Hosoe J, Miya F, Kadowaki H, et al. - In the present study, the researchers sought to examine the clinical utility of a comprehensive genetic testing system consisting of targeted next-generation sequencing (NGS) with phenotype-driven bioinformatics analysis in patients with monogenic diabetes that uses patient genotypic and phenotypic data to prioritize potentially causal variants. A targeted NGS of 383 genes associated with monogenic diabetes or common forms of diabetes was performed in 13 Japanese patients with suspected (n = 10) or previously diagnosed (n = 3) monogenic diabetes or severe insulin resistance. The authors diagnosed 3 patients as subtypes of monogenic diabetes due to disease-associated variants of INSR, LMNA, and HNF1B among the patients suspected having monogenic diabetes or insulin resistance. This comprehensive approach might aid in the early diagnosis of patients with monogenic diabetes and promote the provision of tailored therapy.