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Bloody tears from lacrimal sac rhinosporidiosis

      Lacrimal sac rhinosporidiosis is an uncommon infection and to our knowledge has not been reported in Canada. Rhinosporidium seeberi is a fungus-like parasite with an evolutionary origin near the animal-fungal divergence that causes chronic infection of the mucous membranes of the upper respiratory tract.
      • Herr R.A.
      • Ajello L.
      • Taylor J.W.
      • Arseculeratne S.N.
      • Mendoza L.
      Phylogenetic analysis of Rhinosporidium seeberi's 18S small-subunit ribosomal DNA groups this pathogen among members of the protoctistan Mesomycetozoa clade.
      It is endemic in India and Southeast Asia, but rarely a cause of disease outside of this region.
      • Arseculeratne S.N.
      Recent advances in rhinosporidiosis and Rhinosporidium seeberi.
      A healthy 35-year-old female presented with acute onset unilateral bloody tears. There was no history of trauma, recent travel, or other constitutional symptoms. She resided in Bangladesh before immigration to Canada 14 years earlier. Examination revealed copious bloody, mucopurulent discharge from the left upper and lower puncta, with spurting during manual lacrimal sac compression. There was tenderness in the medial canthal region and mild swelling, but no erythema or warmth. Ocular, orbital, and nasal speculum examinations were otherwise normal. The patient began a course of oral cephalexin and the bloody tears resolved at 1 week, but the discomfort persisted. The improvement could have been related to clearance of bacterial superinfection. The lacrimal system was patent to irrigation. A computed tomography scan (Fig. 1) showed a left lacrimal sac mass, without bone destruction. The mass showed central gadolinium enhancement with magnetic resonance imaging, with loculated fluid peripheral to the mass, and dilatation of the upper part of the left nasolacrimal duct. An open excisional biopsy was performed. Frozen sections showed fungal elements and no evidence of neoplasm; therefore a left external dacryocystorhinostomy was performed. Permanent sections revealed features pathognomonic for R. seeberi infection (Fig. 2). Innumerable cystic structures, many filled with amorphous material surrounded by a thick, sometimes birefringent, capsule were seen. Occasional cysts contained spores, with the majority of cysts either impinging on the overlying mucosa or present in the immediate submucosa. The postoperative course was unremarkable. Infectious disease consultation deemed systemic dapsone treatment unnecessary. The patient has been followed for more than 5 years without recurrence.
      Figure thumbnail gr1
      Fig. 1Axial computed tomographic scan with contrast showing a heterogeneous mass in the area of the left lacrimal sac, with enhancement of the rim and no bone invasion.
      Figure thumbnail gr2
      Fig. 2Multiple thick-walled trophocytes (arrowheads) and a ruptured sporangium releasing its numerous endospores at the mucosal surface (arrow) confirming the diagnosis of Rhinosporidium seeberi (hematoxylin phloxine saffron, original magnification ×150).
      Ocular rhinosporidiosis accounts for 15% of R. seeberi infections, with conjunctival involvement in the majority of cases.
      • Arseculeratne S.N.
      Recent advances in rhinosporidiosis and Rhinosporidium seeberi.
      Lacrimal sac involvement is rare.
      • Pushker N.
      • Kashyap S.
      • Bajaj M.S.
      • et al.
      Primary lacrimal sac rhinosporidiosis with grossly dilated sac and nasolacrimal duct.
      Although rarely encountered in Western countries historically, rhinosporidiosis has become more frequent with immigrants from endemic areas acquiring the infection in their native countries.
      • Harissi-Dagher M.
      • Robillard N.
      • Corriveau C.
      • Mabon M.
      • Allaire G.S.
      Histopathologically confirmed ocular rhinosporidiosis in two Canadians.
      There is no evidence that rhinosporidiosis is transmissible between humans.
      • Arseculeratne S.N.
      Recent advances in rhinosporidiosis and Rhinosporidium seeberi.
      Most cases are sporadic, with transmission presumed to be from exposure of traumatized epithelium to water contaminated with R. seeberi.
      • Arseculeratne S.N.
      Recent advances in rhinosporidiosis and Rhinosporidium seeberi.
      Only two other cases of ocular rhinosporidiosis, both involving conjunctiva, have been reported previously in Canada.
      • Harissi-Dagher M.
      • Robillard N.
      • Corriveau C.
      • Mabon M.
      • Allaire G.S.
      Histopathologically confirmed ocular rhinosporidiosis in two Canadians.
      These cases, along with an additional report of nasal rhinosporidiosis occurring in a dog from Ontario,
      • Hoff B.
      • Hall D.A.
      Rhinosporidiosis in a dog.
      support the possibility of the organism existing locally in our temperate climate.
      Reported features of lacrimal sac rhinosporidiosis include painless, soft, fluctuant swelling of the lacrimal sac with partial nasolacrimal duct obstruction, and epistaxis, although our patient did have tenderness that is atypical. The dilated lacrimal sac often contains polypoidal growths or diverticula.
      • Pushker N.
      • Kashyap S.
      • Bajaj M.S.
      • et al.
      Primary lacrimal sac rhinosporidiosis with grossly dilated sac and nasolacrimal duct.
      Excisional biopsy is the treatment of choice, with recurrences possible from incomplete excision or seeding of endospores into adjacent tissue during removal.

      References

        • Herr R.A.
        • Ajello L.
        • Taylor J.W.
        • Arseculeratne S.N.
        • 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: 2750-2754
        • Arseculeratne S.N.
        Recent advances in rhinosporidiosis and Rhinosporidium seeberi.
        Indian J Med Microbiol. 2002; 20: 119-131
        • Pushker N.
        • Kashyap S.
        • Bajaj M.S.
        • et al.
        Primary lacrimal sac rhinosporidiosis with grossly dilated sac and nasolacrimal duct.
        Ophthal Plast Reconstr Surg. 2009; 25: 234-235
        • Harissi-Dagher M.
        • Robillard N.
        • Corriveau C.
        • Mabon M.
        • Allaire G.S.
        Histopathologically confirmed ocular rhinosporidiosis in two Canadians.
        Can J Ophthalmol. 2006; 41: 226-229
        • Hoff B.
        • Hall D.A.
        Rhinosporidiosis in a dog.
        Can Vet J. 1986; 27: 231-232