Cleft Palate Population Research Articles

Surgical treatment of velopharyngeal dysfunction: Incidence and associated factors in the Swedish cleft palate population

IntroductionSpeech in children with cleft palate can be affected by velopharyngeal dysfunction, which persists after primary palate repair. The incidence of surgery to correct velopharyngeal dysfunction in this patient group has previously been reported as 2.6–37%. We aimed to investigate the incidence of velopharyngeal dysfunction surgery in Swedish children with cleft palate and to examine potential associations of independent variables with this incidence. MethodsIn this cohort study, we analysed data from the Swedish cleft lip and palate quality registry for 1093 children with cleft palate with or without cleft lip. Kaplan–Meier analysis was used to estimate the risk of having velopharyngeal dysfunction surgery. Multivariable Cox proportional hazards models were used to estimate the associated effect of cleft subtype, additional diagnoses, gender, and age at and number of stages for primary palate repair on the primary outcome. ResultsThe risk of having velopharyngeal dysfunction surgery was 25.6%. Complete primary palate repair after the age of 18 months or in more than one stage was associated with a higher risk, but it could not be determined which of these was the more significant factor. Cleft soft palate was associated with a significantly lower risk than other cleft subtypes. ConclusionsPrimary palate repair at a higher age or in more than one stage may increase the risk of having velopharyngeal dysfunction surgery. Further analysis of potential unknown confounding factors and the association between the incidence of velopharyngeal dysfunction and surgery to correct this condition is needed.

Early special educational needs provision and its impact on unplanned hospital utilisation and school absences in children with isolated cleft lip and/or palate: a demonstration target trial emulation study protocol using ECHILD.

Special educational needs (SEN) provision is designed to help pupils with additional educational, behavioural or health needs; for example, pupils with cleft lip and/or palate may be offered SEN provision to improve their speech and language skills. Our aim is to contribute to the literature and assess the impact of SEN provision on health and educational outcomes for a well-defined population. We will use the ECHILD database, which links educational and health records across England. Our target population consists of children identified within ECHILD to have a specific congenital anomaly: isolated cleft lip and/or palate. We will apply a trial emulation framework to reduce biases in design and analysis of observational data to investigate the causal impact of SEN provision (including none) by the start of compulsory education (Year One - age five year on entry) on the number of unplanned hospital utilisation and school absences by the end of primary education (Year Six - age ten/eleven). We will use propensity score-based estimators (inverse probability weighting (IPW) and IPW regression adjustment IPW) to compare categories of SEN provision in terms of these outcomes and to triangulate results obtained using complementary estimation methods (Naïve estimator, multivariable regression, parametric g-formula, and if possible, instrumental variables), targeting a variety of causal contrasts (average treatment effect/in the treated/in the not treated) of SEN provision. This study will evaluate the impact of reasonable adjustments at the start of compulsory education on health and educational outcomes in the isolated cleft lip and palate population by triangulating complementary methods under a target-trial framework.

Healthcare use and direct medical costs in a cleft lip and palate population: an analysis of observed and protocolized care and costs

This study was performed to describe observed healthcare utilization and medical costs for patients with a cleft, compare these costs to the expected costs based on the treatment protocol, and explore the additional costs of implementing the International Consortium for Health Outcomes Measurement (ICHOM) Standard Set for Cleft Lip and Palate (CL/P). Forty patients with unilateral CL/P between 0 and 24 years of age, treated between 2012 and 2019 at Erasmus University Medical Center, were included. Healthcare services (consultations, diagnostic and surgical procedures) were counted and costs were calculated. Expected costs based on the treatment protocol were calculated by multiplying healthcare products by the product prices. Correspondingly, the additional expected costs after implementing the ICHOM Standard Set (protocol + ICHOM) were calculated. Observed costs were compared with protocol costs, and the additional expected protocol + ICHOM costs were described. The total mean costs were highest in the first year after birth (€5596), mainly due to surgeries. The mean observed total costs (€40,859) for the complete treatment (0–24 years) were 1.6 times the expected protocol costs (€25,198) due to optional, non-protocolized procedures. Hospital admissions including surgery were the main cost drivers, accounting for 42% of observed costs and 70% of expected protocol costs. Implementing the ICHOM Standard Set increased protocol-based costs by 7%.

