Key result
Medical management including octreotide and cornstarch successfully treated 1 neonate with congenital hyperinsulinemic hypoglycaemia due to a homozygous ABCC8 mutation, avoiding surgery.
Case Report (n=1)
Highlights the role of genetic diagnosis and medical management in congenital hyperinsulinism to avoid surgery and prevent neurological sequelae.
Suggests medical options may avert surgery in congenital hyperinsulinism; leaves open need for prospective validation.
Hyperinsulinemic hypoglycaemia (HH) is a heterogeneous disorder causing persistent hypoketotic hypoglycaemia in neonates and infants. Congenital hyperinsulinism (CHI) is a rare cause of HH, resulting from inappropriate insulin secretion by pancreatic β-cells due to genetic defects in key genes, notably ABCC8 and KCNJ11, which encode the SUR1 and Kir6.2 components of the K ATP channels, respectively. We present a case of a neonate with congenital HH with persistent hypoglycaemia since birth, which was managed with high-dose glucose infusions, diazoxide and octreotide. A homozygous pathogenic missense variant, c4253G>A (p.Arg1418His) in Exon 35 of the ABCC8 gene was identified in the neonate, confirming CHI. Despite initial refractoriness to treatment, the infant responded to octreotide therapy, cornstarch and careful management with regular feeds and monitoring, avoiding the need for surgical intervention. This case underscores the critical role of genetic diagnosis and timely management in preventing long-term neurological sequelae.
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Prabha et al. (2025) conducted a case report in Congenital hyperinsulinemic hypoglycaemia (n=1). Medical management (glucose infusions, diazoxide, octreotide, cornstarch) was evaluated. Medical management including octreotide and cornstarch successfully treated 1 neonate with congenital hyperinsulinemic hypoglycaemia due to a homozygous ABCC8 mutation, avoiding surgery.
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