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Congenital cleft lip and palate in Singapore

Fong PH, Yeap CL, Lee ST

Ann Acad Med Singap, 1983 Apr;12(2 Suppl):363-5.
PMID: 6625521

Cleft lip and palate is the most common congenital abnormality seen in the Department of Plastic Surgery, Singapore General Hospital. A total of 461 operative cases seen over a period of 5 years (1977 to 1981) is analysed. There is a relatively high incidence in the Singapore population of 2.0 per 1000 live births. In both cleft lip and cleft palate deformities, there is no significant difference in the sex distribution. The cleft patterns show that 78% of the cases are unilateral clefts, 53% are left sided, 25% are right sided and 22% of the cases are bilateral clefts.

Matched MeSH terms: Cleft Lip/epidemiology*
Assessment of complete unilateral cleft lip and palate patients: Determination of factors effecting dental arch relationships

Arshad AI, Alam MK, Khamis MF

Int J Pediatr Otorhinolaryngol, 2017 Jan;92:70-74.
PMID: 28012538 DOI: 10.1016/j.ijporl.2016.11.006

OBJECTIVES: The purpose of the current study was to assess the treatment outcome of non-syndromic patients having complete unilateral cleft lip and palate (CUCLP) by using GOSLON index and to determine any association of pre and/or postnatal factors with the treatment outcome.
MATERIALS AND METHODS: One hundred and one sets of dental models of patients having CUCLP were assessed in this retrospective study. Five examiners that were blinded to case-specific information scored the dental models at two instances with an interval of two weeks to ensure memory bias elimination (5 × 101 × 2 = 1010 observations). Calibration courses were conducted prior to scoring and each examiner was provided with scoring sheets, pictures of GOSLON reference models and flowcharts explaining the scoring method.
RESULTS: According to GOSLON index, a mean (SD) GOSLON score of 3.04 (1.25) was determined. Based on treatment outcome groups, 62 patients had favorable (grade 1, 2, and 3) and 39 cases had unfavorable (grade 4 and 5) treatment outcome. Chi-square tests revealed a significant association of gender (P = 0.002), cheiloplasty (P = 0.001) and palatoplasty (P

Matched MeSH terms: Cleft Lip/surgery*
Mutation screening of IRF6 among families with non-syndromic oral clefts and identification of two novel variants: review of the literature

Salahshourifar I, Wan Sulaiman WA, Halim AS, Zilfalil BA

Eur J Med Genet, 2012 Jun;55(6-7):389-93.
PMID: 22440537 DOI: 10.1016/j.ejmg.2012.02.006

Non-syndromic oral clefts share the main clinical features of Van der Woude Syndrome (VWS), with the exception of the lower lip pit. Thus, about 15% of VWS cases are indistinguishable from cases with non-syndromic oral clefts. IRF6 mutations are the major cause of VWS; however, variants in this gene show strong association with non-syndromic oral clefts, with a higher increased risk among cases with cleft lip only (CLO). A total of 39 individuals, including 16 patients with CLO and 23 patients with a family history of cleft, were examined for IRF6 mutations in the present study. Seven variants, including five known (c.-75-4 A>; G, c.-73T>; C, c.459G>; T 5, c.820G>; A, and c.1060 + 37C>; T) and two novel (c.-75-23G>; C and c.1380G>; T), were found. Both novel variants were inherited from non-affected parents and we did not find also in the 120 control chromosomes. In silico analysis revealed that both c.1380G>; T and c.-75-23G>; C variants may disrupts a putative exonic splicing enhancer and intronic splicing binding site for SC35, respectively. Taken together, the presence of deleterious IRF6 variants in patients with non-syndromic oral clefts could be most likely an evidence for VWS. While, IRF6 variants could, at best, contribute to clefting as part of a complex inheritance pattern, with both additional genes and environmental factors having a role.

Matched MeSH terms: Cleft Lip/genetics*
Transforming growth factor-alpha and nonsyndromic cleft lip with or without palate or cleft palate only in Kelantan, Malaysia

Rahman RA, Ahmad A, Rahman ZA, Mokhtar KI, Lah NA, Zilfalil BA, et al.