Assessment of auditory processing skills in adolescents with cleft lip and palate

Objective Auditory deprivation due to middle ear defects caused by congenital malformations in the cleft lip and palate population may be a long-term risk factor for auditory processing disorder. The aim of the study is to assess the auditory processing skills in adolescents with cleft lip and palate by considering also maturation effect. Methods Thirty (10–17 years old) adolescents with operated, non-syndromic cleft lip and palate and thirty (10–17 years old) adolescents with normal development were included in the study. While both groups had bilateral normal peripheral hearing levels, study group had a history of chronic otitis media due to congenital malformations. In addition, participants were also evaluated in subgroups, under and over the age of 12, based on 12 years of age at which adult-like responses were obtained for auditory processing. Frequency pattern, duration pattern and Turkish matrix tests were used to assess auditory processing skills. Result The mean scores of the study group in frequency pattern, duration pattern and Turkish matrix tests were lower than the control group (p = 0.000). When the participants were evaluated as under and over the age of 12, while under the age of 12 showed lower performance than over the age of 12 for the study group (p < 0.05), this difference was not observed in the control group (p > 0.05). Conclusion Adolescents with cleft lip and palate may be disadvantaged in their auditory processing skills compared to their peers and they still may not catch up with their peers, even if improvement is observed with the maturation effect.

Reconstruction of Previously Failed Alveolar Bone Grafts with Medial Femoral Condyle Flap in Pediatric Cleft Lip and Palate Patients

Alveolar cleft reconstruction is important to increase the quality of life of cleft lip and palate patients. Usually, alveolar clefts can be reconstructed using bone grafts. However, bone grafting can be insufficient, and other alternatives may be necessary in wide and recalcitrant clefts. The medial femoral condyle (MFC) flap may be the solution for alveolar clefts that are impossible to reconstruct with bone grafting. In this study, the reconstruction of alveolar clefts in the pediatric cleft lip and palate population, using the MFC flap, is described.This study examined 9 pediatric patients whose alveolar clefts were reconstructed prospectively using MFC flap in 2015 and 2019. The age, gender, follow-up times, independent parameters, and existence of concomitant vestibulonasal fistulas of the patients were recorded. Computerized tomography images of the patients were evaluated to detect defect characteristics and evaluate the volume of flap postoperatively. Flap viability was confirmed with bone scintigraphy, and donor area morbidity was evaluated with the Dynamic Gait Index (DGI) in the postoperative period.The study included 7 male and 2 female patients. The mean age of the patients was 13. In addition to an alveolar cleft, 6 patients also had vestibulonasal fistula. It was observed that the volume of the flaps had not changed one year after the operation. The DGI score of all the patients was 24.Existing techniques may be inadequate in the reconstruction of wide and recalcitrant alveolar clefts. MFC flap may be the start of a new era for the treatment of alveolar clefts.

Does the Rigid External Distraction Device Alter Maxillary Pitch in Cleft Maxillary Distraction?

