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Table of Contents 
CASE REPORT
Year : 2020  |  Volume : 65  |  Issue : 3  |  Page : 204-207
Disseminated cutaneous rhinosporidiosis: Revisited


From the Department of Dermatology, IPGME and R and SSKM Hospital, Kolkata, West Bengal, India

Date of Web Publication14-Apr-2020

Correspondence Address:
Widhi Agrawal
Room No: 342, SSKM Doctors Hostel, 242 AJC Bose Road, Kolkata - 700 020, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijd.IJD_419_19

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   Abstract 


A vegetative growth in the nasal mucosa and nasopharynx present for a considerable period of time raises the possibility of rhinosporidiosis. Such presentation in the Indian subcontinent is not rare but erythematous cauliflower-like or tumor-like growths often reaching large size present on various areas of the skin without necessarily involving the nasal apparatus are often confusing to the attending clinician. A dermatologist may suspect the disseminated form of cutaneous rhinosporidiosis and perform a diagnostic histopathology. Early institution of treatment may give significant relief to the patient.


Keywords: Cutaneous, disseminated, rhinosporidiosis


How to cite this article:
Sen S, Agrawal W, Das S, Nayak PS. Disseminated cutaneous rhinosporidiosis: Revisited. Indian J Dermatol 2020;65:204-7

How to cite this URL:
Sen S, Agrawal W, Das S, Nayak PS. Disseminated cutaneous rhinosporidiosis: Revisited. Indian J Dermatol [serial online] 2020 [cited 2020 May 30];65:204-7. Available from: http://www.e-ijd.org/text.asp?2020/65/3/204/282457





   Introduction Top


Rhinosporidiosis is a chronic granulomatous disease of infective etiology previously thought to be caused by Rhinosporidium seeberi but now a cyanobacterium Microcystis aeruginosa has been proposed as the causative agent. The disease is endemic in India and Sri Lanka. The most common site of involvement is the nasal mucosa and nasopharynx (70–75%). Cutaneous lesions in rhinosporidiosis, though reported, are very rare. There have been less than twenty cases of disseminated cutaneous rhinosporidiosis reported till date. When present they have a multifaceted and varied appearance, thus posing a diagnostic dilemma.


   Case Report Top


A 37-year-old housewife of rural background from eastern India presented with generalized tumor-like lesions since last 6 months. The lesions started in the neck and gradually spread to the trunk, upper and lower extremities. They were gradually increasing in size and were painful. She had history of nasal polyp 2 years back, which extended to the nasopharynx and oropharynx, and was operated 1 year back. There was a history of similar polyp in the nasal cavity 20 years back which had been operated at that time. However, at the time of presentation, there was no nasal polyp. There was no history of diabetes mellitus, hypertension, or tuberculosis. There was a history of occasional use of pond water. On examination, multiple skin-colored tender tumor-like swellings, some sessile, some pedunculated, varying in 1 to 12 cm in size were present over the neck, anterior chest, right arm, right thigh, and right hip [Figure 1]. The lesion over the right hip showed ulceration and necrosis and was friable [Figure 2]. Anterior and posterior rhinoscopy revealed no mass or polyp. Systemic examination revealed no abnormality. Chest X-Ray and ultrasonography abdomen were normal. Serology for HIV, hepatitis, and syphilis was negative. Potassium hydroxide smear from the nodule showed numerous globular cysts of varying sizes representing sporangia [Figure 3]. Some of the cysts were in ruptured condition with release of endospores. Histopathology showed hyperplastic stratified squamous epithelium with underlying subcutis enclosing many well defined thick-walled sporangia. The sporangia showed many endospores inside it [Figure 4]. Periodic acid Schiff staining further confirmed the diagnosis [Figure 5]. A final diagnosis of rhinosporidiosis with cutaneous dissemination was made. All the cutaneous lesions were excised [Figure 6] and the patient was started on oral dapsone 100 mg daily. The patient was keeping well 6 months after removal of the masses and there was no sign of relapse.
Figure 1: Patient with multiple tumor-like growths on various areas of skin

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Figure 2: Large cauliflower-like friable mass with ulceration and necrosis over right hip

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Figure 3: KOH mount under 40× showing sporangium containing spores

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Figure 4: H and E stained smear under 10× and 40× showing sporangia containing endospores

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Figure 5: PAS stained smear under 10× and 40× showing sporangia containing endospores

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Figure 6: After excision of mass over neck and right hip

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   Discussion Top


Rhinosporidiosis has been known to mankind over the years. It was first described in Argentina. In India, the disease is reported to have a higher prevalence in West Bengal and Tamil Nadu. It is more commonly seen in men than in women. Bathing in water bodies like lakes filled with stagnant water in endemic areas has been considered as a major risk factor. Rhinosporidiosis can manifest in various forms—nasal, ocular, mucosal, cutaneous, or disseminated. Cutaneous lesions are very rare and were first described by Forsyth. It usually occurs as a part of disseminated cutaneous rhinosporidiosis. Three types of cutaneous lesions have been described: satellite nodules around nasal polyps, disseminated lesions with visceral involvement, and cutaneous lesions without mucosal involvement. Dissemination is a rare phenomenon and possible modes of transmission postulated are autoinoculation, hematogenous, and direct implantation. The cutaneous lesion in rhinosporidiosis may have varied modes of presentation. A thorough review of all the reported cases of disseminated cutaneous rhinosporidiosis till date [Table 1] showed polymorphic and multi-faceted appearance, tumor-like nodules,[1] warty papules,[2],[3] plaques,[4],[5],[6] and pedunculated mass.[7] Prasad et al. reported a case with multiple cutaneous plaque-like lesions with nasal obstruction. A polypoidal lesion was also present at the base of oral cavity.[6] Priyadarshini et al. presented a 22-year-old student with multiple recurrent subcutaneous swellings.[1] Diagnosis can be easily obtained by aspiration cytology and examining the smear with 10% potassium hydroxide or Papanicolau stain. Giemsa imprint can also be used. However, the organism cannot be grown in culture. Histopathology is the gold standard for diagnosis. Biopsy shows characteristic sporangia, seen as globular cysts of various sizes lined by well-defined wall. Inside the sporangia are present numerous endospores. The disease remains a therapeutic challenge to the clinician. Various antimicrobials and antifungals have been tried but proved ineffective. Dapsone has been used to decrease the frequency of recurrences. It is not curative. Surgical excision is the treatment of choice.
Table 1: Compilation of reported cases of Disseminated Cutaneous Rhinosporidios

