Actinomycetes canaliculitis complicating congenital nasolacrimal duct obstruction in an infant.

Actinomyces israelii is a Gram-positive anaerobic organism commonly associated with canaliculitis in adults. Pediatric canaliculitis is relatively rare, especially in infancy. We report the case of an 11-month-old boy who presented with co-existing canaliculitis and congenital nasolacrimal obstruction. The presenting signs included epiphora, discharge, conjunctival congestion, and matting of lashes. On examination, punctual pouting, regurgitation, and yellow canaliculiths were noted. A punctoplasty and canalicular curettage were performed along with nasolacrimal probing. Microbiological tests confirmed the organisms to be A. israelii. We discuss the clinical features and management of Actinomyces-associated canaliculitis and review the available literature on pediatric canaliculitis.

Actinomyces israelii is a Gram-positive anaerobic bacterium that is difficult to isolate and identify. It typically causes infections of hollow spaces such as the canaliculi, with the formation of canaliculiths and is associated with a chronic, purulent, granulomatous infection with the presence of yellowish sulfur granules.[1] The mainstay of treatment in canaliculitis is punctoplasty and curettage with a low incidence of postsurgical epiphora.[1234] Preexisting nasolacrimal duct (NLD) obstruction with canaliculitis is difficult to diagnose, although it has been observed in few adult cases following successful curettage.[2] There have been reports of pediatric canaliculitis in literature, but to the best of our knowledge, co-existent congenital nasolacrimal duct obstruction (CNLDO) with canaliculitis has not been previously described in literature.

Case Report

An 11-month-old male child presented with symptoms of watering and discharge in the left eye since birth. The complaints had persisted even after lacrimal sac compression. Redness and discharge in left eye had increased for 3 weeks. In the clinic, the child did not cooperate for an examination with a hand-held slit lamp. A fluorescein dye disappearance test was performed which showed delayed clearance in the left eye. With a diagnosis of the left-sided CNLDO, an examination under anesthesia with irrigation and probing was scheduled.

On examination, the right eye was unremarkable with a patent lacrimal system. The left eye conjunctiva showed some congestion, the cornea was clear and anterior segment, and fundus examinations were normal. Copious discharge was noted over left upper punctum [Fig. 1a]. Using two cotton tip applicators, the canaliculus was squeezed – from the distal part, gradually upward toward the punctum and multiple small yellowish granules were expressed out [Fig. 1b and c]. A vertical incision was made through the posterior wall of the punctum and vertical canaliculus followed by a horizontal incision along a portion of the horizontal canaliculus. All the discharge and granular material were curetted out. The lower punctum was normal, and irrigation through lower canaliculus showed regurgitation through upper punctum suggesting co-existent CNDLO. A hard stop was felt during the irrigation further confirming the location of the obstruction. On probing through lower punctum, a membranous obstruction was encountered at lower nasolacrimal duct opening, which was then overcome. Nasal endoscopy confirmed the presence of probe through NLD opening. Subsequent irrigation was patent. Microbiology of the expressed material showed delicate, branched, Gram-positive filaments irregularly arranged in a background of amorphous material suggestive of Actinomyces species [Fig. 2]. The species was subsequently identified as A. israelii on the anaerobic blood agar plates. The child was administered fortified cefazolin eye drops for 2 weeks. At 8-month follow-up, the child was asymptomatic.

Figure 1

The upper punctum could not be clearly visualized owing to overlying discharge (black arrow in Figure 1a). On clearing the discharge, a yellowish canaliculolith (black arrow in Figure 1b) was seen plugging the pouting punctal opening. Using two cotton tip applicators, the canaliculus was squeezed, and multiple yellow, small, firm bits of granular material were expressed out (black arrow in Figure 1c)

Figure 2

Photomicrograph showed irregularly arranged, delicate, branched, gram-positive filaments of Actinomyces israelii (Gram's stain, ×100)

Discussion

Symptoms of canaliculitis typically include epiphora, chronic conjunctivitis, swelling over the medial canthus, a “pouted” or everted punctum, and purulent discharge.[2] The presence of “yellow sulfur granules” at the punctum is a pathognomonic feature of Actinomyces canaliculitis. Pediatric canaliculitis is uncommon, and a review of the available English literature showed only 11 previous cases, the findings of which are tabulated in Table 1.[1345678910]

Table 1

A summary of previously reported cases of pediatric canaliculitis

The different treatment options for canaliculitis described in the literature include curettage with punctoplasty (one snip to enlarge the punctum) and canaliculotomy (enlarging the punctual incision along the canaliculus).[112] However, scarring and dysfunction of the lacrimal pump can occur following canaliculotomy; therefore, some authors have also tried canaliculoplasty (narrowing the dilated canaliculus using a 6–0 polyglactin suture) along with lacrimal intubation using Crawford stents.[8] Our technique of management described in this communication is similar to the one described by Perumal and Meyer. Which consisted of a 2-mm vertical canaliculotomy with sharp-tipped scissors followed by retrograde expression of the canalicular contents by compressing the canaliculus medial to lateral with 2 cotton-tipped applicators.[13] Conservative treatment in the form of punctal dilatation, canalicular expression, and topical antibiotics has also been tried for treating canaliculitis.[11] However, conservative treatment alone results in incomplete resolution often necessitating additional procedures such as punctoplasty with canalicular curettage.[11] In principle, enlarging the punctum and a thorough curettage of all concretions followed by comprehensive antibiotic coverage based on the sensitivity of the cultured organism is essential to treat canaliculitis.

Actinomyces are normal commensal bacteria in humans and primarily cause opportunistic infections during immunosuppressive state or when loss of continuity of epithelial lining in mucosa occurs.[31214] In our case, there seems to be no certain predisposing factor for Actinomyces infection to occur. In our case, it is difficult to establish conclusively if the canalicular infection had any role to play in the development of nasolacrimal duct obstruction (NLDO). It has been postulated that the presence of bacteria in the lacrimal system could initiate an inflammatory response and result in fibrosis and subsequently cause NLDO.[15] However, the classical history of epiphora soon after birth, the typical membranous obstruction felt during probing which could be easily overcome and visualized endoscopically; and the uneventful recovery after probing suggests that in our case, the NLDO was congenital in origin and the co-existence of canaliculitis was largely fortuitous.

Kaliki et al. reported that in canaliculitis, the mean duration of symptoms in until diagnosis is 10 months (range = 1 month–5 years) in their cohort, which predominantly included adults.[11] In addition, while this series had pediatric cases from age 8 upward, details of individual cases were not provided. However, in children, literature suggests that the mean duration of symptoms until a diagnosis is significantly higher at 27.7 months (range = 2 months–7 years) [Table 1]. Furthermore, among the reported cases, the most common condition that the patients were diagnosed as having before definitive diagnosis and treatment for canaliculitis was conjunctivitis (4/6; 66.7%).

Conclusion

Canaliculitis in children is uncommon, and literature suggests that in children, NLDO and chalazion are common conditions that canaliculitis masquerades as. Typically, epiphora, excessive discharge, and conjunctivitis along with eyelid swelling that does not respond to conventional treatment should raise the clinical suspicion of canaliculitis.

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Conflicts of interest

There are no conflicts of interest.