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Home All issues Volume 32 / No 1 (2026) J Oral Med Oral Surg, 32 1 (2026) 7 Full HTML
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J Oral Med Oral Surg
Volume 32, Number 1, 2026
Article Number 7
Number of page(s) 12
DOI https://doi.org/10.1051/mbcb/2025042
Published online 10 April 2026

© The authors, 2026

Licence Creative CommonsThis is an Open Access article distributed under the terms of the Creative Commons Attribution License (https://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Introduction

Rhinosporidiosis is a chronic granulomatous disease caused by Rhinosporidium seeberi, an aquatic protistan parasite that primarily affects mucous membranes of the nasal cavity and nasopharynx. However, several orofacial sites including the oral cavity, maxillary sinus, and buccal mucosa are often involved which presents with unique diagnostic and therapeutic challenges [1–4]. The disease is endemic in certain tropical and subtropical regions, specifically in India, Sri Lanka, and parts of South America, where environmental exposure to stagnant water bodies plays a significant role in this transmission [5–8].

The prevalence and occasional atypical presentations revealed from epidemiological data, which is gathered from rural and endemic regions, has emphasized the public health relevance of this disease [9–12]. Moreover, there are reports of cases of rhinosporidiosis occurring in different anatomical sites apart from the classical nasal and nasopharyngeal sites. Thus, this signifies the importance of awareness among clinicians to consider it a possible differential diagnosis of orofacial masses, particularly in endemic areas [13–17]. However, the etiology, clinical presentation, and optimal management strategies for orofacial manifestations remain unknown in the scientific literature when compared to nasal rhinosporidiosis. Some systematic reviews have summarized such cases geographically or focused on classical nasal presentations [6,7]. However, there is a gap in a comprehensive synthesis that addresses orofacial rhinosporidiosis, its pathogenesis, diverse clinical presentations, diagnostic challenges, and therapeutic algorithms. There is also limited agreement on management strategies for atypical or disseminated cases, and the role of adjunctive pharmacotherapy remains controversial [18–21]. Moreover, novel surgical innovations and diagnostic modalities have not been systematically reviewed in the context of orofacial involvement.

Clinically, orofacial rhinosporidiosis characteristically manifests as polypoidal masses or nodular swellings, which may mimic other benign or malignant lesions, complicating early diagnosis [14,22]. Uncommon presentations such as large oral cavity masses and facial swellings have been reported, highlighting the variability of clinical features and the need for accurate clinical acumen [2,3,13]. Imaging studies often produce non-specific findings which further leads to the diagnostic dilemma, especially when the lesion involves uncommon sites like the maxillary sinus or causes orbital complications such as proptosis [4,23,24]. Histopathological examination remains the gold standard for definitive diagnosis, demonstrating characteristic sporangia filled with endospores within a hyperplastic mucosal stroma [12,25,26].

The management of rhinosporidiosis primarily includes surgical excision of the lesion combined with cauterization of the base to minimize recurrence which is significant clinically [18,27,28]. Adjuvant medical therapies like administration of dapsone, have been used with variable success, however complications such as autoimmune hemolytic anemia have been reported, necessitating cautious use [19]. Disseminated and recurrent rhinosporidiosis cases have therapeutic challenges which require customized approaches [18,19,28]. Novel surgical modalities and reconstructive techniques have been proposed to improve outcomes in difficult anatomical sites [29].

Orofacial rhinosporidiosis has been minimally discussed in literature with fragmented evidence scattered across few isolated case reports or series. The lack of a consolidated overview restricts the clinical understanding and obstructs the development of evidence-based strategies. This is due to the differences in presentation, progression, and prognosis of the disease from its nasal variant. This systematic review addresses these gaps by synthesizing published literature to establish a structured framework for diagnosis and management. The objectives are to clarify pathogenetic mechanisms contributing to orofacial disease, describe the range of clinical manifestations including uncommon presentations, assess the utility and accuracy of available diagnostic tools, and evaluate the role of surgical and pharmacological interventions. By integrating these domains, the review aims to support clinicians in making informed therapeutic decisions, minimize recurrence, and propose standardized care pathways that can serve as a reference for future research and clinical practice.

Methodology

Research Question and PICO/PICOS Framework: This systematic review has been registered at International Prospective Register of Systematic Reviews (PROSPERO) with PROSPERO ID CRD420251107401. It was guided by the research question: “What are the pathogenetic mechanisms, clinical features, and effective management strategies for orofacial rhinosporidiosis?” The review employed the PICO framework to structure the question as follows: Population (P): patients diagnosed with orofacial rhinosporidiosis; Intervention (I): various management approaches including surgical and medical treatments; Comparison (C): different treatment modalities or no treatment; and Outcomes (O): clinical resolution, recurrence rates, and complication incidence. Study design (S), incorporating case reports and case series to ensure comprehensive coverage of available evidence.

