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 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 8  |  Issue : 2  |  Page : 88-91

Branchial cleft cyst: A case report and literature review


Department of Oral Pathology, SRM Dental College, Chennai, Tamil Nadu, India

Date of Web Publication8-Jun-2017

Correspondence Address:
R Swarna Lakshmi
Department of Oral Pathology, SRM Dental College, Chennai, Tamil Nadu
India
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DOI: 10.4103/srmjrds.srmjrds_70_16

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  Abstract 

Branchial cleft cysts are the most common congenital neck masses which originate from remnants of the branchial arches or branchial pouches. The lesion presents as an asymptomatic circumscribed movable mass on the anterior border of the sternocleidomastoid muscle. The dentist should be aware of this entity, because it can be easily confused with an odontogenic infection or parotid pathology, especially, if it develops high up in the neck. In this article, we report a case of infected branchial cyst in the left side of the neck region along with its literature review.

Keywords: Branchial arches, branchial cleft cyst, cervical lymphoepithelial cyst


How to cite this article:
Lakshmi R S, Nandhini G, Jayant V S, Rajkumar K. Branchial cleft cyst: A case report and literature review. SRM J Res Dent Sci 2017;8:88-91

How to cite this URL:
Lakshmi R S, Nandhini G, Jayant V S, Rajkumar K. Branchial cleft cyst: A case report and literature review. SRM J Res Dent Sci [serial online] 2017 [cited 2019 Nov 17];8:88-91. Available from: http://www.srmjrds.in/text.asp?2017/8/2/88/207653


  Introduction Top


Branchial cleft cysts are the most common congenital neck masses, which occurs on the lateral aspect of the neck. It was first described by Hunczovsky in the year 1785.[1] These cysts originate from remnants of the branchial arches or branchial pouches of which 95% of these cysts are believed to arise from the second branchial arch and 5% originating from first, third and fourth branchial arches. The most common site is upper lateral neck along the anterior border of sternocleidomastoid muscle and usually presents as an asymptomatic circumscribed movable mass.[2],[3]


  Case Report Top


A 49-year-old female patient reported to the Department of Oral Medicine, in 2014 with a chief complaint of swelling in the left side of the neck region for the past 5 years. History revealed that the swelling was initially small in size and gradually increased in size for the past 5 years to attain the present size [Figure 1]. The patient also gave a history of being treated elsewhere three times for the same complaint and which had recurred again.
Figure 1: Well-circumscribed oval swelling in the neck

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On clinical examination, a single well-circumscribed oval swelling was seen in the left upper part of the neck. The swelling extended anteriorly from the angle of the mandible and posteriorly up to the tragus of the ear in relation to the anterior border of sternocleidomastoid muscle and measured approximately 3.5 cm × 2 cm in size. The skin covering the swelling appeared normal as that of the adjacent area. Palpation revealed that the swelling was firm in consistency, pinchable and was nontender. Based on history and clinical findings, differential diagnosis of neurofibromatosis, lipomatosis, lymphadenitis, or branchial cyst was made.

Complete blood investigations were done which were under normal limits and also the possibility of HIV and tuberculosis were ruled out.

Fine needle aspiration cytology (FNAC) was done, and it revealed nonspecific chronic inflammatory cells such as lymphocytes, plasma cells, few neutrophils, and eosinophils in an eosinophilic proteinaceous background suggestive of cystic smear [Figure 2].
Figure 2: Fine needle aspiration cytology, ×10 showing lymphocytic infiltrate

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Under general anesthesia, the lesion was surgically removed [Figure 3] and fixed in 10% formalin and sent for histopathological diagnosis [Figure 4].
Figure 3: Surgically exposed lesion

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Figure 4: Gross specimen of the excised lesion

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Microscopic examination of the excised tissue revealed multiple dilated cystic areas lined by pseudostratified ciliated epithelium, which was thrown into papillary folds into the lumen. The cystic areas were surrounded by dense sheets of lymphoid follicles consisting of abundant lymphocytes, plasma cells, and histiocytes and also showed germinal centers [Figure 5],[Figure 6],[Figure 7].
Figure 5: H and E, ×4 showing cystic lesion with lining epithelium with underlying connective tissue wall showing germinal center with lymphoid aggregate

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Figure 6: H and E, ×10 showing cystic cavity lined by pseudostratified ciliated epithelium with the underlying connective tissue wall showing abundant lymphoid tissue aggregate

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Figure 7: H and E, ×40 showing cystic cavity lined by pseudostratified ciliated epithelium

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Correlating with the patient's history, clinical, FNAC and histopathological findings the swelling was diagnosed as branchial cyst and as there was abundant inflammatory cell infiltrate the final diagnosis was given as infected branchial cyst. The patient recovered well and had no recurrence after 2-year follow-up.


