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Fulltext Incidence and management of middle ear effusion in cleft palate patients

Lokman S, Loh T, Said H, Omar I

Med J Malaysia, 1992 Mar;47(1):51-5.
PMID: 1387450

For a complete overall rehabilitation of cleft palate patients a multi-disciplinary approach should be adopted. Plastic and Head and Neck Surgeons in whom most of the treatment are entrusted should be concerned not only at achieving palatal function and cosmetic acceptability but also the various other problems associated with cleft palate especially hearing loss. In this study, 66 patients with repaired and unrepaired cleft palates were examined for the presence of hearing loss due to middle ear effusion. The incidence of middle ear effusion was high (57.6%). It was also found that only eight of these patients (12.1%) ever complained of hearing loss or any associated symptoms and repair of the cleft palate did not influence the incidence of middle ear effusion. As such, screening should be done in all cleft palates and otolaryngologists should therefore play an important role in the multi-disciplinary team which should comprise the paediatrician, plastic surgeon, speech therapist, orthodontist and dental specialist.

Matched MeSH terms: Cleft Palate/complications*
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 Palate/surgery*
Fulltext Development and virtual validation of a novel digital workflow to rehabilitate palatal defects by using smartphone-integrated stereophotogrammetry (SPINS)

Farook TH, Jamayet NB, Asif JA, Din AS, Mahyuddin MN, Alam MK

Sci Rep, 2021 04 19;11(1):8469.
PMID: 33875672 DOI: 10.1038/s41598-021-87240-9

Palatal defects are rehabilitated by fabricating maxillofacial prostheses called obturators. The treatment incorporates taking deviously unpredictable impressions to facsimile the palatal defects into plaster casts for obturator fabrication in the dental laboratory. The casts are then digitally stored using expensive hardware to prevent physical damage or data loss and, when required, future obturators are digitally designed, and 3D printed. Our objective was to construct and validate an economic in-house smartphone-integrated stereophotogrammetry (SPINS) 3D scanner and to evaluate its accuracy in designing prosthetics using open source/free (OS/F) digital pipeline. Palatal defect models were scanned using SPINS and its accuracy was compared against the standard laser scanner for virtual area and volumetric parameters. SPINS derived 3D models were then used to design obturators by using (OS/F) software. The resultant obturators were virtually compared against standard medical software designs. There were no significant differences in any of the virtual parameters when evaluating the accuracy of both SPINS, as well as OS/F derived obturators. However, limitations in the design process resulted in minimal dissimilarities. With further improvements, SPINS based prosthetic rehabilitation could create a viable, low cost method for rural and developing health services to embrace maxillofacial record keeping and digitised prosthetic rehabilitation.

Matched MeSH terms: Palate/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 Palate/genetics*
Fulltext Effects of uvulopalatopharyngoplasty: a seven year review

Aneeza WH, Marina MB, Razif MY, Azimatun NA, Asma A, Sani A

Med J Malaysia, 2011 Jun;66(2):129-32.
PMID: 22106693 MyJurnal

To review the long term outcome of Uvulopalatopharyngoplasty (UPPP) for obstructive sleep apnoea syndrome in a tertiary referral centre. 38 records were traced where UPPP was done from July 2000 to December 2007. 14 patients were followed up for one to seven years where the Epworth sleepiness scale was scored, long term side effects documented and post operative muller's manoeuvre done. Success of UPPP is defined as a reduction in apnoea hypopnea index (AHI) more than 50%. Sixty percent (60%) were successfully treated with UPPP in the long term. Mean ESS was significantly reduced from 12 +/- 6 to 7 +/- 4. 11 out of 14 patients (78.5%) were reported to develop long term side effects of UPPP, the highest being velopharyngeal insufficiency (42.8%). In conclusion, UPPP is effective in improving symptoms of OSA in the long term. However, in view of its side effects, uvula preserving surgery should be considered as a surgical option.

Matched MeSH terms: Palate/surgery*
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 Palate/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 Palate/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 Palate/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 Palate/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 Palate/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 Palate/surgery*
Proximal femoral focal deficiency as a manifestation of Antley-Bixler syndrome: a case report

Sulaiman AR, Nawaz H, Munajat I, Sallehudin AY

J Orthop Surg (Hong Kong), 2007 Apr;15(1):84-6.
PMID: 17429125

We report a case of the Antley-Bixler syndrome in an 11-year-old girl. She presented with bilateral proximal femoral focal deficiency, right clubfoot, left radiohumeral synostosis, bilateral ear hypoplasia, cleft palate, tongue tie, missing teeth, congenital heart disease, a pelvic kidney with hydronephrosis, and mental retardation. Proximal femoral focal deficiency has never been reported before as a manifestation of Antley-Bixler syndrome. Her mother was exposed to radiation during an intravenous urogram done in the first trimester of pregnancy. Exposure to radiation has not been implicated as a cause of Antley-Bixler syndrome.

