Cleft Lip Cases Research Articles

Cleft lip repair: are outcomes between unilateral and bilateral clefts comparable?

This study sought to compare patient demographics, operative course, and peri-operative outcomes between unilateral and bilateral cleft patients. Primary cleft lip repairs were isolated from the National Surgical Quality Improvement Program Pediatric Database (NSQIP-P). Unilateral and bilateral cases of primary cleft lip were identified by ICD codes. Demographics, comorbidities, and post-operative outcomes were compared between cohorts. Patients were propensity matched to control for differences before repeating the analysis. About 4550 cleft lip repairs were evaluated over the 5-year period. Of the cases where the cleft type was identifiable, 75.5% were unilateral clefts and 24.5% were bilateral clefts. The bilateral cleft population had significantly more comorbidities including higher rates of ventilator dependence (1.0% versus 0.4%, p = 0.02), asthma (1.6% versus 0.7%, p = 0.011), tracheostomy (1.6% versus 0.5%, p < 0.001), gastrointestinal disease (16.9% versus 12.7%, p < 0.001), previous cardiac surgery (3.6% versus 2.2%, p = 0.015), developmental delay (9.9% versus 4.6%, p < 0.001), structural central nervous system abnormalities (5.0% versus 2.5%, p < 0.001), and nutritional support (8.0% versus 3.2%, p < 0.001). Following propensity matching, there were no significant differences in complications, readmissions, or reoperations between the cohorts. Patients with bilateral cleft lip have significantly more comorbidities than unilateral cleft lip patients. However, peri-operative outcomes are comparable between the groups.

EVALUATION OF ASSOCIATION OF CLEFT LIP AND CLEFT PALATE WITH CERVICAL VERTEBRAL ANOMALIES: A LATERAL CEPHALOGRAPH STUDY

The precise origin of cervical vertebral anomalies is still unstated, but it has been suggested that the association between abnormal development of cervical vertebrae and the maxilla and the mandible might be caused by a developmental fault of the mesenchyme as these structures might be dependent on the similar para-axial mesoderm. Hence it is appropriate to focus on this area of research and to consider the craniofacial morphology as an important diagnostic tool in Orthodontic treatment planning. The present study was planned to evaluate the association of Cleft lip and Cleft palate with cervical vertebral anomalies.&#x0D; The present study was planned in Department of Pediatric and Preventive Dentistry, Buddha Institute of Dental Sciences &amp; Hospital, Patna, Bihar, India. Thirty cases of Cleft Lip and Cleft Palate (CLCP) patients were enrolled in the present study. The age of the patients ranged from 5 years to 15 years. The cleft sample were subdivided into patients with CP only, and unilateral cleft lip and palate (UCLP) and bilateral cleft lip and palate (BCLP). Radiographs were examined on a film viewer by a single examiner. The profile of first four cervical vertebrae and Atlanto Occipital Articulation were structurally traced on an acetate paper with 3H lead pencil under optimum illumination and Cervical Vertebral Anomalies (CVA) were registered and categorized into Posterior Arch Deficiencies - PAD (dehiscence and spina bifid) and fusion.&#x0D; The present study concludes the association between cleft lip and palate and Cervical Vertebral Anomalies indicating that CVA may be implicated as the etiology of cleft lip and palate. The present study showed a specific relation between the Cleft Palate and cervical anomalies and the vertebral anomalies following a specific pattern in different types of cleft was found to be PAD which occurred more frequently in UCLP and CP only and fusion occurring significantly more often in BCLP.&#x0D; Keywords: Cleft Lip and Cleft Palate; Cervical Vertebral Anomalies; Dehiscence; Fusion; Lateral Cephalograph

How Should We Decide Whether and When Some Care Is Better Than No Care?

This case of cleft lip and palate repair by a surgical mission team is common. Low-risk, single-procedure surgical interventions requiring minimal follow-up with substantial quality of life improvement are well suited for this type of mission. However, cleft repair can also be quite complex and require multiple surgeries and other care over time, postoperative surveillance, and speech therapy. These benefits and burdens prompt us to investigate, from clinical and ethical perspectives, whether and when some surgical care is better than none. We argue that, when performing surgical interventions in low- and middle-income countries, mission teams should consider a systems-based approach that emphasizes collaboration, context, and sustainability.

