An unusual case of biphasic stridor in an infant: Suprasternal bronchogenic cyst
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INSIGHTS AN UNUSUAL CASE OF BIPHASIC STRIDOR IN AN INFANT: SUPRASTERNAL BRONCHOGENIC CYST Bronchogenic cysts are congenital lesions resulting from ab- errant budding of the embryonic foregut. Although they may occur at any point along the tracheobronchial tree, symptom- atic suprasternal bronchogenic cysts are extremely rare. A 7-month-old male was referred to the Otolaryngology– Head and Neck Surgery service with a presumptive diagnosis of laryngomalacia based on a 5-month history of progressive stri- dor, dysphagia, and poor weight gain. The stridor was biphasic, low-pitched, and associated with moderate tracheal tugging. A round mass was seen to prolapse above the suprasternal notch and left clavicular head during neck extension and crying. Flex- ible endoscopic laryngoscopy was unremarkable. A chest x-ray revealed a widened mediastinum with significant tracheal devi- ation to the right (Figure 1; available at www.jpeds.com). A computed tomography (CT) scan demonstrated a large homo- geneous cystic lesion originating from the posterior mediastinum (Figure 2). A 4.0 5.0 cm fluid-filled cyst was excised from the lateral wall of the mid-trachea through a partial median sternot- omy with cervical extension (Figure 3). Histopathology revealed a cyst lined by pseudostratified ciliated columnar epithelium with irregular fascicles of smooth muscle, focal islands of hyaline cartilage, scattered seromucinous glands, and fibrovascular stroma (Figure 4; available at www.jpeds.com). These features were consistent with a diagnosis of bronchogenic cyst. A history of progressive stridor, dysphagia, and failure to thrive is of concern and warrants an immediate airway investigation. The biphasic nature of the stridor indicated a fixed airway obstruction. This must be differentiated from laryngomalacia, which is associated with an inspiratory, high- pitched stridor. CT scans are diagnostic in more than half of the cases, but magnetic resonance imaging may be required to elucidate the cystic nature of the lesion. 1 Surgical excision is the definitive treatment to alleviate the airway obstruction and prevent possible infection and acute airway obstruction. 2 The prognosis is excellent with surgical excision. Philip Lai, MD Lily H.P. Nguyen, MD, FRCSC Peter C.W. Kim, MD, PhD, FRCSC Paolo Campisi, MD, FRCSC, FAAP Department of Otolaryngology–Head and Neck Surgery Department of Surgery Hospital for Sick Children, Toronto, Ontario, Canada REFERENCES 1. McAdams H, Kirejczyk W, Rosado-de-Christenson M, Matsumoto S. Broncho- genic cyst: imaging features with clinical and histopatho- logic correlation. Radiology 2000;217:441-46. 2. Ustundag E, Iseri M, Keskin G, Yayla B, Muezzi- noglu B. Cervical broncho- genic cysts in the head and neck region. J Laryngol Otol 2005;119:419-23. Reprint requests: Dr. Paolo Campisi, De- partment of Otolaryngology-Head and Neck Surgery, 555 University Avenue, 6th Floor, Elm Wing, Hospital for Sick Children, Toronto, Ontario, M5G 1X8, Canada. E- mail: [email protected]. 0022-3476/$ - see front matter Copyright © 2006 Mosby Inc. All rights reserved. 10.1016/j.jpeds.2006.03.039 Figure 2. Axial CT scan showing a large unilocular, homogenous cystic lesion originating from the posterior mediastinum. Figure 3. Intraoperative view of the cystic lesion. (Available in color at www.jpeds.com.) 424
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Transcript of An unusual case of biphasic stridor in an infant: Suprasternal bronchogenic cyst
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INSIGHTS
AN UNUSUAL CASE OF BIPHASIC STRIDOR IN AN INFANT: SUPRASTERNAL
BRONCHOGENIC CYSTfilptetpp
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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.24
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: [email protected].
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
