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Distal lower extremity manifestations in spina bifida patients of the Yucatan Peninsula: A 24-year retrospective case series

Posted on December 31, 2020 | Comments Off on Distal lower extremity manifestations in spina bifida patients of the Yucatan Peninsula: A 24-year retrospective case series

by Alexandra Heidtmann, BS1; Lahari Madulapally, BS, MA1; Luis Rodriguez Anaya, DPM2*; Daniel Cawley, DC, MS2

The Foot and Ankle Online Journal 13 (4): 8

Spina Bifida, a rare congenital disorder with an incidence of 7.85 per 10,000 births in Mexico. It results from the failed closure of the neural tube leading to the incomplete development of the neural arches. This case series is part of the Yucatan Crippled Children’s Project that began in 1996 by Charles Southerland, Doctor of Podiatric Medicine and former professor of Barry University’s School of Podiatric Medicine. All patients in this study were assessed and treated at the Red Cross hospital in the city of Merida, Yucatan, Mexico. Attendings, residents and medical students travel to the Yucatan Peninsula four times a year for a period of one week. Given that this study was built from reports of medical mission trips that occur four times a year with limited resources and time, the lack of documentation of treatment plans and follow-ups made it difficult to identify surgical procedures and assess the success of surgeries. Additionally, we did not have access to the patients birth records or their mothers medical records to accurately determine the etiology of their deformities. Based on our data, we conclude that intervention should be considered as early as possible in any flexible deformity to prevent them from becoming rigid.

Keywords: Spina Bifida, talipes equinovarus, clubfoot, adductovarus, calcaneo valgus, Ponseti, osteomyelitis

ISSN 1941-6806
doi: 10.3827/faoj.2020.1304.0008

1 – Fourth Year Medical Student at Barry University School of Podiatric Medicine, Miami, Florida.
2 – Assistant Professor Barry University School of Podiatric Medicine
* – Corresponding author: LARodriguez@barry.edu

Spina Bifida, a rare congenital disorder with an incidence of 7.85 per 10,000 births in Mexico. It results from the failed closure of the neural tube leading to the incomplete development of the neural arches [1]. Spina bifida is the result of genetic and non-genetic factors that interfere with the folding and closure of the neural tube. In its most severe form, meningomyelocele, the neurons of the spinal cord are exposed to amniotic fluid resulting in neuronal death. In addition, the spinal cord and meninges protrude through the midline bony defect of the back [2].

The clinical manifestation of a meningomyelocele is dependent on the spinal level of involvement and the presence of cerebral involvement and hydrocephalus [3]. Sensory and motor impairments are commonly present below the level of the lesion causing alterations in the bowel and bladder function, muscle paresis and paralysis, and sensory loss. Impairment is classified by the level of neurosegmental involvement determined by the strength of specific muscle groups [3]. Nearly all patients with spina bifida will experience manifestations in their feet, especially those cases involving the thoracic and lumbar spinal regions [4,5]. Previous studies have reported that the most common manifestation of spina bifida in the feet are talipes equinovarus, equinus, vertical talus, calcaneal deformities, and cavovarus [4,6,7]. The aim of this study is to analyze the incidence of various distal lower extremity manifestations and their long-term effects on spina bifida patients of the Yucatan Peninsula.

Methods

This case series is part of the Yucatan Crippled Children’s Project that began in 1996 by Charles Southerland, Doctor of Podiatric Medicine and former professor of Barry University’s School of Podiatric Medicine [8]. All patients in this study were assessed and treated at the Red Cross hospital in the city of Merida, Mexico. Attendings, residents and medical students travel to the Yucatan Peninsula four times a year for a period of one week.

From 1999 to 2020, we retrospectively analyzed 1,489 patients that were seen by physicians from the Yucatan Crippled Children’s Project. Among the total, we identified 25 patients, 17 male and 8 female, with history of Spina Bifida and concomitant lower extremity deformities. From the 25 patients, 15 patients had bilateral lower extremity deformities and 10 patients had unilateral deformities, leading to a total of 40 limbs. The ages ranged from 3 months old to 43 years old, with a total average age of 11.33 years. The mean age for rigid deformities was 15.4 years, while the mean age for flexible deformities was 8.8 years.

We analyzed 3 cases of patients with a history of spina bifida and lower extremity deformities according to the clinical notes collected from the Yucatan Crippled Children’s Project.

Case Presentation 1

Case 1 is a 2-year-old male who presented to the Yucatan Crippled Children’s project clinic in March of 2007 with a chief complaint of difficulty ambulating. Patient’s family reports past medical history of birth at 38 weeks and significant time spent in the NICU due to hydrocephalus and spina bifida. Upon initial assessment, the patient was alert and oriented and showed no additional symptoms. Patient was diagnosed with bilateral flexible clubfoot deformity, as seen in Figures 1 and 2. Considering the age and the flexibility of the deformity, the conservative Ponseti serial casting technique was performed on the patient.

Figures 1 and 2 Plantar and dorsal views of bilateral clubfoot prior to Ponseti method.

Figures 3 and 4 Patient at 12 years old, 10 years after Ponseti method. AP Radiograph of bilateral feet. Pre-operative clinical picture of bilateral feet.

Figures 5, 6, 7 Intraoperative picture of left foot before and after external fixator application. AP radiograph of left foot.

The patient and his family were instructed to follow – up with the local doctor. The patient returned to the clinic in November of 2017, at 12 years old, with chief complaint of continued difficulty ambulating due to the progression of the windswept deformity (Figures 3 and 4). After assessment, the left foot was diagnosed with adductovarus and talipes equinovarus deformity. The right foot was diagnosed with forefoot adduction, midfoot abduction, and calcaneovalgus which are the three components of complex skew foot. Given the Ponseti technique applied ten years ago had failed, the deformity has worsened and progressed from flexible to rigid. The procedure consisted of application of external fixation with medial motor for gradual correction of adductovarus deformity on the left foot (Figures 5-7).

Figure 8 Bilateral flexible cavovarus deformity with ulcer on dorsolateral aspect of right foot.

Figures 9 and 10 Nine year follow-up shows rigid bilateral cavovarus deformity. The patient is confined to a wheelchair.

The patient presents to the clinic in February of 2018 for a 3 month postoperative visit after application of external fixation. It was noted that the toes were not fixated during the external fixator surgery and they developed flexion contractures within the reduction frame. The patient developed clinodactyly of all five digits of the left foot. At this date, the frame was removed and the patient began physical therapy in an attempt to reduce flexion contractures.

Case Presentation 2

Case 2 is an 8-year-old female with a past medical history of spina bifida and sensory neuropathy bilaterally. The patient presented to the clinic in July of 2011 with a chief complaint of a wound on the right foot. Upon physical exam, an open ulcer was noted on the dorsolateral aspect of the right foot along the cuboid-5th metatarsal joint (Figure 8). The wound has a beefy red base with friable granulation tissue and a circumferential macerated periwound with suspected areas of hyperkeratotic tissue. Mild hyperpigmentation and erythema is noted proximally towards the dorsum of the ankle. The patient was diagnosed with a pressure ulcer on the right foot and bilateral flexible cavovarus deformity. Ulcer was managed during the patient’s first visit prior to any surgical intervention. Upon healing of the ulcer, surgery was performed; however, the surgical technique was not recorded.

The patient was virtually contacted during Covid-19 2020 Pandemic, and sent Figures 9 and 10. The patient stated she still has insensate feet and is unable to ambulate. Deformity has progressed to rigid and she is waiting until the next Yucatan Medical Mission Trip to possibly undergo another surgery that would allow her to ambulate.

Case Presentation 3

Case 3 is a 24-year-old male with a past medical history of spina bifida, insensate feet, and chronic lymphangitis. The patient presented to the Yucatan Project Clinic in April of 2005 with a chief complaint of wounds on the right foot. Upon physical exam, ulcer on the lateral aspect of the head of the 5th metatarsal of the right foot was noted to have a 50/50 granular fibrotic base with slough in the center. The periwound consisted of hyperkeratotic tissue on the plantar aspect of the 5th metatarsophalangeal joint. Hyperpigmentation is present extending proximally on the dorsum of the foot. The second wound, located on the lateral aspect of the 5th metatarsal tuberosity of the right foot, appeared to have 75/25 fibrotic granular base with regular borders. The patient was diagnosed with active infected ulcers and bilateral cavovarus deformity (Figures 11 and 12). Initial treatment consisted of ulcer debridement and offloading of the right foot with a 3D Walker. The patient was seen in November of 2005, 7 months after initial treatment. The right foot still remained dysfunctional with chronic non-healing wounds. Radiographs show radiolucency from mid-shaft of 4th and 5th metatarsal distally to the 4th and 5th distal phalanges and thickened periosteum of the proximal end of the mid shafts of 4th and 5th metatarsals on Figure 13, suggesting osteomyelitis.

Figures 11 and 12 Dorsolateral view of right foot showing ulcer along 5th metatarsal and medial view of the right foot showing cavovarus deformity.

Figure 13 AP Radiograph of bilateral feet.

Figures 14 and 15 Preoperative view of the right foot. Intraoperative picture of the right foot after Lisfranc amputation.

Figure 16 Dorsolateral view of right foot after amputation.

Due to the lack of access to other diagnostic tools, combined with the request from the patient for a permanent solution, a LisFranc amputation was performed on the right foot and osteoset beads with vancomycin were inserted to treat the infection (Figures 14 and 15).

The patient was seen in February of 2006, 3 months after LisFranc amputation of the right foot. The surgical site healed well with good results (Figure 16). However, the cavovarus deformity remained on the left foot (Figure 17).

The patient was seen again in November of 2006 for the last time, 12 months after LisFranc amputation of the right foot. The patient redeveloped an equinus deformity of the right foot and ulcers under styloid processes bilaterally (Figure 18). The patient was treated with well-padded plastazote ankle foot orthosis (AFO).

Figure 17 Dorsolateral view of left foot.

Figure 18 Ulcer under styloid process of the right foot after amputation.

Results

The most common lower extremity manifestation was ulcerations. In 17 ulcerated limbs, 8 were insensate and 5 developed osteomyelitis. Out of the 40 limbs, 5 ulcerated limbs had no reported gross deformity, and therefore were not included in the graphs. The remaining 35 limbs were biomechanically classified as rigid and flexible. In the 13 rigid limbs, there were 4 equinus, 3 talipes calcaneus, 7 cavus, and 2 planus feet. These were further subclassified into 6 more categories: equinovarus, pes cavocalcaneus, cavovalgus, cavovarus, pes planovalgus and no additional deformity. Rigid deformities subgroups can be seen in Graph 1. In the 22 flexible limbs, there were 9 equinus, 5 talipes calcaneus, 6 cavus, and 2 planus feet.

Graph 1 Rigid deformity of the foot.

Graph 2 Flexible deformity of the foot.

They were then subclassified into 5 more categories: calcaneovalgus, calcaneovarus, equinovarus, cavovarus, and no additional deformity. Flexible deformities are illustrated in Graph 2.

Discussion

Lower extremity manifestations due to spina bifida are difficult to be classified and the rate of misdiagnosis and mistreatment is high [9]. Similarly to previous findings, the most frequent foot deformity in our study was flexible equinus [4,5,10]. However, we found that rigid pes cavus was the second most predominant foot deformity in the Yucatan Peninsula, contrary to previous reports. Based on the limited medical access in the area and the higher average age of patients presenting with rigid deformities (15.4 years) when compared to flexible deformities (8.8 years), we suggest that this is possibly due to years of leaving the deformity untreated.

The prevalence of spina bifida was 7.85 per 10,000 births or 0.0785% in the country of Mexico [1]. However, in the Yucatan Peninsula the prevalence was found to be significantly higher. In this study, out of the 1,489 total cases analyzed from years 1999 to 2020, 25 patients with spina bifida were identified. This shows a prevalence rate of 167.90 per 10,000 births or 1.68%. Folic acid is a nutrient that is essential to the development of the fetus. Spina bifida and other birth defects form within the initial 28 days after conception. These congenital deformities can be prevented by ensuring sufficient blood folate levels in the mother during fertile years and early fetal development [11]. In the literature, North America has been shown to have the lowest incidence of spina bifida while Asia has the highest incidence. This could be due to Canada and the United States being the first countries to mandate folic acid fortification. In addition, even though mandatory fortification with folate has been implemented in many countries, it might not be enough folic acid to reach the daily recommended dosage of 400 micrograms. Therefore, it is important for mothers before conception and in the early fetal developmental months to supplement their folic acid intake [12].

Case 1 illustrated a patient with talipes equinovarus, also known as clubfoot deformity on the left foot. This triplanar deformity includes 3 components: ankle equinus, hindfoot varus, and forefoot adduction. Traditionally, there is a higher prevalence of clubfoot in males with a ratio of 2:1 to female and approximately 50% of the cases are bilateral. A few etiologies have been described in the literature, mainly divided into idiopathic and non-idiopathic. Idiopathic consists of limited intrauterine position due to a larger size of the fetus or smaller frame of mothers, while non-idiopathic includes a history of congenital deformities such as spina bifida, cerebral palsy, and meningitis. Clinical presentation of patient 1 at birth predisposed him to a higher risk of developing clubfoot given he is a male with a history of spina bifida. We do not have additional history of patient 1 such as birth weight and height, however, these factors could have also played a role in the patient developing clubfoot [13]. The Ponseti technique, a conservative treatment, was attempted when the patient was 2 years old; however, this technique is only proven to be successful in patients with flexible clubfoot up to 120 days of age [14]. On the right foot, the patient has had a long-standing complex skewfoot with forefoot adduction, midfoot abduction, and calcaneovalgus. The unsuccessful result of the Ponseti method on the left foot, combined with years of the patient not returning for medical assistance, led the bilateral deformity to become rigid on both feet.

Case 2 presented a 2-year-old female with history of spina bifida, insensate feet, active ulcer on the right foot, and flexible bilateral cavovarus deformity. Cavovarus involves a high longitudinal plantar arch, hindfoot varus, forefoot equinus, and pronated first ray in the stance phase of gait. If this deformity is present bilaterally, the most likely etiology is a neurological condition; however, if it is present unilaterally the etiology can be related to trauma such as pilon fractures or talar neck fractures [15]. In a flexible cavovarus foot, surgical correction could be achieved through extensive plantar release and metatarsal osteotomies. However, at the time of the patient’s first visit, physicians from the Yucatan Project prioritized the management of the ulcer prior to correcting any gross deformity. Due to the long period of treatment for ulcer management and limited medical access in the Yucatan Peninsula, the patient did not seek medical help for many years. Recent literature has described that if left untreated, cavovarus deformity can progress into fibrosis of the plantar fascia, shortening and tightening of the achilles tendon leading to excess pressure under metatarsal heads, overloading of the lateral aspect of the foot leading to stress fractures of the 5th metatarsal and more rarely, the cuboid. In addition, it can cause inadequacy of the lateral ligaments and tendons leading to instability of anterolateral ankle and lateral talus [15]. During the 2020 Covid-19 pandemic, we reached out through social media and discovered the patient was no longer ambulating. The patient described a rigid deformity with insensate feet and showed interest in undergoing another surgery, so she could possibly walk again. In a mature foot, surgical intervention might require aggressive techniques including midtarsal osteotomies, calcaneal osteotomies and triple arthrodesis [16]. Final decision for a surgical procedure will only be done in person once full updated history and radiographs are taken.

Case 3 showed the most severe result that could come from insensate lower extremity in spina bifida patients if left untreated for long periods of time: amputation. This patient was first seen at 24 years old, when his rigid cavovarus deformity was present since birth. This deformity caused chronic non-healing wounds that developed into osteomyelitis. Osteomyelitis can be defined as an infectious agent which causes inflammation of the bone. The hallmark of chronic osteomyelitis is the progression of inflammation to tissue necrosis and destruction of bone trabeculae and bone matrix caused by an infectious agent. This is usually accompanied by fragments of bone lacking blood supply which can become separated to form sequestra and continues to host and spread bacteria despite antibiotic treatment. The fifth metatarsal, first metatarsal, calcaneus, and first digit distal phalanx are the four structures with the highest incidence of developing osteomyelitis in the foot [17]. This case emphasizes the need of spina bifida patients with concomitant lower extremity deformities to seek medical help at a young age to avoid the progression of the deformity and consequently loss of a limb.

Given that this study was built from reports of medical mission trips that occur four times a year with limited resources and time, the lack of documentation of treatment plans and follow-ups made it difficult to identify surgical procedures and assess the success of surgeries. Additionally, we did not have access to the patients birth records or their mothers medical records to accurately determine the etiology of their deformities.

Conclusion

The types of foot and ankle deformities seen in spina bifida are diverse in etiology, age and gender of the patients. We discovered the most common lower extremity manifestations of spina bifida in the Yucatan Peninsula are flexible equinus and rigid pes cavus. The mean age of patients with rigid deformities was almost twice as the mean age of the patients with flexible deformities. Zang, et al., concluded that equinovarus requires immediate treatment while valgus deformities can have delayed intervention [15]. Based on our data, we conclude that intervention should be considered as early as possible in any flexible deformity to prevent them from becoming rigid.

Acknowledgements

We would like to thank all attendings, residents and students involved in the Yucatan Crippled Children Project along with the International Foot & Ankle Foundation for Education and Research. Additionally, we would like to thank the local Red Cross Hospital in the city of Merida.

