Evidence map›Paper›PMID 38711434›Full record

ArticleArXiv2024

GestaltMML: Enhancing Rare Genetic Disease Diagnosis through Multimodal Machine Learning Combining Facial Images and Clinical Texts.

Da Wu, Jingye Yang, Cong Liu, Tzung-Chien Hsieh, Elaine Marchi, Justin Blair, Peter Krawitz, Chunhua Weng, Wendy Chung, Gholson J Lyon and 3 more

Abstract readPreprint
In one paragraph

Article in ArXiv, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

0numbers the graph read from it
0cells of the map it votes in
0citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

0 citing papers in PubMed.

No citing paper in PubMed yet.

4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

13 authors.

Da WuRaymond G. Perelman Center for Cellular and Molecular Therapeutics, Children's Hospital of Philadelphia, Philadelphia, PA 19104, USA.
Jingye YangRaymond G. Perelman Center for Cellular and Molecular Therapeutics, Children's Hospital of Philadelphia, Philadelphia, PA 19104, USA.
Cong LiuDepartment of Biomedical Informatics, Columbia University Irving Medical Center, New York, NY 10032, USA.
Tzung-Chien HsiehInstitute for Genomic Statistics and Bioinformatics, University Hospital Bonn, Rheinische Friedrich-Wilhelms-Universität Bonn, Bonn, Germany.
Elaine MarchiDepartment of Human Genetics, New York State Institute for Basic Research in Developmental Disabilities, Staten Island, NY, USA.
Justin BlairDivision of Human Genetics, Children's Hospital of Philadelphia, Philadelphia, PA 19104, USA.
Peter KrawitzInstitute for Genomic Statistics and Bioinformatics, University Hospital Bonn, Rheinische Friedrich-Wilhelms-Universität Bonn, Bonn, Germany.
Chunhua WengDepartment of Biomedical Informatics, Columbia University Irving Medical Center, New York, NY 10032, USA.
Wendy ChungDepartment of Pediatrics, Boston Children's Hospital, Harvard Medical School, Boston, MA, USA.
Gholson J LyonDepartment of Human Genetics, New York State Institute for Basic Research in Developmental Disabilities, Staten Island, NY, USA.
Ian D KrantzDivision of Human Genetics, Children's Hospital of Philadelphia, Philadelphia, PA 19104, USA.
Jennifer M KalishDivision of Human Genetics, Children's Hospital of Philadelphia, Philadelphia, PA 19104, USA.
Kai WangRaymond G. Perelman Center for Cellular and Molecular Therapeutics, Children's Hospital of Philadelphia, Philadelphia, PA 19104, USA.

Funding

The Intellectual and Developmental Disabilities Research Center (IDDRC) at CHOP/PennP50HD105354 · NICHD · CHILDREN'S HOSP OF PHILADELPHIA · PI ERIC D MARSH, ROBERT Thomas SCHULTZ · 2021 to 2026
$9.2M
RESCUE: Rare Disease Detection and Escalation Support via a Learning Health SystemR01HG012655 · NHGRI · COLUMBIA UNIVERSITY HEALTH SCIENCES · PI Cong Liu · 2022 to 2026
$4.1M
Fair Phenotype Annotation and Genomic ReinterpretationR01HG013031 · NHGRI · COLUMBIA UNIVERSITY HEALTH SCIENCES · PI Wendy K Chung, CHUNHUA WENG · 2023 to 2026
$3.5M
Amino-terminal acetylation of proteins in mammalian biology and diseaseR35GM133408 · NIGMS · INSTITUTE FOR BASIC RES IN DEV DISABIL · PI LYON, GHOLSON · 2019 to 2023
$2.4M
NHGRI NIH HHS R01 HG012655NHGRI NIH HHS R01 HG013031NICHD NIH HHS P50 HD105354NIGMS NIH HHS R35 GM133408
6 · The paper itself

Abstract

Individuals with suspected rare genetic disorders often undergo multiple clinical evaluations, imaging studies, laboratory tests and genetic tests, to find a possible answer over a prolonged period of time. Addressing this "diagnostic odyssey" thus has substantial clinical, psychosocial, and economic benefits. Many rare genetic diseases have distinctive facial features, which can be used by artificial intelligence algorithms to facilitate clinical diagnosis, in prioritizing candidate diseases to be further examined by lab tests or genetic assays, or in helping the phenotype-driven reinterpretation of genome/exome sequencing data. Existing methods using frontal facial photos were built on conventional Convolutional Neural Networks (CNNs), rely exclusively on facial images, and cannot capture non-facial phenotypic traits and demographic information essential for guiding accurate diagnoses. Here we introduce GestaltMML, a multimodal machine learning (MML) approach solely based on the Transformer architecture. It integrates facial images, demographic information (age, sex, ethnicity), and clinical notes (optionally, a list of Human Phenotype Ontology terms) to improve prediction accuracy. Furthermore, we also evaluated GestaltMML on a diverse range of datasets, including 528 diseases from the GestaltMatcher Database, several in-house datasets of Beckwith-Wiedemann syndrome (BWS, over-growth syndrome with distinct facial features), Sotos syndrome (overgrowth syndrome with overlapping features with BWS), NAA10-related neurodevelopmental syndrome, Cornelia de Lange syndrome (multiple malformation syndrome), and KBG syndrome (multiple malformation syndrome). Our results suggest that GestaltMML effectively incorporates multiple modalities of data, greatly narrowing candidate genetic diagnoses of rare diseases and may facilitate the reinterpretation of genome/exome sequencing data.

Indexed as

Artificial IntelligenceFacial phenotypingHuman Phenotype OntologyLarge Language ModelsMultimodal Machine LearningRare Genetic Disorders

Identifiers

PMID38711434
PMCPMC11071539

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Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.