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Year : 2017  |  Volume : 5  |  Issue : 2  |  Page : 97-99

A rare case of lacrimal sac diverticulum associated with rhinosporidiosis


Department of Ophthalmology, Calcutta National Medical College and Hospital, Kolkata, West Bengal, India

Date of Submission14-Jan-2016
Date of Acceptance17-Jun-2016
Date of Web Publication25-Apr-2017

Correspondence Address:
Chandana Chakraborti
A/1/1, Pearl Apartment, 50B, Kailas Bose Street, Kolkata - 700 006, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2320-3897.205184

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  Abstract 

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.[1] It may be of congenital, inflammatory, or traumatic in origin.[2] 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.[3],[4]

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.[3],[4],[5]


  Case Report Top


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.
Figure 1: Swelling below the left lower eyelid

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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.
Figure 6: Fistula with discharging spores on the 5th postoperative day

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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.
Figure 7: One month after second surgery, showing apposed healthy wound

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  Discussion Top


Rhinosporidiosis of the eye and its adnexa has been reported to involve conjunctiva (69%), lacrimal sac (24%), canaliculi (4%), and lid and sclera (4%).[6] Once infected, it usually remains asymptomatic in the tissue for as long as 30 years, only becoming symptomatic when secondarily infected.[2] The primary site appears to be lacrimal sac, and nasal involvement is due to downward spread through nasolacrimal duct.[3] 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.[2]

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.[7],[8] 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.[9],[10]

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.[11] 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.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Duke-Elder S, MacFaul PA. The ocular adnexa: Lacrimal, orbital, and para-orbital diseases. Cysts and diverticula. In: Duke-Elder S, editor. System of Ophthalmology. Vol. 13. St. Louis: CV Mosby; 1974. p. 733-5.  Back to cited text no. 1
    
2.
Varshney S, Bist SS, Gupta P, Gupta N, Bhatia R. Lacrimal sac diverticulum due to rhinosporidiosis. Indian J Otolaryngol Head Neck Surg 2007;59:353-6.  Back to cited text no. 2
    
3.
Krishnan MM, Kawatra VK, Rao VA, Ratnakar C. Diverticulum of the lacrimal sac associated with rhinosporidiosis. Br J Ophthalmol 1986;70:867-8.  Back to cited text no. 3
    
4.
Ormrod JN. Diverticulum of the lacrimal sac. Br J Ophthalmol 1958;42:526-8.  Back to cited text no. 4
    
5.
Fredricks DN, Jolley JA, Lepp PW, Kosek JC, Relman DA. Rhinosporidium seeberi: A human pathogen from a novel group of aquatic protistan parasites. Emerg Infect Dis 2000;6:273-82.  Back to cited text no. 5
    
6.
Jain MR, Sahai R. Rhinosporidiosis of lacrimal sac. Indian J Ophthalmol 1974;22:29-30.  Back to cited text no. 6
[PUBMED]  [Full text]  
7.
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;37:2750-4.  Back to cited text no. 7
    
8.
Arseculeratne SN. Recent advances in rhinosporidiosis and Rhinosporidium seeberi. Indian J Med Microbiol 2002;20:119-31.  Back to cited text no. 8
[PUBMED]  [Full text]  
9.
Kim JH, Chang HR, Woo KI. Multilobular lacrimal sac diverticulum presenting as a lower eyelid mass. Korean J Ophthalmol 2012;26:297-300.  Back to cited text no. 9
    
10.
Arseculeratne SN, Atapattu DN, Balasooriya P, Fernando R. The effects of biocides (antiseptics and disinfectants) on the endospores of Rhinosporidium seeberi. Indian J Med Microbiol 2006;24:85-91.  Back to cited text no. 10
[PUBMED]  [Full text]  
11.
Mukherjee B, Mohan A, Sumathi V, Biswas J. Infestation of the lacrimal sac by Rhinosporidium seeberi: A clinicopathological case report. Indian J Ophthalmol 2013;61:588-90.  Back to cited text no. 11
[PUBMED]  [Full text]  


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]



 

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