<scp>Health‐related</scp> quality of life in adults with osteogenesis imperfecta

Murali, Chaya N.; Slater, Brady; Musaad, Salma; Cuthbertson, David; Nguyen, Dianne; Turner, Alicia; Azamian, Mahshid S.; Tosi, Laura L.; Rauch, Frank; Sutton, V. Reid; Lee, Brendan; Nagamani, Sandesh C.S.

doi:10.1111/cge.13939

Cited by 8 publications

(13 citation statements)

References 36 publications

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“…However, in contrast to this classification, we found that the prevalence of chronic pain in the entire sample and the degree of pain interference in adults were similar across OI types. This finding agrees with previous publications, where pain intensity and bodily pain did not correlate with the severity of the disease (Arponen et al, 2018;Murali et al, 2021;Smith, 2015).…”

Section: Discussionsupporting

confidence: 93%

“…To comprehensively understand the pain in OI, we also analyzed the pain content of questions we considered substantially related to pain from PODCI in children and the SF‐12v2 in adults (Table 1). The global scores of these two questionnaires by OI type have already been published (Murali et al, 2020; Murali et al, 2021). Additionally, all the participants were asked if they were under active IV BPs treatment (yes or no).…”

Section: Methodsmentioning

confidence: 99%

“…Ideally, pain assessment should be multidimensional and incorporate patient‐reported outcome measures (PROMs; Boitor et al, 2019; McGrath et al, 2008). Recently, two studies have focused on the validity of measuring the physical function and QoL using Pediatric Outcome Data Collection Instrument (PODCI) and Short Form‐12 items version 2 (SF‐12v2) in children and adults with OI (Murali et al, 2020, 2021). Those studies reported pain/comfort level and bodily pain for the different OI types.…”

Section: Introductionmentioning

confidence: 99%

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A multicenter study to evaluate pain characteristics in osteogenesis imperfecta

Celin

Kruger

Caudill

et al. 2022

American J of Med Genetics Pt A

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The objective was to describe pain characteristics and treatments used in individuals with varying severity of osteogenesis imperfecta (OI) and investigate pain‐associated variables. This work was derived from a multicenter, longitudinal, observational, natural history study of OI conducted at 12 clinical sites of the NIH Rare Diseases Clinical Research Network's Brittle Bone Disorders Consortium. Children and adults with a clinical, biochemical, or molecular diagnosis of OI were enrolled in the study. We did a cross‐sectional analysis of chronic pain prevalence, characteristics, and treatments used for pain relief and longitudinal analysis to find the predictors of chronic pain. We included 861 individuals with OI, in 41.8% chronic pain was present, with similar frequency across OI types. Back pain was the most frequent location. Nonsteroidal anti‐inflammatory drugs followed by bisphosphonates were the most common treatment used. Participants with chronic pain missed more days from school or work/year and performed worse in all mobility metrics than participants without chronic pain. The variables more significantly associated with chronic pain were age, sex, positive history of rodding surgery, scoliosis, other medical problems, assistive devices, lower standardized height, and higher body mass index. The predictors of chronic pain for all OI types were age, use of a wheelchair, and the number of fractures/year. Chronic pain is prevalent in OI across all OI types, affects mobility, and interferes with participation. Multiple covariates were associated with chronic pain.

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Section: Discussionsupporting

confidence: 93%

Section: Methodsmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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A multicenter study to evaluate pain characteristics in osteogenesis imperfecta

Celin

Kruger

Caudill

et al. 2022

American J of Med Genetics Pt A

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“…Unsurprisingly, physical HR-QOL was most impaired in those diagnosed with type 3 OI. Four studies in this review reported signi cantly lower PCS scores in patients living with type 3 compared with type 1 OI (26,27,30,31) . A study by Yonko et al also reported lower respiratory-related QOL in patients living with type 3 compared with type 1 OI.…”

Section: Impact Of Oi Type On Hr-qolmentioning

confidence: 80%

“…In addition, one study reported physical but not mental domains of the SF-36 (30) and another used an abbreviated version called the SF-12 (31) . There was signi cant heterogeneity in how the results of the SF-36 were reported.…”

Section: Hr-qol Assessment Usedmentioning

confidence: 99%

Systematic Review of Health Related-Quality of Life in Adults with Osteogenesis Imperfecta

