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Table of Contents
ORIGINAL ARTICLE
Year : 2022  |  Volume : 24  |  Issue : 3  |  Page : 89-92

Clinical features and surgical outcomes of branchial cleft cyst at a tertiary care hospital, Saudi Arabia, a retrospective study


1 Department of Otolaryngology, King Faisal Specialist Hospital and Research Center; Department of Otolaryngology, College of Medicine, Alfaisal University; Department of Otolaryngology, King Saud University, Riyadh, Saudi Arabia
2 Department of Otolaryngology, King Faisal Specialist Hospital and Research Center; Department of Otolaryngology, College of Medicine, Alfaisal University, Riyadh, Saudi Arabia

Date of Submission27-Apr-2022
Date of Decision03-Jun-2022
Date of Acceptance04-Jun-2022
Date of Web Publication12-Aug-2022

Correspondence Address:
Dr. Nasser K AlMutairi
PO 11597, Riyadh 11211, Kingdom of Saudi Arabia

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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/sjoh.sjoh_17_22

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  Abstract 


Background: Branchial cleft anomalies are the second-most common congenital malformation in the head-and-neck region. They arise from persistent ruminants of pharyngeal clefts and pouches and are classified into four distinct types; the most prevalent one is type 2, which involves the sternocleidomastoid muscles (SCM). The definitive treatment for the four types is complete surgical excision of the anomaly. Objective: The present study describes the clinical features and surgical outcomes for both unilateral and bilateral branchial cleft cysts at a tertiary care hospital in Saudi Arabia. Design and Setting: This retrospective study involved collecting data from patients' records of King Faisal Specialist Hospital, Saudi Arabia, who had surgical excision for branchial cleft cyst in the duration between January 2010 and September 2020. The collected data included patients' demographics, characters of the branchial anomaly, complications, and characters of the procedure. Statistical analysis was performed using IBM SPSS v. 20. for both descriptive and comparative analysis. Results: A total of 18 cases were eligible for inclusion. Of the whole cohort, 55.6% were females. All cases were type 2, with the majority being in the anterior border of sternoclenomastoid (83.3%) and unilateral (77.8%). Discharge was reported as the most common complication, occurring among half of the patients. The diagnostic investigation was only performed in situations of recurrence (22.2%). The prevalence of the bilateral cyst is 22.2%. There was no statistically significant association between the associated genetic disease and the unilateral or bilateral cysts (P = 0.054). Conclusion: Type 2 unilateral cysts are the most prevalent for the examined population, specifically on the anterior border of SCM. A low recurrence rate and low incidence of complications propose an effective management strategy.

Keywords: Saudi Arabia, summary, unilateral branchial cleft cyst bilateral branchial cleft cyst


How to cite this article:
AlMutairi NK, AlMofada HS, AlaEdin JB. Clinical features and surgical outcomes of branchial cleft cyst at a tertiary care hospital, Saudi Arabia, a retrospective study. Saudi J Otorhinolaryngol Head Neck Surg 2022;24:89-92

How to cite this URL:
AlMutairi NK, AlMofada HS, AlaEdin JB. Clinical features and surgical outcomes of branchial cleft cyst at a tertiary care hospital, Saudi Arabia, a retrospective study. Saudi J Otorhinolaryngol Head Neck Surg [serial online] 2022 [cited 2022 Dec 1];24:89-92. Available from: https://www.sjohns.org/text.asp?2022/24/3/89/353715




  Introduction Top


The primary etiology of branchial cleft anomalies is the incomplete obliteration of branchial clefts and pouches during embryogenesis.[1] These anomalies usually arise when the pouches, arches, or grooves of the branchial apparatus fail to resolve and fuse, leading to disconnection of the mucosa to the skin.[2] Cysts, fistulas, sinuses, or cartilaginous remnants develop in the early stages of life; however, the severity of the anomalies varies among patients.[1]

Branchial cleft anomalies represent about 30% of all congenital neck diseases.[3] Nevertheless, this number could be underestimated as some anomalies may remain undiagnosed until adulthood, while most cases are already diagnosed in early adulthood.[1] This delayed diagnosis is more attributed to cysts than sinuses or fistulae, which can be explained by the more observable skin lesions requiring almost chronic drainage via cervical sinus openings.[4],[5]

Branchial cleft anomalies are classified into four distinct groups, with type 2 being the most prevalent, accounting for between 69% and 90% of all branchial anomalies worldwide.[6],[7] Type 2 branchial cleft anomalies are defined as an anomaly occurring in the sternocleidomastoid muscles (SCM), most commonly along the anterior border, lined by squamous epithelium.[4] The clinical presentation of this type of branchial anomaly is commonly a stable, nontender, lateral mass in the neck that slowly enlarges. However, the mass enlargement may worsen during upper respiratory tract infections, leading to dysphagia torticollis and respiratory compromise.[5]

Complete excision of the branchial cleft anomaly showed positive prognostic outcomes.[4] Accordingly, any surgical intervention should be assessed and tailored to the type and origin of the anomaly. The stepladder surgical technique with two separate transverse incisions is currently recommended as the definitive cure.[8] It should be mentioned that inadequate presurgical assessments or incomplete excisions can result in unfavorable outcomes in terms of a high recurrence rate.[7]

This study aims to describe the characteristics of unilateral and bilateral branchial cleft cysts and surgical outcomes for patients operated at the King Faisal Specialist Hospital and Research Centre (KFSHRC), Saudi Arabia.


