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CASE REPORT
Year : 2020  |  Volume : 66  |  Issue : 1  |  Page : 38-41

Calvarial involvement in disseminated rhinosporidiosis – A case report and literature review


Department of Dermatology, JLN Hospital and Research Centre, Bhilai, Chhattisgarh, India

Date of Submission03-Mar-2019
Date of Acceptance06-Sep-2019
Date of Web Publication13-Jan-2020

Correspondence Address:
M Dewangan
Department of Dermatology, JLN Hospital and Research Centre, Bhilai, Chhattisgarh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jpgm.JPGM_131_19

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 :: Abstract 


Rhinosporidiosis is a chronic granulomatous infection caused by Rhinosporidium seeberi and mainly involves nasal and ocular mucosa. Bony involvement in rhinosporidiosis is very rare. A young male, previously operated for nasal rhinosporidiosis, presented with two bony swellings on the forehead and multiple subcutaneous lesions on the right lower limb. The diagnosis of disseminated cutaneous rhinosporidiosis with frontal bone involvement was made with the help of fine needle aspiration cytology (FNAC), histopathology, and computed tomography (CT) scan head. Wide excision of the bony lesion was performed. To the best of our knowledge, this is the first radiologically proven case of frontal bone involvement in disseminated rhinosporidiosis. Early diagnosis can be established with a good clinicopathological and radiological correlation. It also emphasizes the importance of CT scan for the evaluation of any subcutaneous skull lesion.


Keywords: Calvarial rhinosporidiosis, disseminated cutaneous rhinosporidiosis, frontal bone rhinosporidiosis, osseous rhinosporidiosis


How to cite this article:
Dewangan B, Naik R, Membally R, Dewangan M. Calvarial involvement in disseminated rhinosporidiosis – A case report and literature review. J Postgrad Med 2020;66:38-41

How to cite this URL:
Dewangan B, Naik R, Membally R, Dewangan M. Calvarial involvement in disseminated rhinosporidiosis – A case report and literature review. J Postgrad Med [serial online] 2020 [cited 2020 Jul 3];66:38-41. Available from: http://www.jpgmonline.com/text.asp?2020/66/1/38/274712





 :: Introduction Top


Rhinosporidiosis is caused by Rhinosporidium seeberi, an aquatic protistan parasite of the Mesomycetozoea class. It usually involves nasal mucosa and conjunctiva, but mucus membranes at other sites may also be affected.[1] Osseous involvement is a very rare occurrence in this disease. Calvarial involvement in disseminated rhinosporidiosis is not described in the literature. We present here the first radiologically as well as histopathologically proven case of frontal bone involvement in disseminated rhinosporidiosis.


 :: Case Report Top


A 27-year-old man from Chhattisgarh presented with painless swelling over the right side of the forehead since 6 months and multiple swellings on the right leg since 2 months. He had undergone excision of a nasal mass 5 years back in a private hospital and histopathology from the excised lesion was not done. He was a farmer and a regular user of the village pond for bathing which was also shared by cattle.

Physical examination revealed two well-defined hard, nontender subcutaneous swellings over the right side of forehead just above the eyebrow varying from 5 × 5 to 2 × 2 cm in size [Figure 1]a. There was central crackling sensation on one of the swellings. There were three lesions on the right leg. The two were well-defined subcutaneous nodules − 3 × 4 and 2 × 3 cm on the medial aspect and one was diffuse cystic swelling on the lateral aspect [Figure 1]b. History of shared pond bathing and surgery for nasal mass excision in the past raised the suspicion of disseminated rhinosporidiosis. Fine needle aspiration of all the swelling was suggestive of rhinosporidiosis [Figure 2]a. Roentgenography of the skull and lower limbs appeared normal. Computed tomography (CT) scan of the head showed erosion of the frontal bone at two corresponding sites [Figure 3]a, [Figure 3]b, [Figure 3]c, [Figure 3]d, [Figure 3]e, [Figure 3]f.
Figure 1: (a) Two subcutaneous swellings on right side of the forehead(arrows) (b) Subcutaneous lesions on the right leg (arrows) (c) After 1 year of surgery(arrows)

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Figure 2: (a) Microphoto of Fine Needle Aspiration Cytology smears showing endospores-laden sporangia and empty sporangia of Rhinosporidosis seeberi. Background shows dispersed endospores, lymphocytes, macrophages and degenerated cell debris (hematoxylin and eosin; original magnification 40×). (b) Histopathology showing multiple sporangia (large arrow) of R. seeberi. The sporangia are seen in the marrow spaces of the trabecular (frontal) bone. Few foreign body giant cells are seen in the sections studied (hematoxylin and eosin; original magnification 40×)