Median cleft lip: an uncommon and unique anomaly

Median or midline cleft lip [MCL] is an uncommon anomaly characterized by a midline vertical cleft through the upper lip and are either isolated or part of multiple anomalies. It can involve the pre-maxilla, the nasal septum, and the central nervous system. MCL includes Complete (42%), Incomplete (49%), and Minor forms (9%). The three main groups distinguished were: 1. Isolated MCL; 2. MCL with craniofacial malformations; and 3. MCL with extra-facial malformations. To analyze two operated cases of median cleft lip and review the relevant literature. The details of two cases of median cleft lip that were operated in 2017 were analysed. Both cases underwent wedge excision with the classical inverted V incision and muscle reconstruction with satisfactory result. Both the patients had no syndromic association or associated anomaly. All cases of MCL require evaluation for associated abnormalities. Isolated MCL can be repaired surgically with a good outcome.

Breaking the News: Parents' Experiences of Receiving an Antenatal Diagnosis of Cleft Lip.

In high-income countries, many cases of cleft lip are now identified at the 18- to 21-week pregnancy scan. The manner in which a diagnosis is communicated is vital for long-term parental well-being, yet previous studies have been indicative of parental dissatisfaction. The aims of the present study were to examine the experiences of parents who received an antenatal diagnosis of cleft lip in their unborn child and to offer pragmatic suggestions for improving the diagnostic experience in practice. An online, mixed-methods survey was designed and distributed by the Cleft Lip and Palate Association. Data from 574 eligible parents were analyzed using descriptive statistics and inductive content analysis. Although survey responses indicated positive diagnostic experiences overall, respondents perceived a lack of sensitivity among sonographers, long waiting times between referrals, and a lack of appropriate and reliable information. Respondents also reported a number of misconceptions about cleft lip and/or palate and its prognosis, as well as a variety of initial concerns about their own ability to cope with the anticipated challenges. Findings emphasize the importance of providing accurate and individualized information to prospective parents, in a sensitive manner, so they can adjust to their child's diagnosis and prepare for the birth appropriately. Given that antenatal screening for cleft lip is becoming more fully integrated into routine practice, more training for health-care professionals, improved access to reliable information in a variety of formats, and stronger links between local hospitals and specialist cleft services may be needed.

The prenatal diagnosis and classification of cleft palate: the role and value of magnetic resonance imaging.

The aim of this study was to evaluate the value of MRI in the prenatal diagnosis and classification of cleft palate (CP). We collected 94 fetal cases that were suspected of cleft palate with or without cleft lip by prenatal ultrasound (US) and then carried out further MRI to examine the entire body of each fetus within 1week. The diagnoses resulting from MRI and US examination were compared separately with the final diagnoses obtained from postnatal physical examination or fetal autopsy. The diagnostic accuracy between MRI and US was then determined. During the follow-up period, the results for 6 fetuses (6.38%) were lost. Of the remaining 88 cases, the final diagnoses identified 23 cases of cleft lip (CL), 45 cases of unilateral cleft lip with cleft palate (UCLP), 4 cases of median cleft lip with cleft palate (MCLP), 12 cases of bilateral cleft lip with cleft palate (BCLP), 3 cases of unilateral cleft lip and cleft alveolus (CLA), and 1 case of isolated cleft palate (CPO). The total accuracy rate of US was 59.09%, while that of MRI was 92.05%. More importantly, 81 cases were accurately identified by MRI; the accuracy rate for CL, UCLP, MCLP, BCLP, CLA, and CPO was 86.96%, 95.56%, 100%, 91.67%, 66.67%, and 100%, respectively. Our results suggest that MRI could be a useful adjunct to US examination in the prenatal diagnosis of fetuses with cleft palate, and further demonstrates the classification and degree of involvement of the cleft palate. • MRI is a useful adjunct to prenatal ultrasound. • MRI has a higher accuracy rate for CP. • The accurate classification of CP diagnosed by MRI can guide clinical management.

