IJD
Indian Journal of Dermatology
  Publication of IADVL, WB
  Official organ of AADV
Indexed with Science Citation Index (E) , Web of Science and PubMed
 
Users online: 1183  
Home About  Editorial Board  Current Issue Archives Online Early Coming Soon Guidelines Subscriptions  e-Alerts    Login  
    Small font sizeDefault font sizeIncrease font size Print this page Email this page


 
Table of Contents 
CASE REPORT
Year : 2015  |  Volume : 60  |  Issue : 5  |  Page : 500-502
Subcutaneous zygomycosis: A report of one case responding excellently to potassium iodide


1 Department of Dermatology, STD and Leprosy, Burdwan Medical College, Burdwan, West Bengal, India
2 Department of Pathology, Burdwan Medical College, Burdwan, West Bengal, India

Date of Web Publication4-Sep-2015

Correspondence Address:
Abhijit Saha
46/4, Swarnamoyee Road, P.O: Berhampore, Murshidabad - 742 101, West Bengal
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0019-5154.164376

Rights and Permissions

   Abstract 

Subcutaneous Zygomycosis is a rare opportunistic fungal infection caused by Basidiobolus ranarum. Though this entity is endemic in South India, limited numbers of cases have been reported from this part of the country. We report a case of subcutaneous zygomycosis in a 25 year old lady who presented with a nontender, firm to hard swelling over the upper-left arm. Finger was easily inserted below the indurated edge. Histopathology revealed suppurative granuloma with aseptate hyphae. Patient responded excellently to saturated solution of potassium iodide in subsequent visits.


Keywords: Basidiobolus ranarum, potassium iodide, subcutaneous zygomycosis


How to cite this article:
Mondal AK, Saha A, Seth J, Mukherjee S. Subcutaneous zygomycosis: A report of one case responding excellently to potassium iodide. Indian J Dermatol 2015;60:500-2

How to cite this URL:
Mondal AK, Saha A, Seth J, Mukherjee S. Subcutaneous zygomycosis: A report of one case responding excellently to potassium iodide. Indian J Dermatol [serial online] 2015 [cited 2021 Aug 5];60:500-2. Available from: https://www.e-ijd.org/text.asp?2015/60/5/500/164376

What was known?
Subcutaneous zygomycosis caused by Basidiobolus ranarum presents as firm, nontender swelling over the extremities or trunk of healthy individuals and responds well to potassium iodide and azoles.



   Introduction Top


Zygomycosis is a group of fungal infections caused by a number of fungi belonging to the class zygomycetes. The class includes two orders of medical importance mucorales and entomophthorales. Mucorales are responsible for mucormycosis, more severe form of the disease, seen mainly in immunocompromised host.On the other hand, entomophthoromycosis caused by fungi of the order entomophthorales are of two types: Subcutaneous zygomycosis (B. ranarum) and rhinofacial Zygomycosis (Conidiobolus coronatus). B. ranarum are saprophytes found in soil, decaying vegetables, and gastrointestinal tract of frogs. Basidiobolomycosis clinically manifests as chronic infection of subcutaneous tissue in immunocompetent host involving trunk and extremities. Here we present a case of subcutaneous zygomycosis in a 25-year old immunocompetent lady who responded dramatically to saturated solution of potassium iodide.


   Case Report Top


A 25-year old lady was brought to Dermatology OPD with a gradually increasing painless swelling over the upper-left arm of 8 month duration. The lesion appeared insidiously as an erythematous nodule which gradually increased in size to attain the present configuration. There was no preceding history of trauma. The patient was nondiabetic and no history suggestive of any major illness in the past especially history regarding immunosuppression could be elicited. On cutaneous examination, we revealed a well-defined, firm to hard, painless, nontender, indurated swelling of approximate 6.5 × 7.5 cm 2 in size with surface ulceration and crusting at places [Figure 1]. Lesion was freely mobile over the underlying structures. The margin was well defined and indurated with positive finger insinuation test. Patient was afebrile and regional lymph nodes were not palpable. Rest of the general survey and systemic examinations were within normal limit.
Figure 1: Well-defined mass with surface crusting and ulceration and positive finger insinuation test

