Tumoral Rhinosporidioma- a Peripheral Space Occupying Lesion Presentation | Research Square window.SnipcartSettings = { analytics: { enabled: false } }; (function() { var accessVector = localStorage.getItem('access_vector') || ''; window.dataLayer = window.dataLayer || []; if (accessVector) { window.dataLayer.push({ user: { profile: { profileInfo: { snid: accessVector } } } }); } })(); (function(w,d,s,l,i){w[l]=w[l]||[];w[l].push({'gtm.start':new Date().getTime(),event:'gtm.js'});var f=d.getElementsByTagName(s)[0],j=d.createElement(s),dl=l!='dataLayer'?'&l='+l:'';j.async=true;j.src='https://www.googletagmanager.com/gtm.js?id='+i+dl;f.parentNode.insertBefore(j,f);})(window,document,'script','dataLayer','GTM-K279D39R'); Browse Preprints In Review Journals COVID-19 Preprints AJE Video Bytes Research Tools Research Promotion AJE Professional Editing AJE Rubriq About Preprint Platform In Review Editorial Policies Our Team Advisory Board Help Center Sign In Submit a Preprint Cite Share Download PDF Case Report Tumoral Rhinosporidioma- a Peripheral Space Occupying Lesion Presentation Snehasis Das, Karthik Kanna Venkatesh This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-6536212/v1 This work is licensed under a CC BY 4.0 License Status: Posted Version 1 posted You are reading this latest preprint version Abstract Rhinosporidiosis is a granulomatous disorder caused by the aquatic parasite Rhinosporidium seeberi, which is transmitted through contaminated soil and water. This condition predominantly affects the nasopharynx and nasal cavity, while systemic or widespread lesions are uncommon, and soft tissue manifestations are exceedingly rare. This report presents the case of a 24-year-old male who suffered from recurrent nasal rhinosporidiosis for over a decade, which was treatable through surgical excision. However, this ultimately progressed to a recurrence in the form of a soft tissue tumor. It is imperative to recognize common lesions with distinctive presentations to mitigate the potential for increased morbidity and mortality. Surgery Infectious Diseases Disseminated rhinosporidiosis Rhinosporidioma Figures Figure 1 Figure 2 Figure 3 Figure 4 Figure 5 Figure 6 INTRODUCTION Rhinosporidiosis represents a significant pathology within the nasopharyngeal region, with numerous case reports detailing both primary and secondary dissemination to various anatomical sites [ 1 ]. The clinical manifestations can vary widely, ranging from localized cutaneous lesions, typically due to hematogenous spread, to systemic cutaneous disseminations, which may arise from hemo-lymphatic spread or, more commonly, direct inoculation [ 2 ]. While this condition can present in multiple forms, the occurrence of a soft tissue mass lesion is a notably rare phenomenon within this context, with fewer than 20 cases documented in the medical literature to date [ 3 ]. This report seeks to highlight this atypical tumor presentation of a prevalent condition, which may pose diagnostic challenges for less experienced clinicians. CASE PRESENTATION A 24-year-old male with no significant medical history presented with a complaint of swelling in the right leg persisting for one year (Fig. 1 and Fig. 2), accompanied by swelling in the right thigh for the past eight months, and multiple nodular lesions on the head, neck, and face for four months. The patient had a notable medical history of experiencing sudden onset epistaxis 12 years prior, which led to a diagnosis of rhinosporidiosis in the right nasal cavity; he subsequently underwent surgical resection. Despite multiple recurrences within the nasal cavity, necessitating surgical intervention eight times over the last 12 years, he was initiated on Dapsone therapy a decade ago but discontinued it two years prior to this presentation. On physical examination, the patient was alert and oriented, exhibiting no signs of anemia or generalized lymphadenopathy. Notable findings included multiple nodular swellings on the face, earlobes, chest wall, and abdomen. Vital signs were stable, and systematic examination was unremarkable. A local examination revealed a polypoidal lesion in the right nasal cavity. Furthermore, examination of the right lower limb demonstrated a firm swelling measuring 15x10 cm, with an additional 10x5 cm swelling observed in the right thigh, accompanied by dilated veins. Laboratory investigations revealed a hemoglobin level of 11.5 mg/dl without evidence of leukocytosis. Renal function tests remained within normal limits. Fine needle aspiration cytology (FNAC) performed on lesions from the right thigh, calf, neck, and chest wall indicated features consistent with disseminated rhinosporidiosis. This diagnosis was corroborated by excisional biopsy of an abdominal wall lesion, which revealed histopathological features characteristic of