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Record W4318916705 · doi:10.1016/j.gim.2022.11.012

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

2023· review· en· W4318916705 on OpenAlexafffund
Erik Boot, Sólveig Óskarsdóttir, Joanne C. Y. Loo, T. Blaine Crowley, Ani Orchanian‐Cheff, Danielle M. Andrade, Jill M. Arganbright, René M. Castelein, Christine Cserti‐Gazdewich, Steven de Reuver, Ania Fiksinski, Gunilla Klingberg, Anthony E. Lang, Maria R. Mascarenhas, Edward Moss, Beata Nowakowska, Erwin Oechslin, Lisa D. Palmer, Gabriela M. Repetto, Nikolai Gil D. Reyes, Maude Schneider, Candice Silversides, Kathleen E. Sullivan, Ann Swillen, Thérèse van Amelsvoort, Jason Van Batavia, Claudia Vingerhoets, Donna M. McDonald‐McGinn, Anne S. Bassett

Bibliographic record

VenueGenetics in Medicine · 2023
Typereview
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicCongenital heart defects research
Canadian institutionsCentre for Addiction and Mental HealthMount Sinai HospitalToronto Western HospitalUniversity of TorontoToronto General HospitalUniversity Health Network
FundersUniversity of Toronto
KeywordsDeletion syndromeMedicineClinical PracticeFamily medicineGeneticsBiologyPhenotypeGene

Abstract

fetched live from OpenAlex

This review aimed to update the clinical practice guidelines for managing adults with 22q11.2 deletion syndrome (22q11.2DS). The 22q11.2 Society recruited expert clinicians worldwide to revise the original clinical practice guidelines for adults in a stepwise process according to best practices: (1) a systematic literature search (1992-2021), (2) study selection and synthesis by clinical experts from 8 countries, covering 24 subspecialties, and (3) formulation of consensus recommendations based on the literature and further shaped by patient advocate survey results. Of 2441 22q11.2DS-relevant publications initially identified, 2344 received full-text review, with 2318 meeting inclusion criteria (clinical care relevance to 22q11.2DS) including 894 with potential relevance to adults. The evidence base remains limited. Thus multidisciplinary recommendations represent statements of current best practice for this evolving field, informed by the available literature. These recommendations provide guidance for the recognition, evaluation, surveillance, and management of the many emerging and chronic 22q11.2DS-associated multisystem morbidities relevant to adults. The recommendations also address key genetic counseling and psychosocial considerations for the increasing numbers of adults with this complex condition.

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 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)0.002
metaresearch head score (Gemma)0.001
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesMeta-epidemiology (narrow)
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Not applicable · Consensus signal: none
GenreCandidate signal: Review · Consensus signal: Review
Teacher disagreement score0.982
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0020.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0000.001
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.001
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 head0.136
GPT teacher head0.498
Teacher spread0.361 · 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 teacher head, not a consensus.

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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