Cleft Palate Craniofac J, 2008 Nov;45(6):583-6.
PMID: 18956930 DOI: 10.1597/07-020.1

To determine the frequency of the transforming growth factor-alpha (TGFalpha) Taq1 polymorphism in nonsyndromic cleft lip with or without cleft palate (CL+/-P) and cleft palate only (CP) in Kelantan, Malaysia.

Matched MeSH terms: Cleft Lip/genetics*
Maternal uniparental heterodisomy of chromosome 6 in a boy with an isolated cleft lip and palate

Salahshourifar I, Halim AS, Sulaiman WA, Zilfalil BA

Am J Med Genet A, 2010 Jul;152A(7):1818-21.
PMID: 20583164 DOI: 10.1002/ajmg.a.33526

We describe a chromosome 6 uniparental disomy (UPD6) in a boy, discovered during a screening for the genetic cause of cleft lip and palate. In the medical literature, almost all documented cases of UPD6 are paternal in origin, and only four were maternal. We present here a report of complete maternal chromosome 6 uniparental heterodisomy. Haplotype analysis was performed using highly polymorphic short tandem repeat (STR) markers that span both arms of chromosome 6. Analysis of these markers revealed the presence of two maternal alleles but no paternal allele, indicating an instance of maternal uniparental heterodisomy. Chromosome analysis of peripheral blood lymphocytes confirmed a normal male karyotype. Advanced maternal age at the time of the infant's birth and heterodisomy of markers around the centromere favors a meiosis-I error. No specific phenotype has been reported for maternal UPD6. Therefore, the cleft lip and palate in the present case probably occurred due to other risk factors. This report provides further evidence that maternal UPD6 has no specific clinical consequences and adds to the collective knowledge of this rare chromosomal finding.

Matched MeSH terms: Cleft Lip/genetics*
A three-dimensional computed tomographic analysis of the cervical spine in unoperated infants with cleft lip and palate

Rajion ZA, Townsend GC, Netherway DJ, Anderson PJ, Yusof A, Hughes T, et al.

Cleft Palate Craniofac J, 2006 Sep;43(5):513-8.
PMID: 16986980

To investigate anatomical variations and abnormalities of cervical spine morphology in unoperated infants with cleft lip and palate.

Matched MeSH terms: Cleft Lip/radiography*
Fulltext Contribution of 6p24 to non-syndromic cleft lip and palate in a Malay population: association of variants in OFC1

Salahshourifar I, Halim AS, Sulaiman WA, Zilfalil BA

J Dent Res, 2011 Mar;90(3):387-91.
PMID: 21297019 DOI: 10.1177/0022034510391798

Non-syndromic cleft lip, with or without cleft palate, is a heterogeneous, complex disease with a high incidence in the Asian population. Several association studies have been done on cleft candidate genes, but no reports have been published thus far on the Orofacial Cleft 1 (OFC1) genomic region in an Asian population. This study investigated the association between the OFC1 genomic region and non-syndromic cleft lip with or without cleft palate in 90 Malay father-mother-offspring trios. Results showed a preferential over-transmission of a 101-bp allele of marker D6S470 in the allele- and haplotype-based transmission disequilibrium test (TDT), as well as an excess of maternal transmission. However, no significant p-value was found for a maternal genotype effect in a log-linear model, although single and double doses of the 101-bp allele showed a slightly increased cleft risk (RR = 1.37, 95% CI, 0.527-3.4, p-value = 0.516). Carrying two copies of the 101-bp allele was significantly associated with an increased cleft risk (RR = 2.53, 95% CI, 1.06-6.12, p-value = 0.035). In conclusion, we report evidence of the contribution of the OFC1 genomic region to the etiology of clefts in a Malay population.

Matched MeSH terms: Cleft Lip/genetics*
De novo interstitial deletion of 1q32.2-q32.3 including the entire IRF6 gene in a patient with oral cleft and other dysmorphic features

Salahshourifar I, Halim AS, Sulaiman WA, Ariffin R, Naili Muhamad Nor N, Zilfalil BA

Cytogenet Genome Res, 2011;134(2):83-7.
PMID: 21447942 DOI: 10.1159/000325541

Microdeletion of the Van der Woude syndrome (VWS) critical region is a relatively rare event, and only a few cases have been reported in the medical literature. The extent of the deletion and the genotype-phenotype correlation are 2 crucial issues.

