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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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Foot Infections in the Veterans Health Administration

Posted on September 30, 2015 | Comments Off on Foot Infections in the Veterans Health Administration

by Priya P. Sundararajan DPM, FACFAS1, Barbara M. Porter DPM2, Keith A. Grant Ph.D3, Jeffrey M. Robbins DPM4pdflrg

The Foot and Ankle Online Journal 8 (3): 1

BACKGROUND: Foot infections represent a major health concern in the Veterans Health Administration as they often may lead to limb loss. A majority of these infections are associated with diabetes in the form of diabetic foot ulcers. The diabetic foot infection is associated with a substantial mortality rate and often requires amputation to fully address the nidus of infection.
METHODS: A retrospective chart analysis of all surgeries to treat foot infections in an 18-month period was conducted. Multiple variables- patient location, preventative primary care diabetic foot screenings, routine follow-up by a foot-care specialist, and pre-operative hospital admission- were reviewed and recorded. The data was analyzed using a one-tailed z-test and chi-squared tests. The one-tailed z-test provided a facility-specific data analysis highlighting areas which may benefit from education or assistance in terms of resource allocation. The chi-squared tests reveal generalizable findings regarding the association among primary care diabetic foot screenings, routine follow-up by a foot-care specialist, and the need for pre-operative admission.
RESULTS: Results show an absence of routine follow-up by a foot-care specialist is associated with a statistically higher rate of patients requiring pre-operative admission. Conversely, those patients with routine follow-up required fewer admissions. Though not significant at conventional levels, a higher percentage of patients without the primary care diabetic foot exams also lacked specialty follow-up and necessitated pre-operative hospital admission when compared to patients with the screenings.
CONCLUSION: This study provides an example of methodology reviewing pedal infection-related surgical data to perform effective limb loss prevention in the VHA setting. The generalizable results elucidate the role of the primary care and foot-care specialists in preventative medicine thereby avoiding a hospital admission. The current study suggests that a close, collaborative, patient-centered approach between primary care and podiatry results in better outcomes for patients.

Key words infection, ulcer, diabetic foot, veteran, amputation

ISSN 1941-6806
doi: 10.3827/faoj.2015.0803.0001

Address correspondence to: Priya P. Sundararajan DPM, FACFAS
[1] Director of Podiatry, Wilmington VA Medical Center, Department of Surgery, 302-994-2511, 1601 Kirkwood Highway Wilmington, DE 19805, Priya.Sundararajan@va.gov
[2] Podiatric Surgeon, Wilmington VA Medical Center, Department of Surgery,  302-994-2511, 1601 Kirkwood Highway Wilmington, DE 19805, Barbara.Porter3@va.gov
[3] Assistant Professor, James Madison University Department of Political Science, 540-568-4336, 91 E Grace St., MSC 7705 Harrisonburg, VA 22807, GrantKA@jmu.edu
[4] Director, Podiatry Service Veterans Affairs Central Office; Professor of Podiatric Medicine, Kent State University College of Podiatric Medicine; Clinical Assistant Professor, Case Western Reserve University School of Medicine; 216-791-3800, Louis Stokes VA Medical Center, 10701 East Boulevard Cleveland, OH 44106, Jeffrey.Robbins@va.gov

Foot infections are a major health issue in the Veterans Health Administration as they often jeopardize limb preservation and shorten the patient’s lifespan. A majority of these infections are associated with diabetes in the form of diabetic foot ulcers (DFU). The excessively high 5-year mortality rate associated with patients with diabetic ulcers reaches upwards of 55% [1]. With chronicity, the DFU transitions to bone infection. A festering oste-omyelitis further propagates the pedal nidus of infec-tion resulting in a statistically higher rate of fatal sys-temic disease such as heart attack or stroke [2,3,4]. Consequently, 45% of all patients with a diabetic ulcer require surgery, often times a pedal amputation, to address the nidus of infection and reach resolution of symptoms [5]. Effective preventative care can maximize limb preservation and improve life expectancy.

As the single largest health care system in the United States, the Veterans Health Administration (VHA) is working to meet the complex needs of this dramatically increasing pathology [6]. Primary care providers, podiatric surgeons, general surgeons, vascular surgeons, infectious disease physicians, and wound care nurses are integrated in the treatment of the diabetic foot infection.  In the enormity of the VHA system, providers can be oblivious to the amputation-related statistics that may improve patient outcomes.  A facility-specific assessment allows providers to better understand the events leading up to the amputation and prevent long-term loss of follow-up. Such evidence can inform future strategies to effect better prevention and management of the DFU pathology. The aim of this study is two-fold: 1) to provide an example of a retrospective statistical analysis assessing facility-specific data regarding preventative care and patient outcomes for the benefit of other VHA facilities and 2) to understand the associations among preventative primary care diabetic (PC DM) foot exams, routine follow-up by a foot-care specialist, and pre-operative hospital admission in the VHA setting.

