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Second Branchial Cleft Cyst and Fistula

Burton M. Gold1

Cysts and fistulous tracts of branchial origin are unusual while the internal opening was not radiologically demon-
entities seldom seen by radiologists. While this entity has strated, it was clear that the patient had a complete fistuba
been described in the surgical and medical literature, this because she was able to taste the contrast material intro-
is, to the author’s knowledge, only the second case of a duced during the fistubograrn.
fistula from the second branchial cleft reported in the English In a series of 239 branchiab apparatus anomalies reported
radiobogic literature. by Neel and Pernberton [1 ], the ratio of cysts to fistulas and
sinuses was 2 : 1 . Among the fistubas and sinuses, 50% were
external-draining sinuses, 39% ware complete fistubas, and
Case Report
1 1 % were internal-draining fistulas. Cysts, fistulas, and si-
M. V., a 1 9-year-old white woman, was admitted to The New nuses of the second branchial cleft are definitely the most
York Hospital with a history of bilateral hearing loss, a small mass common [2, 3]. While fistubograrns ofthe second cleft fistubas
on the left side of the neck, and intermittent drainage from a site on have been demonstrated in the surgical literature [2, 4-6],
the right side of the neck since early childhood. Physical examina- no examples were shown in the radiologic literature until the
tion revealed small bilateral preauricular cysts, a 2 x i cm cystic recent report by Agbezudor [7].
mass overlying the anterior border of the left sternocleidomastoid
There has been considerable controversy regarding the
muscle, a draining fistula opening at the anterior border of the right
four theories of origin of branchial anomalies [8], and a
sternocleidomastoid muscle, a deviated nasal septum, bilateral
detailed description of the embryology is beyond the scope
sensorineural hearing loss, a retrognathic mandible, and a high
arched palate. of this article. Anatomically [2-4, 6], the typical second
Radiographic examinations revealed a shorter than normal hard branchial cleft fistula has its external opening at the anterior
palate, abnormal development of the mallaus and incus on the right, border of the junction of the middle and lower thirds of the
bilateral cervical ribs, and hypoplasia of the left second rib. A small sternoclaidornastoid muscle. The tract than runs deep to the
pediatric urethral catheter was introduced into the fistula on the platysrna muscles, ascends along the carotid sheath (deep
right side of the neck with partial inflation of the balloon, and a few to the external and superficial to the internal carotid artery),
milliliters of water-soluble contrast material was injected. Antero- and crosses over the hypoglossal and gbossopharyngeal
posterior and lateral radiographs (fig. 1) showed a fistulous tract
nerves and stylopharyngeus muscle. It then extends upward
which went medially and cephalad to the tonsillar region. There was
along the posterior aspect of the tonsil to the posterior
also an area of cystic dilatation just deep to the external orifice.
palatine arch and ends in the upper half of the posterior
During the study, the patient had a slight choking sensation and felt
that she could taste the contrast material, suggesting drainage into faucial pillar, the supratonsillar fossa, or directly on the
the pharynx at the base of the tongue. The study was believed to tonsillar surface. Among branchial cleft cysts, 64% are
be diagnostic of a cyst and fistula of the second right brachial cleft. found in the upper third of the neck anterior to the sterno-
At surgery both praauricular cysts (first branchial cleft cysts), the cleidornastoid muscle, and the remainder are found in the
left second branchial cleft cyst, and the right second branchial cleft middle and lower thirds ofthe neck, the parotid, the pharynx,
cyst and fistula were removed. Surgery was aided by intraoperative and the posterior triangle of the neck [8].
injection of methylene blue and insertion of a small catheter into the Fistulas of branchial cleft origin are almost always present
fistulous tract. Pathologic examination was confirmatory.
at birth with a small pinpoint eternab opening (although they
may go unnoticed for years if there is no drainage). Some
patients also have conductive and sensorinaural deafness
Discussion
as well as other anomalies of the first and second branchial
Developmental anomalies arising from the branchiab ap- arch derivatives (as illustrated by this case) [9]. The fistubas
paratus include cysts, external sinuses, internal sinuses, may be unilateral or bilateral, and have been found in several
and complete fistulas. Cysts may exist independently or members of the same family [3, 4, 8, 9]. Symptoms usually
anywhere along the course of a sinus or fistuba. In this case, consist of continuous or intermittent mucoid drainage and

Received July 27, 1 979; accepted after revision December 1 3, 1979.