Matched MeSH terms: Cleft Lip/genetics
Unusual clinical presentation of a severely protruding premaxilla in patient with bilateral clefts

Burezq H, Bang RL, George A, Mukhtar A

J Craniofac Surg, 2007 Jul;18(4):971-4.
PMID: 17667698

Prominent premaxilla is one of the problems encountered when dealing with bilateral complete cleft lip and palate patients. Secondary alveolar bone grafting with these patients would achieve filling of the osseous defect, supports the alar base, eliminate the oro-nasal fistula and enhance the maxillary instability. This article describes the management of a bilateral cleft lip and palate patient with an extremely protruding premaxilla done in one stage surgery. To our knowledge, this is the first report of such degree of severity in the English literature.

Matched MeSH terms: Cleft Lip/surgery*
Modified Activation Technique for Nasal Stent of Nasoalveolar Molding Appliance for Columellar Lengthening in Bilateral Cleft Lip/Palate

Patil PG, Nimbalkar-Patil SP

J Prosthodont, 2018 Jan;27(1):94-97.
PMID: 27002917 DOI: 10.1111/jopr.12464

Bilateral cleft lip/cleft palate is associated with nasal deformities typified by a short columella. The presurgical nasoalveolar molding (NAM) therapy approach includes reduction of the size of the intraoral alveolar cleft as well as positioning of the surrounding deformed soft tissues and cartilages. In a bilateral cleft patient, NAM, along with columellar elongation, eliminates the need for columellar lengthening surgery. Thus the frequent surgical intervention to achieve the desired esthetic results can be avoided. This article proposes a modified activation technique of the nasal stent for a NAM appliance for columellar lengthening in bilateral cleft lip/palate patients. The design highlights relining of the columellar portion of the nasal stent and the wire-bending of the nasal stent to achieve desirable results within the limited span of plasticity of the nasal cartilages. With this technique the vertical taping of the premaxilla to the oral plate can be avoided.

Matched MeSH terms: Cleft Lip/surgery*
Postnatal treatment factors affecting craniofacial morphology of unilateral cleft lip and palate (UCLP) patients in a Japanese population

Alam MK, Iida J, Sato Y, Kajii TS

Br J Oral Maxillofac Surg, 2013 Dec;51(8):e205-10.
PMID: 23099108 DOI: 10.1016/j.bjoms.2012.10.001

We have evaluated the craniofacial morphology of Japanese patients with unilateral cleft lip and palate (UCLP) and assessed the various postnatal factors that affect it. Lateral cephalograms of 140 subjects (mean (SD) aged 7 (2) years) with UCLP were taken before orthodontic treatment. Surgeons from Hokkaido University Hospital had done the primary operations. The craniofacial morphology was assessed by angular and linear cephalometric measurements. Cheiloplasty, palatoplasty, and preoperative orthopaedic treatment were chosen as postnatal factors. To compare the assessments of the postnatal factors, we made angular and linear cephalometric measurements for each subject and converted them into Z scores in relation to the mean (SD) of the two variables. Subjects treated by the modified Millard cheiloplasty had larger sella-nasion-point A (SNA) and nasion-point A-pogonion (NA-POG) measurements than subjects treated by the modified Millard with a vomer flap cheiloplasty. Two-stage palatoplasty showed consistently better craniofacial morphology than the other palatoplasty. Subjects who had preoperative orthopaedic treatment with a Hotz plate had significantly larger upper incisor/sella-nasion (U1-SN) measurements than who had no preoperative orthopaedic treatment or an active plate. We conclude that in subjects treated by a modified Millard type of cheiloplasty, a two-stage palatoplasty, and a Hotz plate there were fewer adverse effects on craniofacial morphology.

Matched MeSH terms: Cleft Lip/pathology; Cleft Lip/surgery*
Haplotype diversity in 11 candidate genes across four populations

Beaty TH, Fallin MD, Hetmanski JB, McIntosh I, Chong SS, Ingersoll R, et al.

