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Record W4318928085 · doi:10.1016/j.gim.2022.11.006

Updated clinical practice recommendations for managing children with 22q11.2 deletion syndrome

2023· review· en· W4318928085 on OpenAlexafffund
Sólveig Óskarsdóttir, Erik Boot, T. Blaine Crowley, Joanne C. Y. Loo, Jill M. Arganbright, Marco Armando, Adriane L. Baylis, Elemi Breetvelt, René M. Castelein, Madeline Chadehumbe, Christopher M. Cielo︎, Steven de Reuver, Stéphan Eliez, Ania Fiksinski, Brian J. Forbes, Emily R. Gallagher, Sarah Hopkins, Oksana A. Jackson, Lorraine Levitz-Katz, Gunilla Klingberg, Michele P. Lambert, Bruno Marino, Maria R. Mascarenhas, Julie S. Moldenhauer, Edward Moss, Beata Nowakowska, Ani Orchanian‐Cheff, Carolina Putotto, Gabriela M. Repetto, Erica Schindewolf, Maude Schneider, Cynthia Solot, Kathleen E. Sullivan, Ann Swillen, Marta Unolt, Jason Van Batavia, Claudia Vingerhoets, Jacob Vorstman, Anne S. Bassett, Donna M. McDonald‐McGinn

Bibliographic record

VenueGenetics in Medicine · 2023
Typereview
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicCongenital heart defects research
Canadian institutionsCentre for Addiction and Mental HealthHospital for Sick ChildrenUniversity of TorontoUniversity Health Network
FundersUniversity of Toronto
KeywordsExpert opinionMultidisciplinary approachPsychosocialMEDLINEClinical PracticeMedicineRelevance (law)Data extractionSystematic reviewFamily medicineMedical educationPsychologyPsychiatry

Abstract

fetched live from OpenAlex

This review aimed to update the clinical practice guidelines for managing children and adolescents with 22q11.2 deletion syndrome (22q11.2DS). The 22q11.2 Society, the international scientific organization studying chromosome 22q11.2 differences and related conditions, recruited expert clinicians worldwide to revise the original 2011 pediatric clinical practice guidelines in a stepwise process: (1) a systematic literature search (1992-2021), (2) study selection and data extraction by clinical experts from 9 different countries, covering 24 subspecialties, and (3) creation of a draft consensus document based on the literature and expert opinion, which was further shaped by survey results from family support organizations regarding perceived needs. Of 2441 22q11.2DS-relevant publications initially identified, 2344 received full-text reviews, including 1545 meeting criteria for potential relevance to clinical care of children and adolescents. Informed by the available literature, recommendations were formulated. Given evidence base limitations, multidisciplinary recommendations represent consensus statements of good practice for this evolving field. These recommendations provide contemporary guidance for evaluation, surveillance, and management of the many 22q11.2DS-associated physical, cognitive, behavioral, and psychiatric morbidities while addressing important genetic counseling and psychosocial issues.

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)0.006
metaresearch head score (Gemma)0.015
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: none
GenreCandidate signal: Review · Consensus signal: Review
Teacher disagreement score0.007
Threshold uncertainty score0.031

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0060.015
Meta-epidemiology (narrow)0.0010.001
Meta-epidemiology (broad)0.0030.002
Bibliometrics0.0070.005
Science and technology studies0.0010.001
Scholarly communication0.0020.002
Open science0.0030.001
Research integrity0.0040.004
Insufficient payload (model declined to judge)0.0060.003

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 head0.140
GPT teacher head0.499
Teacher spread0.359 · 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

Citations88
Published2023
Admission routes2
Has abstractyes

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