Nostril Morphometry Evaluation before and after Cleft Lip Surgical Correction: Clinical Evidence.
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ABSTRACT:
SUBMITTER: Feijo MJ
PROVIDER: S-EPMC4296992 | biostudies-other | 2014 Apr
REPOSITORIES: biostudies-other
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Project description:The relapse of nasal deformity is a challenge for modern correction of cleft lip. A comprehensive understanding in the biomechanical perspective of both the formation and correction of the cleft lip nasal deformity would lead to improved stability of the corrective outcome. In this study, a finite element model of secondary cleft lip nasal deformity was constructed, on which two critical corrective maneuvers were mimicked in the form of force-loading. The intercrural suture was simulated by a force loaded at the intermediate crus of the alar cartilage directing anteriorly and medially, and the suture suspending the alar cartilage to the upper lateral cartilage was simulated by a force loaded at the lateral crus directing superiorly and medially. The equivalent von-mises stress and the total deformation consequent to different patterns of loading were captured. Our biomechanical analyses suggested that the intercrural suture at the nasal tip might be more effective in generating widespread morphological change than the suspension suture, but left much higher level of stress within the skin envelope if placed too high. Synergistic effect was observed between the two sutures in both the resultant deformation and stress. In addition, our simulations were partially supported by clinical photogrammetry data.
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Project description:BackgroundCleft lip and palate repair requires a deep and small surgical field and is usually performed by surgeons wearing surgical loupes. Surgeons with loupes can obtain a wider surgical view, although headlights are required for the deepest procedures. Surgical microscopes offer comfort and a clear and magnification-adjustable surgical site that can be shared with the whole team, including observers, and easily recorded to further the education of junior surgeons. Magnification adjustments are convenient for precise procedures such as muscle dissection of the soft palate.MethodsWe performed a comparative investigation of 18 cleft operations that utilized either surgical loupes or microscopy. Paper-based questionnaires were completed by staff nurses to evaluate what went well and what could be improved in each procedure. The operating time, complication rate, and scores of the questionnaire responses were statistically analyzed.ResultsThe operating time when microscopy was used was not significantly longer than when surgical loupes were utilized. The surgical field was clearly shared with surgical assistants, nurses, anesthesiologists, and students via microscope-linked monitors. Passing surgical equipment was easier when sharing the surgical view, and preoperative microscope preparation did not interfere with the duties of the staff nurses.ConclusionsSurgical microscopy was demonstrated to be useful during cleft operations.
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Project description:Upper lip scars are at risk of hypertrophy. Our center therefore uses microporous tape and silicone sheeting for postoperative scar care following cleft lip repair. However, some babies have previously ingested their silicone sheeting, which has the potential for respiratory compromise or gastrointestinal obstruction. Self-dry silicone gel is reportedly also effective for preventing hypertrophic scars. Hence, we sought to test whether silicone gel, which cannot be ingested whole, might be non-inferior to silicone sheeting for controlling against upper lip scar hypertrophy. This was a mixed prospective and retrospective case-controlled clinical trial involving patients undergoing unilateral cleft lip repair, 29 of whom received standard postoperative silicone sheeting (control group) and another 33 age-matched consecutive patients who received self-dry silicone instead. The Vancouver scar scale, visual analogue scale and photographically assessed scar width assessments were the same in both groups at six months after surgery. In conclusion, silicone gel appears to be non-inferior to silicone sheeting for postoperative care of upper lip scars as judged by scar quality at six months, but silicone sheeting has the safety disadvantage that it can be swallowed whole by babies. It is thus recommended that silicone gel be used for upper lip scar management in babies.
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Project description:Management of cleft lip and palate requires a unique understanding of the various dimensions of care to optimize outcomes of surgery. The breadth of treatment spans multiple disciplines and the length of treatment spans infancy to adulthood. Although the focus of reconstruction is on form and function, changes occur with growth and development. This review focuses on the surgical management of the primary cleft lip and nasal deformity. In addition to surgical treatment, the anatomy, clinical spectrum, preoperative care, and postoperative care are discussed. Principles of surgery are emphasized and controversies are highlighted.
