed. New York: Demos Medical, 2010.
  4. Pinzur MS. Amputation level selection in the diabetic foot. Clin Orthop. 1993; 296:68-70
  5. Waldvogel FA, Medoff G, Swartz MN. Osteomyelitis: a review of clinical features, therapeutic considerations and unusual aspects (first of three parts) N Engl J Med. 1970 Jan 22;282(4):198–206.
  6. Waldvogel FA, Medoff G, Swartz MN. Osteomyelitis: a review of clinical features, therapeutic considerations and unusual aspects (Second of Three Parts) N Engl J Med. 1970 Jan 29;282(5):260–266.
  7. Waldvogel FA, Medoff G, Swartz MN. Osteomyelitis: a review of clinical features, therapeutic considerations and unusual aspects (Third of Three Parts) N Engl J Med. 1970 Feb 5;282(6):316–22.
  8. Ciampolini J, Harding KG. Pathophysiology of chronic bacterial osteomyelitis. Why do antibiotics fail so often? Postgrad Med J. 2000 Aug; 76(898):479-83.
  9. Clark J, Mills JL, Armstrong DG. A method of external fixation to offload and protect the foot following reconstruction in high-risk patients: the SALSAstand. Eplasty. 2009;9:e21. Published 2009 Jun 4.
  10. Cooper P, Polysois V, Zgonis T. External Fixators Of The Foot And Ankle. Wolters Kluwer Health, Chapter 2. Published 2015.
  11. Akkurta MO, Ismail  D, Öznur A. Partial  calcanectomy  and Ilizarov external fixation may reduce amputation need in severe diabetic calcaneal ulcers. Diabetic Foot Ankle, 2017 8(1), 1264699
  12. Bollinger M, Thordarson DB. Partial calcanectomy: an alternative to below knee amputation. Foot Ankle Int. 2002;23(10):927-932.
  13. Nather A, Chionh SB, Han YY, Chan PL, Nambiar A. Effectiveness of Vacuum-assisted Closure (VAC) Therapy in the Healing of Chronic Diabetic Foot Ulcers. Ann Acad Med Singapore.  2010;39:353–8
  14. Driver V, Blume P. Evaluation of Wound Care and Health-Care Use Costs in Patients with Diabetic Foot Ulcers Treated with Negative Pressure Wound Therapy versus Advanced Moist Wound Therapy. Journal of the American Podiatric Medical Association: 2014;104(2):147-153.
  15. Dalla Paola L, Brocco E, Ceccacci T, Ninkovic S, Sorgentone S, Marinescu MG, Volpe A. Limb salvage in Charcot foot and ankle osteomyelitis: combined use single stage/double stage of arthrodesis and external fixation. Foot Ankle Int 30:1065–1070, 2009.
  16. Germann G. Invited discussion: the simple and effective choice for treatment of chronic calcaneal osteomyelitis: neurocutaneous flaps. Plast Reconstr Surg 2003; 111:761–2.

Intraosseous ganglion of the third metatarsal: A case report

by Bryn Rowe DPM PGY21*, Jeffrey Christensen DPM FACFAS2, Daniel Lowinger DPM FACFAS3

The Foot and Ankle Online Journal 13 (3): 5

Intraosseous ganglia are benign, non-neoplastic lesions of bones that are histologically similar to their soft tissue equivalents. They most often occur in the femoral head, proximal tibia, and carpal bones. A 70-year-old female presented with a complaint of pain to the right dorsal forefoot. The patient was diagnosed with a third metatarsal intraosseous ganglion based on multiple imaging modalities and confirmed by histopathology. A case report and review of the literature is presented.

Keywords: bone cyst, ganglion, subchondral cyst, geode

ISSN 1941-6806
doi: 10.3827/faoj.2020.1303.0005

1 – Swedish Medical Center Resident – Swedish Medical Center, Seattle, WA, PGY-2
2 – Attending Physician – Swedish Medical Center, Seattle, WA
3 – Attending Physician – Swedish Medical Center, Seattle, WA
* – Corresponding author- Bryn.Rowe@swedish.org


Intraosseous ganglia are benign cystic lesions that typically occur in long bones. These lesions should be an included differential for any cystic osteolytic lesion seen in the foot or ankle. Intraosseous ganglia can be painful and have the potential to cause pathologic fractures. The standard treatment includes curettage with bone grafting, with or without internal fixation.

