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BRANCHIAL CYSTS : A CASE REPORT OF A BENIGN LYMPHOEPITHELIAL CYST IN THE NECK WITH REVIEW OF LITERATURE

Pranava Sinha, Shivkumar S Utture

*Surgical Registrar; **Honorary Associate Professor, Dept. of General Surgery, Grant Medical College, Sir JJ Group of Hospitals, Mumbai.

Branchial cysts or lymphoepithelial cysts have been known to occur in neck anterior to the upper third of the sternomastoid. Although they have been reported to occur in the other regions of the anterior triangle of the neck, a cyst in the posterior triangle of the neck is extremely rare. This is a report of such a case with review of the available literature.

INTRODUCTION

Branchial cysts are the commonest cysts to arise in the neck. In the early descriptions they were described as to arise anterior to the sternocleidomastoid muscle. However there have been a number of case reports describing cysts which were found in areas other than the classical position. Just as there have been various sites for the cysts, there have been a number of theories proposed as possible aetiologies. This is a case report of a branchial cyst found in the posterior triangle of the neck with a review of the available literature.

CASE REPORT

A 23 year old female presented with a solitary swelling in the left side of the neck of 6 months duration. Initially the swelling was of the size of pea, and gradually increased with time to attain the size approximately equal to the size of the patient’s fist. There was no pain in the swelling. On examination an 8 cm x 7 cm swelling was found in the left posterior triangle of the neck. It extended from the anterior border of the left sternomastoid to the anterior border of the left trapezius, anteroposteriorly and from the level of the thyroid prominence superiorly to about 3 cm medial to acromion process inferiorly. The smooth, well-defined swelling was fluctuant and brilliantly transluminant. On operation a well-circumscribed unilocular cyst was found without any connecting tract or cord to the skin or the pharynx. The cyst contained clear yellowish fluid. Microscopic examination of the cyst wall revealed a focally preserved flattened cuboidal epithelial lining with lymphocyte aggregates in the underlying collagenous wall.

REVIEW OF LITERATURE

Lateral cysts of the neck were first described by Hunczovsky in 1785. [1] Since then these swellings have been described by various names and various aetiological hypotheses have been proposed for them.

These hypotheses can be conveniently classified as the "Congenital theories" and the "Lymph node theories". Rathke described pharyngeal pouches in 1828. Following this Ascherson proposed the "Branchial theory", suggesting the imperfect obliteration of the pharyngeal cleft as the cause of these cysts. But in 1886 he proposed his "Precervical Sinus Theory" saying that these cysts were related to the cervical sinus rather than the pharyngeal clefts or pouches. [1]

Fig 1
Fig 1 : Branchial cyst in the left posterior triangle - As seen from the front

Wenglowski in 1912 in his dissection of cadavers found that no pharyngeal cleft tissue was found below the level of the hyoid bone and thus pointed out the inability of the "branchial theory" in explaining the cysts in the lower neck. He proposed the possibility of incomplete obliteration of the thymopharyngeal duct.[1]

Based on the findings of Lucke and Luschka, King proposed the "Lymph node theory". Bhasker and Bernier, after their histological study of cysts concluded that these cysts developed due to cystic transformation of cervical lymph nodes. Maran and Buchanan arrived at the same conclusion however they also pointed out that such a transformation was not known to occur in any other site. [1]

Fig 2
Fig 2 : Branchial cyst in the left posterior triangle - Lateral view

There was a lot of speculation about the possible origin of the epithelium within the lymph node that led to the cystic transformation of the node and eventually formed the lining of the cyst. Bhasker and Bernier suggested 3 possible sources, the branchial cleft, the pharyngeal pouch and the parotid gland. [1-4]

Wilde and Mischke [5] , [6] studied the keratin content of the epithelial lining of the cysts and found it homologous to the keratin content of the upper digestive tract epithelium, particularly that of the palatine tonsils. This was further confirmed by Stoll7 who injected blue dye into the tonsillar fossa of 3 cases preoperatively and found the distribution of stain within the capsule of the cyst during surgery. Hosemann after his study of the histopathological characteristics of the cyst epithelium, found it similar to the tonsillar epithelium. [8]

Fig 3
Fig 3 : Brillant Transillumination of the cyst

Although both the branchial cysts and fistulas were regarded to be of branchial origin now it is believed that "branchial cysts" have a non-branchial origin. [9-13]