Methods

A retrospective analysis of all surgeries to address pedal ulceration infections between January 1, 2013 and June 30, 2014 were analyzed using one-tailed z-tests and chi-squared tests. The following data was collected for each infection-related pedal surgery: chronological surgery number, chronological patient number, location following the patient, whether a preventative PC DM foot exam was performed, whether the patient’s condition required pre-operative hospital admission, if so the date of admission and the reason necessitating admission, dates of podiatric/surgical/wound care follow-ups the patient had prior to admission or surgery (in the case of no admission), whether the patient was routinely followed or not followed by a foot-care specialist prior to surgery, the date of surgery, and an update regarding the patient’s condition.  Patients who went on to have further limb amputation or endured further complication related to the pedal infection were classified as “poor prognosis.” On the contrary, patients who healed the surgical sites were classified as “healed surgical site.” A description of the data collected is detailed and summarized in Table 1 (see supplement within PDF). Table 1 was analyzed using both one-tailed z-tests (Table 2) to understand facility-specific trends and chi-squared tests (Table 3-5) to examine the association between PC DM foot screenings, routine follow-up by a foot-care specialist, and pre-operative hospital admissions.

The locations from which the patient was referred included the main medical center: Wilmington, surrounding community based outpatient clinics (CBOC) A, B, C, and D, and a nursing home: Community Living Center (CLC). The CBOC facility location was withheld for this publication. Some patients were also referred from the neighboring Coatesville VA medical center.  Patient follow-up data was not readily available from this facility, leading to the exclusion of patients originating from this location from the analysis. The variables (PC DM foot screening, specialty follow-up, admission, and surgery) measured in each facility were compared against each location’s outpatient population share as the base value (Table 2). Additional analysis was also performed to test for dependencies between the variables: preventative PC DM foot exams, specialty follow-up prior to surgery, and pre-operative hospital admissions (Tables 3-5).

The PC DM foot exam is a clinical reminder to be completed by the primary care provider as required by “VA/DoD Clinical Practice Guidelines for the Management of Diabetes Mellitus in Primary Care“ [7]. This reminder ensures that DFU prevention is performed in the primary care sector. This alert is only activated at the anniversary of the patient’s last exam. The alert remains active until the test is performed by the provider at which point the test is de-activated for another calendar year.  If the PC DM foot exam was either not performed or performed within a week of admission or surgery, the exam was considered non-preventative as it served no preventative use once the patient required surgical intervention.

table2

Table 2 One-tailed test comparing the variables measured in each location. Statistical significant findings are in bold.  Down-arrow: Findings are statistically lower than expected. Up-arrow: Findings are statistically higher than expected.

table3

Table 3 Χ2 = 9.9676, p = 0.008.  A statistically significant relationship was found between patients who were not followed by a foot-care specialist and those who were admitted.

The specialty follow-up dates, (as listed in column 5 in Table 1), dictated if the patient was adequately followed by a foot-care specialist (as noted in the adjacent column, column 6). By recording the patients’ last 3 podiatry, surgery, or wound care visits, the investigators were able to assess if the patient had regular follow-ups prior to surgery.  At these visits, all components of the diabetic foot exam were assessed. ADA guidelines suggest that a high-risk patient with a history of amputation or ulceration be seen by a specialist every 1-2 months [8]. To give the patients and providers some leeway, the patient was considered “not followed” if he/she was not seen within 3 months preceding admission or surgery.

table4

Table 4 Χ2 = 2.0563, p=0.152. No statistically significant association was found between patients who did not have a PC DM foot screening and those who were not followed by a foot-care specialist. However a higher percentage of patients who had a PC DM foot exam were also followed by a foot-care specialist. The converse also held true.

 table5

Table 5 Χ2 = 1.6067, p=0.205. No statistically significant association was found between patients who did not have a PC DM foot screening and those who were admitted. However a higher percentage of patients with no PC DM foot exam were admitted compared to patients with a PC DM foot exam. Similarly, most of the patients who were not admitted had a prior PC DM foot screening.

