PMID- 23843578
OWN - NLM
STAT- MEDLINE
DCOM- 20140716
LR  - 20220309
IS  - 2191-0251 (Electronic)
IS  - 0334-018X (Print)
IS  - 0334-018X (Linking)
VI  - 26
IP  - 11-12
DP  - 2013
TI  - A novel atypical presentation of insulin autoimmune syndrome (Hirata's disease) 
      in a child.
PG  - 1163-6
LID - 10.1515/jpem-2013-0215 [doi]
AB  - Insulin autoimmune syndrome (IAS) or Hirata's disease is a rare cause of 
      hyperinsulinemic hypoglycemia. We report the case of a child with a mild, 
      atypical presentation of IAS. A previously healthy girl, aged 7 years old, 
      developed non-ketotic fasting hypoglycemia during treatment for pneumonia. 
      Laboratory evaluation during hypoglycemia showed the following results: serum 
      glucose, 32 mg/dL (1.8 mmol/L); insulin, 5.6 muIU/mL (38.9 pmol/L); C-peptide, 1.4 
      ng/mL (0.47 nmol/L); anti-insulin antibody, 6.2% (normal, <2.4%); absence of 
      ketonuria; and positive glucagon stimulation test result. Search for mutation in 
      genes ABCC8, KCNJ11, GLUD1 and MEN1 was negative. Human leukocyte antigen (HLA) 
      typing was HLA-DRB1*1104. Computed tomography scan of the abdomen showed a normal 
      result. The patient evolved with spontaneous resolution of the hypoglycemia, 
      within 30 days, with normalization of serum anti-insulin titers. The serum levels 
      of insulin and anti-insulin antibodies in the patient of this report were not 
      extremely high as previously reported. This novel, mild, or forme fruste 
      presentation of IAS expands the previously reported spectrum of this disease.
FAU - Alves, Cresio
AU  - Alves C
FAU - Constanca, Julia
AU  - Constanca J
FAU - De Leon, Diva D
AU  - De Leon DD
FAU - Snider, Kara
AU  - Snider K
FAU - Stanley, Charles
AU  - Stanley C
LA  - eng
GR  - R37 DK056268/DK/NIDDK NIH HHS/United States
PT  - Case Reports
PT  - Journal Article
PL  - Germany
TA  - J Pediatr Endocrinol Metab
JT  - Journal of pediatric endocrinology & metabolism : JPEM
JID - 9508900
RN  - 0 (Insulin Antibodies)
SB  - IM
MH  - Adolescent
MH  - Autoimmune Diseases/*diagnosis/immunology/physiopathology
MH  - Child
MH  - Child, Preschool
MH  - Female
MH  - Humans
MH  - Infant
MH  - Insulin Antibodies/*immunology
MH  - Male
PMC - PMC4361817
MID - NIHMS669306
EDAT- 2013/07/12 06:00
MHDA- 2014/07/17 06:00
PMCR- 2015/03/17
CRDT- 2013/07/12 06:00
PHST- 2013/03/28 00:00 [received]
PHST- 2013/06/03 00:00 [accepted]
PHST- 2013/07/12 06:00 [entrez]
PHST- 2013/07/12 06:00 [pubmed]
PHST- 2014/07/17 06:00 [medline]
PHST- 2015/03/17 00:00 [pmc-release]
AID - /j/jpem.ahead-of-print/jpem-2013-0215/jpem-2013-0215.xml [pii]
AID - 10.1515/jpem-2013-0215 [doi]
PST - ppublish
SO  - J Pediatr Endocrinol Metab. 2013;26(11-12):1163-6. doi: 10.1515/jpem-2013-0215.