PMID- 30862762
OWN - NLM
STAT- MEDLINE
DCOM- 20190805
LR  - 20190805
IS  - 2191-0251 (Electronic)
IS  - 0334-018X (Linking)
VI  - 32
IP  - 4
DP  - 2019 Apr 24
TI  - Pitfalls in the diagnosis of insulin autoimmune syndrome (Hirata's disease) in a 
      hypoglycemic child: a case report and review of the literature.
PG  - 421-428
LID - 10.1515/jpem-2018-0441 [doi]
AB  - Background Insulin autoimmune syndrome (IAS) is a rare cause of hyperinsulinemic 
      hypoglycemia (HH) not addressed as a potential differential diagnosis in current 
      pediatric guidelines. We present a case of IAS in a child with no previous 
      history of autoimmune disease, no previous intake of triggering medications and 
      absence of genetic predisposition. Case presentation A 6-year-old boy presented 
      with recurrent HH (blood glucose of 26 mg/dL [1.4 mmol/L] and insulin of 686 
      muU/mL). Abdominal imaging was normal. After multiple therapeutic failures, we 
      hypothesized misuse of exogenous insulin and factitious hypoglycemia. Council of 
      Guardianship had the child separated from his mother, but insulin levels remained 
      high. A chromatography test was then performed which showed high titers of 
      endogenous insulin autoantibody (IAA) with early dissociation from the insulin 
      molecule. The human leukocyte antigen (HLA) test showed a DRB1 *13:01/*08:02 
      genotype. The patient was advised to control food intake and physical activity 
      routines. During a 5-year follow-up, hypoglycemic episodes were sparse, despite 
      high insulin levels. Conclusions Misdiagnosis of IAS with factitious hypoglycemia 
      may happen if IAS is not considered as a differential diagnosis, leading to 
      potential traumatic consequences. Further efforts should be made to increase 
      awareness of IAS as a differential diagnosis of hypoglycemia and to include it in 
      pediatric guidelines.
FAU - Dos Santos, Tiago Jeronimo
AU  - Dos Santos TJ
AUID- ORCID: 0000-0001-9682-0289
AD  - Pediatric Endocrinology Unit, Instituto da Crianca, Hospital das Clinicas, 
      Faculdade de Medicina, Universidade de Sao Paulo, Sao Paulo/SP, Brazil.
FAU - Passone, Caroline Gouvea Buff
AU  - Passone CGB
AD  - Pediatric Endocrinology Unit, Instituto da Crianca, Hospital das Clinicas, 
      Faculdade de Medicina, Universidade de Sao Paulo, Sao Paulo/SP, Brazil.
FAU - Ybarra, Marina
AU  - Ybarra M
AD  - Pediatric Endocrinology Unit, Instituto da Crianca, Hospital das Clinicas, 
      Faculdade de Medicina, Universidade de Sao Paulo, Sao Paulo/SP, Brazil.
FAU - Ito, Simone Sakura
AU  - Ito SS
AD  - Pediatric Endocrinology Unit, Instituto da Crianca, Hospital das Clinicas, 
      Faculdade de Medicina, Universidade de Sao Paulo, Sao Paulo/SP, Brazil.
FAU - Teles, Milena Gurgel
AU  - Teles MG
AD  - Unidade de Diabetes/Unidade de Genetica (LIM/25), Disciplina de Endocrinologia, 
      Faculdade de Medicina, Universidade de Sao Paulo (USP), Sao Paulo/SP, Brazil.
AD  - Diabetes Center, Fleury Institute, Sao Paulo/SP, Brazil.
FAU - Manna, Thais Della
AU  - Manna TD
AD  - Pediatric Endocrinology Unit, Instituto da Crianca, Hospital das Clinicas, 
      Faculdade de Medicina, Universidade de Sao Paulo, Sao Paulo/SP, Brazil.
FAU - Damiani, Durval
AU  - Damiani D
AD  - Pediatric Endocrinology Unit, Instituto da Crianca, Hospital das Clinicas, 
      Faculdade de Medicina, Universidade de Sao Paulo, Sao Paulo/SP, Brazil.
LA  - eng
PT  - Case Reports
PT  - Journal Article
PT  - Review
PL  - Germany
TA  - J Pediatr Endocrinol Metab
JT  - Journal of pediatric endocrinology & metabolism : JPEM
JID - 9508900
RN  - 0 (Insulin)
RN  - 0 (Insulin Antibodies)
MH  - Autoimmune Diseases/blood/*diagnosis/etiology
MH  - Child
MH  - *Diagnostic Errors
MH  - Humans
MH  - Hypoglycemia/*complications
MH  - Insulin/administration & dosage/*blood
MH  - Insulin Antibodies/*blood/immunology
MH  - Male
MH  - Prognosis
OTO - NOTNLM
OT  - hyperinsulinemia
OT  - hypoglycemia
OT  - insulin antibody
OT  - insulin autoimmune syndrome
EDAT- 2019/03/14 06:00
MHDA- 2019/08/06 06:00
CRDT- 2019/03/14 06:00
PHST- 2018/10/17 00:00 [received]
PHST- 2019/01/29 00:00 [accepted]
PHST- 2019/03/14 06:00 [pubmed]
PHST- 2019/08/06 06:00 [medline]
PHST- 2019/03/14 06:00 [entrez]
AID - /j/jpem.ahead-of-print/jpem-2018-0441/jpem-2018-0441.xml [pii]
AID - 10.1515/jpem-2018-0441 [doi]
PST - ppublish
SO  - J Pediatr Endocrinol Metab. 2019 Apr 24;32(4):421-428. doi: 
      10.1515/jpem-2018-0441.