Literature DB >> 29666464

An adolescence-onset male leukoencephalopathy with remarkable cerebellar atrophy and novel compound heterozygous AARS2 gene mutations: a case report.

Qing Dong1, Ling Long1, Yan-Yu Chang1, Yan-Jun Lin1, Mei Liu1, Zheng-Qi Lu2.   

Abstract

Mutations in the mitochondrial alanyl-transfer (t)RNA synthetase 2 (AARS2; OMIM 612035) have been linked to leukoencephalopathy recently. Until now, there have been only 13 cases reported in the literature. Hence, the clinical and genetic characteristics of this disease are not fully understood. Here, we reported an adolescence-onset male leukoencephalopathy patient characterized by progressive limb tremor at the age of 17 years. He had no signs of a cardiomyopathy. Magnetic resonance imaging scanning demonstrated severe cerebellar atrophy and white matter abnormalities involving descending tracts. Focused exome sequencing revealed he had novel compound heterozygous mutations in AARS2 gene (c.2265dupA; p.Arg756fs and c.650C>T; p.Pro217Leu). The patient was diagnosed with AARS2 mutation-related leukodystrophy (AARS2-L). We report a case with novel AARS2 gene mutations with developed striking cerebellar atrophy and leukoencephalopathy, which helps to further understand the clinical and genetic heterogeneity of AARS2-L.

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Year:  2018        PMID: 29666464     DOI: 10.1038/s10038-018-0446-7

Source DB:  PubMed          Journal:  J Hum Genet        ISSN: 1434-5161            Impact factor:   3.172


  14 in total

Review 1.  Childhood ataxia with CNS hypomyelination/vanishing white matter disease--a common leukodystrophy caused by abnormal control of protein synthesis.

Authors:  Raphael Schiffmann; Orna Elroy-Stein
Journal:  Mol Genet Metab       Date:  2006-01-18       Impact factor: 4.797

Review 2.  Update on several/certain adult-onset genetic leukoencephalopathies: clinical signs and molecular confirmation.

Authors:  Ilaria Di Donato; Maria Teresa Dotti; Antonio Federico
Journal:  J Alzheimers Dis       Date:  2014       Impact factor: 4.472

3.  Novel AARS2 gene mutation producing leukodystrophy: a case report.

Authors:  Laszlo Szpisjak; Nora Zsindely; Jozsef I Engelhardt; Laszlo Vecsei; Gabor G Kovacs; Peter Klivenyi
Journal:  J Hum Genet       Date:  2016-10-13       Impact factor: 3.172

Review 4.  Cognitive dysfunction and symptoms of movement disorders in adult-onset leukoencephalopathy with axonal spheroids and pigmented glia.

Authors:  Takeshi Ikeuchi; Naomi Mezaki; Takeshi Miura
Journal:  Parkinsonism Relat Disord       Date:  2017-08-16       Impact factor: 4.891

5.  Exome sequencing identifies mitochondrial alanyl-tRNA synthetase mutations in infantile mitochondrial cardiomyopathy.

Authors:  Alexandra Götz; Henna Tyynismaa; Liliya Euro; Pekka Ellonen; Tuulia Hyötyläinen; Tiina Ojala; Riikka H Hämäläinen; Johanna Tommiska; Taneli Raivio; Matej Oresic; Riitta Karikoski; Outi Tammela; Kalle O J Simola; Anders Paetau; Tiina Tyni; Anu Suomalainen
Journal:  Am J Hum Genet       Date:  2011-05-05       Impact factor: 11.025

6.  The first Japanese case of leukodystrophy with ovarian failure arising from novel compound heterozygous AARS2 mutations.

Authors:  Mio Hamatani; Naoto Jingami; Yoshinori Tsurusaki; Shino Shimada; Keiko Shimojima; Megumi Asada-Utsugi; Kenji Yoshinaga; Norihito Uemura; Hirofumi Yamashita; Kengo Uemura; Ryosuke Takahashi; Naomichi Matsumoto; Toshiyuki Yamamoto
Journal:  J Hum Genet       Date:  2016-06-02       Impact factor: 3.172

7.  Adult-Onset Leukoencephalopathy with Axonal Spheroids and Pigmented Glia Caused by a Novel R782G Mutation in CSF1R.

Authors:  Nicola Foulds; Reuben J Pengelly; Simon R Hammans; James A R Nicoll; David W Ellison; Adam Ditchfield; Sarah Beck; Sarah Ennis
Journal:  Sci Rep       Date:  2015-05-15       Impact factor: 4.379

Review 8.  The Mitochondrial Aminoacyl tRNA Synthetases: Genes and Syndromes.