Results

Over the 18-month period, 53 surgeries were performed to treat foot infections on 44 patients. Of these surgeries, 92% were amputations (n=49). Fifty-six percent of the surgeries (n=30) required pre-operative admission. Of the admissions, 95.8% occurred secondary to a foot infection. Only 3.7% of the surgeries were performed on non-diabetic patients (n=2). Forty-four percent of the surgeries were performed on patients who were not followed regularly (<3 months). As a result of foot infection, 7.5% of the pedal surgeries (n=4) were associated with further limb amputation. Five of the surgeries were classified as “poor prognosis”, i.e. the patient was expected to or did lose limb or life and was associated with an unresolved pedal infection. One of these patients, healed the surgical site but subsequently developed severe hypotension, multiple bodily pressure lesions, and died from septic shock.

The one-tailed z-test was used to identify patterns within the variables that were disproportionate to that facility’s population share.  For example, a CBOC serving 15% of the population would be expected to account for 15% of the performed surgeries.  This location-specific analysis demonstrates significantly fewer infection-related pedal surgeries, missing PC DM foot exams, and pre-operative admissions out of the Wilmington facility than would be expected relative to its population share alone (table 1).  In contrast, CBOC A has a significantly higher rate of surgeries, missing PC DM foot exams, and admissions than its population share would suggest.  CBOC C also has more admissions than would be expected, but the number of surgeries and missing PC DM foot exams are not overly disproportionate to its population. Additionally, a higher than expected number of patients were regularly followed in CBOC C prior to surgery. As expected with the typical nursing home population, the CLC has a higher rate of surgery, specialty follow-up, pre-operative admissions, and poor prognosis (60%).  No significant findings were noted in CBOC B and D.

Although the above results are idiosyncratic to the Wilmington medical center and surrounding CBOCs, patterns identified in the aggregate data are generalizable to other VHA systems. Chi-squared tests were used to assess bivariate statistical dependencies in which the presence or absence of one factor influences the rate with which another factor occurs. Analysis confirmed a significant relationship (p=0.008) between patients who were not followed by a foot-care specialist to those who necessitate pre-operative admission (table 2). The observed relationship suggests that high-risk patients who are not routinely followed by a foot-care specialist are more likely to require admission than those who are routinely followed. In fact, the odds of a patient without routine specialty follow-up requiring pre-operative admission is roughly 7.5 times higher than for a followed patient.  No statistically significant relationship was found between patients without PC DM foot screenings and those followed (p=0.152) and admitted (p=0.205) at conventional levels (table 3, 4). However based on percentages, certain trends among these variables seem apparent.  Patients without the preventative PC DM foot screenings tended to also lack follow-up by a foot-care specialist (table 3). The converse also held true. Similarly, a higher percentage of the patients without the PC DM foot exam required pre-operative hospital admission when compared to patients with the screening (Table 4).

The Wilmington facility was associated with statistically fewer infection-related pedal surgeries, fewer missing PC DM foot exams, and fewer admissions than its population share would suggest. This site had fewer adverse events preceding the patient’s surgery and overall fared better in the preventative arena than its CBOC counterparts. These comparatively better outcomes coincided with the most resource-intensive location. As a result, the Wilmington facility assisted in the evaluation in slow or non-healing ulcer patients from the CBOC facilities.

The overlap between CBOC C patients who required surgery and those were admitted was 100%. Moreover, 85% of these surgeries were associated with routine follow-up prior to surgery. These clinical outcomes are suggestive of a lack of efficacy in preventative care in this location.  In CBOC A, 87.5% of surgeries required pre-operative admission, which is significantly higher than would be expected based on its population share. Our solution was to request the foot-care specialists in both CBOC A and C to send all non-healing ulcers with a duration greater than 3 months to Wilmington for evaluation and possible treatment.  In terms of resource allocation, funds for part-time nail technician were requested for CBOC A and C to allow the providers to focus on the higher risk patient population. Additionally, 75% of surgeries out of CBOC A did not have preventative PC DM foot evaluations in the year prior to surgery. Our remedy was to present a facility-wide educational lecture discussing these results and the importance of preventative care in the treatment of DFU.

As expected, patients residing in the CLC were associated with a higher rate of pedal surgery with subsequent limb amputation. With its census of patients who are elderly, immobilized, poorly-vascularized, non-responsive, or systemically complicated, a proper treatment addressing the nidus of infection is often not accomplished. We advised the dedicated CLC wound care nurse who performs weekly wound assessments to consult podiatric or general surgery for new wounds in a timely manner. In addition, the Wilmington wound care nurses have assisted in CLC management and prevention of ulcers.

