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CASE REPORT
Year : 2020  |  Volume : 21  |  Issue : 3  |  Page : 229-231

Subcutaneous phycomycosis involving the anterior abdominal wall in an immunocompetent boy successfully treated with potassium iodide


Department of Dermatology, SVS Medical College, Mahbubnagar, Telangana, India

Date of Submission20-Feb-2019
Date of Decision20-Feb-2019
Date of Acceptance26-Apr-2020
Date of Web Publication30-Jun-2020

Correspondence Address:
Dr. Angoori Gnaneshwar Rao
Department of Dermatology, SVS Medical College, Mahbubnagar, Telangana
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijpd.IJPD_17_19

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  Abstract 


Subcutaneous phycomycosis is a chronic deep fungal infection caused by Basidiobolus ranarum, which usually occurs in children, predominantly affecting males. Herein, we report subcutaneous phycomycosis in a 3-year-old boy who presented with a gradually progressive painless swelling over the anterior abdomen of 20-day duration. Lesional biopsy confirmed the diagnosis of subcutaneous phycomycosis which revealed multiple well-defined granulomas composed of histiocytes, epithelioid cells, and multinucleate giant cells with broad nonseptate hyphae stained with Gomori methenamine silver stain. The child was managed with a saturated solution of potassium iodide which completely melted the swelling.

Keywords: Basidiobolus ranarum, broad nonseptate hyphae, phycomycosis, potassium iodide


How to cite this article:
Rao AG, Reddy VS, Tejal M, Divya M. Subcutaneous phycomycosis involving the anterior abdominal wall in an immunocompetent boy successfully treated with potassium iodide. Indian J Paediatr Dermatol 2020;21:229-31

How to cite this URL:
Rao AG, Reddy VS, Tejal M, Divya M. Subcutaneous phycomycosis involving the anterior abdominal wall in an immunocompetent boy successfully treated with potassium iodide. Indian J Paediatr Dermatol [serial online] 2020 [cited 2020 Aug 9];21:229-31. Available from: http://www.ijpd.in/text.asp?2020/21/3/229/288493




  Introduction Top


Phycomycosis is caused by species of Absidia, Rhizopus, Mucor, and Mortierella and by Basidiobolus ranarum, Entomophthora coronate, and Hypomyces destruens.[1] It is a chronic granulomatous infection of the skin and subcutaneous tissue characterized by firm to woody hard painless swellings. It usually involves the extremities and trunk.


  Case Report Top


A 3-year-old boy was brought to the dermatology department with painless swelling over the abdomen for 20 days, not associated with fever. There was no history of injury. Examination revealed a well circumscribed round hyperpigmented swelling over umbilical region 3 cms in diameter, firm to hard in consistency and non-tender. The umbilicus was pushed to the right by the swelling [Figure 1]. Finger insinuation sign was positive. The skin was not pinchable over the swelling. He was provisionally diagnosed as subcutaneous phycomycosis, and the desmoid tumor was considered in the differential diagnosis. Routine hematological and biochemical investigations were normal. Ultrasonography of the swelling revealed an abscess in the subcutaneous fat planes extending from the left lumbar region to the umbilical region measuring 3.8 cm × 2 cm which showed increased flow on color Doppler study. Fine-needle aspiration cytology showed suppurative inflammation. The smear from the aspirate and culture on Sabouraud's dextrose agar was negative for fungi. Serology for human immunodeficiency viruses 1 and 2 was nonreactive. Histopathological examination of a biopsy from the swelling revealed the multiple well-defined granulomas composed of histiocytes, epithelioid cells, and multinucleate giant cells with broad nonseptate hyphae [Figure 2]. The fungal elements were better visualized after Gomori methenamine stain which confirmed the diagnosis of phycomycosis [Figure 3]. Consequently, the child was treated with a saturated solution of oral potassium iodide 30 mg/kg/day and was monitored for thyroid function and early signs of iodism. The swelling resolved completely in 12 weeks, and medication was stopped and the boy is under regular follow-up [Figure 4].
Figure 1: (Original) round well-circumscribed swelling in the umbilical region pushing the umbilicus to the right

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Figure 2: (Original) histopathological examination of biopsy and multiple well-defined granulomas composed of histiocytes, epithelioid cells, and multinucleate giant cells with broad nonseptate hyphae (H and E, ×10)

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Figure 3: (Original) histopathological examination of biopsy. Broad septate hyphae (Gomori methenamine stain, ×100)

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Figure 4: (Original) complete resolution of swelling following potassium iodide treatment

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  Discussion Top


Zygomycosis includes both deep and subcutaneous mycoses 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. The fungus resides in decaying vegetable matter and as a saprophyte in the gut of insectivorous reptiles (lizards), amphibians, and fish.[2]

Zygomycosis caused by B. ranarum usually involves the subcutaneous tissue of the limbs, chest, back, or buttocks, characterized by localized woody swelling. However, the involvement of the abdominal wall in the index case is noteworthy. It predominantly occurs in healthy individuals with no predisposing factors. Similarly, the case under study is also immunocompetent. It is most commonly encountered among boys and usually presents with a single lesion. However, cases of subcutaneous phycomycosis with a single as well as multiple swellings have been reported in the literature [Table 1].[3],[4],[5],[6] The lesion begins as slowly spreading, painless, and hard subcutaneous nodule, well-circumscribed with rounded edges, sometimes mistaken for soft-tissue tumor.[5] Characteristically, one can insinuate fingers behind the swelling as observed in the index case. Diagnosis is by characteristic histopathology of thin-walled, broad septate hyphae with an eosinophilic material “Splendore–Hoeppli” phenomenon.[2] The hyphae stain positive with Gomori's stain, methenamine silver, and Masson's trichrome and faintly with periodic acid–Schiff.[7] The organism responds best to potassium iodide and is administered as a saturated solution of potassium iodide at a dose of 30 mg/kg/day. However, itraconazole was also used with success in some cases.
Table1: (Original) Subcutaneous phycomycosis among children reported in literature

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In conclusion, a high index of suspicion is imperative for the diagnosis of this condition to avoid unnecessary surgical intervention as it often resembles a soft-tissue tumor. Fungal culture and biopsy along with fungal stains can aid in the diagnosis.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Burkitt DP, Wilson AM, Jelliffe DB. Subcutaneous phycomycosis: A review of 31 cases seen in Uganda. Br Med J 1969;1:1669-72.  Back to cited text no. 1
    
2.
Sujatha S, Sheeladevi C, Khyriem AB, Parija SC, Thappa DM. Subcutaneous zygomycosis caused by Basidiobolus ranarum a case report. Indian J Med Microbiol 2003;21:205-6.  Back to cited text no. 2
[PUBMED]  [Full text]  
3.
Sackey A Ghartey N, Gyasi R. Subcutaneous basidiobolomycosis: A case report. Ghana Med J 2017;51:43-6.  Back to cited text no. 3
    
4.
Chintagunta SR, Arakkal G, Damarla SV, Kumar VA. Subcutaneous phycomycosis presenting with multiple swellings. Indian J Ped Dermatol 2016;17:58-61.  Back to cited text no. 4
    
5.
Thotan SP, Ashish VK, Ashwin G Mallya, Rao MS. 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.
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. 6
    
7.
Yangco BG, Okafor JI, TeStrake D.In vitro susceptibilities of human and wild-type isolates of Basidiobolus and Conidiobolus species. Antimicrob Agents Chemother 1984;25:413-6.  Back to cited text no. 7
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]
 
 
    Tables

  [Table 1]



 

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