Abstract
First branchial cleft anomalies are uncommon, and only sporadic case reports are published in the literature. They account for 1% to 8% of all the branchial abnormalities. The often variable presentation and tract siting of first arch fistulae have led to misdiagnosis. The misdiagnosis results in inappropriate/ineffective treatment and recurrence of the sinus tract. We present a 19-year-old woman who presented to the ENT outpatient department with episodic discharge from a long-standing fistula anterior to the left sternomastoid muscle. This was associated with repeated episodes of ipsilateral tonsillitis. In relation to the history and because of the position of the fistula, a diagnosis of second branchial arch fistula was made. An attempt at excision was unfortunately followed by early recurrence of discharge. At review following the procedure, a defect of the left tympanic membrane in the form of a fibrous band was noted, and a revised diagnosis of first branchial arch sinus was made. Wide surgical excision of the tract with partial parotidectomy was performed. An uneventful postoperative course followed, with no recurrence of symptoms after 24 months of review. We discuss the case, the diagnostic pathway, and the wide local excision technique used for removal of branchial fistulae.
1
Introduction
The branchial system plays a significant role in the embryologic development of the many internal and external human body structures. With development, the planes separating arches obliterate leaving seamless junctions. Failure of complete obliteration of these junctions and the interposition of rest cells within the tissues is considered to be the likely cause for the branchial system anomalies . The chance of malformations occurring in and around the ear, including the parotid gland, is possible due to the developmental nature of the cleft, that is, from ventral to dorsal and the interaction with the developing hillocks of Hiss.
First branchial arch fistulae are the rarest of the recognized branchial fistulae occurring in between 1% and 8% of all types . Second arch is the most common, with a tract classically seen with an external opening at the junction of the lower third of the anterior border of sternomastoid muscle.
Complete excision is the only treatment for fistulae , and any residual tract has a chance of recurrence varying from 3% in primary cases to 20% in revision cases .
Because of the recurrence rate, many surgical techniques are described to excise sinuses and fistulas in head and neck site . This includes the sinus/fistula recurrence after Sistrunk procedure for thyroglossal tract sinuses and for preauricular sinuses. We describe a wide local excision technique to reduce recurrence in first branchial fistula. This technique has been used successfully in managing recurrent thyroglossal cyst sinuses after a Sistrunk procedure.
2
Case report
A 19-year-old woman patient presented to the ENT outpatient department with discharging pit on the left side of her neck. She was extremely concerned about this because she planned to go to college to study performing arts, and this unsightly lesion would reduce her chances of progress in the chosen field. Associated with this was a history of repeated episodes of tonsillitis, which she described as being worse on the left side. Initially, she sought help from her general practitioner who treated the problem with oral antibiotics, which reduced the discharge for a few weeks before recurrence.
On examination, the patient had a discharging pit lateral to the midline on the left side at the level of the thyroid cartilage. Full ENT examination was undertaken, and no other abnormality could be detected. In view of a classical history and examination, a diagnosis of second branchial fistula was made. The episodes of left tonsillitis were thought to concur with second arch fistula, as the internal opening would be within the tonsil fossa.
The patient underwent tonsillectomy and excision of the left branchial sinus tract under general anesthesia. Although the operation was uneventful, the tract was followed to a blind end approximately 5 mm from the ipsilateral tonsil but was not found to communicate with the tonsillar fossa. The tonsil was also removed, and the pillars were sutured together. The patient was discharged home the following day.
In the prevailing weeks, the patient had further episodes of neck swelling ( Fig. 1 ) and discharge. At review, incomplete excision was considered and a further ENT examination revealed a defect in the left tympanic membrane described as a bar extending from the floor of the ear canal to the tympanic membrane ( Fig. 2 ). A methylene blue fistula test was undertaken, which revealed communication with this defect and the neck pit. A second diagnosis of first arch fistula was made; this was confirmed with magnetic resonance imaging fistulography. Wide local excision of the fistulous tract with cuff of healthy tissue was undertaken. The postoperative period was uneventful.
Stay updated, free articles. Join our Telegram channel
Full access? Get Clinical Tree
