Table of Contents  
CASE REPORT
Year : 2014  |  Volume : 6  |  Issue : 2  |  Page : 125-128

Branchial cleft cyst - A case report with review of literature


1 Department of Oral Medicine and Radiology, Panineeya Dental College and Hospital, Hyderabad, Telangana, India
2 Department of Oral Medicine and Radiology, Dental College and Hospital, Gujarat, India
3 Department of Oral Medicine and Radiology, Dr. D.Y. Patil Dental College & Hospital, Pune, Maharashtra, India

Date of Web Publication16-Oct-2014

Correspondence Address:
Mamatha Boringi
V2-503, TNR Shakunthala Apartment, Saroornagar Road, LB Nagar, Hyderabad, Telangana
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0975-8844.143058

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  Abstract 

Branchial cleft cysts are the most common congenital neck masses. The other name for Branchial cleft cyst is Benign cervical lymphoepithelial cyst, which occurs on the lateral aspect of the neck. These cysts originate from remnants of the branchial arches or branchial pouches. The lesions presents as an asymptomatic circumscribed movable mass on the anterior border of the sternocledomastoid muscle. Many cases have been reported at the angle of the mandible, in the submandibular area and even in the pre-auricular and parotid areas. 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.
A solitary, 1 month old swelling on the right submandibular region, in a 13-year-old girl, caused diagnostic dilemma with clinical presentation. Diagnosis was done after all the investigations and treated accordingly.

Keywords: Branchial arches, MRI, sternocledomastoid muscle


How to cite this article:
Boringi M, Bontha SC, Kaur M, Shireen A. Branchial cleft cyst - A case report with review of literature . J Orofac Sci 2014;6:125-8

How to cite this URL:
Boringi M, Bontha SC, Kaur M, Shireen A. Branchial cleft cyst - A case report with review of literature . J Orofac Sci [serial online] 2014 [cited 2017 Jul 21];6:125-8. Available from: http://www.jofs.in/text.asp?2014/6/2/125/143058


  Introduction Top


The branchial cleft cyst is a developmental cyst of the lateral neck region. These are the most common congenital neck masses. [1] Other names are, Congenital hydrocoele of the neck, Hygroma colli, Branchial cyst, tumor of the branchial cleft, Lateral lympho-epithelial cyst, Benign cystic lymph nodes, Dermoid cyst of the sheath of the internal jugular vein, and deep seated atheromatous tumor. [2] Lateral cysts of the neck were first described by Hunczovsky in 1785. [2],[3] Ninety-five percent of branchial anomalies are second branchial anomalies. These anomalies present as sinuses, fistulae and cysts. Branchial cleft cysts comprise approximately 75% to 80% of all branchial anomalies. 95% of these cysts are believed to arise from the second branchial arch. [4] 5% originating from first, third and fourth branchial arches. Branchial cleft cyst most commonly occurs, in the upper lateral neck along the anterior border of the sternocledo mastoid muscle. Proctor has shown that second branchial cleft cysts occur three times more often than second branchial sinuses or fistulas. [3]


  Case Report Top


A 13-year-old girl came with a swelling on the right side of the lower jaw since 1 month [Figure 1]. Patient had difficulty in mastication. Mild pain associated with swelling, which aggravated at the night time, mainly at the back of the neck. Patient had pain in right lower back deciduous tooth, 2 weeks back, which was exfoliated after 1week. On examination, solitary diffused swelling was noted on the right submandibular region. It was non-pulsatile, with smooth surface texture. Surrounding skin was normal. It was not moving with protrusion of the tongue or on deglutation. On palpation, surface temperature was not raised, non-tender, soft in consistency, fluctuant, compressible, and not reducible. No secondary changes were noted.
Figure 1: Showing ovoid Swelling on the right submandibular region

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Provisional diagnosis was given as residual right Submandibular Space infection. Differential Diagnosis considered as Plunging ranula, Tubercular lymphadenitis, Suppurative lymphadenitis, Hodgkin's Lymphomas, Metastatic neoplasia, Teratoma, Ectopic salivary tissue.

The OPG shows [Figure 2] normal features with erupting first premolars and canines and second molars. Mantoux test was negative. FNAC suggestive of Mucocele, because of thick mucosal fluid. Ultrasonogram suggested as Cystic Sol [Figure 3]. MRI gives evidence of fairly large irregular cystic lesion measuring about 7 × 22 × 35 mm 3 in size, noted at right submandibular region, anterolateral to right sternocledomatoid muscle. The lesion is hyper in T2 [Figure 4] and Hypo in T1 [Figure 5] with minimal heterogenous signal posteriorly. Anteriorly, the lesion is extending close to the myelohyoid muscle and ramus of the mandible. Posteriorly the lesion is extending, anterior to the upper third region of S.C.M muscle. The submandiblar gland and muscles in the region are displaced medially. MRI suggested of cystic lesion in right submandibular region. Submandibular incision was given and complete enucleation of the cyst had been done. And the cystic lining was sent for histopathological examination.
Figure 2: OPG showing erupting first premolars and canines and second molar

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Figure 3: Ultra sound showing well defined hypoechoeic area

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Figure 4: Showing T2 weighted image showing hyperintance lesion

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Figure 5: T1 weighted image showing hypodence area

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Histopathology revealed that the cyst wall has flattened lining epithelium underlying fibro vascular connective tissue and diffuse infiltrated with lymphocytes suggestive of brancheal cyst [Figure 6].
Figure 6: Histologic picture showing lymphocytic infiltration

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  Discussion Top


Rathke described pharyngeal pouches in 1828. The cervical lymphoepithelial or branchial cleft cyst has a disputed pathogenesis. The etiological hypotheses are as follows:

  1. Congenital theories: The classic theory that the cyst develops from remnants of the embryonic gill apparatus.
  2. Lymph node theories: In 1949, King concluded that the cyst arises from cystic changes in parotid epithelium that becomes entrapped in the upper cervical lymph nodes during embryonic life.
  3. Branchial theory: Cyst develops from imperfect obliteration of the pharyngeal cleft.
  4. Pre-Cervical Sinus Theory: The cyst develops from investigates of the cervical sinus rather than of the pharyngeal clefts or pouches. [3]


King's criteria is that, any cyst arising outside the midline of the neck and having lymphoepithelial characteristics should be regarded as a branchial cyst. [3],[5]

Amot proposed the first classification for anomalies of the first branchial cleft.