The rigid external distraction (RED) device is reported to have the ability to three-dimensionally reposition the maxilla. The purpose of this study is to assess the ability of RED to intentionally alter the maxillary pitch. Retrospective cohort study. Institutional practice. A retrospective chart review was completed over the past 12 years and a total of 50 patients met the inclusion criteria. Cephalometric changes and alteration in palatal plane angle. Cephalometric analysis of standardized landmarks was completed on calibrated, standardized lateral cephalograms. Pre-distraction and post-consolidation variables were compared via a two-tailed paired t-test. The mean age at surgery of 12.2 ± 3.2 years. Through distraction osteogenesis (DO), the maxilla was moved anteriorly with a mean distraction distance of 8.4 ± 4.8 mm. The mean change in the angles sella-nasion-A-point angle (SNA), sella-nasion-B-point angle (SNB), and A-point-nasion-B-point angle (ANB) were 10.2 ± 4.8, 0.9 ± 2.7, and 9.3 ± 4.1, respectively. The mean change in the palatal plane angle was −4.4 ± 3.7. The mean change in the vertical position of the anterior nasal spine (ANS) and posterior nasal spine (PNS) in relation to the Frankfort horizontal (FH) were −2.0 ± 4.1 mm and 1.7 ± 3.8 mm, respectively. This study documents short-term findings of RED in a large cleft lip and palate (CLP) population. Despite positioning of distraction eyelets superior to the theoretical center of resistance, a counterclockwise (CCW) rotation of the palatal plane occurred. This suggests that adjunctive methods of vector control should be considered if clockwise (CW) rotation of the palatal plane is intended with the RED device.

Closing the Gap: A Systematic Review and Meta-Analysis of Enhanced Recovery After Surgery Protocols in Primary Cleft Palate Repair.

Assess the evidence for Enhanced Recovery After Surgery (ERAS) protocols in the cleft palate population. A systematic review of MEDLINE, Embase, Cochrane, and CINAHL databases for articles detailing the use of ERAS protocols in patients undergoing primary palatoplasty. New York-Presbyterian Hospital. Patients with cleft palate undergoing primary palatoplasty. Meta-analysis of reported patient outcomes in ERAS and control cohorts. Methodological quality of included studies, opioid use, postoperative length of stay (LOS), rate of return to emergency department (ED)/readmission, and postoperative complications. Following screening, 6 original articles were included; all were of Modified Downs & Black (MD&B) good or fair quality. A total of 354 and 366 were in ERAS and control cohorts, respectively. Meta-analysis of comparable ERAS studies showed a difference in LOS of 0.78 days for ERAS cohorts when compared to controls (P < .05). Additionally, ERAS patients utilized significantly less postoperative opioids than control patients (P < .05). Meta-analysis of the rate of readmission/return to ED shows no difference between ERAS and control groups (P = .59). However, the lack of standardized reporting across studies limited the power of meta-analyses. ERAS protocols for cleft palate repair offer many advantages for patients, including a significant decrease in the LOS and postoperative opioid use without elevating readmission and return to ED rates. However, this analysis was limited by the paucity of literature on the topic. Better standardization of data reporting in ERAS protocols is needed to facilitate pooled meta-analysis to analyze their effectiveness.

Self-Esteem and Oral Health-Related Quality of Life within a Cleft Lip and/or Palate Population: A Prospective Cohort Study.

(1) Objective: To investigate the oral health-related quality of life (OHRQoL) and self-esteem (SE) of a population with cleft lip and/or palate (CLP) and to compare it with a non-affected control cohort. (2) Materials and methods: This study comprised 91 CLP patients and a control group of 790 individuals, seeking orthodontic treatment. OHRQoL and SE were assessed by the Child’s Perception Questionnaire (CPQ) and the Dutch adaptation of the Harter’s Self-Perception Profile for Adolescents. Treatment need and self-perception of oral aesthetic were assessed using the Index of Orthodontic Treatment Need (IOTN) and the Oral Aesthetic Subjective Impact Scale (OASIS). Patients’ expectations and motivation for treatment were also scored. Linear models were used for statistical comparisons between groups. (3) Results: The cleft group scored higher in all domains of the CPQ, OASIS, IOTN and regarding SE for the domains of scholastic competence, athletic competence, physical appearance and behavioral conduct. The cleft group was not only more motivated and expected less discomfort during treatment but also had higher expectations for the treatment outcome. (4) Conclusions: The OHRQoL of CLP patients is strongly correlated with the presence of an oral cleft, while SE remains a personal resource not influenced by the malocclusion or medical condition.

Child Protective Services Referral in a Cleft Lip and/or Palate Population: Assessment of Prevalence, Indications, and Outcomes.

