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Rhinosporidiosis of the lacrimal sac masquerading as chronic dacryocystitis: a rare presentation

ABSTRACT

Rhinosporidiosis is a chronic infection of the mucous membrane caused by the Rhinosporiduim seeberi, which infects through transepithelial penetration. Although described worldwide, this entity is mostly found in the western hemisphere, afflicting young people, predominantly males, associated in many cases with recreational or professional contact with bath in ponds, rivers, or stagnant waters. The clinical features are varied depending on the affected membrane, in some cases mimicking other diseases postponing the correct diagnosis. Although nasal obstruction and epistaxis are the common clinical presentations in sinonasal rhinosporidiosis, patients with epiphora without a nasal mass often challenge the diagnosis. In the present case, we have documented a case of isolated lacrimal sac rhinosporidiosis masquerading as chronic dacryocystitis, which was successfully managed by endoscopic excision, accompanied by a literature review.

Keywords
Lacrimal Apparatus Diseases; Eye Diseases; Rhinosporidiosis; Mesomycetozoea infections; Endemic Diseases

INTRODUCTION

Rhinosporidiosis is a chronic granulomatous disease caused by Rhinosporidium seeberi, frequently encountered in the southern zone of India and Sri Lanka.11 Kumari R, Laxmisha C, Thappa DM. Disseminated cutaneous rhinosporidiosis. Dermatol Online J. 2005;11(1):19. PMid:15748560. The nose and nasopharynx are the most common site affected by the disease and patients usually present with a painless mass with a history of nasal bleeding.22 Thappa DM, Venkatesan S, Sirka CS, Jaisankar TJ, Gopalkrishnan, Ratnakar C. Gopalkrishnan, Ratnakar C. Disseminated cutaneous rhinosporidiosis. J Dermatol. 1998;25(8):527-32. http://dx.doi.org/10.1111/j.1346-8138.1998.tb02449.x. PMid:9769599.
http://dx.doi.org/10.1111/j.1346-8138.19...
It often presents as a polypoidal mass in the nasal cavity33 Gonazalez G, Viada J, Escalona A, Na’quira N. Nasal rhinosporidiosis - four cases relate literature review. Int Arch Otorhinolaryngol. 2007;11:428-9.. Although nasal obstruction and epistaxis are the common clinical features, the epiphora as the single symptom is a challenging diagnosis. In the present case, we have documented a case of isolated lacrimal sac rhinosporidiosis masquerading as chronic dacryocystitis, which was successfully managed by endoscopic excision.

CASE REPORT

A 25-year-male patient presented to the outpatient clinic with a swelling below the medial canthus of the right eye involving the lower eyelid for 12 months (Figure 1A). A history of epiphora was present for the last six months. There was no history of nasal obstruction or epistaxis in the past. Anterior rhinoscopy did not reveal any abnormality in the nasal cavity. Nasal patency was found to be equal in both nasal cavities. A non-contrast CT scan was suggestive of a soft tissue density in the lacrimal sac region without any erosion of the lacrimal bone (Figure 1B).

Figure 1
A – External view of the patient’s face showing the swelling below the medial canthus of the right eye; B – Non-contrast CT scan (Coronal plane) revealed a soft tissue density in the lacrimal sac without bony erosion (arrowhead).

The visual acuity was normal in both eyes. Syringing the right lacrimal passage revealed a hard stop, consistent with the obstruction of the right nasolacrimal duct. After the informed written consent, the patient was planned for endoscopic dacryocystorhinostomy (DCR). After drilling out of the ascending process of maxilla, a dilated lacrimal sac was detected. On incising the sac, a polypoidal bleeding mass was detected obliterating the whole lumen. The mass was completely excised after a gentle dissection of the sac wall. Endoscopic dacryocystorhinostomy was performed as an adjunctive procedure to maintain the lacrimal patency. The histopathology of the specimen was confirmed to be rhinosporidiosis (Figure 2).

