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Record W4225012659 · doi:10.1038/s41574-022-00662-x

Interdisciplinary management of FGF23-related phosphate wasting syndromes: a Consensus Statement on the evaluation, diagnosis and care of patients with X-linked hypophosphataemia

2022· review· en· W4225012659 on OpenAlexaff
Andrea Trombetti, Nasser M. Al‐Daghri, Maria Luisa Brandi, Jorge B. Cannata‐Andía, Etienne Cavalier, Manju Chandran, Catherine Chaussain, Lucia Cipullo, Cyrus Cooper, Dieter Haffner, Pol Harvengt, Nicholas C. Harvey, M K Javaid, Famida Jiwa, John А. Kanis, Andrea Laslop, Michaël R. Laurent, Agnès Linglart, Andréa Marques, Gabriel T. Mindler, Salvatore Minisola, María Concepción Prieto Yerro, Mário Miguel Rosa, Lothar Seefried, Mila Vlaskovska, María Belén Zanchetta, René Rizzoli

Bibliographic record

VenueNature Reviews Endocrinology · 2022
Typereview
Languageen
FieldMedicine
TopicParathyroid Disorders and Treatments
Canadian institutionsOsteoporosis Canada
FundersMedical Research CouncilNational Institute for Health and Care ResearchRadius HealthUltragenyx PharmaceuticalDanoneAlexion PharmaceuticalsServierAmgenPfizerWorld Health OrganizationEli Lilly and Company
KeywordsMedicineRicketsOsteomalaciaQuality of life (healthcare)PediatricsWastingIntensive care medicineFibroblast growth factor 23Vitamin D and neurologyDiseasePhysical therapyInternal medicine

Abstract

fetched live from OpenAlex

X-linked hypophosphataemia (XLH) is the most frequent cause of hypophosphataemia-associated rickets of genetic origin and is associated with high levels of the phosphaturic hormone fibroblast growth factor 23 (FGF23). In addition to rickets and osteomalacia, patients with XLH have a heavy disease burden with enthesopathies, osteoarthritis, pseudofractures and dental complications, all of which contribute to reduced quality of life. This Consensus Statement presents the outcomes of a working group of the European Society for Clinical and Economic Aspects of Osteoporosis, Osteoarthritis and Musculoskeletal Diseases, and provides robust clinical evidence on management in XLH, with an emphasis on patients’ experiences and needs. During growth, conventional treatment with phosphate supplements and active vitamin D metabolites (such as calcitriol) improves growth, ameliorates leg deformities and dental manifestations, and reduces pain. The continuation of conventional treatment in symptom-free adults is still debated. A novel therapeutic approach is the monoclonal anti-FGF23 antibody burosumab. Although promising, further studies are required to clarify its long-term efficacy, particularly in adults. Given the diversity of symptoms and complications, an interdisciplinary approach to management is of paramount importance. The focus of treatment should be not only on the physical manifestations and challenges associated with XLH and other FGF23-mediated hypophosphataemia syndromes, but also on the major psychological and social impact of the disease. This Consensus Statement provides robust clinical evidence on the multidisciplinary management of children and adults with X-linked hypophosphataemia, with an emphasis on patients’ experiences and needs. It is the outcome of a working group of the European Society for Clinical and Economic Aspects of Osteoporosis, Osteoarthritis and Musculoskeletal Diseases.

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.

How this classification was reachedexpand

Full frame machine prediction

Teacher imitation

Not 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.

metaresearch head score (Codex)0.023
metaresearch head score (Gemma)0.031
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Not applicable · Consensus signal: Not applicable
GenreCandidate signal: Review · Consensus signal: none
Teacher disagreement score0.023
Threshold uncertainty score0.122

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0230.031
Meta-epidemiology (narrow)0.0020.001
Meta-epidemiology (broad)0.0030.005
Bibliometrics0.0030.002
Science and technology studies0.0020.002
Scholarly communication0.0030.002
Open science0.0060.006
Research integrity0.0110.010
Insufficient payload (model declined to judge)0.0030.002

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 head0.070
GPT teacher head0.385
Teacher spread0.315 · 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

Classification

machine, unvalidated

Machine predicted; a candidate call from one source (direct Gemma or distilled Codex), not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designNot applicable
Domainnot available
GenreReview

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".

Quick stats

Citations118
Published2022
Admission routes1
Has abstractyes

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