| Symptomatic liver involvement in neonatal hereditary hemorrhagic telangiectasia. | |
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MedLine Citation:
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PMID: 21536610 Owner: NLM Status: MEDLINE |
Abstract/OtherAbstract:
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High-flow hepatic vascular anomalies with arteriovenous shunting commonly manifest during the neonatal period with signs and symptoms of congestive heart failure, but to our knowledge, they have never been described in patients with hereditary hemorrhagic telangiectasia (HHT). We report here our experience with 3 patients with hepatic arteriovenous malformations (AVMs) who presented with symptoms of high-output congestive heart failure during the neonatal period and were subsequently diagnosed with HHT. Imaging showed large hypervascular lesions and multiple hepatic arteriovenous shunts that differentiated these lesions from liver hemangiomas. Transcatheter embolization was performed in all cases. One infant died of sepsis shortly after embolization; follow-up at the age of 2.5 years of the surviving infants revealed involution of the vascular lesions and no evidence of symptom recurrence. We conclude that severe symptoms related to hepatic AVMs in HHT can occur in the neonatal period and that HHT should therefore be included in the differential diagnosis of symptomatic neonatal hepatic vascular malformations. Imaging plays a key role in differentiating hepatic AVMs from hemangiomas, because the latter require additional pharmacologic treatments. Early transcatheter embolization seems to be effective, but long-term outcomes still need to be assessed. |
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Authors:
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Suhail Al-Saleh; Philip R John; Michelle Letarte; Marie E Faughnan; Jaques Belik; Felix Ratjen |
Publication Detail:
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Type: Case Reports; Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't Date: 2011-05-02 |
Journal Detail:
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Title: Pediatrics Volume: 127 ISSN: 1098-4275 ISO Abbreviation: Pediatrics Publication Date: 2011 Jun |
Date Detail:
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Created Date: 2011-06-02 Completed Date: 2011-08-09 Revised Date: 2012-09-19 |
Medline Journal Info:
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Nlm Unique ID: 0376422 Medline TA: Pediatrics Country: United States |
Other Details:
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Languages: eng Pagination: e1615-20 Citation Subset: AIM; IM |
Affiliation:
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University of Toronto, Toronto, Ontario, Canada. |
Export Citation:
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APA/MLA Format Download EndNote Download BibTex |
| MeSH Terms | |
Descriptor/Qualifier:
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Angiography Diagnosis, Differential Embolization, Therapeutic / methods Female Follow-Up Studies Humans Infant, Newborn Liver / blood supply*, radiography, ultrasonography Liver Diseases / diagnosis, etiology*, therapy Male Telangiectasia, Hereditary Hemorrhagic / complications* Vascular Malformations / complications*, diagnosis, therapy |
| Grant Support | |
ID/Acronym/Agency:
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//Canadian Institutes of Health Research |
| Comments/Corrections | |
From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine
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