Search Strategy: The systematic search was conducted following the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines to ensure transparency and reproducibility. Electronic databases were searched comprehensively, including PubMed, Scopus, Web of Science. Duplicates or irrelevant results were manually screened and removed, with all searches managed according to PRISMA 2020 recommendations. The timeline for literature retrieval spanned from 2012 to 2025. Key search terms included “rhinosporidiosis”, “orofacial rhinosporidiosis”, “oral cavity rhinosporidiosis”, “maxillary sinus rhinosporidiosis”, “buccal mucosa rhinosporidiosis”, “palatal rhinosporidiosis”, “nasopharyngeal rhinosporidiosis”, “oropharyngeal rhinosporidiosis”, “gingival rhinosporidiosis”, “tongue rhinosporidiosis”, “facial swelling rhinosporidiosis”, “management”, “treatment”, “surgical excision rhinosporidiosis”, “dapsone therapy rhinosporidiosis”, “recurrence rhinosporidiosis”, “disseminated rhinosporidiosis”, “diagnosis”, “histopathology rhinosporidiosis”, “clinical features”, and “pathogenesis”. Boolean operators such as AND, OR, and NOT were used to combine keywords strategically; for example, (“rhinosporidiosis” OR “orofacial rhinosporidiosis”) AND (“management” OR “treatment”).

Eligibility Criteria: Inclusion criteria comprised peer-reviewed articles, case reports and case series, focusing on orofacial rhinosporidiosis with clearly documented clinical, diagnostic, or therapeutic information. Case reports and series were included because orofacial rhinosporidiosis is uncommon, and these papers are most commonly available as clinical sources. These reports help to clarify diagnostic features and guide the management where large comparative studies are unavailable. Articles published in English were considered to ensure accurate interpretation. Exclusion criteria involved studies that focused exclusively on nasal or nasopharyngeal rhinosporidiosis without orofacial involvement, review articles without original data, conference abstracts lacking full-text availability, and non-human studies.

Selection Criteria: The initial screening of titles and abstracts was conducted independently by two reviewers (KC and AD) to exclude irrelevant studies. Full texts of potentially eligible articles were then retrieved and assessed for inclusion by the same reviewers. Any discrepancies were resolved through discussion or, if needed, adjudicated by a third reviewer (CS) to ensure impartial selection and consensus. A PRISMA flow diagram was applied to document the number of records identified, screened, included, and excluded at each stage, along with reasons for exclusion.

Data Extraction, Synthesis, and Quality Assessment: Out of a total of 238 articles, only 16 were included in the review after proper screening of the data (Fig. 1). Data extraction was performed independently by reviewers using a pre-designed standardized form capturing study characteristics, patient demographics, clinical presentation, diagnostic methods, treatment modalities, outcomes, and follow-up details. Case reports and case series were appraised using the Joanna Briggs Institute (JBI) critical appraisal checklist [30].

Risk of Bias and Quality Assessment: Risk of bias was assessed using the Joanna Briggs Institute (JBI) checklist, with scores interpreted as low, moderate, or high; studies with higher bias contributed only to narrative synthesis without affecting the main interpretations. Specific attention was paid to selection bias, reporting bias, and outcome ascertainment owing to the predominance of case reports and series in this rare condition. Studies with high risk of bias or methodological limitations were identified and their influence on the overall synthesis was considered [31]. The use of standardized appraisal tools facilitated objective evaluation.

Thumbnail: Fig. 1 Refer to the following caption and surrounding text. Fig. 1

PRISMA flowchart for the review.

Results

Table I shows that most patients with orofacial rhinosporidiosis were young to middle-aged men, with only two female cases reported [18,27]. The age of affected individuals ranged from 10 to 65 yr [4,10,29,32–36]. Male predominance is commonly linked to environmental or occupational exposure, such as swimming or bathing in stagnant water, while female cases were less frequent [6,10,18,27,32,34]. The nasal cavity, maxillary sinus, and nasopharynx were the most frequently involved sites [4,6,27,29,32], whereas the oral cavity including the buccal mucosa, palate, and oropharynx was affected less often [2,3,35,36]. Rare disseminated cases involved subcutaneous tissues or lungs [33]. Clinically, lesions were typically polypoid, reddish, friable, and “strawberry-like” in appearance [2,3,6,27,35,36], with symptom duration ranging from a few weeks to 20 yr [4,29,32,36]. Long-standing lesions sometimes caused facial deformity, proptosis, or recurrent epistaxis [10,18,34], highlighting the need for careful diagnosis in endemic regions [23,31,34].

