Jul 6, 2014 - can cause partial or total obstruction of the lacrimal drainage system whereas the ... remain asymptomatic with patent nasolacrimal ducts [5].
Congenital lacrimal fistula: a case report Zeynep Duru1 , Mualla Hamurcu1 , Süleyman Boynueğri2 , Necati Duru3 (necatiduru at gmail dot com) #, Murat Sinan Sarıcaoğlu1 , Ayten Kocaman Bulut1 , Ahmet Karakurt1 1 Ankara Numune Education and Research Hospital, Department of Ophthalmology, Ankara, Turkey. 2 Ankara Numune Education and Research Hospital, Department of Ear, Nose, and Throat, Ankara, Turkey. 3 Ankara Atatürk Education and Research Hospital, Department of Ophthalmology, Ankara, Turkey # : corresponding author DOI http://dx.doi.org/10.13070/rs.en.1.939 Date 20140706 Cite as Research 2014;1:939 License CCBY
Abstract
Congenital lacrimal fistula, with an abnormal connection from the skin to the lacrimal passage, is a
rare developmental anomaly of the nasolacrimal excretory system. In this article, we aimed to report a case who presented with congenital lacrimal fistula. A 15yearold female admitted to our clinic with lacrimal fistula located inferonasal to the medial canthal angle on the right eye. When lacrimal irrigation through the lower punctum was performed, fluid passed into the nose and the lacrimal passage was found to be patent. However, there was no fluid regurgitation from the ostium of lacrimal fistula. The fluorescein dye disappearance test was normal. In the nasal endoscopy, there was no defect in the nasal passage. In the absence of significant symptoms, we did not feel the necessity of any treatment for this lacrimal fistula.
Introduction Morphogenesis of the lacrimal system begins at sixth week of gestation. If development is affected during this time, many congenital anomalies of the lacrimal system may occur. The most common anomalies are punctal atresia, canalicular atresia, dacryostenosis, sac diverticula, and congenital fistula [1] [2] [3]. Congenital lacrimal fistula is a rare developmental abnormality of the lacrimal system with an estimated incidence of one in 2,000 births [4]. There is no apparent pattern related to sex or race. In congenital lacrimal fistula, an epitheliumlined tract connecting the skin to the lacrimal sac, common canaliculus, or nasolacrimal duct is present from birth. Most of the fistulas are unilateral and typically located inferonasal to the medial canthal angle [5] [6] [7]. This anomaly can cause partial or total obstruction of the lacrimal drainage system whereas the majority of these anomalies remain asymptomatic with patent nasolacrimal ducts [5]. In this article, we aimed to report a case who was diagnosed congenital lacrimal fistula with characteristic findings. Case report
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A 15yearold female presented to our clinic with an opening in the lower medial canthal angle of her right eye. She had no complaints and was in good health. There was no past history of systemic disease, eyelid surgery, trauma, or any relevant family history. Slit lamp examination of the anterior and posterior segment were unremarkable. Ophthalmic examination revealed uncorrected visual acuity of 1.0 in both eyes. External inspection showed an ostium 7 mm from medial canthal angle on the right (Figure 1). There were no Figure 1. Opening of right congenital lacrimal fistula. signs of infection or discharge in this area, and there were protruding bristles from the opening of the fistula (Figure 2). When lacrimal irrigation through the lower punctum was performed, fluid passed into the nose and the lacrimal passage was found to be patent. However, there was no fluid regurgitation from the ostium of lacrimal fistula. The fluorescein dye
disappearance test was normal. In the nasal endoscopy, there was no defect in the nasal passage (Figure 3). A dacryocystogram to visualize the lacrimal system was not performed at this time because of the clear characteristic findings of lacrimal fistula. As she was not significantly symptomatic, we kept our patient under observation without any treatment.
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Discussion Congenital lacrimal fistula is a rare developmental disorder of the nasolacrimal excretory system, and it was first noted by Rasor in 1675 [5]. This anomaly usually is located inferonasally to the medial canthus. Although congenital lacrimal fistula is often unilateral, bilateral cases have been reported [6]. Bilateral cases are occasionally familial, possibly transmitted as an autosomal dominant trait, and sometimes occur as part of a syndrome such as Crouzon’s disease and Goldenhar’s
syndrome [5] [6] [8] [9]. Tract of a fistula is generally lined with stratified squamous epithelium. It connects the skin to the common canaliculus and often to the lacrimal sac. Sometimes the tract may Figure 2. View of the protruding bristles.
end blindly in the subcutaneous tissue near the lacrimal sac [5] [6] [7] [9]. Most of these fistulas are asymptomatic and therefore usually remain undetected [5]. Treatment of lacrimal fistula only becomes necessary when significant epiphora or discharge is present. Treatment modalities for symptomatic fistula have been discussed in the literature. These include nasolacrimal duct probing, cauterization of the external ostium, and surgical excision with or without dacryocystorhinostomy [5] [8]. Our patient was not symptomatic as a result of this fistula. There was no dye retention in the tear meniscus and there was no reflux from the fistula on nasolacrimal sac irrigation. This is probably because of the origination of the fistula from the common canaliculus rather than than the lacrimal sac. In the present case, we kept our patient under observation without any treatment. Conclusion In conclusion, a physician has
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to keep the possibility of congenital lacrimal fistula in mind in patients presenting with lacrimal symptoms. Also, it is important to know clinical characteristics of these cases to make differential diagnosis. In addition, patients that are not symptomatic or have only minimal symptoms, like our case, should be merely kept under observation.
Figure 3. View of the fluorescein solution in the nasal passage.
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