References

  1. Gunay H, Sozbilen MC, Gurbuz Y, Altinisik M, Buyukata B. Incidence and type of foot deformities in patients with spina bifida according to level of lesion. Childs Nerv Syst. 2016;32(2):315-319. doi:10.1007/s00381-015-2944-7.
  2. Swaroop VT, Dias L. Orthopaedic management of spina bifida-part II: foot and ankle deformities. J Child Orthop. 2011;5(6):403-414. doi:10.1007/s11832-011-0368-9.
  3. Sharrard WJ, Grosfield I. The management of deformity and paralysis of the foot in myelomeningocele. J Bone Joint Surg Br. 1968;50(3):456-465.
  4. Atta CA, Fiest KM, Frolkis AD, et al. Global Birth Prevalence of Spina Bifida by Folic Acid Fortification Status: A Systematic Review and Meta-Analysis. Am J Public Health. 2016;106(1):e24-e34. doi:10.2105/AJPH.2015.302902.
  5. Frischhut B, Stöckl B, Landauer F, Krismer M, Menardi G. Foot deformities in adolescents and young adults with spina bifida. J Pediatr Orthop B. 2000;9(3):161-169. doi:10.1097/01202412-200006000-00005.
  6. Krähenbühl N, Weinberg MW. Anatomy and Biomechanics of Cavovarus Deformity. Foot Ankle Clin. 2019;24(2):173-181. doi:10.1016/j.fcl.2019.02.001.
  7. Westcott MA, Dynes MC, Remer EM, Donaldson JS, Dias LS. Congenital and acquired orthopedic abnormalities in patients with myelomeningocele. Radiographics. 1992;12(6):1155-1173. doi:10.1148/radiographics.12.6.1439018.
  8. Yucatán Crippled Children’s Project. (2012). Retrieved from (https://www.internationalfootankle.org/philanthropy/crippled-childrens-project/).
  9. Mandell JC, Khurana B, Smith JT, Czuczman GJ, Ghazikhanian V, Smith SE. Osteomyelitis of the lower extremity: pathophysiology, imaging, and classification, with an emphasis on diabetic foot infection. Emerg Radiol. 2018;25(2):175-188. doi:10.1007/s10140-017-1564-9.
  10. Cavalheiro S, da Costa MDS, Moron AF, Leonard J. Comparison of Prenatal and Postnatal Management of Patients with Myelomeningocele. Neurosurg Clin N Am. 2017;28(3):439-448. doi:10.1016/j.nec.2017.02.005.
  11. McCluskey WP, Lovell WW, Cummings RJ. The cavovarus foot deformity. Etiology and management. Clin Orthop Relat Res. 1989;(247):27-37.
  12. Feldkamp, M., Sanchez, E., & Canfield, M. (2014). International Clearinghouse of Birth Defects Surveillance and Research – Annual Report 2014.
  13. Awang M, Sulaiman AR, Munajat I, Fazliq ME. Influence of Age, Weight, and Pirani Score on the Number of Castings in the Early Phase of Clubfoot Treatment using Ponseti Method. Malays J Med Sci. 2014;21(2):40-43.
  14. Copp AJ, Adzick NS, Chitty LS, Fletcher JM, Holmbeck GN, Shaw GM. Spina bifida. Nat Rev Dis Primers. 2015;1:15007. Published 2015 Apr 30. doi:10.1038/nrdp.2015.7.
  15. Zang J., Qin S., Shi L. (2020) Lower Limb Deformity Caused by Spina Bifida Sequelae and Tethered Cord Syndrome. In: Qin S., Zang J., Jiao S., Pan Q. (eds) Lower Limb Deformities. Springer, Singapore.
  16. Kancherla V. Countries with an immediate potential for primary prevention of spina bifida and anencephaly: Mandatory fortification of wheat flour with folic acid. Birth Defects Res. 2018;110(11):956-965. doi:10.1002/bdr2.1222.
  17. Broughton NS, Graham G, Menelaus MB. The high incidence of foot deformity in patients with high-level spina bifida. J Bone Joint Surg Br. 1994;76(4):548-550.

 

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Emphysematous osteomyelitis of the foot: A case report

Posted on September 30, 2020 | Comments Off on 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.

 

 

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Arthroscopy of septic ankle and subtalar joints: A case report

Posted on March 31, 2020 | Comments Off on Arthroscopy of septic ankle and subtalar joints: A case report

by Karl Dunn, DPM, FACFAS1; Jordan Haber2*

The Foot and Ankle Online Journal 13 (1): 1

Infection of the ankle and subtalar joints is a serious and potentially debilitating condition. Of the few reported cases, results have varied. Traditionally open procedures have been performed, some of which even led to amputation. In the case presented, the authors demonstrate a successful result utilizing arthroscopic techniques in combination with intravenous antibiotic therapy. At two-year follow-up the patient has remained infection free.

Keywords: arthroscopy, infection, osteomyelitis, septic joint, pyarthrosis

ISSN 1941-6806
doi: 10.3827/faoj.2018.1301.0001

1 – Mid-Michigan Orthopaedic Institute, A Division of Compass Healthcare; East Lansing, MI 
2 – Undergraduate Researcher of the Honors College of Michigan State University; East Lansing, MI
* – Corresponding author: haberjor@msu.edu

Joint sepsis is a severe condition with an annual incidence of 2-10 per 100,000 individuals per year [1]. Possible morbidity following joint sepsis includes chronic pain, irreversible joint damage, dysfunction, disability and a mortality rate in 11.5% of cases [2]). The prognosis is directly influenced by the promptness of the diagnosis and initiation of antibiotics [3]. Although administration of culture specific antibiotics with joint decompression are the cornerstones of treatment in a septic joint, debate in the literature exists between open versus arthroscopic approaches.

Larger, over smaller, joints seem to have a predilection, with the hip and knee constituting the majority (60%) of all septic joints [4]. Several risk factors are associated with the development of a septic joint: history of joint prostheses, rheumatoid arthritis, intravenous drug abuse, alcoholism, diabetes, previous intra-articular corticosteroid injection, immunosuppression therapy, among others [5]. 

Interestingly, the false perception is that pyarthrosis is overwhelmingly monoarthritic, as the literature supports polyarticular sepsis 22% of the time [6]. Pyarthrosis has also been reported within the ankle [7,8], metatarsal-phalangeal joints [9], and interphalangeal joints [10]. The presence of joint sepsis in the subtalar joint is extremely rare, as only two other cases have been recorded in the medical literature [5,11]. To the authors’ knowledge, the case presented is the only polyarticular septic arthritis report in which arthroscopic methods were utilized in successful treatment of the subtalar joint.

Case Study

A 68-year-old female presented to the author’s clinic, in May 2016, for evaluation of a tender and swollen left foot and ankle. She reported 9 days prior to arrival, while living in her retirement home in Florida, she suffered a severe and sudden amount of pain to her left foot and ankle while attempting to stand. As a result of the pain, she then fell to the ground. She denied a history of trauma as the inciting event, as the pain occurred prior to her fall. Due to her inability to bear weight, the patient presented to an urgent care clinic, in which radiographs were noted to be negative for fracture. She was placed in a supportive ankle brace and was given non-steroidal anti-inflammatories. She states over the next several days it was difficult to bear weight while her ankle became more edematous. She states she was concerned about her condition and elected to drive back to her permanent residence. Seven days following her first episode of pain she stopped en route to Michigan at a major university hospital in Ohio due to increased pain during her travels. Standard repeat ankle radiographs were negative and she was started on an oral methylprednisolone course. A venous Doppler was negative for deep venous thrombosis.

Upon arrival in the office, the patient was non-diaphoretic, afebrile and in pain. Her past medical history was pertinent for rheumatoid arthritis, treated with bi-weekly injections of adalimumab. Physical examination revealed a mildly edematous foot and ankle, without calor or erythema. Neurovascular status was intact. Range of motion to her ankle and midtarsal joints was severely painful and limited. There was diffuse tenderness of her entire foot and ankle, with maximum tenderness to her lateral foot and posterior ankle. As a result, a lower extremity magnetic resonance image (MRI) was ordered for the patient.

Two days after her MRI, she reported a dramatic increase in pain and presented to her closest emergency department. The patient was afebrile, and her vitals and lab markers revealed no findings of sepsis or leukocytosis. The emergency department physicians’ primary differential diagnosis was continued pain due to ankle sprain. She was discharged from the hospital and was sent directly to our office again as the MRI report was still pending. Upon review of the MRI the radiologist was unable to determine if the large joint effusions were infectious or aseptic due to lack of contrast in the imaging study (Figure 1). There were joint effusions and reactive synovium noted at the ankle, as well as the subtalar joint, with a large expansion noted at the posterior recess (Figure 2). As a result, a joint aspiration was performed from the anterior aspect of the ankle joint. Greater than 3 cc of yellow purulent material was aspirated and the patient was directly admitted to the hospital upon findings of a septic joint. 

Figure 1 Axial MRI T2 weighted image displaying posterior abscess.

Figure 2 Sagittal MRI image which displays reactive bone marrow edema consistent with early osteomyelitis, large posterior fluid collection adjacent to subtalar and ankle joint consistent with abscess.

Upon arrival at the hospital, broad spectrum antibiotics were initiated with previous cultures pending. Infectious disease was consulted for management of antibiotics. Pertinent laboratory findings revealed a white blood count (WBC) of 9,300 WBC/mcL, a C-reactive protein (CRP) of 11.1 mg/L, and an erythrocyte sedimentation rate (ESR) of 47 mm/hr. Except for a small abrasion to her index finger two weeks prior, the patient did not have any other portals of infection. Blood cultures were taken upon presentation and were negative. Due to the patient’s immunosuppression on adalimumab, the medication was stopped indefinitely as this was thought to have predisposed the patient to the infection. She was taken to the operating room the next day for an arthroscopic debridement of the septic joints.

Initial Surgery

After general anesthesia was administered, a well-padded thigh tourniquet was placed. The ipsilateral thigh was placed in a thigh holder and the limb was held in a gravity dependent position. The limb was prepped, draped, and the tourniquet was inflated to 325 mmHg. The subtalar joint was first approached with aspiration to confirm the presence of joint sepsis in this location. An 18-gauge needle was placed on a leur-locked syringe, and the subtalar joint was first accessed for aspiration. The subtalar joint was approached from the sinus tarsi, upon which purulent fluid was aspirated (Figure 3). We traced the distal aspect of the fibula posteriorly, and the 18-gauge needle was placed from posterior to anterior just lateral to the Achilles tendon. Approximately 5 mL of purulent fluid was extracted from the posterior recess of the subtalar joint. Arthroscopic portals were then established, two to the lateral hindfoot at the level of the sinus tarsi, with a third at the posterior recess for an auxiliary suction portal. Upon initial evaluation, the joint was cloudy with purulent material. The joint had elements of infected synovium which was debrided. After establishing a view of the interosseous ligament, the debridement then continued from anterior to posterior and into the posterior facet. There was noted to be loose and degraded cartilage, which exposed the underlying subchondral bone. All loose material was debrided, and the subchondral bone was noted to be firm without obvious signs of infection. After a thorough debridement, 3 liters of fluid was used for irrigation until the joint fluid appeared translucent.

Attention was then directed to the ankle joint for arthroscopy. Anteromedial and anterolateral portals were established and a standard 21-point examination ensued. 

Figure 3 Subtalar joint aspiration with purulence; the accessory portal during arthroscopic irrigation and debridement placed in the same posterior recess.

The joint fluid was initially cloudy, with a lesser degree of loose bodies as the subtalar joint. The ankle cartilage was largely intact, and less irrigation was needed than the subtalar joint to establish a lucent joint and a clear picture with the arthroscopic equipment. Following the thorough irrigation, debridement, and removal of infected synovium, all equipment was removed from the ankle. The portals from the subtalar and ankle joints were left open to allow for joint decompressions and continue to drain.

On postoperative day one, the patient reported an immediate reduction in pain and no postoperative events recorded. The patient remained improved on post-operative day two as well. Culture results from the initial aspiration yielded a Methicillin-resistant Staphylococcus aureus (MRSA) pathogen which was sensitive to vancomycin. She reported a mild increase in pain on postoperative day three and as a result a repeat MRI was ordered with and without contrast to further scrutinize the septic joints. There was noted contrast enhancement of the synovium of the ankle and subtalar joints was noted with joint effusion, with new findings of marrow edema and contrast enhancement of the joint surfaces of the subtalar and ankle joints indicative of acute osteomyelitis. Following a lengthy discussion with the patient regarding possible limb salvage potential with a conversation regarding further arthroscopic versus an open approach, the patient agreed to a second arthroscopic approach to her ankle and subtalar joints.

Second Surgery

The subtalar joint was again approached first with an attempted aspiration. No fluid or purulence was able to be extracted from the sinus tarsi nor the posterior recess of the joint. Arthroscopic equipment was re-introduced in the similar approach, with the posterior drainage portal established as well. There was continued loose cartilage and infected synovium, and an aggressive debridement was performed to the joint. Following copious irrigation, the equipment was removed and the ankle was inspected as well. The joint fluid remained relatively clear, and the cartilage was intact. Following an aggressive synovectomy and irrigation all instrumentation was removed. The portals were again left open to decompress the joint.

Recovery

The patient recovered well following the second arthroscopic procedure, and was prescribed a 6-week course of intravenous vancomycin. Two days following the second procedure, the patient was discharged home with a peripherally inserted central catheter (PICC) line for continued antibiotic administration. Inflammatory lab markers were followed on a weekly basis. Four weeks after discharge, her CRP and ESR normalized and did not re-elevate following her course of antibiotics. Her skin incisions healed without complications by three weeks succeeding the second surgery.

The patient was gradually able to increase her weight bearing status, first with a fixed walking boot for 4 weeks following her hospitalization, and transitioning to an ankle brace thereafter. The patient was sent for extensive physical therapy to aid in her recovery back to full weight bearing. She was eventually able to forego any bracing or assistance 6 months following her hospitalizations with mild pain which was treated with oral NSAIDs. Upon most recent follow-up at fourteen months, she was able to return to all pre-injury activities. No continued signs of infection were noted on clinical exam.

Discussion

The subtalar joint is a highly complex articulation of the talus and calcaneus. There are usually three distinct facets (anterior, middle, posterior) which may differ in the context of a tarsal coalition. There is a degree of variability in the ligamentous contents and joint capsular attachments as well. Arthroscopic evaluation of the subtalar joint remains a lesser utilized procedure, likely due to the complexity of the joint and the relatively small surface area which may be difficult for an unfamiliar arthroscopist to visualize. 

To the authors’ knowledge, no other cases have been reported in the literature in the successful arthroscopic treatment of a septic subtalar joint. Two other cases were identified in the literature but utilized an open approach in combination with antibiotic therapy. A Danish report displayed an isolated septic subtalar joint caused by Neisseria gonorrhoeae [5]. A second, more recent case study depicted an open approach to an infection caused by Mycoplasma hominis [12]. In both cases the infection was rapidly identified, treated, and the patients recovered fully. In the present case, two major risk factors were identified which likely predisposed the patient to her septic joint: rheumatoid arthritis and concomitant immunosuppression therapy. Patients with RA are more likely to develop joint sepsis both due to the disease process itself and due to the immunosuppressive therapy that they receive. Dissimilar to the previous reports is the fact that the patient was successfully treated with an arthroscopic approach, which did not require an open method with antibiotics, to cure the infection.

A joint aspiration of a joint is considered the gold standard in diagnosis of a septic joint, and should be performed with a low threshold when pyarthrosis is considered as a differential diagnosis. Once the diagnosis has been confirmed, rapid initiation of antibiotics should be employed. Arthroscopic and open approaches are useful tools in the irrigation and debridement of the septic joint. Advantages are inherent to an arthroscopic versus open approach: decreased soft tissue damage, which may lead to a shorter hospital stay, possible overall faster patient recovery. Repeat open versus arthroscopic lavage and debridement may be required until the infection has been controlled. This especially holds true when there has been a delay in the diagnosis and initiation of antibiotics from time of initial presentation, such as the case presented.

Conclusion

Due to the proximity of the ankle and subtalar joint, which share a capsular connection, the authors advocate that the subtalar joint may be affected more commonly than previously thought when a septic ankle is encountered. Prognosis of a septic ankle shows probable infection of the subtalar joint, causing a septic subtalar joint. The pyarthrosis of the septic ankle can be polyarthritic [6]. The infection can spread from the septic ankle to the subtalar joint through the sinus tarsi, causing infection of the subtalar joint. Performing a procedure to prevent infection transmission through the sinus tarsi may hinder the ease of the infection to spread; the procedure would counteract the close proximity of the ankle and subtalar joints, preventing a monoarthritis diagnosis from developing into a polyarthritic case.

Given the case encountered, an arthroscopic approach may be beneficial over an open approach when a septic subtalar joint is encountered. An arthroscopic approach is less invasive, leading to a less cumbersome recovery and quicker recovery time for the patient. Arthroscopic aspiration of purulent fluid is more effective in comparison to an open approach. A late diagnosis of a septic subtalar joint is more effectively combated by an arthroscopic approach, as multiple open lavages and debridements are required to accomplish what could be completed by a single arthroscopic approach. Although the literature does not have copious studies regarding open irrigation vs. arthroscopic irrigation, when comparing both methods used during treatment of acute native knee septic arthritis, 71% of the open treatment group required repeat irrigation compared to 50% in the arthroscopic treatment group [12]. Lowered chance of repeated irrigation serves as an assuring factor that an arthroscopic approach would withhold the need of multiple joint irrigations, leading to a quickened overall patient recovery.

The septic subtalar joint cases described in the literature used a combination of an arthroscopic and open procedure to eradicate infection. This is the first described case of a 68-year-old patient with a septic ankle and subtalar joint that was successfully treated with an arthroscopic approach alone. Open procedures are not necessary for proper treatment of septic arthritis. An open procedure combined with antibiotic therapy will likely lead to a more arduous path for patient recovery. Although up to physician preference, research is spreading the belief that arthroscopic procedures are less invasive and more efficient for patient recovery.  

Acknowledgements: None

Funding Declaration: None

Conflict of Interest Declaration: None

References

  1. Kaandorp CJE, Dinant HJ, van de Laar MAFJ, Moens HJB, Prins APA, Dijkmans BAC. Incidence and sources of native and prosthetic joint infection: a community based prospective survey. Ann Rheum Dis. 1997;56:470–475
  2. Weston VC, Jones AC, Bradbury N, Fawthrop F, Doherty M. Clinical features and outcome of septic arthritis in a single UK Health District 1982–1991. Ann Rheum Dis. 1999;58:214–219.
  3. Weston VC, Jones AC, Bradbury N, et al. Clinical features and outcome of septic arthritis in a single UK Health District 1982–1991. Ann Rheum Dis 1999; 58:214–219.
  4. Mathews CJ, Coakley G. Septic arthritis: current diagnostic and therapeutic algorithm. Curr Opin Rheumatol. 2008;20(4):457-62.
  5. Wynes J, Harris IV W, Hadfield RA, Malay DS. Subtalar Joint Septic Arthritis in a Patient with Hypogammaglobulinemia. Journal Foot and Ankle Surgery 2013; 52:242-248.
  6. Dubost JJ, Fis I, Denis P, et al. Polyarticular septic arthritis. Medicine (Baltimore) 1993; 72:296–310.
  7. Margaretten ME, Kohlwes J, Moore D, Bent S. Does this adult patient have septic arthritis?. JAMA. 2007;297(13):1478-88.
  8. Newman JH. Review of septic arthritis throughout the antibiotic era. Ann Rheum Dis 35:198–205, 1976.
  9. Velilla-Moliner J, Martinez-Burgui JA, Cobeta-Garcia JC, Fatahi-Bandpey ML. Podagra, is it always gout? Am J Emerg Med 22:320–321, 2004.
  10. Brower AC. Septic arthritis. Radiol Clin North Am 34:293–309, 1996.
  11. Larsen LS, Nielsen DC, Terslev L, Amris K. Infectious monoarthritis in the subtalar joint due to N. gonorrhoeae. Ugeskr Laeger 169:324–325, 2007.
  12. Johns, B P, et al. “Open Compared with Arthroscopic Treatment of Acute Septic Arthritis of the Native Knee.” PubMed.gov, NCBI, 15 Mar. 2017, www.ncbi.nlm.nih.gov/pubmed/28291

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Case study of rare incidence of gas gangrene caused by Raoultella Ornithinolytica

Posted on December 31, 2018 | Comments Off on Case study of rare incidence of gas gangrene caused by Raoultella Ornithinolytica

by Edward Mirigliano DPM, MBA1, Kyle Hopkins DPM2, Samantha Banga, DPM3

The Foot and Ankle Online Journal 11 (4): 1

Gas gangrene is a bacterial infection that produces gas in tissues.  It is fast spreading, potentially life-threatening and needs to be addressed promptly.  In this case report, we present a patient that presented with gangrene of his left foot.  He was first seen in the emergency room where blood cultures and x-rays were obtained.  The patient was then promptly treated with OR debridement of the site and the cultures obtained intraoperatively revealed a rare organism, Klebsiella ornithinolytica (currently called Raoultella ornithinolytica).  In addition to the OR debridement, the patient was treated with 6 weeks of IV antibiotics.