The etiology of intraosseous ganglion is unknown and multiple theories exist. They are most widely accepted to be primary lesions and may arise de novo with no imaging or pathological evidence of degenerative process in neighboring joints [1]. Differential diagnoses for intraosseous ganglia include any well circumscribed cystic lesion with marginal sclerosis located in the subchondral bone adjacent to a joint, such as a subchondral bone cyst, giant cell tumor, enchondroma, chondrosarcoma/blastoma, Brodie’s abscess, and fibrous dysplasia among others [3]. Intraosseous ganglia can be differentiated from other potential diagnoses based on the age of the patient as well as characteristics seen on radiographs and other imaging modalities. Diagnosis, however, can only be confirmed by histopathological analysis.

Intraosseous ganglia appear as well-defined lytic lesions with marginal sclerosis on radiographs. They can regularly be distinguished as they often do not often exhibit calcifications, cortical expansion, or cortical destruction. They can be unilocular or multilocular. Magnetic resonance imaging (MRI) of these lesions shows low intensity signal in T1 and high intensity signal in T2. Computed Tomography (CT) imaging is useful to determine intra articular involvement or pathologic fracture not easily identified on radiographs [7].

Conservative treatment includes non-operative observation and symptom relief. Sclerosing injections have been performed however their effectiveness have not been reported in literature. The gold standard treatment is surgical curettage of the cyst and surrounding zone of sclerosis with or without bone grafting. The deficit is often packed with bone cement, autograft, or allograft either with or without internal fixation [5]. Pathologic fracture is a reported complication caused by these lesions. Arthroscopy has also been performed but is more commonly done in the wrist. There are no current studies comparing the outcomes between arthroscopy versus open curettage. Recurrence rate for intraosseous ganglion after surgical curettage is around 6.1%, however there is a variable overall reported recurrence rates ranging from 7-43% in the literature [8]. The recurrence is thought to not necessarily be from inadequate excision but rather from connective tissue metaplasia after surgery [1].

Grossly, intact cysts are round or oval in shape and appear opalescent and blue-grey in color. The walls of the cyst do not contain any epithelial or synovial lining but are rich in collagenous fibers. The unroofed cyst is made up of a largely fibrous membrane containing mucinous material. Acid mucopolysaccharide storage within the cyst leads to the myxoid degeneration seen within the ganglion wall [2]. Intraosseous and extraosseous ganglion are indistinguishable histologically. The diagnosis is confirmed by histopathological analysis.

Intraosseous ganglia are benign, non-neoplastic lesions of bones that are histologically similar to their soft tissue equivalents. They most often occur in the femoral head, proximal tibia, and carpal bones. A 70-year-old female presented with a complaint of pain to the right dorsal forefoot. The patient was diagnosed with a third metatarsal intraosseous ganglion based on multiple imaging modalities and confirmed by histopathology. A review of the literature found only one reported case of an intraosseous ganglion in the third metatarsal and two other reports occurring in the first metatarsal [2, 6]. She was treated operatively by curettage with bone allograft with no recurrence at one year follow-up. Of note, the patient’s postoperative course was complicated by delayed consolidation of the bone allograft with subsequent malunion of the third metatarsal base with dorsal angulation.

Figure 1-3 AP, MO, and lateral radiographs of the right foot with subtle marginal sclerosis noted to the base of the third metatarsal.

Case Report

The patient is a 70-year-old female who presented with chief complaint of pain with activity on the central and dorsal aspect of her right foot. The pain had been present for two years with gradual onset. There was no history of trauma. Of note, the patient also complained of first metatarsal phalangeal joint pain with a progressively worsening bunion deformity causing shoe irritation. On physical exam there was a moderate hallux valgus deformity and mild tenderness to palpation of the dorsal aspect of the 3rd metatarsal base. Radiographs of the right foot demonstrated a long 1st metatarsal, mild narrowing of the 1st metatarsophalangeal joint, and slight increase of the 1st and 2nd intermetatarsal angle.

Figure 4 Sagittal T2-weighted MR imaging with high signal intensity in the base of the third metatarsal extending distally within the metatarsal shaft.

There was a subtle discrete radiolucent area in the base of 3rd metatarsal accompanied by marginal sclerosis laterally and proximally, most pronounced on the medial oblique view (Figures 1-3).

An MRI was ordered to further assess the irregular sclerotic area as well as to rule out potential stress fracture or arthropathy. T2-weighted MR images demonstrated high intraosseous signal intensity in the proximal third metatarsal base extending to the articular surface of the third tarsometatarsal joint as well as marrow edema extending distally in the metatarsal shaft (Figure 4). The area measured 1.8 x 1.1 cm. There was no cortical irregularity, calcification, or periosteal reaction. A CT scan demonstrated a unilocular mixed lytic and lucent intramedullary lesion at the proximal aspect of the third metatarsal with no visualized tarsometatarsal joint involvement (Figs. 5-6).

