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Background: Long-term mental health support is not routinely offered to families of neonatal intensive care unit (NICU) graduates treated for non-cardiac congenital anomalies. This may reflect the paucity of evidence-based guidance for clinical practice in these diagnostic groups.Objective: This systematic review aimed to explore long-term mental health outcomes of NICU graduates and their parents, following treatment for non-cardiac congenital anomalies.Methods: A systematic review of empirical literature was conducted for the following diagnoses: congenital diaphragmatic hernia, oesophageal atresia, gastroschisis, exomphalos, Pierre Robin sequence, and Vein of Galen aneurysmal malformations. Embase, MEDLINE, and PsycINFO were comprehensively searched to identify relevant articles. Risk of bias assessments were conducted using the QualSyst quality assessment tool for articles meeting the following inclusion criteria: English- language articles published in peer-reviewed journals on or before 9 May 9 2025, that used validated quantitative mental health measures.Results: Of the 45 included articles, 38 examined child mental health and 14 investigated parental mental health (seven studies explored both child and parent mental health). Twenty-six measures were used, most commonly the Child Behavior Checklist for child mental health and the Short Form Health Survey for parental mental health. Although methodological heterogeneity yielded conflicting results, overall, the evidence summarised suggested both the child and parents were at an increased risk of long-term mental health difficulties.Conclusion: The evidence presented in this review highlights the value of incorporating routine mental health follow-up into the standard clinical care of children and families affected by congenital anomalies.