Keywords: gas gangrene, osteomyelitis, amputation

ISSN 1941-6806
doi: 10.3827/faoj.2018.1104.0001

1 – Staff Podiatrist, Department of Podiatric Services, Department of Veterans Affairs Medical Center, Lebanon, PA
2 – Chief Podiatric Resident, Department of Podiatric Services, Department of Veterans Affairs Medical Center, Lebanon, PA
3 – Staff Podiatrist, Department of Veterans Affairs Medical Center, Lebanon, PA
* – Corresponding author: EDWARD.MIRIGLIANO2@va.gov

Gas gangrene is a form of tissue necrosis that can be life-threatening. It often occurs at the site of trauma, or recent surgical site, however, can present without an irritating event. Populations at high risk for developing gas gangrene include those with diabetes and atherosclerosis. If suspicion for gangrene is present, it is imperative to obtain tissue and fluid cultures, blood cultures, x-ray, CT scan or MRI of the area. Surgery should be performed quickly to remove dead and infected tissue. Although it is well established that Clostridium spp. causes gas gangrene; non-clostridial involvement is possible. In the present case, x-ray findings indicated gas formation and additionally, Klebsiella ornithinolytica was recovered from surgical specimens. Based on both radiological and microbiological evidence, the diagnosis of Klebsiella Ornithinolytica gas gangrene was established. The patient was treated for 6 weeks with IV antibiotics.

Case Study

A 56-year-old Caucasian male presented to the ER with a 3-day history of foul-smelling discharge from his right foot after having a 5th met resection 2 months ago at a civilian facility. The patient was having constant pain in the foot over the past 2 weeks. Over the last 3 days, he also noticed a redness that was going up the leg along with bluish discoloration of the fourth digit accompanied by edema. There was tenderness of the 4th digit on palpation along with the metatarsal head dorsally despite patients self-described neuropathy of his feet. The patient said that over the last day he had developed fever, chills, and nausea. His medical history was significant for uncontrolled diabetes, hypertension, cocaine abuse, and tobacco abuse.

Figure 1 Demonstrating Gas Gangrene of 4th proximal phalanx.

Figure 2 Post-operative radiographs of Partial 4th ray resection.

Figure 3 Post-operative radiographs of transmetatarsal amputation.

The patient had a 5th ray resection from an outside facility and was unable to give us any other details nor records from that hospitalization.

Inspection at the time of his first visit revealed a 3.5cm x 1.5 cm opening to the fifth metatarsal resection surgical site of the right foot.  There was surrounding erythema to the surgical site and forefoot with lymphangitis streaking accompanied by a purplish discoloration to the fourth digit with pain to palpation.  Also noted were calor and malodor. The dorsum of the foot was erythematous and edematous over the 4th digit and fourth metatarsal. There was a local increase in skin temperature on the dorsal aspect of the left foot compared to that of the contralateral foot. The dorsalis pedis and tibialis posterior pulses were easily palpated and capillary return was within normal limits. His initial temporal temperature was 100.7. The inguinal lymph node palpation was negative. The chest radiograph obtained displayed   no evidence of an acute cardiopulmonary abnormality. Significant lab findings were an erythrocyte sedimentation rate of 37, white blood cell (WBC) count of 16.8, Glucose of 305 and cultures of the wound obtained in the emergency room revealed Klebsiella ornithinolytica, Enterococcus faecalis and Streptococcus anginosus. Blood cultures were taken in the emergency room and the results were negative.

Routine radiographs performed at our center showed lucency through the medial base of the residual 5th metatarsal could represent a nondisplaced fracture or residual osteomyelitis within the bone.  Also seen was soft tissue air consistent with gas was seen medial to the 4th proximal phalanx. There was no adjacent osseous erosive change to suggest osteomyelitis within the 4th toe (Figure 1). Because of the presence of gas on the x-ray, the plan was to bring the patient the same day to the operating room for resection of 4th ray and incision and drainage of the same area without closure followed by IV antibiotics.

Under general anesthesia and with the use of an ankle tourniquet, utilizing a 4-cm racket incision that incorporated the existing lateral surgical incision, the fourth digit was disarticulated at the fourth metatarsophalangeal joint and sent to pathology.  Deep tissue cultures were obtained in this area. Necrotic tissue was noted to the plantar aspect, and there was a foul smell noted without purulence. All necrotic tissue was removed and the distal aspect of the fourth metatarsal was freed of all soft tissue attachments and utilizing a sagittal saw, the bone was resected at the mid-shaft of the fourth metatarsal. The resected bone was sent to pathology.  The remaining bone of the fourth metatarsal was noted to be firm, and there was no surrounding necrotic tissue. The surrounding areas were probed, and no abscesses were noted. The operative site was copiously lavaged with Betadine-infused saline. Betadine-soaked Iodoform gauze packing was used to fill the void in the 4th metatarsal with a 3cm area remaining opened for drainage. After surgery, he was placed on IV antibiotics that included Zosyn 3.375 gram every 6 hours and Vancomycin 1 gram every 12 hours for the suspected osteomyelitis. Pathologically, the examination revealed acute osteomyelitis of the toe and metatarsal head with the bone margin free from osteomyelitis and the soft tissue margins of amputation were free from acute inflammation. Cultures taken intra-operatively displayed Klebsiella ornithinolytica [1,2,3,4,5,6,7,8] which was susceptible to Zosyn.

For the next 6 days, the patient stayed as an in-patient without complications while the pain diminished, constitutional symptoms of infection disappeared and WBC improved to 8.8. He was discharged after 6 days and sent home on Augmentin for two weeks and seen as an outpatient in the podiatry clinic. Over the next 6 weeks, the patient had no local or constitutional signs of infection while the incision site closed.

Discussion

Raoultella ornithinolytica (formerly Klebsiella ornithinolytica) is a Gram-negative aerobic bacillus which belongs to the family Enterobacteriaceae. However, human infections caused by bacteria of the genus Raoultella are uncommon. A search of the available literature shows only a handful of documented infections with this presentation.  It is likely due to the patient’s history of poorly controlled diabetes, with a history of cocaine and continued tobacco use, that caused a compromise to his immune system. Due to the dusky appearance of his fourth digit as well as gas on the radiographs, it was medically necessary for a partial ray resection of the fourth metatarsal. After the operative procedure, the patient was started on IV vancomycin and IV Zosyn.  After cultures returned patient was switched to Augmentin.

We have described an unusual presentation of bony involvement with soft tissue gas which was a result of an unusual organism. Though infection is rare, Raoultella ornithinolytica can cause significant and possibly limb and life-threatening infection.  As previously stated there are only a handful of cases where this level of destruction has been noted due to this particular organism. It is important as clinicians to remember that patients who have an immunocompromised status may encounter organisms that are out of the ordinary and may require increased care.  Even though this organism is rare the treatment for the resulting gas gangrene is straightforward. We are fortunate that this patient presented to the emergency department when he did. We establish a definitive treatment plan in order to prevent a more aggressive amputation. Unfortunately, do to the sequela of this procedure the patient was left with an unstable forefoot which ultimately led to a transmetatarsal amputation.  The transmetatarsal amputation site healed uneventfully.

References   

  1. Walckenaer E, Poirel L, Leflon-Guibout V, et al. Genetic and Biochemical Characterization of the chromosomal class A β-lactamases of Raoultella (formerly Klebsiella) planticola and Raoutella ornithinolytica. Antimicrob Agents Chemother. 2004;48(1):305–312.
  2. Kanki M, Yoda T, Tsukamoto T, Shibata T. Klebseilla pneumoniae produces no histamine: Raoutella plantico and Raoutella ornithinolytica strains are histamine producers. Appl Environ Microbiol. 2002;68(7):3462–3466.
  3. Ferran M, Yébenes M. Flushing associated with scombroid fish poisoning. Dermatol Online J. 2006;12:15.
  4. Morais VP, Daporta MT, Bao AF, Campello MG, Andrés GQ. Enteric fever-like syndrome caused by Raoultella ornithinolytica (Klebsiella ornithinolytica) J Clin Microbiol. 2009;47(3):868–869.
  5. Solak Y, Gul EE, Atalay H, Genc N, Tonbul HZ. A rare human infection of Raoultella ornithinolytica in a diabetic foot lesion. Ann. Saudi Med. 2011;31(1):93–94.
  6. Hadano Y, Tsukahara M, Ito K, Suzuki J, Kawamura I, Kurai H. Roultella ornithinolytica bacteremia in cancer patients: report of three cases. Intern Med. 2012;51(22):3193–3195.
  7. Hostacká A, Klokocníková Antibiotic susceptibility, serum response and surface properties of Klebsiella species. Microbios. 2001;104:115–124.
  8. Hoshide RR, Chung H, Tokeshi J. Emergence of community-acquired extended-spectrum beta-lactamase Escherichia coli (ESBLEC) in Honolulu: A case series of three individuals with community-acquired ESBLEC bacteriuria. Hawaii Med J. 2011;70(9):193–195.

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Fusarium osteomyelitis of the foot in non-immunocompromised patient: A case report

Posted on September 30, 2015 | Comments Off on Fusarium osteomyelitis of the foot in non-immunocompromised patient: A case report

by Dr. P. Ashok Kumar1, Dr. P. Guru Prasad2, Dr. P. Sravya Teja3, Dr M. Satish4, Dr. V. Sri Lakshmi5pdflrg

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

Fusarium is a large genus of filamentous fungi, part of a group often referred to as hyphomycetes, widely distributed in soil and associated with plants. Most species are harmless saprobes, and are relatively abundant members of the soil microbial community. Some species produce mycotoxins in cereal crops that can affect human and animal health if they enter the food chain. Some species may cause a range of opportunistic infections in humans. Human infections are usually precipitated by local / systemic predisposing factors and disseminated infection is associated with impaired immune responses.  Fusarium causes serious morbidity and mortality and may mimic Aspergillosis. Here we report a case of fusarium osteomyelitis in a 40 year old immunocompetent male patient, who is agricultural laborer. In this case, patient is suffering from Fusarium osteomyelitis of the foot.

Keywords Fusarium, osteomyelitis, non-immunocompromised, Sabouraud’s dextrose agar

ISSN 1941-6806
doi: 10.3827/faoj.2015.0803.0003

Address correspondence to: Dr. P. Ashok Kumar
[1] Professor of Orthopaedics, Dept of orthopaedics, King George hospital, Visakhapatnam, Andhra Pradesh, India. drsravyapaleti89@gmail.com
[2] Professor of Dermatology and Venereal medicine,  Dept of  Dermatology and Venereal medicine, King George hospital, Visakhapatnam, Andhra Pradesh, India.
[3] Juniour resident, Dept of Orthopaedics,  King George hospital, Visakhapatnam, Andhra Pradesh, India.
[4] Senior resident, Dept of Orthopaedics, King George hospital, Visakhapatnam, Andhra Pradesh, India.
[5] Junior resident, Dept of Dermatology and Venereal medicine, King George hospital, Visakhapatnam, Andhra Pradesh, India.

Foot infections are among the most common and serious in immunocompromised patients. Osteomyelitis involving the foot is more common in immunocompromised patients such as patients with diabetes mellitus. Fusarium is known to cause skin infections, infections of bone, endophthalmitis and keratitis [1,2]. The main route of acquisition of the pathogen is through direct inoculation / inhalation of spores. Here, we present a case of fusarium osteomyelitis of the foot in a young patient without any comorbid conditions or immunocompromise.

CASE REPORT

A 40-year-old male patient presented with multiple discharging sinuses of the right foot of one year duration.  There is no history of trauma. The patient had a history of pain and swelling which progressed gradually. There were multiple discharging sinuses over the plantar and dorsal aspects of the foot.

Patient was febrile with white granular discharge from the foot. On examination, the foot was swollen with multiple sinuses. There was induration of skin and subcutaneous tissue. There was obliteration of the normal curvature of the foot (Figure 1). The discharged granules were sent to microbiology department for diagnosis confirmation. Plain radiograph of the foot showed multiple lytic lesions of all tarsal bones (Figure 2). There was no evidence of any sclerosis and all the tarsal bones appear coalescent. He had raised ESR of 45mm/1st hour, with leukocytosis. His viral screening and other blood parameters were normal. The patient was kept on antibiotic therapy for 2 weeks for which patient did not respond.

1

Figure 1 Swollen Foot with multiple discharging sinuses.

2

Figure 2 Radiograph showing the gross destruction of tarsal and metatarsal bones.

In India the most common causes of multiple discharging sinuses over foot are mycetoma of foot and tuberculous osteomyelitis. Tuberculosis infection was ruled out as his chest radiographs were normal and his sputum negative for Acid fast bacilli, no bacilli grew on Lowenstein-Jensen medium. The discharging granules were sent for microbiological examination. The fungal culture on Sabouraud’s dextrose agar grew a mold with characteristic pink pigment, which on microscopy had septate macroconidia specific for fusarium genus of mold (Figure 3). With this we had made a diagnosis of fusarium osteomyelitis of the tarsal bones which is a rare entity in non-immunocompromised patients.

3

Figure 3 Microscopic Picture of Fusarium, note macroconidia of Fusarium in LPCB mount (Lacto Phenol Cotton Blue Stain).

The patient was started on oral Itraconazole 200mg twice daily, After 1 week of Itraconazole therapy, local debridement was done. Further Itraconazole was continued for 12 weeks. Our patient responded well on local debridement and oral Itraconazole therapy.  No resection of bones was planned as the patient is able to walk in spite of massive bone destruction.

DISCUSSION

Fusarium species are filamentous fungi present as saprophytes in soil and animals. They can cause two forms of infections, localized and disseminated. Localized infections such as cutaneous infections, onychomycosis, osteomyelitis, and septic arthritis. Local infections are characterized by massive local tissue damage. Disseminated fusarium infections occur in immunocompromised patients and the main portals of entry are respiratory tract, sinuses, and skin [3,4]. Fusarium osteomyelitis can be part of disseminated form in immunocompromised patients [4,5].

Fusarium species have been reported to cause cutaneous infections in patients with diabetes mellitus and renal failure [6,7,8]. Fusarium osteomyelitis has been reported in healthy individuals in whom the predisposing factors were trauma and surgery. Nuovo et al reported a case of fusarium osteomyelitis in a 34 year old healthy male who suffered multiple lower extremities fracture during an automobile accident [9]. Bourguignon et al reported a case of fusarium osteomyelitis in a 7 year old healthy child after a puncture of his right knee with a thorn [10]. Finally, Page et al reported a case of fusarium toe osteomyelitis in a 56 year old black female after surgical resection of the head of proximal phalanx [11].

Our patient was a healthy male without any history of trauma. The diagnosis of fusarium infection may be made on histopathology, fungal culture, blood culture, or serology and doesn’t need any special investigations.

CONCLUSION

The fusarium species which have emerged as human infectious agents emphasize the need for correct etiological identification allowing for appropriate treatment. Though, fusarium species is a plant pathogen, it is an opportunistic infection in immunocompromised patients. Here we present this case because of its rarity in a young healthy patient. As such the fusarium species is uncommon even in immunocompromised patients and much rarer or not reported in the literature.

REFERENCES  

  1. Hospenthal DR. Uncommon fungi. In:Mandell GL, Bennett JL, Dolin R editors, principles and Practices of Infectious Disease, 6th ed, Chapter 267. Vol 2, 2005. p.3068-79.
  2. Mangini C, de Camargo B. Fungal infection due to Fusarium (spp) in children with refractory hematologic malignancies. Med Pediatr Oncol. 1999;32(2):149-50. [PubMed]
  3. Nelson PE, Dignani MC, Anaissie EJ. Taxonomy, biology, and clinical aspects of Fusarium species. Clin Microbiol Rev. 1994;7(4):479-504. [PubMed]
  4. Boutati EI, Anaissie EJ. Fusarium, a significant emerging pathogen in patients with hematologic malignancy: ten years’ experience at a cancer center and implications for management. Blood. 1997;90(3):999-1008. [PubMed]
  5. Brint JM, Flynn PM, Pearson TA, Pui CH. Disseminated fusariosis involving bone in an adolescent with leukemia. Pediatr Infect Dis J. 1992;11(11):965-8. [PubMed]
  6. Nucci M, Anaissie E. Cutaneous infection by Fusarium species in healthy and immunocompromised hosts: implications for diagnosis and management. Clin Infect Dis. 2002;35(8):909-20. [PubMed]
  7. Van dijk E, Van den berg WH, Landwehr AJ. Fusarium solani infection of a hypertensive leg ulcer in a diabetic. Mykosen. 1980;23(11):603-6. [PubMed]
  8. English MP. Invasion of the skin by filamentous non-dermatophyte fungi. Br J Dermatol. 1968;80(5):282-6. [PubMed]
  9. Nuovo MA, Simmonds JE, Chacho MS, Mckitrick JC. Fusarium solani osteomyelitis with probable nosocomial spread. Am J Clin Pathol. 1988;90(6):738-41. [PubMed]
  10. Bourguignon RL, Walsh AF, Flynn JC, Baro C, Spinos E. Fusarium species osteomyelitis. Case report. J Bone Joint Surg Am. 1976;58(5):722-3. [PubMed]
  11. Page JC, Friedlander G, Dockery GL. Postoperative fusarium osteomyelitis. J Foot Surg. 1982;21(3):174-6. [PubMed]

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Acute Multifocal Hematogenous Osteomyelitis in a 13 year-old: A Case Report

Posted on April 29, 2013 | Comments Off on Acute Multifocal Hematogenous Osteomyelitis in a 13 year-old: A Case Report

by M.F. Madu, MD1, T. Klok, MD2, L. Sijbrandy, MD PhD3, J. Bekhof, MD4pdflrg

The Foot and Ankle Online Journal 6 (4): 1

Background: Acute hematogenous osteomyelitis (AHOM) is a serious pyogenic infection that is generally caused by bacteria and is most commonly found in children. A multifocal presentation of the disease is rare, mostly seen in the newborn and only in a small percentage of older children. We report a case of multifocal AHOM in a previously healthy, 13-year old girl, who presented herself with several painful and swollen joints, accompanied by high fever. The history, clinical course, radiologic findings and management rationale are presented in this report.
Methods: A review of the clinical, laboratory, radiological and microbiological data of the patient was done.
Results: Arthrocentesis of a swollen ankle, done under suspicion of septic arthritis, yielded clear and sterile synovial fluid. Magnetic resonance imaging scans of affected joints later confirmed AHOM close to the joint, causing the sterile joint effusion.
Discussion: A lesson to be learned from this case is that in case of a clinically suspected septic arthritis, a negative joint puncture could mean an osteomyelitis close to the joint.