Operative Technique

The patient was placed under general anesthesia in a supine position. A linear incision was made over the third tarsometatarsal joint after the position of the bone lesion was triangulated with fluoroscopy. Sharp dissection was carried down to the extensor tendons which were retracted. The periosteum over the base of the third metatarsal was incised longitudinally and reflected medially and laterally. A dorsal rectangular shaped window was created in the cortex of the base of the third metatarsal (Figure 7).

Figures 5-6 Sagittal and coronal CT imaging demonstrating the third metatarsal lesion with intact cortex present at the 3rd metatarsal-cuneiform joint with no apparent intraarticular extension.

Figure 7 Intraoperative image of the third metatarsal base after the dorsal cortical window was removed, revealing a soft gray mass and cystic cavity.

A well-encapsulated gelatinous gray tissue mass was visualized in the marrow cavity and excised completely with a curette. There was no apparent intra-articular involvement or extension in the tarsometatarsal joint noted. The cortical wall was curetted and bone allograft was packed into the deficit. The cortical window was press fit into the graft and positioned in its original position. The graft was not fixated as it was stable after positioning. The gross pathology report described the mass as a 1.6 x 0.8 x 0.7 cm disrupted pale, tan-red cyst, containing mucoid material. The final diagnosis was benign intraosseous ganglion.

Discussion

Intraosseous ganglions are benign non-neoplastic bone lesions that are histologically similar to their soft tissue homologues. They are most commonly found in the epiphyseal-metaphyseal area of long bones with higher occurrences in the medial malleolus, femoral head, and carpal bones. There are few case reports in literature describing intraosseous ganglia of metatarsals, with only one case being reported in the third metatarsal and two additional cases reported in the first metatarsal [1,6]. This may be due to underreporting and confusing nomenclature leading to this lesion being named under a different pathologic entity. Peak incidence for both males and females in the fourth or fifth decade. They infrequently occur in skeletally immature individuals or the elderly, although cases have been reported in the literature [5]. Communication with surrounding joints and extension into the soft tissues are frequently reported, however, there are variable occurrence rates in literature due to differences in which imaging modality was used. In a study of patients with confirmed intraosseous ganglia, there was soft tissue extension present in 38% and intra-articular involvement present in 17% of the 29 patients who underwent MRI [7]. These lesions were evaluated using T1-weighted, T2-weighted, and short tau inversion recovery (STIR) sequences. In a similar study of 17 patients, there was 24% soft tissue extensions present. The authors also noted that among their patients, 12% had associated osteoarthritis and 18% had pathological fracture [4]. Preventing potential pathologic fracture in patients with intraosseous ganglion is an important rationale for surgical treatment

Although benign and uncommon, intraosseous ganglia should be considered in the potential diagnosis of any bony subchondral cystic lesion in the foot or ankle. Without proper clinical observation and potential treatment, intraosseous ganglia have the potential to cause significant pain, cause pathologic fracture, and exhibit secondary pathological effects by displacing surrounding soft tissues. Ideally curettage with grafting would be performed for both treatment and diagnostic purposes.

References

  1. Feldman F, Johnston A. Intraosseous ganglion. Am J Roentgenol Radium Ther Nucl Med. 1973;118(2):328-43.
  2. Helwig, U., Lang, S., Baczynski, M., & Windhager, R. (1994) The intraosseous ganglion. Archives of Orthopaedic and Trauma Surgery, 114: 14-17
  3. Murff, R. & Ashry, H. (1994) Intraosseous ganglia of the foot. The Journal of Foot and Ankle Surgery, 33 (4): 396-401.
  4. Sakamoto, A., Oda, Y., & Iwamoto, Y. (2013) Intraosseous ganglia: a series of 17 treated cases. Biomed Research International, 2013, Article ID 462730, 4 pages, http://dx.doi.org/10.1155/2013/462730
  5. Sedeek, S., Choudry, Q., & Garg, S. (2014) Intraosseous ganglion of the distal tibia: clinical, radiological, and operative management. Case Reports in Orthopedics, 2015, Article ID 759257, 4 pages, https://doi.org/10.1155/2015/759257.
  6. Wakabayashi, I., Okada, K., Hashimkoto, M., & Sageshima, M. (1999) Intraosseous ganglion of the metatarsal bone. Journal of Computer Assisted Tomography, 23 (5): 727-729. doi: 10.1097/00004728199909000-00017
  7. Williams, H., Davies, A., Allen, G., Evans, N., & Mangham, D. (2004) Imaging features of intraosseous ganglia: a report of 45 cases. European Radiology, 14: 1761-1769
  8. Yu, K., Shao, X., Tian, D., Bai, J., Zhang, B., & Zhang, Y. (2016) Therapeutic effect of bone cement injection in the treatment of intraosseous ganglion of the carpal bones. Experimental and Therapeutic Medicine, 12: 1537-1541