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Project description:BackgroundCongenital heart disease (CHD) frequently occurs in conjunction with extracardiac developmental anomalies, including cleft malformations. The clinical impact of concomitant cleft disease on the surgical management of CHD has not been studied. We evaluated cardiac surgical outcomes in patients with concomitant CHD and cleft lip and/or palate (CL/P).MethodsPatients with CHD + CL/P managed at our institution between January 2004 and December 2018 were included. Demographic, operative, and follow-up data were retrospectively collected and analyzed using SAS 9.4. Chi-square tests were used for categorical variables and t test or Wilcoxon rank sum tests for continuous variables. Significance of P < .05 was used.ResultsThere were 127 patients with CHD + CL/P; 63 (50%) were boys. Compared to the general CHD population, patients with CHD + CL/P demonstrated an enrichment of atrial septal defects (10.5% vs 34%), tetralogy of Fallot/double outlet right ventricle (6.4% vs 15.7%), arch defects (4.5% vs 10.2%), truncus arteriosus (1.2% vs 3.1%), and total anomalous pulmonary venous return (1.0% vs 2.4%). Of 63 patients who underwent CHD repair, 58 (92%) did so prior to CL/P repair at 21.5 (6-114) days of age. Compared to CHD lesion-matched patients undergoing cardiac surgical repair at our institution, patients with CL/P had a 2- to 3.7-fold longer intensive care stay, 1.8- to 2.6-fold longer hospital stay, and 6- to 13.5-fold increase in major morbidity, without a significant difference in mortality.ConclusionsCardiac outflow tract defects are particularly overrepresented in CL/P patients. The presence of CL/P increases the complexity of postoperative care after CHD surgery, without a significant impact on mortality.
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Project description:Background and Objectives: Previous literature has disclosed that facial attractiveness affects the esthetic evaluation of nose and lip deformity on frontal and lateral photographs. However, it has never been debated if the removal of the external facial features on three-dimensional (3D) models ("cropped assessment bias") could provide a considerable usefulness in the interpretation and comparison of the results. Additionally, it has been assumed on two-dimensional (2D) studies that esthetic assessment biases with respect to observer gender, and it is not acknowledged if and to the extent that "gender assessment bias" may be influenced by a three-dimensional layout. The aim of this study is to investigate if facial traits and observers' gender may affect the esthetic ratings of unilateral cleft lip and palate (UCLP) patients after soft tissue reconstruction. Materials and Methods: Three-dimensional images of ten UCLP patients' images were acquired before the intervention (T0), one-month (T1) and six-months (T2) postoperative. Geomagic® software (version 2014; 3D Systems, Rock Hill, SC, USA) was used to remove the external facial features of 3D surface models. Five-point scale developed by Asher-McDade et al. was used to rate both nasolabial attractiveness and impairment for full-face (FF) and cropped-face (CF) 3D images. Forty-three judges (21 males, 22 females) were enrolled for the esthetic evaluation. Intraclass correlation coefficient (ICC) was used to test intra- and inter-examiner reliability; a value of 0.7 was set as the minimum acceptable level of reliability. Results: When comparing the 2 sets of observations (FF and CF), the ICC ranged from 0.654 to 0.823. Concerning gender assessment bias, the ICC ranged from 0.438 to 0.686 and from 0.722 to 0.788 for males and females, respectively. Concerning inter-examiner reliability, ICC for questions 2-7 ranged from 0.448 to 0.644 and from 0.659 to 0.817 at T0 and T2, respectively. Conclusions: The removal of external facial features provides subtle differences on the esthetic assessment of UCLP patients. Moreover, based on our data, examiners' gender differences may affect esthetic assessment of UCLP patients. Despite the subjectivity of esthetic judgments, a reliable, validated and reproducible scoring protocol should consider the influence of gender differences on 3D esthetic assessment of UCLP patients.