Keywords: AHOM, pediatric, child, acute, multifocal, hematogenous, osteomyelitis, staphylococcal

Accepted: March, 2013
Published: April, 2013

ISSN 1941-6806
doi: 10.3827/faoj.2013.0604.001

Address correspondence to: J. Bekhof, MD, Isala Klinieken, Department of Pediatrics. PO Box 10400, 8000 GK Zwolle, The Netherlands. j.bekhof@isala.nl

1-4Department of Pediatrics, Isala Klinieken, Zwolle, The Netherlands.

Acute hematogenous osteomyelitis (AHOM) is a serious pyogenic infection that is generally caused by bacteria and is most commonly found in children.[1] Usually, antibiotics and the careful use of surgery can effectively treat the disease, but when managed poorly it can be life threatening, or at best, a debilitating and crippling illness.[2] AHOM is frequently caused by S. aureus, which is responsible for over 80-90% of culture positive cases.[5] These cases amount to 20-90% of all cases.[1]

AHOM typically presents with local pain and fever that has lasted for approximately 3 days on presentation in the hospital.[3] It is most commonly found in tubular bones. The femur and tibia are affected in one third of the cases, followed by the humerus.[4,5] Infection of pelvic bones, which was seen in the following case, is rare.[6] Multifocal presentation of acute hematogenous osteomyelitis occurs even less frequently in older children.

hetosteoFig

Figure 1
a. Anterior posterior radiograph of the left ankle, demonstrating soft tissue swelling (arrow) and lytic bone lesions in the distal fibula (arrowheads).
b. Coronal T-1 weighted, fat suppressed magnetic resonance imaging (MRI) scan of the left ankle, showing a large skin and subcutaneous defect (arrow), where incision and drainage of the abscess was performed.
c. The same image after intravenous contrast (gadolinium), showing contrast enhancement in the distal fibula, corresponding with osteomyelitis (arrow). Abscess formation (arrowheads) and edema in the surrounding subcutaneous tissue can be seen.

While a multifocal presentation is common in neonates, where around 50% of the cases involve multiple bones[7], this is unusual in older children, occurring in 6-9% of those cases.[8,9] Multifocal osteomyelitis can also be caused by chronic recurrent multifocal osteomyelitis (CRMO), a rare disease that presents with sterile osteomyelitis with an unclear, presumably autoimmune etiology.[19]

We report an unusual case of multifocal AHOM in a 13-year old girl. The aim of this study is to emphasize that this diagnosis should be considered when a child presents with multifocal bone pain and/or painful joints, resembling a multifocal arthritis.

Case report

A 13 year-old girl was referred to our hospital with several painful joints since two days, accompanied by vomiting and fever. Physical examination showed a swollen right knee and a red, swollen proximal interphalangeal joint of the third digit of the left hand.

The patient was not severely ill with a non-febrile temperature. Blood tests showed a slight elevation of inflammatory and hematological markers: erythrocyte sedimentation rate (ESR) 24 mm/hour, C-reactive protein (CRP) 31 mg/l, serum ferritin 160μg/l, leukocyte count 12.6*109/l, neutrophil count 10.3*109/l.

The working diagnosis was reactive arthritis. The differential diagnosis consisted of juvenile idiopathic arthritis and septic arthritis, although low levels of inflammatory markers and a non-critically ill patient made these diagnoses unlikely. Diclofenac 50 mg was prescribed and the patient was sent home.

Over the next week, joint pain had not subsided and the right shoulder and right side of the pelvis were now involved. Furthermore, during the nighttime, the patient had developed a fever in excess of 40 degrees Celsius (104 degrees Fahrenheit), with a normal temperature during the day. Physical examination showed a swollen, warm and red area superior of the left lateral malleolus, as well as swollen digits 2 and 3 of the left hand. In addition, a painful area on the right iliac crest and slightly swollen knees were seen. Blood tests showed rising inflammatory markers (ESR 90 mm/hour, CRP 158 mg/l, serum ferritin 279 μg/l) and a rising leukocyte (15.2*109/l) and neutrophil count (11.6*109/l). Under suspicion of septic arthritis or osteomyelitis, the patient was admitted to the pediatric ward for further examination and treatment. A blood culture was done and the orthopedic surgeon performed a joint puncture of the ankle, which yielded clear liquid. The patient was treated with indomethacin and intravenous amoxicillin/clavulanic acid 2000/200mg three times daily. Three days after admittance, results of the joint puncture and blood culture were available. No bacterial growth was detected in the synovial fluid. The blood culture was positive for Staphylococcal aureus, after which antibiotics were switched to intravenous flucloxacillin 1000mg, four times daily.

In the meantime, her body temperature had dropped to normal levels; however an increase in pain and swelling of the left ankle was reported, with accompanying rising inflammatory markers (ESR 98 mm/hour, CRP 178 mg/l, neutrophil count 16.1*109/l and leukocyte count 18.7*109/l). Ultrasound of the left ankle showed a fluctuating, hypodense lesion, cranial to the lateral malleolus. Since an abscess was suspected, an incision was performed, after which a large amount of pus was drained. The periosteum had been damaged and underlying bone was visible, but still firm. The wound was thoroughly cleansed and was allowed to heal by second intention.

Further radiological evaluation was performed. Ultrasound of the pelvis showed a fluid collection lateral of the right anterior superior iliac spine (ASIS). Magnetic resonance imaging (MRI) scans of the pelvis showed bone edema in the right ASIS, and high signal intensity after intravenous contrast injection, corresponding with osteomyelitis. Pathological fluid pockets were seen in the soft tissue surrounding the affected bone, with capsular enhancement after contrast injection, corresponding with multiple micro-abscesses. MRI scans of the left ankle showed bone edema in the distal fibula and high signal intensity after intravenous contrast injection, corresponding with osteomyelitis. Pathological fluid pockets with capsular enhancement after contrast injection were also seen, pointing to micro-abscesses in the soft tissue surrounding the distal fibula. (Fig. 1)

Since multiple sites were affected, the presumptive diagnosis was multifocal acute hematogenous osteomyelitis.

Pain and limitation of range of motion gradually decreased over the next six days, with blood tests showing ever decreasing inflammatory markers (ESR 54 mm/hour and CRP 9 mg/l) and leukocyte and neutrophil numbers within the normal range. Three weeks after the onset of symptoms, the patient was discharged from our hospital with oral antibiotic therapy consisting of oral clindamycin 450 mg four times per day, which was to be continued for 4 weeks. At the outpatient clinic she showed complete recovery after 6 weeks.

Discussion

Diagnosis of multifocal AHOM can present a challenge because of the atypical presentation and similarity to septic arthritis, juvenile idiopathic arthritis and/or reactive arthritis. When AHOM presents in the pelvis, diagnosis can be especially difficult because pain is referred to the hip, thigh or abdomen because of the deep localization of the infection.[10] In one study, only 12 out of 82 patients suffering from pelvic AHOM were admitted with the correct diagnosis.[11]

Presenting clinical features of AHOM are pain, fever and in pelvic AHOM, limping. Laboratory findings include elevated inflammatory markers (ESR and CRP), which are found in over 90% of cases.[12] Peripheral white cell count is not a very reliable indicator of AHOM[1], however, white cell count should be performed, as leukemia is a differential diagnosis of AHOM. In this case, septic arthritis, juvenile idiopathic arthritis and reactive arthritis as well as leukemia were considered. Based on the not very ill patient and elevated inflammatory markers, the working diagnosis was reactive arthritis. Leukemia could be ruled out easily, based on the white cell count and differentiation, which is routinely performed in our hospital in similar cases.

Two important imaging modalities in the diagnosis of AHOM are bone scintigraphy and magnetic resonance imaging (MRI). These techniques are complementary. Hot spots on bone scintigraphy can guide high-resolution imaging modalities such as MRI, which have a limited field of view compared to a bone scan[10], but a higher sensitivity and specificity (93% and 96%).[13] Initial MRI is recommended in clearly localized disease, initial bone scintigraphy in diffuse, not clearly localized disease.[10] Since in this case, although disease was multifocal, symptoms were clearly localized, an indication for initial MRI scanning was present.

Bone or abscess aspiration to confirm the diagnosis is strongly recommended by experts. However, this is becoming increasingly controversial because of the high sensitivity and specificity of radiologic techniques to diagnose AHOM and S. aureus as causative pathogen in the majority of cases. Steer, et al., recommend incision and drainage when certain criteria are met, such as delayed presentation, an immunocompromised patient, abscess formation, underlying malignancy or delayed response to antibiotics.[1] In this case, abscess formation was a reason for incision and drainage of the ankle abscesses, which served as a combined interventional and diagnostic approach. Arthrocentesis of the ankle joint, performed under suspicion of septic arthritis yielded sterile synovial fluid.

A lesson to be learned from this is that AHOM close to a joint can clinically mimic septic arthritis. Therefore, when arthrocentesis is performed in case of a clinically suspected septic arthritis and sterile fluid is obtained, the clinician should consider osteomyelitis close to the joint.

Antibiotic therapy is initially based on the most likely pathogen causing AHOM in children, which is S. aureus. Single β-lactamase-resistant penicillins such as flucloxacillin have shown their effectiveness against methicillin-susceptible staphylococci and streptococci.[14] After identification of the organism, antimicrobial therapy can be changed according to bacterial susceptibility. When blood/bone culture turns out negative, empirical antibiotic therapy can be continued, as long as clinical response is observed. Duration of antibiotic therapy in AHOM differs from that in adults. Adults are usually treated with prolonged intravenously therapy (mostly six weeks). Children can be treated for shorter courses and with parenteral-oral sequential therapy[1], in which intravenous administration is followed by oral continuation of antibiotic therapy. The efficacy and safety of this regimen has been proven in numerous studies, some of which measuring bone concentrations of oral antimicrobials.[15,16] The total duration of therapy and timing of the switch to oral therapy differs greatly between studies, but generally, intravenous therapy for 3-7 days and further continuation of oral therapy for 3-4 weeks is most common[17,18], although this has never been thoroughly tested in a randomized controlled trial.[19]

Conclusion

In our opinion, two important lessons can be learned from the aforementioned case. Firstly, alternative diagnoses such as multifocal AHOM should be considered when patients present with atypical symptoms, including bone or joint pain and fever. Secondly, when arthrocentesis is performed under suspicion of septic arthritis and sterile synovial fluid is obtained, this can very well mean AHOM close to the apparently symptomatic joint. This case underscores the importance of careful consideration of different etiological entities and multidisciplinary diagnosis and treatment when a patient presents with atypical symptoms and signs.

References

1.  Steer AC. Carapetis JR. Acute hematogenous osteomyelitis in Children: Recognition and Management. Pediatr Drugs 2004 6: 333-346. [PubMed]
2.  Mollan R, Piggot J. Acute osteomyelitis in children. JBJS  1977 59B: 2-7. [PubMed]
3.  Karwowska A, Davies HD, Jadavji T. Epidemiology and outcome of osteomyelitis in the era of sequential intravenous-oral therapy. Pediatr Infect Dis  1998 17: 1021-1026. [PubMed]
4.  Tröbs R, Möritz R, Bühligen U, Bennek J, Handrick W, Hörmann D, Meier T. Changing pattern of osteomyelitis in infants and children. Pediatr Surg Int 1999 15: 363-372.
5.  Craigen MAC, Watters J, Hackett JS. The changing epidemiology of osteomyelitis in children. JBJS 1992 74B: 541-545. [PubMed]
6.  Corti N, Sennhauser FH, Nadal D, Stauffer UG, Nadal D. Fosfomycin for the initial treatment of acute hematogenous osteomyelitis. Arch Dis Child 2003 88: 512-516. [PubMed]
7.  Nelson JD. Acute osteomyelitis in children. Infect Dis Clin North Am 1990 4: 513-522. [PubMed]
8.  Nixon G. Acute hematogenous osteomyelitis. Pediatr Ann 1976 5: 64-81. [PubMed]
9.  Labbé JL, Peres O, Leclair O, Goulon R, Scemama P, Jourdel F, Menager C, Duparc B, Lacassin F.  Acute osteomyelitis in children: the pathogenesis revisited? Orthopaedics Traum Surg Res 2010 96 268-275. [PubMed]
10.  Weber-Chrysochoou C, Corti N, Goetschel P, Altermatt S, Huisman TA, Berger C. Pelvic osteomyelitis: a diagnostic challenge in children. J Ped Surg 2007 42 553-557. [PubMed]
11.  Mustafa MM, Sáez-Llorens X, McCracken GH Jr, Nelson JD. Acute hematogenous pelvic osteomyelitis in infants and children, Pediatr Infect Dis  1990 9: 416-421. [PubMed]
12.  Unkila-Kallio L, Kallio MJT, Eskola J, Peltola H. Serum C-reactive protein, erythrocyte sedimentation rate and white blood cell count in acute hematogenous osteomyelitis in children. Pediatrics 1994 93: 59-62.  [PubMed]
13.  Davidson D, Letts M, Khoshhal K. Pelvic osteomyelitis in children: a comparison of decades from 1980-1989 with 1990- 200. J Pediatr Orthop 2003 23: 514- 521. [PubMed]
14.  Krogdstad P, Smith AL. Osteomyelitis and septic arthritis, Textbook of pediatric infectious diseases. 4th Edition. Philadelphia (PA), WB Saunders, 1998.
15.  Bryson YJ, Connor JD, LeClerc M, Giammona ST.
High dose oral dicloxacillin treatment of acute staphylococcal osteomyelitis in children. J Pediatr 1979 94: 673-675. [PubMed]
16.  Feigin RD, Pickering LK, Anderson D, Keeney RE, Shackleford PG. Clindamycin treatment of osteomyelitis and septic arthritis in children. Pediatrics 1975 55: 213-223. [PubMed]
17.  Peltola H, Unkila-Kallio L, Kallio MJ. Simplified treatment of acute staphylococcal osteomyelitis of childhood, Pediatrics 1997 99: 846-850. [PubMed]
18.  Syrogiannopolous GA, Nelson JD. Duration of antimicrobial therapy for acute suppurative osteoarticular infections.  Lancet 1988 I (8575-6): 37-40. [PubMed]
19.  Ferguson PJ, Sandu M. Current understanding of the pathogenesis and management of chronic recurrent multifocal osteomyelitis. Curr Rheumatol Rep 2012 14: 130-141. [PubMed]

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15 Diagnoses, 9 Surgical Procedures, 1 Device – Multiple Applications of the MiniRail

Posted on August 1, 2012 | Comments Off on 15 Diagnoses, 9 Surgical Procedures, 1 Device – Multiple Applications of the MiniRail

by Thomas Merrill, DPM1, Mario Cala, DPM2, Victor Herrera, DPM3, Alan E. Sotelo DPM4

The Foot and Ankle Online Journal 5 (8): 2

The last decade has seen an increase in the successful application and use of external fixators. More complex procedures involving the ankle and leg require Ilizarov circular or ring fixators for stability and strength. Procedures involving the forefoot and rearfoot (excluding the ankle) do very well with the application of MiniRail fixators. The use of Unilateral MiniRail External Fixation System has grown in popularity over the last decade and has seen a high degree of success. From 2009 to 2011 and application of 29 MiniRail External Fixators were placed on 26 patients ranging in age from 23 to 79. A total of 9 different procedures were performed on the population with 15 different diagnoses. Mini-Rail External Fixators have also been found to be successful in the presence of comorbidities such as in patients with diabetes mellitus, smokers, osteomyelitis and avascular necrosis and have been shown to be definitive in cases where internal fixation has failed. All patients went on to full recovery with no complications or recurrences.

Key words: MiniRail, external fixator, Charcot arthropathy, osteomyelitis, nonunion

Accepted: July, 2012

Published: August, 2012

ISSN 1941-6806
doi: 10.3827/faoj.2012.0508.0002

Unilateral MiniRail External Fixation System is a device used in the treatment of bone conditions and deformities of the foot. In the last decade this application has evolved and become more popular with the development of systems that allow a greater understanding of this technique. External fixation is divided into two main categories: circular frames and unilateral rails;[1,2] however the use of Ilizarov-type circular frames is reserved for more complex deformities in the foot and ankle as well as the distal leg. MiniRail external fixators have been described in the use of a variety of procedures, more commonly used in forefoot surgery.[1,3,4] In this paper we put together a total of nine different surgical procedures used to treat fifteen different foot conditions and deformities as well as trauma. All conditions were treated with unilateral external fixation system with excellent results. These procedures include arthrodesis of the first metatarsal cuneiform joint (with two revisions of this procedure not previously using a MiniRail), medial column fusion, open and closed reduction of Lisfranc fracture-dislocation injuries, metatarsal callus distraction, correction of first and fifth metatarsal fractures, sliding calcaneal osteotomy, first metatarsal cuneiform fusion and first metatarsal phalangeal joint fusion.

External fixation systems have been shown to be advantageous over internal fixation for various reasons. Placement of an external fixator is done percutaneously thus eliminating any unnecessary incisions and risk of infection.[1] Dehiscence and the need for wound care are thus prevented. Associated treatments such as dressing changes, skin grafting, bone grafting and irrigation are possible without disturbing the correction or fixation. Any post-operative condition that may arise such as ulcerations or pin-track infections may be easily accessed and cared for; a luxury not enjoyed by a plate or screw. External fixators can also be applied in the presence of a bone infection. The percutaneous placement of pins eliminates guesswork involved in deciding how to correct the deformity since the pins can be placed at a safe distance from the infection. External fixators can also be safely used in the presence of comorbidities such as diabetes mellitus, smokers, osteomyelitis and avascular necrosis.[1-3]

As with any external fixation system, early weight bearing is not only allowable but encouraged as this expedites bone healing. Immediate motion of the proximal and distal joints is also allowed aiding in the reduction of edema and preventing capsular fibrosis, joint stiffening, muscle atrophy and osteoporosis. When it comes to multi-planar deformities, external fixators provide neutralization and stabilization with adjustable amounts of compression or distraction. This allows correction (compression or distraction) throughout the post-operative period through a minimally invasive procedure and a multifunctional correction.[1,2] Finally, once the desired correction has been achieved, the pins are removed and the patient is left with no internal hardware that may cause pain in the future.