To determine the prevalence of, reasons for, and outcomes related to Child Protective Services (CPS) referral in an isolated and syndromic cleft lip/palate population. Retrospective cohort study. Tertiary Children's Hospital. Any patient <18 years of age attending the multidisciplinary cleft team for care at our institution with a history of referral to CPS by the cleft team during the study period 2009 to 2014. The number of children with CPS referrals, reasons for CPS referrals, outcomes of CPS referrals, associated psychosocial risk factors potentially predictive of CPS referral; demographics and cleft-related surgical history was also reviewed for each patient. Of 1392 patients, 25 (1.8%) were identified with a history of referral to CPS. Average age at referral was 11 months; 76.0% of patients were <1 year of age. Most referrals (64.0%) were directly associated with issues related to cleft care. Identified psychosocial risk factors included financial strain, mental illness/cognitive disability, transportation issues, and inadequate social support. Nine families ultimately lost custody of their children temporarily (n = 5) or permanently (n = 4). Cleft team family referral to CPS involves long-term patient care challenges requiring maximal medical and social support. Families are most commonly referred for issues related to medical neglect, which can lead to failure to thrive, delays in care, and ultimate removal from the home. Identifying families with known psychosocial risk factors and providing increased support may potentially help avoid referrals to CPS.

Prevalence of Reading Difficulties in 9- to 10-Year Old Children in Sweden Born With Cleft Palate.

To investigate the prevalence of reading difficulties in children born with cleft palate at ages 9 and 10 in Sweden. Using a cross-sectional design, a parental questionnaire assessing dyslexia-like reading difficulties (Short Dyslexia Scale, SDS) was administered together with separate questions regarding background data. Families with a child born with overt cleft palate with or without cleft lip in 4 regions of Sweden. A total of 245 families were approached of which 138 families responded. Data from 136 (56%) were complete with information on cleft type and could be analyzed. Twenty-two percent (95% CI, 15-30) of the whole study group displayed risk for dyslexic reading difficulties on the SDS corresponding to the 7th to 10th percentiles in the population. Children with cleft palate only had a significantly higher prevalence of reading difficulties (37%) compared to children with unilateral cleft palate (19%) and bilateral cleft palate (10%). The frequency of reading difficulties in participants with comorbidity was 32%. Among a subgroup with reported comorbidity in areas of attention, language, and learning problems, there was a 2.5 times higher risk of reading disability compared to participants without this reported comorbidity. The prevalence of reading difficulties in the cleft palate population was higher than in the general population. Results showed that co-occurring difficulties were common in the cleft group and that reading difficulties often appear together with other, co-occurring neurodevelopmental difficulties.

Long Term Speech Outcomes Following Midface Advancement in Syndromic Craniosynostosis.

Midface advancement by distraction osteogenesis (DO) is commonly performed in patients with craniosynostosis for indications including midface hypoplasia, exorbitism, obstructive sleep apnea, class III malocclusion, and overall aesthetic facial deficiency. There is evidence to suggest that maxillary LeFort I advancement increases the risk of velopharyngeal dysfunction in the cleft palate population, yet few studies have investigated changes in speech following LeFort III or monobloc midface advancement in patients with syndromic craniosynostosis. The purpose of this study was to examine the effect of midface DO on speech as indicated by the Pittsburgh Weighted Speech Score in patients with Apert, Crouzon, and Pfeiffer Syndrome. Among 73 midface advancement cases performed during the study period, 19 cases met inclusion criteria. Overall, the highest post-advancement Pittsburgh Weighted Speech Score (PWSS) was significantly higher than the pre-advancement PWSS (0.52 versus 2.42, P = 0.01), indicating an acute worsening of VPI post-advancement. Specifically, the PWSS components nasal emission and nasality were significantly higher post-advancement than pre-advancement (nasal emission: 1.16 versus 0.21, P = 0.02) (nasality: 0.68 versus 0.05, P = 0.04). However, there was no significant difference between pre-advancement PWSS and the latest post-advancement PWSS (P = 0.31). Midface distraction is associated with an acute worsening of VPI post-operatively that is followed by improvement, and often resolution over time. Future work with additional patient accrual is needed to determine the effect of different advancement procedures and syndromes on VPI rates and profundity.

