|Year : 2017 | Volume
| Issue : 2 | Page : 97-99
A rare case of lacrimal sac diverticulum associated with rhinosporidiosis
Chandana Chakraborti, Nabanita Barua, Rosy Kahakashan Chishti, Sheuli Kumar
Department of Ophthalmology, Calcutta National Medical College and Hospital, Kolkata, West Bengal, India
|Date of Submission||14-Jan-2016|
|Date of Acceptance||17-Jun-2016|
|Date of Web Publication||25-Apr-2017|
A/1/1, Pearl Apartment, 50B, Kailas Bose Street, Kolkata - 700 006, West Bengal
Source of Support: None, Conflict of Interest: None
A 13-year-old female presented with gradually increasing painless swelling of the left lower eyelid for 2 years with no history of watering, ocular trauma, or sinus infection. The swelling (10 mm × 5 mm) was soft, nontender, and free from the skin. The left nasolacrimal duct was patent on syringing. There was regurgitation of serosanguinous fluid from lower puncta on firm pressure over the swelling. Computed tomography scan showed a soft tissue-enhancing mass near the left medial canthus. Dacryocystography revealed diverticulum of the left lacrimal sac. The diverticulum was excised leaving the sac behind. Histopathological examination of the excised mass was found to be infected with rhinosporidiosis. A fistula developed postoperatively discharging spores. Dacryocystectomy with fistulectomy and electrocauterization of the area was done. No recurrence was seen till 12 months follow-up. After PubMed search, we have found only one such case report. We would like to discuss the clinical presentation and management of this rare case.
Keywords: Dacryocystography, fistula, lacrimal sac diverticulum, rhinosporidiosis
|How to cite this article:|
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
|How to cite this URL:|
Chakraborti C, Barua N, Chishti RK, Kumar S. A rare case of lacrimal sac diverticulum associated with rhinosporidiosis. J Clin Ophthalmol Res [serial online] 2017 [cited 2020 Jun 6];5:97-9. Available from: http://www.jcor.in/text.asp?2017/5/2/97/205184
Diverticulum of the lacrimal sac is a rare condition characterized by an outpouching from canaliculi or sac. It may be of congenital, inflammatory, or traumatic in origin. It arises mostly from lateral wall of the sac as it is covered by periorbita only. The diverticula may or may not be connected with the sac. It mostly stays asymptomatic unless infected secondarily. Inflammatory engorgement of vessel wall may interfere with the drainage resulting in recurrent dacryocystitis.,
Rhinosporidiosis is caused by Rhinosporidium seeberi, typically producing mucosal disease, mostly secondary to posttraumatic inoculation. Lacrimal sac diverticulum infected with Rhinosporidium is a rare entity.,,
| Case Report|| |
A 13-year-old female presented with gradually increasing, painless swelling of the left lower eyelid for 2 years. There was no history of watering from eyes or any other discharge or trauma around that region. On examination, vision was 20/20 in both eyes; anterior and posterior segments were within normal limits. A soft, nontender, 10 mm × 5 mm size swelling was found, extending from below the left inner canthus up to the medial two-third of the lower eyelid [Figure 1]. It was free from the skin but fixed to underlying structure without any sign of inflammation. On syringing, fluid passed fairly easily without alteration in size of the swelling. On firm pressure over the swelling, serosanguinous discharge came out through the lower puncta.
Microbiological examination with Gram stain, Giemsa stain, and KOH preparation of the discharge showed no microorganisms. Computed tomography scan of the orbit and paranasal sinuses revealed soft tissue-enhancing mass near the medial canthus of left orbit-partially eroding nasal bone and indenting left eyeball [Figure 2]. Paranasal sinuses were normal. Ultrasonography B-scan diagnosed cystic lesion in lacrimal sac area. Clinically, it was diagnosed as a case of lacrimal sac diverticulum and a dacryocystography was advised which confirmed it the presence of a diverticulum [Figure 3].
