Osteoporosis pathology in people with severe motor and intellectual disability

Sakai, Tomoko; Honzawa, Shiho; Kaga, Makiko; Iwasaki, Yuji; Masuyama, Tatsuo

doi:10.1016/j.braindev.2019.12.010

Cited by 13 publications

(10 citation statements)

References 23 publications

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“…Osteoporosis is one of the most common metabolic bone diseases which results from the disrupted balance between bone formation and resorption 1 . With the progressive aging of the global population, the incidence of osteoporosis is increasing dramatically which leads to an increased risk of fractures and exerts a strong impact on morbidity, and even mortality 2,3 .…”

Section: Introductionmentioning

confidence: 99%

Long non-coding RNA MIR22HG promotes osteogenic differentiation of bone marrow mesenchymal stem cells via PTEN/ AKT pathway

et al. 2020

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Osteoporosis is a prevalent metabolic bone disease characterized by low bone mineral density and degenerative disorders of bone tissues. Previous studies showed the abnormal osteogenic differentiation of endogenous bone marrow mesenchymal stem cells (BMSCs) contributes to the development of osteoporosis. However, the underlying mechanisms by which BMSCs undergo osteogenic differentiation remain largely unexplored. Recently, long non-coding RNAs have been discovered to play important roles in regulating BMSC osteogenesis. In this study, we first showed MIR22HG, which has been demonstrated to be involved in the progression of several cancer types, played an important role in regulating BMSC osteogenesis. We found the expression of MIR22HG was significantly decreased in mouse BMSCs from the osteoporotic mice and it was upregulated during the osteogenic differentiation of human BMSCs. Overexpression of MIR22HG in human BMSCs enhanced osteogenic differentiation, whereas MIR22HG knockdown inhibited osteogenic differentiation both in vitro and in vivo. Mechanistically, MIR22HG promoted osteogenic differentiation by downregulating phosphatase and tensin homolog (PTEN) and therefore activating AKT signaling. Moreover, we found MIR22HG overexpression promoted osteoclastogenesis of RAW264.7 cells, which indicated that MIR22HG played a significant role in bone metabolism and could be a therapeutic target for osteoporosis and other bone-related diseases.

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Section: Introductionmentioning

confidence: 99%

Long non-coding RNA MIR22HG promotes osteogenic differentiation of bone marrow mesenchymal stem cells via PTEN/ AKT pathway

et al. 2020

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“…Age was a predictor of bone quality, and current physical ability was the strongest predictor. We reported previously that bone resorption markers in female patients with SMID increased beyond 50 years of age, possibly representing a sign of primary osteoporosis ( 4 ). The existence of primary osteoporosis could be an influencing factor, although it is unclear in this study.…”

Section: Discussionmentioning

confidence: 95%

“…This study investigated calcaneal QUS measures of osteoporosis in patients with SMID. In a cohort of bedridden patients from a different institution, we had previously reported that the bone quality examined by QUS of patients with SMID was consistently low from a young age ( 4 ).…”

Section: Discussionmentioning

confidence: 96%

“…No significant differences were observed among the groups regarding the tube feeding status. We reported previously that tube feeding was a substantial predictor for low bone quality in bedridden patients with SMID ( 4 ). Many reports have found that tube feeding is a risk factor for low bone quality in patients with CP ( 7 , 8 ).…”

Section: Discussionmentioning

confidence: 98%

“…In cases involving primary osteoporosis, lower levels of female hormones during the postmenopausal stage in female patients increase the rate of bone resorption, resulting in decreased bone density ( 3 ). In patients with SMID, bone quality examined by quantitative ultrasonography (QUS) does not increase with growth and remains low throughout the person’s lifetime ( 4 ).…”

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confidence: 99%

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Bone quality in adults with severe motor and intellectual disabilities

Sakai

Shirai²,

Oishi³

2021

J Rehabil Med

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Objective To determine bone quality in adults with severe motor and intellectual disabilities. Design A retrospective cohort study. Patients Bone quality of 60 patients with severe motor and intellectual disabilities (28 men, 32 women; mean age 57 years) at a long-term care facility for adults was examined retrospectively. Methods Quantitative ultrasonography was used to measure the stiffness index, T-score and Z-score of the calcaneus. A multiple linear regression model, including sex, age, anti-epileptic drug use, tube-feeding status, and current and peak physical abilities, was used to identify significant predictors of T-scores. Results Quantitative ultrasonography revealed that all patients had lower bone quality (based on T-scores, Z-scores, and stiffness index), and all patients had T-scores with standard deviations (SD) below 1.8. Current physical ability, age, and anti-epileptic drug use were significant factors in T-score determination, while tube-feeding and peak physical ability were not. The ability to walk without assistance was the most significant predictor in quantitative ultrasonography. Conclusion Severely low bone quality is observed in patients with severe motor and intellectual disabilities; and it is strongly associated with current physical activity. It is important that patients with severe motor and intellectual disabilities preserve their physical abilities to prevent osteoporosis- related fractures. LAY ABSTRACT Fractures are common in adults with severe motor and intellectual disabilities. This study assessed the bone quality of 60 individuals from young adult to advanced age. All patients had low bone quality from a young age, and current physical ability, age, and anti-epileptic drug use were factors affecting bone quality, while tube-feeding and peak physical ability were not. Severely low bone quality in patients with severe motor and intellectual disabilities is strongly associated with current physical activity levels. It is very important for patients with severe motor and intellectual disabilities to increase and preserve their physical activities and abilities, respectively, to prevent osteoporosis-related fractures.

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