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Record W4406739936 · doi:10.1093/jbmrpl/ziaf012

Qualitative analysis of pain impact in adult patients with X-linked hypophosphatemia

2025· article· en· W4406739936 on OpenAlex

Why this work is in the frame

A frame that forgets how it found something cannot be audited. These are the routes that admitted this work.

fundA Canadian funder is recorded on the work.
no affNo Canadian affiliation: this work is invisible to an affiliation-only frame.
No Canadian affiliation. An affiliation-only frame, the usual design, would never have seen this work. It is one of the works that make the case for inverting the frame.

Bibliographic record

VenueJBMR Plus · 2025
Typearticle
Languageen
FieldMedicine
TopicParathyroid Disorders and Treatments
Canadian institutionsnot available
FundersUniversity of TorontoHospital for Sick ChildrenConnecticut Children's Medical Center
KeywordsHypophosphatemiaMedicineInternal medicine

Abstract

fetched live from OpenAlex

We previously reported pain as a predominant finding in adult patients with X-linked hypophosphatemia (XLH). This study explored health-related quality of life using the 36-Item Short Form Survey Instrument (SF-36v1) and pain experiences of adults with XLH through qualitative analysis of interviews with 15 patients (11 females, 4 males). Age-adjusted differences using SF-36 were lower than the general population in all domains of health-related quality of life, with significant differences notably related to physical function and pain. Ten themes emerged from semi-structured interviews: (1) chronic and variable pain: patients reported persistent pain of varying intensity and location; (2) impact on mobility and daily life: pain severely restricted daily activities, affecting lifestyle, employment, and independence; (3) pain management: patients used medications, physical therapy, lifestyle changes, alternative therapies, and assistive devices; (4) healthcare and accessibility challenges: access to appropriate care and treatments was limited by insurance issues and healthcare providers' lack of XLH knowledge; (5) emotional and psychological impact: chronic pain and limitations led to social isolation, depression, and emotional burden; (6) desire for improved information and support: patients sought better information, treatment, and community support; (7) challenges with physical inactivity: staying active was essential but difficult, as inactivity worsened pain; (8) navigating healthcare services: accessing therapy and insurance was often challenging; (9) long-term outlook and concerns: patients worried about disease progression and future health; and (10) advocacy and awareness: increased public and medical awareness was needed to improve care. Although pain is not a universal experience within this population, it is a significant issue for many individuals with XLH. These data illustrate the profound impact of XLH on multiple life aspects underscoring the need for effective pain management strategies, better healthcare accessibility, supportive tools, and a more knowledgeable medical community to improve quality of life. Insights from this study will guide the development of a digital pain self-management intervention tailored to XLH patients to address these needs.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

Full frame distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.007
Threshold uncertainty score0.349

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.001
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0000.000

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.

Opus teacher head0.012
GPT teacher head0.364
Teacher spread0.353 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it