Diazoxide toxicity in a child with persistent hyperinsulinemic hypoglycemia of infancy: mixed hyperglycemic hyperosmolar coma and ketoacidosis.

Abstract

BACKGROUND:

Persistent hyperinsulinemic hypoglycemia of infancy (PHHI), also known as congenital hyperinsulinism, has been known to go into spontaneous remission, with patients developing diabetes in later life. A temporary phase of hyperglycemia is, however, rarely reported.

CASE PRESENTATION:

We describe a 16-month-old child, a known case of diazoxide responsive PHHI, presenting with mixed hyperglycemic hyperosmolar coma and ketoacidosis with rhabdomyolysis while on diazoxide treatment. The patient required temporary cessation of diazoxide and initiation of insulin infusion, followed by a relapse of hypoglycemia again necessitating diazoxide therapy.

CONCLUSIONS:

Hyperosmolar coma with ketoacidosis is a rare side-effect of diazoxide therapy, documented even in patients with persistent hyperinsulinemic hypoglycemia of infancy.

• Authors: Pragya Mangla, Khalid Hussain, Sian Ellard, Sarah E Flanagan, Vijayalakshmi Bhatia
• Journal: Journal of pediatric endocrinology & metabolism : JPEM (J Pediatr Endocrinol Metab) Vol. 31 Issue 8 Pg. 943-945 (Aug 28 2018) ISSN: 2191-0251 [Electronic] Germany
• PMID: 29958183 (Publication Type: Case Reports)

Chemical References

• Antihypertensive Agents
• Diazoxide

Topics

• Antihypertensive Agents (adverse effects)
• Congenital Hyperinsulinism (drug therapy)
• Diazoxide (adverse effects)
• Humans
• Hyperglycemic Hyperosmolar Nonketotic Coma (chemically induced, pathology)
• Infant
• Ketosis (chemically induced, pathology)
• Male
• Treatment Outcome