Genetics, 2005 Sep;171(1):259-67.
PMID: 15965248

Analysis of haplotypes based on multiple single-nucleotide polymorphisms (SNP) is becoming common for both candidate gene and fine-mapping studies. Before embarking on studies of haplotypes from genetically distinct populations, however, it is important to consider variation both in linkage disequilibrium (LD) and in haplotype frequencies within and across populations, as both vary. Such diversity will influence the choice of "tagging" SNPs for candidate gene or whole-genome association studies because some markers will not be polymorphic in all samples and some haplotypes will be poorly represented or completely absent. Here we analyze 11 genes, originally chosen as candidate genes for oral clefts, where multiple markers were genotyped on individuals from four populations. Estimated haplotype frequencies, measures of pairwise LD, and genetic diversity were computed for 135 European-Americans, 57 Chinese-Singaporeans, 45 Malay-Singaporeans, and 46 Indian-Singaporeans. Patterns of pairwise LD were compared across these four populations and haplotype frequencies were used to assess genetic variation. Although these populations are fairly similar in allele frequencies and overall patterns of LD, both haplotype frequencies and genetic diversity varied significantly across populations. Such haplotype diversity has implications for designing studies of association involving samples from genetically distinct populations.

Matched MeSH terms: Cleft Lip/ethnology; Cleft Lip/genetics
Comparing Modified Huddart-Bodenham Scoring System and GOSLON Yardstick to Assess Dental Arch Relationships in Unilateral Cleft Lip and Palate Patients

Yakob M, Hassan YR, Tse KL, Gu M, Yang Y

Cleft Palate Craniofac J, 2018 Jan 01.
PMID: 28092164 DOI: 10.1597/16-191

Objective To test the reliability of the modified Huddart-Bodenham (MHB) numerical scoring system and its agreement with the GOSLON Yardstick categorization for assessing the dental arch relationships in unilateral cleft lip and palate (UCLP) cases. Design A retrospective study. Setting Faculty of Dentistry, The University of Hong Kong. Patients Forty-one nonsyndromic UCLP consecutive patients attending the Joint Cleft Lip/Palate Clinic at Faculty of Dentistry in the University of Hong Kong were selected. Interventions Study models at 8 to 10 years old (T1) and 10 to 12 years old (T2) were obtained from each patient. Main Outcome Measures Models were rated with the MHB scoring system and GOSLON Yardstick index. The intra- and interexaminer reliabilities as well as correlation of both scoring systems were evaluated. Furthermore, to investigate the outcome measurements consistency, the MHB scoring system and GOSLON Yardstick were independently used to compare the dental arch relationships from T1 to T2, with the samples split into intervention and nonintervention groups. Results The MHB scoring system presented good intra- and interexaminer agreement, which were comparable to those of the GOSLON Yardstick. The correlation between the MHB scoring system and GOSLON Yardstick scores was good. Both scoring systems showed similar results when assessing the change in the dental arch relationships from T1 to T2. Conclusions The MHB scoring system can be used as an alternative method to the commonly used GOSLON Yardstick for assessing dental deformities in UCLP patients. Both scoring systems showed similar results in assessing the improvement in dental arch relationships.

Matched MeSH terms: Cleft Lip
Fulltext Oral health knowledge, attitude and practice of parents, and frequency of cariogenic food intake among their non syndromic cleft lip with or without cleft palate children in North-east Peninsular Malaysia

Rathmawati Ahmad, Normastura Abd Rahman, Ruhaya Hasan, Azizah Yusoff

Malays J Nutr, 2017;23(2):239-252.
MyJurnal

Introduction: Appropriate oral health knowledge is essential to enable parents to be responsible for the oral health care of their children especially those with special needs. This study aimed to determine the oral health knowledge, attitude and practice (KAP) of parents and cariogenic food intake of their non-syndromic cleft lip with or without cleft palate children (NSCLP) compared to non-cleft children.

Methods: A comparative cross-sectional study was conducted on randomly selected parents with children aged 1 to 6 years at Hospital Universiti Sains Malaysia and Kota Bharu Dental Clinic. A face-to-face interview using a validated questionnaire on oral health KAP and cariogenic food intake of their children was done. Data were analysed by using IBM SPSS version 22.0.