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This article has been presented to highlight the rarity of the disease and to emphasize its polymorphic presentations, which may at times confuse the physician. A keen suspicion can alert the clinician leading to early diagnosis and prompt treatment.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the forms, the patient has giver her consent for her images and other clinical information to be reported in the journal. The patient understands that her name and initials will not be published and due efforts will be made to conceal her identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.[17]



 
   References Top

1.
Priyadarshini GR, Srinivasa KS, Parijatham BO, Ganapathy H, Subhashree AR. A rare case of disseminated cutaneous rhinosporidiosis. J Clin Diagn Res 2015;9:EL01-2.  Back to cited text no. 1
    
2.
Verma R, Vasudevan B, Pragasam V, Deb P, Langer V, Rajagopalan S. A case of disseminated cutaneous rhinosporidiosis presenting with multiple subcutaneous nodules and a warty growth. Indian J Dermatol Venereol Leprol 2012;78:520.  Back to cited text no. 2
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3.
Thappa DM, Venkatesan S, Sirka CS, Jaisankar TJ, Gopalkrishnan, Ratnakar C. Disseminated cutaneous rhinosporidiosis. J Dermatol 1998;25:527-32.  Back to cited text no. 3
    
4.
Kumari R, Nath AK, Rajalakshmi R, Adityan B, Thappa DM. Disseminated cutaneous rhinosporidiosis: Varied morphological appearances on the skin. Indian J Dermatol Venereol Leprol 2009;75:68-71.  Back to cited text no. 4
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5.
Ravindran Sandhya B, Jisy SR, Nandakumar G. Cutaneous rhinosporidiosis with varied clinical presentations in an immunocompetent adult. J Int Med Dent 2016;3:42-6.  Back to cited text no. 5
    
6.
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. 6
[PUBMED]  [Full text]  
7.
Salim T, Komu F. Varied presentations of cutaneous rhinosporidiosis: A report of three cases. Indian J Dermatol 2016;61:209-12.  Back to cited text no. 7
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8.
Nayak S, Rout TK, Acharjya B, Patra MK. Subcutaneous rhinosporidiosis. Indian J Dermatol 2008;53:41-3.  Back to cited text no. 8
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Anoop TM, Rajany A, Deepa PS, Sangamithra P, Jayaprakash R. Disseminated cutaneous rhinosporidiosis. J R Coll Physicians Edinb 2008;38:123-5.  Back to cited text no. 9
    
10.
Idirisinghe KAP, Sumanasena JAMB, Madarasinghe N. Disseminated rhinosporidiosis following spontaneous regression of the possible primary lesion. J Diag Pathol 2016;11:34-7.  Back to cited text no. 10
    
11.
Sarker MM, Kibria AKMG, Haque MM. Disseminated subcutaneous rhinosporidiosis: A case report. TAJ 2006;19:31-3.  Back to cited text no. 11
    
12.
Mandal PK, Pandey A, Mukhopadhyay S, Roy S. Disseminated cutaneous rhinosporidiosis: A tumor like lesion with therapeutic challenge. Iran J Pathol 2014;9:277-80.  Back to cited text no. 12
    
13.
Pai S, Pai K. Disseminated rhinsosporidiosis with cutaneous involvement. Egypt Dermatol Online J 2014;10:11.  Back to cited text no. 13
    
14.
Sahu B, Sahu P, Puhan MR. Disseminated cutaneous rhinosporidiosis: A polymorphic presentation in an immunocompetent patient. Indian J Dermatol 2015;60:218.  Back to cited text no. 14
    
15.
Bhartiya R, Agarwal P, Mehra R. Cutaneous rhinosporidiosis mimicking soft tissue tumour: A case report with review of literature. Ann Pathol Lab Med 2017;4:102-5.  Back to cited text no. 15
    
16.
Tolat SN, Gokhale NR, Belgaumkar VA, Pradhan SN, Birud NR. Disseminated cutaneous rhinosporidiomas in an immunocompetent male. Indian J Dermatol Venereol Leprol 2007;73:343-5.  Back to cited text no. 16
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17.
Kaushal S, Mathur SR, Mallick SR, Ramam M. Disseminated cutaneous, laryngeal, nasopharyngeal, and recurrent obstructive nasal rhinosporidiosis in an immunocompetent adult: A case report and review of literature: Disseminated cutaneous rhinosporidiosis. Int J Dermatol 2011;50:340.  Back to cited text no. 17
    


    Figures

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

  [Table 1]



 

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