Table II shows that histopathology was consistently used to confirm orofacial rhinosporidiosis, demonstrating sporangia with endospores in all reported cases. Imaging modalities, such as CT scans, X-rays, and nasal endoscopy, were variably employed to delineate lesion extent [4,6,13,19,29,33,36], while some cases relied solely on clinical assessment [2,10]. Surgical excision with electrocauterization or endoscopic techniques was the primary intervention [3,6,13,27,34,35], often supplemented with Dapsone for recurrence prevention or as adjunctive therapy [4,13,27,29,36]. Recurrences were uncommon but occurred in select cases, particularly with disseminated disease or incomplete excision [4,29,32,33]. Follow-up durations ranged from 3 to 36 months [2,3,27,33], and complications were minimal, with rare instances of Dapsone-induced hemolytic anemia [33]. Overall, combined surgical and medical management achieved favorable outcomes [3,4,27,35].

Table III presents the methodological quality assessment of 16 case reports using a standardized tool for evaluating case reports and series [31]. Scores were based on selection, ascertainment (A1, A2), causality (C1–C4), and reporting criteria. Four studies achieved a high-quality rating [3,4,33,35], scoring 7/8 due to comprehensive patient selection, detailed diagnostic confirmation, and thorough follow-up. Most remaining reports were rated moderate-high [2,6,10,13,27,29,34,36], demonstrating adequate methodology but with occasional gaps such as incomplete causality assessment or missing follow-up details. Five studies were rated moderate [4,10,18,19,32], mainly due to partial reporting or limited selection criteria. This analysis highlights variability in case report quality, emphasizing the need for standardized reporting to improve reproducibility, diagnostic clarity, and outcome assessment in orofacial rhinosporidiosis.

Table IV presents the risk of bias assessment of individual case reports on orofacial rhinosporidiosis, evaluated using the Joanna Briggs Institute (JBI) Critical Appraisal Checklist for Case Reports. The tool evaluates methodological rigor across ten items, covering patient selection, diagnostic methods, intervention details, outcomes, and follow-up. Across the included studies, most reports scored “ + +” (low risk of bias) in Items 1–3 (patient demographics, clinical history, and diagnostic methods), indicating well-documented case descriptions. Item 4 (clear reporting of diagnostic tests) varied, with some studies marked as “NA” where imaging was not performed [3,29,36]. Item 5 (intervention clearly described) showed occasional gaps, particularly in older studies [29,34]. For Item 6 (adverse events reporting), several reports failed to provide details, receiving “–” [4,6,13,27,36]. Item 7 (takeaway message/conclusion) was consistently well-addressed, while Item 8 (follow-up adequacy) varied, with some studies limited by short-term follow-up [1,5,7]. Items 9 and 10 (outcomes measured and reported) were generally well-documented, showing consistent strengths across most reports. The majority of studies demonstrated moderate-to-high quality, with comprehensive patient details, diagnostic confirmation via histopathology, and clear outcome reporting. Methodological limitations were mainly linked to incomplete reporting of adverse events and variable follow-up duration. This suggests that while evidence from case reports remains valuable, future reports should strengthen consistency in documenting interventions, complications, and long-term outcomes to minimize bias.

Table I

Demographic and clinical characteristics of reported case reports of orofacial rhinosporidiosis.

Table II

Diagnostic modalities, therapeutic interventions, and outcomes in reported cases of orofacial rhinosporidiosis.

Table III

Quality assessment for case reports done using the tool for evaluating the methodological quality of case reports and case series [31].

Table IV

Risk of bias assessment individual studies - JBI's tool for case series.

Discussion

Rhinosporidiosis is a chronic granulomatous disease caused by Rhinosporidium seeberi, characterized by the formation of vascular, friable polyps in mucocutaneous regions, predominantly in the nose and oropharynx. The rare infection continues to pose diagnostic and therapeutic challenges due to its relapsing course and its ability to mimic other pathologies. The present review synthesized evidence from published case reports of orofacial rhinosporidiosis, with particular focus on clinical presentation, diagnostic approaches, management strategies, and recurrence patterns. Case reports and series were included because orofacial rhinosporidiosis is rare disease and most cases present the findings in an elaborate manner. These publications are the main clinical source for uncommon disease presentations and give practical information when there is deficient data from other sources [31].

