Literature DB >> 20661168

Bone mass in Rett syndrome: association with clinical parameters and MECP2 mutations.

Jay R Shapiro1, Genila Bibat, Girish Hiremath, Mary E Blue, Shilpa Hundalani, Theodore Yablonski, Aditi Kantipuly, Charles Rohde, Michael Johnston, Sakkubai Naidu.   

Abstract

Rett syndrome (RTT) is an X-linked neurodevelopmental disorder caused by mutations in the MECP2 gene. In 49 female RTT children, aged 1.9-17 y, bone mass was assessed and correlated with clinical parameters and mutations involving the MECP2 gene. We also studied five adult females, aged 20-33 y, and one male child, aged 6 y. Lumbar spine bone mineral content (BMC) and bone mineral density (BMD) were correlated with weight, height, BMI, clinical severity, degree of scoliosis, use of anticonvulsants, and ambulatory status. L1-L4 BMD and BMC showed that 48.9% of them had BMD values >2 SD below age-related norms. BMD values were in the osteoporotic range in the five adult females with RTT. Eleven percent of the children and adults with RTT experienced fractures. Low bone mass was correlated with marginal significance to clinical severity and ambulation but not to scoliosis or anticonvulsant use. Lowest bone mass occurred in patients with T158M or R270X mutations but without statistical significance. Studies in a murine model of RTT confirmed low bone mass as an inherent component of this syndrome. MECP2 mutations and clinical parameters impact bone mass in RTT, but an association with a specific mutation was not demonstrable.

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Year:  2010        PMID: 20661168      PMCID: PMC3074246          DOI: 10.1203/PDR.0b013e3181f2edd2

Source DB:  PubMed          Journal:  Pediatr Res        ISSN: 0031-3998            Impact factor:   3.756


  30 in total

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Journal:  J Child Neurol       Date:  2005-09       Impact factor: 1.987

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  11 in total

1.  Low bone mineral mass is associated with decreased bone formation and diet in girls with Rett syndrome.

Authors:  Kathleen J Motil; Judy O Barrish; Jeffrey L Neul; Daniel G Glaze
Journal:  J Pediatr Gastroenterol Nutr       Date:  2014-09       Impact factor: 2.839

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Journal:  Nat Rev Neurol       Date:  2016-12-09       Impact factor: 42.937

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Journal:  J Pediatr Gastroenterol Nutr       Date:  2012-09       Impact factor: 2.839

Review 4.  Rett syndrome: think outside the (skull) box.

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Journal:  Fac Rev       Date:  2021-06-29

5.  Biomechanical properties of bone in a mouse model of Rett syndrome.

Authors:  Bushra Kamal; David Russell; Anthony Payne; Diogo Constante; K Elizabeth Tanner; Hanna Isaksson; Neashan Mathavan; Stuart R Cobb
Journal:  Bone       Date:  2014-10-24       Impact factor: 4.398

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Journal:  Open Biol       Date:  2018-02       Impact factor: 6.411

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Authors:  Neeti Vashi; Monica J Justice
Journal:  Mamm Genome       Date:  2019-02-28       Impact factor: 2.957

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Authors:  Carla Caffarelli; Stefano Gonnelli; Maria Dea Tomai Pitinca; Silvia Camarri; Antonella Al Refaie; Joussef Hayek; Ranuccio Nuti
Journal:  BMC Med Genet       Date:  2020-01-31       Impact factor: 2.103

9.  Clinical Guidelines for Management of Bone Health in Rett Syndrome Based on Expert Consensus and Available Evidence.

Authors:  Amanda Jefferson; Helen Leonard; Aris Siafarikas; Helen Woodhead; Sue Fyfe; Leanne M Ward; Craig Munns; Kathleen Motil; Daniel Tarquinio; Jay R Shapiro; Torkel Brismar; Bruria Ben-Zeev; Anne-Marie Bisgaard; Giangennaro Coppola; Carolyn Ellaway; Michael Freilinger; Suzanne Geerts; Peter Humphreys; Mary Jones; Jane Lane; Gunilla Larsson; Meir Lotan; Alan Percy; Mercedes Pineda; Steven Skinner; Birgit Syhler; Sue Thompson; Batia Weiss; Ingegerd Witt Engerström; Jenny Downs
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Authors:  Anne-Sophie Lambert; Anya Rothenbuhler; Perrine Charles; Sylvie Brailly-Tabard; Séverine Trabado; Elisabeth Célestin; Emmanuel Durand; Isabelle Fontaine; Lotfi Miladi; Philippe Wicart; Nadia Bahi-Buisson; Agnès Linglart
Journal:  PLoS One       Date:  2017-10-26       Impact factor: 3.240

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