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AN UNUSUAL CASE OF BIPHASIC STRIDOR IN AN INFANT: SUPRASTERNAL

BRONCHOGENIC CYST

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ronchogenic cysts are congenital lesions resulting from ab-rrant budding of the embryonic foregut. Although they mayccur at any point along the tracheobronchial tree, symptom-tic suprasternal bronchogenic cysts are extremely rare.

A 7-month-old male was referred to the Otolaryngology–ead and Neck Surgery service with a presumptive diagnosis of

aryngomalacia based on a 5-month history of progressive stri-or, dysphagia, and poor weight gain. The stridor was biphasic,

ow-pitched, and associated with moderate tracheal tugging. Aound mass was seen to prolapse above the suprasternal notchnd left clavicular head during neck extension and crying. Flex-ble endoscopic laryngoscopy was unremarkable. A chest x-rayevealed a widened mediastinum with significant tracheal devi-tion to the right (Figure 1; available at www.jpeds.com). Aomputed tomography (CT) scan demonstrated a large homo-eneous cystic lesion originating from the posterior mediastinumFigure 2).

A 4.0 � 5.0 cm fluid-filled cyst was excised from theateral wall of the mid-trachea through a partial median sternot-my with cervical extension (Figure 3). Histopathology revealedcyst lined by pseudostratified ciliated columnar epithelium with

rregular fascicles of smooth muscle, focal islands of hyalineartilage, scattered seromucinous glands, and fibrovasculartroma (Figure 4; available at www.jpeds.com). These featuresere consistent with a diagnosis of bronchogenic cyst.

A history of progressive stridor, dysphagia, and failureo thrive is of concern and warrants an immediate airwaynvestigation. The biphasic nature of the stridor indicated a

igure 2. Axial CT scan showing a large unilocular, homogenous cystic

2esion originating from the posterior mediastinum.

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xed airway obstruction. This must be differentiated fromaryngomalacia, which is associated with an inspiratory, high-itched stridor. CT scans are diagnostic in more than half ofhe cases, but magnetic resonance imaging may be required tolucidate the cystic nature of the lesion.1 Surgical excision ishe definitive treatment to alleviate the airway obstruction andrevent possible infection and acute airway obstruction.2 Therognosis is excellent with surgical excision.

Philip Lai, MDLily H.P. Nguyen, MD, FRCSC

Peter C.W. Kim, MD, PhD, FRCSCPaolo Campisi, MD, FRCSC, FAAP

Department of Otolaryngology–Head and Neck SurgeryDepartment of Surgery

Hospital for Sick Children, Toronto, Ontario, Canada

REFERENCES. McAdams H, Kirejczyk

, Rosado-de-Christenson, Matsumoto S. Broncho-

enic cyst: imaging featuresith clinical and histopatho-

ogic correlation. Radiology000;217:441-46.. Ustundag E, Iseri M,eskin G, Yayla B, Muezzi-oglu B. Cervical broncho-enic cysts in the head andeck region. J Laryngol Otol

Reprint requests: Dr. Paolo Campisi, De-partment of Otolaryngology-Head andNeck Surgery, 555 University Avenue, 6thFloor, Elm Wing, Hospital for Sick Children,Toronto, Ontario, M5G 1X8, Canada. E-mail: philip.lai@utoronto.ca.

0022-3476/$ - see front matter

Copyright © 2006 Mosby Inc. All rightsreserved.

igure 3. Intraoperative view of the cystic lesion. (Available in color atww.jpeds.com.)

005;119:419-23.

10.1016/j.jpeds.2006.03.039

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igure 1. Posterior–anterior chest x-ray showing a widened mediastinumith marked tracheal deviation to the right.

igure 4. A, The cyst wall contains irregular fascicles of smooth muscle and focal islands of hyaline cartilage consistent with a bronchogenic cyst. B, The

yst is lined by pseudostratified ciliated columnar epithelium.

ai et al 424.e1