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An extremely rare case of adult with first branchial cleft fistula: Case report.

ABSTRACT:

Introduction

Anomalies of the first branchial cleft are rare and represent 10% of lateral cervical malformations. Very few cases have been described in literature. In our research, we didn't find cases of adults with first branchial cleft fistulas. First branchial cleft fistulas in adults are difficult to diagnosticate, and the surgery of the parotid gland to extract the fistula requires to be precautious.

Case presentation

The aim of this work is to present an extremely rare case of a 65 years-old patient with first branchial cleft fistula. Fistula excision surgery completed with superficial parotidectomy were performed. No sign of recurrency were found after 6 months surveillance.

Discussion

First branchial cleft fistula is hard to diagnosticate, especially with adults. Very few cases of adult male are described in the literature. It is often misdiagnosed with parotid tumours or as otitis with ear drainage.The treatment of First branchial cleft fistula is surgical. A complete resection of the tract is necessary to prevent recurrency. It usually requires a superficial parotidectomy and the fistula tract passes under the parotid gland.

Conclusion

First branchial cleft fistulas are rare, and forms with adults are extremely rare to encounter. Imagery can't always help to diagnosis. Surgery is indicated in the management of this pathology, with preservation of the facial nerve.

SUBMITTER: Lendoye W

PROVIDER: S-EPMC8463818 | biostudies-literature |

REPOSITORIES: biostudies-literature

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Project description:ObjectivesTo describe the prevalence and significance of first branchial cleft anomalies in children with congenital aural atresia.MethodsRetrospective cohort study and case series. Patients were included if they had ICD-10 code Q16.0, Q16.1, Q16.9, Q17.2, or Q17.9 in their medical record and were seen at UCSF Benioff Children's Hospital from 2012 to 2020 for aural atresia. Children were categorized as having aural atresia and first branchial cleft anomalies if the presence of a first branchial cleft anomaly was noted in otolaryngology provider notes; otherwise, they were categorized as aural atresia alone. Patients with aural atresia and first branchial cleft anomalies were included in the case series.ResultsAmong 125 children with congenital aural atresia, 5 (4%) were identified with first branchial cleft anomalies. In all cases, an epithelialized tract was noted to originate from the inferior aspect of the middle ear cleft, exiting the temporal bone in an inferomedial position adjacent to the stylomastoid foramen, with a cutaneous exit point inferior to the expected location of the native ear canal. There was no association with sex, microtia grade, or laterality of atresia; however, children with aural atresia and first branchial cleft anomalies were significantly more likely to have syndromes such as Goldenhar and Treacher Collins (p = .04) than those with aural atresia alone.3/5 (60%) children with aural atresia and first branchial cleft anomalies presented with cholesteatoma compared with 1/120 (0.8%) children with aural atresia alone, a significant difference (p < .001). All four children over the age of two have undergone surgical management. In two of these, excision of the branchial cleft anomaly could be combined with atresiaplasty, with normal hearing results in both cases.ConclusionsAural atresia can be associated with comorbid anomalies of the head and neck. First branchial cleft anomalies can be suspected based on characteristic clinical appearance and confirmed with computed tomography showing a typical course through the temporal bone. When present in the context of congenital aural atresia, first branchial cleft anomalies are associated with a significantly increased risk for cholesteatoma, often necessitating surgical management with favorable hearing outcomes.Level of evidence4.