The median age of presentation of these cysts is in the 3rd decade. They are found to occur more commonly in females. [1] , [14] They have classically been described to occur anterior to the upper third of the sternomastoid. [15] However they have been reported to occur in the other areas of the neck, [1], [16]as well as in the oral cavity,17-19 within the salivary glands, [16], [20-23] the thyroid24 and even in the mediastinum 25 and within the pancreas. [26]

Within the neck they occur most commonly in the region anterior to the upper third of the sternomastoid, followed by the region to the middle and lower third of the sternomastoid. [15]It very rarely occurs in the posterior triangle of the neck. Most of the cysts occur deep to the investing layer of the deep cervical fascia and none of them have a cord or tract attached leading to the skin or the pharynx. [1], [11], [12], [14]

On microscopic examination these cysts are composed of a wall of lymphoid tissue lined with squamous or columnar cells. [1], [8] , [14]Due to the variability of the position King suggested that any cyst arising outside the midline, with the histological features as above should be regarded as a lymphoepithelial or a branchial cyst. [1]

TABLE 1
Sites of branchial cysts in the neck

Site

Titchener and Allison

Golledge and Ellis

Neel and Pemberton

King

Bhasker and Bernier

Ant. to Upper 1/3 of SCM

35

85

73

74

41

Ant. to Mid and Lower 1/3 of SCM

 

10

22

7

15

Submandibular r Region

7

5

0

3

10

Parotid

0

0

0

3

Anterior Neck

 

0

0

0

15

Posterior Triangle

 

0

0

0

3

 

Langlois [27] has described pancreatic tissue in a lateral cervical cyst, while Gosain and Wildes[28] found gastric epithelium within a branchial cyst. There have also been reports of secondaries from papillary thyroid cancers [29]and tonsillar cancers masquerading as branchial cysts, [30]but such cases are rare. Most cases present with a painless, soft, cystic and brilliantly transcluscent swelling in the neck. Rarely is a cyst painful. [14]

Although clinical diagnosis of a cyst in the classical position is relatively simple. They are seldom correctly diagnosed preoperatively at other sites. Titchener and Allison [14] could make a correct preoperative diagnosis in only 22 out of their 42 cases, and have stressed on the role of preoperative ultrasonography of the neck and FNAB for evaluation of such cases. Earl31 has also found USG to be very helpful in the diagnosis of cystic neck swellings. The role of FNAB and preoperative CT scan of the neck has also been discussed by Rossell. [32]

Most patients are cured by complete surgical excision 9 of the cyst and usually never get a recurrence [11] and surgery remains the mainstay in the treatment of lateral cervical cysts. Unlike cystic hygromas there are no reports of any use of sclerosing agents or other drugs like OK432 or bleomycin in this condition.

DISCUSSION

As per King’s criteria any cyst arising outside the midline of the neck and having lymphoepithelial characteristics should be regarded as a branchial cyst. Such cysts are found more commonly in females and usually occur in the 2nd or 3rd decade of life. They are most commonly found in the anterior triangle of the neck anterior to the upper third of the sternomastoid. A cyst occupying the posterior triangle is extremely rare. However these cysts have been reported to occur in all the regions of the neck, and even in the mediastinum and the abdomen. Hence they should be suspected in all the cystic swellings of the neck except the median ones. Ultrasonography and FNAB definitely help in arriving at the diagnosis and is especially recommended for patients in the older age group to rule out cystic secondaries from head and neck malignancies. On operation a unilocular cyst with clear fluid, deep to the investing fascia and without a connecting stalk more or less makes the diagnosis certain. The histological picture is classical and confirmatory.

The ‘Branchial theory’ has now fallen into disfavour and the most appropriate hypothesis explaining the aetiology of these cysts is the "Lymph node inclusion theory" with he palatine tonsils as the most likely source of the enclosed epithelium.

Complete surgical removal remains the only acceptable form of treatment.

REFERENCES

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2. Bhasker SW, Bernier JL. Lympho-epithelial lesions of salivary glands. Cancer 1958; 11 : 1156-79.

3. Bhasker SW, Bernier JL. Histogenesis of branchial cysts - a report of 468 cases. American Journal of Pathology 1959; 407-14.

