Literature DB >> 20857844

Anemia and neutropenic fever with high dose diazoxide treatment in a case with hyperinsulinism due to Munchausen by proxy.

A Ozon1, H Demirbilek, A Ertugrul, S Unal, F Gumruk, N Kandemir.   

Abstract

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.

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Year:  2010        PMID: 20857844     DOI: 10.1515/jpem.2010.23.7.719

Source DB:  PubMed          Journal:  J Pediatr Endocrinol Metab        ISSN: 0334-018X            Impact factor:   1.634


  4 in total

1.  Diazoxide-Induced Neutropenia and Long-Term Follow-up in a Patient with Hyperinsulinemia-Hyperammonemia due to GLUD1 Mutation.

Authors:  D Vuralli; S Aytac Eyupoglu; N Kandemir; A Ozon; N Gonc; A Alikasifoglu
Journal:  Acta Endocrinol (Buchar)       Date:  2021 Jul-Sep       Impact factor: 0.877

Review 2.  Munchausen by proxy syndrome mimicking systemic autoinflammatory disease: case report and review of the literature.

Authors:  Helmut Wittkowski; Claas Hinze; Sigrid Häfner-Harms; Vinzenz Oji; Katja Masjosthusmann; Martina Monninger; Ulrike Grenzebach; Dirk Foell
Journal:  Pediatr Rheumatol Online J       Date:  2017-04-05       Impact factor: 3.054

Review 3.  Long-term medical treatment in congenital hyperinsulinism: a descriptive analysis in a large cohort of patients from different clinical centers.

Authors:  Alena Welters; Christian Lerch; Sebastian Kummer; Jan Marquard; Burak Salgin; Ertan Mayatepek; Thomas Meissner
Journal:  Orphanet J Rare Dis       Date:  2015-11-25       Impact factor: 4.123

Review 4.  Hyperinsulinemic hypoglycemia in children and adolescents: Recent advances in understanding of pathophysiology and management.

Authors:  Maria Gϋemes; Sofia Asim Rahman; Ritika R Kapoor; Sarah Flanagan; Jayne A L Houghton; Shivani Misra; Nick Oliver; Mehul Tulsidas Dattani; Pratik Shah
Journal:  Rev Endocr Metab Disord       Date:  2020-12       Impact factor: 6.514

  4 in total

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