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 Table of Contents  
ORIGINAL ARTICLE
Year : 2020  |  Volume : 8  |  Issue : 2  |  Page : 96-101

A Clinical Study of Second Branchial Cleft Anomalies: Our Experience in a Teaching Hospital in Northern India


1 Department of ENT, FH Medical College, Agra, Uttar Pradesh, India
2 Department of Surgery, FH Medical College, Agra, Uttar Pradesh, India
3 Department of Radiology, FH Medical College, Agra, Uttar Pradesh, India

Date of Submission12-Jun-2020
Date of Decision30-Jun-2020
Date of Acceptance11-Jul-2020
Date of Web Publication8-Dec-2020

Correspondence Address:
Ram Avtar
Department of Surgery, FH Medical College, Etmadpur, Agra - 283 202, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jhnps.jhnps_28_20

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  Abstract 


Background: Branchial cleft anomalies are of five types. Among these, second branchial cleft anomalies are the most common developmental disorders. The aim of the study was to evaluate the clinical data in various subsets among the patients who had been diagnosed and treated for second branchial anomalies in the past 2 years. Materials and Methods: The present study was a retrospective study conducted from January 2018 to January 2020 in our institute. We collected the clinical data from patient’s medical records, radiological reports, surgical files, and histopathological reports. Results: A total of 22 patients presented with second branchial cleft anomalies. Among these, 18 (81.8%) were branchial cysts and 4 (18.2%) were branchial sinuses. There was no branchial fistula. The highest number of cases belonged to the second decade (72.7%). The mean age was found to be 16.8 years. The male-to-female ratio was 1.75:1. The rural-to-urban ratio was 3.4:1. There was no proclivity to any one side of the neck. All 22 patients underwent surgical excision as the treatment. There was no recurrence. Conclusion: We concluded that branchial cysts are the main type among second branchial cleft anomalies. Most patients belonged to the second decade of life. Early detection and reaching correct diagnosis are the major factors influencing treatment outcome. Preferably, surgical excision has to be performed in noninfected neck. Meticulous dissection helps in avoiding recurrence. Genetic disorders such as branchio-oto-renal syndrome have to be kept in mind in the evaluation of any branchial cleft anomalies.

Keywords: Anomalies, branchial cyst, branchial sinus, excision, fistula, second branchial cleft


How to cite this article:
Anoop M, Avtar R, Akhtar W, Yadav Y. A Clinical Study of Second Branchial Cleft Anomalies: Our Experience in a Teaching Hospital in Northern India. J Head Neck Physicians Surg 2020;8:96-101

How to cite this URL:
Anoop M, Avtar R, Akhtar W, Yadav Y. A Clinical Study of Second Branchial Cleft Anomalies: Our Experience in a Teaching Hospital in Northern India. J Head Neck Physicians Surg [serial online] 2020 [cited 2021 Jan 18];8:96-101. Available from: https://www.jhnps.org/text.asp?2020/8/2/96/302624




  Introduction Top


In embryonic period, the branchial arches and the mesenchyma are easily recognizable by the 4th week of embryonic life. The branchial arches are derived from the neural crest cells. The mesenchyma developed from the lateral mesoderm.

Ectodermal clefts and endodermal pouches which are five pairs each, separate the six branchial arches by a closing membrane, which is located at the junction between pouches and clefts[1],[2] [Line Diagram 1] and [Line Diagram 2].



Ascherson coined the term “branchial cyst” in 1832. He found that branchial cysts results due to impaired obliteration of branchial clefts.[3]

The word “Branchia” is a Greek word. Its meaning is gill. The word denotes the anomalies owing to their resemblance to gills of certain species of fish.

Branchial arches contain connective tissue that forms cartilage, muscle, bone, and blood vessels. Persistent or incomplete embryonic development of arches leads to several defects in the neck region.