4. Hirota J, Maeda Y, Ueta E, Osaki T. Immunohistochemical and histological study of cervical lymphoepithelial cysts. Journal of Oral Pathological Medicine 1989; 18 : 202-5.

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6. Wiid G, Willie G, Mischke D. Lateral cervical cyst epithelia express upper digestive tract-type cytokeratins. Poly clonal antibody study. Ann Otol Rhinol Laryngol 1988; 97 (4) : Pt 1 : 364-72.

7. Stoll W, Huttenbrink KB. The lateral cervical cyst as a cystic lesion of a lymph node. Laryngol Rhinol Otol (Stuttg) 1982; 61 (5) : 272-5.

8. Hosemann W, Wigand ME. Are lateral neck cysts true derivatives of cervical lymph nodes? HNO 1988; 36 (4) : 140-46.

9. Vannineuse A, Dor P. Latero-cervico branchial Cysts and fistulas. Acta Otorhinolaryngol (Belg) 1976; 30 (3) : 299-307.

10. Stoll W. Lateral cysts and fistulae : two different cervical lesions. Laryngol Rhinol Otol (Stuttg) 1980; 59 (9) : 585-95.

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13. Howie AJ, Proops DW. The definition of branchial cysts, sinuses and fistulae. Clinical Otolaryngology 1952; 7 : 51-7.

14. Titchener GW, Allison RS. Lateral cervical cysts : a review of 42 cases. NZ Med J 1989; 102 (877) : 536-37.

15. Branchial apparatus and its abnormalities. In : Bailey and Loves’ Short Practice of Surgery : Chapman and Hall. 1996; 497.

16. Takita M, Hamaguchi H, Lin YT, Machiya T, Matsumoto R, Iwamoto M, Kawamoto T. Lymphoepithelial cyst of the upper neck : report of a case. Osaka Daigaku Shigaku Zasshi. 1989; 34 (2) : 431-37.

17. Buchner A, Hansen LS. Lymphoepithelial cysts of the oral cavity. A clinicopathologic study of 38 cases. Oral Surg Oral Med Oral Pathol 1980; 50 (5) : 441-49.

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21. Gnepp DR, Sporck FT. Benign lymphoepithelial parotid cyst with sebaceous differentiation-cystic sebaceous lymphadenoma. American Journal of Clinical Pathology 1980; 74 (5) : 683-87.

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24. Ryska A, Vokurka J, Michal M, Ludvikova M. Intrathyroidal lymphoepithelial cyst. A report of two cases not associated with Hashimoto’s thyroiditis. Pathol Res Pract 1997; 193 (11) : 777-81.

25. Tanaka H, Igarashi T, Teramoto S, Yoshida Y, Abe S. Lymphoepithelial cyst in the mediastinum with an opening in the trachea. Respiration 1995; 62 (2) : 110-13.

26. Yamamoto K, Fujimoto K, Matsushiro T, Ota K. Lymphoepitheial cyst in the pancreas : a case report. Gastroenterol Jpn 1990; 25 (6) : 758-61.

27. Langlois NE, Krukowski ZH, Miller ID. Pancreatic tissue in a lateral cervical cyst attached to the thyroid gland - a presumed foregut remnant. Histopathology 1997; 31 (4) : 378-80.

28. Gosain AK, Wildes TO. Lateral cervical cyst containing gastric epithelium. Archives of Pathological Laboratory Medicine 1988; 112 : 96-98.

29. Tovi F, Zirkin H. Solitary lateral cervical cyst : presenting symptom of papillary thyroid adenocarcinoma. Ann Otol Rhinol Laryngol 1983; 92 (5) Pt 1 : 521-24.

30. Miller H, Andreassen UK. Lateral cervical cyst as a primary manifestation of tonsillar cancer. Ugeskr laeger 1992; 154 (50) : 3597-99.

31. Earl PD, Ward Booth RP. A case of branchial cyst, illustrating the value of ultrasound in the diagnosis of cervical swellings. Br J Oral Maxillofac Surg 1985; 23 (4) : 292-97.

32. Rosell Cervilla A, Raboso Garcia Baquero E, Onrubia Parra T, Martinez Vidal A. Lateral cervical branchial cysts : a retrospective study. Acta Otorrinolaringol Esp 1998; 49 (1) : 51-56.


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