Literature DB >> 29806077

Intrathecal baclofen in metachromatic leukodystrophy.

Nikki van der Veldt1, Diane F van Rappard2, Laura A van de Pol2, Marjo S van der Knaap2, Willem J R van Ouwerkerk3, Jules G Becher1, Nicole I Wolf2, Annemieke I Buizer1.   

Abstract

Metachromatic leukodystrophy (MLD) is a rare progressive neurological disorder, often accompanied by motor impairments that are challenging to treat. In this case series, we report the course of treatment with intrathecal baclofen (ITB), aimed at improving daily care and comfort in children and young adults with MLD. All patients with MLD in our centre on ITB treatment for a minimum of 6 months were included (n=10; 4 males, 6 females; mean age 10y 8mo [range 6-24y]). Eight patients had MLD with a predominant spastic movement disorder (sMLD) and two were mainly dyskinetic. Patients with sMLD were compared with matched patients with spastic cerebral palsy (CP). Complication rates related to ITB treatment were similar in both groups. ITB treatment course in the first 6 months after pump implantation appears to show more dose increase in most patients MLD, compared to patients with spastic CP. This may be due to the progressive disease in MLD. ITB is a feasible therapy to improve daily care and comfort in patients with MLD and should therefore be considered early. WHAT THIS PAPER ADDS: Intrathecal baclofen (ITB) is a feasible therapy to improve comfort and daily care in children and young people with metachromatic leukodystrophy (MLD). In the first 6 months of ITB treatment, MLD seems to show more dose increase compared to spastic cerebral palsy.
© 2018 The Authors. Developmental Medicine & Child Neurology published by John Wiley & Sons Ltd on behalf of Mac Keith Press.

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Year:  2018        PMID: 29806077     DOI: 10.1111/dmcn.13919

Source DB:  PubMed          Journal:  Dev Med Child Neurol        ISSN: 0012-1622            Impact factor:   5.449


  4 in total

Review 1.  Peripheral neuropathy in metachromatic leukodystrophy: current status and future perspective.

Authors:  Shanice Beerepoot; Stefan Nierkens; Jaap Jan Boelens; Caroline Lindemans; Marianna Bugiani; Nicole I Wolf
Journal:  Orphanet J Rare Dis       Date:  2019-11-04       Impact factor: 4.123

Review 2.  A Proposed Diagnostic Algorithm for Inborn Errors of Metabolism Presenting With Movements Disorders.

Authors:  Juan Darío Ortigoza-Escobar
Journal:  Front Neurol       Date:  2020-11-13       Impact factor: 4.003

3.  Modified Delphi procedure-based expert consensus on endpoints for an international disease registry for Metachromatic Leukodystrophy: The European Metachromatic Leukodystrophy initiative (MLDi).

Authors:  Daphne H Schoenmakers; Shanice Beerepoot; Sibren van den Berg; Laura Adang; Annette Bley; Jaap-Jan Boelens; Francesca Fumagalli; Wim G Goettsch; Sabine Grønborg; Samuel Groeschel; Peter M van Hasselt; Carla E M Hollak; Caroline Lindemans; Fanny Mochel; Peter G M Mol; Caroline Sevin; Ayelet Zerem; Ludger Schöls; Nicole I Wolf
Journal:  Orphanet J Rare Dis       Date:  2022-02-14       Impact factor: 4.123

Review 4.  Metachromatic Leukodystrophy: Diagnosis, Modeling, and Treatment Approaches.

Authors:  Alisa A Shaimardanova; Daria S Chulpanova; Valeriya V Solovyeva; Aysilu I Mullagulova; Kristina V Kitaeva; Cinzia Allegrucci; Albert A Rizvanov
Journal:  Front Med (Lausanne)       Date:  2020-10-20
  4 in total

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