Different types of neck masses present to the ear, nose, and throat (ENT) surgeon in their clinical practice. Congenital cervical anomalies have to be given importance in the differential diagnosis of head and neck masses in children and adults.[4]

In pediatric population, 20% of cervical masses are attributed to branchial cleft cysts and fistulae.[4] In 1% of the cases, they are bilateral, without laterality to one side (left or right) in which they may develop.[5]

The branchial cysts were classified into four types. Ninety-five percent of branchial anomalies were attributed to the second branchial cleft.[6],[7],[8],[9] Mostly these cysts were noticed along the anterior border of upper third of the sternocleidomastoid muscle, close to the muscle [Figure 1]. However, in some cases, these cysts might present anywhere along the course of a second branchial fistula, which starts from the skin of the lateral neck, between the internal and external carotid arteries and ends in to the palatine tonsil[10] [Table 1]. Therefore, second branchial cyst had to be kept as the differential diagnosis of para-pharyngeal masses.
Figure 1: Clinical photograph of right side branchial cyst

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Table 1: Openings and course of different types of branchial fistulae

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Our teaching institute is situated in a rural background. Most of the patients belonged to low socioeconomic status. In the last couple of years, the number of patients presented with neck swelling was found to be increasing in our outpatient department. We noticed that most of the neck swellings were of branchial cleft origin. Hence, we took up our present clinical study to analyze its various patterns in our regional population and compare it with the current literature available.

The main objectives of our present clinical study were as follows:

  1. To analyze the various clinical types of second branchial cleft anomalies in our institute
  2. To know which type of anomalies were most common among them
  3. To analyze the age distribution and sex pattern
  4. To observe regional variation among population
  5. To know any proclivity to any one side of neck
  6. To analyze treatment outcome in terms of recurrence.



  Materials and Methods Top


We took up this clinical study on second branchial cleft anomalies in our institute. The study period was of 2 years, i.e., between January 2018 and January 2020.

The present study was a retrospective one in which we included 22 patients, who attended the ENT department of our hospital. The study analyzed the patient’s clinical and surgical files, radiological and histopathological examinations.

All patients were having developmental anomalies pertaining to second branchial arch, i.e., branchial cysts and branchial sinuses (in our study period of 2 years no patients with branchial fistula were reported). Patients presenting with first or third branchial cleft anomalies were excluded from the study. Those patients who underwent previous neck surgery were also excluded. Each patient was clinically assessed by detailed history and local examination. Relevant investigations including radiological one were conducted before taking up surgical procedure. Each patient underwent ultrasonography (GE Voluson S8) as primary investigation followed by computed tomography (CT) (Toshiba, 16 Slice, contrast as well as noncontrast study) as definitive cross-sectional imaging investigation. They were analyzed by the same radiologist. Every patient’s histopathological reports were analyzed, and patients were regularly followed up for 6 months to 1 year. The statistical analysis of our clinical data was done using the SPSS software version 16.0 (IBM corporation, Chicago, IL, USA) to find out mean, median, mode, standard deviation, coefficient of variation, mean deviation, and P value (P < 0.05 was considered to be statistically significant). The statistical findings are summarized along with the results.


  Results Top


The results of the present clinical study are briefly summarized below:

Branchial cysts were the most common type of second branchial cleft anomalies [Table 2]. It accounted for 81.8% of second brachial cleft anomalies, whereas branchial sinus accounted for 18.2% of total cases. None of the patient presented with branchial fistula. The branchial cyst: Branchial sinus: Branchial fistula ratio was 4.5:1:0.
Table 2: Types of second branchial cleft anomalies

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The most common age group was 11–20 years [Table 3]. The mean age was 16.8 years. The median was 16.1 years and mode was 15.6 years. The standard deviation was 5.13. The coefficient of variation and mean deviation were 30.53 and 0.67, respectively. The P value was found to be 0.0006 (<0.05) and is statistically significant.
Table 3: Age distribution

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Mild male preponderance was noticed. Males consisted of 63.6% of the total cases, and rest were females [Table 4]. The male-to-female ratio was 1.75:1. Rural population contributed majority of patients. Rural patients consisted of 77.3% and urban as 22.7% of the total cases [Table 5]. Rural-to-urban ratio was 3.4:1. Any proclivity to any one side of the neck was not observed. All patients underwent surgical excision as treatment. There was no recurrence observed among any of our patients.
Table 4: Sex distribution

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Table 5: Regional distribution

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  Discussion Top


The earliest documentation of branchial apparatus has been attributed to Von Baer in 1827. Cervicoaural or collaural fistula was identified for the first time by Sir James Paget in 1878. Rathke, Ascherson, and Virshow are the other prominent names who had given immense contribution in the study of branchial cleft and its anomalies.

