{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,9]],"date-time":"2026-10-09T05:52:00Z","timestamp":1791525120760,"version":"4.3.4"},"reference-count":32,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2026,10,9]],"date-time":"2026-10-09T00:00:00Z","timestamp":1791504000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Dent. Med."],"abstract":"<jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Mowat\u2013Wilson syndrome (MWS) is a rare genetic disorder characterized by intellectual disability, distinctive craniofacial features, behavioral impairments, and congenital anomalies. Although oral and craniofacial abnormalities are frequently reported, their impact on oral health-related quality of life (OHRQoL) has not been investigated. This study aimed to evaluate OHRQoL in children with MWS and compare it with peers with autism spectrum disorder (ASD) and neurotypical controls. Secondary analyses explored caregiver functional limitations and dental care characteristics.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>\n                      A cross-sectional questionnaire-based study was conducted involving caregivers of children aged 6\u201318 years with MWS (\n                      <jats:italic>n<\/jats:italic>\n                      \u2009=\u200919), ASD (\n                      <jats:italic>n<\/jats:italic>\n                      \u2009=\u200919), and neurotypical controls (\n                      <jats:italic>n<\/jats:italic>\n                      \u2009=\u200938). OHRQoL was assessed using the short-form Parental-Caregiver Perception Questionnaire (\n                      <jats:italic>P<\/jats:italic>\n                      -CPQ) and Family Impact Scale (FIS). Additional questions explored socioeconomic characteristics, dental care setting and attendance, and parental perceptions of oral health. Group comparisons were performed using non-parametric and parametric tests, with significance set at\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&amp;lt;\u20090.05.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      Children with MWS showed significantly higher total P-CPQ scores than neurotypical controls (37.11\u2009\u00b1\u20098.65 vs. 26.23\u2009\u00b1\u20095.05;\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&amp;lt;\u20090.001), indicating poorer OHRQoL. The greatest impairments were observed in the functional limitation domain, particularly regarding chewing difficulties, mouth breathing, slower eating, and sleep disturbances (all\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&amp;lt;\u20090.001). Halitosis and food impaction on the palate were also significantly more frequent in the MWS group. Parents of children with MWS reported a greater impact of oral conditions on wellbeing than both ASD and neurotypical groups (\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&amp;lt;\u20090.05). Although OHRQoL impairment was comparable between MWS and ASD participants, ASD was associated with greater family burden, reflected by higher FIS scores (16.42\u2009\u00b1\u20097.09 vs. 21.58\u2009\u00b1\u20094.85;\n                      <jats:italic>p<\/jats:italic>\n                      \u2009=\u20090.016). Both MWS and ASD groups exhibited higher combined P-CPQ\u2009+\u2009FIS scores than neurotypical controls (\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&amp;lt;\u20090.01).\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>Both MWS and ASD were associated with poorer OHRQoL than neurotypical status, although distinct patterns of impairment emerged. MWS was primarily characterized by functional oral limitations and a greater impact of oral conditions on wellbeing, whereas ASD was associated with greater psychosocial and family burden. These findings support the existence of a syndrome-specific profile of caregiver-perceived unmet oral health needs and functional limitations in children with complex healthcare conditions and emphasize the importance of individualized, multidisciplinary dental care strategies.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.3389\/fdmed.2026.1953280","type":"journal-article","created":{"date-parts":[[2026,10,9]],"date-time":"2026-10-09T05:33:24Z","timestamp":1791524004000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":0,"title":["Caregiver-reported oral health-related quality of life in children with Mowat\u2013Wilson syndrome: a comparative study"],"prefix":"10.3389","volume":"7","author":[{"given":"Silvia","family":"Cirio","sequence":"first","affiliation":[{"name":"Department of Biomedical, Surgical and Dental Sciences, University of Milan","place":["Milan, Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Claudia","family":"Salerno","sequence":"additional","affiliation":[{"name":"Department of Biomedical, Surgical and Dental Sciences, University of Milan","place":["Milan, Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aesha","family":"Allam","sequence":"additional","affiliation":[{"name":"Department of Biomedical, Surgical and Dental Sciences, University of Milan","place":["Milan, Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Giulia","family":"Zambon","sequence":"additional","affiliation":[{"name":"Department of Biomedical, Surgical and Dental Sciences, University of Milan","place":["Milan, Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Araxi","family":"Balian","sequence":"additional","affiliation":[{"name":"Dental and Stomatology Unit, Surgical Area Department, Cittadella Hospital","place":["Cittadella, Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maria Grazia","family":"Cagetti","sequence":"additional","affiliation":[{"name":"Department of Biomedical, Surgical and Dental Sciences, University of Milan","place":["Milan, Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2026,10,9]]},"reference":[{"key":"B1","first-page":"431","article-title":"Early childhood caries and quality of life: child and parent perspectives","volume":"25","author":"Filstrup","year":"2003","journal-title":"Pediatr Dent"},{"key":"B2","doi-asserted-by":"publisher","first-page":"4407","DOI":"10.3390\/ijerph17124407","article-title":"The effect of dental treatment under general anesthesia on quality of life and growth and blood chemistry parameters in uncooperative pediatric patients with compromised oral health: a pilot 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