Title: Fractures in people with intellectual disabilities: comparison with the general population and development of a fracture risk calculator specific to these patients.
Funder: Department of Health and Social Care

Abstract: Background A high rate of osteoporosis and fractures, and a relatively young age at fracture, have been consistently reported in people with intellectual disabilities (ID- aka learning disabilities) by numerous studies over the past 30 years. A recent Canadian study including 30,000 people with ID and 1.5 million general population subjects, showed a low-trauma fracture rate three times higher in those with ID. Our long-term clinical experience is consistent with the literature and highlights the devastating effects of fractures in patients in whom immobilisation and post-fracture rehabilitation are extremely difficult. Fractures are also very expensive, particularly in those making a poor recovery, among whom people with ID are likely over-represented. Nevertheless, these patients increased fracture risk is largely overlooked, both in primary and secondary care. Neither of the fracture risk assessment tools recommended by the National Institute for Health and Care Excellence (NICE), Frax and Qfracture-2016, take into account risk factors for fractures frequent in people with ID. Furthermore, even in the latest NICE Clinical Knowledge Summary and Quality Standard on Osteoporosis (dated 2016 and 2017) patients with ID are not identified as a group at risk, and the yearly routine primary care health check for these patients does not address bone health at all. The lack of awareness of the increased risk of fractures in patients with ID is likely due to three key factors. First, the absence of UK specific epidemiological data. Second, the fact that the limited information on risk factors for fractures in this group has remained confined to the scientific literature or unpublished clinical observations without reaching the majority of clinicians or health policy makers. Third, the intellectual disabilities themselves, which prevent patients from expressing and raising concerns about their health problems. Aims 1) To compare the rate of fractures between people with ID and the general UK population 2) To investigate the risk factors for fractures and develop a fracture risk scoring system specific to people with ID 3) To raise awareness of the increased risk of fractures in people with ID so that appropriate prevention strategies can be adopted Research questions 1) What is the incidence, and the age and sex distribution of fractures in people with ID compared to the general UK population? 2) What are the risk factors for fractures in people with ID? Plan We will compare fracture rates between people with ID (estimated 300,000) and general population subjects (up to 15,000,000) in the Clinical Practice Research Datalink (CPRD), a large UK database containing anonymised data from people registered with General Practitioners. Linkage of CPRD to Hospital Episode Statistics databases will allow comprehensive collection of fracture data. The sheer size of the databases will allow in-depth analysis of potential risk factors for fractures in the ID population. We will investigate all the risk factors included in QFracture-2016 and additional factors relevant to people with ID such as cause of ID (e.g. Down s syndrome), associated illnesses (e.g. cerebral palsy), endocrine abnormalities (e.g. hypogonadism), medication (e.g. antipsychotics), vitamin D deficiency and others. We will then construct a fracture risk scoring system based on a derivation cohort and assess it in a validation cohort within the CPRD. Potential benefits to patients and the NHS Identification of patients with ID at risk of fractures could allow treatment with available medication, and lead to adopting methods to reduce the risk of falls. The scoring system, following future validation in an additional cohort, could be made available as a fracture risk calculator for widespread clinical use. Ultimately, a reduction in fracture incidence could be achieved, which would benefit patients and potentially reduce NHS costs.

Lay abstract: Background People with intellectual disabilities (ID) have poorer health than the general population. We aim to reduce this inequality by tackling fractures, which are serious, costly but often preventable events. A recent survey from Canada on 30,000 individuals with ID and 1.5 million general population subjects showed a fracture rate three times higher in ID people, confirming findings of smaller studies over the past 30 years. However, current guidelines on fractures don t identify ID patients as a high-risk group and their routine health check doesn t address bone health. The neglect of these patients fracture risk probably stems from the lack of UK specific data. Additionally, the data on fractures have not filtered from the scientific literature to clinicians, health policy makers, patients and carers. Study proposal We will compare fracture rates between patients with ID and the general UK population and examine risk factors for fractures in these patients. We ll use a large general practice database, containing data from approximately 15 million people including 300,000 with ID. We ll design a fracture risk scoring system specific for people with ID. This is important because the currently available fracture risk assessment tools may not include risk factors for fractures which are common in these patients. Our study results will help identify ID patients at risk of fractures. Available prevention strategies (e.g. improvements in the living environment to reduce the risk of falls, addition of or changes in medication) could then be used to reduce the incidence of fractures in these patients. This is our study s ultimate aim. Patient and Public involvement A carer of a patient with ID is part of our research group. The study is endorsed by the three major relevant charities (Mencap, Down s Syndrome Association, National Osteoporosis Society), whose support will be essential particularly for the dissemination of results.

Research activity codes: 7.3 MANAGEMENT AND DECISION MAKING;2.4 SURVEILLANCE AND DISTRIBUTION
Health categories: MENTAL HEALTH;INJURIES AND ACCIDENTS;MUSCULOSKELETAL