Homozygosity Mapping on Homozygosity Haplotype Analysis to Detect Recessive Disease-Causing Genes from a Small Number of Unrelated, Outbred Patients
2011

Homozygosity Mapping to Identify Disease-Causing Genes

Sample size: 60 publication Evidence: high

Author Information

Author(s): Hagiwara Koichi, Morino Hiroyuki, Shiihara Jun, Tanaka Tomoaki, Miyazawa Hitoshi, Suzuki Tomoko, Kohda Masakazu, Okazaki Yasushi, Seyama Kuniaki, Kawakami Hideshi

Primary Institution: Saitama Medical University

Hypothesis

Can homozygosity mapping on homozygosity haplotype analysis effectively identify recessive disease-causing genes from a small number of unrelated patients?

Conclusion

The study demonstrates that homozygosity mapping on homozygosity haplotype analysis is a powerful technique for identifying disease-causing genes in recessive conditions using a small number of patients.

Supporting Evidence

  • The analysis accurately identified the disease-causing gene locus from data of any 2 patients.
  • Using synthetic data, the analysis performed well for IBD fragments derived from a most recent common ancestor who existed less than 60 generations ago.
  • The method is effective for identifying recessive genes in small populations.

Takeaway

Researchers found a way to identify genes that cause diseases by looking at DNA from just a few unrelated patients, which can help find rare diseases faster.

Methodology

The study used homozygosity mapping combined with haplotype analysis to identify disease-causing genes in patients with Siiyama-type α1-antitrypsin deficiency.

Limitations

The method is unsuitable for genes with a frequency in the general population greater than 0.1.

Participant Demographics

The study involved 6 unrelated patients with Siiyama-type α1-antitrypsin deficiency and 54 healthy subjects.

Digital Object Identifier (DOI)

10.1371/journal.pone.0025059

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