Cleft Palate with or without Cleft Lip: The Role of Retronasal Triangle View and Maxillary Gap at 11–14 Weeks

Objective: To evaluate the presence of maxillary gap (MG) and abnormal retronasal triangle (RT) as markers of cleft palate (CP) with and without cleft lip in the first trimester and to assess their association with the type of orofacial cleft (OC). Methods: The RT and the mid-sagittal view of the face were evaluated retrospectively by two operators in 26 fetuses with OC and in 80 normal controls to detect abnormal RT and/or MG. The agreement between operators was calculated. Results: Amongst the 26 fetuses, there were 15 cases of bilateral, 6 cases of unilateral, and 4 cases of median cleft lip and palate, and 1 case of CP alone. The MG was observed in 18 cases by operator 1 and in 17 cases by operator 2; an abnormal RT was detected in 21 cases by operator 1 and in 22 cases by operator 2. Great agreement between operators was obtained. In controls, MG or abnormal RT was suspected in 6 and 2–4% of cases, respectively. Conclusions: RT seems to be more sensitive compared to MG; however, the latter showed an additional diagnostic ability when the secondary palate was involved. Both approaches in combination could be useful in detecting OC in the first trimester.

A case of cleft lip and palate performed a comprehensive oral rehabilitation combined dental implants with orthognathic surgery

A case of cleft lip and palate performed a comprehensive oral rehabilitation combined dental implants with orthognathic surgery

The prevalence of developmental anomalies among school children in Southern district of Andhra Pradesh, India.

Background:The developmental anomalies of oral cavity are malformations affecting dental and oral tissues. Anomalies of teeth can be associated with primary, mixed or adult dentitions. Anomalies are the results of perturbations in the developmental stages of tissues which may be influenced by genetic and/or environmental factors.Aim and Objectives:The primary objective of this study was to estimate the prevalence of oral and dental anomalies among school attending children in Chittoor and Kadapa districts of Andhra Pradesh. The secondary objective of this study was to compare occurrence of anomalies based on the age stratification to denote primary, mixed and adult dentitions.Materials and Methods:A total of 5000 school children, aged 3–15 years were invited to participate in the study. Information regarding age, sex, level of school education, brushing and hygiene habits were collected using a questionnaire. Intra- and extra-oral examinations were conducted by trained dental surgeons. Clinical data were collected by a single examiner and the details of these anomalies were recorded on the data sheet of the study. The obtained data were statistically analyzed using Chi-square test.Results:Overall prevalence of developmental anomalies was 11.40% and documented 14 types of anomalies. The prevalence of documented anomalies is as follows: tongue-tie 197 (3.90%), dental fluorosis 171 (3.40%), high frenal attachments 156 (3.10%), cusp of Carabelli 14 (0.30%), supernumerary teeth 11 (0.20%), microdontia 4 (0.10%), congenitally missing teeth 4 (0.10%), lip pits 3 (0.08%), fusion 2 (0.04%), retained deciduous teeth 2 (0.04%) and one case of angular cheilitis, cleft lip and cleft palate, talon cusp, amelogenesis imperfecta (0.02%). The prevalence of dental anomalies was 18.10% in 3–5 years, 52.30% among 6–12 years and 29.6% in 13–15 years. Chi-square test was statistically significant (P = 0.003).Conclusions:Tongue-tie was the most frequent oral tissue developmental anomaly and fluorosis was the most common developmental anomaly affecting dental tissue. The prevalence rate of the study was compared with studies published from other geographical regions in India. The variations in the reported prevalence of developmental anomalies are probably related to genetic and environmental conditions.

Median cleft lip – A case report of rare congenital anomaly

Median cleft lip is a midline vertical cleft through the upper lip in the absence of a prolabial remnant. This is a rare anomaly with very few cases described in the literature. This may occur as a sporadic event or as a part of an inherited sequence of anomalies. It arises embryologically from incomplete fusion of the medial nasal prominences. The author presents a rare case of median cleft lip, broad nose, and wide ala with hypertelorism. The embryology, presentation, and surgical technique for treating these cases are discussed.

