CASE REPORT Unilateral lacrimal fistula in a patient with uterus didelphys and renal agenesis Ece Turan-Vural • Fatih Atmaca • Cihan Unlu • Gurkan Erdogan • Huseyin Bayramalar Received: 16 January 2011 / Accepted: 26 December 2011 / Published online: 19 January 2012 Ó Springer Science+Business Media B.V. 2012 Abstract A 30-year-old female patient presented to our clinic because of lacrimation from an orifice close to the left lower eyelid. Ocular examinations and analyses revealed uterus didelphys and unilateral renal agenesis associated with a left lacrimal fistula. The patient underwent fistulectomy and external dacryocystorhi- nostomy. We decided to report on this patient owing to the unusual concurrent systemic abnormalities. Keywords Lacrimal fistula Á Uterus didelphys Á Renal agenesis Case A 30-year-old female patient had an ongoing complaint of eye watering in her left eye and an additional discharge from the lower medial portion of the left eye since childhood. External ophthalmic examination revealed a fistula located medially 3 mm below the punctum. A mucoid discharge and a fluid flow from this location were observed during lacrimal lavage (Fig. 1). There was a reflux of dye from the external opening of the lacrimal fistula (Fig. 1). There was no reflux of the right eye. Bilateral lacrimal lavage showed patent passage. There was no other ophthalmic system abnor- mality. Systemic examination and analyses were per- formed. Abdominal computer tomography revealed that the patient had left renal agenesis (Fig. 2) and uterus didelphys. Renal scintigraphy showed no func- tional kidney tissue on the left side and a fully functional kidney on the right side (Fig. 2). The patient had no other systemic abnormality. Hysterosalpingo- gram confirmed the uterus didelphys (Fig. 3). Congen- ital lacrimal fistulas are routinely managed with fistulectomy and dacryocystorhinostomy. Due to excessive eye watering in the patient, we also carried out fistula excision and external dacryocystorhinos- tomy. The patient was followed-up for 6 months during which no complaint or complication was observed. Discussion Congenital lacrimal fistula is a rare abnormality that is encountered in every 1/2000 births [1]. It was first described by Rasor in 1965 which was followed by hundreds of case reports in the literature [1]. The most common concurrent abnormalities are reported to be associated with punctum and canaliculi [1, 2]. How- ever, recently there are reports on accompanying systemic development abnormalities such as preauric- ular fistulas [3], thalassemia [2, 3], Down’s syndrome E. Turan-Vural (&) Ophthalmology Clinic, Haydarpasa Numune Education and Research Hospital, Istanbul, Turkey e-mail: dreceturan76@yahoo.com F. Atmaca Á C. Unlu Á G. Erdogan Á H. Bayramalar Ophthalmology Clinic, Umraniye Education and Research Hospital, Istanbul, Turkey 123 Int Ophthalmol (2012) 32:67–69 DOI 10.1007/s10792-011-9510-4