Assessing speech at three years of age in the cleft palate population: a scoping review of assessment practices.

There is no consensus in the UK regarding the types of speech samples or parameters of speech that should be assessed at 3 years of age in children with cleft palate ± cleft lip (CP±L), despite cleft units routinely assessing speech at this age. The standardization of assessment practices would facilitate comparisons of outcomes across UK cleft units; earlier identification of speech impairments-which could support more timely treatments; and more reliable recording of therapy impacts and surgical interventions. To explore assessment practices used to assess speech in 3-year-old children with CP±L, including speech parameters, methods of assessment and the nature of the speech sample used. A broad examination of the literature was undertaken through the use of a scoping review conducted in accordance with Joanna Briggs Institute guidelines. Search terms were generated from a preliminary search and then used in the main search (Medline, CINAHL, Embase, AMED and PsycINFO). A combination of approaches (medical, linguistic, developmental and functional) is required to assess CP±L speech at age 3. A developmental approach is recommended at this age, considering the complexity of speech profiles at age 3, in which typically developing speech processes may occur alongside cleft speech characteristics. A combined measure for both nasal emission and turbulence, and an overall measure for velopharyngeal function for speech, show potential for assessment at this age. Categorical ordinal scales are frequently used; the use of continuous scales has yet to be fully explored at age 3. Although single-word assessments, including a subset of words developed for cross-linguistic comparisons, are frequently used, more than one type of speech sample may be needed to assess speech at this age validly. The lack of consensus regarding speech samples highlights a need for further research into the types of speech samples 3-year-olds can complete; the impact of incomplete speech samples on outcome measures (particularly relevant at this age when children may be less able to complete a full sample); the impact of different speech samples on the validity of assessments; and the reliability of listener judgements. Whilst a medical model and linguistic approaches are often central in assessments of age-3 cleft speech, this review highlights the importance of developmental and functional approaches to assessment. Cross-linguistic single-word assessments show potential, and would facilitate the comparison of UK speech outcomes with other countries. Further research should explore the impact of different speech samples and rating scales on assessment validity and listener reliability.

Optimal Timing of Alveolar Cleft Bone Grafting for Maxillary Clefts in the Cleft Palate Population.

Timing of bone grafting for maxillary alveolar clefts is not standardized. Secondary bone grafting is often performed; however, consensus does not exist regarding use of chronologic versus dental age to guide treatment. Several authors suggest an early chronologic age is associated with greater success. Available literature was systematically reviewed for evidence for optimal timing for grafting maxillary alveolar clefts. PubMed, MEDLINE, and Cochrane Central Registrar of Controlled Trials databases were queried for manuscripts pertaining to maxillary alveolar cleft bone grafting. Inclusion criteria included manuscripts with level of evidence 4 or greater. Studies not using bone graft, lacking postoperative follow up, and clinical reports were excluded. Seventeen articles met criteria. Nine manuscripts recommended grafting based on dental age prior to eruption of the permanent canines, while 8 recommended grafting between ages 7 to 12. The most commonly reported complication was wound dehiscence, followed by graft-site infection. Ten studies used perioperative treatment protocols, 8 of which included preoperative and/or postoperative orthodontia for maxillary expansion. Correlation between chronologic age and success was not significant, but trended towards greater success with increasing age. Success of secondary grafting is high, but significant variability exists in the timing of grafting. Evidence is lacking to support specific chronologic age; rather, perioperative protocols, systematic surgical technique, and a multidisciplinary discussion are likely more substantial in achieving success, and may be confounders in studies where an early age at grafting appears associated with success. The timing of bone grafting for maxillary alveolar clefts would benefit from a prospective randomized study.