  Materials and Methods Top


Study design

This retrospective study included patients who had a surgical excision by stepladder incisions with help of methylene blue local injection for suspected co-occurrence of fistula either unilateral or bilateral branchial cleft cyst at KFSHRC, Riyadh-Saudi Arabia, in the duration between January 1, 2010, and September 30, 2020.

Data collection

The research team collected data retrospectively from patients' records. The collected data included information about patients' demographics, clinical characters of the anomaly (type, location, and side), presence or absence of sinus, the incidence of complications, investigations, surgical technique, and recurrence.


  Statistical analyses Top


Collected information was entered into an excel sheet, which was predesigned for data collection. Continuous data were expressed as a mean and associated standard deviation, and categorical data were expressed as either absolute values or percentages. Comparison between variables was conducted via either a t-test (for normally distributed variables), Mann–Whitney test (for nonnormally distributed variables) for numerical dependent variables, or Chi-square for categorical dependent variables. A value of P ≤ 0.05 was considered statistically significant. Data were analyzed via IBM SPSS v. 20 IBM Corporation, Armonk (N.Y., USA).

Ethical considerations

Patient privacy and medical confidentiality were maintained through de-characterization of patient records, with initial screening and anonymity being insured by hospital staff.


  Results Top


This study included 18 patients; a description of their clinical features is described below.

Demographics of patients and characters of anomalies

The included patients' age ranged between 1–47 years old, with a mean age of 11.83 ± 14.093 years. More than 50% of patients were females 10 (55.6%). As for the clinical features of the cysts, all patients had second branchial cleft cyst 18 (100%), and the majority, 15 (83.3%), had the cyst anterior to the border of SCM. Most patients have unilateral branchial cleft cysts 14 (77.8%). The sinus was present among 11 (61.1%) patients.

Concerning complications, 50% 9 (50%) reported discharge, and 7 (38.9%) had no fever. No one was suffering difficulty in breathing or swallowing, and no one had a family history, but only one patient (5.6%) reported an associated genetic disease. The vast majority suffered no complications (94.4%), whereas only one patient (5.6%) experienced blood loss of 50 ml.

Furthermore, diagnostic investigations were performed for only 4 (22.2%) of patients; of these, 3 (16.7%) performed computed tomography (CT) scans, and only one patient (5.6%) performed both CT scans and magnetic resonance imaging (MRI). More than half of the patients were treated with the use of methylene blue (61.1%), and 3 (16.7%) underwent complementary tonsillectomy and tie and all of them were unilateral cases. Most of the patients suffered no recurrence (77.8%), as described in [Table 1]. While the recurrence rate of 22.2% which was attributed to inadequate surgical excision of the tract for which they were scheduled for revision surgery later on.
Table 1: Demographics and clinical characteristics of the patients

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Correlation between location of the cyst and other factors

Using Chi-square testing, the site of cysts has been compared over family history and the presence/absence of associated genetic disease. It was found that there was no statistically significant association between the associated genetic disease and the unilateral or bilateral cysts (P = 0.05), as described in [Table 2].
Table 2: Correlations between factors and cysts

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  Discussion Top


Although branchial cleft cysts are among the most frequent neck and head congenital malformations, the clinical features and surgical management of branchial anomalies in the gulf area, particularly in Saudi Arabia, are still very sparse. Consequently, the present study aimed to describe the outcomes, complications, and recurrence of branchial cleft cysts, either on unilateral or bilateral sites, after surgical excision in Saudi Arabia.

The present study demonstrated that the mean age for the included cohort was 11.83 years old, with females showing slightly higher presentations than males. This age was higher than the average reported in a previous study that focused on patients presenting with different malformations, where the average age was 3.6 years, 4.1 years, and 2.6 years for sinus, cysts, and fistulas, respectively.[9]

This difference can be attributed to multiple reasons. In the present study, age was recorded in patients' records at the time of admission, which might not reflect the actual age at the time of diagnosis. In addition, the small sample size in the present study might have affected the mean age calculation. Furthermore, some patients may have had their surgery delayed or postponed for multiple reasons. Turning to gender distribution, a similar representation for females was also identified in another study, which reported a 2:1 ratio for gender selectivity.[10]

Clinical features of the branchial cleft cysts were extensively examined. The present study demonstrated that all patients had type 2 branchial anomalies supported by other studies that reported type 2 branchial anomalies as the dominant type.[9],[11] Nevertheless, some studies also classified sinus as the most prevalent type.[12] In addition, the location of the cysts varied among unilateral and bilateral sites, where unilateral cysts at the anterior border of the SCM were the prevalent disposition in the present study. This location has also been supported by other published literature.[13],[14]

The diagnosis and surgical management were also evaluated. The present study revealed that only 22.2% had a record of imaging investigations, with four patients requiring a CT scan and one patient requiring an MRI. Methylene blue was administered in over half of the included cohort, and tonsillectomy and tie were the most frequent surgeries performed due to fistula occurrence which estimated to be 16.7%. These figures are compliant with the medical literature, where methylene blue is well established to facilitate surgery; however, the injection of methylene blue does not necessarily demonstrate the entire extent of the anomalies as it may be blocked by secretions.[15] Hence, using methylene blue seemed to reduce the recurrence in our cohort.