rhinosporidiosis. An MRI of the right leg, including thigh screening, demonstrated a large circumscribed enhancing lesion in the subcutaneous plane of the leg posterolaterally and within the rectus femoris in the upper thigh, consistent with rhinosporidiomas (Fig. 3 and Fig. 4). DISCUSSION Rhinosporidium seeberi, a protozoan, is the etiological agent responsible for the granulomatous disease known as rhinosporidiosis. While this infection is predominantly found in India and Sri Lanka, cases have also been documented in Iran, South America, and the United States, with higher prevalence in the southern and central regions of India. The disease is believed to be transmitted through contaminated water and soil, particularly among rural populations that frequently swim in ponds, resulting in exposure to stagnant water as a likely source of infection. Rhinosporidiosis is characterized by its persistence, often recurring after surgical intervention and, in rare instances, spreading beyond its initial site of infection. Although the nasal cavity is the primary area affected, other sites including the skin, lips, palate, uvula, maxillary antrum, larynx, trachea, bronchus, ear, scalp, vulva, vagina, penis, and rectum may also be involved. Cutaneous disseminated rhinosporidiosis, although acknowledged, remains exceptionally rare. Hematogenous spread is considered to be the primary mechanism behind distant manifestations. The clinical presentation of this disease is diverse, posing a significant diagnostic challenge. Dermatologists primarily encounter initial presentations as subcutaneous nodules, plaques, sessile polyps, or ulcerated lesions, while tumoral presentations are exceedingly uncommon. Notably, Aravindan et al. reported the first case of tumoral rhinosporidiosis in 1989, presenting as a left scapular mass initially misdiagnosed as chondrosarcoma. Subsequently, Date et al. introduced the term "rhinosporidioma" in 1995 to describe this cutaneous tumoral form of the disease, which remains infrequently documented in the literature. Histopathological examination is regarded as the gold standard for diagnosing such surgical dilemmas preoperatively. Fine needle aspiration cytology has also proven effective in providing adequate cellular information to establish a diagnosis of rhinosporidiosis. Microscopic evaluations of the lesions typically reveal numerous thick-walled sporangia containing abundant endospores, alongside an inflammatory infiltrate predominantly composed of lymphocytes and plasma cells. In certain cases, a foreign body giant cell response may be observed, influenced by the maturation stage of the sporangia. The Grocott methenamine silver stain is utilized to elucidate the pathognomonic contrast of the cellular walls of sporangiospores and mature sporangia, aiding in diagnostic challenges. In cases necessitating surgical intervention, MRI imaging of the lesion's anatomy is instrumental in facilitating surgical planning. Although there have been reports of spontaneous regression, such occurrences are exceedingly rare and insufficient for routine application. It is plausible to view tumoral lesions as components of the broader disease process, as the majority of reported patients have a history of nasal mucosal involvement. In terms of therapeutic approaches, dapsone is commonly considered the primary treatment modality, particularly when used in conjunction with antibiotic therapy for disseminated cases. Surgical excision, especially via wide local excision with electrocoagulation of the base, remains the primary treatment for accessible lesions. In this particular case, the patient's history of recurrent nasal lesions contributed to systemic dissemination, ultimately resulting in the emergence of a soft tissue-like lesion. Both cytological analysis and histopathological assessment were instrumental in arriving at the diagnosis. Despite the functional impairments caused by the lesions, the patient's unwillingness to undergo further surgical procedures led to a conservative treatment strategy. The patient was initiated on dapsone therapy, resulting in progressive improvement, and a six-month follow-up revealed decrease in the size of the lesion with no compressive symptoms on the surrounding structures (Fig. 5 and Fig. 6). CONCLUSION Tumoral rhinosporidiosis is an exceedingly rare condition that can mimic an aggressive soft tissue sarcoma. The management of this disease primarily resides within the medical domain, with Dapsone typically recognized as the preferred pharmacological agent. Surgical intervention is rarely necessary, except in cases of significant mass effect, distal neurovascular compromise, functional impairment, or for cosmetic reasons. Consequently, it is imperative for surgeons to maintain a high index of suspicion to facilitate the accurate