Speech and language characteristics in individuals with nonsyndromic submucous cleft palate-A systematic review.

Up to 80% of individuals with unrepaired submucous cleft palate (SMCP) experience speech difficulties secondary to velopharyngeal insufficiency. Language delays are reported in the broader cleft lip and/or palate population, suggesting that individuals with SMCP may also be at risk. However, contemporary understanding of this population remains limited as there has been no systematic examination of the literature. This review aims to systematically review and document the speech and language characteristics of individuals with nonsyndromic SMCP and, in addition, to identify factors reported to impact speech and language outcomes. This review followed the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines. Five databases were comprehensively searched using keywords and indexed headings. Included studies had to report speech or language outcomes of individuals with nonsyndromic SMCP. Risk of bias and methodological design quality were examined using tools from the Scottish Intercollegiate Guidelines Network. Relevant data were extracted for analysis. Eighteen studies met inclusion criteria, yielding 598 participants. Study results showed that individuals with unrepaired nonsyndromic SMCP may have speech difficulties secondary to velopharyngeal insufficiency including increased nasal resonance and palatalized or glottal articulation. Lower age at primary surgical repair led to better postsurgical speech outcomes. There is a paucity of literature outlining motor or phonological aspects of speech and receptive or expressive language abilities of this population. Individuals with nonsyndromic SMCP present with speech difficulties similar to those experienced by individuals with overt cleft palate. Health care professionals should be aware of possible presenting symptoms and consider early SMCP diagnoses where appropriate. Further research is needed to specify the broader communication profile in this population.

Breastfeeding Practices in Newborns with Cleft Lip and or Palate

Background: The Surgeon General, AAP, and WHO are all encouraging increases in the exclusive use of breast milk for at least 6 months through direct breastfeeding. The Surgeon General underscores the specific need to empower the medical community to support breastfeeding mothers through education, encouragement, and professional assistance. There is scant literature on the rates of breast milk provision for infants with medical conditions other than very low birth weight and prematurity. One group in which breastfeeding is often discouraged is the cleft lip and palate population. These …

Incidence and Outcomes of Child Protective Services Referral in a Cleft Lip and Palate Population

Incidence and Outcomes of Child Protective Services Referral in a Cleft Lip and Palate Population

Incidence and Outcomes of Child Protective Services Referral in a Cleft Lip and Palate Population

Background: Having a child affected by cleft lip and/or palate can create numerous challenges for a family. Some patients have few medical issues while others have significant needs requiring close attention and monitoring to ensure appropriate growth and development. The cleft team occasionally encounters families who are not able to meet these challenges well. When all conservative prompts and supports provided by the team have proved unsuccessful, referring the family to Child Protective Services for intervention may help ensure appropriate care is provided to the patient. …

Desempenho cognitivo-linguístico de crianças com fissura labiopalatina em fase de aquisição da leitura e escrita

RESUMO Objetivo: identificar as competências cognitivo-linguísticas necessárias ao aprendizado da leitura e da escrita de crianças com fissura labiopalatina. Métodos: participaram 120 crianças, de 05 e 06 anos, cursando o Jardim II e o 1º ano do ensino fundamental, compondo dois grupos: G1, formado por 60 crianças com fissura labiopalatina, e G2, com 60 crianças sem alterações no desenvolvimento. Empregaram-se as Matrizes Progressivas Coloridas de Raven, o Perfil de Habilidades Fonológicas e o Boston Naming Test. Resultados: comparando os desempenhos dos grupos, verificou-se diferença estatística nas funções intelectuais, indicando prejuízos nas habilidades cognitivas do G1 (p=0,019). As competências cognitivo-linguísticas foram classificadas “Sob Atenção” para ambos os grupos, sendo mais representativa a defasagem no G1, com 66,7%. Conclusão: as competências linguísticas necessárias para a aquisição da leitura e escrita mostraram-se deficitárias na população fissurada, com níveis de desempenho cognitivo inferior ao esperado para a idade e escolaridade, em grande parte das modalidades avaliadas. Fatores ambientais limitantes e o baixo nível sociocultural da amostra foram sugestivos de interferência adicional à aquisição das pré-competências cognitivo-linguísticas, necessárias ao desempenho satisfatório de tarefas acadêmicas como a leitura e escrita.