 

Emphysematous osteomyelitis of the foot: A case report

by Igor Dukarevich, DPM1*; Victoria Chirman, DPM2; Mahin Siddiqui, DPM3

The Foot and Ankle Online Journal 13 (3): 4

Emphysematous osteomyelitis is a rare life-threatening infection requiring early recognition and immediate surgical intervention. The condition is usually caused by anaerobes, gram negative rods, or is polymicrobial. It presents in immunocompromised hosts with comorbidities such as diabetes mellitus, thalassemia major, sickle cell disease, alcohol abuse, and exogenous immunosuppression. This infection can be either of contiguous or hematogenous spread, and has been previously reported in both the axial and the appendicular skeleton. Intraosseous gas is frequently overlooked on plain radiographs but is easily diagnosed by CT scan. We describe a case of direct extension emphysematous osteomyelitis involving the foot of a 52-year-old male with poorly controlled diabetes mellitus type 2. We emphasize the need for a high index of suspicion, early diagnosis via CT scan, and immediate surgical intervention. We also underscore the utility of the Symes amputation, used in our case as an alternative to transtibial amputation for diabetic limb salvage.

Keywords: emphysematous, foot, gas, intraosseous, osteomyelitis

ISSN 1941-6806
doi: 10.3827/faoj.2020.1303.0004

1 – Podiatry Residency Director, Loretto Hospital, 645 S Central Ave, Chicago, IL 60644
2 – Podiatry Resident, Loretto Hospital, 645 S Central Ave, Chicago, IL 60644
3 – Podiatry Resident, Loretto Hospital, 645 S Central Ave, Chicago, IL 60644
* – Corresponding author: dukarevichi@gmail.com


Emphysematous osteomyelitis is a rarely-reported condition, previously not described in the podiatric literature.  It was first noted by Ram PC, et al., in 1981, when a CT scan demonstrated gas within the medullary cavity of the involved bone [1]. In their case series, all plain radiographs were negative and there was no clinical suspicion of the severity of the infection until a CT scan was obtained. The CT scan findings significantly changed the management of the patients.

Since the initial report, the majority of the cases described have been limited to the axial skeleton with suspected hematogenous spread [2]. A hand-full of cases have been described in the appendicular skeleton, both of contiguous and hematogenous extension, with emphysematous osteomyelitis presenting in the femur, the tibia, and the foot. In the majority of the reported cases, the patients have multiple comorbidities including diabetes mellitus, use of immunosuppressive medication, malignancy, alcohol abuse, thalassemia major, or sickle cell disease [2-4]. In many cases, the X-rays were negative for soft tissue gas and the diagnosis was made only with prompt CT imaging [1].  We report a case of contiguous spread emphysematous osteomyelitis in the foot, emphasizing the need for a high-index of suspicion, prompt advanced imaging, and aggressive treatment for this rare but life-threatening condition.

Case Report

A 52-year-old African American male, with a past medical history of polysubstance abuse, poorly controlled diabetes mellitus type 2, iron deficient anemia, seizure disorder, peripheral neuropathy, history of chronic ulcerations, had underwent treatment in our facility from 12/2018 through 01/2019 for emphysematous osteomyelitis of the right foot.  The patient presented to the emergency department on December 6, 2018 with a chief complaint of right foot pain and swelling.

Figure 1 Clinical appearance.

He previously underwent a partial right first ray amputation at a different hospital in 08/2018, with delayed healing of the surgical wound.  The patient was unable to provide a detailed history of his condition at the time of the admission. The patient had no known drug allergies. Family history was non-contributory. Review of systems was unremarkable, with exception of the chief complaint.

On examination, the patient was noted to be a well-nourished, well-developed male in no apparent distress. The vital signs were stable, with the exception of a low-grade fever at 99.4 degrees Fahrenheit and a pulse of 126 bpm.  Significant findings on the physical exam included moderate edema and erythema to the right foot. A partially healed amputation site of the first ray of the right foot was appreciated with a necrotic ulceration on the dorsum of the foot probing directly to bone and tendinous structures. Mild serous drainage was noted from the wound, but no obvious fluctuance, purulence, or soft tissue crepitus was appreciated (Figure 1).  Pedal pulses were faintly palpable bilaterally with capillary refill times less than four seconds to the remaining digits of the right foot. Neurologically, light touch and sharp/dull sensation was diminished distal to the mid-leg level of bilateral lower extremities.