Among all the cystic swellings in the neck region, branchial cyst presents commonly in the second decade of life, but can occur in any age with equal frequency in both sexes. It presents as a swelling in the upper part of the neck anterior to sternocleidomastoid muscle.

Mainly four theories are there regarding the origin of branchial cleft anomalies. They are branchial theory, precervical theory, thymo-pharyngeal duct origin theory, and theory of cystic lymph node degeneration.[11]

In view of Ascherson, incomplete obliteration of branchial cleft mucosa which remains dormant is stimulated to grow in the later part of life resulting in cyst formation. Wenglowski was of the view that cystic degeneration of cervical lymph nodes was the main mechanism of origin of lateral cervical cyst. Bhaskar and Bernier came out with the theory of cystic alteration of cervical lymph node which is stimulated by entrapped epithelium.

At present, to clarify the exact origin, immunohistochemistry is used at the molecular level. The nature of epithelial cells and their relationship to other cell types elsewhere in the body are studied.[12]

Clinically, the swelling is smooth, oval, fluctuant, nontender, and nontransilluminant. At times, painful increase in the size of swelling can occur during upper respiratory tract infections.

Among all the branchial arch anomalies, second branchial arch anomalies are the most common in clinical practice. Branchial sinus and fistula are rare compared to branchial cyst. Second branchial sinus and fistula are more common than third branchial sinus and fistula. A second arch branchial sinus has as an external opening at the junction of lower and middle of the anterior border of sternocleidomastoid and it may exude mucoid discharge. When there is an internal opening, it opens in the tonsillar fossa. When both internal and external opening are present, it is named as branchial fistula.[13]

Typical course is noted in the second branchial fistula. This knowledge of the tract is useful for the operating surgeon for total surgical extirpation of the tract[13] [Table 1]. The branchial fistula tract ascends up just deep to deep cervical fascia along the carotid artery. Then, it passes deep to second arch structures but superficial to third arch structures. It passes deep to external carotid artery, stylohyoid, and posterior belly of digastric which are second arch structures. It passes superficial to internal carotid artery which is third arch structure, and also it is superficial to hypoglossal nerve. Finally, it pierces pharyngeal wall and ends in tonsillar fossa. Complete excision of the tract can be accomplished by step-ladder pattern incisions.

Third branchial cleft sinus and fistula are uncommon. Its external opening is at the same place as second cleft sinus, but internal opening is situated in pyriform sinus. The third arch fistulous tract passes behind both internal and external carotid vessels but is superficial to vagus and hypoglossal nerves.[13]

About 75% of all second branchial cleft anomalies are cysts,[14] and it presents when an individual is between 10 and 40 years old. Second branchial cleft sinuses and fistulas are less common and usually present during the first decade of life.[15],[16] No gender predilection has been noticed.[17]

In our study, nearly 81.8% of the patients had branchial cysts, and the rest 18.2% had branchial sinuses (there was no case of branchial fistula). These finding were consistent with the specialty literature data that show a greater incidence of branchial cysts than that of branchial sinuses and fistulae. In the present study, 72.7% of patients belonged to the second decade. Specialty literature data show a higher incidence of branchial cysts in adult patients (decade of two and three).

There was mild male preponderance in our study, with 63.6% patients were males and 36.4% were females. There was no predisposition of a certain side of the neck (right or left). The findings of our study are shown in [Table 2],[Table 3],[Table 4],[Table 5] and in [Diagram 1],[Diagram 2],[Diagram 3],[Diagram 4].



Second branchial cleft anomalies are usually small but can become large to cause difficulty in swallowing and breathing. Other symptoms such as hoarseness and cranial nerve palsies are extremely rare.[18],[19]

In ultrasonography, a second branchial cleft cyst seen as a sharply marginated, ovoid to round, centrally anechoic mass with a thin peripheral wall that displaces the surrounding soft tissues. The mass is compressible and shows distinct acoustic enhancement.

