Noncompaction cardiomyopathy in Hirschsprung's disease: a case report

S. D. Visona, G. Thiene, S. Mannarino, G. Corana, A. Osculati, A. Angelini, S. Rizzo

Research output: Contribution to journalArticle

Abstract

Noncompaction cardiomyopathy is a rare disorder, often associated with cardiac and noncardiac malformations. Hirschsprung's disease, a well-known aganglionosis, is associated with congenital heart diseases and has been reported to be due to impairment migration and differentiation of neural crest cells. Here, we present an 8-month-old male infant who died for cardiogenic shock after surgical resection of the involved bowel segment. The child was affected by both noncompaction cardiomyopathy and Hirschsprung's disease, two entities which can share a common neural crest-derived etiology.
Original languageEnglish
Pages (from-to)51-53
Number of pages3
JournalCardiovascular Pathology
Volume27
DOIs
Publication statusPublished - Mar 1 2017

Fingerprint

Hirschsprung Disease
Neural Crest
Cardiomyopathies
Cardiogenic Shock
Heart Diseases

Keywords

  • Cardiomyopathies/complications/genetics/pathology
  • Fatal Outcome
  • Hirschsprung Disease/complications/genetics/pathology
  • Humans
  • Infant
  • Infant, Newborn
  • Male
  • Hirschsprung's disease
  • Neural crest
  • Noncompaction cardiomyopathy

Cite this

Noncompaction cardiomyopathy in Hirschsprung's disease: a case report. / Visona, S. D.; Thiene, G.; Mannarino, S.; Corana, G.; Osculati, A.; Angelini, A.; Rizzo, S.

In: Cardiovascular Pathology, Vol. 27, 01.03.2017, p. 51-53.

Research output: Contribution to journalArticle

@article{f058d461ee4c4b1a867967005e3dacd9,
title = "Noncompaction cardiomyopathy in Hirschsprung's disease: a case report",
abstract = "Noncompaction cardiomyopathy is a rare disorder, often associated with cardiac and noncardiac malformations. Hirschsprung's disease, a well-known aganglionosis, is associated with congenital heart diseases and has been reported to be due to impairment migration and differentiation of neural crest cells. Here, we present an 8-month-old male infant who died for cardiogenic shock after surgical resection of the involved bowel segment. The child was affected by both noncompaction cardiomyopathy and Hirschsprung's disease, two entities which can share a common neural crest-derived etiology.",
keywords = "Cardiomyopathies/complications/genetics/pathology, Fatal Outcome, Hirschsprung Disease/complications/genetics/pathology, Humans, Infant, Infant, Newborn, Male, Hirschsprung's disease, Neural crest, Noncompaction cardiomyopathy",
author = "Visona, {S. D.} and G. Thiene and S. Mannarino and G. Corana and A. Osculati and A. Angelini and S. Rizzo",
note = "LR: 20170321; CI: Copyright (c) 2017; JID: 9212060; OTO: NOTNLM; 2016/08/26 00:00 [received]; 2016/11/11 00:00 [revised]; 2016/12/29 00:00 [accepted]; 2017/01/29 06:00 [pubmed]; 2017/03/23 06:00 [medline]; 2017/01/29 06:00 [entrez]; ppublish",
year = "2017",
month = "3",
day = "1",
doi = "S1054-8807(16)30095-3 [pii]",
language = "English",
volume = "27",
pages = "51--53",
journal = "Cardiovascular Pathology",
issn = "1054-8807",
publisher = "Elsevier Inc.",

}

TY - JOUR

T1 - Noncompaction cardiomyopathy in Hirschsprung's disease: a case report

AU - Visona, S. D.

AU - Thiene, G.

AU - Mannarino, S.

AU - Corana, G.

AU - Osculati, A.

AU - Angelini, A.

AU - Rizzo, S.

N1 - LR: 20170321; CI: Copyright (c) 2017; JID: 9212060; OTO: NOTNLM; 2016/08/26 00:00 [received]; 2016/11/11 00:00 [revised]; 2016/12/29 00:00 [accepted]; 2017/01/29 06:00 [pubmed]; 2017/03/23 06:00 [medline]; 2017/01/29 06:00 [entrez]; ppublish

PY - 2017/3/1

Y1 - 2017/3/1

N2 - Noncompaction cardiomyopathy is a rare disorder, often associated with cardiac and noncardiac malformations. Hirschsprung's disease, a well-known aganglionosis, is associated with congenital heart diseases and has been reported to be due to impairment migration and differentiation of neural crest cells. Here, we present an 8-month-old male infant who died for cardiogenic shock after surgical resection of the involved bowel segment. The child was affected by both noncompaction cardiomyopathy and Hirschsprung's disease, two entities which can share a common neural crest-derived etiology.

AB - Noncompaction cardiomyopathy is a rare disorder, often associated with cardiac and noncardiac malformations. Hirschsprung's disease, a well-known aganglionosis, is associated with congenital heart diseases and has been reported to be due to impairment migration and differentiation of neural crest cells. Here, we present an 8-month-old male infant who died for cardiogenic shock after surgical resection of the involved bowel segment. The child was affected by both noncompaction cardiomyopathy and Hirschsprung's disease, two entities which can share a common neural crest-derived etiology.

KW - Cardiomyopathies/complications/genetics/pathology

KW - Fatal Outcome

KW - Hirschsprung Disease/complications/genetics/pathology

KW - Humans

KW - Infant

KW - Infant, Newborn

KW - Male

KW - Hirschsprung's disease

KW - Neural crest

KW - Noncompaction cardiomyopathy

U2 - S1054-8807(16)30095-3 [pii]

DO - S1054-8807(16)30095-3 [pii]

M3 - Article

VL - 27

SP - 51

EP - 53

JO - Cardiovascular Pathology

JF - Cardiovascular Pathology

SN - 1054-8807

ER -