Click here to view


Routine investigations including complete hemogram, renal, and liver function tests did not reveal any abnormality. She was seronegative for HIV by ELISA. X-ray of upper-left arm showed soft tissue shadow without any bony involvement. Biopsy sample was taken and sent for histopathology and culture. On direct microscopy with 10% potassium hydroxide mount, we found broad, irregular aseptate hyphae. Histopathology from skin lesion showed dense infiltrate of eosinophils, small cluster of epithelioid cells, foci of necrosis with few broad aseptate fungal hyphae surrounded by eosinophilic material (Splendore-Hoeppli phenomenon) [Figure 2] and [Figure 3]. Culture on Sabouraud's dextrose agar demonstrated luxurious, creamy white, membranous, centrally heaped up, radially folded colony with peripheral satellite growth. Bacteriological culture yielded no growth. Lactophenol cotton blue (LPCB) test could not be performed due to lack of facility.
Figure 2: Scanner view showing eosinophilic granuloma with cluster of epithelioid cells and foci of necrosis

Click here to view
Figure 3: ×40: Showing broad aseptate fungal hyphae surrounded by eosinophilic material (Splendore-Hoeppli phenomenon)

Click here to view


After confirming the diagnosis as Basidiobolomycosis with the help of microscopy and culture, the patient was put on saturated solution of potassium iodide with strict vigilance of thyroid function and signs of iodism. The patient responded remarkably within 2 months. During the first follow up after 1month, lesion reduced to a great extent [Figure 4] and on subsequent visit, it almost disappeared leaving some hyperpigmentation [Figure 5].
Figure 4: First follow-up after 1 month

Click here to view
Figure 5: Second follow up after 2 months

Click here to view



   Discussion Top


Zygomycetes constitute a class of ubiquitous organism which causes clinically relevant fungal infection in humans. Class zygomycetes can be subdivided into two orders, mucorales, and entomophthorales. Members of the order entomophthorales such as B. ranarum are associated with chronic infection of cutaneous and subcutaneous tissue and usually do not disseminate to internal organs. [1],[2] On the contrary, the order mucorales tend to cause invasive disease such as pulmonary, rhinocerebellar and gastrointestinal infection in immunocompromised host. [1],[2] Few reports state mucorales as the culprit organism causing cutaneous/subcutaneous infection in immunocompetent individuals. [3]

The first case of subcutaneous zygomycosis had been reported from Indonesia in 1956. [4] Human disease with B. ranarum is prevalent in tropical countries. As far as Indian scenario is concerned, disease is more concentrated in south India. [5],[6] The fungus is a saprophytic one and present in decaying vegetables, intestine of amphibians, reptiles, fish, and insectivorous bat. [7] Subcutaneous zygomycosis results from inoculation of fungal spore into the dermis or subcutis. Possible mode of transmission is minor trauma which may be through insect bite, intravenous catheter, or even intramuscular injection. [6],[8] Clinically, the disease starts as hardened nodule on extremities or trunk which if untreated may spread and expand locally to form hard, painless, nontender swelling. The mass can be raised up by inserting fingers underneath its smooth and rounded edge. Although ulceration of the nodule may be a possible fate, dissemination is extremely rare. Previously, clinical isolates of basidiobolus were classified into B. ranarum, B. haptosporus, and B. meristosporus. But recent taxonomic studies using sophisticated techniques such as restriction enzyme analysis prove that B. ranarum is the sole agent causing all the human disease.

Histopathologically, subcutaneous Zygomycosis is characterized by small foci of suppurative granuloma distributed all over the dermis and subcutis. Though different types of cells including lymphocytes, histiocytes, plasma cell, and multinucleated giant cells contribute to the composition, eosinophils play the major role. Dominance of eosinophils could be attributed to the release of IL4 and IL10 which help in recruiting eosinophils to the target site. [9] The presence of eosinophilic infiltrate in the granuloma is so characteristic that it is also called eosinophilic granuloma. Degranulation of eosinophils leads to formation of eosinophilic sheath (Splendore-Hoppelli phenomenon) surrounding aseptate or infrequently septate thin walled hyphae. Though direct examination of the biopsy sample may suggest a diagnosis of this entity, culture is gold standard for confirmation.

