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CASE REPORT
Year : 2020  |  Volume : 38  |  Issue : 3  |  Page : 478-480
 

An unexpected host in a soft-tissue lesion of thigh


Department of Pathology, IPGMER and SSKM Hospital, Kolkata, West Bengal, India

Date of Submission29-May-2020
Date of Decision28-Jul-2020
Date of Acceptance07-Aug-2020
Date of Web Publication4-Nov-2020

Correspondence Address:
Dr. Moumita Sengupta
244 AJC Bose Road, Kolkata - 700 020, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijmm.IJMM_20_236

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 ~ Abstract 


Rhinosporidiosis is an enigmatic entity and poses a major health problem in the developing countries of South-East Asia. A soft friable polypoid nasal mass is the most common presentation, while sparse literature is available on extranasal involvement. We describe the case of a 35-year-old female patient who presented with a slow-growing soft-tissue swelling with ulceration over the thigh. On clinical and radiological examination, a provisional diagnosis of soft-tissue neoplasm was made. After resection, histopathological sections showed a closely packed cyst with innumerable endospores. The present case report documents the rare occurrence of an incidentally detected cutaneous rhinosporidiosis causing diagnostic difficulty.


Keywords: Histopathology, rhinosporidiosis, soft-tissue swelling


How to cite this article:
Jain K, Sarfaraj S M, Sengupta M, Datta C, Chatterjee U. An unexpected host in a soft-tissue lesion of thigh. Indian J Med Microbiol 2020;38:478-80

How to cite this URL:
Jain K, Sarfaraj S M, Sengupta M, Datta C, Chatterjee U. An unexpected host in a soft-tissue lesion of thigh. Indian J Med Microbiol [serial online] 2020 [cited 2020 Nov 28];38:478-80. Available from: https://www.ijmm.org/text.asp?2020/38/3/478/299818





 ~ Introduction Top


Rhinosporidiosis, a rare granulomatous entity, is known to be hyperendemic in the southern part of India and Sri Lanka.[1] This lesion caused by Rhinosporidium Seeberi, taxonomically under Mesomycetozoa, commonly affects the mucosal membrane of the nasal cavity and nasopharynx. Males are commonly affected.[2] Only a handful of case reports have described cutaneous and soft-tissue involvement.[3],[4],[5] The mainstay of treatment is excision, but the disease has a tendency to recur posing a challenge. Here, we describe a rare case of cutaneous rhinosporidiosis in an immunocompetent female misdiagnosed initially as a soft-tissue neoplasm.


 ~ Case Report Top


A 35-year-old female of low socioeconomic status from rural India presented in the surgery outpatient department with a solitary large soft-tissue lesion over the outer aspect of the right thigh for 6 months. The lesion progressively increased in size over the last 3 months. Central ulceration was noted. There was no history of any underlying systemic illness, transplantation or use of immunosuppressive drugs. Her fasting blood sugar was 90 g/dl. A clinical diagnosis of soft-tissue neoplasm was made.

FNAC from the nodule in the thigh region was done. It was suggestive of an inflammatory lesion. Aspiration cytology showed the presence of plenty basophilic endospores morphologically resembling Rhinosporidium [Figure 1]a. Consequently, it was surgically excised. Gross examination revealed a single soft-tissue mass measuring 7 cm × 7 cm × 4 cm. Central ulceration was identified measuring 3 cm × 3 cm × 3 cm [Figure 1]b. Representative sections were taken and sent for further processing.
Figure 1: (a) Aspiration cytology showed the presence of many basophilic endospores (May–Grunwald–Giemsa, ×100). (b) Gross examination revealed a single soft-tissue mass with central ulceration. (c) Microphotograph showed many well-defined thick-walled sporangia in the dermis and subcutaneous area (H and E, ×100). (d) Histopathological sections showed closely packed cyst with innumerable endospores (periodic acid–Schiff, ×100)

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Histopathological examination of the haematoxylin and eosin (H and E)- and periodic acid–Schiff (PAS)-stained sections from nodular lesion of the thigh showed hyperplastic stratified squamous epithelium with many well-defined thick-walled sporangia in the underlying dermis and subcutaneous area. The sporangia were in different stages of development and showed many endospores inside [Figure 1]c and [Figure 1]d. Final diagnosis of cutaneous rhinosporidiosis was made.

During review of the case, the patient informed that she had a nasal bleed 2 years ago and was operated for some lesion. She had lost all documents, but she was prescribed 100 mg of daily dapsone which she stopped mid-course. On further investigation, multiple subcutaneous nodules of small-to-medium size were detected over the neck, anterior chest, arm and gluteal region. All lesions were removed, and after histopathological examination, final diagnosis was revised from solitary cutaneous to disseminated cutaneous rhinosporidiosis. On enquiry, the patient reported to have a history of pond bathing. The patient was referred to the department of otorhinolaryngology for repeat nasal and nasopharyngeal examination. She was administered dapsone 100 mg/day for 6 months. No mucosal lesions were noted. X-ray chest was also within normal limits. The patient is doing fine on 1-year follow-up and has not reported any new lesions anywhere in the body.


