Literature DB >> 23430529

Therapeutic efficacy of magnesium valproate in succinic semialdehyde dehydrogenase deficiency.

Elena Vanadia1, K Michael Gibson, Phillip L Pearl, Emanuele Trapolino, Salvatore Mangano, Francesca Vanadia.   

Abstract

Succinic semialdehyde dehydrogenase deficiency (SSADHD), a disorder of γ-aminobutyric acid (GABA) metabolism, manifests typically as a nonprogressive neurodevelopmental disorder with cognitive deficiency, neuropsychiatric morbidity and epilepsy. Therapy targets symptomatic seizures and neurobehavioral disturbances. We report an adolescent female with SSADHD whose unresponsiveness to a broad spectrum of antiepileptics was circumvented with magnesium valproate (MgVPA). Epilepsy remains well controlled in our patient, with concomitant improvements in behavioral symptoms and an absence of adverse symptoms. MgVPA intervention may have utility in SSADHD.

Entities:  

Year:  2012        PMID: 23430529      PMCID: PMC3565633          DOI: 10.1007/8904_2012_170

Source DB:  PubMed          Journal:  JIMD Rep        ISSN: 2192-8304


  22 in total

1.  Epilepsy in succinic semialdehyde dehydrogenase deficiency, a disorder of GABA metabolism.

Authors:  Phillip L Pearl; Lovy Shukla; William H Theodore; Cornelis Jakobs; K Michael Gibson
Journal:  Brain Dev       Date:  2011-06-12       Impact factor: 1.961

2.  Vigabatrin therapy in six patients with succinic semialdehyde dehydrogenase deficiency.

Authors:  K M Gibson; C Jakobs; H Ogier; L Hagenfeldt; K E Eeg-Olofsson; O Eeg-Olofsson; F Aksu; H P Weber; E Rossier; B Vollmer
Journal:  J Inherit Metab Dis       Date:  1995       Impact factor: 4.982

3.  Absence seizures in succinic semialdehyde dehydrogenase deficient mice: a model of juvenile absence epilepsy.

Authors:  M A Cortez; Y Wu; K M Gibson; O C Snead
Journal:  Pharmacol Biochem Behav       Date:  2004-11       Impact factor: 3.533

4.  Monitoring of 4-hydroxybutyric acid levels in body fluids during vigabatrin treatment in succinic semialdehyde dehydrogenase deficiency.

Authors:  Katrin Ergezinger; Reinhard Jeschke; Georg Frauendienst-Egger; Herbert Korall; K Michael Gibson; Volker H Schuster
Journal:  Ann Neurol       Date:  2003-11       Impact factor: 10.422

Review 5.  Gamma-hydroxybutyric acid: neurobiology and toxicology of a recreational drug.

Authors:  C Guin Ting Wong; Katherine F Y Chan; K Michael Gibson; O Carter Snead
Journal:  Toxicol Rev       Date:  2004

Review 6.  Vigabatrin and newer interventions in succinic semialdehyde dehydrogenase deficiency.

Authors:  Andrea Gropman
Journal:  Ann Neurol       Date:  2003       Impact factor: 10.422

Review 7.  Succinic semialdehyde dehydrogenase deficiency in children and adults.

Authors:  Phillip L Pearl; Edward J Novotny; Maria T Acosta; Cornelis Jakobs; K Michael Gibson
Journal:  Ann Neurol       Date:  2003       Impact factor: 10.422

8.  Clinical spectrum of succinic semialdehyde dehydrogenase deficiency.

Authors:  P L Pearl; K M Gibson; M T Acosta; L G Vezina; W H Theodore; M A Rogawski; E J Novotny; A Gropman; J A Conry; G T Berry; M Tuchman
Journal:  Neurology       Date:  2003-05-13       Impact factor: 9.910

9.  [Mechanism of action of an anticonvulsant, sodium dipropylacetate].

Authors:  S Simler; L Ciesielski; M Klein; S Gobaille; P Mandel
Journal:  C R Seances Soc Biol Fil       Date:  1981

10.  Mutational spectrum of the succinate semialdehyde dehydrogenase (ALDH5A1) gene and functional analysis of 27 novel disease-causing mutations in patients with SSADH deficiency.

Authors:  Shinjiro Akaboshi; Boris M Hogema; Andrea Novelletto; Patrizia Malaspina; Gajja S Salomons; George D Maropoulos; Cornelis Jakobs; Markus Grompe; K Michael Gibson
Journal:  Hum Mutat       Date:  2003-12       Impact factor: 4.878

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  8 in total

Review 1.  Thirty years beyond discovery--clinical trials in succinic semialdehyde dehydrogenase deficiency, a disorder of GABA metabolism.

Authors:  Kara R Vogel; Phillip L Pearl; William H Theodore; Robert C McCarter; Cornelis Jakobs; K Michael Gibson
Journal:  J Inherit Metab Dis       Date:  2012-06-28       Impact factor: 4.982

Review 2.  Inherited disorders of gamma-aminobutyric acid metabolism and advances in ALDH5A1 mutation identification.

Authors:  Phillip L Pearl; Mahsa Parviz; Kara Vogel; John Schreiber; William H Theodore; K Michael Gibson
Journal:  Dev Med Child Neurol       Date:  2014-12-29       Impact factor: 5.449

3.  Disorders of GABA metabolism: SSADH and GABA-transaminase deficiencies.

Authors:  Mahsa Parviz; Kara Vogel; K Michael Gibson; Phillip L Pearl
Journal:  J Pediatr Epilepsy       Date:  2014-11-25

Review 4.  Epilepsy in inherited neurotransmitter disorders: Spotlights on pathophysiology and clinical management.

Authors:  Mario Mastrangelo
Journal:  Metab Brain Dis       Date:  2020-10-23       Impact factor: 3.584

Review 5.  Succinic semialdehyde dehydrogenase deficiency (SSADHD): Pathophysiological complexity and multifactorial trait associations in a rare monogenic disorder of GABA metabolism.

Authors:  P Malaspina; J-B Roullet; P L Pearl; G R Ainslie; K R Vogel; K M Gibson
Journal:  Neurochem Int       Date:  2016-06-14       Impact factor: 3.921

6.  Metabolic Stroke: A Novel Presentation in a Child with Succinic Semialdehyde Dehydrogenase Deficiency.

Authors:  Sangeetha Yoganathan; Gautham Arunachal; Lisa Kratz; Mugil Varman; Maya Thomas; Sniya Valsa Sudhakar; Samuel Philip Oommen; Sumita Danda
Journal:  Ann Indian Acad Neurol       Date:  2020 Jan-Feb       Impact factor: 1.383

Review 7.  Understanding the Molecular Mechanisms of Succinic Semialdehyde Dehydrogenase Deficiency (SSADHD): Towards the Development of SSADH-Targeted Medicine.

Authors:  Henry H C Lee; Gabrielle E McGinty; Phillip L Pearl; Alexander Rotenberg
Journal:  Int J Mol Sci       Date:  2022-02-26       Impact factor: 5.923

Review 8.  Succinic Semialdehyde Dehydrogenase Deficiency: An Update.

Authors:  Miroslava Didiášová; Antje Banning; Heiko Brennenstuhl; Sabine Jung-Klawitter; Claudio Cinquemani; Thomas Opladen; Ritva Tikkanen
Journal:  Cells       Date:  2020-02-19       Impact factor: 6.600

  8 in total

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