Figure 2
Photomicrograph of the surgical specimen. A – showing globular cysts representing the sporangia containing daughter spores in different stages of development (H&E 100x); B – Shows sporangia which are surrounded by dense chronic inflammatory infiltrate comprising of lymphocytes and plasma cells (H&E 200x).

The patient is on regular follow in the rhinology clinic and the ophthalmology department for the past 12 months without any recurrence of the disease.

DISCUSSION

Rhinosporidiosis is a chronic granulomatous disease affecting both humans and animals, caused by Rhinosporidium seeberi. Although it was first classified under fungal disease under ICD10, it is now considered an aquatic protistan parasite belonging to the class Mesomycetozoa44 Herr RA, Ajello L, Taylor JW, Arseculeratne SN, Mendoza L. Phylogenetic analysis of Rhinosporidium Seeberi’s 18S Small-Subunit Ribosomal DNA groups this pathogen among members of the protoctistan mesomycetozoa clade. J Clin Microbiol. 1999;37(9):2750-4. http://dx.doi.org/10.1128/JCM.37.9.2750-2754.1999. PMid:10449446.
http://dx.doi.org/10.1128/JCM.37.9.2750-...
. The fish and the aquatic amphibians are considered the natural host of the parasite, Humans acquire the disease by accidental contact or bathing in ponds, rivers, or working in stagnant water.55 Karthikeyan P, Vijayasundaram S, Pulimoottil DT. A retrospective epidemiological study of rhinosporidiosis in a Rural Tertiary Care Centre in Pondicherry. J Clin Diagn Res. 2016;10(5):MC04-08. http://dx.doi.org/10.7860/JCDR/2016/17465.7788. PMid:27437252.
http://dx.doi.org/10.7860/JCDR/2016/1746...
Transmission from animals to humans often occurs through water contact. Although already encountered worldwide, it notably endemic in the Southern part of India and Sri Lanka.66 Morelli L, Polce M, Piscioli F, et al. Human nasal rhinosporidiosis: an Italian case report. Diagn Pathol. 2006;1(1):25.http://dx.doi.org/10.1186/1746-1596-1-25. PMid:16945122.
http://dx.doi.org/10.1186/1746-1596-1-25...
Rhinosporidiosis was first identified in 1892, but comprehensively described in 1900 by Guillermo Seeber in a farm-worker in Argentina with impaired breathing by a nasal mass. Kuriakose77 Kuriakose ET. Oculosporidiosis: rhinosporidiosis of the eye. Br J Ophthalmol. 1963;47(6):346-9. http://dx.doi.org/10.1136/bjo.47.6.346. PMid:14189701.
http://dx.doi.org/10.1136/bjo.47.6.346...
first described the ocular rhinosporidiosis in 1963. The nose and nasopharynx are the primary sites to be affected by the infection (78%), followed by the conjunctiva (15%).55 Karthikeyan P, Vijayasundaram S, Pulimoottil DT. A retrospective epidemiological study of rhinosporidiosis in a Rural Tertiary Care Centre in Pondicherry. J Clin Diagn Res. 2016;10(5):MC04-08. http://dx.doi.org/10.7860/JCDR/2016/17465.7788. PMid:27437252.
http://dx.doi.org/10.7860/JCDR/2016/1746...
The lacrimal system becomes affected in 14.3% to 59.6% of cases of ocular rhinosporidiosis.88 Pushker N, Kashyap S, Bajaj MS, et al. Primary lacrimal sac rhinosporidiosis with grossly dilated sac and nasolacrimal duct. Ophthal Plast Reconstr Surg. 2009;25(3):234-5. http://dx.doi.org/10.1097/IOP.0b013e3181a39495. PMid:19454941.
http://dx.doi.org/10.1097/IOP.0b013e3181...

9 Shrestha SP, Hennig A, Parija SC. Prevalence of rhinosporidiosis of the eye and its adnexa in Nepal. Am J Trop Med Hyg. 1998;59(2):231-4. http://dx.doi.org/10.4269/ajtmh.1998.59.231. PMid:9715938.
http://dx.doi.org/10.4269/ajtmh.1998.59....

