Anemia and Neutropenic Fever with High Dose Diazoxide Treatment in a Case with Hyperinsulinism Due to Munchausen by Proxy

作者：Ozon A^*; Demirbilek H; Ertugrul A; Unal S; Gumruk F; Kandemir N

来源：Journal of Pediatric Endocrinology & Metabolism, 2010, 23(7): 719-723.

• factitious hyperinsulinism
• Munchausen by proxy
• hypoglycemia
• diazoxide
• bone marrow suppression

Background: The etiology of hyperinsulinemic hypoglycemia in adolescents is similar to that of adults. Patients resistant to medical treatment may undergo pancreatectomy. Diazoxide is the mainstay of medical treatment. Rarely bone marrow suppression is reported due to diazoxide.
Patient: An adolescent with severe hyperinsulinemic hypoglycemia was referred for pancreatectomy after she was treated with high doses of diazoxide, octreotide and glucose. She developed anemia and febrile neutropenia in the course of diazoxide treatment that resolved with cessation of medication. The cause of the hyperinsulinemia proved to be classical Munchausen by proxy.
Conclusion: This is the first report of bone marrow suppression involving erythroid series by diazoxide. Follow-up of blood count may be considered in patients on high dosages since anemia may be dose dependent. Munchausen by proxy poses a serious threat to children with significant morbidity and mortality. Awareness and a high index of suspicion in clinical settings with unusual causes are the mainstay for the diagnosis.

• 出版日期2010-7