Indian Journal of Medical Microbiology, (2006) 24 (1):58-60 Case Report

CUTANEOUS : NECROTISING FASCITIS DUE TO

*IJ Padmaja, TV Ramani, S Kalyani Abstract

Saksenaea vasiformis is an emerging human pathogen, most often associated with cutaneous or subcutaneous lesions following trauma. This is the report of a case of subcutaneous zygomycosis from which Saksenaea vasiformis was isolated on culture. As the patient developed acute interstitial nephritis, amphotericin B could not be administered in full dose. Surgical debridement was carried out, but the patient deteriorated gradually and died. To the best of our knowledge, this is the first reported case of Zygomycosis due to Saksenaea vasiformis from Visakhapatnam.

Key words: Saksenaea vasiformis, zygomycosis, lactophenol cotton blue, Sabouraud dextrose agar, potassium hydroxide.

Among the zygomycetes there are two orders that contain are discharged thorough the neck after the dissolution of an genera and species of medical importance – the apical mucilaginous plug. and Entomophthorales. In general, fungi in the order Mucorales, cause more severe forms of disease.1 Most of the Saksenaea vasiformis is most often associated with species known to cause human or animal infections belong cutaneous or subcutaneous lesions after trauma. It has also to a few genera within the family Mucoraceae. Members of been associated with rhinocerebral infection, cranial infection, several other genera belonging to other families such as osteomyelitis, necrotising cellulitis and disseminated type of Cunninghamellaceae, Mortierellaceae and Saksenaeceae are infection. also occasionally reported. Since the 1980s, nosocomial Case Report zygomycosis manifesting as primary skin and wound infections caused by Rhizopus spp, Absidia, Cunninghamella, A male patient aged 35 years, working as a constable in a Saksenaea vasiformis and Apophysomyces elegans are often tribal area of Visakhapatnam district was brought to the being reported. emergency service of King George Hospital with complaints of pain abdomen, vomitings and fever of one day duration. A Zygomycosis may manifest as rhinocerebral, pulmonary, history of similar complaint in the past was present and a abdominal, pelvic, cutaneous and disseminated forms. The provisional diagnosis of acute appendicitis was made. The monotypic genus Saksenaea was described by Saksenaea in patient was admitted to surgical unit and an emergency 1953 based on isolates from soil in India.2 It has been isolated appendisectomy was done under spinal anaesthesia. A in soil samples from other geographic areas. Vasiformis is the gangrenous appendix with minimal periappendicular only species. The first human infection with Saksenaea collection was noticed, he was under an antibiotic coverage vasiformis was described by Ajello et al in 1976. of i.v metrogyl and ciprofloxacin pre and post operatively. The key features of Saksenaea vasiformis are the The postoperative period was uneventful till the fourth formation of flask shaped sporangia and failure to sporulate postoperative day except for a mild soiling of the dressing at on primary isolation media. Sporulation may be induced by the drain site on the fifth post operative day. Induration and the use of nutrient deficient media or Czapek Dox agar or by oedema were noticed at the incision site and the patient using agar block method described by Ellis and Ajello. complained of pain. Sutures were removed on the seventh Colonies are fast growing, white with no pigment on the postoperative day and the incision site showed induration and reverse, Hyphae are broad, aseptate and the sporangia are oedema extending for about 7cms around the incision. There typically flask shaped with a distinct spherical center and long was blistering of the skin, the skin edges were becoming neck arising singly or in pairs from dichotomously branched gangrenous and a gaping wound with white cottony growth darkly pigmented rhizoids. Columellae are dome shaped and was observed (Fig. a). By the tenth postoperative day a rapid sporangiospores are small, oblong 1-2 x 3-4 mm in size and spread involving skin and subcutaneous tissue and areas of necrosis were noticed. *Corresponding author (email: ) Department of Microbiology, Andhra Medical College, The patient was afebrile, there was complaint of pain and Visakhapatnam – 530 002, Andhra Pradesh, India burning sensation at the wound site. An emergency slough Received: 04-06-04 excision was done and bits of excised skin and subcutaneous Accepted: 18-10-05 tissue were sent for bacteriological and fungal culture and for