10 Chowdhury RK, Behera S, Bhuyan D, Das G. Oculosporidiosis in a tertiary care hospital of western Orrisa, India: a case series. Indian J Ophthalmol. 2007;55(4):299-301. http://dx.doi.org/10.4103/0301-4738.33045. PMid:17595481.
http://dx.doi.org/10.4103/0301-4738.3304...
-1111 Suseela V, Subramaniam KS. Rhinosporidiosis and the eyes. Indian J Ophthalmol. 1976;23(4):1-4. PMid:1031152. The ocular involvement is mostly presented by a conjunctival polyp (77.6%), either in the upper or lower lid of the fornix. Approximately 26% of the cases of ocular rhinosporidiosis are associated with either nasal or conjunctival involvement.1212 Basu SK, Bain J, Maity K, et al. Rhinosporidiosis of lacrimal sac: an interesting case of orbital swelling. J Nat Sci Biol Med. 2016;7(1):98-101. http://dx.doi.org/10.4103/0976-9668.175102. PMid:27003980.
http://dx.doi.org/10.4103/0976-9668.1751...
Isolated lacrimal sac rhinosporidiosis is very rare in the clinical practice with only a few reports in the literature.99 Shrestha SP, Hennig A, Parija SC. Prevalence of rhinosporidiosis of the eye and its adnexa in Nepal. Am J Trop Med Hyg. 1998;59(2):231-4. http://dx.doi.org/10.4269/ajtmh.1998.59.231. PMid:9715938.
http://dx.doi.org/10.4269/ajtmh.1998.59....
,1010 Chowdhury RK, Behera S, Bhuyan D, Das G. Oculosporidiosis in a tertiary care hospital of western Orrisa, India: a case series. Indian J Ophthalmol. 2007;55(4):299-301. http://dx.doi.org/10.4103/0301-4738.33045. PMid:17595481.
http://dx.doi.org/10.4103/0301-4738.3304...
It is always a challenge to suspect a case of isolated lacrimal sac rhinosporidiosis as the clinical feature always resembles a chronic dacryocystitis. Radiological evaluation (CT scan /MRI) can be undertaken in selective cases to rule out a similar pathology.1313 Ashworth JH. On RhinosporidiumSeeberi (Wernicke 1903), with special reference to its sporulation and affinities. Trans R Soc Edinb. 1923;53:30142. We undertook a literature review based on the PubMed database using “rhinosporidiosis and lacrimal sac” as the keywords. A total of 29 articles were retrieved, which comprised publications from 1973 to 2019. 156 cases of lacrimal sac rhinosporidiosis have been reported and are summarized in Table 1, including the predominant symptoms, the primary site of involvement, and the various treatment protocols. From the 156 cases, the main symptom was lacrimal sac swelling, which was observed in almost all cases. Epiphora was seen in 10.25% of the cases. In our case, epiphora and lacrimal swelling were the only presenting complaints that misled the working diagnosis toward a dacryocystitis/dacryocystocele. Epiphora can be continuous,44 Herr RA, Ajello L, Taylor JW, Arseculeratne SN, Mendoza L. Phylogenetic analysis of Rhinosporidium Seeberi’s 18S Small-Subunit Ribosomal DNA groups this pathogen among members of the protoctistan mesomycetozoa clade. J Clin Microbiol. 1999;37(9):2750-4. http://dx.doi.org/10.1128/JCM.37.9.2750-2754.1999. PMid:10449446.
http://dx.doi.org/10.1128/JCM.37.9.2750-...
,1717 Rajesh Raju G, Sandeep S. Lacrimal sac rhinosporidiosis and surgical management by transnasal endoscopic excision: a case series. Laryngoscope. 2018;128(12):2693-6. http://dx.doi.org/10.1002/lary.27322. PMid:30194840.
http://dx.doi.org/10.1002/lary.27322...