Disadvantages of the unilateral MiniRail system are mainly due to the complexity of the system and difficulty in application and manipulation. This difficulty can be overcome, as with any technical and mechanical difficulty, through surgeon education, training and experience. Another disadvantage of external fixator systems is the cost of the equipment, including the tools needed for application and removal. Although major incisions are avoided as well as placement of internal hardware, the risk of pin track infection and possible neurovascular damage continue to be a realistic risk.[1-3] As stated earlier however, most of these problems can be solved by early detection, quick action and by surgeon education and experience.

Materials and Methods

A total of 29 Orthofix MiniRail external fixators were placed on 26 patients with 15 different diagnoses who underwent 9 different surgical procedures. The patients ranged in age from 23 to 79 years with 8 male (30%) and 18 female (70%) patients. Each patient was educated at length about both internal and external fixation. All advantages and disadvantages including complications as well as recovery time and weight bearing status after surgery were discussed with the patients in detail. All patients who opted for the MiniRail external fixator received pre-operative and post-operative instructions for careful management of the MiniRail. All patients received prophylactic intravenous antibiotic therapy 30 minutes pre-operative and post-operative weekly pin care (cleansing and dressing changes).

In the study, compression-stabilization techniques were used in 26 out of the 29 procedures, within these cases 20 were Arthrodesis and 5 were fracture management techniques. One sliding osteotomy with fixation and 3 distraction- stabilization procedures were also performed (Table 1). All patients had weekly post-operative adjustments of the mini-rail except for the callus distraction patient who performed his own adjustments daily. Intra-operative x-rays were performed to confirm position and stabilization with follow-up x-rays performed at 3 weeks and 8 weeks post-operative. The average post-operative period with the MiniRail was 8 weeks with weight bearing beginning as early as one week post-operative with the aid of a surgical shoe and crutches.

Ten Lapidus fusions were performed with 4 pins placed perpendicular to the long axis of the bone: 2 in the medial cuneiform and 2 in the shaft of the first metatarsal. Prior to pin insertion, the first metatarsal cuneiform joint was prepared under fluoroscopy with temporary fixation through the use of 0.45 Kirschner wire. After placement of the MiniRail, compression was then achieved with an Allen wrench.

     

Figure 1 Examples pre-operative evaluation of 1st and 2nd metatarsophalangeal joint (MPJ) with varus deformity (A and B). First MPJ fusion with MiniRail (C and D). Post-operative evaluation after 1st MPJ fusion and external fixator removal (E and F).

Two revisional Lapidus fusions were performed after failed procedures with internal fixation resulted in non-union. The screws were removed and the joint was prepared once again for the Lapidus procedure described above. Three medial column fusions were performed with a talo-navicular joint fusion involving the use of 2 pins in each bone and compression through the rail with early weight bearing after 1 week and post-operative adjustments made every other week.

Table 1 Summary of diagnoses and procedures performed.

 

Figure 2 Example pre-operative (A) and Post-operative (B) radiograph evaluation of metatarsal cuneiform fusion and brachymetatarsia deformity.

 

Figure 3 Example metatarsal cuneiform joint fusion.

Three Lisfranc’s fracture-dislocations, two fifth metatarsal fractures and one first metatarsal fracture-dislocation were reduced with a total of 6 MiniRails with compression through fracture defect. Five first metatarsal-phalangeal joint fusions were performed with 4 pin compression at the joint through the neck of the first of the metatarsal and the first proximal phalanx.

Two Brachymetatarsia callus distraction procedures were performed with MiniRail placement along the metatarsal shaft. Daily adjustments of the MiniRail were performed for callus distraction by the patient at home. One sliding Calcaneal osteotomy procedure was performed and a MiniRail external fixator was used for compression and stabilization of the osteotomy.

   

Figure 4 Example midfoot osteoarthritis.

 

Figure 5 Example brachymetatarsia with MiniRail external fixator and k-wire fixation.

Results

Of the 29 procedures, all patients went on to full recovery with no complications or recurrence. The average time of duration with MiniRail external fixator was 8 weeks with removal at that time +/- one week. After removal of the MiniRail external fixator, all patients had an average recovery period of approximately 3 weeks at which time patients were allowed to transition out of their post-operative shoes and into athletic shoes. By one month following removal patients were cleared to return to all normal pre-operative activity without restrictions. Physical therapy was highly recommended to all patients to regain muscle strength and balance and averaged 3 weekly physical therapy sessions for 3 weeks. Most patients were allowed to begin physical therapy a week after removal of the external fixator. To date there have no recurrences and patient satisfaction has been overall positive with results. There was no need for further corrective procedures and all patients went on to full recovery.

Discussion

The use of external fixation devices has been in practice for many years. Today the use of external fixators has become a popular methodology for treating a great variety of conditions with minimally invasive procedures. While the larger ring fixators are reserved for more complex conditions (ankle fractures, limb lengthening, Charcot reconstructions), MiniRail external fixators have been a staple for the minimally invasive surgical correction of various forefoot and midfoot conditions as well as some Calcaneal and rear foot conditions.

  

Figure 6 Example Lisfranc fracture dislocation pre-operative (A) and post-operative (B), and follow-up evaluation (C).

 

Figure 7 Example fifth metatarsal fracture non-union repair using MiniRail.

   

Figure 8 Example Tarsal coalition fixed with MiniRail.

In this study, 26 patients underwent a total of 9 different surgical procedures with application of 29 MiniRail external fixators to correct conditions in 15 different diagnostic categories. All patients received pre-operative education and weekly post-operative adjustments and pin care with follow up x-rays at 3 and 8 weeks. The patient population ranged in age from 23-79 years of age with females outnumbering males 18 to 8 respectively.

Since 2009 we have found MiniRail external fixators to be superior over internal fixation for the various reasons listed above. The success rate is exceptionally high with patients able to ambulate very early after surgery and the ability to perform any necessary adjustments post-operative make the MiniRail system a very useful device.[4,5] It should also be noted that satisfaction is overall very positive considering they are able to ambulate early on and they have the peace of mind knowing that any correction needed can be easily adjusted at any time. Patient complaints are minimal and are generally geared toward the bulky dressings and uncomfortable post-operative shoe gear however any complaints of pain or discomfort are virtually non-existent. Finally, it is of importance to note once again that this device can be safely and successfully used on patients with comorbidities that would otherwise lead failure with internal fixation such as patients who are smokers, have bone infections or suffer from chronic system illnesses such as diabetes mellitus.[5,6]

We will continue to use MiniRail external fixators for future cases and hope to broaden the scope of indication for the device. Although this study has a very small sample population, the degree of success we have experienced thus far will propel us forward to continue.

References

1. LaBianco GL, Vito GR, Rush SM. External fixation. In: Banks AS, Downey MS, Martin DE, Miller SJ, (editors). McGlamry’s Comprehensive Textbook of Foot and Ankle Surgery. Vol 1. 3rd edition. Philadelphia: Lippincott, Williams & Wilkins; 2001. p. 107–38.
2. Seibert FJ, Fankhauser F, Elliott B, Stockenhuber N, Peicha G. External fixation in trauma of the foot and ankle. Clinics in Podiatric Medicine and Surgery 20(1): 159-180, 2003. [PubMed]
3. Treadwell JR. Rail external fixation for stabilization of closing base wedge osteotomies and Lapidus procedures: a retrospective analysis of sixteen cases. J Foot Ankle Surg 2005 44: 429-436. [Website]
4. Hamilton GA, Mullins S, Schuberth JM, Rush SM, Ford L. Revision Lapidus arthrodesis: rate of union in 17 cases. J Foot Ankle Surg 2007 46: 447-450. [PubMed]
5. Gamble J, Decker S, Abrams RC: Short first ray as a complication of multiple metatarsal osteotomies. Clin Orthop 1982 164: 241–244. [PubMed]
6. Levine SE, Davidson RS, Dormans JP, et al: Distraction osteogenesis for congenitally short lesser metatarsals. Foot Ankle Int 1995 16:196-200. [PubMed]

Address correspondence to: Thomas Merrill, DPM, Barry University/ Mercy Hospital, Miami, FL

1Diplomate, American Board of Podiatric Surgery
2Sport Medicine Fellow at Barry University/ Mercy Hospital, Miami, FL
3Senior Resident at Barry University/ Mercy Hospital, Miami, FL
4Resident at Barry University/ Mercy Hospital, Miami, FL

© The Foot and Ankle Online Journal, 2012

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Dextrose Prolotherapy Treatment for Unresolved “Morton’s Neuroma” Pain

Posted on June 1, 2012 | Comments Off on Dextrose Prolotherapy Treatment for Unresolved “Morton’s Neuroma” Pain

by Ross A. Hauser, MD1, Wayne A. Feister2, DO, Debra K. Brinker, RN3

The Foot and Ankle Online Journal 5 (6): 1

This study investigates the effectiveness of Dextrose Prolotherapy injections on a group of patients with “Morton’s neuroma.” These patients had failed previous conservative therapies, including surgical and non-surgical procedures as well as steroid injections. In this study, seventeen patients with neuroma pain were treated for six months. Every month, 10 to 20 injections containing 0.5 to 1 milliliter of Dextrose solution were given based on patient response. Pre- and post-treatment surveys utilized both objective data (i.e., solutions used, length and number of treatments, etc.) and subjective data (post-treatment visual analog scale or VAS ratings of pain relief/reduction). The results of this short-term study suggest that Prolotherapy, using injections of Dextrose into weakened ligaments, tendons, and joints, is a promising option among current treatment choices. Prolotherapy works by stimulating the body to repair these soft tissues. Future studies must confirm not only the efficacy but also the reduced risks of Dextrose Prolotherapy for one of the most common foot ailments.

Key words: Morton’s neuroma, neuralgia, metatarsalgia, paresthesias, intermetatarsal bursitis, inflammatory arthritis, osteomyelitis, rheumatoid arthritis, localized vasculitis, ischemia, tarsal tunnel syndrome, peripheral neuritis, synovitis, tendonitis, avascular necrosis, metatarsophalangeal joint capsulitis, Hackett-Hemwall Dextrose Prolotherapy

Accepted: March, 2012
Published: June, 2012

ISSN 1941-6806
doi: 10.3827/faoj.2012.0506.0001

Morton’s neuroma (MN) is a painful condition that affects the ball of the foot. First described in the 1800s, this affliction continues to be a common cause of forefoot pain [1]. Seemingly benign, MN pain can cause extreme discomfort, making it difficult to walk. Those affected become so cautious that they are afraid to place the afflicted foot (or feet) on the ground to take a step.

The word “neuroma” suggests a tumor of the nerve; however, the term is actually a misnomer since the condition is not necessarily an abnormal growth of the nerve [2, 3]. Also, the term neuroma does not describe what is seen with a microscope. Over time, other terms have been used to describe aspects of this pathology. Based on the shape, size, and structure (morphology) of tissues noted under the microscope, other terms may apply: perineural fibrosis, endoneural edema, neurofibromata, angioneurofibromata, local demyelination, and local vascular degeneration [4, 5] (Fig. 1).

Figure 1 Possible tissue pathologies that explain interdigital pain.

What circumstances give rise to the onset of neuromas in the foot? Chronic irritation, trauma, or excessive motion induces a severe, intermittent pain between a pair of the five metatarsal heads in the bones of the fore foot. MN pain may then radiate through the nerves to the tip of the toes [6]. The shooting pain follows a path from that web space to the touching halves of adjacent toes. Seen most commonly in the second and third web space—any interdigital space between toes can be affected [7-9] (Fig. 2).

Figure 2 Interdigital spaces.

Typical symptoms in the region of the intermetatarsal spaces include sharp pains, burning sensations, and paresthesias (abnormal sensation) with weight-bearing activity. (Fig. 3) In fact, the sensation is often described as walking with a stone in the shoe or on a folded or creased sock. As the condition progresses, the pain becomes debilitating; and walking becomes more apprehensive, even to an observer. Noting these typical symptoms, an accurate diagnosis can then be made after a thorough review of the patient’s history and a physical assessment.

Figure 3 Inflamed interdigital nerve.

Evidence on the frequency of this condition is minimal; however, a foot clinic computed the incidence of patients diagnosed with a “neuroma” at a rate of 9.3% of 4000 patients who complained of foot pain [10-12]. Although neuromas in both feet and multiple neuromas in one foot occur, both conditions are rare [13, 14]. Furthermore, neuromas are seen among patients of all ages; even so, they are more prevalent in middle-aged adults [15-17]. The condition most often affects women who frequently wear pointed, high-heeled, close-toed, ill-fitting shoes poorly designed for foot mechanics [18]. Footwear that transfers body weight to the metatarsal heads may be the reason women suffer from MN more frequently than men at a documented rate of eighteen to one [19]. The pain generally intensifies with walking, weight-bearing movement, and tight-fitting shoes.

The discomfort, however, eases with rest and the removal or change of footwear [20, 21] At the onset of the condition, additional relief may be gained by removing the shoe, massaging the foot, and wiggling the toes.

The etiology (cause) of Morton’s neuroma is controversial. A longstanding entrapment theory maintains that the third digital nerve, which is large and formed by a branch of the medial and lateral plantar nerves, is compromised by mechanical irritation. With dorsiflexion—when the toes or foot are bent upward toward the nose—the unyielding transverse ligament fixates the proximal end of the digital nerve [22, 23] (Fig. 4). However, this pinching does not always occur in one nerve; other intermetatarsal spaces can be affected [24]. Since it is not a true neuroma (tumorous nerve), some refer to the condition as Morton’s metatarsalgia [25]. Metatarsalgia is pain related to the metatarsal bones of the foot [26]. Another explanation for the pain is an ischemia or lack of blood flow through the plantar digital artery, which precedes a fibrous thickening around the nerve, called a perineural fibrosis [27]. In addition, a pathophysiological theory for MN claims that the intermetatarsal bursa—distally located to the transverse metatarsal ligament and close to the neurovascular bundles—is irritated. Thus inflamed, secondary fibrosis in the bursa can lead to the symptoms of neuroma. Lateral compression of the foot will then invariably cause pain, probably due to the inflamed bursa—not the nerve—being squeezed between the metatarsal heads [28] (Fig. 4).

Figure 4 Cross sectional view of the fore foot displaying the interdigital points of irritation/inflammation.

The inflamed and enlarged bursa causes a click when the metatarsals are squeezed. This distinctive click, called “Mulder sign,” can be used when diagnosing Morton’s neuroma [29].

A clear knowledge of conditions that affect the metatarsal region is critical to making a definitive diagnosis. Initially, possible diagnoses include metatarsal stress fracture, intermetatarsal bursitis, inflammatory arthritis, osteomyelitis, rheumatoid arthritis, localized vasculitis, ischemia, tarsal tunnel syndrome, peripheral neuritis, synovitis, tendonitis, avascular necrosis, metatarsophalangeal joint capsulitis, and others [30-32] (Fig. 5).

Figure 5 Diagnoses to consider when interdigital pain is the main symptom.

Many treatments have been developed for relief of the symptoms of Morton’s neuroma, but initially, non-surgical approaches are preferred. Among these conservative treatments from simple to complex are changing the footwear; avoiding high-heeled shoes; resting the feet; applying ice; elevating the foot; taking anti-inflammatory medications; taping and strapping, padding, and immobilizing the foot; receiving physical therapy; wearing orthotics or other shoe gear; and injecting steroids. When conservative approaches are unsuccessful, surgery is generally sought as the next step. Surgical approaches include resection, transection, decompression, excision of the involved nerve, and cryogenic nerve ablation.

Another conservative treatment for Morton’s neuroma pain is Prolotherapy, which has a longstanding record of success with hypermobility, when joints are unusually loose or abnormally flexible.

If the goal of padding and strapping is to reduce forefoot motion and pain, it is reasonable to utilize a treatment, such as Prolotherapy, that not only reduces hypermobility, but also results in joint stabilization [33].

Additionally, recent studies demonstrate that injection therapy, utilizing 4% sclerosing alcohol, has success rates of 84 – 89% [34-36]. Dextrose Prolotherapy injections will induce a proliferative response without the risk of alcohol infiltrating the surrounding tissue. The overall purpose of this study was to record the outcomes of Dextrose Prolotherapy on a group of patients with Morton’s neuroma in a private pain clinic.

Patients and Methods

In the study, an attending physician treated seventeen patients with Morton’s neuroma at a private medical clinic. All subjects signed a consent form, stating that a minimum of three and a maximum of six monthly treatments might be needed.

To meet the criteria for inclusion in the study, patients had to be at least 18-years-old, to have suffered unresolved Morton’s neuroma at any intermetatarsal space, and to have failed previous conservative treatment.

At the clinic, a search of electronic medical records (EMR) to find patients with the diagnosis of Morton’s neuroma was conducted.

To be included in the study, two criteria were paramount: 1) a diagnosis of Morton’s neuroma, which had to be the primary condition, and 2) a six-month time lapse, since the patient’s last Prolotherapy injections. The search revealed 31 patients diagnosed with Morton’s neuroma; of these, five could not be contacted by phone (three attempts were made before discontinuing phone calls). Two patients chose not to participate.

Five patients were excluded because of multiple foot problems that took priority over Morton’s neuroma: previous surgeries, osteoarthritis, and ankle problems. Two patients were excluded because not enough time had elapsed—at least six months—since their last Prolotherapy session.

Patients selected for the study had to complete preliminary oral, written, and visual surveys. Demographic information was obtained. Then the patient completed a visual analog scale (VAS), which includes ratings of pain at rest; pain with normal activities; pain while walking barefoot; ability to walk distances without pain; as well as stiffness and numbness/burning (Fig. 6). Finally, an assessment interview with clinical staff members collected both subjective and objective data, such as the type and duration of symptoms, previous treatments and tests, limitations to activity, and previous medical opinion.

Figure 6 Questionnaire used by patients to assess levels of pain.

Next, a physical examination determined objective data by checking for the following: the precise location of the pathology or point of maximal tenderness by palpating (light and/or deep touch) the affected web space; the presence or absence of Mulder’s click; and the severe pain that results with lateral compression of the forefoot.

The end of the Prolotherapy treatment was determined when patients indicated a zero to 1 on the pain scale, or their personal goals for pain relief or for the ability to function were met. Although some had little pain, their main goal was to eradicate numbness, which they found disturbing. Some patients wanted to achieve a zero to 1 level of pain while walking, even with level 4 pain while jumping. Therefore, they would stop treatment with a low level of walking discomfort.

Following treatment, interviews and surveys were completed on a monthly basis. Monthly data collection included the total percentage of improvement; VAS score of pain; level of pain intensity; level of stiffness; degree of crepitation (grating sensations from a joint); range of motion; ability to perform the ADLs (activities of daily living) and to exercise the affected body part.

