Refractory hypoglycemia in a pediatric patient with desmoplastic small round cell tumor
-
Abdullah Almaghraby
,
Wendy J. Brickman
Abstract
Background
Tumor-induced hypoglycemia is a rare and serious complication that is usually a consequence of either excessive insulin secretion (insulinoma) or because of non-islet cell tumor hypoglycemia (NICTH). NICTH is a rare phenomenon seen most often in adult patients. It is associated with different tumor types. Here, we report the first case to the best of our knowledge in the literature of a pediatric patient with NICTH associated with desmoplastic small round cell tumor (DSRT).
Case presentation
This is a 15-year-old girl who presented with symptomatic hypoglycemia and abdominal mass. She required an intravenous glucose infusion rate as high as 9 mg/kg/min in addition to glucose containing oral supplements in order to maintain her blood glucose above 60 mg/dL. Computed tomography (CT) scan of the chest, abdomen and pelvis showed multiple hepatic lesions with an intraperitoneal soft tissue mass which subsequently was diagnosed as DSRT. When the blood glucose was 45 mg/dL, the insulin, growth hormone (GH) and insulin-like growth factor-1 (IGF-1) levels were suppressed with an appropriate elevation of cortisol. Subsequently, an insulin-like growth factor-2 (IGF-2) level was sent and the IGF-2:IGF-1 ratio was found to be elevated >10 consistent with NICTH. After the first dose of chemotherapy, hypoglycemia improved, and she was weaned off glucose containing fluids.
Conclusions
NICTH should be considered in all cancer patients regardless of their age with refractory hypoglycemia.
Author contributions: Abdullah Almaghraby: Dr. Almaghraby took direct care of the patient, conducted the literature review, drafted the initial manuscript and edited the manuscript according to feedback of the other authors. Wendy J. Brickman: Dr. Brickman took direct care of the patient, provided feedback and reviewed and revised the manuscript. Jeffery A. Goldstein: Dr. Goldstein reviewed the histopathology to assist with confirmation of the diagnosis and created the histopathology figures and legends. Reema L. Habiby: Dr. Habiby took direct care of the patient, provided feedback and critically reviewed the manuscript. All the authors have accepted responsibility for the entire content of this submitted manuscript and approved submission.
Research funding: None declared.
Employment or leadership: None declared.
Honorarium: None declared.
Competing interests: The funding organization(s) played no role in the study design; in the collection, analysis, and interpretation of data; in the writing of the report; or in the decision to submit the report for publication.
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©2018 Walter de Gruyter GmbH, Berlin/Boston
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Artikel in diesem Heft
- Frontmatter
- Original Articles
- Urinary bisphenol-A levels in children with type 1 diabetes mellitus
- The relationship between metabolic syndrome, cytokines and physical activity in obese youth with and without Prader-Willi syndrome
- Association of anthropometric measures and cardio-metabolic risk factors in normal-weight children and adolescents: the CASPIAN-V study
- The effect of obesity and insulin resistance on macular choroidal thickness in a pediatric population as assessed by enhanced depth imaging optical coherence tomography
- Hereditary 1,25-dihydroxyvitamin D-resistant rickets (HVDRR): clinical heterogeneity and long-term efficacious management of eight patients from four unrelated Arab families with a loss of function VDR mutation
- Long-term thyroid disorders in pediatric survivors of hematopoietic stem cell transplantation after chemotherapy-only conditioning
- Screening for autoimmune thyroiditis and celiac disease in minority children with type 1 diabetes
- Resistance exercise alone improves muscle strength in growth hormone deficient males in the transition phase
- Sequential measurements of IGF-I serum concentrations in adolescents with Laron syndrome treated with recombinant human IGF-I (rhIGF-I)
- Symptomatic Rathke cleft cyst in paediatric patients – clinical presentations, surgical treatment and postoperative outcomes – an analysis of 38 cases
- Molecular genetics of tetrahydrobiopterin deficiency in Chinese patients
- Single center experience of biotinidase deficiency: 259 patients and six novel mutations
- Next-generation sequencing as a second-tier diagnostic test for newborn screening
- Case Reports
- Thyroid storm after choking
- Three new Brazilian cases of 17α-hydroxylase deficiency: clinical, molecular, hormonal, and treatment features
- Diazoxide toxicity in a child with persistent hyperinsulinemic hypoglycemia of infancy: mixed hyperglycemic hyperosmolar coma and ketoacidosis
- Refractory hypoglycemia in a pediatric patient with desmoplastic small round cell tumor