The analysis of published case reports revealed that most patients with orofacial rhinosporidiosis were young to middle-aged men (with two cases reported in females) (Tab. I). This demographic pattern has been consistently observed in larger series from endemic regions, where male predominance was more than females with a ratio ≥ 2:1 [1,6,14,21]. The predominance of male is commonly attributed to environmental and occupational exposure, since swimming or bathing in stagnant pond water is a recognized risk factor for acquiring the infection. This review found fewer female cases, supporting earlier reports that the disease is less common in women but not limited to men [14]. The age distribution, which ranged from the 2nd to the 6th decade, also matches the trend reported in institutional data where most cases occur in the younger age groups, possibly revealing greater outdoor exposure during these years [8,13].

The commonest sites of involvement are the nasal cavity, paranasal sinuses, and nasopharynx which is consistent with the well-established predilection of R. seeberi for the upper respiratory tract mucosa, where trauma and microabrasions facilitate implantation of endospores [19,37]. Oral cavity involvement, though less frequent, was documented in some cases, confirming previous reports that oral mucosa can be directly infected or secondarily involved from contiguous spread [10,34]. Disseminated cases involving the lungs, skin, or subcutaneous tissue represent the extreme clinical spectrum and are rarely reported, but they highlight the organism's potential for hematogenous spread in advanced or recurrent disease [33,37]. Clinically, the lesions described in this review were characteristically polypoid, reddish, and friable, often likened to a strawberry in appearance. This vascular, granulation-like morphology is pathognomonic and has been consistently described across decades of literature [3,6,27,34]. The duration of symptoms varied widely, from weeks to several years, reflecting the slow-growing yet persistent nature of the infection. Long-standing lesions were often associated with facial deformity, proptosis, or recurrent epistaxis, depending on the site and size of the mass [2,8,32]. These findings emphasize the importance of considering rhinosporidiosis in the differential diagnosis of chronic nasal and oropharyngeal masses, especially in endemic settings, since the disease can mimic benign polyps, papillomas, or even malignancies [4,6,37].

The gold standard for diagnosis in all reviewed cases was histopathology, which demonstrated sporangia of varying sizes containing numerous endospores. This feature is highly distinctive and remains the only reliable diagnostic method, as attempts to culture R. seeberi have been unsuccessful [38–41]. In clinical practice, the inability to culture the organism emphasizes the critical role of biopsy and histopathological examination. The constant dependence on histology in both case reports and institutional studies reinforces its indispensability. The use of imaging modalities such as CT or MRI was different across the reviewed cases. Some reports included detailed radiological assessments to delineate lesion extent, particularly in sinonasal and orbital disease [3,6,29,42], whereas others relied solely on endoscopic or gross examination findings. The varying use of imaging reflects differences in reporting standards and resource availability, but it also creates inconsistency in how completely the disease was evaluated. In the quality assessment, reports that included imaging and complete clinical description scored higher for diagnostic clarity compared to those that relied exclusively on histopathology without detailing preoperative evaluation [26,43]. Another important aspect identified was the incomplete reporting of follow-up. While some reports documented outcomes for more than a year, others mentioned only short-term recovery. This variation restricts the dependability of recurrence data, since rhinosporidiosis is known for late recurrences (sometimes several years after surgery) [4,29]. Similarly, adverse event reporting was found to be often absent, with few reports addressing complications related to surgery or Dapsone therapy. This represents a significant methodological gap, because Dapsone, while useful in reducing recurrence, carries risks such as hemolytic anemia and methemoglobinemia [33,44]. Adverse event reporting was often missing, which reduced the value of these case reports. The methodological quality ranged from moderate to high. Reports with complete demographic data, diagnostic methods, surgical details, and long-term outcomes scored higher on JBI. Those with incomplete follow-up or poor safety reporting scored lower. Consistent use of standardized guidelines, such as the CARE checklist, can improve the reliability and comparability of published cases [30].

