Bilateral hydrosalpinx in adolescent girls with Hirschsprung's disease: association of two rare conditions

OBJECTIVE: The purpose of our study was to illustrate three cases of bilateral hydrosalpinx in postpubertal girls operated on for Hirschsprung's disease and to discuss the possible cause: iatrogenic or congenital. We identified bilateral hydrosalpinx in three postpubertal sexually inactive girls with Hirschsprung's disease treated, respectively by Duhamel, Soave pull-through, and Martin procedures. No history of surgical complications or pelvic inflammation had been reported. CONCLUSION: Hirschsprung's disease is rare in girls and bilateral hydrosalpinx is also extremely uncommon in sexually inactive adolescents. We think there may be a possible common cause: either a postsurgical complication or a congenital defect of the autonomous innervation in the context of a neurocristopathy. Because of the rarity of both conditions, the association is unlikely to be coincidental. The cause of this association is unclear and further studies are required to find its prevalence and to estimate the possible impact on fertility.