Radiographs of the right foot were obtained and were suggestive of osteomyelitis of the second metatarsal base, however no evidence of significant osseous destruction or soft tissue gas was noted. Vascular calcifications were appreciated. (Figure 2). Significant neutrophilic leukocytosis was noted with WBC at 14.4. Blood cultures were positive for Strep. Pyogenes.  Lactic acid was 2.1.

Figure 2 Radiographs of the right foot, suggestive of osteomyelitis of the second metatarsal base.

Figure 3 CT of foot, showing small foci of subcutaneous gas were also noted in the tissues.

The last HbA1C was 13.7%. Albumin was 1.4. Deep wound cultures were obtained at the time of admission. The patient was started on IV fluids and Vancomycin and transferred to the hospital loor for further evaluation and management. Infectious disease and a podiatry consult was requested.

Infectious disease and podiatry recommended the addition of piperacillin/tazobactam and metronidazole to broaden the antibiotic coverage. A CT scan of the right foot was obtained. The CT scan demonstrated multiple foci of intraosseous gas in the midfoot including navicular, cuboid and cuneiform bones, as well as the bases of second, third, fourth, fifth metatarsals. Small foci of subcutaneous gas were also noted in the tissues (Figure 3). The findings were consistent with the “pumice stone” pattern previously reported by Small JE, et al., and diagnostic for emphysematous osteomyelitis.

Given the findings, an emergent incision and drainage of the right foot with a guillotine amputation at the Chopart level was performed. Clearance fragments were obtained from the distal talus and the calcaneus. Following surgical intervention, the patient continued to improve with resolution of leukocytosis and fever. Blood cultures were negative.  Wound culture results revealed growth of Staphylococcus Aureus, Klebsiella, Enterobacter Aerogenes, and Streptococcus Pyogenes Group A. Empiric antibiotic therapy was narrowed to clindamycin and penicillin, per sensitivity report and infectious disease recommendations.

Figure 4 Radiographs after Chopart level amputation.

Arterial doppler studies of the lower extremities confirmed no significant peripheral arterial disease of the right lower extremity with biphasic waveforms throughout. Follow-up radiographs and CT scan demonstrated no proximal spread of emphysematous osteomyelitis (Figure 4). Pathology analysis of the resected foot displayed skin and subcutaneous tissue showing necrosis and gangrene; bone with underlying acute and chronic osteomyelitis. Clearance fragments from the distal talus and calcaneus were negative for osteomyelitis.

In the subsequent days revision of the amputation and delayed primary closure was performed. Due to fair right lower extremity arterial perfusion, a decision was made to attempt distal limb salvage with a Syme’s amputation, as opposed to a below-the-knee amputation. A Syme’s amputation was performed per standard technique and the patient tolerated the procedure well (Figure 5). The remaining hospitalization course was uneventful and the amputation flap was healing well. The patient was discharged to an extended care facility. The patient missed his first two postoperative appointments and was seen in the outpatient clinic for follow-up about one month after the surgery.  The patient was noted to have partial dehiscence and necrosis of the lateral one-third of the incision with the remainder of the incision healing well. The patient was readmitted for IV antibiotic therapy, vascular evaluation, and debridement.   An angiogram of the right lower extremity confirmed no significant disease in the bilateral common internal and external iliac arteries and there was noted to be a two-vessel runoff to the foot without any significant disease. The patient underwent further debridement and wound care. The patient had successful healing of the Syme’s amputation stump via secondary intention without further setbacks.

Figure 5 Radiographs after Syme’s level amputation.

Discussion

Emphysematous osteomyelitis is a rare but potentially life-threatening condition [1-5]. About thirty cases have been described thus far in literature; the majority presenting with predominantly hematogenous spread in the spine, pelvis, and hip [1-5]. Only three cases have been previously described affecting the foot [2-4].

Our case of emphysematous osteomyelitis in the foot was similar in presentation to those previously reported by Mautone et al and Abdelbaki et al [3-4].  The spread of the infection was contiguous from a chronic ulceration persisting from delayed healing of a partial foot amputation. Khanduri et al reported the only case of hematogenous spread to the foot, with the source likely being a urinary tract infection [2].

As in the previously reported cases of emphysematous osteomyelitis of the foot, our patient was immunocompromised with multiple comorbidities. Clinical findings and X-rays were fairly benign and underestimated the extent of the infection. A prompt CT scan allowed for accurate diagnosis and appropriate emergent treatment. The finding of intraosseous “pumice stone” pattern of gas formation on CT scan was diagnostic for emphysematous osteomyelitis [5]. The CT scan allowed for clear visualization of the extent of the infection and helped to guide the level of the amputation.