The findings of this review confirmed that surgical excision, often combined with electrocauterization of the base, remains the cornerstone of management for orofacial rhinosporidiosis. This is supported by previous reports, since meticulous removal of the lesion is essential to prevent recurrence [3,16,19,32]. However, surgical excision alone is not always curative, especially in cases where complete clearance is difficult due to anatomical constraints or lesion size. Adjunctive therapy with Dapsone was widely reported in the reviewed cases, with most authors justifying its use based on the drug's ability to inhibit maturation of sporangia and promote fibrosis of lesion stroma [8,10,21,34]. In many cases, patients who received post-operative Dapsone remained recurrence-free during follow-up. However, recurrences still occurred in some cases despite medical therapy, which may reflect incomplete surgical removal or variability in individual drug responsiveness [4,20,29]. This highlights that Dapsone is not a substitute for surgery but serves as a valuable adjunct. The exceptional cases of disseminated disease revealed an even greater therapeutic challenge. In such patients, conventional surgery is impractical, and pharmacological approaches have been explored. Some reports described attempts with antifungals or broad-spectrum antibiotics, but outcomes were inconsistent, and no drug has proven reliably effective in systemic disease [15,33,38]. These rare but severe cases emphasize the urgent need for research into alternative therapeutic strategies beyond the current surgical-medical model. An important observation in this review was the inadequacy of long-term follow-up in many reports. Since recurrence is a hallmark of rhinosporidiosis, often occurring months or years after treatment, short-term follow-up cannot be equated with cure. Studies that documented outcomes for more than a year provided more meaningful insights into recurrence rates, while those with only brief follow-up risked underestimating the true burden of relapse [17,41]. This highlights the significance of structured, long-term monitoring in affected patients, with follow-up extending beyond the immediate postoperative period.

The synthesis of case reports in this review reaffirms the established principles of rhinosporidiosis management but also exposes persistent gaps in evidence quality. The dependence on histopathology for diagnosis remains unchallenged, and surgery with adjunctive Dapsone continues to represent the most effective strategy for localized disease. However, recurrence persists as a challenge, partly due to incomplete excision, difficulty accessing deep sites, or inadequate follow-up. The review highlights the importance of suspecting rhinosporidiosis in patients presenting with chronic nasal or orofacial polyps, particularly in endemic regions. The classical strawberry-like morphology should prompt biopsy, and clinicians should aim for complete excision with electrocauterization, followed by medical therapy when appropriate. Structured follow-up extending beyond one year should be standard practice, given the risk of late recurrence. This review also highlights several unmet needs. Future case reports should adhere to standardized reporting frameworks to ensure completeness and reproducibility. Systematic documentation of adverse events, particularly with Dapsone, is necessary to balance efficacy with safety. Innovative therapeutic options for disseminated rhinosporidiosis warrant investigation, as conventional management remains inadequate for systemic disease.

Strengths and limitations: This review systematically synthesized evidence on orofacial rhinosporidiosis and provided the first structured framework on various clinical and management aspects. The strengths include comprehensive databases searched, strict eligibility criteria, and standardized quality assessment using the JBI checklist, which ensured methodological rigor. The review highlights consistent patterns of clinical features, confirms histopathology as the diagnostic gold standard, and evaluates combined surgical and pharmacological strategies. However, limitations arise from dependence on only case reports and small case series, which are prone to reporting bias, incomplete follow-up, and underreporting of adverse events. The geographic concentration of cases in endemic regions also restricts the generalizability. Future multicenter research is crucial to refine management strategies and improve outcomes in this neglected infection. Future studies should define clear diagnostic criteria, test treatment methods across centres, and set up regional records to monitor cases and patient recovery.

Conclusion

Hence, the present review reports published evidence on orofacial rhinosporidiosis and shows its relevance in routine clinical practice. Clinicians should consider the disease when patients present with strawberry-like polyps in the oral or sinonasal region and confirm diagnosis through histopathology, as imaging alone often proves inconclusive. Surgical excision with cauterization remains the mainstay of management, and adjunctive Dapsone may reduce recurrence, though safety monitoring is essential. The review highlights certain challenges, including late recurrences, inadequate reporting of adverse events, and the absence of effective options for disseminated disease. Standardized reporting using the CARE checklist, structured long-term follow-up, and exploration of novel pharmacological or immunological therapies are strongly recommended.

Funding

This study was self-funded.

Conflicts of interest

The authors declare that they have no conflict of interest.

Data availability statement

Not applicable as the topic is a systematic review and all the data used is available on the internet.

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Cite this article as: Chandrasekaran K, Sridhar C, Deshpande A, 2026. Etiology, clinical features, and management strategies in orofacial rhinosporidiosis: a systematic review. J Oral Med Oral Surg. 32: 7. https://doi.org/10.1051/mbcb/2025042

All Tables

Table I

Demographic and clinical characteristics of reported case reports of orofacial rhinosporidiosis.

In the text
Table II

Diagnostic modalities, therapeutic interventions, and outcomes in reported cases of orofacial rhinosporidiosis.

In the text
Table III

Quality assessment for case reports done using the tool for evaluating the methodological quality of case reports and case series [31].

In the text
Table IV

Risk of bias assessment individual studies - JBI's tool for case series.

In the text

All Figures

Thumbnail: Fig. 1 Refer to the following caption and surrounding text. Fig. 1

PRISMA flowchart for the review.
In the text

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