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CASE REPORT
Korean J Pediatr 2012 January;55(1) :29-33.
doi:https://doi.org/10.3345/kjp.2012.55.1.29
PHACE association with intracranial, oropharyngeal hemangiomas, and an atypical patent ductus arteriosus arising from the tortuous left subclavian artery in a premature infant
Do-Hyun Kim (Kim DH), Jang Hwan Choi (Choi JH), Jung Ha Lee (Lee JH), Hee Sup Kim (Kim HS)
Department of Pediatrics, Dongguk University Ilsan Hospital, Goyang, Korea
Corresponding Author: Do-Hyun Kim ,Tel: +82-31-961-7184, Fax: +82-31-961-7182, Email: dayeong1@dumc.or.kr
Copyright © 2012 by The Korean Pediatric Society
ABSTRACT
PHACE association is a rare neurocutaneous condition in which facial hemangiomas associate with a spectrum of posterior fossa malformations, arterial cerebrovascular anomalies, cardiovascular anomalies, and eye anomalies. We reported a case of PHACE association in a premature infant showing facial, intracranial, and oropharyngeal hemangiomas with evidence of the Dandy-Walker variant and complicated cardiovascular anomalies, including a right-sided aortic arch and an atypical patent ductus arteriosus arising from a tortuous left subclavian artery. To our knowledge, intracranial hemangiomas are rare in PHACE association, and a concomitant oropharyngeal hemangioma has not been previously reported in the PHACE association literature. In infants presenting with large, plaque-like facial hemangiomas, it is important to conduct active cardiovascular and neurological evaluations. Special attention should be given to the laryngoscopic examination to search for additional hemangiomas in the airway.
Keywords: PHACE association | Central nervous system cavernous hemangioma | Patent ductus arteriosus | Premature infants
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