In CT scan, these cysts are typically well circumscribed, homogeneously hypo-attenuated masses surrounded by a uniformly thin wall.[19] The mural thickness increases after infection. The cyst typically displaces the sternocleidomastoid muscle posteriorly or posterolaterally, pushing the vessels of the carotid space medially or posteromedially, and displaces the submandibular gland anteriorly.[20] It may also be seen more medially within the parapharyngeal space after extending through the stylomandibular tunnel and middle constrictor muscle[21] [Figure 2].
Figure 2: Computed tomography scan of right side branchial cyst

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Magnetic resonance imaging (MRI) better depicts the deep tissue extent of second branchial cleft cysts, which is useful in preoperative planning. The cyst fluid varies from hypointense to slightly hyperintense relative to muscle on T1-weighted images and is usually hyperintense on T2-weighted images.[22]

Histopathological characteristics of branchial cysts are usually filled with a turbid, yellowish fluid that may contain cholesterol crystals. Their walls are thin and lined with stratified squamous epithelium overlying lymphoid tissue.[23] Columnar respiratory epithelium occasionally present.

The histopathological examinations of the operated specimens of second branchial cleft anomalies have accurately determined the diagnosis, specifically showing the presence of stratified squamous epithelium.

The management of second branchial cleft anomalies is surgical excision[24] [Figure 3]. In ideal settings, surgery should be performed on noninfected neck. All the patients in our study (22 patients) underwent surgery as treatment, and there was the absence of recurrence observed within 6 months to 1-year follow-up period. The operative procedures were namely cervicotomies with branchial cyst/sinus excision.
Figure 3: Peroperative photograph of branchial cyst excision

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  Conclusion Top


Branchial cysts are missed and forgotten at times, in the differential diagnosis of neck swellings. Branchial cysts should be suspected as a differential diagnosis in any patient presenting with swelling in the lateral part of neck, painful, or painless.

Second branchial cleft anomalies are presented to clinicians most often in the first and second decades. There is no proclivity of the position to one side of the neck (right or left). There is mild male preponderance. Ultrasonography followed by CT scan and MRI scan is the reliable radiological investigations. Histopathological examination confirms the correct diagnosis. The definite treatment for branchial anomalies is a complete surgical excision in a noninfected neck. General anesthesia is preferred because of complexity of the dissection. Meticulous dissection during the procedure will avoid the recurrence of the branchial anomalies. Genetic disorders such as Branchio-oto-renal syndrome have to be kept in mind in the evaluation of any branchial cleft anomalies.

Acknowledgments

We authors express our gratitude to the chairman and the management of F.H. Medical College Etmadpur Agra for allowing us to conduct this clinical study.

We sincerely thank our professor and Head of Department of ENT F. H. Medical College Etmadpur Agra, Dr. Gaurav Khandelwal and Dr. Anuj Goyal, professor of ENT department F.H. Medical college Etmadpur, Agra, for constant guidance given during this clinical study.

We authors thank Mr. Dinesh Kumar and Miss Sapna Samanta for their help in typing and other computer works for our article.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

Disclosure

This material has never been published and is not currently under evaluation in any other peer reviewed publication.

Ethical approval

The permission was taken from Institutional Ethics Committee prior to starting the project. All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.

Informed consent

Informed consent was obtained from all individual participants included in the study.



 
  References Top

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2.
Subhadra Devi V. Pharyngeal arches. In: Inderbir Singh’s Text Book of Human Embryology. 11th ed.. New Delhi: Jaypee; 2018. p. 126-36.  Back to cited text no. 2
    
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Langmann J. Medical Embryology. 3rd ed. Baltimore: Willams & Wilkins; 1975.  Back to cited text no. 3
    
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Acierno SP, Waldhausen JH. Congenital cervical cysts, sinuses and fistulae. Otolaryngol Clin North Am 2007;40:161-76.  Back to cited text no. 4
    
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Doshi J, Anari S. Branchial cyst side predilection: Fact or fiction? Ann Otol Rhinol Laryngol 2007;116:112-4.  Back to cited text no. 5
    