Donald

Donnell

Martin-Grace

et al. 2022

Preprint

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BackgroundOsteogenesis imperfecta (OI) is a rare, connective tissue disorder characterised by bone fragility, resulting in recurrent fractures and skeletal deformities. Extra-skeletal manifestations include dentinogenesis imperfecta, hearing abnormalities and restrictive lung disease. These co-morbidities combined with recurrent fractures can exert a significant impact on health-related quality of life (HR-QOL). It is important to have a clear understanding of HR-QOL throughout adulthood as some OI specific complications increase with age.MethodsPubMed, EMBASE and CENTRAL databases were searched in February 2022 to identify studies reporting quantitative assessments of HR-QOL in adults with OI. The primary endpoint was to determine the impact of a diagnosis of OI on an adult’s HR-QOL. Secondary endpoints were (i) to examine how frequently various HR-QOL assessment tools were used (ii) identify differences in HR-QOL between OI types and (iii) to investigate the determinants of HR-QOL in adults with OI. Search results were exported to Endnote where two reviewers independently conducted title/abstract and full text reviews. Data from accepted studies were extracted into Microsoft Excel. A narrative synthesis was then undertaken. ResultsThe review identified 17 studies with a total of 1,648 adults. The Short Form-36 (SF-36) was the most widely reported HR-QOL assessment tool, used in nine studies. Physical HR-QOL was reduced in adults with OI. Physical component scores (PCS) or individual physical domains of the SF-36 were lower in eight of nine studies. Mental component scores (MCS) were preserved in all six studies, however individual mental health domains of the SF-36 were reduced in some studies. The prevalence of anxiety/ depression was relatively low in adults with OI. Those with type 3 OI, had lower physical and respiratory HR-QOL but preserved mental HR-QOL compared with type 1. The prevalence of fatigue and pain were higher in adults with OI. Age and cardio-pulmonary co-morbidities were associated with lower HR-QOL. ConclusionOI has an extensive and wide ranging impact on adult HR- QOL. Physical and respiratory HR-QOL were lower, while the prevalence of pain and fatigue were higher than reference populations. Mental HR-QOL was relatively preserved. Age and cardio-pulmonary co-morbidities were associated with lower HR-QOL.

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Evaluation of the Nordic Musculoskeletal Questionnaire for Measuring Prevalence and the Consequence of Pain in a Danish Adult OI Population: A Pilot Study

Horn,

Jensen,

Hartvigsen

et al. 2024

Calcif Tissue Int

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Pain is a challenge in persons with OI and causes much concern in the Osteogenesis Imperfecta (OI) population. We aim to evaluate the usability of the Nordic Musculoskeletal Questionnaire (NMQ) to identify painful sites in adults with OI and to describe the occurrence of musculoskeletal (MSK) pain and its impact on their work and daily activities. This cross-sectional pilot study uses the OI-NMQ to study MSK pain prevalence in nine separate anatomical regions (neck, upper back, lower back, shoulder, elbow, hand/wrist, hip, knee, and ankle/foot) and its impact on regular work and daily activities in adults with OI. The questionnaire was distributed among participants of the 2023 annual meeting of The Danish OI Society. The response rate was 68%, and all participants considered the OI-NMQ helpful in assessing the presence of pain and its consequences. The analysis included 27 adults with OI type I, III, or IV above 18 years. Among all 27 participants, MSK pain was present in 15–56% of the 9 sites within the last 7 days and 33–89% of the nine anatomical regions during the last 12 months. In 7–48% of all the participants, their regular work and daily activities had been affected by the presence of MSK pain. The OI-NMQ was feasible in assessing MSK pain among adults with OI and displayed a high prevalence of MSK pain with a moderate impact on their regular work and daily activities in this OI population. A larger and repeated measurement of MSK pain in adults with OI is needed to confirm these results.

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Health‐related quality of life in adults with osteogenesis imperfecta

Cited by 8 publications

References 36 publications

A multicenter study to evaluate pain characteristics in osteogenesis imperfecta

A multicenter study to evaluate pain characteristics in osteogenesis imperfecta

Systematic Review of Health Related-Quality of Life in Adults with Osteogenesis Imperfecta

Evaluation of the Nordic Musculoskeletal Questionnaire for Measuring Prevalence and the Consequence of Pain in a Danish Adult OI Population: A Pilot Study

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