Nonsyndromic bilateral second branchial cleft fistulae: A case report

• Main Authors: Cameron P. Worden, Kenan C. Michaels, William P. Magdycz
• Format: Article
• Language: English
• Published: Elsevier 2021-09-01
• Series: Otolaryngology Case Reports
• Online Access: http://www.sciencedirect.com/science/article/pii/S2468548821000485

Branchial cleft anomalies are rare congenital malformations that result from the abnormal persistence of branchial clefts during embryogenesis and manifest clinically as cysts, sinuses, or fistulae. In greater than 95% of cases, branchial cleft anomalies originate from remnants of the second branchial cleft. Identification of branchial cleft anomalies, particularly branchial cleft fistulae, are clinically important as these findings may be part of a larger syndromic clinical presentation such as the branchiootorenal syndrome, which necessitates further workup. Branchial cleft anomalies are bilateral in approximately one percent of cases; however, bilateral second branchial cleft fistulae are, for unknown reasons, much rarer. To the best of our knowledge, there have been less than ten cases of nonsyndromic, bilateral second branchial cleft fistulae recorded in the literature. In this report, we present the CASE of a 50-year-old woman with recent left-sided pain, drainage, and swelling in the lower one-third of her neck. The patient reported a history of bilateral “cysts” in the lower one-third of her neck for most of her adult life, which frequently become infected. She denied a personal or family history of renal anomalies or hearing loss. Computed tomography scan with intravenous contrast of the soft tissues of the neck revealed bilateral soft tissue tracts beginning in the region of the tonsillar fossae and extending bilaterally along the anterior borders of the sternocleidomastoid muscle (SCM) down to the skin surface near the level of the thyroid gland, consistent with bilateral second branchial cleft fistulae.