diagnosis of this complex surgical challenge. Declarations 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. Disclaimers: All images and case details are available with the authors of this case report Sources of support: There was no source of support Conflict of Interest: No authors have any conflict of interest regarding this case report Patient Consent: The authors of this article have received and archived written patient consent References Kumari R, Laxmisha C, Thappa DM (2005) Disseminated cutaneous rhinosporidiosis. Dermatol Online J 11: 19. Arseculeratne SN (2002) Recent advances in rhinosporidiosis and Rhinosporidium seeberi. Indian J Med Microbiol 20: 119-131. Nath N, Subba S, Giri SK, Pant P, Kanungo A, Mishra A, Mishra P. Tumoral cutaneous rhinosporidiosis: Case report and review of literature. Indian J Pathol Microbiol [Epub ahead of print] [cited 2022 Jun 23]. Chung KG, Bennett JE. Medical Mycology. London: Philadelphia, Lea & Febiger; 1992. p. 695-704. Grover S. Rhinosporidiosis. J Ind Med Assoc 1975;64:93-5. Arjun R, Khan F, Aravind B. Disseminated rhinosporidiosis masquerading as sarcoma. J Assoc Physicians India 2016;64:86-7. Shenoy MM, Girisha BS, Bhandari SK, Peter R (2007) Cutaneous rhinosporidiosis. Indian J Dermatol Venerol Leprol 73: 179-181. Aravindan KP, Viswanathan MK, Jose L. Rhinosporidioma of bone. A case report. Indian J Pathol Microbiol 1989;32:312-3. Date A, Ramkrishna B, Lee VN, Sundraraj GD (1995) Tumoral rhinosporidiosis. Hiatopathology 27: 288-290. Additional Declarations The authors declare no competing interests. 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Also discoverable on Platform About Our Team In Review Editorial Policies Advisory Board Help Center Resources Author Services Accessibility API Access RSS feed Manage Cookie Preferences © Research Square 2026 | ISSN 2693-5015 (online) Privacy Policy Terms of Service Do Not Sell My Personal Information {"props":{"pageProps":{"initialData":{"identity":"rs-6536212","acceptedTermsAndConditions":true,"allowDirectSubmit":true,"archivedVersions":[],"articleType":"Case Report","associatedPublications":[],"authors":[{"id":448428050,"identity":"9c043752-a92a-40a6-9d53-cab2338bcbd3","order_by":0,"name":"Snehasis 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(Antero-posterior view)\u003c/p\u003e","description":"","filename":"Figure5.jpg","url":"https://assets-eu.researchsquare.com/files/rs-6536212/v1/8be04f69f4958661e9282990.jpg"},{"id":81952999,"identity":"ff0f7493-9ab0-4c1e-9ba4-22e3976807ba","added_by":"auto","created_at":"2025-05-05 09:37:59","extension":"jpg","order_by":6,"title":"Figure 6","display":"","copyAsset":false,"role":"figure","size":2101715,"visible":true,"origin":"","legend":"\u003cp\u003ePost treatment follow up image- complete regression of the swelling (Lateral view)\u003c/p\u003e","description":"","filename":"Figure6.jpg","url":"https://assets-eu.researchsquare.com/files/rs-6536212/v1/28b4291e583a5d2e3b693b4e.jpg"},{"id":81956836,"identity":"c1a9b4e9-310a-4e38-a681-a6af7dfae63e","added_by":"auto","created_at":"2025-05-05 10:02:06","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":7833491,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-6536212/v1/1dee5e62-a13c-49a3-90de-0cf76c6f2f69.pdf"}],"financialInterests":"The authors declare no competing interests.","formattedTitle":"\u003cp\u003eTumoral Rhinosporidioma- a Peripheral Space Occupying Lesion Presentation\u003c/p\u003e","fulltext":[{"header":"INTRODUCTION","content":"\u003cp\u003eRhinosporidiosis represents a significant pathology within the nasopharyngeal region, with numerous case reports detailing both primary and secondary dissemination to various anatomical sites [\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e]. The clinical manifestations can vary widely, ranging from localized cutaneous lesions, typically due to hematogenous spread, to systemic cutaneous disseminations, which may arise from hemo-lymphatic spread or, more commonly, direct inoculation [\u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e]. While this condition can present in multiple forms, the occurrence of a soft tissue mass lesion is a notably rare phenomenon within this context, with fewer than 20 cases documented in the medical literature to date [\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e]. This report seeks to highlight this atypical tumor presentation of a prevalent condition, which may pose diagnostic challenges for less experienced clinicians.\u003c/p\u003e"},{"header":"CASE PRESENTATION","content":"\u003cp\u003eA 24-year-old male with no significant medical history presented with a complaint of swelling in the right leg persisting for one year (Fig.\u0026nbsp;1 and Fig.