As in other reported cases of emphysematous osteomyelitis, the infection in our case was polymicrobial. As such, empiric antibiotic therapy should be broad-spectrum and should include anaerobic coverage, with later narrowing based on culture and sensitivity results. As with gas gangrene of the soft tissues, the primary treatment for emphysematous osteomyelitis is emergent surgical debridement with amputation of all infected structures. Input and intervention from internal medicine, interventional cardiology, and infectious disease specialists is also critical in the successful management.

As with other diabetic foot infections, the long-term treatment goal should be distal limb salvage with rapid return to functional activity [7]. Previous studies have demonstrated the utility of the Syme’s amputation, with advantage of a more natural gait resulting in decreased metabolic expenditure and cardiac stress [6-7]. The literature also suggests lower morbidity and mortality rates after a Syme’s amputation in comparison to transtibial amputations [6-7]. We believe that it remains a viable alternative for limb salvage.

We describe a case of emphysematous osteomyelitis, previously not reported in the podiatric literature, managed with a Syme’s amputation. We emphasize the need for a high-index of suspicion in immunocompromised patients with long-standing post-surgical ulcerations, as well as early use of advanced imaging. The use of a CT scan helps to determine the extent of infection and the level of amputation. We also note that the Syme’s amputation remains an alternative to transtibial amputations for distal limb preservation. Severe diabetic foot infections such as emphysematous osteomyelitis, are a challenging entity, requiring prompt intervention by a multidisciplinary team to achieve a successful outcome.

References

  1. PC Ram, S Martinez, M Korobkin, RS Breiman, HR Gallis, JM Harrelson. CT detection of intraosseous gas: a new sign of osteomyelitis. AJR Am J Roentgenol, 137 (1981), pp. 721-723
  2. Sachin Khanduri, Meenu Singh, Aakshit Goyal, Simran Singh. Emphysematous osteomyelitis: Report of two cases and review of literature. Indian Journal of Radiology and Imaging. 2018;(1):78.
  3. Mautone M, Gray J, Naidoo P. A Case of Emphysematous Osteomyelitis of the Midfoot: Imaging Findings and Review of the Literature. Case Reports in Radiology. January 2014:1-4.
  4. Abdelbaki A, Bhatt N, Gupta N, Li S, Abdelbaki S, Kumar Y. Emphysematous osteomyelitis of the forefoot. Proceedings (Baylor University Medical Center). 2017;31(1):100-101.
  5. Small JE, Chea P, Shah N, Small KM. Diagnostic Features of Emphysematous Osteomyelitis. Curr Probl Diagn Radiol. 2018 Jun 1
  6. Pinzur MS. Amputation level selection in the diabetic foot. Clin Orthop. 1993; 296:68-70.
  7. Yu G, Meszaros A, Schinke T. Syme’s amputation: A retrospective review of 10 cases. Podiatry Institute Update, Chapter 14, Podiatry Institute, Tucker, GA, 2005, pp. 78–88.

 

 

Ankle arthrodiastasis in conjunction with treatment for acute ankle trauma

by Nunzio Misseri, DPM¹; Hayley Iosue, DPM¹; Elizabeth Sanders, DPM¹; Amber Morra, DPM¹; Mark Mendeszoon, DPM2,3

The Foot and Ankle Online Journal 13 (3): 3

Arthrodiastasis has been described as an alternative joint sparing procedure for more advanced stages of arthritis. The use of joint distraction has been gaining popularity in foot and ankle surgery, especially with regards to post-traumatic ankle arthritis. Less is known about the effects of arthrodiastasis in cases of acute ankle trauma. This case series presents four cases of intra-articular ankle trauma that were treated with arthrodiastasis using external fixation along with reduction with/without internal fixation. The external fixators were kept on for at least 6 weeks with follow-up of at least 1-2 years for each case. These cases represent high impact injuries that were destined for post-traumatic arthritis that would eventually result in a joint destructive procedure. The results were promising in all cases, by at least delaying the need for a joint fusion or replacement in one case and foregoing the need for such procedures in the other 3 cases within our follow-up period.

Keywords: Arthrodiastasis, ankle, diastasis, arthritis, trauma, post traumatic, external fixation

ISSN 1941-6806
doi: 10.3827/faoj.2020.1303.0003

1 – University Hospitals Regional Hospitals, Surgical Fellow
2 – University Hospitals Regional Hospitals, Fellowship Director; faculty
3 – Precision Orthopaedic Specialities Inc.


The incidence of people with post-traumatic arthritis accounts for nearly 12% of those with symptomatic lower extremity arthritis [1]. Among those with ankle joint osteoarthritis, previous trauma is the most common etiology ranging from 20% to 78% incidence [2-4]. These patients usually end up with joint destructive procedures such as joint fusion or replacement.

