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Background: Little is known about father's involvement in the care of children born with perinatal risk factors. This study aimed to understand father's involvement in the care of children born preterm, low birth weight (LBW) and/or with hypoxic ischemic encephalopathy (HIE) in rural Rwanda and assess child and home environment factors associated with father involvement. Methods: A cross‐sectional study of children born preterm, LBW or with HIE who were discharged from Kirehe District Hospital neonatal unit from May 2015 to April 2016 and those enrolled in a neonatal unit follow‐up programme from May 2016 to November 2017. Interviews were conducted when the children were ages 24–47 months in the child's home. Primary caregivers reported on father involvement in parenting, home environment, child disability, and child development outcomes. Children's nutritional status were directly measured. Only children whose fathers were living in the home were included in the sample. Bivariate analyses were conducted using Fisher's exact test and Wilcoxon Rank Sum test. Results: A total of 236 children aged 24–47 months were included in this study, 66.4% were born preterm or LBW with a mean age of 33.3 months. 73.5% of children were at risk of disability and 77.7% had potential delay in overall child development. 15.5% of fathers reported engaging in four or more activities with their child in the last 3 days. Factors associated with father involvement included smaller household size (p = 0.004), mother engaged in decision‐making (p = 0.027), being on‐track in developmental milestones for problem solving (p = 0.042) and mother's involvement in learning activities (p = 0.043); the number of activities a father engaged in was significantly associated with the child's overall development (p = 0.032). Conclusion: We found that father involvement in activities to support learning was low amongst children born preterm/LBW and/or with HIE. Programme interventions should encourage fathers to engage with their children given the benefits for children's development. [ABSTRACT FROM AUTHOR]