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Reversible left ventricular noncompaction caused by hypertensive hydrocephalus: a pediatric case report

BACKGROUND: Left ventricular noncompaction cardiomyopathy (LVNC) is characterized by prominent ventricular trabeculations on cardiovascular imaging. Acquired reversible LVNC has not been reported in pediatrics without a genetic background. CASE PRESENTATION: A 9-year-old girl with a ventriculoperito...

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Autores principales: Kato, Riko, Taneichi, Hiromichi, Takarada, Shinya, Okabe, Mako, Miyao, Nariaki, Nakaoka, Hideyuki, Ibuki, Keijiro, Ozawa, Sayaka, Adachi, Yuichi, Yoshimura, Naoki, Saito, Kazuyoshi, Ichida, Fukiko, Hirono, Keiichi
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8080348/
https://www.ncbi.nlm.nih.gov/pubmed/33910553
http://dx.doi.org/10.1186/s12887-021-02680-6
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author Kato, Riko
Taneichi, Hiromichi
Takarada, Shinya
Okabe, Mako
Miyao, Nariaki
Nakaoka, Hideyuki
Ibuki, Keijiro
Ozawa, Sayaka
Adachi, Yuichi
Yoshimura, Naoki
Saito, Kazuyoshi
Ichida, Fukiko
Hirono, Keiichi
author_facet Kato, Riko
Taneichi, Hiromichi
Takarada, Shinya
Okabe, Mako
Miyao, Nariaki
Nakaoka, Hideyuki
Ibuki, Keijiro
Ozawa, Sayaka
Adachi, Yuichi
Yoshimura, Naoki
Saito, Kazuyoshi
Ichida, Fukiko
Hirono, Keiichi
author_sort Kato, Riko
collection PubMed
description BACKGROUND: Left ventricular noncompaction cardiomyopathy (LVNC) is characterized by prominent ventricular trabeculations on cardiovascular imaging. Acquired reversible LVNC has not been reported in pediatrics without a genetic background. CASE PRESENTATION: A 9-year-old girl with a ventriculoperitoneal (VP) shunt for neonatal posthemorrhagic hydrocephalus was referred due to exacerbation of hydrocephalus caused by VP shunt dysfunction. Transthoracic echocardiography (TTE) revealed depressed left ventricular (LV) systolic function and thick prominent trabeculae in the LV, predominantly in the apex, suggesting LVNC. Following treatment with extraventricular drainage for hydrocephalus, prominent trabeculation of the LV was diminished on TTE within 3 months. Genetic testing using next-generation sequencing was performed, and no significant variants were identified. CONCLUSIONS: We revealed for the first time a pediatric case of reversible LVNC without genetic predisposition. This case report provides valuable information on the pathogenesis of acquired LVNC and suggests that detailed evaluation is required to elucidate the diagnosis of this wide spectrum of etiologic–pathogenetic disorders. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1186/s12887-021-02680-6.
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spelling pubmed-80803482021-04-29 Reversible left ventricular noncompaction caused by hypertensive hydrocephalus: a pediatric case report Kato, Riko Taneichi, Hiromichi Takarada, Shinya Okabe, Mako Miyao, Nariaki Nakaoka, Hideyuki Ibuki, Keijiro Ozawa, Sayaka Adachi, Yuichi Yoshimura, Naoki Saito, Kazuyoshi Ichida, Fukiko Hirono, Keiichi BMC Pediatr Case Report BACKGROUND: Left ventricular noncompaction cardiomyopathy (LVNC) is characterized by prominent ventricular trabeculations on cardiovascular imaging. Acquired reversible LVNC has not been reported in pediatrics without a genetic background. CASE PRESENTATION: A 9-year-old girl with a ventriculoperitoneal (VP) shunt for neonatal posthemorrhagic hydrocephalus was referred due to exacerbation of hydrocephalus caused by VP shunt dysfunction. Transthoracic echocardiography (TTE) revealed depressed left ventricular (LV) systolic function and thick prominent trabeculae in the LV, predominantly in the apex, suggesting LVNC. Following treatment with extraventricular drainage for hydrocephalus, prominent trabeculation of the LV was diminished on TTE within 3 months. Genetic testing using next-generation sequencing was performed, and no significant variants were identified. CONCLUSIONS: We revealed for the first time a pediatric case of reversible LVNC without genetic predisposition. This case report provides valuable information on the pathogenesis of acquired LVNC and suggests that detailed evaluation is required to elucidate the diagnosis of this wide spectrum of etiologic–pathogenetic disorders. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1186/s12887-021-02680-6. BioMed Central 2021-04-28 /pmc/articles/PMC8080348/ /pubmed/33910553 http://dx.doi.org/10.1186/s12887-021-02680-6 Text en © The Author(s) 2021 https://creativecommons.org/licenses/by/4.0/Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) . The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/ (https://creativecommons.org/publicdomain/zero/1.0/) ) applies to the data made available in this article, unless otherwise stated in a credit line to the data.
spellingShingle Case Report
Kato, Riko
Taneichi, Hiromichi
Takarada, Shinya
Okabe, Mako
Miyao, Nariaki
Nakaoka, Hideyuki
Ibuki, Keijiro
Ozawa, Sayaka
Adachi, Yuichi
Yoshimura, Naoki
Saito, Kazuyoshi
Ichida, Fukiko
Hirono, Keiichi
Reversible left ventricular noncompaction caused by hypertensive hydrocephalus: a pediatric case report
title Reversible left ventricular noncompaction caused by hypertensive hydrocephalus: a pediatric case report
title_full Reversible left ventricular noncompaction caused by hypertensive hydrocephalus: a pediatric case report
title_fullStr Reversible left ventricular noncompaction caused by hypertensive hydrocephalus: a pediatric case report
title_full_unstemmed Reversible left ventricular noncompaction caused by hypertensive hydrocephalus: a pediatric case report
title_short Reversible left ventricular noncompaction caused by hypertensive hydrocephalus: a pediatric case report
title_sort reversible left ventricular noncompaction caused by hypertensive hydrocephalus: a pediatric case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8080348/
https://www.ncbi.nlm.nih.gov/pubmed/33910553
http://dx.doi.org/10.1186/s12887-021-02680-6
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