www.ijmm.org January 2006 Padmaja et al – Cutaneous Zygomycosis 59 histopathological examination. The material was processed by planned, but the patient was not willing for high risk and left potassium hydroxide (KOH) mount, lactophenol cotton blue the hospital against medical advice. He was later admitted to (LPCB) mount, Gram stain and culture for bacteria and a private hospital where he passed away. . KOH and LPCB preparations showed broad, aseptate fungal hyphae and fat globules. Grams stain revealed aseptate, Discussion broad hyphae and no bacteria were seen. Bacterial culture Cutaneous infections account for 16% of all forms of yielded no growth. There was no growth on Sabouraud zygomycosis, with an associated mortality of 16%, compared dextrose agar (SDA) with actidione, however, SDA without o to 67% for rhinocerebral, 83% for pulmonary and 100% for actidione at 22 C showed growth after 48 hours. The fungal disseminated infection. Saksenaea vasiformis and colony was cottony and white. Apophysomyces elegans differ from other pathogenic A LPCB mount from this growth showed only broad zygomycetes species by their failure to sporulate on media aseptate hyphae. No sporulation could be observed. Subculture routinely used in mycology laboratory like SDA, potato on cornmeal agar and fresh SDA also failed to accomplish dextrose agar and corn meal agar. Czapek dox agar and agar 3 sporulation. Saksenaea / Apophysomyces were considered as block culture have been used for their identification. a possibility and subculture was made on to 1% saline agar Most of the reported infections caused by Saksenaea and 1% water agar. vasiformis are subcutaneous, with only two being 4 Agar block technique was adopted to achieve sporulation disseminated and one rhinocerebral infection reported. by floating the agar block containing subculture in distilled Cutaneous lesions may be primary or secondary, representing water. Mycelial growth could be observed extending from the haematogenous dissemination from some other primary site. agar block into distilled water in 72 hours. LPCB mount from In the primary disease, the infection may occur at the site of agar block showed broad aseptate ribbon like hyphae a barrier break such as in surgery or an indwelling catheter. characteristic of Zygomycetes and typical flask shaped In an immunosuppressed patient infection may become sporangium with columella short sporangiophore and darkly rapidly disseminated and require much more aggressive pigmented rhizoids (Fig. b). Depending on the typical management. Fatal disseminated infection with Saksenaea morphology the fungus was identified as Saksenaea vasiformis in an immunocompromised woman has been 5 vasiformis. Haematoxylene and eosin staining of tissue section reported by Torell J et al and in a immunocompetent child 6 also showed broad aseptate ribbon like hyphae and areas of by Hay RJ et al . Subcutaneous infections have been reported necrosis (Fig. c). in a three month old infant and an eleven year old thalassemic child, who were successfully treated. Cases of tissue invasion The patient remained afebrile throughout. The infection with Saksenaea vasiformis following traumatic injury have extended from the McBurneys incision to infraclavicular been reported by several workers.7 The first case of region. His blood sugar and blood counts were within normal subcutaneous zygomycosis caused by Saksenaea vasiformis limits. His serum tested nonreactive for antibodies to HIV. in India was reported by Padhye et al8 in a rice mill worker Serum creatinine was 4 gms and there was persistant in 1988. The infection involved the foot with multiple sinuses. hypertension. He was referred to nephrology where he was Amputation of the fore-part of the foot followed by a split diagnosed as having acute interstitial nephritis and was put thickness graft and treatment with potassium iodide cured the on 50% dose of amphotericin B. Emergency debridement was infection. Primary cutaneous zygomycosis due to Saksenaea

Figure: (a) A large gaping wound of anterior abdominal wall with white cottony growth, (b) Lactophenol cotton blue mount of the fungus showing flask shaped sporangium (x400), (c) H & E staining of tissue section showing broad aseptate hyphae(x400).

www.ijmm.org 60 Indian Journal of Medical Microbiology vol. 24, No. 1 vasiformis has subsequently been reported from Chandigarh Mycology. 9th edn, 1998. p. 247–63. in 1997.9 2. Rippon JW. Zygomycosis. Chapter 25 in Medical Mycology 3rd In an immunocompetent patient infection usually remains edn. (W.B Saunder’s Company): 1988. p. 684–5. localized around the site of initial trauma and responds well 3. Mogbil Al Wedaithy. Cutaneous zygomycosis due to Saksenaea to local debridement and antifungal therapy. The present case vasiformis; case report and literature review. Ann Saudi Med has been an acute infection with lesions appearing and 1998;18:482–31. progressing rapidly. There was no systemic illness which might have predisposed to the present condition and there was 4. Chander J. Zygomycosis. Chapter 24 A text book of Medical no evidence of immunosupression. Sporulation was induced Mycology: first edn. Inter print: New Delhi; 1996. p. 158–9. by the agar block method. Though an early diagnosis was 5. Torell J, Cooper BH, Helgeson NG. Disseminated Saksenaea made and debridement carried out amphotericin B could not vasiformis infection. Am J Clin Pathol 1981;76:116–21. be administred in full dose because of the acute interstitial nephritis and the patient was lost. Disseminated infection due 6. Hay RJ, Campbell CK, Marshall WM, Rees BI, Pincott J. to Saksenaea vasiformis in an immunocompetent adult which Disseminated zygomycosis () caused by was fatal has been reported by Solano et al in 2000.10 Saksenaea vasiformis. J Infect 1983;7:162–5.

Saksenaea vasiformis is increasingly being reported as a 7. Dean DF, Ajello L, Irwin RS, Woelk WK, Skarulis GJ. Cranial cause of subcutaneous zygomycosis. Cutaneous zygomycosis zygomycosis caused by Saksenaea vasiformis. Case report. J generally has a favourable outcome, zygomycosis due to Neurosurg 1977; 46:97–103. Saksenaea vasiformis has also been reported to have a 8. Padhye AA, Koshi G, Anandi V, Ponniah J, Sitaram V, Jacob favourable outcome after treatment in most of the cases, M, et al. Division of Mycotic Diseases, Centers for Disease though at times it can cause a fatal infection, as in the present Control, Atlanta, GA 30333. First case of subcutaneous case. Hence when a zygomycetes species which fails to zygomycosis caused by Saksenaea vasiformis in India. Diagn sporulate on routine media is isolated, the isolate should be Microbiol Infect Dis 1988;9:69–77. cultured on nutritionally deficient media to induce sporulation 9. Chakrabarti A, Kumar P, Padhya AA, Chatha L, Singh SK, Das so as to enable quick identification and to start treatment A, et al. Department of Medical Microbiology, Postgraduate properly. In an immunocompetent individual with localized Institute of Medical Education and Research, Chandigarh, India. cutaneous lesions, surgical debridement alone is often Primary cutaneous zygomycosis due to Saksenaea vasiformis sufficient, but aggressive debridement and i.v amphotericin and Apophysomyces elegans. Clin Infect Dis 1997;24:580–3. B are definitely indicated in severe cases. 10. Solano T, Atkins B, Tambosis E, Mann S, Gottlieb T. References Department of Intensive Care, Concord Hospital, Sydney, Australia. Disseminated mucormycosis due to Saksenaea 1. Ellis DH. The zygomycetes. Chapter 15. In: Topley & Wilsons vasiformis in an immunocompetent adult. Clin Infect Dis Microbiology and microbial infections, Vol 4 Medical 2000;30:942–3.

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