Literature DB >> 23828127

Osteoporosis in Rett syndrome: a case study presenting a novel management intervention for severe osteoporosis.

M Lotan1, R Reves-Siesel, R S Eliav-Shalev, J Merrick.   

Abstract

The present article describes a successful novel therapeutic intervention with Aredia with one child with Rett syndrome, after suffering from six pathological fractures within less than 3 years due to severe osteoporosis. Since the initiation of the treatment (3 years ago), the child has not suffered any fractures. Patients with chronic diseases and those with disabilities or on anticonvulsant medications are at risk for low bone density and possibly for the resultant pathologic fractures that define osteoporosis in children. Individuals with Rett syndrome (RS) have been shown to have low bone mineral density (or osteopenia) at a young age. If osteoporosis occurs in a girl with RS, it can inflict pain and seriously impair the child's mobility and quality of life. The present article describes a case study of a child with RS (showing an average of 1.75 fractures annually for the 4 years preceding the treatment) before and after a treatment with Aredia. Patient received 30 mg/day for 3 days on a once every 3-month cycle. There was a 45 % improvement in bone mass density (BMD) values from pre-post-intervention. The child had no fractures in the 3 years posttreatment. This finding is significant (p < 0.03). The BMD Z-scores of the child showed severe osteoporosis (Z-score of -3.8) at pre-intervention and are elevated to osteopenia levels (Z-score of -1.3) at post-intervention measurements. All measurements suggest that the treatment successfully reversed the osteoporotic process and prevented further fractures. This change caused great relief to the child and her family and an improvement in their quality of life. The findings support the ability (in one case) to reverse the progression of osteoporosis in individuals with Rett syndrome showing severe osteoporosis with multiple fractures.

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Year:  2013        PMID: 23828127     DOI: 10.1007/s00198-013-2423-5

Source DB:  PubMed          Journal:  Osteoporos Int        ISSN: 0937-941X            Impact factor:   4.507


  17 in total

1.  Bone density and other possible predictors of fracture risk in children and adolescents with spastic quadriplegia.

Authors:  R C Henderson
Journal:  Dev Med Child Neurol       Date:  1997-04       Impact factor: 5.449

2.  Fractional calcium absorption is increased in girls with Rett syndrome.

Authors:  Kathleen J Motil; Rebecca J Schultz; Steven Abrams; Kenneth J Ellis; Daniel G Glaze
Journal:  J Pediatr Gastroenterol Nutr       Date:  2006-04       Impact factor: 2.839

3.  Pathological fractures in patients with cerebral palsy.

Authors:  R Brunner; L Doderlein
Journal:  J Pediatr Orthop B       Date:  1996       Impact factor: 1.041

4.  Osteopenia in Rett syndrome.

Authors:  R H Haas; S D Dixon; D J Sartoris; M J Hennessy
Journal:  J Pediatr       Date:  1997-11       Impact factor: 4.406

5.  Fractures caused by falling from a wheelchair in patients with neuromuscular disease.

Authors:  B Gray; J D Hsu; J Furumasu
Journal:  Dev Med Child Neurol       Date:  1992-07       Impact factor: 5.449

6.  Bone quantitative ultrasound and nutritional status in severely handicapped institutionalized children and adolescents.

Authors:  C Hartman; R Brik; A Tamir; J Merrick; R Shamir
Journal:  Clin Nutr       Date:  2004-02       Impact factor: 7.324

7.  [Contribution to studies of seizures in Rett's syndrome. Analysis of critical forms of four cases].

Authors:  M Nieto; F Aguilar; R Candau; M L Siljestrom; P Prieto
Journal:  Rev Neurol       Date:  1995 Nov-Dec       Impact factor: 0.870

Review 8.  Osteoporosis in children and adolescents.

Authors:  Maria Luisa Bianchi
Journal:  Bone       Date:  2007-07-18       Impact factor: 4.398

Review 9.  Current advances in bone health of disabled children.

Authors:  Margaret Zacharin
Journal:  Curr Opin Pediatr       Date:  2004-10       Impact factor: 2.856

10.  Continuous therapy with pamidronate, a potent bisphosphonate, in postmenopausal osteoporosis.

Authors:  I R Reid; D J Wattie; M C Evans; G D Gamble; J P Stapleton; J Cornish
Journal:  J Clin Endocrinol Metab       Date:  1994-12       Impact factor: 5.958

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  8 in total

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Authors:  Helen Leonard; Stuart Cobb; Jenny Downs
Journal:  Nat Rev Neurol       Date:  2016-12-09       Impact factor: 42.937

2.  Bone microarchitecture in Rett syndrome and treatment with teriparatide: a case report.

Authors:  M B Zanchetta; M F Scioscia; J R Zanchetta
Journal:  Osteoporos Int       Date:  2016-04-11       Impact factor: 4.507

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Authors:  Gemma Marcucci; Maria Luisa Brandi
Journal:  Clin Cases Miner Bone Metab       Date:  2015-10-26

4.  Teriparatide in the treatment of recurrent fractures in a Rett patient.

Authors:  Carla Caffarelli; Jussef Hayek; Ranuccio Nuti; Stefano Gonnelli
Journal:  Clin Cases Miner Bone Metab       Date:  2015-12-29

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Authors:  Nicolas Garcelon; Antoine Neuraz; Rémi Salomon; Nadia Bahi-Buisson; Jeanne Amiel; Capucine Picard; Nizar Mahlaoui; Vincent Benoit; Anita Burgun; Bastien Rance
Journal:  Orphanet J Rare Dis       Date:  2018-05-31       Impact factor: 4.123

Review 6.  Bone fragility in patients affected by congenital diseases non skeletal in origin.

Authors:  L Masi; S Ferrari; M K Javaid; S Papapoulos; D D Pierroz; M L Brandi
Journal:  Orphanet J Rare Dis       Date:  2021-01-06       Impact factor: 4.123

7.  Subclinical inflammatory status in Rett syndrome.

Authors:  Alessio Cortelazzo; Claudio De Felice; Roberto Guerranti; Cinzia Signorini; Silvia Leoncini; Alessandra Pecorelli; Gloria Zollo; Claudia Landi; Giuseppe Valacchi; Lucia Ciccoli; Luca Bini; Joussef Hayek
Journal:  Mediators Inflamm       Date:  2014-01-06       Impact factor: 4.711

8.  Lower incidence of fracture after IV bisphosphonates in girls with Rett syndrome and severe bone fragility.

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

  8 in total

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