Arthrodiastasis is an innovative treatment for ankle arthritis to enhance ankle joint range of motion, diminish pain, and potentially delay or forego ankle joint destructive procedures. Arthrodiastasis of the ankle has been described as an alternative and/or adjunctive salvage procedure for arthritis in patients not amenable to ankle joint replacement or arthrodesis [5]. The procedure is not technically demanding for the surgeon and, long-term, can cost less than arthrodesis or arthroplasty.

Various theories exist to explain how arthrodiastasis has a positive effect on joints. A theory by Gavril Ilizarov suggests applying tension to tissues with distraction increases micro-vascularity to articular cartilage, therefore assisting in cartilage repair [6]. This tension creates a hypervascular state which increases synthesis of nutrients, proteoglycans and in turn helps stimulate chondrocyte formation [6].

Lafeber described a theory in which joint unloading with resulting fluctuations in intra-articular pressure from joint distraction along with concomitant weight bearing, the activity of chondrocytes increases which creates proteoglycans that have the ability to repair articular cartilage and stimulate pluripotent mesenchymal cells to differentiate into articular cartilage [7,8]. This concept of mechanical offloading with continuing pressure changes was shown to increase proteoglycan synthesis by 50% in osteoarthritis knee condyles undergoing arthrodiastasis [7,9]. This process also decreases the inhibition of proteoglycan synthesis by mononuclear inflammatory cells, decreases production of catabolic cytokines and provides increased nutrition delivery to chondrocytes [7].

Both theories predicate the notion that osteoarthritic ankle cartilage is capable of regeneration. Arthrodiastasis has been used over the years with chronic osteoarthritis of the ankle with good results. A review by Dr. Rodriguez-Merchan published in 2017 looked at 14 articles that included patients with end stage osteoarthritis undergoing ankle joint distraction. A total of 249 patients were included in this review with follow up ranging from 1-12 years. Overall 73-91% of patients had good results within their follow up and 6.2-44% of patients ended up with either a joint fusion or replacement [10]. This review serves as a good foundation on the results of ankle joint arthrodiastasis in chronic cases of osteoarthritis, however little is known on its effects during its application in acute trauma. We present a series of acute ankle trauma in which we employ external fixation for arthrodiastasis. In these cases studies, each patient suffered from an intra articular ankle fracture. In the acute setting, the fractures were reduced and an external fixator was applied. Ankle joint diastasis of 5-10mm was applied to the ankle joint utilizing the external fixator. The external fixators were left in place for six to eight weeks.

Case 1

A 30 year-old male sustained an open bimalleolar fracture while operating his horse-drawn lawn mower. Upon presentation to the emergency department, he was evaluated and subsequently taken to the operating room for wound washout, flap closure, application of a delta frame for stability with percutaneous kirschner wire fixation to the medial malleolus. Once the soft tissue envelope was stable nine days later, open reduction and internal fixation was performed. The same delta frame remained intact and the ankle joint was distracted in an attempt to preclude ankle arthritic changes. The frame remained in place for six weeks allowing for ankle joint arthrodiastasis during this time. The patient was seen in the office 1.5 years after surgery and was clinically doing well. He is ambulating without orthoses and able to perform his daily activities without issues. Radiographic images revealed a healed fracture with the ankle mortise in good alignment, without signs of degenerative arthritis.

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Figure 1 Open bimalleolar fracture of a 21-year-old Amish male sustained while operating a horse-drawn lawn mower. Case 1.

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Figure 2 Post-operative radiographs status-post wound washout and closure, open bimalleolar fracture reduction, percutaneous fixation, application of delta frame. Status-post bimalleolar fracture open reduction and internal fixation, syndesmotic repair, and re-application of delta frame to obtain arthrodiastasis at the ankle joint.

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Figure 3 Six weeks status-post bimalleolar fracture open reduction and internal fixation, postoperative day 0 of delta frame removal.

Case 2

A 56-year-old female presented after a motor vehicle accident where she sustained a right closed comminuted talar fracture. Radiographs and a CT scan revealed a Hawkins type IV talus fracture. She was subsequently taken to the operating room after full evaluation in the emergency department. Closed reduction was attempted with a calcaneal pin but was not possible. Therefore, a lateral sinus tarsi approach incision was made from the tip of the fibula extending dorsally over the 4th metatarsal to expose the talus. The talus was reduced and fixed percutaneously with Kirschner wires and a delta frame was applied.

