BackgroundChildren with autism spectrum disorder (ASD) face unique challenges related to oral health, which can negatively impact their day‐to‐day lives, significantly compromising their overall quality of life. Primary caregivers of children with ASD have a critical role in delivering and seeking oral health care. Hence, it is vital to study their perspective towards their children's oral health and its impact on their quality of life.AimTo explore the parental perception of oral health‐related quality of life in children with autism.MethodsA systematic electronic and manual search was conducted in Medline (via PubMed), Embase, Google Scholar, Scopus, and LILACS of articles published from January 2003 to May 2023 using appropriate MeSH terms, keywords, and other terms. A four‐phase study selection process was followed according to PRISMA guidelines, and data extraction and synthesis were performed using an extraction form. The selected studies were critically appraised using the QATSDD and Crombie's assessment tool. The inter‐reviewer agreement was assessed using the kappa with a linear weighting coefficient.ResultsOut of the 885 results, 15 studies were included in the review after the two selection phases, and the study characteristics were summarized in tabular form. Study quality varied considerably, and out of a total possible QATSDD score of 42, scores for the individual studies ranged from 14 to 40. The risk of bias for the seven criteria was found to be low.ConclusionThe parental perception of the OHRQoL in children with ASD is poor, and the most significant perceived impact is on the child's functional and social well‐being aspects. Parental‐Caregiver Perception Questionnaire was the most commonly used. The most frequent symptoms include bad breath, food lodgment, mouth breathing and night grinding. The familial impact and influence on the OHRQoL of siblings were also studied.