Matched MeSH terms: Cleft Palate/etiology
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 Palate/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 Palate/ethnology; Cleft Palate/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 Palate
Fulltext Prevalence of oral tori and exostosis in Malaysian population - A cross-sectional study

Kumar Singh A, Sulugodu Ramachandra S, Arora S, Dicksit DD, Kalyan CG, Singh P

J Oral Biol Craniofac Res, 2017 Sep-Dec;7(3):158-160.
PMID: 29123992 DOI: 10.1016/j.jobcr.2017.08.008

Introduction: Oral tori and exostosis are non-pathological bony protuberances seen on the alveolar surfaces of the jaw bones. These are commonly seen on the palatal surfaces of the maxilla [torus palatinus (TP)] and around the premolars in the lingual surface of the mandible [torus mandibularis (TM)]. The aim of this cross-sectional study was to determine the prevalence of tori/exostosis in the Malaysian population.

Methodology: A total of 2666 patients were examined for the presence of tori and exostosis in the maxilla and mandible and were categorized into TP, TM, and exostosis (facial/labial). Collected data was analysed for obtaining descriptive statistics.

Results: 882 subjects were noticed with oral tori/exostosis among the population studied with a prevalence rate of 33%. TP was seen more in females (35%), compared to males (20%), and this difference was statistically significant (p value

Matched MeSH terms: Palate
Fulltext Mandibular advancement by distraction Osteogenesis for obstructive sleep apnea in patient with Pierre Robin Sequence: a case report

Mohamed Hisham Mohamed Jali @ Yunos, Shaifulizan Abdul Rahman, Ramizu Shaari

International Medical Journal Malaysia, 2017;16(21):28-28.
MyJurnal

Pierre Robin Sequence (PRS) is a condition consists of set of anomalies, which are
cleft palate, micrognathia and glossoptopsis. Management of patients with PRS addresses two main
problems, namely airway obstruction and feeding difficulties. Airway obstruction may lead to
obstructive sleep apnea (OSA). Treatment modalities for OSA are based on the causes. There are
surgical and non-surgical methods. Non-surgical methods such as diet, medication, oral appliances
and continuous positive airway pressure (CPAP) can only be employed in moderate cases. Surgical
method such as maxillo-mandibular advancement or expansion can be achieved by orthognathic
surgery or distraction osteogenesis. (Copied from article).

Matched MeSH terms: Cleft Palate
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 Palate
Fulltext Alveolar restoration following rapid maxillary expansion with and without corticotomy: A microcomputed tomography study in sheep

Le MHT, Noor Hayaty AK, Zaini ZM, Dom SM, Ibrahim N, Radzi ZB

Korean J Orthod, 2019 Jul;49(4):235-245.
PMID: 31367578 DOI: 10.4041/kjod.2019.49.4.235

Objective: This study examined bone microstructure restoration after rapid maxillary expansion (RME) with and without corticotomy over multiple retention periods.
Methods: Eighteen male Dorper sheep were randomly distributed into three groups (n = 6 each group): group 1, RME with corticotomy on the buccal and palatal sides; group 2, conventional RME treatment; and group 3, no treatment. Post-RME, trabecular bone microstructure and new bone formation were evaluated by using microcomputed tomography (microCT) and histomorphometry after a 4- or 12-week retention period. Intergroup differences in bone quality and bone remodeling were analyzed by using two-way analysis of variance with Bonferroni post-hoc test.
Results: The bone volume fraction (bone volume [BV]/total volume [TV]) values relative to the control in groups 1 and 2 were 54.40% to 69.88% after the 4-week retention period and returned to approximately 80% after the 12-week retention period. The pooled BV/TV values of the banded teeth in groups 1 and 2 were significantly lower than those of the control after the 4-week retention period (p < 0.05). However, after the 12-week retention period, the pooled BV/TV values in group 2 were significantly lower than those in groups 1 and 3 (p < 0.05). Histomorphological analysis showed that the new bone formation area in group 1 was approximately two to three times of those in group 2 and control.
Conclusions: Corticotomy significantly enhanced the restoration of bone quality after the retention periods for banded teeth. This benefit might result from the increased new bone formation after corticotomy.

Matched MeSH terms: Palate
Fulltext Evaluation of Mandibular Incisive Canal using Cone Beam Computed Tomography in Malaysians

Lim JTS, Kang WJ, Ajit Bapat R, Kanneppady SK, Pandurangappa R

J Maxillofac Oral Surg, 2019 Dec;18(4):596-603.
PMID: 31624443 DOI: 10.1007/s12663-018-1168-2

Objectives: The risk of damaging the mandibular incisive canal (MIC) during surgery in the anterior mandible should not be overlooked. Hence, preoperative radiographic assessment is essential to avoid complications. This study was aimed to estimate the length of the MIC in the interforaminal safe zone, to analyse its course in relation to the lingual and the buccal cortical plates of the mandible using cone beam computed tomography (CBCT) scans and to relate the above findings to age, gender, dental status and Malaysian races.
Methods: Retrospective analysis of 100 CBCT scans (n = 200) was performed on both sides of the mandible. Cross-sectional and panoramic images were reconstructed. The length of the MIC and the horizontal distances between the MIC and the buccal and the lingual cortical plates were measured at the three different points (starting, mid-, end points). Independent samples t-test and one-way ANOVA test were used to analyse the variation in the length and course of the MIC in gender, age, dental status and Malaysian races.
Results: The mean length of the MIC was 11.31 ± 2.65 mm, with the Malays having the longest MIC, followed by the Chinese and the Indians (p

Matched MeSH terms: Palate, Hard

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