Discussion

The current study demonstrates the value of collaboration between primary care and specialty care for the treatment of diabetic foot infections in the VHA setting. It is the first in its class to present an example of methodology reviewing pedal amputation and infection-related surgical data for limb loss prevention in the integrated VHA system. This facility-specific research focusing on the circumstances surrounding surgery was conducted to assess the efficacy of preventative measures and effect change to better patient outcomes. As it stands today, data collection and analysis for the purpose of limb preservation is not a routine occurrence in the VHA. The present study uses the data collected to highlight areas of concern and allow implementation of minor changes to effectively manage high-risk diabetic patients.  This methodology can be applied in any facility and may directly impact departmental reorganization, resource allocation, and provider or patient education. The present research is also suggestive of a collaborative relationship between of primary care and foot-care specialists in the management and mitigation of diabetic pedal infections. Prior to this study, the associations of these variables and the need for pre-operative hospital admission were not evident. Our results encourage a partnership between primary care providers and foot-care specialists, including podiatrists, general surgeons, and wound care specialists for early detection of pedal infections, thereby minimizing the need for pre-operative hospital admissions in VHA facilities.

Results indicate CBOC A was associated with a higher rate of surgical interventions for foot infections as well as a lower rate of completed preventative PC DM foot exams. One explanation suggests that fewer providers examining the diabetic foot may lead to undetected foot ulcers, propagate the infection, and result in an amputation. Previous studies have indicated that an increased number of providers examining the diabetic foot resulted in fewer infection-related surgeries [9,10]. A study originating in Sweden demonstrates a lower amputation rate in a region in which patients were referred by a variety of providers in contrast to only referrals from general practitioners, suggesting that the more providers examine the diabetic foot, the earlier infection is treated [9]. Another analysis documents the reduced rate of amputation with early detection of DFU [11]. With the addition of nail technicians, we increase the number of providers examining the diabetic foot. Along with the current study, these investigations illustrate the importance of cross-collaboration between specialties for the early detection and subsequent referral to a specialized diabetic wound care team.

Patients originating from CBOC C were routinely followed prior to surgery but nonetheless required admission prior to surgical intervention. This finding questions the efficacy of preventative treatment received in this facility and is suggestive of the need for education, resources, or further referral to a more specialized team. Similarly, CBOC A was associated with a significantly higher than expected rate of surgeries and admissions. As a hospital admission rather than an outpatient consult usually confers a more serious infection, the presumption that superficial infections are permitted to devolve into deeper more consequential infections is suggested. One plausible hypothesis to explain the higher rate of amputations is that care may not be adequately appropriated for the higher risk patients. Often times, VA podiatric providers are inundated with the lower risk routine nail patients leaving limited resources available for the higher risk patients with ulcers.  The American Diabetes Association task force recommends that high-risk patients (history of ulceration/amputation) be evaluated by a foot-care specialist every 1-2 months, whereas low risk diabetic patients may be evaluated annually by a primary care provider or specialist when necessary [8,12-14]. The addition of a nail technician in CBOC A and C could offload the low-risk patients allowing the providers to focus on the patients at a higher risk for amputation. Moreover, the request for the CBOC facilities to refer their long-standing DFU (> 3months) to the Wilmington facility benefits the CBOC patients. With the Wilmington facility having statistically lower rates of infection-related surgeries and admissions, the patients in the lesser performing facilities are likely to have more positive clinical outcomes with an earlier referral.

The purpose of the study was not necessarily to avoid pedal amputation but to maintain optimal compliance in the events preceding the surgery. Many providers have associated the word “amputation” with a negative connotation as in the case of “amputation prevention.” However evidence-based medicine suggests that patients who avoid amputation and live with chronic osteomyelitis generate a chronic inflammatory response by triggering vascular atherosclerosis [3,15]. A population-based study in a cohort of 23 million studied the relationship between chronic osteomyelitis and coronary heart disease [15]. Once the researchers controlled for age, gender, hypertension, diabetes, hyperlipidemia, and stroke between the control and chronic osteomyelitis cohorts, they found a significantly elevated risk of heart disease- a 95% increase- as compared to the control population [15]. Similar findings were supported in a meta-analysis study evaluating the association of the DFU and cardiovascular mortality [3]. Results showed a substantially increased risk of all-cause mortality, fatal myocardial infarction, and fatal stroke in patients with DFU [3]. These studies are among the growing number of studies that support a timely resolution of the DFU thereby preventing limb loss and increasing life expectancy [3,15-20]. The 30-day mortality rate, cardiovascular outcomes, and pulmonary events associated with a pedal amputation is substantially lower (4x) than below-knee or above knee amputations [17-20]. The goal is not simply to avoid amputation but to recognize the time-sensitivity of reaching a permanent resolution, thereby broadening our perspective to prioritize limb and life preservation.

