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Idiopathic hemiconvulsion-hemiplegia syndrome

MedGen UID:
639806
Concept ID:
C0549118
Disease or Syndrome
Synonym: Hemiplegia-hemiconvulsion-epilepsy syndrome
SNOMED CT: Hemiconvulsion-hemiplegia-epilepsy syndrome (230407006); H-H-E syndrome (230407006); Hemiplegia-hemiconvulsion-epilepsy syndrome (230407006)
 
Monarch Initiative: MONDO:0019485
Orphanet: ORPHA86908

Definition

A rare acute encephalopathy with inflammation-mediated status epilepticus characterized by infancy-onset of refractory unilateral, mainly clonic status epilepticus during or shortly after a febrile episode without evidence of central nervous system infection, followed by permanent or transient hemiplegia with a minimum duration of one week. The majority of children develop pharmaco-resistant epilepsy a few months later. Brain imaging shows edematous swelling of the affected hemisphere at the time of the initial status, followed by hemiatrophy that does not correlate with any vascular territory. [from ORDO]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • Idiopathic hemiconvulsion-hemiplegia syndrome
Follow this link to review classifications for Idiopathic hemiconvulsion-hemiplegia syndrome in Orphanet.

Recent clinical studies

Etiology

Barcia G, Desguerre I, Carmona O, Barnerias C, Chemaly N, Gitiaux C, Brunelle F, Dulac O, Boddaert N, Nabbout R
Dev Med Child Neurol 2013 Dec;55(12):1150-8. Epub 2013 Aug 8 doi: 10.1111/dmcn.12233. PMID: 23924283
Nabbout R, Vezzani A, Dulac O, Chiron C
Lancet Neurol 2011 Jan;10(1):99-108. doi: 10.1016/S1474-4422(10)70214-3. PMID: 21163447

Diagnosis

Nabbout R
Epilepsia 2013 Sep;54 Suppl 6:54-6. doi: 10.1111/epi.12278. PMID: 24001074
Barcia G, Desguerre I, Carmona O, Barnerias C, Chemaly N, Gitiaux C, Brunelle F, Dulac O, Boddaert N, Nabbout R
Dev Med Child Neurol 2013 Dec;55(12):1150-8. Epub 2013 Aug 8 doi: 10.1111/dmcn.12233. PMID: 23924283
Dulac O, Takahashi T
Handb Clin Neurol 2013;111:681-9. doi: 10.1016/B978-0-444-52891-9.00070-1. PMID: 23622215
Nabbout R
Epilepsia 2012 Sep;53 Suppl 4:58-62. doi: 10.1111/j.1528-1167.2012.03614.x. PMID: 22946722
Auvin S, Bellavoine V, Merdariu D, Delanoë C, Elmaleh-Bergés M, Gressens P, Boespflug-Tanguy O
Eur J Paediatr Neurol 2012 Sep;16(5):413-21. Epub 2012 Feb 14 doi: 10.1016/j.ejpn.2012.01.007. PMID: 22341151

Therapy

Nasir M, Jehangir M, Ahmed RN, Khan E, Ahmad A
J Pak Med Assoc 2018 Apr;68(4):656-659. PMID: 29808062
Shimakawa S, Nomura S, Ogino M, Fukui M, Kashiwagi M, Tanabe T, Tamai H
Brain Dev 2015 Aug;37(7):733-7. Epub 2014 Dec 1 doi: 10.1016/j.braindev.2014.11.003. PMID: 25476246
Barcia G, Desguerre I, Carmona O, Barnerias C, Chemaly N, Gitiaux C, Brunelle F, Dulac O, Boddaert N, Nabbout R
Dev Med Child Neurol 2013 Dec;55(12):1150-8. Epub 2013 Aug 8 doi: 10.1111/dmcn.12233. PMID: 23924283
Nabbout R
Epilepsia 2012 Sep;53 Suppl 4:58-62. doi: 10.1111/j.1528-1167.2012.03614.x. PMID: 22946722

Prognosis

Shimakawa S, Nomura S, Ogino M, Fukui M, Kashiwagi M, Tanabe T, Tamai H
Brain Dev 2015 Aug;37(7):733-7. Epub 2014 Dec 1 doi: 10.1016/j.braindev.2014.11.003. PMID: 25476246
Dulac O, Takahashi T
Handb Clin Neurol 2013;111:681-9. doi: 10.1016/B978-0-444-52891-9.00070-1. PMID: 23622215

Clinical prediction guides

Kim DW, Lim BC, Kim KJ, Chae JH, Lee R, Lee SK
Epilepsy Res 2013 Oct;106(3):440-5. Epub 2013 Aug 1 doi: 10.1016/j.eplepsyres.2013.06.012. PMID: 23916143
Nabbout R, Vezzani A, Dulac O, Chiron C
Lancet Neurol 2011 Jan;10(1):99-108. doi: 10.1016/S1474-4422(10)70214-3. PMID: 21163447

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