The Brain in Oral Clefting: A Systematic Review With Meta-Analyses

Sándor-Bajusz, Kinga Amália; Sadi, Asaad; Varga, Eszter; Csábi, Györgyi; Antonoglou, Georgios N.; Lohner, Szimonetta

doi:10.3389/fnana.2022.863900

Cited by 3 publications

(1 citation statement)

References 62 publications

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“…It is often unclear whether syndromic participants were excluded from these studies (Gallagher and Collett, 2019), and may include a mixed population of both syndromic and non-syndromic forms (Nopoulos et al, 2000(Nopoulos et al, , 2002. Underlying genetic abnormalities-which are present in syndromic oral clefts-often affect proper brain development and function (McDonald-McGinn et al, 2015;Berg et al, 2016) and may therefore misrepresent the non-syndromic population (Rincic et al, 2016;Sándor-Bajusz et al, 2022).…”

Section: Introductionmentioning

confidence: 99%

Cognitive functioning and clinical characteristics of children with non-syndromic orofacial clefts: A case-control study

Sándor-Bajusz

Dergez²,

Molnár³

et al. 2023

Front. Psychol.

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IntroductionThe higher rate of neuropsychiatric disorders in individuals with non-syndromic orofacial clefts has been well documented by previous studies. Our goal was to identify children with non-syndromic orofacial clefts that are at risk for abnormal neurodevelopment by assessing their developmental history and present cognitive functioning.Materials and methodsA single-center, case-controlled study was carried out at the Department of Pediatrics of the University of Pécs in Hungary. The study consisted of three phases including questionnaires to collect retrospective clinical data and psychometric tools to assess IQ and executive functioning.ResultsForty children with non-syndromic oral clefts and 44 age-matched controls participated in the study. Apgar score at 5 min was lower for the cleft group, in addition to delays observed for potty-training and speech development. Psychiatric disorders were more common in the cleft group (15%) than in controls (4.5%), although not statistically significant with small effect size. The cleft group scored lower on the Continuous Performance Test. Subgroup analysis revealed significant associations between higher parental socio-economic status, academic, and cognitive performance in children with non-syndromic orofacial clefts. Analyzes additionally revealed significant associations between early speech and language interventions and higher scores on the Verbal Comprehension Index of the WISC-IV in these children.DiscussionChildren with non-syndromic orofacial clefts seem to be at risk for deficits involving the attention domain of the executive system. These children additionally present with difficulties that affect cognitive and speech development. Children with non-syndromic orofacial clefts show significant skill development and present with similar cognitive strengths as their peers. Longitudinal studies with larger sample sizes are needed to provide more conclusive evidence on cognitive deficits in children with non-syndromic orofacial clefts at risk for neurodevelopmental difficulties.

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Section: Introductionmentioning

confidence: 99%

Cognitive functioning and clinical characteristics of children with non-syndromic orofacial clefts: A case-control study

Sándor-Bajusz

Dergez²,

Molnár³

et al. 2023

Front. Psychol.

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Divergent growth of the transient brain compartments in fetuses with nonsyndromic isolated clefts involving the primary and secondary palate

Masse,

Brumfield,

Ahmad

et al. 2024

Cerebral Cortex

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Cleft lip/palate is a common orofacial malformation that often leads to speech/language difficulties as well as developmental delays in affected children, despite surgical repair. Our understanding of brain development in these children is limited. This study aimed to analyze prenatal brain development in fetuses with cleft lip/palate and controls. We examined in utero MRIs of 30 controls and 42 cleft lip/palate fetal cases and measured regional brain volumes. Cleft lip/palate was categorized into groups A (cleft lip or alveolus) and B (any combination of clefts involving the primary and secondary palates). Using a repeated-measures regression model with relative brain hemisphere volumes (%), and after adjusting for multiple comparisons, we did not identify significant differences in regional brain growth between group A and controls. Group B clefts had significantly slower weekly cerebellar growth compared with controls. We also observed divergent brain growth in transient brain structures (cortical plate, subplate, ganglionic eminence) within group B clefts, depending on severity (unilateral or bilateral) and defect location (hemisphere ipsilateral or contralateral to the defect). Further research is needed to explore the association between regional fetal brain growth and cleft lip/palate severity, with the potential to inform early neurodevelopmental biomarkers and personalized diagnostics.

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Longitudinal Speech Outcome at 5 and 10 Years in UCLP: Influence of Speech Therapy and Secondary Velopharyngeal Surgery

Persson,

Davies,

Havstam

et al. 2024

The Cleft Palate Craniofacial Journal

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Objective To investigate speech development of children aged 5 and 10 years with repaired unilateral cleft lip and palate (UCLP) and identify speech characteristics when speech proficiency is not at ‘peer level’ at 10 years. Estimate how the number of speech therapy visits are related to speech proficiency at 10 years, and what factors are predictive of whether a child's speech proficiency at 10 years is at ‘peer level’ or not. Design Longitudinal complete datasets from the Scandcleft project Participants 320 children from nine cleft palate teams in five countries, operated on with one out of four surgical methods. Interventions Secondary velopharyngeal surgery (VP-surgery) and number of speech therapy visits (ST-visits), a proxy for speech intervention. Main Outcome Measures ‘Peer level’ of percentage of consonants correct (PCC, > 91%) and the composite score of velopharyngeal competence (VPC-Sum, 0–1). Results Speech proficiency improved, with only 23% of the participants at ‘peer level’ at 5 years, compared to 56% at 10 years. A poorer PCC score was the most sensitive marker for the 44% below ‘peer level’ at 10-year-of-age. The best predictor of ‘peer level’ speech proficiency at 10 years was speech proficiency at 5 years. A high number of ST-visits received did not improve the probability of achieving ‘peer level’ speech, and many children seemed to have received excessive amounts of ST-visits without substantial improvement. Conclusions It is important to strive for speech at ‘peer level’ before age 5. Criteria for speech therapy intervention and for methods used needs to be evidence-based.

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The Brain in Oral Clefting: A Systematic Review With Meta-Analyses

Cited by 3 publications

References 62 publications

Cognitive functioning and clinical characteristics of children with non-syndromic orofacial clefts: A case-control study

Cognitive functioning and clinical characteristics of children with non-syndromic orofacial clefts: A case-control study

Divergent growth of the transient brain compartments in fetuses with nonsyndromic isolated clefts involving the primary and secondary palate

Longitudinal Speech Outcome at 5 and 10 Years in UCLP: Influence of Speech Therapy and Secondary Velopharyngeal Surgery

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