,3232 Watve JK, Mane RS, Mohite AA, Patil BC. Lacrimal sac Rhinosporidiosis. Indian J Otolaryngol Head Neck Surg. 2006;58(4):399-400. http://dx.doi.org/10.1007/BF03049611. PMid:23120364.
http://dx.doi.org/10.1007/BF03049611...
intermittent,1818 Girish N, Prathima. Rhinosporidiosis of lacrimal sac: a case report. Int Clin Pathol J. 2017;4(4):85-6. http://dx.doi.org/10.15406/icpjl.2017.04.00099.
http://dx.doi.org/10.15406/icpjl.2017.04...
,2323 Sah BP, Chettri ST, Si S, Kandel DR, Ir D. Lacrimal sac rhinosporidiosis: an unusual case report. Am J Med Case Rep. 2014;2(4):84-6. http://dx.doi.org/10.12691/ajmcr-2-4-4.
http://dx.doi.org/10.12691/ajmcr-2-4-4...
,2929 Ghosh A, Saha S, Srivastava A, Mishra M. Rhinosporidiosisunusualpresentations. Indian J Otolaryngol Head Neck Surg. 2008;60(2):159-62. http://dx.doi.org/10.1007/s12070-008-0003-3. PMid:23120527.
http://dx.doi.org/10.1007/s12070-008-000...
,3333 Nerurkar NK, Bradoo RA, Joshi AA, Shah J, Tandon S. Lacrimal sac rhinosporidiosis: a case report. Am J Otolaryngol. 2004;25(6):423-5. http://dx.doi.org/10.1016/j.amjoto.2004.04.012. PMid:15547812.
http://dx.doi.org/10.1016/j.amjoto.2004....
bloody2424 Guru RK, Pradhan DK. Rhinosporidiosis with special reference to extra nasal presentation. J Evol Med Dental Sci. 2014;3(22):6189-99. http://dx.doi.org/10.14260/jemds/2014/2721.
http://dx.doi.org/10.14260/jemds/2014/27...
,2727 Mithal C, Agarwal P, Mithal N. Ocular and adnexal rhinosporidiosis: the clinical profile and treatment outcomes in a tertiary eye care centre. Nepal J Ophthalmol. 2012;4(1):45-8. http://dx.doi.org/10.3126/nepjoph.v4i1.5849. PMid:22343995.
http://dx.doi.org/10.3126/nepjoph.v4i1.5...
, mucopurulent,1818 Girish N, Prathima. Rhinosporidiosis of lacrimal sac: a case report. Int Clin Pathol J. 2017;4(4):85-6. http://dx.doi.org/10.15406/icpjl.2017.04.00099.
http://dx.doi.org/10.15406/icpjl.2017.04...
,3434 Thakur SKD, Sah SP, Badhu BP. Oculosporidiosis in Eastern Nepal: a report of five cases. Southeast Asian J Trop Med Public Health. 2002;33(2):362-4. PMid:12236438. or sometimes it is absent.55 Karthikeyan P, Vijayasundaram S, Pulimoottil DT. A retrospective epidemiological study of rhinosporidiosis in a Rural Tertiary Care Centre in Pondicherry. J Clin Diagn Res. 2016;10(5):MC04-08. http://dx.doi.org/10.7860/JCDR/2016/17465.7788. PMid:27437252.
http://dx.doi.org/10.7860/JCDR/2016/1746...
,1515 Suneer R, Sivasankari L. Clinical profile of rhinosporidiosis in a tertiary care centre of an endemic area. Int J Otorhinolaryngol Head Neck Surg. 2018;4(3):659-62. http://dx.doi.org/10.18203/issn.2454-5929.ijohns20181382.
http://dx.doi.org/10.18203/issn.2454-592...
,1919 Chakraborti C, Barua N, Chishti RK, Kumar S. A rare case of lacrimal sac diverticulum associated with rhinosporidiosis. J Clin Ophthalmol Res. 2017;5:97-9.,2020 Jamison A, Crofts K, Roberts F, Gregory ME. Educational report: a case of lacrimal sac rhinosporidiosis. Orbit. 2016;35(5):254-7. http://dx.doi.org/10.1080/01676830.2016.1193529. PMid:27541939.
http://dx.doi.org/10.1080/01676830.2016....
,2525 Mukherjee PK, Shukla IM, Deshpande M, Kher P. Rhinosporidiosis of the lacrimal sac. Indian J Ophthalmol. 1982;30:513.,3636 Suseela V, Subramaniam KS. Rhinosporidiosis and the eyes. Indian J Ophthalmol. 1975;23(4):1-4. PMid:1031152.,3838 Bain J, Basu SK, Maity K, et al. Rhinosporidiosis of lacrimal sac: an interesting case of orbital swelling. J Nat Sci Biol Med. 2016;7(1):98-101. http://dx.doi.org/10.4103/0976-9668.175102. PMid:27003980.