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Figure 3: Transaxial enhanced computed tomography images of the brain. (a) Right supraorbital homogenous enhancing soft tissue swelling in subcutaneous plane (arrow). (b) Bone window showing adjacent irregular osseous destruction (arrow) extending up to inner table and lateral wall of right frontal sinus. (c) Enhancing soft tissue swelling extending to right frontal extracalvarial region (arrow). (d) Bone window shows mild osseous erosion of the outer table with thick solid spiculated periosteal reaction (sunburst appearance) (arrow). (e) Small focal enhancing lobular soft tissue in midline high frontal subcutaneous plane (arrow). (f) Bone window showing adjacent punched out osteolytic destruction (arrow)

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The final diagnosis of disseminated cutaneous rhinosporidiosis with frontal bone involvement was made. Tablet dapsone 100 mg daily was started, but there was no change after 2 months of therapy. Infact, the size of his forehead lesion has gradually increased to cover the upper half of the orbit and eye opening was restricted. Surgical management was planned. The two subcutaneous nodules of leg were subjected to excisional biopsy along with electrocoagulation of the margins. Aspiration of the diffuse swelling on the lateral aspect of right leg yielded serosanguinous fluid and was left after decompression for further observation.

On exploration of the forehead lesion, there was fragile granulation tissue with serosanguinous fluid over the eroded area of the frontal bone. Fluid was drained and the granulation tissue was curetted out. The content was sent for histopathological examination which confirmed rhinosporidiosis. Both outer and inner table of the frontal bone was deficient in the bigger lesion whereas inner table was intact in the smaller one [Figure 4]. Dura was not breached in either cases. One centimeter margin of the bone was excised all around and subjected for histopathological examination. Histology confirmed the involvement of bone with the presence of sporangia in the marrow space [Figure 2]b. Postoperative recovery was uneventful and he was discharged on tablet dapsone 100 mg once daily. After 1 year of follow-up, there are no signs of recurrence [Figure 1]c.
Figure 4: Intraoperative view showing bony erosions of frontal bone (arrows)

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 :: Discussion Top


Rhinosporidiosis has been reported from all over the world but it is more common in tropics. The disease prevalence is high in some parts of India and Sri Lanka.[1] In India, the disease is mainly confined to the coastal states of south India where prevalence is as high as 4.7% and Chhattisgarh region with a prevalence rate of 1%.[2]

Four forms of the rhinosporidiosis are recognized as per the anatomical localization – nasopharyngeal (70%–90%), ocular (15%), cutaneous, and disseminated.[1],[3] Cutaneous and systemic dissemination is quite rare.[3] Occasionally lips, palate, uvula, maxillary antrum, epiglottis, pharynx, larynx, trachea, bronchus, ears, vulva, vagina, urethra, penis, rectum, scalp, and skin are involved.[1],[2] Bony involvement is extremely rare. Only 18 cases have been reported so far in the literature till date, but frontal bone involvement has been reported once only [Table 1].[4],[5],[6],[7],[8],[9],[10],[11],[12],[13],[14],[15],[16],[17],[18],[19],[20]
Table 1: Description of all the cases of osseous involvement in rhinosporidiosis described in literature with respect to involved bone, associated lesions and treatment

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Bone involvement in rhinosporidiosis can occur in three different clinical settings: 1). Local invasion of the skull bone by nasal rhinosporidiosis. 2) Bone involvement is a part of disseminated rhinosporidiosis as seen in our case. 3) Bone involvement is primary without any evidence of lesions elsewhere.[4] Hematogenous spread could be the possible mode of spread to the distant sites.[21] Frontal bone, as in our case could have been reached by this route only.

The sole previous report by Madhavan et al. described isolated frontal bone involvement in 1978, but the radiological evidence was lacking and the diagnosis of osseous involvement was made postoperatively after histopathological examination.[6] Our case is unique because the frontal bone affliction was the part of disseminated rhinosporidiosis and was radiologically evident. Furthermore, the frontal bone erosion was not depicted by the routine X-ray. It was revealed only on CT scan. Hence, we recommend CT scan for the evaluation of any skull lesion in disseminated cutaneous rhinosporidiosis to detect underlying bony involvement.

CT imaging features of surrounding bony involvement in rhinosporidiosis has been described as irregularity, rarefaction, complete, or partial erosion.[22] The sunburst or spiculated pattern of periosteal reaction as seen in our case has not been described in the literature previously. These CT imaging features can easily be confused with bony tumors such as osteosarcoma, multiple myeloma, and metastasis from unknown primary. Therefore, a good clinicopathologial and radiological correlation is necessary to diagnose calvarial rhinosporidiosis preoperatively.

Treatment of choice is excision and electrocoagulation of the base of subcutaneous lesion and wide excision of the bony lesion. We have done excision of bony margin by one centimeter. Dapsone was restarted in postoperative period as this is the only recommended antimicrobial agent to prevent recurrences.[23] Patient is symptom free after 1-year follow-up.

To conclude, almost all the bones are within the reach of R. seeberi, probably through the hematogenous route. It produces osteolytic lesions mimicking various bony lesions including tumors. High index of suspicion will pick up the lesions early, especially in areas with high prevalence. Diagnosis can easily be confirmed by FNAC and histopathology. Excision of the lesion as wide as possible is the treatment of choice. Tablet dapsone is recommended to prevent recurrence.

