Literature DB >> 11266018

Phenotype assignment in symptomatic female carriers of X-linked adrenoleukodystrophy.

S Schmidt1, F Träber, W Block, E Keller, C Pohl, J von Oertzen, H Schild, U Schlegel, T Klockgether.   

Abstract

However, only very few studies have systematically investigated the extent and distribution of nervous and endocrine system involvement in female carriers of X-ALD. To define the phenotype in symptomatic female carriers of X-ALD we performed a prospective study including eight symptomatic women who were followed for a mean period of 3.4+/-1.8 years (range 1-6) using standardized clinical examination protocols, magnetic resonance imaging and spectroscopy, evoked potential studies including visual, brainstem auditory, somatosensory and magnetic evoked potentials, neurographic recordings and endocrine studies. Spastic paraparesis and decreased vibration sense in the lower extremities were the most frequent clinical findings. Slightly hyperintense symmetric parieto-occipital white matter lesions on magnetic resonance imaging were detectable in two of seven cases, and the N-acetylaspartate/choline ratios on magnetic resonance spectroscopy were decreased in three of seven patients. P40 latencies were abnormal in all patients, and central motor conduction times to the lower extremities in seven of eight patients. Prolonged latencies of brainstem auditory evoked potential waves III-V or interpeak latencies of waves I-III, I-V and III-V were detectable in all patients. The degree of walking impairment was positively correlated with the duration of clinical disease (r=0.58, P < 0.05) and inversely correlated with the N-acetylaspartate/choline ratios (r=0.85; P < 0.05). Neurographic recordings revealed only subtle abnormalities, suggesting that nervous system involvement in symptomatic female carriers of X-ALD is confined mainly to the central nervous system. No evidence of adrenal insufficiency was detected in any of the patients.

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Year:  2001        PMID: 11266018     DOI: 10.1007/s004150170267

Source DB:  PubMed          Journal:  J Neurol        ISSN: 0340-5354            Impact factor:   4.849


  10 in total

1.  Heterozygous X-linked adrenoleukodystrophy-associated myelopathy mimicking primary progressive multiple sclerosis.

Authors:  Massimiliano Di Filippo; Elisa Luchetti; Paolo Prontera; Emilio Donti; Piero Floridi; Maria Di Gregorio; Nicola Tambasco; Paola Sarchielli; Paolo Calabresi
Journal:  J Neurol       Date:  2010-08-31       Impact factor: 4.849

2.  Granny trips down: is she carrying the big bad wolf?

Authors:  L Tremolizzo; M Patassini; G Uziel; B Castellotti; C Gellera; C Ferrarese; I Appollonio
Journal:  Neurol Sci       Date:  2012-05-17       Impact factor: 3.307

3.  Progression rate of myelopathy in X-linked adrenoleukodystrophy heterozygotes.

Authors:  Clarissa Troller Habekost; Fernanda Santos Pereira; Carmen Regla Vargas; Daniella Moura Coelho; Vitor Torrez; Jean Pierre Oses; Luis Valmor Portela; Pedro Schestatsky; Vitor Torres Felix; Ursula Matte; Vanessa Leotti Torman; Laura Bannach Jardim
Journal:  Metab Brain Dis       Date:  2015-04-30       Impact factor: 3.584

Review 4.  Adrenoleukodystrophy - neuroendocrine pathogenesis and redefinition of natural history.

Authors:  Stephan Kemp; Irene C Huffnagel; Gabor E Linthorst; Ronald J Wanders; Marc Engelen
Journal:  Nat Rev Endocrinol       Date:  2016-06-17       Impact factor: 43.330

5.  Preferential expression of mutant ABCD1 allele is common in adrenoleukodystrophy female carriers but unrelated to clinical symptoms.

Authors:  Ettore Salsano; Silvia Tabano; Silvia M Sirchia; Patrizia Colapietro; Barbara Castellotti; Cinzia Gellera; Marco Rimoldi; Viviana Pensato; Caterina Mariotti; Davide Pareyson; Monica Miozzo; Graziella Uziel
Journal:  Orphanet J Rare Dis       Date:  2012-01-26       Impact factor: 4.123

6.  Neurological impairment among heterozygote women for X-linked Adrenoleukodystrophy: a case control study on a clinical, neurophysiological and biochemical characteristics.

Authors:  Clarissa Troller Habekost; Pedro Schestatsky; Vitor Felix Torres; Daniella Moura de Coelho; Carmen Regla Vargas; Vitor Torrez; Jean Pierre Oses; Luis Valmor Portela; Fernanda dos Santos Pereira; Ursula Matte; Laura Bannach Jardim
Journal:  Orphanet J Rare Dis       Date:  2014-01-13       Impact factor: 4.123

7.  Disease progression in women with X-linked adrenoleukodystrophy is slow.

Authors:  Irene C Huffnagel; Marcel G W Dijkgraaf; Georges E Janssens; Michel van Weeghel; Björn M van Geel; Bwee Tien Poll-The; Stephan Kemp; Marc Engelen
Journal:  Orphanet J Rare Dis       Date:  2019-02-07       Impact factor: 4.123

8.  X-linked adrenoleukodystrophy caused by maternal ABCD1 mutation and paternal X chromosome inactivation.

Authors:  Zhen Li; Guangrui Lai
Journal:  Exp Ther Med       Date:  2022-07-12       Impact factor: 2.751

9.  A Large Family with p.Arg554His Mutation in ABCD1: Clinical Features and Genotype/Phenotype Correlation in Female Carriers.

Authors:  Rosa Campopiano; Cinzia Femiano; Maria Antonietta Chiaravalloti; Rosangela Ferese; Diego Centonze; Fabio Buttari; Stefania Zampatti; Mirco Fanelli; Stefano Amatori; Carmelo D'Alessio; Emiliano Giardina; Francesco Fornai; Francesca Biagioni; Marianna Storto; Stefano Gambardella
Journal:  Genes (Basel)       Date:  2021-05-19       Impact factor: 4.096

10.  Familial skewed x chromosome inactivation in adrenoleukodystrophy manifesting heterozygotes from a Chinese pedigree.

Authors:  Zhihong Wang; Aizhen Yan; Yuxiang Lin; Haihua Xie; Chunyan Zhou; Fenghua Lan
Journal:  PLoS One       Date:  2013-03-01       Impact factor: 3.240

  10 in total

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