‘Department of Radiology, State University of New York, Stony Brook, NY 1 1 794.
AJR 134:1067-1069, May I 980 036 1 -803x/80/ 1 345- 1067 $00.00 © American Roentgen Ray Society
 1068 CASE REPORTS AJR:134, May 1980

Fig. 1 .-Anteropostenior (A) lateral

(B) views of contrast injection of second
right brachial cleft fistula. Cystic area
just deep to external orifice.
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recurrent attacks of inflammation that often follow an upper A sinograrn or fistulogram may be obtained to confirm the
respiratory infection or mild trauma. Frank cellulitis as well clinical diagnosis and is especially useful to show the length
as abscess formation may occasionally occur and necessi- and location of the tract as well as the possible presence of
tate incision and drainage. With very large fistulas, ingested an associated cyst. The tract may be injected with either
material may regurgitate through the tract [2-4]. The exter- oily or water-soluble contrast medium. The injection can be
nal opening can be seen to rise with deglutition when a made using a urethral catheter held in place by its own
complete fistula is present [4]. Occasionally, a cordlike balloon (as in this case), by a purse-string suture [3, 4], or
structure extending upward from the external orifice may be by a suction technique [7]. It may also be injected with a
palpable [3, 5]. Probing the tract sometimes produces symp- fine-nozzled syringe. Radiographs reveal a smoothly mar-
toms of coughing, palpitations, pallor, and vomiting because ginated tract of variable width following the anatomic path
of the tract’s proximity to the vagus nerve [2-4]. Pathobogi- described above. In contrast, the walls of a tubercubous
cally, the fistula lining consists of squamous and/or col- fistula are very irregular [2, 4]. The length of the visualized
umnar epithelium [3, 4]. tract is dependent on whether a sinus or fistula is present,
Cysts deriving from the branchiab clefts make their first as well as on the filming technique and pressure and volume
appearance later in life with the peak incidence during the of contrast material injected. Aspiration of cyst contents and
third decade [8]. They range from 1 to 1 0 cm in diameter injection of radiopaque contrast medium has been used to
[9], with 60% found in males and 40% in females [8]. About aid the preoperative diagnosis of branchial cleft cysts
2% are bilateral; two-thirds are found on the left and one [10, 11].
third on the right [8]. The most common symptoms are If the fistula is asymptomatic, surgery is usually not mdi-
continuous swelling (80%), pain (30%), intermittent swelling cated [4]. However, most cases are symptomatic and sur-
(20%), infection (1 5%), and pressure symptoms (7%) [8]. gery is done to avoid the morbidity of recurrent infections
On palpation, 70% are cystic and 30% are solid [8]. Sudden as well as the cosmetic problem of a draining sinus or fistula
appearance or enlargement is often associated with upper [9]. Sclerosing solutions have been used for obliteration of
respiratory tract infections or trauma [2, 4, 5]. The differ- cysts and fistulas to avoid scar formation. However, these
ential diagnosis includes inflammatory and neoplastic solutions are rarely used now because of the danger of
lymphadenopathy, thyroid nodule, parotid tumor, carotid marked inflammatory reaction and necrosis with perforation
body tumor, cystic hygroma, neurofibroma, and Iipoma, as into the pharynx [2, 4]. Total surgical excision using a
well as other far less common entities [9]. Pathologically, stepladder technique with two
separate transverse incisions
the cysts are usually thin-walled and unibocular, with a lining is recommended for a definite cure [4]. Surgery is often
of squamous or columnar epithelium and occasional gran- facilitated by the preoperative injection of the tract with
ulation tissue or ectopic salivary tissue [9]. More than 90% methylene blue or paraffin, or by the insertion of a thin
have subepithelial lymphoid tissue [8]. catheter [3, 4]. Cysts should be excised to clarify the diag-
 AJR:134, May 1980 CASE REPORTS 1069

nosis [9], to prevent the recurrence of infection, and for 4. ProctorB. Lateral vestigial cysts and fistulas of the neck.
cosmetic reasons [5]. Surgery is best done when the cyst is Laryngoscope 1955;65 : 355-401
not infected and should consist of complete excision by an 5. McPhail N, Mustard RA. Branchial cleft anomalies: a review of
external or an appropriate intraoral approach [8]. Partial 87 cases treated at Toronto General Hospital. Can Med Assoc
J 1966;94:174-179
aspiration of the cyst contents may facilitate complete re-
6. Albers GD. Branchial anomalies. JAMA 1 963; 1 83 : 399-409
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moval [4].
7. Agbezudor P. A rare muco-cutaneous branchial fistulogram
using the McCallum technique. Radiography 1 976;42 : 221-
224
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DR. Branchial cysts, sinuses and fis-
tulae. Clin 1978;3 : 77-92
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1 . Neal HB, Pemberton J. Lateral cervical (branchial) cysts and 9. Deane SA, Telander AL. Surgery for thyrogbossal duct and
fistulas. Surgery 1945;1 8 : 267-286 branchial cleft anomalies. Am J Surg 1 978:1 36 : 348-353
2. Simpson RA. Lateral cervical cysts and fistulas. Laryngoscope 1 0. Newman J. Diagnosis and treatment of congenital cysts of the
1969;79: 30-59 neck. Eye Ear Nose Throat Monthly 1 966;45 : 43-46
3. Fitz-Hugh GS, Camp RJ. Branchial cysts and fistulas: report of 11. Wangensteen OH. Differentiation of branchial from other car-
an unusual case. South MedJ 1963;56:232-240 vical cysts by x-ray examination. Ann Surg I 931 93 : 790-792