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Record W2261933367 · doi:10.1371/journal.pone.0146824

Clinical Guidelines for Management of Bone Health in Rett Syndrome Based on Expert Consensus and Available Evidence

2016· review· en· W2261933367 on OpenAlexaff
Amanda Jefferson, Helen Leonard, Aris Siafarikas, Helen Woodhead, Sue Fyfe, Leanne M. Ward, Craig F. Munns, Kathleen J. Motil, Daniel Tarquinio, Jay R. Shapiro, Torkel B. Brismar, Bruria Ben‐Zeev, Anne‐Marie Bisgaard, Giangennaro Coppola, Carolyn Ellaway, Michael Freilinger, Suzanne Geerts, Peter Humphreys, Mary Jones, Jane B. Lane, Meir Lotan, Alan K. Percy, Mercédes Pineda, Steven A. Skinner, Birgit Syhler, Sue Ann Thompson, Batia Weiss, Ingegerd Witt Engerström, Jenny Downs

Bibliographic record

VenuePLoS ONE · 2016
Typereview
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicGenetics and Neurodevelopmental Disorders
Canadian institutionsChildren's Hospital of Eastern OntarioUniversity of Ottawa
FundersEunice Kennedy Shriver National Institute of Child Health and Human DevelopmentSchool of Medicine, Johns Hopkins UniversityNational Health and Medical Research CouncilUniversity of California, San FranciscoNational Institutes of HealthAriel UniversityTexas Children's HospitalUniversità degli Studi di SalernoCardiff UniversityUniversität WienInternational Rett Syndrome FoundationMedizinische Universität WienKarolinska InstitutetInstituto de Salud Carlos IIIJohns Hopkins UniversityRigshospitaletAgricultural Research ServiceCivitan InternationalUniversity of California, San DiegoU.S. Department of Agriculture
KeywordsRett syndromeMedicineDelphi methodPsychological interventionOsteoporosisPhysical therapyBone healthPediatricsBone mineralIntensive care medicinePathologyPsychiatryComputer science

Abstract

fetched live from OpenAlex

OBJECTIVES: We developed clinical guidelines for the management of bone health in Rett syndrome through evidence review and the consensus of an expert panel of clinicians. METHODS: An initial guidelines draft was created which included statements based upon literature review and 11 open-ended questions where literature was lacking. The international expert panel reviewed the draft online using a 2-stage Delphi process to reach consensus agreement. Items describe the clinical assessment of bone health, bone mineral density assessment and technique, and pharmacological and non-pharmacological interventions. RESULTS: Agreement was reached on 39 statements which were formulated from 41 statements and 11 questions. When assessing bone health in Rett syndrome a comprehensive assessment of fracture history, mutation type, prescribed medication, pubertal development, mobility level, dietary intake and biochemical bone markers is recommended. A baseline densitometry assessment should be performed with accommodations made for size, with the frequency of surveillance determined according to individual risk. Lateral spine x-rays are also suggested. Increasing physical activity and initiating calcium and vitamin D supplementation when low are the first approaches to optimizing bone health in Rett syndrome. If individuals with Rett syndrome meet the ISCD criterion for osteoporosis in children, the use of bisphosphonates is recommended. CONCLUSION: A clinically significant history of fracture in combination with low bone densitometry findings is necessary for a diagnosis of osteoporosis. These evidence and consensus-based guidelines have the potential to improve bone health in those with Rett syndrome, reduce the frequency of fractures, and stimulate further research that aims to ameliorate the impacts of this serious comorbidity.

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)0.076
metaresearch head score (Gemma)0.141
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Systematic review · Consensus signal: none
GenreCandidate signal: Review · Consensus signal: none
Teacher disagreement score0.076
Threshold uncertainty score0.400

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0760.141
Meta-epidemiology (narrow)0.0030.002
Meta-epidemiology (broad)0.0030.006
Bibliometrics0.0160.008
Science and technology studies0.0030.003
Scholarly communication0.0060.006
Open science0.0100.007
Research integrity0.0110.008
Insufficient payload (model declined to judge)0.0080.005

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 head0.303
GPT teacher head0.415
Teacher spread0.112 · 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 designSystematic review
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
Published2016
Admission routes1
Has abstractyes

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Same venuePLoS ONESame topicGenetics and Neurodevelopmental DisordersFrench-language works237,207