 ~ Discussion Top


Rhinosporidiosis is a chronic granulomatous disease that mostly affects nasal, nasopharyngeal and conjunctival mucosa.[2] It has been rarely reported in areas such as brain, bone, liver, lung, spleen, anal canal and genitourinary tract.[6] The disease is mainly prevalent in hot tropical climates like India and Sri Lanka, though few cases have been reported in developed countries like the USA and Europe.[7],[8]

Depending on the clinical manifestation, it can be broadly classified into mucosal, solitary cutaneous and disseminated cutaneous lesions. Cutaneous rhinosporidiosis may mimic other tumoural and non-tumoural diseases and produce a clinical dilemma [Table 1]. Cutaneous lesions occur due to haematogenous spread or auto-inoculation. These may occur with or without nasal involvement. Tumoural cutaneous rhinosporidiosis is rarer, with only a handful of reported cases in the literature.[9],[10],[11],[12]
Table 1: Tumoural cutaneous presentation of rhinosporidiosis

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In our case, the female from low socioeconomic status initially presented only with a solitary lesion over the inner aspect of the thigh. There was no documented lesion anywhere else at that time. Considering the consistency and rapidly enlarging mass, an initial diagnosis of a soft-tissue neoplasm was made. It was after aspiration cytology and histology that a final diagnosis of rhinosporidiosis was made. PAS stain on biopsy samples also complemented the light microscopic diagnosis. None of the previous cases reported rhinosporidiosis initially mimicking soft-tissue neoplasm located solely in the thigh.[9],[10],[11],[12] In addition, as elaborated in [Table 1], warty lesion, nodule or ulceroproliferative lesions were the initial presentation in other literatures. Aravindan et al. reported a case of enlarging scapular mass in an elderly male misdiagnosed clinically and radiologically as chondrosarcoma.[9] On histological examination of the excised tumour, the lesion was found to be a case of rhinosporidiosis. There was a past history of excision of nasal polyp 20 years ago. Avadhani et al. reported another case of cutaneous rhinosporidiosis from India which presented as a progressive vascular soft-tissue mass on the leg and was provisionally diagnosed as a soft-tissue sarcoma.[5] In the present case, the middle-aged woman presented with large soft-tissue lesions over the thigh. The clinical diagnosis of soft-tissue tumour was made. After histopathological confirmation using H and E as well as PAS stain, the case was treated as cutaneous rhinosporidiosis.

Treatment of the disease on accessible sites is complete excision with or without the help of electrocautery. The treatment of disseminated and recurrent cases is very challenging. Failure of drug therapy may also occur due to impermeability of the sporangial wall. Hence, awareness of the varied presentations and critical appraisal of cytology and histological findings are essential for early identification and management of rhinosporidiosis, especially in countries where the disease is endemic.

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 initial s 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.
Herr RA, Ajello L, Taylor JW, Arseculeratne SN, Mendoza L. Phylogenetic analysis of Rhinosporidiumseeberi's18S small–subunitribosomal DNA groups this pathogen among members of the protoctistanmesomycetozoa clade. J Clin Microbiol 1999;37:2750-4.  Back to cited text no. 1
    
2.
Saha J, Basu AJ, Sen I, Sinha R, Bhandari AK, Mondal S. Atypical presentations of rhinosporidiosis: A clinical dilemma? Indian J Otolaryngol Head Neck Surg 2011;63:243-6.  Back to cited text no. 2
    
3.
Vijaikumar M, Thappa DM, Karthikeyan K, Jayanthi S. A verrucous lesion of the palm. Postgrad Med J 2002;78:302, 305-6.  Back to cited text no. 3
    
4.
Angunawela P, De Tissera A, Dissanaike AS. Rhinosporidiosis presenting with two soft tissue tumors followed by dissemination. Pathology 1999;31:57-8.  Back to cited text no. 4
    
5.
Avadhani A, Pai K, Mohanty SP. Cutaneous rhinosporidiosis clinically masquerading as a soft tissue sarcoma- a rare occurrence. Int J Dermatol 2008;47:1153-4.  Back to cited text no. 5
    
6.
Sudarshan V, Goel NK, Gahine R, Krishnani C. Rhinosporidiosis in Raipur, Chhattisgarh: A report of 462 cases. Indian J Pathol Microbiol 2007;50:718-21.  Back to cited text no. 6
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7.
Vélez A, Jiménez G, Hidrón A, Talero S, Agudelo CA. Rhinosporidiosis in Colombia: Case series and literature review. Trop Doct 2018;48:289-93.  Back to cited text no. 7
    
8.
Van Der Coer J, Marres, Wlelinga E, Wong-Alcala L. Rhinosporidiosis in Europe. J Laryngol Otology 1992;106:440-3.  Back to cited text no. 8
    
9.
Aravindan KP, Viswanathan MK, Jose I. Rhinosporidioma of man, a case report. Indian J Pathol Microbiol 1989;32:312-3.  Back to cited text no. 9
    
10.
Das C, Das SK, Chatterjee P, Bandyopadhyay SN. Series of atypical rhinosporidiosis: Our experience in western part of West Bengal. Indian J Otolaryngol Head Neck Surg 2019;71:1863-70.  Back to cited text no. 10
    
11.
Singh G, Bajpai RK, Gambhir R. Rhinosporidiosis: An unusual presentation: A case report. Med J Armed Forces India 1997;53:140-1.  Back to cited text no. 11
    
12.
Prasad K, Veena S, Permi HS, Teerthanath S, Shetty KP, Shetty JP. Disseminated cutaneous rhinosporidiosis. J Lab Physicians 2010;2:44-6.  Back to cited text no. 12
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