Document Detail


Risk for PHACE syndrome in infants with large facial hemangiomas.
MedLine Citation:
PMID:  20643720     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
OBJECTIVES: This study was conducted to determine the prevalence of posterior fossae of the brain, arterial anomalies, cardiac anomalies, and eye anomalies (PHACE) in infants with large facial hemangiomas. The extracutaneous manifestations of PHACE may be associated with significant morbidity, and the prevalence of PHACE in patients with facial hemangiomas has not previously been reported. METHODS: A multicenter prospective study was conducted with 108 infants who had large facial hemangiomas and were systematically evaluated for manifestations of PHACE. The prevalence of PHACE and its extracutaneous manifestations in this cohort was calculated. The relationship between hemangioma distribution and the manifestations of PHACE was analyzed. RESULTS: Thirty-three (31%) of 108 had PHACE. Thirty of the 33 patients with PHACE had >1 extracutaneous finding. The risk for PHACE syndrome was higher in infants with larger hemangiomas and in those with hemangiomas that encompassed >1 facial segment. The most common extracutaneous anomalies observed in infants with PHACE were of the arteries of the cerebrovasculature (91%) and cardiac anomalies (67%). Upper face (frontotemporal and frontonasal) hemangiomas were commonly observed in infants with PHACE; isolated maxillary hemangiomas were rarely associated with PHACE. CONCLUSIONS: In infants with large facial hemangiomas, one-third have extracutaneous manifestations consistent with the diagnosis of PHACE syndrome, most commonly cerebrovascular and cardiovascular anomalies. The high prevalence of arterial anomalies in this cohort has implications for clinical management and future research regarding the pathophysiology of PHACE.
Authors:
Anita N Haggstrom; Maria C Garzon; Eulalia Baselga; Sarah L Chamlin; Ilona J Frieden; Kristen Holland; Sheilagh Maguiness; Anthony J Mancini; Catherine McCuaig; Denise W Metry; Kimberly Morel; Julie Powell; Susan M Perkins; Dawn Siegel; Beth A Drolet
Publication Detail:
Type:  Journal Article     Date:  2010-07-19
Journal Detail:
Title:  Pediatrics     Volume:  126     ISSN:  1098-4275     ISO Abbreviation:  Pediatrics     Publication Date:  2010 Aug 
Date Detail:
Created Date:  2010-08-03     Completed Date:  2010-09-02     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  0376422     Medline TA:  Pediatrics     Country:  United States    
Other Details:
Languages:  eng     Pagination:  e418-26     Citation Subset:  AIM; IM    
Affiliation:
Department of Dermatology and Pediatrics, Division of Biostatistics, Indiana University, Indianapolis, Indiana, USA. ahaggstr@iupui.edu
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MeSH Terms
Descriptor/Qualifier:
Abnormalities, Multiple / pathology
Airway Obstruction / complications
Cerebrovascular Disorders / complications*,  pathology*
Ear Diseases / complications*
Eye Diseases / complications*
Facial Neoplasms / complications*,  pathology*
Female
Gestational Age
Heart Defects, Congenital / complications*
Hemangioma / complications*,  pathology*
Humans
Infant
Infant, Newborn
Male
Neurocutaneous Syndromes / complications*,  pathology*
Prospective Studies
Syndrome

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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