Six months after the last visit, patients were called to obtain information and answered detailed questions. Interviews provided data on the level of foot/toe pain (VAS scale), percent of overall improvement, limitations/improvements in activities and walking, duration of post-treatment pain relief, and assessment of the treatment by the patient.

For data analysis, patient responses were collected, calculated, and compared at three different times: prior to Prolotherapy, during monthly visits, and in phone interviews conducted six months after Prolotherapy. Statistical analysis using Graph Pad Software calculated the paired student t-test before and after Prolotherapy.

Technique

The Hackett-Hemwall technique of Prolotherapy (www.hacketthemwall.org) was used. Each patient received 10 – 20 injections of 15% Dextrose, 0.2% Procaine, and a 10% Sarapin solution, for a total of 10 to 20 cubic centimeters of solution per foot. Each injection consisted of 0.5 to 1 cubic centimeter of solution and used a two-inch, 27-gauge needle. Injected areas were web spaces one through four—with attention given to metatarsophalangeal joints, dorsal and plantar surfaces, and joint capsules and ligaments (Fig. 7). If applicable, patients were advised to reduce or discontinue non-steroidal anti-inflammatory (NSAID), steroidal and narcotic medications, and other therapies. Prolotherapy treatments were discontinued, once a patient reached a clinical resolution of symptoms.

Figure 7 Prolotherapist injecting the third interdigital space with sclerosant solution.

Results

The final study group included 17 patients but 19 feet, since some patients suffered from MN in both feet. Ten right feet and nine left feet were treated. The average age of the 17 patients was 57 years: eleven were women, and six were men.

Before introducing Prolotherapy, study patients reported previous treatments. No one used pain medications for their symptoms. Some patients had tried wide-toed shoes, orthotics, padding, chiropractics, acupuncture, and steroid injections. Some patients had had MRI and radiographic diagnosis. One of seventeen had seen a podiatrist. A physician told three patients that surgery was required, but only one had surgery to remedy the pain on the other foot.

From patient questionnaires, averages were determined for periods of time. The average length of time patients experienced the pain of Morton’s neuroma was 20 months before entering the clinic. Patients received an average of 3.7 Prolotherapy treatments.

The average time of follow-up was 13.3 months. To determine the efficacy of treatments, only those patients with follow-up more than 6 months were included.

Patients’ subjective experience of pain offers the best measure for statistical accuracy. Patients were asked to rate their pain levels on a scale of 0 to 10—with 0 being no pain and 10 being severe crippling pain. All 17 patients reported pain as a symptom. Thus, patients were asked to report pain levels before and after Prolotherapy in these four categories: 1) pain at rest; 2) pain with normal activities; 3) pain with exercise, and 4) pain while walking barefoot.

Concerning 1) pain at rest: prior to Prolotherapy treatment, VAS pain levels averaged 4.68. None of the patients had a starting pain of less than three. After Prolotherapy treatment, VAS pain levels averaged 0.95.

Concerning 2) pain with normal activity and mobility: prior to Prolotherapy treatment, 15 of the 17 participants reported walking with some degree of pain, and a VAS pain level of 6.89. Eleven of 17 patients were unable to walk fifty feet without pain; 14 of 17 could not walk a half-mile without pain. Four of 17 patients reported an inability to walk barefoot. After Prolotherapy, all patients reported improvements in walking without pain, and a VAS pain level of 1.89. Fourteen of the 17 participants walked normally again and rated their pain relief at greater than 74%. Sixteen of the 17 could walk one block or more.

Concerning 3) pain with exercise: prior to Prolotherapy, 15 of the 17 patients reported decreased ability to exercise, and a VAS pain level of 7.27. Of those 15, eight were totally compromised and unable to exercise; five were moderately (only 30 to 60 minutes possible) to severely compromised (only 0 to 30 minutes possible). Nearly half of the patients were totally compromised in their athletic abilities prior to treatment. After Prolotherapy, 5 of the 17 patients reported being able to exercise as much as they wanted without impediments and with satisfaction, with a VAS pain level of 1.73. Other physical improvements occurred, notably, decreases in stiffness and numbness (burning). Thirteen to 14 patients reported a 100% improvement in the activities of daily living that continued to the end of the study. None reported an inability to exercise.

Concerning 4) pain while walking in bare feet: prior to Prolotherapy treatment, 10 of 17 patients could not walk barefooted without severe pain at levels eight, nine, or ten, and an average VAS pain level of 6.47. Furthermore, 12 of 17 patients could walk less than 50 feet before they experienced noticeable pain, with or without shoes. Only 3 of the 17 patients could walk more than a half-mile without pain.

After Prolotherapy, all patients had a pain level of four or less walking barefooted, and a VAS pain level of 1.65. As for walking distances without pain, all patients could walk at least one block or more. One patient was restricted to walking between 50 feet and one block. Among the 19 treated feet of the 17 patients in the study, eighteen feet could manage walking a half-mile or more, eight of the treated feet reported no walking restrictions.

When comparing the four previous categories before and after Prolotherapy, all reached a statistically significant outcome with a paired student t-test of p = <0.0001. This p-value confirms that the numerical results, when compared and tallied, exceed the mathematical probability of mere chance.

Thus, this prospective, non-controlled study demonstrates that Hackett-Hemwall Dextrose Prolotherapy decreases pain and improves the quality of life for patients with Morton’s neuroma, which was unresolved by previous therapies, medications, and interventions. Prolotherapy provided a relief of 74% for 14 out of 17 of the patients. Among the three patients who were told they needed surgery, two patients felt sufficient pain relief with Prolotherapy to avoid surgery. After the study period, patients experienced overall improvement in range of motion, ability to walk and exercise, as well as relief of stiffness and numbness/burning (Fig. 8).

Figure 8 Survey responses before and after Prolotherapy on levels of pain with various activities.

Discussion

This study should not be compared to a clinical trial in which a treatment is studied under controlled conditions. Instead, the projected goal was to document the responses of patients with unresolved Morton’s neuroma pain to the Hackett-Hemwall technique of Dextrose Prolotherapy. Clearly, the study’s strength was the number of quality of life parameters examined. Quality of life conditions—such as the ability to walk and exercise, enhanced range of motion, reduced stiffness, enjoyment of activities of daily life, and reduced levels of pain—are all important factors affecting the person with Morton’s neuroma.

Improvements in a large number of variables were most likely the result of Prolotherapy treatment. There is no medical test to quantify pain relief. However, observable, documented changes—such as the ability to walk or walk barefoot, to exercise, to work, and to use less pain therapies—are valid measures of success for patients whose health and vitality have considerably improved.

This study noted two empirical shortcomings. One is the subjective nature of the data gathered by the most reliable methods available. Surveys, for instance, relied on the patients to rate their pain, stiffness, and degree of disability. A second obvious weakness is the small number of patients involved in the study. However, on the positive side, this small study group made it possible to see results in a relatively short time span.

Many treatments for the relief of Morton’s neuroma symptoms have developed over time. Although conservative, non-surgical, and surgical approaches have been used, their effectiveness as a treatment is variable, often leaving patients with mixed results and questionable improvement.

A review of three trials that involved 121 people was not able to determine the effectiveness of conservative, surgical, and non-surgical interventions because, as the authors noted, there was insufficient evidence and research flaws. For instance, there were only three randomized controlled studies of the various treatments. The authors were also unable to find any studies to identify the incidence or prevalence of this condition. In the review of the three trials, researchers found no evidence to support the use of pronation insoles, which are routinely used as a conservative approach. Furthermore, they found no evidence supporting the effectiveness of corticosteroids (non-surgical); and they gave a poor grade to the surgical approach due to high risks of amputation neuroma (minimum of 20%), painful plantar scars, and postoperative complications [37].

Histomorphological findings are accepted as the gold standard for diagnosing Morton’s neuroma. Of consequence is a histomorphologic study of 23 nerve biopsies from patients with typical Morton’s neuroma symptoms compared to 25 plantar nerve autopsies of individuals with no record of forefoot problems. The study revealed that nerve biopsies from MN patients had the same characteristics as those removed from autopsies. Tissue samples were identical and could not be distinguished one from the other. However, none of the excised tissue in this study was found to be normal; all had the pathological features of fibrotic tissue (thickened, scarred) [38]. Another study found identical histology when comparing Morton’s neuroma and control patients, observing the same fibrotic changes in the symptomatic patients as in the asymptomatic patients [39] From this research , the question arises as to whether the “neuroma” is actually the cause of the condition, caused by other conditions, or present in normal plantar nerves?

Searches with magnetic resonance imaging (MRI) for typical pathologies of Morton’s neuroma did not discover any diagnostic features (symptomatology). In a retrospective study of 85 foot MRI examinations, 33% of patients with no clinical evidence of Morton’s neuroma showed diagnostic “lesions” suggestive of the condition [40] In a study of 70 asymptomatic volunteers, 30% were diagnosed with Morton’s neuromas [41].

In MR imaging after neuroma resection, a neuroma was found in 26% of the asymptomatic and 50% in the symptomatic web spaces [42]. Thus, MRI reveals neuroma-like abnormalities in both symptomatic and asymptomatic patients [43].

Another retrospective study of steroid injections showed a 47% improvement in the recipients [44]. A study gauging symptom relief from a series of corticosteroid injections reported that 30% of the patients attested to total symptom relief [45]. In another study involving 60 patients, the results of conservative treatment were considered poor in 73% of the cases; thus, the authors recommended surgery as the initial treatment of choice [46].

Surgical removal of the neuroma is reported to provide satisfactory relief in 76 – 85% of the patients [47, 48]. Nonetheless, there were exceptions. In a study of 56 patients with excised neuromas, two thirds of the satisfied patients continued to have tenderness at the cut end of the common digital nerve; 75% were still limited in their choice of footwear; and 14% failed to demonstrate any notable improvement. Those who did not respond to surgery continued their pre-surgical use of steroids, lidocaine, and broad-toed shoes [49]. Other complications of surgery include numbness of the affected toes, postoperative infection, tenderness at the incision, keratosis (scarring) of the sole of the foot, recurrence of pain, and an amputation neuroma. Nearly 20% of the patients continued to feel pain after the first surgery, and few found pain relief with additional surgery [50]. In view of these findings, patients should be informed of the possible results of surgery, since adverse outcomes are common.

Patients searching for alternatives to the mainstream medical care are prudent to consider Prolotherapy for reasons of which practitioners of Prolotherapy are aware. First, Prolotherapists know that ligaments need to be tighter, shorter, and stronger. If the intermetatarsal ligament is weak and loose, however, the interdigital nerve rises up between the metatarsal heads where they can be compressed and, thereby, traumatized [51]. Abnormal metatarsal mobility that results from such weakened ligaments inflames the bursa (cushioning sac) between the heads, creating a space into which tissue from the plantar side of the foot can enter and is subsequently pinched by the metatarsal heads [52]. A fibrous build-up can occur when the weak ligaments allow tissue between bones to be rubbed and irritated. Because Prolotherapy strengthens weakened ligaments and connective tissues, it is a viable treatment option.

For Prolotherapists, Morton’s “neuroma” is most likely mechanically-induced from excessive motion between the metatarsals, combined with excessive weight-bearing stress on the forefoot [53]. Hypermobility of the forefoot predisposes a person to this condition, and Prolotherapy injections at the plantar and dorsal structures of the affected metatarsals will benefit the patient [54].

Prolotherapy has a long history of being utilized for unresolved foot and toe pain [55]. In a study of 19 patients with unresolved foot and toe pain, 63% of patients noted 75% pain relief from Prolotherapy [56] In a study undertaken precisely to evaluate the effectiveness of Dextrose Prolotherapy on Morton’s neuroma pain, 16 of the 20 patients with chronic plantar fasciitis who had failed previous conservative treatment reported good to excellent results from the Prolotherapy [57].

As a treatment, Prolotherapy has been utilized for approximately 100 years, with its modern injection protocols being formalized by George S. Hackett, MD in the 1950s [58, 59]. Increasingly popular in the US, Prolotherapy is used nationally and internationally in both alternative (integrative) and allopathic (orthodox) medical practice [60] The treatment is simple. When therapeutic solutions are injected into painful and tender ligaments, tendons, and joints—an inflammation develops, which causes healing cells to proliferate and strengthen damaged ligament, tendon, and joint structures [61], These injections improve both joint stability and biomechanics, ultimately decreasing pain [62]. In this way, Prolotherapy is a safe and practical option for hypermobile joints of the foot that cause persistent pain [63].

Conclusion

While the exact cause of Morton’s neuroma (MN) is still debated, this study confirms that the Hackett-Hemwall technique of Dextrose Prolotherapy not only reduces levels of pain for patients with MN, but also enhances other quality of life concerns. Conventional therapies, on the other hand—rest, weight loss, exercises for muscle strengthening, orthotics, massage therapy, physiotherapy, manipulation, analgesics, non-steroidal anti-inflammatory drugs, anti-depressant medications, trigger point and steroid injections, and various surgical treatments—often result in residual pain for the patients [64-66]. Patients with MN, therefore, are searching for alternative treatments to relieve the pain [67]. Patients unable to find relief with traditional treatments are also hesitant to use options like surgery.

Surgery for Morton’s neuroma, for instance, presents these significant risks: numbness of the affected toe, postoperative infection, incisional soreness, scarring, and recurring stump neuromas [68, 69]. Instead of these traditional options, patients dealing with Morton’s neuroma are now trying Prolotherapy [70]

As a promising option, Prolotherapy—using injections of an irritant—tightens, shortens, and strengthens ligaments, tendons, and joints. Prolotherapy works by stimulating the body to repair these soft tissues. The solution starts and accelerates healing through inflammation, triggering a healing cascade of effects. Initially, fibroblasts—immature cells capable of producing collagen fibers—proliferate. Hence, the term Prolotherapy arose from this observable process. Once collagen forms, it is woven (reticulated) into ligament and tendon tissue. In this manner, Prolotherapy has the potential to stop the disease process.

In some cases, preliminary, anecdotal evidence suggests that Prolotherapy can reverse Morton’s neuroma. In one double-blind animal study over a six-week period, for instance, Prolotherapy was shown to increase ligament mass by 44%, ligament thickness by 27%, and ligament-bone attachment by 28% [71]. In human studies on Prolotherapy, biopsies performed after the completion of Prolotherapy showed significant increases in collagen fiber and ligament diameter of 60% [72, 73]. These finding are especially significant since a potential cause of Morton’s neuroma is weakened ligaments [74, 75].

In this prospective study, the Hackett-Hemwall technique of Dextrose Prolotherapy used on patients averaging 1.5 years of unresolved pain with Morton’s neuroma was shown to improve their quality of life, which continued 13.3 months after their last session. The 17 patients treated with Prolotherapy reported significantly less pain, stiffness, disability, or use of other pain therapies, as well as improvements in walking, range of motion, ability to exercise, and performing activities of daily living.

Patients told that there were no other treatments for pain or that surgery was their only option achieved the same positive results. This study justifies the desirability and use of Prolotherapy for Morton’s neuroma pain. Future studies need to further substantiate these findings, especially if Prolotherapy enables Morton’s neuroma sufferers to avoid surgery and its possible adverse effects. Although a study with more patients in a controlled empirical setting is needed to document the efficacy of Hackett-Hemwall Dextrose Prolotherapy, this treatment should be considered, based on the substantial advantages and minimal drawbacks (e.g., aversion to needles), as well as the reduced risks and increased rewards of Prolotherapy over conventional treatments.

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Address Correspondence to: : Ross Hauser, MD, Caring Medical, 715 Lake St., Suite 600, Oak Park, IL 60301

1Medical Director, Caring Medical & Rehabilitation Services; Editor-in-Chief, Journal of Prolotherapy
2Private Practice, Medical Editor, Ohio University Clinical Assistant Professor, Bowling Green State University Adjunct Assistant Professor
3Registered Nurse, Caring Medical & Rehabilitation Services

© The Foot and Ankle Online Journal, 2012

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Diabetic Limb Salvage in the Septic Ankle: Case Studies of Arthrodesis using the Ilizarov Methodology

Posted on October 1, 2011 | Comments Off on Diabetic Limb Salvage in the Septic Ankle: Case Studies of Arthrodesis using the Ilizarov Methodology

by Sutpal Singh, DPM. FACFAS1 , Albert Kim, DPM2, Timothy Dailey, DPM,3
Long Truong, DPM4, Maria Mejia, DPM5

The Foot and Ankle Online Journal 4 (10): 1

Diabetic patients usually have multiple comorbidities resulting in higher complication rates after ankle fractures. In many cases, the patient, through diabetic complications of peripheral neuropathy, may mistakenly ambulate resulting in dislocation or hardware failure if only internal fixation is utilized. Also, impaired wound healing, infection, non-union, mal-union and development of Charcot foot and ankle arthropathy may ensue. This article will present several cases in which open reduction and internal fixation in diabetic ankle fractures failed which then lead to osteomyelitis. This infection with the presence of diabetic neuropathy results in an increased risk for loss of limb. These cases were ultimately salvaged with septic ankle arthrodesis using the Ilizarov Method.

Key words: Diabetic ankle fracture, osteomyelitis, Limb Salvage, Septic Ankle Arthrodesis, Ilizarov Methodology.

Accepted: September, 2011
Published: October, 2011

ISSN 1941-6806
doi: 10.3827/faoj.2011.0410.0001

Treating diabetic ankle fractures is a very complex task and many times lead to multiple complications. The majority of diabetic patients have comorbidities such as peripheral vascular disease, osteoporosis with poor bone stock that can lead to poor healing potential and complications. A few of the complications encountered are ulcerations and wound dehiscence.

It has been well documented in the literature that diabetic patients with ankle fractures who underwent open reduction and internal fixation developed complications of wound infections, below the knee amputations, Charcot arthropathy, malunions, wound necrosis requiring plastic surgery, and deep sepsis. [1,2] When complications are encountered, often, salvage is managed by ankle arthrodesis. Our treatment protocol is to fuse the ankle using the Ilizarov Method.

When performing an ankle fusion there are a variety of different open surgical approaches to exposing the ankle for fusion as well as arthroscopic ankle fusions.

The open surgical approaches are more commonly used than the arthroscopic option. Of the many different open approaches the more common ones are the medial transmalleolar, lateral transmalleolar, anterior, and posterior approaches. A discussion on the various types of ankle arthodesis will be presented followed by the Ilizarov Method in septic ankle joint arthrodesis.

