• Media type: E-Article
  • Title: Excessive Seizure Clusters in an Otherwise Well-Controlled Epilepsy as a Possible Hallmark of Untreated Vitamin B6-Responsive Epilepsy due to a Homozygous PLPBP Missense Variant
  • Contributor: Johannsen, Jessika; Bierhals, Tatjana; Deindl, Philipp; Hecher, Laura; Hermann, Katharina; Hempel, Maja; Kloth, Katja; Denecke, Jonas
  • imprint: Georg Thieme Verlag KG, 2019
  • Published in: Journal of Pediatric Genetics
  • Language: English
  • DOI: 10.1055/s-0039-1685501
  • ISSN: 2146-4596; 2146-460X
  • Keywords: Genetics (clinical) ; Pediatrics, Perinatology and Child Health
  • Origination:
  • Footnote:
  • Description: <jats:title>Abstract</jats:title><jats:p>Recently, mutations in the PLPBP gene were described as a novel cause for vitamin B6-responsive epilepsy. We report the outcome in case of a male adolescent with a novel homozygous missense variant in PLPBP who was never treated with pyridoxine until the age of 16 years. He presented with only mild cognitive impairment and an early-onset, well-controlled epilepsy. In our patient, excessive seizure clusters and anxiety states occurred intermittently, suggesting that the combination might be a hallmark in untreated patients. Thus, mutations in PLPBP should be addressed even in adolescent patients with only mild learning disabilities and relatively good seizure control over the years.</jats:p>
  • Access State: Open Access