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CORRESPONDENCE
Year : 2022  |  Volume : 67  |  Issue : 5  |  Page : 610-613
Cutaneous entomophthoromycosis from Bihar: A report of three cases and review of literature


1 Department of Dermatology, All India Institute of Medical Sciences, Patna, Bihar, India
2 Department of Pathology, All India Institute of Medical Sciences, Patna, Bihar, India

Date of Web Publication29-Dec-2022

Correspondence Address:
Swetalina Pradhan
Department of Dermatology, All India Institute of Medical Sciences, Patna, Bihar
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijd.ijd_439_22

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How to cite this article:
Singh S, Shahid R, Pradhan S, Kumar T, Gupta R. Cutaneous entomophthoromycosis from Bihar: A report of three cases and review of literature. Indian J Dermatol 2022;67:610-3

How to cite this URL:
Singh S, Shahid R, Pradhan S, Kumar T, Gupta R. Cutaneous entomophthoromycosis from Bihar: A report of three cases and review of literature. Indian J Dermatol [serial online] 2022 [cited 2023 Feb 8];67:610-3. Available from: https://www.e-ijd.org/text.asp?2022/67/5/610/366130




Sir,

Cutaneous entomophthoromycosis is endemic in the tropics and subtropical regions (southern India). It usually affects immunocompetent patients, and the infection is of two types: Basidiobolomycosis and conidiobolomycosis. The saprobes of the fungus are present in moist, decomposing, or rotting plant/vegetation and faecal matter of amphibians/reptiles. The infection is often preceded by trauma, although it can occur following inhalation of spores.[1] We are discussing three cases of entomophthoromycosis from Bihar, who presented with varied morphology and were diagnosed based on clinic-histopathological co-relation and successfully treated with a combination of supersaturated potassium iodide (SSKI) plus itraconazole therapy.

In our three cases, one infant and two adults presented with a localized hard plate-like swelling over the scrotum and face without any systemic involvement [Figure 1], [Figure 2], [Figure 3]. All were immunocompetent and given a prior history of trauma. Routine haematological investigations and inflammatory markers (ESR and CRP (Erythrocyte sedimentation rate and C reactive protein)) were within normal limits. Mantoux test was negative. The patients were diagnosed with cutaneous entomophthoromycosis based on clinic-histopathological correlation [Figure 4]. A tissue culture test was unavailable from the department of microbiology because of the COVID situation. Details of the clinical profile and investigation have been illustrated in [Table 1].
Figure 1: (a) Swelling on the central aspect of face covering entire nose, bilateral medical canthus, medial aspect of cheeks up to forehead (b) positive finger insinuation beneath the swelling (c) loss of skin pinchability on the surface

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Figure 2: (a) Erythematous to hyperpigmented swelling over left side of nose (b) positive finger insinuation

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Figure 3: (a) Well-defined swelling over scrotum with nodularity at places (b) positive finger insinuation beneath swelling (c) loss of skin pincability on surface

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Figure 4: (a and b) CT scan at different level showing soft tissue swelling involving perinasal region with no bony involvement (c) X-ray scrotum showing normal soft tissue

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Table 1: Clinical profile and treatment response and side-effect noted with itraconazole and SSKI of all the patients

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All the patients were treated with combination therapy of oral SSKI (dose: 40 mg/kg/day in three divided doses) and itraconazole 5 mg/kg/day. SSKI solution was prepared using KI salt dissolved in sterile water till no further crystals dissolved. One drop of SSKI solution contained approx. 50 mg KI. The dose of SSKI was increased daily until the endpoint was achieved. Clinical pointers for endpoint were lacrimation, metallic taste, and salivation. The biochemical pointer was a deranged thyroid profile. A baseline thyroid profile was done in all cases, along with weekly monitoring. The tapering of SSKI was done after complete improvement at a dose of 1 drop/day (50 mg/day). Itraconazole was continued for 4 more weeks after complete improvement. Liver function test (LFT) monitoring was done every month. Case 3 being infant, the dose of SSKI was maintained at a fixed dose 40 mg/kg/day. Complete improvement was seen in all cases [Table 1] and [Figure 6]. Case 2 developed hypothyroidism after 2 weeks of treatment with SSKI, after which the dose was decreased, and the patient was started with a thyroxine sodium tablet of 50 micrograms. His thyroid profile was monitored and gradually returned to normal during 4 months.
Figure 5: (a) H and E stain: dense infiltrate in the lower dermis and subcutaneous layer ×4 (b) H and E stain: dense infiltrate and granuloma ×10 (c) H and E satin: Langerhans giant cells with lympho-histiocytes infiltrate along with eosinophiles ×40 (d) periodic acid Schiff stain: wide empty looking aeptate hyphae ×400 (red circle)

