000 01875cam a2200265 4500500
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041 _afre
042 _adc
100 1 0 _aHe, Jing
_eauthor
700 1 0 _a Zhou, Wenjing
_eauthor
700 1 0 _a Shi, Jie
_eauthor
700 1 0 _a Zhang, Bingqing
_eauthor
700 1 0 _a Wang, Haixiang
_eauthor
245 0 0 _aA Chinese patient with epilepsy and WWOX compound heterozygous mutations
260 _c2020.
500 _a94
520 _aEarly infantile epileptic encephalopathy type 28 is a refractory epilepsy with early onset, poor prognosis, and hereditary causes. WW domain-containing oxidoreductase ( WWOX) gene mutation can result in epileptic encephalopathy, but the mechanism remains unclear. We present the case of a patient with epilepsy and WWOX compound heterozygous mutations. The seizures manifested as tonic-clonic, convulsive and were refractory to drugs. Magnetic resonance imaging showed a widened subarachnoid space and thin corpus callosum. The patient died from asphyxia at the age of one year and 23 days. Peripheral blood was taken from the patient and his parents, and whole-exome sequencing was investigated to determine possible gene mutation. Two compound heterozygous mutations were identified: c.172+1G>C (with no amino acid change) and c.984C>G (amino acid change: p.Tyr328Ter). The pathophysiology of epileptic encephalopathy related to the WWOX gene remains to be determined, and further studies are required to elucidate possible mechanisms.
690 _aWWOX gene
690 _acompound heterozygous mutations
690 _ainfantile epilepsy
690 _aWhole-exome sequencing
690 _aepileptic encephalopathy
786 0 _nEpileptic Disorders | Vol 22 | 1 | 2020-01-01 | p. 120-124 | 1294-9361
856 4 1 _uhttps://shs.cairn.info/revue-epileptic-disorders-2020-1-page-120?lang=en
999 _c246349
_d246349