Results derived from the full dataset suggest that the more high risk patients are followed by foot-care specialists, the less likely the infection will progress to a degree that necessitates admission (table 2). On the patient-level, routine follow-up generally translates to earlier detection of infection or vascular impairment, fewer systemic complications, and lower potential for nosocomial infections. From the facility standpoint, a substantial financial and economic burden can be obviated for each avoidable hospitalization.  Studies show that on average each hospital admission for a pedal amputation costs the facility is approximately $32,000 [21]. This confirms the role of foot-care specialists in the treatment of diabetic foot infection and limb loss prevention as documented in previous studies [22,23]. The present study also demonstrates a positive trend between PC DM foot screenings and follow-up by a foot-care specialist in the VHA setting (table 3). Thus the domino effect between the absence of PC DM foot screening and patients necessitating pre-operative admission is evident. The direct impact of fewer PC DM foot screenings and a higher rate of admission follows a negative trend, though not statistically significant at a conventional level (table 4). The current study, specific to the VHA system, is among the increasing evidence supporting the interdepartmental collaboration to improve patient outcomes and reduce complications [23-25].

Limitations to this study are inherent to any retrospective analysis in that all variables cannot be examined. Regarding the one-tailed z-test, extraneous variables such a provider methodology, patient non-compliance, reason for lacking specialty follow-up, or location-specific resources such as casts, grafts, or personnel assistance were not assessed. However, these extrinsic factors do not diminish current results highlighting areas that may benefit from assistance or modification. This study provides perspective in regards to the number of surgeries rather than the number of patients. Therefore, some patients had repeat infection-related surgeries; this variable was not assessed.  In regards to the chi-squared tests, the variables studied (specialty follow-up, PC DM foot assessments, and pre-operative admission) are generalizable among the VHA facilities nationwide. However, small sample size biases against statistically significant results. For example, the findings regarding PC DM foot screenings and specialty follow-up or admissions are likely to be significant by conventional standards with a larger sample following the current trends. Future research specific to the treatment of pedal infections or DFU may help determine which strategies and wound therapies will improve amputation prevention in this high-risk population. We encourage all VHA facilities to retrospectively assess the variables affecting patient outcomes and study the associations between these variables to better patient outcomes.

In summary, by focusing on the situations surrounding the surgical treatment of pedal infections or amputation, each facility is able to perform self-assessments to improve patient care. We believe that only with a self-investigative approach can limb preservation be legitimately pursued. By assessing relevant variables we demonstrate the value of foot-care specialists and primary care providers in the treatment of diabetic foot infections in a VHA facility. This patient-centered approach facilitates earlier detection of infection, mitigates systemic complications, decreases the economic burden to the facility, and ultimately minimizes limb loss.  With interdepartmental collaboration, we are able to prioritize limb preservation for veterans who have already sacrificed so much.

References

  1. Moulik PK, Mtonga R, Gill GV: Amputation and morality in new-onset diabetic foot ulcers stratified by etiology. Diabetes Care 26(2): 491-4, 2003. (PubMed)
  2. Bortoletto MS, de Andrade SM, Matsuo T, Haddad MC, González AD, Silva AM: Risk factors for foot ulcers- a cross sectional survey from a primary care setting in Brazil. Prim Care Diabetes 8(1):71-6, 2014. (PubMed)
  3. Brownrigg JRW, Davey J, Holt PJ, Davis WA, Thompson MM, Ray KK, Hinchliffe RJ: The association of ulceration of the foot with cardiovascular and all-cause mortality in patients with diabetes: a meta-analysis. Diabetologia 55(11):2906-2912, 2012. (PubMed)
  4. Subramaniam B, Pomposelli F, Talmor D, Park KW: Perioperative and Long term morbidity and mortality of above-knee and below-knee amputation in Diabetics and Nondiabetics. Anesth Analg 100:1241-7, 2005. (PubMed)
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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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