Healthcare perceptions and concerns among individuals with osteogenesis imperfecta and their caregivers in Germany
Bibliographic record
Abstract
Introduction: The IMPACT Survey explored self-reported experiences of the clinical, humanistic, and economic impact of osteogenesis imperfecta (OI), a rare hereditary connective tissue disorder associated with low bone mass, increased bone fragility and variable secondary features. Here we present findings on perceptions and concerns associated with healthcare among adults with OI and caregivers (CG) of individuals with OI in Germany. Methods: The IMPACT Survey was developed by the Osteogenesis Imperfecta Federation Europe (OIFE), the Osteogenesis Imperfecta Foundation (OIF) and an international steering committee of OI experts. Aimed at adults with OI (presented here), CGs (with or without OI; both presented here), adolescents with OI, and relatives, it was fielded online from July–September 2021 in eight languages. The OIFE and OIF drove global recruitment, with the Deutsche OI-Gesellschaft (DOIG) leading in Germany. Responses were translated into English. Microsoft Excel was used to clean, code, validate and analyse data. Descriptive statistics for the German cohort are presented here. Results: Among 194 German adult respondents, there were 134 adults with OI (including 8 CGs and 126 non-CGs) and 60 CGs without OI. While many individuals felt that care was continuous (44% of adults with OI; 53% of CGs without OI), at least half of the respondents (49% of adults with OI; 66% of CGs without OI) felt the need to coordinate care for themselves or individuals in their care. More CGs without OI (70%) reported receiving sufficient information about OI compared with adults with OI (51%); however, similar proportions of respondents felt that doctors and other healthcare professionals did not understand OI (29% of adults with OI; 22% of CGs without OI). Furthermore, a lower proportion of adults with OI (32%) felt that doctors and other healthcare professionals understood their needs and concerns compared with CGs without OI (50%). Whilst individuals with and without OI shared several worries, a higher proportion of adults with OI worried about access to doctors (82%) and care (76%) compared with CGs without OI (64% and 45%, respectively). Notably, less than a quarter of adults with OI (15%) felt supported in their transition from paediatric to adult care. This was emphasised by the fact that 74% of CGs without OI expressed worry about how individuals in their care would transition from paediatric to adult in the future. Discussion: A significant disparity in the perception of the quality of healthcare was observed when comparing adults with OI and CGs without OI. Insights from the IMPACT Survey emphasise the need for a more coordinated healthcare pathway to address specific concerns and elevate the overall quality of care for individuals with OI.
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How this classification was reachedexpand
Full frame machine prediction
Teacher imitationNot calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.
Distilled classifier scores by category (both heads)
| Category | Codex | Gemma |
|---|---|---|
| Metaresearch | 0.001 | 0.002 |
| Meta-epidemiology (narrow) | 0.000 | 0.000 |
| Meta-epidemiology (broad) | 0.000 | 0.000 |
| Bibliometrics | 0.001 | 0.001 |
| Science and technology studies | 0.001 | 0.001 |
| Scholarly communication | 0.001 | 0.000 |
| Open science | 0.000 | 0.001 |
| Research integrity | 0.000 | 0.000 |
| Insufficient payload (model declined to judge) | 0.001 | 0.000 |
Machine scores (provisional)
The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.
Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.
score_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from itClassification
machine, unvalidatedMachine predicted; a candidate call from one source (direct Gemma or distilled Codex), not a consensus.
How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".