|Figure 2: Computed tomography scan shows soft tissue-enhancing mass near medial canthus with normal paranasal sinuses|
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|Figure 3: Dacryocystogram showing passage of contrast dye from the left lacrimal sac through a fistulous tract and pooling into a pouch|
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It was approached by an anterior orbitotomy via subciliary incision under general anesthesia; the diverticulum was found attached to underlying tissue from which it was dissected meticulously, sealing the neck of diverticulum by purse string suture [Figure 4]. The area was irrigated with povidone iodine, and the diverticulum was removed leaving the sac behind. Histopathological evaluation revealed fibrocollagenous cyst wall lined by tall columnar epithelium. Chronic inflammatory cells infiltrated along with rhinosporangium within mucosa and papilla were seen projecting within lumen [Figure 5]. Retrospectively, she gave a history of repeated bathing in pond. Based on this, we concluded it to be a case of left lacrimal sac diverticulum infected with rhinosporidiosis. The patient was started on tablet amoxicillin/clavulanate potassium (co-amoxiclav) 625 mg three times a day with tablet paracetamol 500 mg once daily for 5 days.
|Figure 4: Steps of dissection of the diverticulum through anterior orbitotomy|
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|Figure 5: Histopathological evaluation of diverticulum showing fibrocollagenous cyst wall lined by columnar epithelium, chronic inflammatory cells infiltration, rhinosporangium within mucosa, and papillary projection suggesting rhinosporidiosis|
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On the 1st postoperative day, the patient developed fistula-discharging spores [Figure 6]. Apart from regular dressing of the wound, ear-nose-throat consultation was done. Nasal endoscopy excluded any nasal spread of infection and she was discharged from the hospital. After 1 month, as the edema subsided, dacryocystectomy (DCT) with fistulectomy with extensive electrocauterization of the area under general anesthesia was done. Sac area was profusely irrigated with 5% povidone iodine during surgery. The patient received tablet amoxicillin/clavulanate potassium (co-amoxiclav) 625 mg three times a day with tablet paracetamol 500 mg once daily for 5 days.
Broad spectrum antibiotics for 5 days along with anti-inflammatory medication were received. Betadine dressing was continued for 1 week which yielded satisfactory apposition [Figure 7]. On 12 months follow-up, the patient was asymptomatic.
| Discussion|| |
Rhinosporidiosis of the eye and its adnexa has been reported to involve conjunctiva (69%), lacrimal sac (24%), canaliculi (4%), and lid and sclera (4%). Once infected, it usually remains asymptomatic in the tissue for as long as 30 years, only becoming symptomatic when secondarily infected. The primary site appears to be lacrimal sac, and nasal involvement is due to downward spread through nasolacrimal duct. In our patient, dacryocystography confirmed the presence of the diverticulum and its communication with the sac. This was the reason for serosanguinous fluid regurgitation on pressure in our patient although she never had complaint of any watering, pain, or redness of the eye. Our patient gave a history of bathing in pond regularly. Mode of infection of Rhinosporidium is thought to be contaminated stagnant water (e.g., river-sand) on abraded skin. Southern India and Sri Lanka are the endemic areas for rhinosporidiosis. The stagnant water, chemical constituents, and synergistic relation with other microbes may be the reason for persistence of this parasite.
The diagnosis of rhinosporidiosis is usually confirmed on histopathology. Proliferation of fibroconnective stroma, inflammatory infiltrate with thick-walled sporangium with many daughter spores in various developmental stage clinches the diagnosis. Stains such as periodic acid–Schiff diastase, Gomori methenamine-silver nitrate stain, and mucicarmine are used for diagnosing rhinosporidiosis., Our tissue also revealed similar features as mentioned in literature.
Medical therapy against Rhinosporidium is challenging as no antifungal/antimicrobial drug is effective against this parasite. Dapsone shows some benefits in preventing recurrence, by inhibiting the spores and inducing fibrosis of stroma. Bacterial infections are the most frequent complications. Antiseptics such as cetrimide-chlorhexidine, povidone iodine, and silver nitrate solutions show some anti-rhinosporidiosis activity. Later two drugs are available for ophthalmic use.,
The only proven treatment is surgical excision of both sac and diverticulum with complete removal of the lesions. A thorough removal of the affected tissue with extensive electrocauterization of the base is needed to prevent recurrence and its spread to nasal passage. We did DCT with fistulectomy with widespread cauterization of the surrounding area under general anesthesia. Postoperatively, dressing with betadine helped in healing. Till 1-year follow-up, there is no recurrence.
Lacrimal sac swelling in the presence of a patent syringing could be because of diverticulum. Serosanguinous discharge through the punctum can be indicative of rhinosporidiosis. The patient may be otherwise asymptomatic. Surgical treatment is the mainstay of treatment as medical management does not cure the disease.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]