Does Type of Cleft Palate Repair Influence Postoperative Eustachian Tube Dysfunction?

Otitis media (OM) is a frequent occurrence in the cleft palate population. Palate repair is thought to positively contribute to Eustachian tube function. The purpose of this study is to determine the proportion of cleft patients in our cohort with OM, and if palatoplasty closure technique influences future OM differently. The authors posited that Z-plasty palate repair would result in improved Eustachian tube function, compared with straight-line repair. Patient records were accessed and data was recorded on palatoplasties from 2005 to 2007 as well as from 2012 to 2014. Type of palatoplasty (straight-line vs. Z-plasty) was recorded and compared to prevalence of pressure equalizer tube placement and antibiotic prescriptions in various assessment intervals following repair 1) <6 months, 2) 6 months to 1 year. Statistical analysis included Welch 2 sample t test. The type of cleft palate was similar between the 2 groups. Prior to palate repair, over 30% of subjects in both groups required either tubes or antibiotics for OME. At the time of surgery, over 50% of patients received ear tubes, in both groups. Following repair, for straight versus Z, 0 versus 11%; and, 27.8 versus 20% required ear tubes for intervals 1) and 2), respectively. P values for both intervals are 0.9183 and 0.328, respectively. Antibiotics during interval one were required in 22.2% versus 23.5% of straight-line closures and Z-plasty, respectively. Antibiotics in the second interval were needed for 33.3% and 30% of straight-line closures and Z-plasty closures, respectively. P values for antibiotic use in the first and second intervals were 0.5353 and 0.4316 respectively. Cleft palate patients have a high prevalence of OM. Palatoplasty technique (straight-line and Z-plasty) does not affect Eustachian tube dysfunction differently following repair as measured by pressure equalizer tube placement and antibiotic use.

Permanent tooth agenesis in non-syndromic Robin sequence and cleft palate: prevalence and patterns

ObjectivesPartial tooth agenesis is frequently observed in Robin sequence. Tooth anomalies are increasingly considered as an extended phenotype of the cleft palate population. The study objective was to compare the prevalence and patterns of tooth agenesis in a group of patients with non-syndromic Robin sequence (ns-RS) and a group with non-syndromic cleft palate (ns-CP).Materials and methodsThe panoramic radiographs of 115 ns-RS and 191 ns-CP patients were assessed for agenesis of the permanent dentition (excluding third molars) and the patterns recorded using the Tooth Agenesis Code.ResultsPartial tooth agenesis was observed in 47.8% of ns-RS and 29.8% of ns-CP patients with a greater prevalence in the mandibula than in the maxilla, particularly in ns-RS. The teeth most frequently absent in both groups were the mandibular second premolars and maxillary lateral incisors. Tooth agenesis was bilateral in two-thirds of affected ns-RS patients and one-half of ns-CP patients. In ns-RS, bilateral agenesis of the mandibular second premolars was more frequently observed in female than that in male patients. Completely symmetrical patterns of hypodontia were found in around 45% of ns-RS patients with tooth agenesis compared to 35% in ns-CP. No association was found between the extent of the palatal cleft and the severity of hypodontia.ConclusionTooth agenesis is more prevalent in ns-RS than that in ns-CP, demonstrates a much greater predilection for the mandible in ns-RS, and bears no relation to the extent of the palatal cleft.Clinical relevanceWhen compared to ns-CP, additional developmental disturbances are likely involved in the etiology of tooth agenesis in ns-RS. Future research could help identify the underlying genetic traits and aid in classifying patients in those with and without expected tooth agenesis in order to facilitate orthodontic management strategies.