The medial approach is performed by using an osteotome or oscillating saw to create a transverse medial malleolar osteotomy at the level of the distal tibial articular surface. Next, the medial malleolus is reflected distally on the deltoid hinge, exposing the ankle joint. A power saw is used to resect the tibial plafond perpendicular to long axis of the tibia. Temporary fixation with Steinmann pins can be utilized to maintain the plantigrade position. [3]

The medial approach provides better visualization of the tibiotalar articulation [3,4,5], the surgical exposure obtained is more subcutaneous and gives better access for preparation of the articular surfaces. Neurovascular complications are also decreased by this approach. Finally, the medial approach allows for visualization in placement of a posterior screw which has to be placed blindly when using the lateral approach. [3]

In the lateral approach of ankle arthrodesis, an osteotomy is performed approximately 2 cm proximal to the level of the ankle joint. In this approach care must be taken not to sever the sural nerve. [3,4,5,6] The lateral approach is preferred over an anterior approach in cases with moderate to severe deformity. Therefore in cases where a severe deformity of the ankle joint is not present, an anterior approach is indicated.

In regards with the anterior approach, care is taken to avoid any damage to the terminal branches of the superficial peroneal nerve, the intermediate and the medial dorsal cutaneous nerves due to the course of these nerves under the incision site. [7] The fixation indicated for the anterior approach is composed of at least 2 screws inserted at 30 degrees with respect to the long axis of the tibia. These screws should cross proximal to the fusion site to maximize stability. In certain cases a third screw can be placed to improve sagittal plane stability. [7] A major advantage of the anterior approach is that the osteotomy of the lateral and medial malleoli is avoided. The other approach which is used but is not discussed in the literature as frequently is the posterior approach.

For the posterior approach, Hanson et al., concluded that using a posterior approach with a 95 degree blade plate is effective in large patients with a mild to moderate hindfoot deformity. [8]

In addition to the open techniques, an ankle fusion can be performed with arthroscopy. In the arthroscopic technique various abraders, curettes and other arthroscopic instrument are used to remove the cartilage from the joint surfaces using a camera and small portals through the ankle joint. After removing the cartilage the ankle is reduced into proper position and finally fixated internally with two transmalleolar screws. With arthroscopic fusion it has been shown that the average time to fusion is significantly less, whereas this produces a faster recovery period. The shorter time to fusion is likely a result of the minimal soft tissue stripping that is performed during the procedure. [4,9]

Regardless of whether an open or an arthroscopic fusion is performed, the position of the foot for fusion is the same. In the literature the correct position for fusion is valgus of the posterior foot varying from 0 to 5 degrees with an external rotation of 5 to 10 degrees, sloping slightly posterior talus relative to tibia and neutral flexion position. [1,2,4,5,6] In order to prevent malposition, the foot should be compared to the rest of the leg and the contralateral limb before fusing it.

Once the proper position is found the next concern is fixation. There are various methods of fixation such as compression with an external fixator, internal fixation using plates and screws, intramedullary fixation, and arthroscopic ankle fusion. [1,2]

In difficult cases of ankle arthrodesis and limb salvage the preferred fixation methods are Intramedullary (IM) nail and external fixation. [1,4,5,10,11] Obtaining a solid fusion can be challenging in compromised bony interfaces, and standard techniques of tibiotalar fixation such as crossed lag screws are often inadequate. An advantage that the Ilizarov technique has over IM nailing and the other internal fixation options is it can be used in cases of infection. [3,4] The Ilizarov method also spares the subtalar joint.

Post operative care is comparably the same in almost all the surgical procedures with non weightbearing in a posterior splint followed with a cast for at least 6 weeks, removal of casts depends on healing noted and once healing progresses the patient is placed in a CAM boot. [4,5]

For patients that were treated using the Ilizarov Method, early weight bearing is permitted. When comparing open arthrodesis with arthroscopic fusions, the arthroscopic patients were hospitalized for an average of 1.6 (1-4 day range) days whereas the open group was hospitalized for average of 3.4 days (1-6 day range). [12]

As discussed earlier, complications include malposition, neurovascular complications. Nonunions and amputations can also occur as a complication depending on the surgical approach. Initially they can be treated with prolonged periods of immobilization and minimal weightbearing. In addition, an external bone stimulator can be used. [11] If prolonged immobilization does not help then bone grafting and external fixation are recommended. [1,4,5,11,13,14,15] In cases in which non unions are painful and they are not able to be resolved with repeated surgical options an amputation is many times the only option. [11,13] Also superficial infection of the surgical incision or the pin sites in external fixation methods has been reported as occurring in 40% to 50% in which local wound care is usually sufficient enough. In cases of deeper infections where osteomyelitis is involved the rate of amputation is as high as 50% which happens more so in the case of fusions performed in an existing septic process. [11] This article will present failed open reduction internal fixation (ORIF) in diabetic patients that were salvaged in case of septic ankle fusion using the Ilizarov method in which both medial and lateral incisions were used.

Case Report

Case # 1

The first case is that of a 70 year-old diabetic, neuropathic, cardiomyopathic, liver transplant patient on dialysis. He had a bimalleolar ankle fracture stabilized with internal fixation. (Figs. 1 and 2) He was referred to our service after undergoing multiple surgeries including wound care and skin graft. He had a large ulcer on the medial and lateral ankle on the left lower extremity. His fibular plate was severely bent and the tibia was exposed on the medial side due to noncompliance and ambulation. An external fixator with several tibial screws and one calcaneal transfixation screw was used to temporarily hold the deformity. This was done prior to being transferred to our service. Cultures revealed Methicillin-resistant Staphylococcus aureus (MRSA) with osteomyelitis at the ankle and the patient was on intravenous (IV) antibiotics. After consulting with infectious disease, internal medicine, cardiology and vascular surgery, the patient was given clearance for limb salvage. The patient had only two choices at the time: (1) below the knee amputation or (2) limb salvage. He chose the latter. The treatment plan included wound care, debridement of the ulcer and removal of the necrotic and infected bone and soft tissue. This was performed 1 week prior to surgery. The patient then underwent a septic ankle arthrodesis using the Ilizarov frame as well as rotational flap to close the ulcer. (Figs. 3-5)

Figure 1  Pre-operative radiograph of failed internal fixation in case #1.

Figure 2  Clinical photograph of the exposed distal tibia and calcaneus in case # 1.

Figure 3   Application of the external fixator, closure of the calcaneal ulcer with a Graft Jacket, and rotational flap to cover the tibial wound at the ankle in case #1.

Figure 4  Showing lateral approach in case #1 with the external fixator.  The anteroposterior radiograph (A),  Lateral radiograph (B) and lateral radiograph after removal of wires from the subtalar joint (C) in case # 1.   All radiographs showing complete consolidation of the tibial talar joint.

Figure 5   Clinical photographs 4 months after surgery showing limb salvage with all wound healed with solid bone consolidation in case #1.

Case # 2

This patient is a 380lb, diabetic, neuropathic, cardiomyopathic patient with a malunion. (Fig. 6) He had an unstable ankle fracture at the fibula with complete rupture of the deltoid ligament. (Fig. 7A and 7B) He was stabilized with open reduction and internal fixation. He ambulated several days after the surgery resulting in malunion and widening of the tibial talar joint. He was seen by our service several months after the initial surgery. He had a large open wound down to the medial tibia with purulent drainage coming from the ankle joint. He presented with osteomyelitis of the ankle. Again, he was cleared for limb salvage.

Figure 6   Clinical photographs of open distal tibia with osteomyelitis in case # 2. (Close-up in inset)

 

Figure 7A and 7B   Anteroposterior  (A)  and lateral (B) radiographic views of a failed internal fixation resulting in a diabetic septic ankle.  (Case # 2)

The treatment plan again included wound care, debridement of the ulcer and removal of the necrotic and infected tissue. One week later, he had a septic ankle arthrodesis and subtalar joint arthrodesis using the Ilizarov frame as well as a rotational flap to close the ulcer at the ankle. (Figs. 8A, 8B, 9A, 9B, 10A, 10B, 11 and 12)

 

Figure 8A and 8B   Clinical photographs of septic ankle arthrodesis using the Ilizarov frame.  Medial view: Note closure of the ankle using a rotational flap (A) and anterior view.  This is the second day after surgery for case #2.

 

Figure 9A and 9B   Lateral (A) and anteroposterior (B) radiographic views 2 days after surgery showing ankle and subtalar joint arthrodesis compressed with an Ilizarov circular external fixator in case # 2.

 

Figure 10A and 10B  Medial (A) and lateral (B) views 3 months after tibial-talar-calcaneal fusion with the Ilizarov frame.  The skin on the medial side has completely healed with the rotational flap in case # 2.

Figure 11   The ulcer has completely healed and the Ilizarov external fixator has been removed in case #2.  The foot is very stable and completely fused at the tibial-talar-calcaneal joint.

  

Figures 12  Post operative radiographs showing complete arthrodesis of the tibial-talar-calcaneal joint and stabilization using percutaneous 6.5 mm fusion Synthes bolts after the Ilizarov frame was removed in case #2.

Case # 3

This patient is a 70 year old diabetic, neuropathic who suffered a severe ankle and foot fracture. She is a chronic tobacco abuser smoking 2 packs per day. She had an ORIF of the right ankle and foot. The patient developed a postoperative infection. She was referred to our service for limb salvage. On initial presentation the patient had a tremendous amount of putrid smelling brown pus coming from the medial ankle. (Fig. 13, 14A and 14B) Culture and sensitivity revealed MRSA. She was on IV Vancomycin. She was taken to the OR and an incision and drainage was performed. The necrotic bone and tissue as well as the hardware at the ankle were removed. The wound was then packed with iodoform and she had daily wound care. One week later, when the infection was controlled, she had a septic ankle arthrodesis using the Ilizarov Method. (Figs. 15, 16A, 16B, 17A, 17B)

Figure 13  Clinical photograph showing the diabetic open septic ankle joint.  The toes are to the upper right and the knee is to the upper left.  (Case #3)

 

Figure 14A and 14B   Severe foot and ankle deformity with sepsis at the tibial talar joint and failed hardware. (A) Note the probe in the medial ankle. (B) Putrid smelling brown pus was noted coming from the medial ankle and tracking across the ankle to the lateral mid leg area in  case #3.

Figure 15  Lateral radiographic view of the septic ankle arthrodesis using the Ilizarov frame several weeks after surgery.  There is good alignment of the tibial talar complex.  There is placement of antibiotic beads in the ankle/lower leg area.  (Case #3)

 

Figure 16A and 16B   There is a valgus rotation of the calcaneus relative to the long axis of the tibia.  (A) The forefoot was in neutral position without any varus or valgus. (B) All incisions have healed.  (Case # 3)

 

Figure 17A and 17B  After removal of the external fixator, insertion of internal splinting with Synthes metaphyseal plate, and calcaneal osteotomy with medial translation.  This shows good alignment of the lower extremity.  The tibial talar joint is completely fused. (A)  Lower leg, ankle and hind foot are in good alignment after the medial calcaneal slide osteotomy. (B) (Case #3)

Surgical Technique and Result

Case # 1: Three tibial rings, each 180 mm with several smooth 1.8 mm wires were applied to the proximal tibial segment. Also two half pins were also inserted and attached to the tibial rings. Then a foot plate was applied using the 1.8 mm wires. Note that the tibial rings and foot plate were not connected at this time. An incision was made on the lateral side. The hardware and the distal fibula were removed. On the medial side, the hardware and distal medial tibia were removed. The ulcer on the medial side was debrided and all necrotic tissue was removed. Then the tibial talar joint was resected until there was good apposition and bleeding. The wound was copiously irrigated with 3 liters of normal saline and bacitracin. The foot plate was manipulated to hold the tibial talar joint in good apposition with the second toe in line with the tibial tuberosity. There was no varus, valgus, dorsiflexion or plantar flexion noted. The tibial talar joint was in neutral position. The foot plate is used to move the foot such that the talus is directly under the tibia and not forward or behind the tibia. Several 0.062 Kirschner wires were inserted to hold the tibial talar joint. (Figs. 3 and 4)

Rods were then applied to the foot plate and tibial rings. Compression was applied in an axial direction. There was good alignment and good compression. Another incision was made above the ulcer and a full thickness rotational flap was performed to close the ulcer where the tibia was exposed. There were also two other ulcers noted which were created by the prior transfixation screw through the calcaneus. These ulcers were debrided to good bleeding tissue and then covered with Graft Jacket and sutured with 3-0 ProleneTM. The rest of the surgical sites were closed using 3-0 VicrylTM for deep tissue and 3-0 ProleneTM for the skin as well as skin staples.

The external fixation was left on for three months until consolidation was seen on radiograph. Then the external fixation was removed and the wires going into the subtalar joint was removed. A CROW boot was then dispensed to the patient to protect the limb. The patient then began ambulating with a walker. At six month and one year follow-up, the patient is still ambulating and without any recurrence. (Fig. 5)

Case # 2: Three 200 mm tibial rings were applied to the patient proximal to the open wound on the lower leg. Then 1.8 mm smooth wires were inserted and tensioned appropriately. Then 4 tibial half pins were inserted into the tibia and attached to the tibial rings.

Then a foot plate was applied to the foot with wires and tensioned appropriately. The tibial rings and the foot plate were not connected. Then an incision was made on the medial and lateral ankle. All the necrotic bone, tissue and the hardware were then removed. The tibia and the talus were then resected to good bleeding tissue and good apposition. The lateral incision was also extended to the subtalar joint and the subtalar joint was then denuded of cartilage. The large ulcer on the medial side was debrided and all necrotic tissue was removed. There was an even larger opening on the medial side after the debridement. The surgical site was irrigated with 3 liters of normal saline with bacitracin. The foot plate with the foot was then manipulated in a manner in which the tibia and second toe was in line. The tibial talar joint was in neutral position without varus or valgus. There was no varus, valgus, dorsiflexion or plantar flexion noted. The tibial talar joint was in neutral position. The foot plate is used to move the foot such that the talus is directly under the tibia and not forward or behind the tibia. Then several 0.062 Kirschner wires were inserted from the calcaneus, through the talus and then into the tibia.

Rods were then used to connect the foot plate to the tibial rings and this was then compressed to fuse the tibial-talar-calcaneal joint. Attention was then directed to the medial large ulcer. Another incision was made at the ulcer and a rotational flap was performed so as to close the ulcer. The surgical site was closed with 3-0 vicryl for the deep tissue, and 3-0 ProleneTM and skin staples for the skin.

The external fixator was left in place for three months until good consolidation was noted. The K wires were removed. Because he was morbidly obese, internal splinting with percutaneous 6.5 bolt screws from Synthes were inserted from plantar calcaneus to the tibia. He was also given custom AFO. (Figs. 8A,8B, 9A, 9B, 10A, 10B, 11 and 12) At six month and one year follow-up, the patient is still ambulating and without any recurrence.

Case # 3: The patient had severe abscess at the medial left ankle with the pus tracking laterally up the leg. (Fig. 13) An incision and drainage was performed on the medial and lateral ankle. The infected tissue, bone and hardware were all removed as well at the distal fibula. The surgical site was irrigated copiously with three liters of normal saline and bacitracin using a pulse lavage system. The surgical site was loosely approximated with 3-0 ProleneTM and skin staples. She then had wound care every day including the use of Betadine® soaked iodoform as well as irrigation with one liter of normal saline and bacitracin for 5 days. Once the infection was controlled, she was then taken back to the OR for a septic ankle arthrodesis.

The patient was taken back to the OR and three 180 mm tibial rings were applied to the left lower leg proximal to the infected area. (Fig. 15) The wires were tensioned appropriately and then 2 half pins were applied. Then a foot plate was applied and tensioned appropriately. The tibial talar joint was then resected and then placed in a neutral position without any varus or valgus. There was no varus, valgus, dorsiflexion or plantar flexion noted at the tibial talar joint. Also note that the foot plate is used to move the foot such that the talus is directly under the tibia and not forward or behind the tibia. Her tibial talar joint was in neutral position. This was then stabilized with several 0.062 K wires. The foot plate with the foot was then connected to the tibial rings with several rods. These were then tightened to compress the tibial talar joint. She did have a valgus tilt of the subtalar joint with the heel being laterally located. (Figs. 16A and 16B) Because of the complexity of the deformity, it was decided to perform a medial calcaneal slide osteotomy at a different time until there was complete consolidation of the tibial talar joint. Antibiotic beads of 1 gm of Vancomycin were made and inserted into the lower leg ankle area.

The external fixator was left in place for four months until good consolidation was noted. The K wires were removed. At this time, because of the severe deformity and possibility of recurrence and BKA, internal splinting with a 10-hole 3.5 mm metaphyseal plate and screws spanning the tibial-talar-calcaneal complex was performed. Also, a medial calcaneal slide osteotomy was also performed to have a more rectus foot and in better alignment of the leg and hindfoot. This was performed by making an incision on the lateral calcaneal area. The incision was deepened to the subcutaneous tissue and then to bone. A sagittal saw was used to perform the osteotomy and the calcaneus was translated medially approximately 2 cm and stabilized with several crossing K wires. (Figs. 17A and 17B)

Two months later, the wires in the calcaneus were removed and a custom Arizona brace was dispensed and she was able to ambulate with a walker.

After six months, she developed an ulcer on the plantar right foot. She had a Charcot foot prior to the severe infection on the left foot. This ulcerated after six months but with proper wound care, this ulcer completely healed. She was also dispensed another Arizona brace for the right lower extremity. At two year follow-up, she is doing well and has both of her legs and feet. (Figs. 18,19)

 

Figures 18  Two years after surgery, the radiographs show good alignment and complete arthrodesis of the tibial-talar-calcaneal bones.   The wires in the posterior calcaneus have all been removed in case #3.

Figure 19   Complete healing of the calcaneal osteotomy in anatomic good position after removal of the internal fixation wires in case #3.

Discussion

Multiple studies have noted that open reduction internal fixation in acute diabetic ankles fracture can be devastating. [16,17,18,19,20] Patients with complications associated with diabetes are at an increased risk for higher rates of in hospital mortality, in hospital post operative complications, length of stay and non-routine discharges. [19] Previous studies has shown mortality rate as high as 8.5% and deep infection of 17% associated with complications of diabetic ankle fracture. [18] Even after anatomical reduction with stable internal fixation, the diabetic neuropathic patients may experience complications such as breaking or bending the fibular plate, malunion, nonunion, and charcot arthropathy. After repeat ORIF of the ankle with stacked one-third tubular plates and several syndesmotic screws, failure can occur. It is noted in previous studies that diabetic neuropathic patients are 5 times more likely to need revision surgery when comparing to patient with uncomplicated diabetes. [20] Salvage by tibiotalocalcaneal fusion with intramedullary rod in this population group also failed due to non compliance. This can ultimately resulted in a below the knee amputation. [2,21] In revisions surgery, the fusion rate is noted to be lower than in primary arthrodesis. [22]

Thus, in our case reports, our protocol is to perform ORIF and then to stabilize the lower extremity with an Ilizarov frame. If the patient has peripheral vascular disease, the Ilizarov frame was applied with
very minimal to no internal fixation. If the patient is severely medically compromised, the surgery was performed under IV sedation using a popliteal block, common peroneal block at the neck of the fibula and saphenous nerve block at the level of the tibial tuberosity.

