Literature DB >> 2608165

Focal changes in the globi pallidi associated with neurological dysfunction in methylmalonic acidaemia.

C de Sousa1, A T Piesowicz, E M Brett, J V Leonard.   

Abstract

Neurological abnormalities are common in patients with organic acidaemias and sometimes these are distinctive. Lesions in various parts of the brain are increasingly being recognised in patients with methylmalonic acidaemia. We present the cases of two patients with methylmalonic acidaemia who became acutely ill with marked metabolic acidosis. CT scan showed bilateral areas of low density involving the globi pallidi. One patient developed dystonic posturing of the upper and lower limbs with rigidity and bradykinesia following her recovery. Treatment with benzhexol and with levodopa and carbidopa had no sustained benefit. Her gait slowly improved but her dystonic posturing remained. The other patient developed marked truncal hypotonia and a variable increase in limb tone following the acute illness. These abnormalities have persisted. It is concluded that structural brain changes, and in particular symmetrical lesions in the globi pallidi, may follow acute illness in patients with methylmalonic acidaemia and be accompanied by neurological symptoms.

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Year:  1989        PMID: 2608165     DOI: 10.1055/s-2008-1071292

Source DB:  PubMed          Journal:  Neuropediatrics        ISSN: 0174-304X            Impact factor:   1.947


  18 in total

1.  Progressive neurological deterioration and MRI changes in cblC methylmalonic acidaemia treated with hydroxocobalamin.

Authors:  G M Enns; A J Barkovich; D S Rosenblatt; D R Fredrick; K Weisiger; C Ohnstad; S Packman
Journal:  J Inherit Metab Dis       Date:  1999-06       Impact factor: 4.982

2.  Selective death of immature neurons in methylmalonic acidemia of the neonate: a case report.

Authors:  J M Sum; J L Twiss; D S Horoupian
Journal:  Acta Neuropathol       Date:  1993       Impact factor: 17.088

Review 3.  Renal transplant in methylmalonic acidemia: could it be the best option? Report on a case at 10 years and review of the literature.

Authors:  Riccardo Lubrano; Marco Elli; Massimo Rossi; Elisabetta Travasso; Claudia Raggi; Paola Barsotti; Claudia Carducci; Pasquale Berloco
Journal:  Pediatr Nephrol       Date:  2007-03-31       Impact factor: 3.714

4.  Neurological outcome of methylmalonic acidaemia.

Authors:  P Nicolaides; J Leonard; R Surtees
Journal:  Arch Dis Child       Date:  1998-06       Impact factor: 3.791

5.  Inhibition of succinate dehydrogenase and beta-hydroxybutyrate dehydrogenase activities by methylmalonate in brain and liver of developing rats.

Authors:  J C Dutra; C S Dutra-Filho; S E Cardozo; C M Wannmacher; J J Sarkis; M Wajner
Journal:  J Inherit Metab Dis       Date:  1993       Impact factor: 4.982

6.  Use of human somatotrophin in the treatment of a patient with methylmalonic aciduria.

Authors:  M D Bain; S S Nussey; M Jones; R A Chalmers
Journal:  Eur J Pediatr       Date:  1995-10       Impact factor: 3.183

7.  Effect of propionic and methylmalonic acids on the high molecular weight neurofilament subunit (NF-H) in rat cerebral cortex.

Authors:  L Vivian; F Dall Bello Pessutto; L M Vieira de Almeida; S de Oliveira Loureiro; P de Lima Pelaez; C Funchal; M Wajner; R Pessoa-Pureur
Journal:  Neurochem Res       Date:  2002-12       Impact factor: 3.996

Review 8.  Methylmalonic acidemia: brain imaging findings in 52 children and a review of the literature.

Authors:  Alireza Radmanesh; Talieh Zaman; Hossein Ghanaati; Sanaz Molaei; Richard L Robertson; Amir A Zamani
Journal:  Pediatr Radiol       Date:  2008-07-18

Review 9.  Shunt surgery for early-onset severe hydrocephalus in methylmalonic acidemia: report on two cases and review of the literature.

Authors:  Minglei Ma; Mingxing Wu; Yanbin Li; Di Wu; Bingke Zhang
Journal:  Childs Nerv Syst       Date:  2018-02-27       Impact factor: 1.475

10.  Ganglioside alterations in the central nervous system of rats chronically injected with methylmalonic and propionic acids.

Authors:  V M T Trindade; A M Brusque; J R Raasch; L E Pettenuzzo; H P Rocha; C M D Wannmacher; M Wajne
Journal:  Metab Brain Dis       Date:  2002-06       Impact factor: 3.584

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