\u0026nbsp;2), accompanied by swelling in the right thigh for the past eight months, and multiple nodular lesions on the head, neck, and face for four months. The patient had a notable medical history of experiencing sudden onset epistaxis 12 years prior, which led to a diagnosis of rhinosporidiosis in the right nasal cavity; he subsequently underwent surgical resection. Despite multiple recurrences within the nasal cavity, necessitating surgical intervention eight times over the last 12 years, he was initiated on Dapsone therapy a decade ago but discontinued it two years prior to this presentation.\u003c/p\u003e \u003cp\u003eOn physical examination, the patient was alert and oriented, exhibiting no signs of anemia or generalized lymphadenopathy. Notable findings included multiple nodular swellings on the face, earlobes, chest wall, and abdomen. Vital signs were stable, and systematic examination was unremarkable. A local examination revealed a polypoidal lesion in the right nasal cavity. Furthermore, examination of the right lower limb demonstrated a firm swelling measuring 15x10 cm, with an additional 10x5 cm swelling observed in the right thigh, accompanied by dilated veins.\u003c/p\u003e \u003cp\u003eLaboratory investigations revealed a hemoglobin level of 11.5 mg/dl without evidence of leukocytosis. Renal function tests remained within normal limits. Fine needle aspiration cytology (FNAC) performed on lesions from the right thigh, calf, neck, and chest wall indicated features consistent with disseminated rhinosporidiosis. This diagnosis was corroborated by excisional biopsy of an abdominal wall lesion, which revealed histopathological features characteristic of rhinosporidiosis. An MRI of the right leg, including thigh screening, demonstrated a large circumscribed enhancing lesion in the subcutaneous plane of the leg posterolaterally and within the rectus femoris in the upper thigh, consistent with rhinosporidiomas (Fig.\u0026nbsp;3 and Fig.\u0026nbsp;4).\u003c/p\u003e"},{"header":"DISCUSSION","content":"\u003cp\u003eRhinosporidium seeberi, a protozoan, is the etiological agent responsible for the granulomatous disease known as rhinosporidiosis. While this infection is predominantly found in India and Sri Lanka, cases have also been documented in Iran, South America, and the United States, with higher prevalence in the southern and central regions of India. The disease is believed to be transmitted through contaminated water and soil, particularly among rural populations that frequently swim in ponds, resulting in exposure to stagnant water as a likely source of infection. Rhinosporidiosis is characterized by its persistence, often recurring after surgical intervention and, in rare instances, spreading beyond its initial site of infection. Although the nasal cavity is the primary area affected, other sites including the skin, lips, palate, uvula, maxillary antrum, larynx, trachea, bronchus, ear, scalp, vulva, vagina, penis, and rectum may also be involved.\u003c/p\u003e \u003cp\u003eCutaneous disseminated rhinosporidiosis, although acknowledged, remains exceptionally rare. Hematogenous spread is considered to be the primary mechanism behind distant manifestations. The clinical presentation of this disease is diverse, posing a significant diagnostic challenge. Dermatologists primarily encounter initial presentations as subcutaneous nodules, plaques, sessile polyps, or ulcerated lesions, while tumoral presentations are exceedingly uncommon. Notably, Aravindan et al. reported the first case of tumoral rhinosporidiosis in 1989, presenting as a left scapular mass initially misdiagnosed as chondrosarcoma. Subsequently, Date et al. introduced the term \"rhinosporidioma\" in 1995 to describe this cutaneous tumoral form of the disease, which remains infrequently documented in the literature.\u003c/p\u003e \u003cp\u003eHistopathological examination is regarded as the gold standard for diagnosing such surgical dilemmas preoperatively. Fine needle aspiration cytology has also proven effective in providing adequate cellular information to establish a diagnosis of rhinosporidiosis. Microscopic evaluations of the lesions typically reveal numerous thick-walled sporangia containing abundant endospores, alongside an inflammatory infiltrate predominantly composed of lymphocytes and plasma cells. In certain cases, a foreign body giant cell response may be observed, influenced by the maturation stage of the sporangia. The Grocott methenamine silver stain is utilized to elucidate the pathognomonic contrast of the cellular walls of sporangiospores and mature sporangia, aiding in diagnostic challenges. In cases necessitating surgical intervention, MRI imaging of the lesion's anatomy is instrumental in facilitating surgical planning.