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CASE REPORT
Year : 2016  |  Volume : 17  |  Issue : 1  |  Page : 58-61

Subcutaneous phycomycosis presenting with multiple swellings


1 Department of DVL, Gandhi Hospital, Hyderabad, Telangana, India
2 Department of DVL, Gandhi Medical College and Hospital, Hyderabad, Telangana, India
3 Department of DVL, Gandhi Medical College, Hyderabad, Telangana, India

Date of Web Publication4-Jan-2016

Correspondence Address:
Sudha Rani Chintagunta
Department of DVL, Gandhi Hospital, Hyderabad, Telangana
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2319-7250.172461

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  Abstract 

Subcutaneous phycomycosis is a chronic fungal infection caused by Basidiobolus ranarum. Phycomycosis due to B. ranarum is sporadic in nature with a few case reports from South India. The disease usually occurs in children, predominantly affecting males. Diagnosis is based on histopathology and culture. We herewith report a case of subcutaneous phycomycosis in a 12-year-old male child who presented with multiple painless swellings involving the right buttock and thigh which was misdiagnosed and treated with surgical resection and antituberculous therapy with no response by local doctors. Subsequently, we made a diagnosis of subcutaneous phycomycosis, and he was started on itraconazole therapy that has resulted in complete resolution.

Keywords: Basidiobolus ranarum, itraconazole, subcutaneous phycomycosis


How to cite this article:
Chintagunta SR, Arakkal G, Damarla SV, Kumar V A. Subcutaneous phycomycosis presenting with multiple swellings. Indian J Paediatr Dermatol 2016;17:58-61

How to cite this URL:
Chintagunta SR, Arakkal G, Damarla SV, Kumar V A. Subcutaneous phycomycosis presenting with multiple swellings. Indian J Paediatr Dermatol [serial online] 2016 [cited 2020 Jul 2];17:58-61. Available from: http://www.ijpd.in/text.asp?2016/17/1/58/172461


  Introduction Top


Phycomycosis due to Basidiobolus ranarum (subcutaneous zygomycosis, subcutaneous phycomycosis, and basidiobolomycosis) is a chronic granulomatous infection of the skin and subcutaneous tissue characterized by firm to woody hard painless swellings. B. ranarum is a saprophytic filamentous fungus belonging to the order Entomophthorales. The disease is widely prevalent in tropical and subtropical regions of the world. Few cases have been reported from India, predominantly from South India.[1],[2],[3] It is more common in male children. It usually involves the extremities, trunk, and rarely other parts of the body. We report a case of subcutaneous phycomycosis in a male child who presented with multiple swellings on the right buttock and thigh.


  Case Report Top


A 12-year-old male child was brought with the complaints of swellings over right thigh and buttock of 6 months duration. It started as small raised lesion over right thigh and gradually increased in size associated with serous discharge. The swelling later became hard with pigmentation of overlying skin. The child was initially seen by a surgeon who did an excision biopsy which revealed nonspecific inflammatory changes. One month later, he developed two more swellings adjacent to the primary lesion. The child was taken to a pediatrician who suspected tuberculosis and performed a biopsy which was reported as granulomatous panniculitis. He was treated with anti-tuberculosis therapy (ATT) for 5 months with no improvement. The child was then referred to DVL Department. Cutaneous examination revealed an ill-defined swelling of size 9 cm × 4 cm over inferior aspect of right buttock and a swelling of size 1 cm × 1 cm with a hypopigmented scar over lateral surface of right thigh [Figure 1]. On palpation, four firm to hard subcutaneous swellings were felt. Two subcutaneous nodules of 5 cm × 4 cm and one nodule of 3 cm × 3 cm in size were present over inferior and superior lateral quadrant of buttock, respectively. Another nodule of size 2 cm × 2 cm was present over lateral aspect of right thigh. They were mobile, firm to hard in consistency, and edges were well-defined. Skin over the swellings was pinchable. Horizontal group of inguinal lymph nodes were enlarged which were nontender, firm, and mobile. General and systemic examination revealed no abnormalities. We considered a differential diagnosis of deep fungal infection, atypical mycobacterial infection, and soft tissue tumor. Routine investigations were normal. Gram stain from the serous fluid showed Gram-positive cocci. Mantoux test was negative. X-ray of chest, spine, and pelvis showed no abnormalities. Incisional skin biopsy specimen was sent for potassium hydroxide (KOH) mount, fungal culture, and histopathological examination.
Figure 1: Ill-defined swelling with scaling on the right buttock with previous biopsy scars

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KOH wet mount showed aseptate hyphae. Culture on Sabouraud's dextrose agar showed creamy white colonies [Figure 2] and lactophenol cotton blue wet mount showed large vegetative hyphae and zygospores with beak-like appendages [Figure 3]. Histopathology revealed inflammatory granulomatous reaction with a predominant eosinophilic cell infiltrate, multinucleated giant cells, and short hyphae with eosinophilic material around the hyphae (Splendore-Hoeppli phenomenon) [Figure 4] and [f 5]. A final diagnosis of basidiobolomycosis was made. The child was started on itraconazole 100 mg twice daily for 12 weeks. There was complete resolution of all the swellings [Figure 6] and the child was under follow-up for the last 6 months.
Figure 2: Creamy brown growth on 14-keto-stypodiol diacetate

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Figure 3: Short and broad aseptate hyphae (PAS, ×40)

