Branchial anomalies in children: A report of 105 surgical cases

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Abstract

Background

Branchial anomalies (BAs) account for 20% of all congenital masses in children. We sought to review the incidence of involvement of individual anomalies, diagnostic methods, surgical treatment, and complications of BAs in children. In addition, we also classified our study and analyzed a congenital lower neck cutaneous fistula near the sternoclavicular joint that was thought to be the skin-side remnant of the fourth BAs.

Methods

We conducted a retrospective analysis of 105 children who were referred to our hospital from June 2009 to December 2016 for the treatment of BAs.

Results

In this series, there were 51 males and 54 females. The age at the time of operation varied from 19 days to 13 years, and the mean age was 4.5 years. A total of 33 (31.4%) cases presented with first BAs, 13 (12.4%) presented with second BAs, and 59 (56.2%) presented with third and fourth BAs, including 6 cases of congenital lower neck cutaneous fistula. Fistulectomy under general anesthesia was performed on all of them. For postoperative complications, 2 cases had temporary facial paralysis, 1 case had permanent facial paralysis, 4 cases had temporary recurrent laryngeal nerve injury. Recurrence occurred in 2 patients with first BAs after medium follow-up time of 3.6 years (6 months–8 years).

Conclusions

BAs are common congenital head and neck lesions in children, and there are four distinct types (first, second, third and fourth anomalies). The incidence of third and fourth BAs in Asia maybe higher when compared with literature reports, second BAs seem rare in this population, but more research is needed to confirm this perspective. Diagnosis is not difficult with a proper knowledge of the anatomy of the BAs. The surgical procedures should be tailored depending on the various types, and complete excision of the fistula is the key to prevent recurrence.

Introduction

Branchial anomalies (BAs) account for 20% of all pediatric congenital masses and are the second-most common mass after thyroglossal duct cysts and sinuses [1]. The incidence rates in males and females are the same. BAs encompasses first, second, third and fourth branchial cleft cysts, fistulas, and sinuses. Although some theories about the origin of BAs have been reported (thymopharyngeal duct theory, parotid gland inclusion theory, degenerative cystic changes of cervical lymph nodes theory) [2]. The most widely accepted theory is the incomplete involution of the branchial apparatus during embryogenesis.

Branchial structures begin to develop early in the fourth week of the embryonic stage. By the end of the fourth week, there are four unequivocal pairs of branchial arches and two extra rudimentary arches that are invisible on the surface of the embryo, and the mesoderm of the arches is separated by endoderm-lined pouches internally and ectoderm-lined clefts externally. By the seventh week of embryonic life, the pouches and clefts are gradually obliterated by invasion of the surrounding mesenchyme. BAs are residuals of the four primary pairs of branchial pouches and branchial clefts that fail to regress or develop normally [3]. When a cleft or pouch fails to obliterate, it may communicate with either the mucosa of the upper airway or skin, forming a sinus. When both a cleft and pouch fail to obliterate, it may form a communication between the skin and mucosa, forming a fistula. When a cleft remnant forms an epithelial-lined space without communication to mucosa or the skin, a cyst is formed [1].

This article retrospectively analyzed 105 cases of BAs operated on at Shanghai Children's Hospital over the past 8 years. In addition, we also classified our study and analyzed a congenital lower neck cutaneous fistula near the sternoclavicular joint that was thought to be the skin-side remnant of the fourth branchial anomalies [4].

Section snippets

Patients and methods

This study was conducted at the pediatric ENT department at Shanghai Children's Hospital, China. All cases who underwent surgery for BAs from June 2009 to December 2016 were included in this study. Eligible patients included those under 18 years of age. The intraoperative course of the fistula tract and pathology results confirmed the diagnosis of BAs. Congenital lower neck cutaneous fistulas were also included. The cases of BAs who just received abscess incision and drainage and who did not

Results

The summary of patients in this series was depicted in Table 1. A total of 105 patients were surgically treated for BAs, and there were 51 males and 54 females. The age at the time of operation varied from 19 days to 13 years, and the mean age was 4.5 years. According to the classification of BAs, 33 (31.4%) presented with first BAs, 13 (12.4%) presented with second BAs, 59 (56.2%) presented with third and fourth BAs, and we included 6 cases of congenital lower neck cutaneous fistula. The BAs

Discussion

Second BAs, which account for 95%, are the most common; first BAs account for 1–4%, and third and fourth branchial anomalies are rare [5]. Y. Bajaj reported 80 cases of BAs in the United Kingdom. Second clefts account for 77.5%, first clefts account for 18.8%, and fourth clefts account for 3.7% [6]. In our study, 31.4% presented with first BAs, 12.4% presented with second BAs, and 56.2% presented with third and fourth BAs; the incidence of the third and fourth BAs was the highest, while that of

Conclusion

BAs are common congenital head and neck lesions in children, and there are four distinct types (first, second, third and fourth anomalies). The incidence of third and fourth BAs in Asia may be higher when compared with literature reports, but more research is needed to confirm this perspective. Diagnosis is not difficult with a proper knowledge of the anatomy of BAs. The surgical procedures should be tailored depending on the various types, and complete excision of the fistula is the key to

Acknowledgements

The organization funded my research.

Youth project of National Natural Science Foundation of China, No: 81500779.

References (21)

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