  Discussion Top


Branchial cleft cyst is a soft tissue swelling usually seen in the neck of young adults. It is also termed as congenital hydrocoele of the neck, hygroma colli, lateral lymphoepithelial cyst, and benign cystic lymph nodes.[4] It usually occurs at the angle of the mandible, in the submandibular area and also in the preauricular and parotid areas.[5]

Clinically, they present as an asymptomatic lump with well-defined margins and are usually slow growing. Sometimes, they present as fistulas due to the sudden growth of the lesion as a result of infection of the cyst with signs of local inflammation.

The etiopathogenesis of branchial cleft cyst is controversial with four main theories of origin which has been postulated till now, such as they may occur due to incomplete obliteration of branchial apparatus, persistence of vestiges of the precervical sinus, thymo-pharyngeal ductal origin, and cystic lymph node origin of which the first one is the most accepted theory.[6]

During embryogenesis at the 4th week of embryonic life, the development of 4 branchial (or pharyngeal) clefts results in 5 ridges known as the branchial (or pharyngeal) arches, which contribute to the formation of various structures of the head, neck, and thorax. The second arch grows caudally and ultimately, covers the third and fourth arches. The buried clefts become ectoderm-lined cavities, which normally involutes around week 7 of development. If a portion of the cleft fails to involute completely, the entrapped remnant forms an epithelium-lined cyst with or without a sinus tract to the overlying skin.[7]

King's criteria state that any cyst arising outside the midline of the neck and having lymphoepithelial characteristics should be regarded as a branchial cyst.[8],[9]

Amot proposed the first classification for anomalies of the first branchial cleft. Type 1 defect as any cyst or sinus in the parotid gland that is lined by squamous epithelium and which presents in early or middle adult life. Type 2 defects are those which, develop during childhood in the anterior triangle of the neck, with a communicating tract to the external auditory canal. A high index of suspicion for the first branchial cleft cyst is needed when the sinus opening is located in the Pochet's triangle.[10]

Bailey classified second branchial cyst into four subtypes:

  • Type I: Localized between the platysma and anterolateral surface of the sternocleidomastoid
  • Type II: Along the anterior surface of the sternocleidomastoid muscle lateral to the carotid space and posterior to the submandibular glands
  • Type III: Between the internal carotid and the external carotid artery, the presence of a “tail” of the cyst between these two vessels is pathognomonic of Type III cysts
  • Type IV: Originate from the pharyngeal mucosa, deeply compared to the palatine tonsils, and often can reach the base of the skull.[11]


The anomalies of the third and fourth branchial cleft are rare, and they are localized at the lower third of the neck. The second branchial cleft cysts are often located between the first and second third of the front surface of the sternocleidomastoid muscle.

The clinical differential diagnosis of branchial cleft cyst is lipomatosis, tuberculosis lymphadenitis, HIV-related lymphadenopathy, cat-scratch disease, sarcoidosis and Hodgkin's lymphoma. Other far less common entities such as vascular neoplasms, carotid body tumor, lymphatic malformation (cystic hygroma), ectopic thyroid tissue, ectopic salivary tissue, and glomus tumor of head and neck are also to be considered.[5],[11],[12]

Both computed tomography (CT) scanning and magnetic resonance imaging are useful in the evaluation of branchial cleft cysts. CT imaging of branchial cleft cyst usually presents as a well-circumscribed homogeneous cystic lesion which may be unilocular or separated if secondarily infected. The typical sonographic appearance of a brachial cleft cyst has been described as an anechoic, well-circumscribed cyst. Some may show faint low-level internal echoes if infected. Others have suggested that the sonographic appearances may range from anechoic to highly echogenic.[13]