\u003c/p\u003e \u003cp\u003eAlthough there have been reports of spontaneous regression, such occurrences are exceedingly rare and insufficient for routine application. It is plausible to view tumoral lesions as components of the broader disease process, as the majority of reported patients have a history of nasal mucosal involvement. In terms of therapeutic approaches, dapsone is commonly considered the primary treatment modality, particularly when used in conjunction with antibiotic therapy for disseminated cases. Surgical excision, especially via wide local excision with electrocoagulation of the base, remains the primary treatment for accessible lesions.\u003c/p\u003e \u003cp\u003eIn this particular case, the patient's history of recurrent nasal lesions contributed to systemic dissemination, ultimately resulting in the emergence of a soft tissue-like lesion. Both cytological analysis and histopathological assessment were instrumental in arriving at the diagnosis. Despite the functional impairments caused by the lesions, the patient's unwillingness to undergo further surgical procedures led to a conservative treatment strategy. The patient was initiated on dapsone therapy, resulting in progressive improvement, and a six-month follow-up revealed decrease in the size of the lesion with no compressive symptoms on the surrounding structures (Fig.\u0026nbsp;5 and Fig.\u0026nbsp;6).\u003c/p\u003e"},{"header":"CONCLUSION","content":"\u003cp\u003eTumoral rhinosporidiosis is an exceedingly rare condition that can mimic an aggressive soft tissue sarcoma. The management of this disease primarily resides within the medical domain, with Dapsone typically recognized as the preferred pharmacological agent. Surgical intervention is rarely necessary, except in cases of significant mass effect, distal neurovascular compromise, functional impairment, or for cosmetic reasons. Consequently, it is imperative for surgeons to maintain a high index of suspicion to facilitate the accurate diagnosis of this complex surgical challenge.\u003c/p\u003e"},{"header":"Declarations","content":"\u003cp\u003e\u003cstrong\u003eDECLARATION OF PATIENT CONSENT: -\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe 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.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eFINANCIAL SUPPORT AND SPONSORSHIP\u003c/strong\u003e: - Nil.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eCONFLICTS OF INTEREST:\u003c/strong\u003e\u0026nbsp;- There are no conflicts of interest.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eDisclaimers:\u003c/strong\u003e All images and case details are available with the authors of this case report\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eSources of support:\u003c/strong\u003e There was no source of support\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConflict of Interest:\u003c/strong\u003e No authors have any conflict of interest regarding this case report\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003ePatient Consent:\u003c/strong\u003e The authors of this article have received and archived written patient consent \u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\n\u003cli\u003eKumari R, Laxmisha C, Thappa DM (2005) Disseminated cutaneous rhinosporidiosis. \u003cem\u003eDermatol Online J \u003c/em\u003e11: 19.\u003c/li\u003e\n\u003cli\u003eArseculeratne SN (2002) Recent advances in rhinosporidiosis and Rhinosporidium seeberi. \u003cem\u003eIndian J Med Microbiol \u003c/em\u003e20: 119-131.\u003c/li\u003e\n\u003cli\u003eNath N, Subba S, Giri SK, Pant P, Kanungo A, Mishra A, Mishra P. Tumoral cutaneous rhinosporidiosis: Case report and review of literature. Indian J Pathol Microbiol [Epub ahead of print] [cited 2022 Jun 23]. \u003c/li\u003e\n\u003cli\u003eChung KG, Bennett JE. Medical Mycology. London: Philadelphia, Lea \u0026amp; Febiger; 1992. p. 695-704. \u003c/li\u003e\n\u003cli\u003eGrover S. Rhinosporidiosis. J Ind Med Assoc 1975;64:93-5.\u003c/li\u003e\n\u003cli\u003eArjun R, Khan F, Aravind B. Disseminated rhinosporidiosis masquerading as sarcoma. J Assoc Physicians India 2016;64:86-7.\u003c/li\u003e\n\u003cli\u003eShenoy MM, Girisha BS, Bhandari SK, Peter R (2007) Cutaneous rhinosporidiosis. \u003cem\u003eIndian J Dermatol Venerol Leprol \u003c/em\u003e73: 179-181.\u003c/li\u003e\n\u003cli\u003eAravindan KP, Viswanathan MK, Jose L. Rhinosporidioma of bone. A case report. Indian J Pathol Microbiol 1989;32:312-3.\u003c/li\u003e\n\u003cli\u003eDate A, Ramkrishna B, Lee VN, Sundraraj GD (1995) Tumoral rhinosporidiosis. \u003cem\u003eHiatopathology \u003c/em\u003e27: 288-290.\u003c/li\u003e\n\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":true,"hideJournal":true,"highlight":"","institution":"Jawaharlal Institute of Postgraduate Medical Education and Research, Puducherry","isAcceptedByJournal":false,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"
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