Authors:  Daria Diodato; Daniele Ghezzi; Valeria Tiranti
Journal:  Int J Cell Biol       Date:  2014-02-04

9.  Analysis of Mutations in AARS2 in a Series of CSF1R-Negative Patients With Adult-Onset Leukoencephalopathy With Axonal Spheroids and Pigmented Glia.

Authors:  David S Lynch; Wei Jia Zhang; Rahul Lakshmanan; Justin A Kinsella; Günes Altiokka Uzun; Merih Karbay; Zeynep Tüfekçioglu; Hasmet Hanagasi; Georgina Burke; Nicola Foulds; Simon R Hammans; Anupam Bhattacharjee; Heather Wilson; Matthew Adams; Mark Walker; James A R Nicoll; Jeremy Chataway; Nick Fox; Indran Davagnanam; Rahul Phadke; Henry Houlden
Journal:  JAMA Neurol       Date:  2016-12-01       Impact factor: 18.302

10.  Novel (ovario) leukodystrophy related to AARS2 mutations.

Authors:  Cristina Dallabona; Daria Diodato; Sietske H Kevelam; Tobias B Haack; Lee-Jun Wong; Gajja S Salomons; Enrico Baruffini; Laura Melchionda; Caterina Mariotti; Tim M Strom; Thomas Meitinger; Holger Prokisch; Kim Chapman; Alison Colley; Helena Rocha; Katrin Ounap; Raphael Schiffmann; Ettore Salsano; Mario Savoiardo; Eline M Hamilton; Truus E M Abbink; Nicole I Wolf; Ileana Ferrero; Costanza Lamperti; Massimo Zeviani; Adeline Vanderver; Daniele Ghezzi; Marjo S van der Knaap
Journal:  Neurology       Date:  2014-05-07       Impact factor: 9.910

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  9 in total

1.  AARS2-Related Leukodystrophy: a Case Report and Literature Review.

Authors:  Xiao Zhang; Jie Li; Yanyan Zhang; Meina Gao; Tao Peng; Tian Tian
Journal:  Cerebellum       Date:  2022-01-27       Impact factor: 3.847

Review 2.  The emerging neurological spectrum of AARS2-associated disorders.

Authors:  Sahyli Perez Parra; Stephan H Heckers; William R Wilcox; Colin David Mcknight; H A Jinnah
Journal:  Parkinsonism Relat Disord       Date:  2021-11-10       Impact factor: 4.891

Review 3.  The Primary Microglial Leukodystrophies: A Review.

Authors:  Isidro Ferrer
Journal:  Int J Mol Sci       Date:  2022-06-06       Impact factor: 6.208

4.  A novel compound heterozygous mutation in AARS2 gene (c.965 G > A, p.R322H; c.334 G > C, p.G112R) identified in a Chinese patient with leukodystrophy involved in brain and spinal cord.

Authors:  Chengyuan Song; Linliu Peng; Shengjun Wang; Yiming Liu
Journal:  J Hum Genet       Date:  2019-08-06       Impact factor: 3.172

Review 5.  Mitochondrial Aminoacyl-tRNA Synthetase and Disease: The Yeast Contribution for Functional Analysis of Novel Variants.

Authors:  Sonia Figuccia; Andrea Degiorgi; Camilla Ceccatelli Berti; Enrico Baruffini; Cristina Dallabona; Paola Goffrini
Journal:  Int J Mol Sci       Date:  2021-04-26       Impact factor: 5.923

6.  Novel Alanyl-tRNA Synthetase 2 Pathogenic Variants in Leukodystrophies.

Authors:  Xingao Wang; Qun Wang; Hefei Tang; Bin Chen; Xiang Dong; Songtao Niu; Shaowu Li; Yuzhi Shi; Wei Shan; Zaiqiang Zhang
Journal:  Front Neurol       Date:  2019-12-17       Impact factor: 4.003

7.  AARS2 leukoencephalopathy: A new variant of mitochondrial encephalomyopathy.

Authors:  Yi Tang; Qi Qin; Yi Xing; Dongmei Guo; Li Di; Jianping Jia
Journal:  Mol Genet Genomic Med       Date:  2019-01-31       Impact factor: 2.183

Review 8.  Mitochondrial aminoacyl-tRNA synthetase disorders: an emerging group of developmental disorders of myelination.

Authors:  Amena Smith Fine; Christina L Nemeth; Miriam L Kaufman; Ali Fatemi
Journal:  J Neurodev Disord       Date:  2019-12-16       Impact factor: 4.025

9.  Case report: 'AARS2 leukodystrophy'.

Authors:  Tobias Melton Axelsen; Tzvetelina Lubenova Vammen; Mads Bak; Nelsan Pourhadi; Christian Midtgaard Stenør; Sabine Grønborg
Journal:  Mol Genet Metab Rep       Date:  2021-07-13
  9 in total

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