Literature DB >> 18197404

Neonatal pyruvate dehydrogenase deficiency due to a R302H mutation in the PDHA1 gene: MRI findings.

João P Soares-Fernandes1, Roseli Teixeira-Gomes, Romeu Cruz, Manuel Ribeiro, Zita Magalhães, Jaime F Rocha, Lara M Leijser.   

Abstract

Pyruvate dehydrogenase (PDH) deficiency is one of the most common causes of congenital lactic acidosis. Correlations between the genetic defect and neuroimaging findings are lacking. We present conventional and diffusion-weighted MRI findings in a 7-day-old male neonate with PDH deficiency due to a mosaicism for the R302H mutation in the PDHA1 gene. Corpus callosum dysgenesis, widespread increased diffusion in the white matter, and bilateral subependymal cysts were the main features. Although confirmation of PDH deficiency depends on specialized biochemical analyses, neonatal MRI plays a role in evaluating the pattern and extent of brain damage, and potentially in early diagnosis and clinical decision making.

Entities:  

Mesh:

Substances:

Year:  2008        PMID: 18197404     DOI: 10.1007/s00247-007-0721-9

Source DB:  PubMed          Journal:  Pediatr Radiol        ISSN: 0301-0449


  8 in total

1.  A new case of pyruvate dehydrogenase deficiency due to a novel mutation in the PDX1 gene.

Authors:  Runu Dey; Manuele Mine; Isabelle Desguerre; Abdelhamid Slama; Loic Van Den Berghe; Michèle Brivet; Bernard Aral; Cécile Marsac
Journal:  Ann Neurol       Date:  2003-02       Impact factor: 10.422

Review 2.  Advanced MR techniques in the term-born neonate with perinatal brain injury.

Authors:  Mary A Rutherford; Phil Ward; Christina Malamateniou; Christine Malamatentiou
Journal:  Semin Fetal Neonatal Med       Date:  2005-10       Impact factor: 3.926

3.  [Pyruvate dehydrogenase deficit associated to the C515T mutation in exon 6 of the E1alpha gene].

Authors:  O Blanco-Barca; C Gomez-Lado; E Rodrigo-Saez; C Curros-Novos; P Briones-Godino; J Eiris-Punal; M Castro-Gago
Journal:  Rev Neurol       Date:  2006 Sep 16-30       Impact factor: 0.870

4.  Females with PDHA1 gene mutations: a diagnostic challenge.

Authors:  Marjolein Willemsen; Richard J T Rodenburg; Alexandra Teszas; Lambert van den Heuvel; Gyorgy Kosztolanyi; Eva Morava
Journal:  Mitochondrion       Date:  2006-05-19       Impact factor: 4.160

5.  Pathophysiology of oligodendroglial excitotoxicity.

Authors:  A Yoshioka; B Bacskai; D Pleasure
Journal:  J Neurosci Res       Date:  1996-11-15       Impact factor: 4.164

6.  In vivo pyruvate detected by MR spectroscopy in neonatal pyruvate dehydrogenase deficiency.

Authors:  Dina J Zand; Erin M Simon; Steven B Pulitzer; D J Wang; Z J Wang; Lucy B Rorke; Michael Palmieri; Gerard T Berry
Journal:  AJNR Am J Neuroradiol       Date:  2003-08       Impact factor: 3.825

7.  Pyruvate dehydrogenase E1alpha subunit deficiency in a female patient: evidence of antenatal origin of brain damage and possible etiology of infantile spasms.

Authors:  Naoko Wada; Toyojiro Matsuishi; Michiko Nonaka; Etsuo Naito; Makoto Yoshino
Journal:  Brain Dev       Date:  2004-01       Impact factor: 1.961

8.  Cranial ultrasound in metabolic disorders presenting in the neonatal period: characteristic features and comparison with MR imaging.

Authors:  L M Leijser; L S de Vries; M A Rutherford; A Y Manzur; F Groenendaal; T J de Koning; M van der Heide-Jalving; F M Cowan
Journal:  AJNR Am J Neuroradiol       Date:  2007-08       Impact factor: 3.825

  8 in total
  8 in total

Review 1.  The spectrum of pyruvate dehydrogenase complex deficiency: clinical, biochemical and genetic features in 371 patients.

Authors:  Kavi P Patel; Thomas W O'Brien; Sankarasubramon H Subramony; Jonathan Shuster; Peter W Stacpoole
Journal:  Mol Genet Metab       Date:  2011-10-07       Impact factor: 4.797

2.  The spectrum of pyruvate dehydrogenase complex deficiency: clinical, biochemical and genetic features in 371 patients.

Authors:  Kavi P Patel; Thomas W O'Brien; Sankarasubramon H Subramony; Jonathan Shuster; Peter W Stacpoole
Journal:  Mol Genet Metab       Date:  2012-07       Impact factor: 4.797

3.  MRI features of 4 female patients with pyruvate dehydrogenase E1 alpha deficiency.

Authors:  Nicholas Ah Mew; Johanna B Loewenstein; Nadja Kadom; Uta Lichter-Konecki; Andrea L Gropman; Jodie M Martin; Adeline Vanderver
Journal:  Pediatr Neurol       Date:  2011-07       Impact factor: 3.372

4.  Neonatal onset of mitochondrial disorders in 129 patients: clinical and laboratory characteristics and a new approach to diagnosis.

Authors:  Tomas Honzik; Marketa Tesarova; Martin Magner; Johannes Mayr; Pavel Jesina; Katerina Vesela; Laszlo Wenchich; Karol Szentivanyi; Hana Hansikova; Wolfgang Sperl; Jiri Zeman
Journal:  J Inherit Metab Dis       Date:  2012-01-10       Impact factor: 4.982

5.  Pyruvate dehydrogenase complex deficiency mimicking congenital cytomegalovirus infection on imaging.

Authors:  Jasmin Rahesh; Rohan Anand; Victor Mendiola; Roy Jacob
Journal:  Proc (Bayl Univ Med Cent)       Date:  2021-11-05

Review 6.  In Vivo NMR Studies of the Brain with Hereditary or Acquired Metabolic Disorders.

Authors:  Erica B Sherry; Phil Lee; In-Young Choi
Journal:  Neurochem Res       Date:  2015-11-26       Impact factor: 3.996

Review 7.  Neonatal neuroimaging findings in inborn errors of metabolism.

Authors:  Andrea Poretti; Susan I Blaser; Maarten H Lequin; Ali Fatemi; Avner Meoded; Frances J Northington; Eugen Boltshauser; Thierry A G M Huisman
Journal:  J Magn Reson Imaging       Date:  2012-05-07       Impact factor: 4.813

8.  Somatic mosaicism for a novel PDHA1 mutation in a male with severe pyruvate dehydrogenase complex deficiency.

Authors:  Kristin K Deeb; Jirair K Bedoyan; Raymond Wang; Leighann Sremba; Molly C Schroeder; George J Grahame; Monica Boyer; Shawn E McCandless; Douglas S Kerr; Shulin Zhang
Journal:  Mol Genet Metab Rep       Date:  2014-08-28
  8 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.