Results: Sixty-four Malay parents with NSCLP (n=33) and non-cleft (n=31) children were recruited. The mean (SD) percentage score of oral health knowledge of parents in both groups (NSCLP and non-cleft) was fair, 59.5%(11.85) and 62.0%(15.00), P=0.4722; oral health attitude was good, 78.0% (9.30) and 80.2% (15.40), P=0.504; but oral health practice was poor 33.8% (5.94) and 36.1 (8.44), P=0.220. The cariogenic food frequency score was significantly higher in noncleft children; 40.2(10.32) compared to NSCLP children, 34.8(9.46), P=0.032.

Conclusions: Parents of both groups had fair knowledge and good attitude but poor oral health practice. Cariogenic foods were consumed more by the non-cleft children. Oral health awareness among the parents needs to be strengthened towards good oral health attitude and practice and non-cariogenic food choices for the betterment of oral health status of their children.

Matched MeSH terms: Cleft Lip
'Pai Syndrome' with anterior alveolar polyp: A variant of a rare clinical entity

Debnath KK, Kanapaty Y, Yong DJ, Chandran S, Bakri A

Med J Malaysia, 2019 Jun;74(3):229-230.
PMID: 31256179

'Pai syndrome' (PS) is a rare congenital syndrome. Presented here, a new-born baby-girl who exhibited the characteristic features of having a midline nasal (septal) polyp, an anterior alveolar process polyp, and a pericallosal lipoma associated with corpus callosum dysgenesis of the brain. Both polyps were lined with stratified-squamous epithelium. The overall features were largely consistent with those described by Pai et al., in 1987. A midline cleft-lip (with or without cleft-alveolus) is one of the most common features of the syndrome which was however absent in this case. Instead, an anterior alveolar polyp is present, which is relatively rare.

Matched MeSH terms: Cleft Lip
Effects of Multiple Factors on Treatment Outcome in the Three-Dimensional Maxillary Arch Morphometry of Children With Unilateral Cleft Lip and Palate

Haque S, Khamis MF, Alam MK, Ahmad WMAW

J Craniofac Surg, 2020 May 04.
PMID: 32371703 DOI: 10.1097/SCS.0000000000006464

Studies have claimed that the maxillary arch dimension of unilateral cleft lip and palate (UCLP) patient is significantly smaller than normal patient. Multiple factors (both congenital and post natal treatment factors) are believed to have an impact on the treatment outcome (maxillary arch retardation) of UCLP patient. The aim of this study was to evaluate the effects of multiple factors on the treatment outcome by assessing the maxillary arch dimension of UCLP Children. Eighty-five Malaysian UCLP children were taken who received cheiloplasty and palatoplasty. Laser scanned 3D digital models of UCLP subjects were prepared before any orthodontic treatment and bone grafting at 7.69 ± 2.46 (mean± SD) years of age. Intercanine width (ICW), intermolar width (IMW), and arch depth (AD) measurements of maxillary arch were measured with Mimics software. Multiple linear regression analyses were used to evaluate the association between multiple factors (gender, UCLP type, UCLP side, family history of cleft, family history of class III malocclusion, techniques of cheiloplasty, and techniques of palatoplasty) and maxillary arch dimensions (ICW, IMW, and AD). P value was set at 5%. Significant association was found between 2 techniques of cheiloplasty and ICW (P = 0.001) and also between 2 techniques of palatoplasty and ICW (P = 0.046) of maxilla. No significant association observed in IMW and AD in relation to all other factors. Modified Millard techniques of Cheiloplasty and Bardach technique of palatoplasty had unfavorable effect on the treatment outcome by assessing the maxillary arch dimension (ICW) using laser scanned 3D digital models in Malaysian UCLP children.

Matched MeSH terms: Cleft Lip
Fulltext Three-dimensional facial analysis of Chinese children with repaired unilateral cleft lip and palate

Othman SA, Aidil Koay NA

Sci Rep, 2016 08 10;6:31335.
PMID: 27507713 DOI: 10.1038/srep31335

We analyzed the facial features of Chinese children with repaired unilateral cleft lip and palate (UCLP) and compared them with a normal control group using a three-dimensional (3D) stereophotogrammetry camera. This cross-sectional study examined 3D measurements of the facial surfaces of 20 Chinese children with repaired UCLP and 40 unaffected Chinese children aged 7 to 12 years old, which were captured using the VECTRA 3D five-pod photosystem and analyzed using Mirror software. Twenty-five variables and two ratios were compared between both groups using independent t-test. Intra- and inter-observer reliability was determined using ten randomly selected images and analyzed using intra-class correlation coefficient test (ICC). The level of significance was set at p