Declaration of patient consent

The authors certify that appropriate patient consent was obtained.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
 :: References Top

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Lupi O, Tyring SK McGinnis MR. Tropical dermatology: Fungal tropical diseases. J Am Acad Dermatol 2005;53:931-51.  Back to cited text no. 1
    
2.
Billore OP. Epidemiology Rhinosporidiosis. 1st ed. Delhi: AITBS Publishers and distributers; 1996. p. 24-6.  Back to cited text no. 2
    
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Acharya S, Prusty N, Naik LK. Disseminated rhinosporidiosis-- A rare case presentation. Asian J Pharm Health Sci 2014;4:895.  Back to cited text no. 3
    
4.
Suryawanshi PV, Rekhi B, Desai S, Desai SM, Juvekar SL, Jambhekar NA. Rhinosporidiosis isolated to the distal clavicle: A rare presentation clinicoradiologically mimicking a bone tumor. Skeletal Radiol 2011;40:225-8.  Back to cited text no. 4
    
5.
Chatterjee PK, Khatua CR, Chatterjee SN, Dastidar N. Recurrent multiple rhinosporidiosis with osteolytic lesions in hand and foot: A case report. J Laryngol Otol 1977;91:729-34.  Back to cited text no. 5
    
6.
Madhavan M, Ratnakar C, Mehdiratta KS. Rhinosporidial infection of the forehead (report of a case). J Postgrad Med 1978;24:235-6.  Back to cited text no. 6
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Sudarsan K, Saify AA, Siddique D, Sudarsan V, Agrawal S. Rhinosporidosis of first metatarsal - A case report. Indian J Orthop 1979;13:172-75.  Back to cited text no. 7
    
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Aravindan KP, Viswanathan MK, Jose L. Rhinosporidioma of bone: A case report. Indian J Pathol Microbiol 1989;32:312-3.  Back to cited text no. 8
    
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Mitra K, Maity PK. Cutaneous rhinosporidiosis. J Indian Med Assoc 1996;94:84.  Back to cited text no. 9
    
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Pai SA, Naresh KN, Shindhe SR, Borges AM. Rhinosporidioma of bone: Diagnosis by fine needle aspiration. Acta Cytol 1996;40:845-6.  Back to cited text no. 10
    
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Adiga BK, Singh N, Arora VK, Bhatia A, Jain AK. Rhinosporidiosis. Report of a case with an unusual presentation with bony involvement. Acta Cytol 1997;41:889-91.  Back to cited text no. 11
    
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Gokhale S, Ohri VC, Subramanya H, Reddy PS, Sharma SC. Subcutaneous and osteolytic rhinosporidiosis. Indian J Pathol Microbiol 1997;40:95-8.  Back to cited text no. 12
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Kavishwar VS, Naik LP, Vora IM. Fine needle aspiration diagnosis of subcutaneous and osteolytic rhinosporidiosis. Cytopathology 1998;9:215-7.  Back to cited text no. 13
    
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Makannavar JH, Chavan SS. Rhinosporidiosis: A clinicopathological study of 34 cases. Indian J Pathol Microbiol 2001;44:17-21.  Back to cited text no. 14
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Dash A, Satpathy S, Devi K, Das BP, Dash K. Cytological diagnosis of rhinosporidiosis with skeletal involvement: A case report. Indian J Pathol Microbiol 2005;48:215-7.  Back to cited text no. 15
    
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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. 16
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Amritanand R, Nithyananth M, Cherian VM, Venkatesh K, Shah A. Disseminated rhinosporidiosis destroying the talus: A case report. J Orthop Surg (Hong Kong) 2008;16:99-101.  Back to cited text no. 17
    
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Mondal S, Chowdhury A. Rhinosporidiosis of the left wrist joint: A case report. J Orthop Surg (Hong Kong) 2013;21:245-8.  Back to cited text no. 18
    
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Kundu AK, Phuljhele S, Jain M, Srivastava RK. Osseous involvement in rhinosporidiosis. Indian J Orthop 2013;47:523-5.  Back to cited text no. 19
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Pal DK, Mallick AA, Bandyopadhyay R. Rhinosporidiosis of distal radius: A case report with review of literature. Ann Trop Med Public Health 2013;6:321-3.  Back to cited text no. 20
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Rajam RV, Viswamatnum GC, Rao AR, Rangain PN, Anguli VC. Rhinosporidiosis, a study with report of a fatal case of systemic dissemination. Ind J Surg 1955;17:269-98.  Back to cited text no. 21
    
22.
Prabhu SM, Irodi A, Khiangte HL, Rupa V, Naina P. Imaging features of rhinosporidiosis on contrast CT. Indian J Radiol Imaging 2013;23:212-8.  Back to cited text no. 22
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23.
Job A, Venkateswaran S, Mathan M, Krisnaswami H, Raman R. Medical therapy of rhinosporidiosis with dapsone. J Laryngol Otol 1993;107:809-12.  Back to cited text no. 23
    


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