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Figure 4: Splendore-Hoeppli phenomenon (H and E, ×20)

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Figure 5: Aseptate hyphae and zygospores with conjugation beaks on lactophenol cotton blue wet mount

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Figure 6: Regression of swelling following treatment

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  Discussion Top


Zygomycosis includes both deep and subcutaneous mycosis caused by fungi belonging to various genera of the class Zygomycetes which has two orders, Mucorales and Entomophthorales. The term mucormycosis refers to infections caused by molds belonging to the order Mucorales, usually seen in immunocompromised hosts as opportunistic infections. Infection with fungi of the order Entomophthorales typically occurs in immunocompetent individuals and subdivided into Basidiobolus and Conidiobolus species which cause basidiobolomycosis and conidiobolomycosis, respectively. They are saprophytes in soil, decaying vegetation, and gastrointestinal tract of frogs.[1] Conidiobolomycosis caused by Conidiobolus coronatus affects young adults, predominantly males than females. Face is the most common site of infection. It is a locally progressive infection of the nasal and perinasal areas.

Zygomycosis caused by B. ranarum is a usually involves the subcutaneous tissue of the limbs, chest, back, or buttocks characterized by localized woody swelling. It predominantly occurs in healthy individuals with no predisposing factors. The lesion begins as slowly spreading, painless, and hard subcutaneous nodule, usually single, well-circumscribed with rounded edges,[4] sometimes mistaken for soft tissue tumor.[5],[6] Rarely present as necrotic papulonodules, ulcerated [2] lesions or chronic discharging sinuses.[7] Diagnosis is based on histopathology and culture. Culture on Sabouraud's dextrose agar shows furrowed, creamy brown, heaped up, radially folded colonies. Lactophenol cotton blue wet mount shows large, broad vegetative hyphae, and thick walled zygospores with beak-like appendages characteristic of Basidiobolus. The typical histopathological feature is the presence of thin-walled, broad aseptate hyphae with an eosinophilic material “Splendore-Hoeppli” phenomenon.[1]

Treatment includes various drugs such as potassium iodide (KI), amphotericin, and oral azoles such as ketoconazole, itraconazole, and fluconazole.[1] KI and itraconazole are commonly used. The treatment of choice is KI in a dose of 30 mg/kg/day as a single daily dose or divided into three doses. Many reports also showed good response to itraconazole. Treatment is usually continued for 6–12 months.[5] The role of surgical resection is controversial, Prasad et al. have reported that surgery may hasten the spread of infection.[8],[9]

Our case initially presented as single painless subcutaneous hard nodule with serous discharge which was wrongly managed with excision and ATT as was documented in other case reports.[7],[8] Later the child developed four swellings in the adjacent area following excision. Surgery may be the cause for spread of infection in this case as observed by Prasad et al.[8]

High index of suspicion is required to diagnose this condition and to avoid unnecessary surgical interventions. The patients respond well to oral KI and itraconazole. Hence, surgical excision may be avoided since it may hasten the spread of infection as in our case.

Financial Support and Sponsorship

Nil.

Conflicts of Interest

There are no conflicts of interest.

 
  References Top

1.
Sujatha S, Sheeladevi C, Khyriem AB, Parija SC, Thappa DM. Subcutaneous zygomycosis caused by Basidiobolus ranarum- A case report. Indian J Dermatol Venereol Leprol 2003;21:205-6.  Back to cited text no. 1
    
2.
Kumari PH, Rao MS, Subbarayudu S, Saritha Y, Kar A. A case study on subcutaneous zygomycosis with ulcer. Int J Curr Microbiol Appl Sci 2013;2:448-451.  Back to cited text no. 2
    
3.
Chokka KM, Reddy SR, Rayi NR, Pullela R. Entomophthoromycosis of face. Indian J Pathol Microbiol 2010;53:564-5.  Back to cited text no. 3
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4.
Naniwadekar MR, Jagtap SV, Nikam BP, Sanghavi KD. Subcutaneous phycomycosis in a child. Online J Health Allied Sci 2009;8:14.  Back to cited text no. 4
    
5.
Thotan SP, Kumar V, Gupta A, Mallya A, Rao S. Subcutaneous phycomycosis-fungal infection mimicking a soft tissue tumor: A case report and review of literature. J Trop Pediatr 2010;56:65-6.  Back to cited text no. 5
    
6.
Anand M, Deshmukh SD, Pande DP, Naik S, Ghadage DP. Subcutaneous zygomycosis due to Basidiobolus ranarum: A case report. J Trop Med 2010;2010:950390.  Back to cited text no. 6
    
7.
Jayanth T, Gaikwad P, Promila M, Muthusami JC. The sinus that breeds fungus: subcutaneous zygomycosis caused by Basidiobolus ranarum at the injection site. Case Rep Infect Dis 2013;2013:534192.  Back to cited text no. 7
    
8.
Prasad PV, Paul EK, George RV, Ambujam S, Viswanthan P. Subcutaneous phycomycosis in a child. Indian J Dermatol Venereol Leprol 2002;68:303.  Back to cited text no. 8
[PUBMED]  Medknow Journal  
9.
Kumar Verma R, Shivaprakash MR, Shanker A, Panda NK. Subcutaneous zygomycosis of the cervicotemporal region: Due to Basidiobolus ranaram. Med Mycol Case Rep 2012;1:59-62.  Back to cited text no. 9
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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