FNAC proves a strong evidence to arrive at a provisional diagnosis if the lesion is a benign cyst or a metastatic carcinoma. In branchial cleft cyst, the aspirant appears as a variably thick, grey-yellow, pus-like fluid. The criteria for diagnosis are the appearance of anucleate, keratinized cells, squamous epithelial cells, mainly mature, some metaplastic, in a background of amorphous debris, and often inflammatory cells, appropriate to anatomical site.[14]

Microscopically, they show either stratified squamous or pseudostratified columnar lining epithelium and ciliated in few cases. Sometimes, the lining may be ulcerated. The connective tissue wall contains numerous lymphoid tissue which may show typical germinal centers. Some authors have noted the presence of hair follicles, sebaceous and sweat glands within the cyst.[5],[15]

The treatment of the branchial cyst involves complete enucleation, and usually, there is no recurrence on follow-up.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Thomaidis V, Seretis K, Tamiolakis D, Papadopoulos N, Tsamis I. Branchial cysts. A report of 4 cases. Acta Dermatovenerol Alp Pannonica Adriat 2006;15:85-9.  Back to cited text no. 1
    
2.
Alexandre A, Giuseppe P, Vincenzo B, Sara B, Luigi C. Absence of adjuvant radiotherapy may be an additional criteria in diagnosing a branchiogenic squamous cell carcinoma: A case report. Open Journal of Stomatology 2012;2:63-7.  Back to cited text no. 2
    
3.
Chavan S, Deshmukh R, Karande P, Ingale Y. Branchial cleft cyst: A case report and review of literature. J Oral Maxillofac Pathol 2014;18:150.  Back to cited text no. 3
[PUBMED]  [Full text]  
4.
Frazer JE. The nomenclature of diseased states caused by certain vestigial structures in the neck. Br J Surg 1923;11:131-6.  Back to cited text no. 4
    
5.
Shear M, Speight PM. Cysts of the Oral and Maxillofacial Regions. 4th ed. John Wiley & Sons; 2008.  Back to cited text no. 5
    
6.
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. 6
    
7.
Little JW, Rickles NH. The histogenesis of the branchial cyst. Am J Pathol 1967;50:533-47.  Back to cited text no. 7
    
8.
Sinha D, Utture SK. Branchial cysts: A case report of a benign lymphoepithelial cyst in the neck with review of literature. Bombay Hosp J 2001;1:4303.  Back to cited text no. 8
    
9.
Bhanote M, Yang GC. Malignant first branchial cleft cysts presented as submandibular abscesses in fine-needle aspiration: Report of three cases and review of literature. Diagn Cytopathol 2008;36:876-81.  Back to cited text no. 9
    
10.
Harnsberge HR, Wiggins RH, Hudgins PA, Michel MA, Swartz J, Mac Donald D, et al. Diagnostic Imaging: Head and Neck. 1st ed. India: Amirsys Publishing; 2009. p. 1-24.  Back to cited text no. 10
    
11.
Boringi M, Bontha SC, Kaur M, Shireen A. Branchial cleft cyst – A case report with review of literature. J Orofac Sci 2014;6:125.  Back to cited text no. 11
  [Full text]  
12.
Somashekara KG, Babu KG, Lakshmi S, Geethamani V, Yashaswi RG, Srinivas CV. Type II first branchial cleft cyst: A case report with review of literature. Indian J Otolaryngol Head Neck Surg 2011;63 Suppl 1:75-7.  Back to cited text no. 12
    
13.
Ahuja AT, King AD, Metreweli C. Second branchial cleft cysts: Variability of sonographic appearances in adult cases. AJNR Am J Neuroradiol 2000;21:315-9.  Back to cited text no. 13
    
14.
Cangiarella J, Breast SA. Orell and Sterrett's Fine Needle Aspiration Cytology. 5th ed. Sydney: Elsevier; 2012. p. 40-1.  Back to cited text no. 14
    
15.
Chen H, Li M, Lin T. Branchial cleft cyst carcinoma: A case report. Chin J Radiol 2005;30:231-4.  Back to cited text no. 15
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]



 

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