Cosmetic Outcome Following Cleft Lip and Palate Repair: A Case Series

Cleft Lip and Palate are the most common congenital abnormalities of the orofacial structures. Along with the cosmetic problems, there may be associated feeding problems, speech problems and dental anomalies. Cleft Repair Surgeries are aimed at restoring the normal anatomy. The aesthetic and cosmetic outcome is a major determinant of the success of Cleft repair surgeries. We hereby present five pictorial cases of Cleft Lip and/or Palate who have undergone Repair surgeries at our institution, with their corresponding long-term cosmetic outcomes.

Association of low-frequency genetic variants in regulatory regions with nonsyndromic orofacial clefts.

Genome-wide scans have shown that common risk alleles for orofacial clefts (OFC) tend to be located in noncoding regulatory elements and cumulatively explain only part of the heritability of OFCs. Low-frequency variants may account for some of the "missing" heritability. Therefore, we scanned low-frequency variants located within putative craniofacial enhancers to identify novel OFC risk variants and implicate new regulatory elements in OFC pathogenesis. Analyses were performed in a multiethnic sample of 1,995 cases of cleft lip with or without cleft palate (CL/P), 221 cases with cleft palate (CP) only, and 1,576 unaffected controls. One hundred and nineteen putative craniofacial enhancers identified from ChIP-Seq studies in craniofacial tissues or cell lines contained multiple low-frequency (0.01-1%) variants, which we genotyped in participants using a custom Illumina panel. Two complementary statistical approaches, sequence kernel association test and combined multivariate and collapsing, were used to test association of the aggregated low-frequency variants across each enhancer region with CL/P and CP. We discovered a significant association between CP and a branchial arch enhancer near FOXP1 (mm60; p-value = .0002). Additionally, we observed a suggestive association between CL/P and a forebrain enhancer near FOXE1 (hs1717; p-value = .001). These findings suggest that low-frequency variants in craniofacial enhancer regions contribute to the complex etiology of nonsyndromic OFCs.

The Effects of Maternal Mirroring on the Development of Infant Social Expressiveness: The Case of Infant Cleft Lip.

Parent-infant social interactions start early in development, with infants showing active communicative expressions by just two months. A key question is how this social capacity develops. Maternal mirroring of infant expressions is considered an important, intuitive, parenting response, but evidence is sparse in the first two months concerning the conditions under which mirroring occurs and its developmental sequelae, including in clinical samples where the infant's social expressiveness may be affected. We investigated these questions by comparing the development of mother-infant interactions between a sample where the infant had cleft lip and a normal, unaffected, comparison sample. We videotaped dyads in their homes five times from one to ten weeks and used a microanalytic coding scheme for maternal and infant behaviours, including infant social expressions, and maternal mirroring and marking responses. We also recorded maternal gaze to the infant, using eye-tracking glasses. Although infants with cleft lip did show communicative behaviours, the rate of their development was slower than in comparison infants. This group difference was mediated by a lower rate of mirroring of infant expressions by mothers of infants with cleft lip; this effect was, in turn, partly accounted for by reduced gaze to the infant's mouth, although the clarity of infant social expressions (indexed by cleft severity) and maternal self-blame regarding the cleft were also influential. Results indicate the robustness of parent-infant interactions but also their sensitivity to specific variations in interactants' appearance and behaviour. Parental mirroring appears critical in infant social development, likely supported by the mirror neuron system and underlying clinical and, possibly, cultural differences in infant behaviour. These findings suggest new avenues for clinical intervention.

Management of cleft lip and palate in Egypt: A National survey.