Eleven days later, after the soft tissue envelope improved, she was taken back to the operating room for subtalar and talonavicular joint arthrodesis in an attempt to maintain blood supply to the talus. The deltoid ligament was repaired and a modified Brostrom augmentation was performed. A ring external fixator was placed to achieve stability as well as arthrodiastasis at the ankle joint. The external fixator was removed two months postoperatively. Minor medial ankle arthritis was noted on postoperative radiographic images which worsened over the years. Two years postoperatively the patient is contemplating joint destructive procedures.

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Figure 4 Radiographs and CAT scan of a Hawkins type IV severely comminuted talus fracture. Case 2.

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Figure 5 Intraoperative findings of a severely comminuted talus fracture. Postoperative clinical photos and radiographs of open reduction and external fixation of a comminuted talus fracture, stabilization with percutaneous Kirschner wires and circular external fixator.

Case 3

A 34-year-old male patient was admitted from an outside hospital two days after a trauma where a car he was repairing fell on his left lower limb. He was noted to have a closed dislocated fracture of the left talus, Hawkins type III, and displaced medial malleolus fracture. Closed reduction and splinting was performed at the previous hospital. After full evaluation at our facility, open reduction of the talus and closed reduction of the medial malleolus was performed followed by the application of a ring external fixator. After adequate reduction, approximately a half centimeter of distraction of the ankle joint was produced. This frame was left in place for four months. Following frame removal, the patient continued physical and functional treatment aimed at strengthening the tibial and foot muscles and was encouraged to increase range of motion of the ankle. The patient was able to return to his normal daily activities and return to work. At his two year follow-up he has not needed to go on to further joint destructive procedures and continues to be able to perform his activities of daily living without issue.

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Figure 6 Radiographs on admission. Case 3.

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Figure 7 Radiographs following Ilizarov frame application and during treatment.

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Figure 8 Radiographs following Ilizarov frame removal 80 days status-post reduction and external fixation of talus fracture.

Case 4

A 15-year-old male patient who presented with chief complaint of right foot and ankle injury sustained after a fall while riding a BMX bike. The patient did have a history of a previous talus fracture 3 years prior to this presentation which was treated non-surgically. Radiographic images revealed a Hawkins type III talar neck fracture which was confirmed and evaluated on CT scan. The patient underwent open reduction with internal fixation of the talus fracture with two cannulated screws from posterior to anterior and application of an external fixation with approximately 6-mm of joint distraction.

The external fixator was removed after 6 weeks and the patient was gradually transitioned from a walking boot and into well-supportive sneakers while undergoing physical therapy. He was able to return to his daily activities, sports and BMX bike. The patient was seen in the office 1.5 years after surgery without any clinical or radiographic signs of post traumatic arthritis.

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Figure 9 Preoperative radiographs and CT scan images; post operative radiographs pre and post removal of external fixation.

Discussion

Acute ankle arthrodiastasis with concomitant ankle fracture, open reduction with internal and/or external fixation, should be considered in an attempt to preclude post-traumatic ankle arthritis. This becomes more crucial in cases of intra-articular ankle trauma, where the rate of post-traumatic arthritis increases. With arthrodiastasis, the changes in hydrostatic pressure provide an environment for chondrocyte repair and regeneration thus decreasing the chances for post-traumatic arthritis and the potential need for a joint fusion or replacement. The combination of mechanical offloading along with the microangiogenesis that is produced with increased tension to the soft tissue structures have shown to aid this process of repair.

Vito, et al., distracted 65 arthritic ankles using the Ilizarov frame for 6 weeks with distraction of 5-10 mm [11]. The patients had marked reduction in pain at 12 months for all patients except two: those two went on to arthrodesis. Valburg, et al., reported an average of two years pain relief following three months of arthrodiastasis with an Ilizarov frame [12]. Ploegmakers, et al., assessed the use of arthrodiastasis in 22 patients and reported 73% of the patients had significant improvement at seven years [13]. Although these series were not in the acute setting, one can assess the benefit these series showed with arthrodiastasis of the ankle joint.

This case series showcased four different cases of intra-articular ankle trauma where ankle diastasis was employed as part of the fixation in the acute setting. Successful outcomes were noted in three patients thus far at one to two years of follow up. One of the patients will require a joint fusion or replacement after 2 years. With the widening list of indications for arthrodiastasis, we believe there are benefits of using joint distraction in acute intra-articular trauma to either forgo or delay post-traumatic arthritis. This review serves as a foundation to pursue further indications for arthrodiastasis, however it does have limitations. The sample size is small at this time due to lack of extended follow-up. The follow-up time period listed for these four cases is 1-2 years. The results may prove to be different in the future with extended follow-up, however ankle joint diastasis remains a viable option in patients with intra-articular trauma to possibly reduce or delay the need for arthrodesis in the future

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