Matched MeSH terms: Cleft Lip
Fulltext Orthodontic treatment of a unilateral cleft lip and palate patient with secondary bone graft and eruption of canine

Mathew, Thomas

Malaysian Dental Journal, 2010;31(2):84-89.
MyJurnal

The cleft lip and palate patients often have constricted maxillary arch, congenitally missing teeth, impacted teeth, and many kinds of malocclusion problems. Secondary bone grafting in cleft lip and palate patients is performed preferably before the eruption of permanent canine in order to provide adequate periodontal support for eruption and preservation of the teeth adjacent to the cleft. Secondary grafting with iliac marrow consistently produces trabecular bone to unify the maxilla and provide odontogenic support. Presented here is a case of unilateral cleft lip and palate, which was treated by expansion by quad helix and straight wire appliance for correction of dental malocclusion. Also discussed in detail the entire range of treatment procedures the boy underwent from 14 years of age, especially the role of secondary bone grafting with iliac marrow to facilitate the eruption of permanent impacted canine.

Matched MeSH terms: Cleft Lip
Fulltext Cleft Lip And Palate Centre: The University Hospital Experience

Ghani, S.H.A., Hussain, R., Hassan, S., Tan, K.K., Ahmad, M.H.

Ann Dent, 1996;3(1):-.
MyJurnal

The Combined Cleft Clinic at the University Hospital, Kuala Lumpur was organised in 1992. The team consists of Plastic Surgeon, Orthodontists, Speech therapist, Ear, Nose and Throat Surgeon, Audiologist, medical officers and the nurses. We attend the clinic on a regular basis. Specialists from other medical and dental disciplines as well as the members of the Cleft Lip and Palate Association of Malaysia (CLAPAM) do occasionally participate in this set-up.The team members formulate treatment plans for each cleft patient, monitor the patient's growth and development and manage the patient at different stages according to the individual needs. To date, the idea of team approach and an establishment of a centre towards management of cleft patients seem to be the most ideal as the patients are benefiting total treatment and care from various specialists from only one place. The experience of University Hospital as a centre for cleft patients is discussed.

Matched MeSH terms: Cleft Lip
Fulltext Facial profile and maxillary arch dimensions in unilateral cleft lip and palate children in the mixed dentition stage

Gopinath VK, Samsudin AR, Mohd Noor SNF, Mohamed Sharab HY

Eur J Dent, 2017 4 25;11(1):76-82.
PMID: 28435370 DOI: 10.4103/ejd.ejd_238_16

OBJECTIVES: The aim of this study was to evaluate the vertical and sagittal facial profile and maxillary arch width, depth, and length of patients with unilateral cleft lip and palate (UCLP) and to compare them with healthy noncleft children in the mixed dentition stage (7-13 years).
MATERIALS AND METHODS: This study is conducted at Hospital Universiti Sains Malaysia. UCLP group comprised 48 patients with nonsyndromic UCLP who have had the lip and palate repaired, whereas the control group comprised 48 healthy noncleft cases. The lateral cephalometrics measurements were used to determine the vertical height, sagittal depth of the face, and cranial base length and angle. Maxillary arch dimensions were measured on the study cast including arch width, depth, and length.
RESULTS: Vertical facial height and sagittal depth measurements showed a significant decrease (P < 0.05) in the mean growth pattern in UCLP group. The anterior cranial base length (S-N) was shorter in UCLP children (P < 0.001), while Ba-N length had no significant difference (P = 0.639). Nasion-Sella Tursica-Basion angle was significantly higher in the UCLP group (P = 0.016). Dental arch width with reference to canine-to-canine and first premolar-to- first premolar distance was significantly larger in control (P = 0.001).
CONCLUSION: Mean vertical and sagittal facial dimensions in the UCLP children who do not undergo orthodontic treatment are significantly lesser in all directions of growth than healthy noncleft children. The maxillary dental arch had a normal depth but constricted in width and arch length.

Matched MeSH terms: Cleft Lip

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