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Record W4406971677 · doi:10.1212/wnl.0000000000209779

Characterization of Factors Associated With Death in Deceased Patients With Mitochondrial Disorders

2025· article· en· W4406971677 on OpenAlexaff
Alina Ivaniuk, Irina Anselm, Aaron B. Bowen, Bruce H. Cohen, Fatma Tuba Eminoğlu, Jane Estrella, Renata C. Gallagher, Rebecca Ganetzky, Jennifer Gannon, Gráinne S. Gorman, Carol L. Greene, Andrea Gropman, Richard Haas, Michio Hirano, Seema Kapoor, Amel Karaa, Mary Kay Koenig, Cornelia Kornblum, Engi̇n Köse, Austin Larson, Uta Lichter‐Konecki, Piervito Lopriore, Michelangelo Mancuso, Robert McFarland, Aye Moe, Éva Morava, Yi Shiau Ng, Russell P. Saneto, Fernando Scaglia, Carolyn M. Sue, Mark A. Tarnopolsky, Melissa Walker, Sumit Parikh, Cheuk Wing Fung, Tsz-sum Wong, Kiran Belaramani, Chun-kong Chan, Wing-ki Chan, Wai-lun Larry Chan, H. M. E. Cheung, Ka-yin Cheung, Richard Shek‐kwan Chang, Sing-ngai Cheung, Tsz-Fung Cheung, Yuk-fai Cheung, Shuk-ching Josephine Chong, Chi-kwan Jasmine Chow, Brian Hon‐Yin Chung, Florence Fan, Wai-ming Joshua Fok, Ka-wing Fong, Tsui-hang Sharon Fung, Kwok-fai Hui, Ting-hin Hui, Joannie Hui, Chun Hung Ko, Min-chung Kwan, Anne Mei Kwun Kwok, Sung-shing Jeffrey Kwok, Moon-sing Lai, Yau-on Lam, Ching‐Wan Lam, Ming-chung Lau, Chun Yiu Law, Hiu-Fung Law, Wing‐Cheong Lee, Han‐Chih Hencher Lee, Kin-hang Leung, Kit-yan Leung, Siu-hung Li, Tsz-ki Ling, Kam Tim Liu, Ivan F. M. Lo, Colin H. T. Lui, Ching-on Luk, Ho‐Ming Luk, Che-Kwan Ma, Karen Ma, Kam-hung Ma, Yuen-ni Mew, Alex Mo, Sui-Fun Hg, Grace Wing-kit Poon, Bun Sheng, Cheuk-ling Charing Szeto, Shuk‐Mui Tai, Choi-ting Alan Tse, Li-yan Lilian Tsung, Ho-ming June Wong, Wing-yin Winnie Wong, Kwok-kui Wong, Suet-na Sheila Wong, Chun-nei Virginia Wong, Wai-shan Sammy Wong, Chi-kin Felix Wong, Shun-Ping Wu, Hiu-fung Jerome Wu, Man-Mut Yau, Eric Kin Cheong Yau, Wai Lan Yeung, Hon‐Ming Jonas Yeung, Kin-keung Edwin Yip, Huijun Wu, Pui-hong Terence Young, Yuet‐Ping Yuen, Chi-lap Yuen

Bibliographic record

VenueNeurology · 2025
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicMitochondrial Function and Pathology
Canadian institutionsMcMaster Children's Hospital
Fundersnot available
KeywordsMitochondrial diseaseMedicineMitochondrial DNABiologyGenetics

Abstract

fetched live from OpenAlex

BACKGROUND AND OBJECTIVES: Mitochondrial disorders are multiorgan disorders resulting in significant morbidity and mortality. We aimed to characterize death-associated factors in an international cohort of deceased individuals with mitochondrial disorders. METHODS: This cross-sectional multicenter observational study used data provided by 26 mitochondrial disease centers from 8 countries from January 2022 to March 2023. Individuals with genetically confirmed mitochondrial disorders were included, along with patients with clinically or genetically diagnosed Leigh syndrome. Collected data included demographic and genetic diagnosis variables, clinical phenotype, involvement of organs and systems, conditions leading to death, and supportive care. We defined pediatric and adult groups based on age at death before or after 18 years, respectively. We used Kruskal-Wallis with post hoc Dunn test with Bonferroni correction and Fisher exact test for comparisons, Spearman rank test for correlations, and multiple linear regression for multivariable analysis. RESULTS: < 0.001). On multivariate analysis, individuals with movement disorders and nuclear gene involvement had increased odds of any respiratory support use (OR 2.42 (95% CI 1.17-5.22) and OR 2.39 (95% CI 1.16-5.07), respectively). DISCUSSION: This international collaboration highlights the importance of respiratory care and infection management and provides a reference for prognostication across different mitochondrial disorders.

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)0.001
metaresearch head score (Gemma)0.003
Version: metacan-v3-hybrid-931329e0061cValidation 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 score0.003
Threshold uncertainty score0.005

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0010.003
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0010.001
Science and technology studies0.0000.000
Scholarly communication0.0010.000
Open science0.0000.001
Research integrity0.0000.001
Insufficient payload (model declined to judge)0.0010.000

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 head0.006
GPT teacher head0.204
Teacher spread0.198 · 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 designObservational
Domainnot available
GenreEmpirical

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

Citations5
Published2025
Admission routes1
Has abstractyes

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