http://dx.doi.org/10.4103/0976-9668.1751...
The absence of lacrimal swelling could be due to the spread of infection through the peri canalicular space of the lacrimal sac without affecting the drainage pathway.1818 Girish N, Prathima. Rhinosporidiosis of lacrimal sac: a case report. Int Clin Pathol J. 2017;4(4):85-6. http://dx.doi.org/10.15406/icpjl.2017.04.00099.
http://dx.doi.org/10.15406/icpjl.2017.04...
,3939 Jain MR, Sahai R. Rhinosporidiosis of the lacrimal sac. Indian J Ophthalmol. 1974;22(3):29-30. PMid:4465292. The etiological agent can reach the lacrimal sac from the nose or conjunctiva,4040 Sood NN, Rao SN. Rhinosporidium granuloma of the conjunctiva. Br J Ophthalmol. 1967;51(1):61-4. http://dx.doi.org/10.1136/bjo.51.1.61. PMid:6018553.
http://dx.doi.org/10.1136/bjo.51.1.61...
unlike the present case where the mass was found only in the lacrimal sac, without a synchronous nasal involvement. Due to the complete obstruction of the lacrimal flow, the patient presented with epiphora resembling a chronic dacryocystitis. There are various schools of thought on the possible route by which rhinosporidiosis involves the lacrimal sac. Some researchers think it spreads through the nasolacrimal duct to the lacrimal sac and others comment that it does not spread through the duct because of the presence of the valve.1616 Prabhu MS, Irodi A, Khiangte HL, Rupa V, Naina P. Imaging features of rhinosporidiosis on contrast CT. Indian J Radiol Imaging. 2013;23(3):212-8. http://dx.doi.org/10.4103/0971-3026.120267. PMid:24347850.
http://dx.doi.org/10.4103/0971-3026.1202...
The imaging examination of the nose and paranasal sinus is considered a key resource to diagnose patients with isolated rhinosporidiosis, as it is often mistaken with chronic dacryocystitis. On computed tomography, it appears as a homogenously enhanced mass in the inferior meatus extending to the lacrimal sac with the erosion of the adjacent bone.4141 Gonazalez G, Viada J, Escalona A, Na’quira N. Nasal rhinosporidiosis - four cases relate literaturereview. Int Arch Otorhinolaryngol. 2007;11:428-9. However, in the present case, the mass was only limited to the lacrimal sac without any radiological evidence of bony erosion. Although the majority of patients with sinonasal rhinosporidiosis are diagnosed on the clinical findings, histopathology is warranted for its confirmation.4242 Karunaratne WA. The pathology of rhinosporidiosis. J Pathol Bacteriol. 1936;42(1):193-202. http://dx.doi.org/10.1002/path.1700420121.
http://dx.doi.org/10.1002/path.170042012...
On histopathology, the lesion presents with a distinctive morphology, consisting of globular cysts representing the thick-walled sporangia containing more than 1000 daughter spores in different stages of development, accompanied by a mixed inflammatory infiltrate. The organism is stained by periodic acid Schiff (PAS), Gomori’s methenamine silver and Mucicarmine.2222 Nuruddin M, Mudhar HS, Osmani M, Roy SR. Lacrimal sac rhinosporidiosis: clinical profile and surgical management by modified dacryocystorhinostomy. Orbit. 2014;33(1):29-32. http://dx.doi.org/10.3109/01676830.2013.844170. PMid:24195743.
http://dx.doi.org/10.3109/01676830.2013....

