Literature DB >> 2242177

Rett syndrome: findings suggesting axonopathy and mitochondrial abnormalities.

S Wakai1, K Kameda, Y Ishikawa, S Miyamoto, M Nagaoka, M Okabe, R Minami, N Tachi.   

Abstract

We report the histopathologic findings of 3 sural nerve biopsies and 1 muscle biopsy from 3 patients with Rett syndrome. The 3 sural nerve biopsies demonstrated a few ultrastructural abnormalities, including the presence of many Pi-granules and mitochondrial changes in the cytoplasm of Schwann cells, occasional bands of Büngner and onion-bulb formations, and mitochondrial alterations in myelinated axons. Morphometric analysis disclosed reduction in the number of large myelinated fibers with normal densities in comparison to those of an age-matched normal control. Light microscopic examination of the biopsied muscle from a 6-year-old patient with Rett syndrome revealed the existence of many small, dark, angulated fibers with NADH-TR staining. Ultrastructural investigation of the muscle confirmed the presence of the dumbbell-shaped mitochondria. Peripheral nerve involvement and the possibility of mitochondrial abnormalities in Rett syndrome were suggested by the results.

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Year:  1990        PMID: 2242177     DOI: 10.1016/0887-8994(90)90028-y

Source DB:  PubMed          Journal:  Pediatr Neurol        ISSN: 0887-8994            Impact factor:   3.372


  11 in total

1.  Oxidative stress in Rett syndrome: natural history, genotype, and variants.

Authors:  Silvia Leoncini; Claudio De Felice; Cinzia Signorini; Alessandra Pecorelli; Thierry Durand; Giuseppe Valacchi; Lucia Ciccoli; Joussef Hayek
Journal:  Redox Rep       Date:  2011       Impact factor: 4.412

2.  Isolated neurodevelopmental delay in childhood: clinicoradiological correlation in 170 patients.

Authors:  P Demaerel; D P Kingsley; B E Kendall
Journal:  Pediatr Radiol       Date:  1993

Review 3.  Does microglial dysfunction play a role in autism and Rett syndrome?

Authors:  Izumi Maezawa; Marco Calafiore; Heike Wulff; Lee-Way Jin
Journal:  Neuron Glia Biol       Date:  2012-04-30

4.  Aberrant mitochondrial function in patient-derived neural cells from CDKL5 deficiency disorder and Rett syndrome.

Authors:  Smita Jagtap; Jessica M Thanos; Ting Fu; Jennifer Wang; Jasmin Lalonde; Thomas O Dial; Ariel Feiglin; Jeffrey Chen; Isaac Kohane; Jeannie T Lee; Steven D Sheridan; Roy H Perlis
Journal:  Hum Mol Genet       Date:  2019-11-01       Impact factor: 6.150

5.  Dysregulation of glutamine transporter SNAT1 in Rett syndrome microglia: a mechanism for mitochondrial dysfunction and neurotoxicity.

Authors:  Lee-Way Jin; Makoto Horiuchi; Heike Wulff; Xiao-Bo Liu; Gino A Cortopassi; Jeffrey D Erickson; Izumi Maezawa
Journal:  J Neurosci       Date:  2015-02-11       Impact factor: 6.167

6.  Downstream targets of methyl CpG binding protein 2 and their abnormal expression in the frontal cortex of the human Rett syndrome brain.

Authors:  Joanne H Gibson; Barry Slobedman; Harikrishnan K N; Sarah L Williamson; Dimitri Minchenko; Assam El-Osta; Joshua L Stern; John Christodoulou
Journal:  BMC Neurosci       Date:  2010-04-26       Impact factor: 3.288

Review 7.  Rett syndrome: think outside the (skull) box.

Authors:  Emilie Borloz; Laurent Villard; Jean-Christophe Roux
Journal:  Fac Rev       Date:  2021-06-29

8.  Genes related to mitochondrial functions, protein degradation, and chromatin folding are differentially expressed in lymphomonocytes of Rett syndrome patients.

Authors:  Alessandra Pecorelli; Guido Leoni; Franco Cervellati; Raffaella Canali; Cinzia Signorini; Silvia Leoncini; Alessio Cortelazzo; Claudio De Felice; Lucia Ciccoli; Joussef Hayek; Giuseppe Valacchi
Journal:  Mediators Inflamm       Date:  2013-12-12       Impact factor: 4.711

Review 9.  Mitochondrial Dysfunction in the Pathogenesis of Rett Syndrome: Implications for Mitochondria-Targeted Therapies.

Authors:  Natalya Shulyakova; Ana C Andreazza; Linda R Mills; James H Eubanks
Journal:  Front Cell Neurosci       Date:  2017-03-14       Impact factor: 5.505

10.  Retention of Mitochondria in Mature Human Red Blood Cells as the Result of Autophagy Impairment in Rett Syndrome.

Authors:  Diego Sbardella; Grazia Raffaella Tundo; Luisa Campagnolo; Giuseppe Valacchi; Augusto Orlandi; Paolo Curatolo; Giovanna Borsellino; Maurizio D'Esposito; Chiara Ciaccio; Silvia Di Cesare; Donato Di Pierro; Cinzia Galasso; Marta Elena Santarone; Joussef Hayek; Massimiliano Coletta; Stefano Marini
Journal:  Sci Rep       Date:  2017-09-26       Impact factor: 4.379

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