Top

Published in:

Open Access 01-12-2021 | Transthoracic Echocardiography | Case report

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

Authors: Riko Kato, Hiromichi Taneichi, Shinya Takarada, Mako Okabe, Nariaki Miyao, Hideyuki Nakaoka, Keijiro Ibuki, Sayaka Ozawa, Yuichi Adachi, Naoki Yoshimura, Kazuyoshi Saito, Fukiko Ichida, Keiichi Hirono

Published in: BMC Pediatrics | Issue 1/2021

Abstract

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.

Available only for authorised users

Gati S, Papadakis M, Papamichael ND, Zaidi A, Sheikh N, Reed M, et al. Reversible de novo left ventricular trabeculations in pregnant women: implications for the diagnosis of left ventricular noncompaction in low-risk populations. Circulation. 2014;130(6):475–83. https://doi.org/10.1161/CIRCULATIONAHA.114.008554.CrossRefPubMed

De Rosa G, Pardeo M, Di Rocco C, et al. Neurogenic stunned myocardium presenting as left ventricular hypertrabeculation in childhood: a variant of Takotsubo cardiomyopathy? Pediatr Crit Care Med. 2011;12(6):e420–3. https://doi.org/10.1097/PCC.0b013e3182192944.CrossRefPubMed

Lindman BR, Stewart WJ, Pibarot P, Hahn RT, Otto CM, Xu K, et al. Early regression of severe left ventricular hypertrophy after transcatheter aortic valve replacement is associated with decreased hospitalizations. JACC Cardiovasc Interv. 2014;7(6):662–73. https://doi.org/10.1016/j.jcin.2014.02.011.CrossRefPubMedPubMedCentral

Akashi YJ, Nakazawa K, Sakakibara M, Miyake F, Musha H, Sasaka K. 123I-MIBG myocardial scintigraphy in patients with “takotsubo” cardiomyopathy. J Nucl Med. 2004;45(7):1121–7.PubMed

Nef HM, Mollmann H, Akashi YJ, Hamm CW. Mechanisms of stress (Takotsubo) cardiomyopathy. Nat Rev Cardiol. 2010;7(4):187–93. https://doi.org/10.1038/nrcardio.2010.16.CrossRefPubMed

Lyon AR, Rees PS, Prasad S, Poole-Wilson PA, Harding SE. Stress (Takotsubo) cardiomyopathy--a novel pathophysiological hypothesis to explain catecholamine-induced acute myocardial stunning. Nat Clin Pract Cardiovasc Med. 2008;5(1):22–9. https://doi.org/10.1038/ncpcardio1066.CrossRefPubMed

Akashi YJ, Anker SD. Takotsubo cardiomyopathy. Int J Cardiol. 2006;112(1):114–5. https://doi.org/10.1016/j.ijcard.2005.07.054.CrossRefPubMed

Neil CJ, Nguyen TH, Sverdlov AL, Chirkov YY, Chong CR, Stansborough J, et al. Can we make sense of takotsubo cardiomyopathy? An update on pathogenesis, diagnosis and natural history. Expert Rev Cardiovasc Ther. 2012;10(2):215–21. https://doi.org/10.1586/erc.11.190.CrossRefPubMed

Gati S, Papadakis M, Van Niekerk N, Reed M, Yeghen T, Sharma S. Increased left ventricular trabeculation in individuals with sickle cell anaemia: physiology or pathology? Int J Cardiol. 2013;168(2):1658–60. https://doi.org/10.1016/j.ijcard.2013.03.039.CrossRefPubMed

Title: Reversible left ventricular noncompaction caused by hypertensive hydrocephalus: a pediatric case report
Authors: Riko Kato
Hiromichi Taneichi
Shinya Takarada
Mako Okabe
Nariaki Miyao
Hideyuki Nakaoka
Keijiro Ibuki
Sayaka Ozawa
Yuichi Adachi
Naoki Yoshimura
Kazuyoshi Saito
Fukiko Ichida
Keiichi Hirono
Publication date: 01-12-2021
Publisher: BioMed Central
Keywords: Transthoracic Echocardiography
Cardiomyopathy
Hydrocephalus
Hydrocephalus
Electrocardiography
Hydrocephalus
Electrocardiography
Published in: BMC Pediatrics / Issue 1/2021
Electronic ISSN: 1471-2431
DOI: https://doi.org/10.1186/s12887-021-02680-6

Keynote webinar | Spotlight on medication adherence

Springer Medicine

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

Abstract

Background

Case presentation

Conclusions

Keynote webinar | Spotlight on medication adherence

Springer Medicine

Abstract

Background

Case presentation

Conclusions

Please log in to get access to this content

Other articles of this Issue 1/2021

Preoperative thyroid hormone levels predict ICU mortality after cardiopulmonary bypass in congenital heart disease patients younger than 3 months old

Effects of nutrition therapy on growth, inflammation and metabolism in immature infants: a study protocol of a double-blind randomized controlled trial (ImNuT)

Duplex kidney complicated with preoperative inferior nephroblastoma rupture in children: a case report and literature review

Facial cleft? The first case of manitoba‐oculo‐tricho‐anal syndrome with novel mutations in China: a case report

Physical activity and sedentary behaviors (screen time and homework) among overweight or obese adolescents: a cross-sectional observational study in Yazd, Iran

End-organ saturations correlate with aortic blood flow estimates by echocardiography in the extremely premature newborn – an observational cohort study