Background:Variable protocols for the management of cleft lip and/or palate (CLP) patients are currently used. However, to our knowledge, there are no previously published data about cleft management and practice in Egypt.Materials and Methods:One-hundred questionnaires were distributed to cleft surgeons attending the annual meeting of the Egyptian Society of Plastic and Reconstructive Surgeons in March 2016 to investigate timing, techniques and complications of cleft surgery. Seventy-two colleagues returned the questionnaire, and the data were analysed using Microsoft Excel software.Results:The majority of cleft lip cases are repaired between 3 and 6 months. Millard and Tennison repairs for unilateral cleft lip, while Millard and Manchester techniques for bilateral cleft lip are the most commonly performed. Cleft palate is usually repaired between 9 and 12 months with the two-flap push-back technique being the most commonly used. The average palatal fistula rate is 20%. Pharyngeal flap is the method of choice to correct velopharyngeal incompetence. Polyglactin 910 is the most commonly used suture material in cleft surgery in the country. Multidisciplinary cleft management is reported only by 16.5% of participants.Conclusion:Management of CLP in Egypt is mainly dependent on personal preference, not on constitutional protocols. There is a lack of multidisciplinary approach and patients’ registration systems in the majority of centres. The establishment of cleft teams from the concerned medical specialties is highly recommended for a more efficient care of cleft patients.

The refinement of the median tubercle of cleft lip.

Aim:Repair of cleft lip aims to bring symmetry and provide normal aesthetics for the lip. Several techniques have been employed; however, the finer aspect of median lip tubercle has not been emphasised in cleft lip repair.Materials and Methods:We have modified cleft lip repair, both unilateral and bilateral, preserving all the tissues of the median tubercle to provide normal-looking median tubercle of the lip. The modified technique was carried out by a single surgeon on 322 cases of unilateral cleft lip and 68 cases of bilateral cleft lip. Follow-up was done for 1–3 years on these children to evaluate the outcome.Results:The evaluation showed excellent results in more than 80% of patients in unilateral cleft lip repair, on 1–3 years of follow-up; 20% had acceptable results. Of 68 patients with bilateral cleft lip, none had any complication, and excellent results were obtained in 70%. Result was rated acceptable in 30%; 15% may need revision surgery for white roll and vermilion adjustment.Conclusion:We present the technique of reconstructing a normal-looking median tubercle in cleft lip repair. The technique was modified based on the study of normal upper lip and embryology of cleft lip, with emphasis on creating better median tubercle of the lip.

Evaluation of the fetal palate at 11 to 13 (+6) weeks of gestation based on an analysis of static ultrasound images using modern IT techniques.

To describe a new computer-based technique to isolate the shape of the fetal palate visible in the midsagittal plane from static ultrasound images routinely used to measure nuchal translucency. This is a retrospective interpretation of images of the midsagittal view of the fetal face at 11 to 13 (+6)weeks of gestation in 7 cases of cleft lip and palate (CLP) and 7 normal controls. The images were subjected to pattern analysis. Proprietary software was applied and for each CLP case, and palatine bones with different forms from those of fetuses in the control group were recorded. In 4 cases, an image of a continuous palatine bone was observed at a threshold of 180, whereas the remaining 3 images were obtained at 128. A continuous palatine bone structure was not observed in any fetus from the CLP group, even at a level of 128, when the surrounding structures were visible. The application of pattern analysis to a 2D frozen image is a new approach for prenatal diagnostics. This technique may be a helpful tool for physicians and could assist in the diagnosis of cleft palate.

ICD‐10 impact on ascertainment and accuracy of oral cleft cases as recorded by the Brazilian national live birth information system

