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Winthrop-University Hospital Mineola, NY 11501
A 27-year-old-man presented to our institution for outpatient fluoroscopic evaluation, with a complaint of discharge from a small opening approximately 2 cm to the right and 2 cm superior to the sternal notch. The patient recalled having such symptoms since early childhood but was never given a specific diagnosis. At physical examination, no visible opening was seen, and only a tiny crusted papule was identified. The patient manually expressed a small amount of clear, mucoid material from this papule by milking the skin and subcutaneous tissues of the anterolateral aspect of the right neck in an inferomedial direction.
We cannulated this superficial pore with a 24-gauge pediatric IV catheter and secured the catheter with adhesive tape. A few milliliters of water-soluble contrast material was injected. Frontal and lateral radiographs obtained under fluoroscopic control revealed a fistulous tract originating from the skin surface near the origin of the sternocleidomastoid muscle, ascending deeply and medially into the soft tissues of the neck. The internal opening was identified in the pharynx (Figs. 2A, 2B), and the patient subsequently tasted the water-soluble contrast material. The findings were diagnostic of a second branchial cleft fistula.
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Congenital abnormalities of the branchial apparatus can result in various abnormal conditions in the neck, including a cyst (a small, partial malformation with no external opening), a sinus (a malformation with one superficial opening), or a fistula (a complete malformation with both internal and external communications). Ninety-five percent of abnormalities of the branchial cleft apparatus arise from the second cleft. At least 75% of all second branchial cleft abnormalities are cysts, which typically present when an individual is between 10 and 40 years old. Second branchial cleft fistulas and sinuses are less common and usually present during the first decade of life [1]. As was the case in our patient, individuals with a branchial cleft fistula typically present with persistent mucoid discharge from a skin pore [2].
Classically, the external opening of a second branchial cleft fistula is along the anterior margin of the sternocleidomastoid muscle at the junction of the middle and inferior thirds of the muscle. The tract initially ascends laterally to the common carotid artery but then courses medially between the external and internal carotid arteries and finally opens into the tonsillar fossa [3].
Our patient had experienced symptoms since early childhood, but the correct diagnosis was not considered until the patient reached adulthood and consulted an otolaryngologist. Although classically the external opening of such a fistula is found at the junction of the middle and inferior thirds of the sternocleidomastoid muscle, the external opening in our patient was in a more inferior location, closer to the sternal notch. Fluoroscopic imaging was especially helpful in establishing the correct diagnosis by revealing the characteristic internal opening of this type of fistula in the oropharynx.
Branchial cleft fistulas are a rare clinical entity, but proper diagnosis can be made by performing a simple fistulogram as we described. The definitive treatment for branchial cleft abnormalities, including fistulas, is surgical excision, with a wide cervicotomy as the incision method of choice [2]. Additional imaging with CT and MR imaging may also be helpful. However, initial fistulograms have been sufficient in most cases and may obviate any additional imaging [4].
References
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Radiology Quiz Case 1: Diagnosis Arch Otolaryngol Head Neck Surg, July 1, 2005; 131(7): 648 - 648. [Full Text] [PDF] |
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