The possibility of misdiagnosing this entity as neoplasm should be kept in mind as it is a great mimicker of soft tissue tumor, synovial sarcoma and Burkitt's lymphoma. [10] Other important differentials are mycobacterial infection, sporotrichosis, and parasitic infection such as onchocerciasis.

Most of the patients of subcutaneous zygomycosis respond excellently to potassium iodide and azoles. In our patient it was potassium iodide which effectively cured the infection.


   Conclusion Top


In a nutshell, early diagnosis and awareness of this entity even in this part of the country is important to prevent disfigurement, misdiagnosis, and avoidance of unnecessary investigations and surgical interventions.

 
   References Top

1.
Ribes JA, Vanover-Sams CL, Baker DJ. Zygomycetes in human disease. Clin Microbiol Rev 2000;13:236-301.  Back to cited text no. 1
    
2.
Mantadakis E, Samonis G. Clinical presentation of zygomycosis. Clin Microbiol Infect 2009;15:15-20.  Back to cited text no. 2
    
3.
Desai RP, Joseph NM, Ananthakrishnan N, Ambujam S. Subcutaneous zygomycosiscaused by Mucor hiemalis in an immunocompetent patient. Australas Med J 2013;6:374-7.  Back to cited text no. 3
    
4.
Burkitt, DP, Wilson AM, Jelliffe DB. Subcutaneous phycomycosis. A Review of 31 cases in Uganda. Br. Med J 1964;1:1669-72.   Back to cited text no. 4
    
5.
Sujatha S, Sheeladevi C, Khyriem AB, Parija SC, Thappa DM. Subcutaneous zygomycosis caused by basidiobolus ranarum -A case report. Ind J Dermatol Venereol Leprol 2003;21:205-6.  Back to cited text no. 5
    
6.
Kamalam A, Thambiah AS. Muscle invasion by Basidiobolus haptosporus following IM injection. Sabouraudia J Med Vet Mycol 1984;22:273-7.  Back to cited text no. 6
    
7.
Ellis DH. Subcutaneous zygomycosis. In: Merz WG, Hay RJ, editors. Topley and Wilson's: Microbiology and microbial infections - Medical mycology. 10 th ed. London: HodderArnold; 2005:348.  Back to cited text no. 7
    
8.
Gugnani HC. A review of zygomycosis due to Basidiobolus ranarum. Eur J Epidemiol 1999;15:923-9.  Back to cited text no. 8
    
9.
Khan ZU, Khoursheed M, Makar R, Al-Waheeb Sal-Bader I, Al Muzaini A. Basidiobolus ranarum as an etiological agent of gastrointestinal zygomycosis. J Clin Microbiol 2001;39:2360-3  Back to cited text no. 9
    
10.
Bittencourt AL, Serra G, Sadigursky M. Subcutaneous zygomycosis caused by Basidiobolus haptosporus: Presentationof a case mimicking Burkitt's lymphoma. Am J Trop Med and Hyg 1982;31:370-3.  Back to cited text no. 10
    

What is new?
Patient responded extremely well only with potassium iodide and did not develop any sign of iodism with strict compliance to therapy. Our case also denotes that this entity is not exclusive only in South India.


    Figures

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



 

Top
Print this article  Email this article
 
 
  Search
 
  
    Similar in PUBMED
 Related articles
    Article in PDF (1,249 KB)
    Citation Manager
    Access Statistics
    Reader Comments
    Email Alert *
    Add to My List *
* Registration required (free)  


    Abstract
   Introduction
   Case Report
   Discussion
   Conclusion
    References
    Article Figures

 Article Access Statistics
    Viewed3002    
    Printed28    
    Emailed0    
    PDF Downloaded84    
    Comments [Add]    

Recommend this journal