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Case Reports
. 2021 Feb 3;11(1):2045894020987117.
doi: 10.1177/2045894020987117. eCollection 2021 Jan-Mar.

Diazoxide-associated pulmonary hypertension in a patient with noncompaction cardiomyopathy

Rachel T Sullivan  1 , Kathryn A Tillman  1 , Steven J Kindel  1 , Stephanie S Handler  1
Affiliations
Case Reports

Diazoxide-associated pulmonary hypertension in a patient with noncompaction cardiomyopathy

Rachel T Sullivan et al. Pulm Circ. .

Abstract

Development of pulmonary hypertension after initiation of diazoxide for the treatment of neonatal hyperinsulinemic hypoglycemia is a rare, but previously described association. Risk factors for development of diazoxide-associated pulmonary hypertension include lower gestational age and congenital heart disease. This novel case report describes an infant with noncompaction cardiomyopathy who developed pulmonary hypertension shortly after initiation of diazoxide for hyperinsulinemic hypoglycemia which resolved upon cessation of the drug. This case highlights the benefit of having pre-treatment knowledge of underlying cardiac anatomy and makes a case for routine echocardiographic screening for neonates initiating diazoxide treatment.

Keywords: cardiomyopathy; pediatric cardiovascular disease; pulmonary hypertension.

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Figures

Fig. 1.
Fig. 1.
Panel (a) demonstrates Echocardiographic evidence of severe pulmonary hypertension after diazoxide initiation with severe interventricular septal flattening (a). Panel (b) shows Complete resolution of pulmonary hypertension within nine months of diazoxide discontinuation with normal interventricular septal configuration.
See this image and copyright information in PMC

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