Surgical excision for the treatment of type 2 branchial anomalies is known to be effective, and significant complications are rare. Only minor complications that do not require surgical intervention were reported, including stitch extrusion, minor wound infections, and seromas.[10],[13] In the present study, only one patient reported a blood loss of 50 ml, indicating that all procedures were competently performed. However, 22.2% of patients suffered a recurrence, which is just over the reported recurrence rate for such procedures in the literature (14%–22%),[16] which requires further assessment in future studies.

It is worth mentioning that the present study was limited by the retrospective nature of the collected data, which might have led to inaccuracies or missing information. Furthermore, the small sample size of the included patients may have affected reaching statistical significance for comparative analysis. These limitations should be taken into consideration in future studies.


  Conclusion Top


Type 2 unilateral SCM branchial cleft cysts were the most common form of branchial cleft anomalies. Surgical interventions with the use of methylene blue showed high levels of effectiveness as well as safety, with no indication of significant complications. There was no significant association between the associated genetic disease and the unilateral or bilateral cysts. Future studies should focus on evaluating postsurgical care. A Robust study design should also be adopted, including multi-center data collection, prospective design, and recruiting a larger sample size.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Li W, Xu H, Zhao L, Li X. Branchial anomalies in children: A report of 105 surgical cases. Int J Pediatr Otorhinolaryngol 2018;104:14-8.  Back to cited text no. 1
    
2.
Goff CJ, Allred C, Glade RS. Current management of congenital branchial cleft cysts, sinuses, and fistulae. Curr Opin Otolaryngol Head Neck Surg 2012;20:533-9.  Back to cited text no. 2
    
3.
Mohamed K, Badawy MK. Pediatric neck masses. Clin Pediatr Emerg Med 2010;11:73-80.  Back to cited text no. 3
    
4.
Magdy EA, Hamza A, Youssef A, Yoneis A. Second branchial cleft fistula/sinus tract endoscopy: A novel intraoperative technique assisting complete surgical resection. Eur Arch Otorhinolaryngol 2021;278:833-8.  Back to cited text no. 4
    
5.
Moroco AE, Saadi RA, Patel VA, Lehman EB, Wilson MN. Postoperative outcomes of branchial cleft cyst excision in children and adults: An NSQIP analysis. Otolaryngol Head Neck Surg 2020;162:959-68.  Back to cited text no. 5
    
6.
Jaka RC, Singh G. Complete congenital third branchial fistula on right side. Otolaryngol Head Neck Surg 2007;137:518-9.  Back to cited text no. 6
    
7.
Rankovic N, Todorovic J, Simic R. Clinical and ultrasound characteristics of pediatric lateral neck masses. PLoS One 2021;16:e0251563.  Back to cited text no. 7
    
8.
Li L, Liu J, Lv D, Shen T, Deng D, Wang J, et al. The utilization of selective neck dissection in the treatment of recurrent branchial cleft anomalies. Medicine 2019;98:e16799.  Back to cited text no. 8
    
9.
Schroeder JW Jr., Mohyuddin N, Maddalozzo J. Branchial anomalies in the pediatric population. Otolaryngol Head Neck Surg 2007;137:289-95.  Back to cited text no. 9
    
10.
Agaton-Bonilla FC, Gay-Escoda C. Diagnosis and treatment of branchial cleft cysts and fistulae. A retrospective study of 183 patients. Int J Oral Maxillofac Surg 1996;25:449-52.  Back to cited text no. 10
    
11.
Waldhausen JH. Branchial cleft and arch anomalies in children. Semin Pediatr Surg 2006;15:64-9.  Back to cited text no. 11
    
12.
Choi SS, Zalzal GH. Branchial anomalies: A review of 52 cases. Laryngoscope 1995;105:909-13.  Back to cited text no. 12
    
13.
Grohmann NC, Herrington HC. Second branchial cleft anomalies. Oper Tech Otolaryngol Head Neck Surg 2017;28:156-60.  Back to cited text no. 13
    
14.
Doshi J, Anari S. Branchial cyst side predilection: Fact or fiction? Ann Otol Rhinol Laryngol 2007;116:112-4.  Back to cited text no. 14
    
15.
Shen LF, Zhou SH, Chen QQ, Yu Q. Second branchial cleft anomalies in children: A literature review. Pediatr Surg Int 2018;34:1251-6.  Back to cited text no. 15
    
16.
Reiter D. Third branchial cleft sinus: An unusual cause of neck abscess. Int J Pediatr Otorhinolaryngol 1982;4:181-6.  Back to cited text no. 16
    



 
 
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  [Table 1], [Table 2]



 

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