DNAH10 encodes dynein axonemal heavy chain 10, a critical component of inner arm dynein complexes in motile cilia and sperm flagella that generates force for ciliary and flagellar motility 1. The protein functions as a minus-end-directed microtubule motor with ATPase activity, producing force through power strokes upon ADP release 1. DNAH10 plays essential roles in sperm flagellar assembly and motility, with evidence suggesting it coordinates sperm head and flagella development during spermiogenesis by interacting with the UCHL3-PACRG complex to facilitate manchette function and proper axonemal protein localization 2. Bi-allelic mutations in DNAH10 cause multiple morphological abnormalities of sperm flagella (MMAF), characterized by absent or shortened flagella, inner dynein arm defects, and severe asthenoteratozoospermia leading to male infertility 345. Additionally, DNAH10 deficiency causes primary ciliary dyskinesia (PCD) with symptoms including sinusitis, bronchiectasis, situs inversus, and hydrocephalus, as demonstrated in both human patients and mouse models 1. The gene also shows associations with metabolic traits, being implicated in adipocyte differentiation and insulin resistance pathways 67. Clinically, patients with DNAH10 mutations can achieve favorable reproductive outcomes through intracytoplasmic sperm injection (ICSI) 3.