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Figure 6: (a) Complete improvement of case 1 post-treatment (b) complete improvement of case 2 post-treatment (c) complete improvement in case 3 post-treatment

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In the current case series, three immunocompetent patients of two adults and one infant, the adults had rhino-facial involvement suggestive of conidiobolomycosis and the infant had scrotum involvement suggestive of basidiobolomycosis. The clinical differentials of long-standing rhino-facial swelling include tuberculosis, leishmaniasis, sporotrichosis, soft tissue tumour, and lymphoma. Similarly, basidiobolomycosis may mimic other tropical infections presenting with subcutaneous swelling such as sporotrichosis, filarial elephantiasis and onchocerciasis, cutaneous tuberculosis, and other diseases, including Burkitt's lymphoma and soft tissue tumour.[2] However, histopathology can help in differentiating the above conditions.

There is no single best therapeutic agent for treatment. Various drugs tried include ketoconazole, itraconazole, fluconazole, potassium iodide, co-trimoxazole, and amphotericin-B. Considering the safety, easy oral administration, the efficacy of itraconazole and SSKI has been used in various reported cases. SSKI contains 1,000 mg of potassium iodide (KI) per ml, with each drop containing 50 mg and a density of 1.72 g/ml. It is stored in a light-resistant tight container at 15–30°C temperature. It is considered as a gold standard as it gets concentrated at the site of microorganisms and increases the myeloperoxidase enzyme activity of neutrophils and macrophages. It is given at a 40–60 mg/kg/day dose with a total of 3 g/day. It is contraindicated in hypersensitivity to KI, nodular thyroid disease, active tuberculosis, and low complement level. The common side effects are nausea, vomiting, and diarrhoea. Less common side effects are angioedema, urticaria, and iodism (burning in mouth, throat, metallic taste, lacrimation, headache, confusion, weakness, arrhythmia, numbness, eye swelling, and irritation).[3] The role of surgical resection is controversial, and surgery may hasten the spread of infection, according to Prasad et al.[4] All patients in our cases were started with an itraconazole dose of 5 mg/kg/day and a SSKI at 40 mg/kg/day. No patients developed deranged liver function tests in our case series, but one patient had deranged thyroid profiles, following which the dose of SSKI was decreased. All patients had complete improvement with the above treatment.

In a resource-poor setting, clinic-histopathological correlation and therapeutic response to treatment are sufficient to confirm the diagnosis of cutaneous entomophthoromycosis. On review of literature, there have been reports of cutaneous entomophthoromycosis from various parts of the world and India with different sites of involvement treated with various treatment regimes. The details of which have been illustrated in [Supplementary Table 1].



Ethical approval/Informed consent

Informed consent taken.

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.
Chowdhary A, Randhawa HS, Khan ZU, Ahmad S, Khanna G, Gupta R, et al. Rhinoentomophthoromycosis due toconidioboluscoronatus. A case report and an overview of the disease in India. Med Mycol 2010;48:870–9.  Back to cited text no. 1
    
2.
Thotten 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. 2
    
3.
Hassan I, Keen A. Potassium iodide in dermatology. Indian J Dermatol Venereol Leprol 2012;78:390-3.  Back to cited text no. 3
  [Full text]  
4.
Prasad PV, Paul EK, George RV, Ambujam S, Viswanthan P. Subcutaneous phycomycosis in a child. Indian J Dermatol Venereol Leprol 2002;68:3034.  Back to cited text no. 4
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