Several surgical techniques are currently accepted for performing primary ankle arthrodesis. These techniques include compression with an external fixator, internal fixation using plates and screws, intramedullary fixation, and arthroscopic ankle fusion. [1,22]

The Ilizarov technique offers several advantages that “traditional” fusion does not offer in patients with complex ankle pathology such as infection, limb-length discrepancy, mal-union, Charcot joints, talar osteonecrosis, and talar absence. [22] Internal fixation and arthroscopic techniques are not suitable methods for infection, bone loss, severe deformities, or failed procedures. [1] Post-operatively, the Ilizarov method allows for adjustments in mechanical control throughout the treatment period that is otherwise impossible with nails, screws, or plates. [1,22]

Potential limitations that can be associated with this technique include pin track problem, the cumbersome frame, and complexity associated with application of the frame. [22]

Pin track infections do occur but usually are managed locally with pin site care and oral antibiotics. However, the advantages outweigh these downfalls. These advantages prove even more invaluable when application is planned for revisions and complex situations. [1]

The Ilizarov technique provides stable fixation and allows application of primary and continuous forces along any axis and direction. [1,22] The dynamic axial fixation maintains bone contact without additional bone grafting and allows excellent bending, shearing, and torsional stability that allows early weightbearing. [1,22] Most patients are bearing partial weight immediately, therefore earlier compression is noted across the surgical site, enhancing fusion rate. [22]

Additionally, due to early ambulation, there is noted improvement in proprioception and reduction in complications such as deep vein thrombosis and deconditioning. [22]

The Ilizarov technique also enables correction in a single plane or in multiple planes. [1,2,22] A well aligned fusion ensures a near normal gait. It is recommended that fusion be position with valgus of the posterior foot varying from 0 to 5 degrees with an external rotation of 5 to 10 degrees, sloping slightly posterior and neutral flexion position. [1,2]

Ankle arthrodesis can be divided by approach as anterior, transmalleolar, or posterior or by method of fixation as external or internal. [1] However, when planning the proper procedure for the high risk diabetic patient, many techniques become less appropriate with frequent complications and difficult to achieve fusion site. In our case reports in this article, we performed septic ankle arthrodesis using the Ilizarov Method for limb salvage. All patients were told that a BKA was eminent. We were able to salvage the limb by the Ilizarov Methodology. It has been noted by Gabriel Ilizarov that osteomyelitis burns in the fury of osteogenesis. Osteogenesis occurs by compression and immobilization of bone using the Ilizarov Methodology. In the presence of infection of the tibiotalar joint, arthrodesis is a reasonable treatment option and in some cases may be the way to prevent amputation at a more proximal level. [2]

Conclusion

The previous cases of diabetic ankle fractures which were fixed with open reduction and internal fixation went on to septic ankle joints. Septic ankle joint is a difficult condition to treat with two viable options limb salvage ankle arthrodesis or below knee amputation. Patients must be aware that ankle arthrodesis may still end up in a BKA. Many different ankle arthrodesis surgical techniques exist with the salvage option.

Each surgeon has his or her preference as to their procedure of choice with each having their advantages and disadvantages. The author’s systematic approach to diabetic ankle fractures is to cast if they are non-displaced, and ORIF with an Ilizarov frame if ankle fracture is displaced. If they go on to a septic ankle joint then the area is debrided and internal hardware is removed and an Ilizarov method is used for ankle arthrodesis. In the authors experience the biggest complication with the Ilizarov frame is pin tract irritations and or infections but these are easily treated by removing the pin and placing a new one. The Ilizarov method is a good option in providing adequate compression and in allowing the patient to bear weight. It is important to follow these patients frequently to make sure the arthrodesis site is healing well and free of infections to prevent a BKA.

References

1. Salem KH, Kinzl L, Schmelz A. Ankle arthrodesis using Ilizarov ring fixators: A review of 22 cases. Foot & Ankle International 2006 27:764-70.
2. Klouche S, El-Masri F, Graff W, Mamoudy P. Arthrodesis with internal fixation of the infected ankle. J Foot & Ankle Surgery 2011 50: 25-30.
3. Schuberth J, Cheung C, Rush S, Blitz N, Roling B. The medial malleolar approach for arthrodesis of the ankle: A report of 13 cases. J of Foot & Ankle Surgery 2005 44:125-132.
4. Easley M. Operative Techniques in Foot and Ankle Surgery. Philadelphia: Lippincott Williams & Wilkins 2011.
5. Coughlin M, Mann R, Saltzman C: Surgery of the Foot and Ankle. Philadelphia. Mosby 2007.
6. Grass R, Rammelt S, Biewener A, Zwipp H: Arthrodesis of the ankle Joint” Clinics Podiatric Medicine Surgery 2004 21:161-178.
7. Karl-Heinz K, Hans-Jörg T, Fusszentrum W. Ankle arthrodesis with an anterior approach. Techniques Foot Ankle Surgery 2007 6: 243-248.
8. Hanson TW, Cracchiolo A 3rd: The use of a 95 degree blade plate and a posterior approach to achieve tibiotalocalcaneal arthrodesis. Foot Ankle International 2002 23:704-710.
9. Glick J, Morgan C, Myerson M, Sampson T, Mann J. Ankle arthrodesis an arthroscopic method: Long-term follow-up of 34 Cases. Arthroscopy 1996 12: 428-434.
10. Fragomen AT, Fragomen AT, Meyers KN, Davis N, Shu H, Wright T, Rozbruch SR. A biomechanical comparison of micromotion after ankle fusion using 2 fixation techniques: Intramedullary arthrodesis nail or Ilizarov external fixator. Foot & Ankle International 2008 29: 334-341.
11. Raikin S, Venkat R. An approach to the failed ankle arthrodesis. Foot Ankle Clinics 2008 13:401-416.
12. O’Brien T, Hart T, Shereff M, Stone J, Johnson J. Open versus arthroscopic ankle arthrodesis A comparative study. Foot Ankle International 1999 20: 368-373.
13. Hagen RJ. Ankle arthrodesis: problems and pitfalls. Clinical Orthopaedics and Related Research. 1986 202: 152-162.
14. Katsenis D, Bhave A, Paley D. Treatment of malunion and nonunion at the site of an ankle fusion with the Ilizarov apparatus. JBJS 2005 87A: 302–309.
15. Morgan CD, Henke JA, Bailey RW, Kaufer H. Long-term results of tibiotalar arthrodesis. JBJS 1985 67A: 546–550.
16. Costigan W, Thordarson D, Debnath U. Operative management of ankle fractures in patients with diabetes mellitus. Foot & Ankle International 2007 28: 32-37.
17. Jones KB, Maiers-Yelden KA, Marsh JL, Zimmerman MB, Estin M, Saltzman CL. Ankle fractures in patients with diabetes mellitus. JBJS 2005 87B: 489-495.
18. McCormack R.G., Leith J.M.: Ankle fractures in diabetics: Complications of Surgical Management. JBJS1998 80B: 689-692.
19. Wukich D, Joseph A, Ryan M, Ramirez C, Irrgang JJ. Outcomes of ankle fractures in patients with uncomplicated versus complicated diabetes. Foot & Ankle International 2011 32:120-30.
20. Kline AJ, Gruen GS, Pape HC, Tarkin IS, Irrgang JJ, Wukich DK. Early complications following the operative treatment of pilon fractures with and without diabetes. Foot & Ankle International 2009 30:1042-1047.
21. Thordarson, D: Ankle fractures in diabetics. Techniques in Foot and Ankle Surgery. 2004 3: 192-197.
22. Eylon S, Porat S, Bor N, Leibner E. Outcome of Ilizarov ankle arthrodesis. Foot & Ankle International. 2007 28: 873-879.

Address correspondence to: Sutpal Singh, DPM. FACFAS, FAPWCA, Chief Ilizarov Surgical Instructor at Doctors Hospital, West Covina, California.

1  Chief Ilizarov Surgical Instructor at Doctors Hospital, West Covina, California. Private practice in Southern California.
2  Resident, Doctors of Podiatric Medicine (R3),
3  Resident, Doctors of Podiatric Medicine (R2),
4,5  Residents, Doctors of Podiatric Medicine (R1).
All residents : Doctors Hospital of West Covina (PM&S-36).

© The Foot and Ankle Online Journal, 2011

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Maduramycosis of the Foot: A case report of Boyd's Amputation as a salvage procedure in late presentation

Posted on March 1, 2011 | Comments Off on Maduramycosis of the Foot: A case report of Boyd's Amputation as a salvage procedure in late presentation

by Mohan Kumar, J.1 , Narayana Gowda, B.S.2

The Foot and Ankle Online Journal 4 (3): 2

With the increased movement of the world population, familiarity with the clinical picture of the Madura foot is of growing importance beyond its original endemic areas. The characteristic triad of symptoms consists of indurated swelling, multiple sinus tracts with purulent discharge filled with grains and localization at the foot. An increasing number of new etiologic agents are recognized today. For a better choice of therapy an adequate diagnostic procedure is essential; a deep biopsy for histology appears to give a more substantial contribution to identification of the causal organism than culture. The treatment which should be started early is at first essentially a drug treatment. However, in spite of high expectations with regard to new antimycotic drugs, amputation or disarticulation is often inevitable even today, particularly when the lesion is caused by Eumycetes. We present a case of eumycotic mycetoma with extensive involvement of foot for which a Boyd’s amputation was done and treated with antifungal therapy with no recurrence.

Key words: Osteomyelitis, amputation, Mycetoma, Madura foot.

Accepted: February, 2011
Published: March, 2011

ISSN 1941-6806
doi: 10.3827/faoj.2011.0403.0002

Mycetoma is a chronic localized infectious and granulomatous disease involving subcutaneous skin and bone. It results in various deforming sequelae. [1] It is a granulomatous infection of the dermal and subcutaneous tissues caused by filamentous aerobic and anaerobic bacteria (actinomycetomas), true fungi (eumycetomas), and true bacteria, such as Staphylococcus aureus and Pseudomonas species (botryomycosis). [2,3] Mycetoma of the foot was first described by Colebrock in 1846 in the Indian district of Madura, and is commonly known as Madura foot. [4]

The infectious organism is presumed to be directly inoculated after penetration of the skin with a sharp object, such as a thorn. Clinically it presents with painless subcutaneous nodules and fistulae from which a purulent exudate may be discharged. Histologically the nodules contain microabscesses and a surrounding granulomatous reaction. The treatment which should be started early is at first a drug treatment. However, in spite of high expectations with regard to new antimycotic drugs, amputation or disarticulation is often inevitable even today, particularly when the lesion is caused by Eumycetes.

Although the clinical picture is characteristic, diagnostic confusion may occur with chronic bacterial osteomyelitis, especially when bone destruction has occurred. Botryomycosis can give a similar picture. This is a chronic bacterial infection caused by gram positive cocci (Staphylococci, Streptococci) and gram negative bacteria (Escherichia coli, Pseudomonas, Proteus) that can lead to subcutaneous swelling and draining fistulas. Like mycetoma, grains (colonies of bacteria) can be found in suppurative discharges and biopsy specimens. In botryomycosis however, organs can be affected too. Neoplasms (benign and malignant) should be excluded as well.

In the foot, amputation between the tarsometatarsal level and the level of the Syme procedure results in an equinus deformity due to imbalance between tendons acting at the ankle. Boyd’s operation retains the calcaneus and fuses it with the tibia in the ankle mortise. [5] It provides an excellent weight-bearing stump with no need for an artificial limb, but it has been discarded because of difficulty in obtaining sound calcaneotibial fusion. [6]

Recent literature suggests that all mycetomas may be amenable to medical treatment, particularly since the introduction of new azole – derivatives like itraconazole and ketoconazole. [7]

Case Report

A 35-year-old woman from Tamil Nadu, India presented with a 24 month history of a steadily growing lump in the region of the first metatarsophalangeal joint of her right foot. She was treated elsewhere with first ray amputation and itraconazole for 2 months, but patient came to us with multiple sinuses with discharging black granules. (Fig. 1) General examination was unremarkable with no lymphadenopathy or other soft tissue masses. Though the clinical picture was characteristic, differential diagnoses of chronic bacterial osteomyelitis, botryomycosis were also considered.

Figure 1 Clinical photograph of Madura foot.

Blood and serum chemistry were unremarkable. Plain radiographs showed a soft tissue swelling with no calcification. (Fig. 2) An Ultrasound showed a hypoechoic lesion containing discrete hyperechoic foci). In the magnetic resonance imaging (MRI) scan, the lesion was seen on T1 and T2 weighted sequences, to be composed of multiple lesions of high signal intensity measuring a few millimeters across. (Fig. 3)

Figure 2 Radiograph of Madura foot  with a multiple lytic lesions in tarsal and metatarsal bones.

Figure 3 MRI showing multiple lesions of high signal intensity measuring a few millimeters across involving all the metatarsal and tarsal bones except the calcaneus.

A biopsy was performed under ultrasound guidance. Histological features were suggestive of an inflammatory condition with no clear evidence of malignancy. Since the disease was chronic in nature and the patient had taken antifungal treatment for a very long time with no signs of resolution, we planned for an amputation. We discussed in detail the patient’s options including below knee, Syme’s and Boyd’s amputation. A wide excision was performed due to extensive soft tissue tumor and Boyd’s amputation was performed. A talectomy and calcaneo-tibial arthrodesis was performed by using a Charnley’s compression device. (Figs. 4A and 4B) Histological examination of the resected tissue revealed chronic inflammation with visible fungal hyphae.

Figure 4A and 4B Intraoperative photo showing tibiocalcaneal arthrodesis stabilized with a Steinnman pin. (A)  Immediate post-operative radiograph showing Charnley’s compression device in place to stabilize the arthrodesis.

The patient was treated with oral itraconazole for 10 months, 200 mg three times daily for one week followed by 200 mg once daily. The patient has been followed for 14 months without evidence of recurrence. The patient was monitored regularly with routine investigations, renal function tests and liver function tests at every three months during treatment. The patient did not develop any of the side effects of long term use of itraconazole. The patient has been followed for 14 months without evidence of recurrence. (Figs. 5 and 6)

Figure 5 Boyd’s amputation 6 months after surgery showing functional os calcis.

Figure 6 Boyd’s amputation 6 months after surgery showing functional stump.

Discussion

Mycetomas are frequent in the tropical zones of America (Mexico and Venezuela), Africa (Senegal, Sudan) and Asia (India), but can also be observed beyond these areas. Bidie and Carter gave a full description of the disease. Dieng, et al., report 130 cases of mycetoma in Senegal from 1983 to 2000.

Treatment was medical for actinomycetoma and surgical for eumycetoma. Lesions were located on the foot in 81 patients. Sixty six patients with actinomycetoma were cured by medical treatment. Distinction between eumycetoma and actinomycetoma is very important for the treatment. [10]

Actinomycetoma is amenable to treatment by antibiotics, preferably by combined drug therapy for long periods. Eumycetoma is usually treated by aggressive surgical excision combined with medical treatment. [11] Without proper treatment, mycetoma can lead to deformity, amputation, and death. [12]

It is essential to start the treatment at an early stage. Several recorded eumycetomas appear to respond well to administration of antifungal therapy. In our case however, there was a recurrence probably due to inadequate clearance and inadequate antifungal therapy. We performed a Boyd’s amputation and instituted antifungal therapy with Itraconazole. Boyd’s operation has advantages over Syme’s amputation in terms of walking, foot stability, and rebalancing. Also, backward migration of the heel fat pad and shortening that may occur long term in Syme’s amputation is not seen in Boyd’s operation. [13]

After 14 months of follow-up, there was no evidence of recurrence. The stump of the Boyd amputation has sound plantar skin with good blood supply and sensation.

References

1. Shadomy HJ, Utz JP. Deep fungal infection. In Fitzpatrick TB, Eisen AZ,Wolff (eds). Dermatology in General Medicine. 4th edition New York, McGraw Hill 1993, 2472-2475.
2. Lupi O, Tyring SK, McGinnis MR. Tropical dermatology: fungal tropical diseases. J Am Acad Dermatol 2005 53: 931-951.
3. Picou K, Batres E, Jarratt M. Botryomycosis: a bacterial cause of mycetoma. Arch Dermatol 1979, 115: 609–610.
4. Magana M, Magana M. Mycetoma. In Demis DJ (ed). Clinical Dermatology. 26th edition. Philadelphia: Lippincott Williams and Wilkins. 1999, section 17–24, 1–22.
5. Boyd HB. Amputation of the foot with calcaneotibial arthrodesis. JBJS 1939 21: 997-1000.
6. Mills KL. Guide to Orthopaedics. Volume I. Edinburgh, Churchill Livingstone Ltd, 1981.
7. Ruxin T, Steck W, Helm T, Bergfeld W, Bolwell B. Pseudallescheria boydii in an immunocompromised host, Successful treatment with debridement and itraconazole. Arch Dermatol 1996, 132: 382 – 384.
8. Cathrine AN, Bhattacharya K, Srinivasan V. Mycetoma leg a–case report. Int J Low Extrem Wounds. 2003 2(3): 171-172.
9. Sundaram C, Umabala P, Laxmi V, Purohit AK, Prasad VS, Panigrahi M, Sahu BP, Sarathi MV, Kaul S, Borghain R, Meena AK, Jayalakshmi SS, Suvarna A, Mohandas S, Murthy JM. Pathology of fungal infections of the central nervous system: 17 years’ experience from Southern India. Histopathology. 2006 49(4): 396-405.
10. Dieng MT, Sy MH, Diop BM, Niang SO, Ndiaye B. Mycetoma: 130 cases. Ann Dermatol Venereol 2003 130(1 Pt 1): 16-9.
11. Fahal AH. Mycetoma: a thorn in the flesh. Trans R Soc Trop Med Hyg 2004 98(1): 3-11.
12. Lichon V, Khachemoune A. Mycetoma: a review. Am J Clin Dermatol 2006 7(5): 315-321.
13. Altindas M, Kilic A. Is Boyd’s Operation a last solution that may prevent major amputations in diabetic foot patients. J Foot & Ankle Surgery 2008 47 (4): 307-312.

Address correspondence to: Dr. Mohan Kumar, PESIMSR, Kuppam,India.
Mob:09703326378

1 Consultant in Arthroscopy & Sports Medicine, PESIMSR,Kuppam,India.
2 Ortho (Diploma in orthopaedics), DNB Ortho (Diplomate of national board)
Assistant professor, Dept of Orthopaedics, PESIMSR, Kuppam, India.

© The Foot and Ankle Online Journal, 2011

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