Evidence map›Paper›PMID 42420566›Full record

ArticleHuman genetics2026

Implementation of exome sequencing for rare undiagnosed diseases in LMICs: the G2MC rare diseases exome sequencing pilot project.

Yasas D Kolambage, Claudia Gonzaga-Jauregui, Guillermo Lay-Son, Rupesh Mishra, Njabulo Christian Mabaso, Sok-Kun Tae, Kristin A Maloney, Carolina I Galaz-Montoya, Dineshani Hettiarachchi, Bronwyn Dillon and 9 more

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Article in Human genetics, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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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

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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

19 authors.

Yasas D Kolambage *Department of Anatomy, Genetics and Biomedical Informatics, Faculty of Medicine, Centre for Genetics and Genomics, University of Colombo, Colombo, Sri Lanka. yasas@med.sab.ac.lk.ORCID https://orcid.org/0000-0002-5066-9840
Claudia Gonzaga-Jauregui *Laboratorio Internacional de Investigación Sobre el Genoma Humano, International Laboratory for Human Genome Research, Universidad Nacional Autónoma de México, Querétaro, México.
Guillermo Lay-SonGenetics Section, Division of Pediatrics, School of Medicine, Pontificia Universidad Católica de Chile, Santiago, Chile.
Rupesh MishraCivil Service Hospital, Kathmandu, Nepal.
Njabulo Christian MabasoDivision of Human Genetics, National Health Laboratory Service and School of Pathology, Faculty of Health Sciences, University of the Witwatersrand, Johannesburg, South Africa.
Sok-Kun TaeDepartment of Paediatrics, Faculty of Medicine, University of Malaya, Kuala Lumpur, Malaysia.
Kristin A MaloneyUniversity of Maryland School of Medicine, Baltimore, MD, USA.
Carolina I Galaz-MontoyaEscuela de Medicina, Ciencias de La Salud, Universidad de Sonora, Hermosillo, Sonora, Mexico.
Dineshani HettiarachchiDepartment of Anatomy, Genetics and Biomedical Informatics, Faculty of Medicine, Centre for Genetics and Genomics, University of Colombo, Colombo, Sri Lanka.
Bronwyn DillonDivision of Human Genetics, National Health Laboratory Service and School of Pathology, Faculty of Health Sciences, University of the Witwatersrand, Johannesburg, South Africa.
Nilam ThakurBir Hospital, Kathmandu, Nepal.
Ludivine de MentenGlobal Genomic Medicine Collaborative, Salt Lake City, UT, USA.
Cecilia MelladoGenetics Section, Division of Pediatrics, School of Medicine, Pontificia Universidad Católica de Chile, Santiago, Chile.
Carol L GreeneUniversity of Maryland School of Medicine, Baltimore, MD, USA.
Toni I PollinUniversity of Maryland School of Medicine, Baltimore, MD, USA.
Amanda KrauseDivision of Human Genetics, National Health Laboratory Service and School of Pathology, Faculty of Health Sciences, University of the Witwatersrand, Johannesburg, South Africa.
Meow-Keong ThongDepartment of Paediatrics, Faculty of Medicine, University of Malaya, Kuala Lumpur, Malaysia.
Alan R Shuldiner *University of Maryland School of Medicine, Baltimore, MD, USA.
Vajira H W Dissanayake *Department of Anatomy, Genetics and Biomedical Informatics, Faculty of Medicine, Centre for Genetics and Genomics, University of Colombo, Colombo, Sri Lanka.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Rare undiagnosed diseases impose a substantial burden on patients, families, and health systems. Collectively they affect an estimated 300-350 million people worldwide. Of these, approximately 250 million live in low- and middle-income countries (LMICs). Many rare diseases have a genetic basis and can be diagnosed using molecular testing. However, patients in LMICs often lack access to advanced diagnostics and specialist care, leading to prolonged diagnostic odysseys and significant distress. To address this gap, the Global Genomic Medicine Collaborative (G2MC) implemented a Rare Disease Pilot Project to enable clinical genomic testing at six LMIC sites (Chile, Malaysia, Mexico, Nepal, South Africa, Sri Lanka). Using trio exome sequencing for 18 selected families, supplemented by chromosomal microarray as indicated, definitive molecular diagnoses were established in 5 families (pathogenic/likely pathogenic variants explaining the phenotype). An additional 8 families had suspected molecular diagnoses with strong phenotype correlation that require functional validation, while 5 families remained undiagnosed at the end of the pilot. The integration of genomic data into clinical decision-making enabled definitive diagnoses, precise management, and genetic counseling for these patients. The pilot also strengthened local capacity building by conducting cross-site case conferences and enabling local reanalysis of sequence data at sites with bioinformatics capabilities. This study demonstrates the feasibility, effectiveness, and adaptability of implementing exome sequencing in resource-limited settings and highlights its potential to transform rare disease diagnosis and care in LMICs.

Indexed as

ExomeExome SequencingRare DiseasesUndiagnosed DiseasesDeveloping CountriesFemaleGenetic TestingHumansMalePilot Projects

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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.