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Waldhausen JH. Branchial cleft and arch anomalies in children. Semin Pediatr Surg 2006;15:64-9.  Back to cited text no. 6
    
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Koeller KK, Alamo L, Adair CF, Smirniotopoulos JG. Congenital cystic masses of the neck: Radiologic-pathologic correlation. Radiographics 1999;19:121-46.  Back to cited text no. 7
    
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Vogl T. Hypopharynx, larynx, thyroid, and parathyroid In: Stark D, Bradley W, editors. Magnetic Resonance Imaging. 2nd ed. New York: Mosby-Yearbook, St Louis; 1992. p. 1184-243.  Back to cited text no. 8
    
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Ang AH, Pang KP, Tan LK. Complete branchial fistula. Case report and review of the literature. Ann Otol Rhinol Laryngol 2001;110:1077-9.  Back to cited text no. 9
    
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Zadvinskis DP, Benson MT, Som PM, Smoker WR. Embryology and congenital cystic lesions. In: Som PM, Curtin HD, editors. Head and Neck Imaging. London: Mosby, St Louis; 1996. p. 754.  Back to cited text no. 10
    
11.
Glosser JW, Pires CA, Feinberg SE. Branchial cleft or cervical lymphoepithelial cysts: Etiology and management. J Am Dent Assoc 2003;134:81-6.  Back to cited text no. 11
    
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Little JW, Rickles NH. The histogenesis of the branchial cyst. Am J Pathol 1967;50:533-47.  Back to cited text no. 12
    
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Dhingra PL, Dhingra S, Neck Masses; Text Book of Diseases of Ear, Nose and Throat and Head and Neck Surgery. 6th ed.. New Delhi: Elsevier; 2014. p. 390-94.  Back to cited text no. 13
    
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Mandell DL. Head and neck anomalies related to the branchial apparatus. Otolaryngol Clin North Am 2000;33:1309-32.  Back to cited text no. 14
    
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Som PM, Sacher M, Lanzieri CF, Solodink P, Cochen BA, Reede DL, et al. Parenchymal cysts of the lower neck. Radiology 1985;157:399-406.  Back to cited text no. 15
    
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Michael AS, Mafee MF, Valvassori GE, Tan WS. Dynamic computed tomography of the head and neck: differential diagnostic value. Radiology 1985;154:413-9.  Back to cited text no. 16
    
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Faerber EN, Swartz JD. Imaging of neck masses in infants and children. Crit Rev Diagn Imaging 1991;31:283-314.  Back to cited text no. 17
    
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Shin JH, Lee HK, Kim SY, Park HW, Khang SK, Choi CG, Suh DC. Parapharyngeal second branchial cyst manifesting as cranial nerve palsies: MR findings. AJNR Am Neuroradiol 2001;22:510-2.  Back to cited text no. 18
    
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Durrant TJ, Sevick RJ, Lauryssen C, MacRae ME. Parapharyngeal branchial cleft cyst presenting with cranial nerve palsies. Can Assoc Radiol J 1994;45:134-6.  Back to cited text no. 19
    
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Harnsberger H. Handbook of Head and Neck Imaging. 2nd ed. London: Mosby-Yearbook, St.Louis; 1995.  Back to cited text no. 20
    
21.
Papay FA, Kalucis C, Eliachar I, Tucker HM. Nasopharyngeal presentation of second branchial cleft cyst. Otolaryngol Head Neck Surg 1994;110:232-4.  Back to cited text no. 21
    
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McClure MJ, McKinstry CS, Stewart R, Madden M. Late presentation of branchial cyst. Ulster Med J 1998;67:129-31.  Back to cited text no. 22
    
23.
Agaton-Bonilla FC, Gay-Escoda C. Diagnosis and treatment of branchial cleft cysts and fistulae. A retrospective study of 183 patients. Int J Oral Maxillof Surg. 1996;25:449-52.  Back to cited text no. 23
    
24.
Choi SS, Zalzal GH. Branchial anomalies: A review of 52 cases. Laryngoscope 1995;105 (9 Pt 1):909-13.  Back to cited text no. 24
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]
 
 
    Tables

  [Table 1], [Table 2], [Table 3], [Table 4], [Table 5]



 

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