Table 1
Shows the review of literature of the lacrimal sac rhinosporidiosis

Surgical excision with electrocautery of the base of the lesion is considered the treatment of choice for patients with sinonasal rhinosporidiosis. The majority of the patients are managed through external dacryocystorhinostomy.2121 Mishra LK, Gupta S, Pradhan SK, Baisakh MR. Lacrimal sac rhinosporidiosis. Plast Aesthet Res. 2015;2(6):353-6. http://dx.doi.org/10.4103/2347-9264.169501.
http://dx.doi.org/10.4103/2347-9264.1695...
,2626 Belliveau MJ, Strube YNJ, Dexter DF, Kratky V. Bloody tears from lacrimal sac rhinosporidiosis. Can J Ophthalmol. 2012;47(5):e23-4. http://dx.doi.org/10.1016/j.jcjo.2012.03.019. PMid:23036556.
http://dx.doi.org/10.1016/j.jcjo.2012.03...
,2828 Rogers S, Waring D, Martin P. Recurrent lacrimal sac rhinosporidiosis involving the periocular subcutaneous tissues, nasolacrimal duct and nasopharynx. Orbit. 2012;31(5):358-60. http://dx.doi.org/10.3109/01676830.2012.700548. PMid:23030407.
http://dx.doi.org/10.3109/01676830.2012....
,3030 Varshney S, Bist SS, Gupta P, Gupta N, Bhatia R. Lacrimal sac diverticulum due to rhinosporidiosis. Indian J Otolaryngol Head Neck Surg. 2007;59(4):353-6. http://dx.doi.org/10.1007/s12070-007-0100-8. PMid:23120472.
http://dx.doi.org/10.1007/s12070-007-010...
,3131 Ghorpade A, Gurumurthy J, Banerjee PK, Banerjee AK, Bhalla M, Ravindranath M. Oculosporidiosis presenting as an under-eye swelling. Indian J Dermatol Venereol Leprol. 2007;73(3):196-7. http://dx.doi.org/10.4103/0378-6323.32749. PMid:17558058.
http://dx.doi.org/10.4103/0378-6323.3274...
,3535 Krishnan MM, Kawatra VK, Rao VA, Ratnakar C. Diverticulum of the lacrimal sac associated with rhinosporidiosis. Br J Ophthalmol. 1986;70(11):867-8. http://dx.doi.org/10.1136/bjo.70.11.867. PMid:3098278.
http://dx.doi.org/10.1136/bjo.70.11.867...
,3737 David SS, Sivaramasubrahmanyam P. Ocular rhinosporidiosis, a study of twenty one cases. Indian J Ophthalmol. 1973;21:204-7. In contrast, in the present case, the mass was excised through a transnasal endoscopic procedure, similar to the standard endoscopic dacryocystorhinostomy, as described by Gupta et al.1414 Gupta N, Singla P, Pradhan B, Gurung U. Lacrimal sac rhinosporidiosis: case report and review of literature with a new grading system to optimize treatment. Saudi Journal of Ophthalmology. 2019;33(3):283-90. http://dx.doi.org/10.1016/j.sjopt.2019.05.002.
http://dx.doi.org/10.1016/j.sjopt.2019.0...
Although medical treatment cannot be denied, its role is very much limited to patients where complete surgical resection is not possible due to the systemic spread of the disease. Dapsone, because of its anti rhinosporidial activity, can be used to arrest the maturation of sporangia and to promote fibrosis in the stroma. It can be used with 100 mg once/twice daily for 3-6 months in the preoperative period to prevent the recurrence.44 Herr RA, Ajello L, Taylor JW, Arseculeratne SN, Mendoza L. Phylogenetic analysis of Rhinosporidium Seeberi’s 18S Small-Subunit Ribosomal DNA groups this pathogen among members of the protoctistan mesomycetozoa clade. J Clin Microbiol. 1999;37(9):2750-4. http://dx.doi.org/10.1128/JCM.37.9.2750-2754.1999. PMid:10449446.
http://dx.doi.org/10.1128/JCM.37.9.2750-...
,1818 Girish N, Prathima. Rhinosporidiosis of lacrimal sac: a case report. Int Clin Pathol J. 2017;4(4):85-6. http://dx.doi.org/10.15406/icpjl.2017.04.00099.
http://dx.doi.org/10.15406/icpjl.2017.04...
,2323 Sah BP, Chettri ST, Si S, Kandel DR, Ir D. Lacrimal sac rhinosporidiosis: an unusual case report. Am J Med Case Rep. 2014;2(4):84-6. http://dx.doi.org/10.12691/ajmcr-2-4-4.
http://dx.doi.org/10.12691/ajmcr-2-4-4...
,2424 Guru RK, Pradhan DK. Rhinosporidiosis with special reference to extra nasal presentation. J Evol Med Dental Sci. 2014;3(22):6189-99. http://dx.doi.org/10.14260/jemds/2014/2721.
http://dx.doi.org/10.14260/jemds/2014/27...
,2929 Ghosh A, Saha S, Srivastava A, Mishra M. Rhinosporidiosisunusualpresentations. Indian J Otolaryngol Head Neck Surg. 2008;60(2):159-62. http://dx.doi.org/10.1007/s12070-008-0003-3. PMid:23120527.
http://dx.doi.org/10.1007/s12070-008-000...
Due to the advancement of the transnasal endoscopic procedures, there has been a decrease in the disease’s recurrence rate due to enhanced visualization ensuring complete removal of the disease, as observed in the present case. Dapsone was not prescribed in our present as the disease was only limited to a lacrimal sac where complete removal was ensured with the use of wide-angle rigid nasal endoscopes. Despite the low recurrence rate, each patient needs a close follow-up with routine nasal endoscopy and lacrimal patency tests to find out the early recurrence of the disease. A high index of suspicion of the disease, appropriate radiological examination, and histopathology are considered the cornerstones for the diagnosis of an isolated rhinosporidiosis of the lacrimal sac. As described in the present case, isolated rhinosporidiosis of the lacrimal sac can be a potential differential diagnosis of chronic dacryocystitis, especially with atypical presentation.