He designated that first branchial anomalies as a 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.

Bailey Classified 2nd Branchial cyst: 4 sub types:

Type I: Is anterior to SCM, beneath platysma muscle.

Type II: Is adjacent to ICA and often adherent to IJV: Most common

Type III: Extends between ICA and ECA to lateral pharyngeal wall

Type IV: Lies against lateral pharyngeal wall and may extend to skull base. [6],[8]

This cyst frequently affects the young adults between the ages of 20 and 40 years. Regarding age at presentation, Telander and Deane found, in their survey that sinuses and fistulas typically arise in the first decade of life and to a lesser extent in the second decade, whereas cysts occur in the adolescent and adult which contradicts with present case. Cyst appears as soft, fluctuant mass, that can be larger from 1 cm to 10 cm in diameter. The pattern of inheritance is consistent with an autosomal gene having incomplete penetrance. 20-40% of patients relate its appearance to a recent upper-respiratory-tract infection, odontogenic infection or even in pregnancy. Infection in the deciduous tooth might trigger the lesion. If large enough, the anomalies can cause asymmetry of the neck, as well as dyspnea, dysphagia and dysphonia. [7] Larger cyst may displace the sternocledo mastoid muscle posterolaterally, and the carotid and internal jugular vein medially. [7] When bilateral cysts/sinuses develop, there seems to be a familial tendency. Cyst appears more commonly in males than in females. 2/3 rd of the branchial cyst occurs on the left side of the neck. 1/3 rd found on the right side.

Investigations

FNAC

Is very helpful in preoperative diagnosis. The criteria for FNA cytology diagnosis of branchial cyst are: a) thick, yellow, pus-like fluid b) a nuclear, keratinizing cells c) squamous epithelial cells of variable maturity and d) a back ground of amorphous debris. [7] Aspirate appears as a straw-colored fluid, that microscopically may exhibit squamous cells, polymorphonuclear cells, lymphocytes and cholesterol crystals. The sonomorphologic findings typically yield a rounded mass that has a uniform low echogenicity lacking internal septation, with no acoustic enhancement or motion. Treatment complete enucleation of the cyst is necessary. No recurrence has been noted. Branchial cyst carcinoma is extremely rare, compared to the far more frequent cystic metastases arising from primary malignancies. [9],[10] But in another situation, 13 cases of malignant second branchial cleft cysts in the literature had been noted that fulfill Khafif's modified criteria. [9]

Histopathology

More than 90% of branchial cleft cysts are lined by stratified squamous epithelium. May or may not be keratinized. The wall of the cyst typically contains lymphoid tissue - germinal centers formation. Some authors have noted the presence of hair follicles and sebaceous and sweat glands within the cyst. [7],[9]


  Conclusion Top


The branchial cleft cyst is a developmental cyst of the lateral neck region. Though it is a common lesion occurring in the head and neck region, the present case has a varied presentation and throws light on the diagnostic aspect for proper treatment planning of a brachial cyst. It is uncommon to suspect a lesion of such a type in dental fraternity and this often leads to mistaken diagnosis and improper treatment planning of the same. Hence, the present case is reported to broaden the differential diagnosis of swellings occurring in the lateral neck region.

 
  References Top

1.
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. 1
    
2.
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. 2
    
3.
Sinha D, Utture SK. Branchial cysts: A case report of a benign lymphoepithelial cyst in the Neck with review of literature. Bombay Hospital Journal.  Back to cited text no. 3
    
4.
Anesi A, Pollastri G, Bondì V, Barberini S, Chiarini L. Absence of adjuvant radiotherapy may be an additional criteria in diagnosing a branchiogenic squamous cell carcinoma: A case report. OJST 2012;2:63-7.  Back to cited text no. 4
    
5.
Bhanote M, Yang GC. Malignant first brancial 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. 5
    
6.
Harnsberge, Wiggins, Hudgins, Michel, Swartz, Davidson Mac Donald, Glanstonbury, Cure, Branstetter. Diagnostic Imaging: Head and Neck. 1 st ed. India: Amirsys publishing; 2009, p. 1-24.  Back to cited text no. 6
    
7.
Lee HJ, Kim EK, Hong S. Sonographic detection of intrathyroidal branchial cleft cyst: A case report. Korean J Radiol 2006;7:149-51.  Back to cited text no. 7
    
8.
Chun RH, Choi SS. First branchial cleft cyst: A rare presentation with mesotympanic extension. Int J Pediatr Otorhinolaryngol Extra 2009;4:80-3.   Back to cited text no. 8
    
9.
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. 9
    
10.
Wasson J, Blaney S, Simo R. A third branhial pouch cyst presenting as stridor in a child. Ann R Coll Surg Engl 2007;89:W12-4.  Back to cited text no. 10
    


    Figures

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



 

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