We compared Brazilian oral cleft (OC) frequencies between the population-based Brazilian System of Live Birth (SINASC) and the hospital-based Latin American Collaborative Study of Congenital Malformations (ECLAMC), trying to understand the paucity of cleft of lip and palate (CLP) in the first system. SINASC uses the International Classification of Disease version 10 (ICD-10) for congenital defects coding, ECLAMC uses ICD-8 with modifications. In SINASC, the CLP frequency was 1.7 per 10,000 (95% confidence limits 1.7-1.8), cleft lip (CL) 1.6 (1.5-1.7), and cleft palate (CP) 2.0 (1.9-2.1). In ECLAMC, the CLP frequency was 10.4 per 10,000 (9.0-12.1), CL 5.5 (4.5-6.7), and CP 4.4. (4.5-6.7). In SINASC, only 33% of the oral clefts were CLP, versus 51% in ECLAMC. Part of this discrepancy may have been due to the relative excess of CP and CL cases. Although congenital defect frequencies are usually lower in population than in hospital-based registries, differences in the proportion of the main OC categories are not expected and are probably due to ICD-10 coding issues, such as lumping of unilateral CL and CL without other specifications. ICD-10 codes, whose deficiency for oral clefts is fully explained in the literature, lack modifiers for severity, or clinical subtypes. This paper shows the practical aspect of the ICD-10 system deficiency in capturing cleft lip and palate (CLP) subtypes, as demonstrated in SINASC covering three million births per year. Such errors are expected to occur in any registry that uses the ICD-10 coding system, and must be adjusted, given its relevance worldwide.

A novel method of obtaining impression from three-dimensionally printed skull and incorporating medical grade silicone elastomer in fabricating silicone palatal feeding obturators for cleft lip and palate cases

This report aims to depict a novel method in impression taking and feeding obturator fabrication for cleft lip and palate patients, utilizing three-dimensionally printed base of the skull from presurgical computerized tomography scan data. This technique uses soft and biocompatible medical grade room temperature vulcanizing silicone elastomer as the material of choice for fabrication procedure and reviews a representative patient case of a 10-month-old female child with bilateral cleft lip and palate deformity. Before final insertion, a strap was attached to the obturator as a safety precaution in case of accidental swallowing. The use of this unique technique satisfies all benefits of conventional feeding obturator with the added benefits of reducing most of its discomfort and difficulties of direct impression taking from the infant and fabricating process.

Demographic distribution and pattern of orofacial clefts in a Nigerian Urban Hospital

Introduction: Clefts lip and palate are the most common deformities in the head-and-neck region. They vary in the severity of presentation from small notches in the lip to clefts that extend through the alveolar ridge in the mouth and involve the floor of the nostrils/palate. They may either occur in isolation (asyndromic) or in association with other congenital anomalies as part of a syndrome as observed. Patients and Methods: This is a retrospective clinic-based study of records of patients seen with orofacial clefts from 2009 to 2014, at the Army Dental Clinic, Bonny Cantonment, Lagos, Nigeria. Results: A total of 43 cases of cleft lip and palate of varying degrees were seen over the study period. There were slightly more males (n = 23, 53.5%) than that of females (n = 20, 46.5%). The age at presentation ranged from 4 days old to 46 years and the median age was 3 years. In terms of age distribution, patients aged 3 years and below (n = 23; 53%) were the most predominant group. This was followed by the 4–7 years (n = 6; 13.9%) age bracket. Patients aged 8–11 years, 12–15 years, and 28–31 years were equally distributed with 3 (6.9%) each. Conclusion: A significant minority of orofacial clefts patients still present in adulthood possibly due to ignorance about treatment options and poverty to access surgical treatment.

Moustache restoration using follicular unit extraction technique for esthetic repair of prolabial alopecia in adult male patients with repaired bilateral cleft lip: An initial report in eight patients

Prolabial alopecia is an inherent feature of bilateral cleft lip cases, occurring in varying degrees which is worsened following cleft lip repair. In the Southeast Asian population, a strong healthy moustache is considered a symbol of pride and hence many patients seek treatment of their alopetic prolabium due to the cleft lip. The alopetic prolabium also adds to the patients dented confidence by adding to the typical cleft lip look. Among the two techniques mentioned in the literature for hair restoration, namely, follicular unit extraction (FUE) and follicular unit transplantation techniques, the FUE technique offers a minimally invasive and virtually scar less option and is often preferred by most of the patients as well as the surgeons. A very few studies have focused on moustache restoration in adult male patients presenting with prolabial alopecia following bilateral cleft lip repair. This article presents the initial report in 8 adult male patients presenting with prolabial alopecia after repaired bilateral cleft lip that underwent moustache restoration using the FUE technique of hair restoration.