CONCLUSION

Although patients with ocular rhinosporidiosis are mostly diagnosed on the clinical findings, i.e., with nasal obstruction and epistaxis, it is often a challenge to diagnose a case where epiphora is the single complaint. In combination with the radiological and histopathological examination, a proper clinical history is mandatory for the accurate diagnosis of atypical rhinosporidiosis.

All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or National research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards. Written informed consent has been taken from the patient prior to surgery and has been informed to the Institute reviewer board.

  • How to cite: Pradhan P, Samal S. Rhinosporidiosis of the lacrimal sac masquerading as chronic dacryocystitis: a rare presentation. Autops Case Rep [Internet]. ano Mês-Mês;v(n):e2020214. https://doi.org/10.4322/acr.2020.214
  • The article was developed in AIIMS, Bhubaneswar, India.
  • Ethics Statement: informed consent has been taken signed by the patient authorizing data and his face's picture for publication.
  • Financial support: none.

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Publication Dates

  • Publication in this collection
    20 Jan 2021
  • Date of issue
    2021

History

  • Received
    29 May 2020
  • Accepted
    21 July 2020
Hospital Universitário da Universidade de São Paulo Hospital Universitário da Universidade de São Paulo, Av. Prof. Lineu Prestes, 2565 - Cidade Universitária, 05508-000 - São Paulo - SP - Brasil, (16) 3307-2068, (16) 3307-2068 - São Paulo - SP - Brazil
E-mail: autopsy.hu@gmail.com