The effect of low magnitude mechanical stimulation (LMMS) on bone density in patients with Rett syndrome: A pilot and feasibility study

Issue title: Bone Health in Children with Special Health Care Needs

Article type: Research Article

Authors: Afzal, Sarah Y.^; | Wender, Anna R.^; | Jones, Mary D. | Fung, Ellen B. | Pico, Elaine L.^{; ;}

Affiliations: Katie's Clinic for Rett Syndrome, Children's Hospital & Research Center Oakland, Oakland, CA, USA | Children's Hospital Oakland Research Institute, Oakland, CA, USA | HEDCO Health Sciences Center, Children's Hospital Oakland Research Institute, Oakland, CA, USA | Division of Pediatric and Rehabilitative Medicine, Children's Hospital & Research Center Oakland, Oakland, CA, USA | Emory University, Atlanta, GA, USA

Note: [] Corresponding author: Elaine L. Pico, Children's Hospital Oakland Research Institute 744 52nd St, Oakland, CA 94609, USA. Tel.: +1 510 601 PICO (7426); Fax: +1 510 923 0378; E-mail: [email protected] or [email protected]

Abstract: PURPOSE: Low magnitude mechanical stimulation (LMMS) has been used successfully to promote bone formation in certain patient populations. This study evaluated the feasibility and effectiveness of LMMS on improving bone mineral density (BMD) in patients with Rett syndrome. METHODS: A 12-month crossover pilot study design of 6 months of intervention with LMMS and 6 months without was studied in 14 subjects divided in two subgroups. BMD was assessed using Dual Energy X-ray Absorptiometry (DXA). The levels of 25-hydroxy vitamin D (25OHD), Parathyroid Hormone (PTH), Insulin-Like Growth Factor 1 (IGF-1), and circulating markers of bone resorption (NTx) were analyzed in blood samples. Health questionnaires and diet logs were obtained at 0, 6, and 12 months. RESULTS: Of the 11 subjects who completed the protocol, 9 had an adherence of > 65% and showed an increase in spine BMD Z-scores from the intervention (Z: −2.51) compared to non-intervention period (Z: −2.27) of 0.23 SD (p=0.048). Following intervention, favorable trends were also observed for IGF-1 (p=0.06) and right distal femur BMD Z-scores (p=0.07). CONCLUSIONS: These preliminary results are promising for a larger, placebo-controlled randomized study of subjects with Rett syndrome.

Keywords: Rett syndrome, MECP2, bone mineral density, low magnitude mechanical stimulation (LMMS), Z-scores, DXA, fracture, osteopenia

DOI: 10.3233/PRM-140286

Journal: Journal of Pediatric Rehabilitation Medicine, vol. 7, no. 2, pp. 167-178, 2014