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Project description:Orofacial clefts are common birth defects and can occur as isolated, nonsyndromic events or as part of Mendelian syndromes. There is substantial phenotypic diversity in individuals with these birth defects and their family members: from subclinical phenotypes to associated syndromic features that is mirrored by the many genes that contribute to the etiology of these disorders. Identification of these genes and loci has been the result of decades of research using multiple genetic approaches. Significant progress has been made recently due to advances in sequencing and genotyping technologies, primarily through the use of whole exome sequencing and genome-wide association studies. Future progress will hinge on identifying functional variants, investigation of pathway and other interactions, and inclusion of phenotypic and ethnic diversity in studies.
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Project description:Nonsyndromic cleft lip with or without cleft palate (NSCLP) is a common birth defect for which only ~ 20% of the underlying genetic variation has been identified. Variants in noncoding regions have been increasingly suggested to contribute to the missing heritability. In this study, we investigated whether variation in craniofacial enhancers contributes to NSCLP. Candidate enhancers were identified using VISTA Enhancer Browser and previous publications. Prioritization was based on patterning defects in knockout mice, deletion/duplication of craniofacial genes in animal models and results of whole exome/whole genome sequencing studies. This resulted in 20 craniofacial enhancers to be investigated. Custom amplicon-based sequencing probes were designed and used for sequencing 380 NSCLP probands (from multiplex and simplex families of non-Hispanic white (NHW) and Hispanic ethnicities) using Illumina MiSeq. The frequencies of identified variants were compared to ethnically matched European (CEU) and Los Angeles Mexican (MXL) control genomes and used for association analyses. Variants in mm427/MSX1 and hs1582/SPRY1 showed genome-wide significant association with NSCLP (p ≤ 6.4 × 10-11). In silico analysis showed that these enhancer variants may disrupt important transcription factor binding sites. Haplotypes involving these enhancers and also mm435/ABCA4 were significantly associated with NSCLP, especially in NHW (p ≤ 6.3 × 10-7). Importantly, groupwise burden analysis showed several enhancer combinations significantly over-represented in NSCLP individuals, revealing novel NSCLP pathways and supporting a polygenic inheritance model. Our findings support the role of craniofacial enhancer sequence variation in the etiology of NSCLP.
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Project description:This study examined the association between markers in transforming growth factor alpha (TGFA) and isolated, non-syndromic cleft lip with/without palate (CL/P) using a case-parent trio design, considering parent-of-origin effects. We also tested for gene-environmental interaction with common maternal exposures, and for gene-gene interaction using markers in TGFA and another recognized causal gene, IRF6. CL/P case-parent trios from four populations (76 from Maryland, 146 from Taiwan, 35 from Singapore, and 40 from Korea) were genotyped for 17 single nucleotide polymorphisms (SNPs) in TGFA. The transmission disequilibrium test was used to test individual SNPs, and the parent-of-origin likelihood ratio test (PO-LRT) was used to assess parent-of-origin effects. We also screened for possible gene-environment interaction using PBAT, and tested for gene-gene interaction using conditional logistic regression models. When all trios were combined, four SNPs showed significant excess maternal transmission, two of which gave significant PO-LRT values [rs3821261: P = 0.004 and OR(imprinting) = 4.17; and rs3771475: P = 0.027 and OR(imprinting) = 2.44]. Haplotype analysis of these two SNPS also supported excess maternal transmission. We saw intriguing but suggestive evidence of G x E interaction for several SNPs in TGFA when either individual SNPs or haplotypes of adjacent SNPs were considered. Thus, TGFA appears to influence risk of CL/P through unconventional means with an apparent parent-of-origin effect (excess maternal transmission) and possible interaction with maternal exposures.
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Project description:Cleft lip with or without cleft palate is present in approximately one in 500 to 700 live births, representing the most common congenital craniofacial anomaly. Previously, the authors developed a unique murine model with compound Pbx deficiency that exhibits fully penetrant cleft lip with or without cleft palate. To investigate the possibility of tissue repair at an early gestational stage, the authors designed a minimally invasive surgical approach suitable for intrauterine repair using Wnt9b-soaked collagen microspheres to restore craniofacial developmental programs for cleft correction.Collagen microspheres with diameters ranging from 20 to 50 ?m were fabricated to serve as a delivery vehicle for Wnt9b. At gestational day 11.5, wild-type and Pbx-deficient murine embryos were isolated. Microspheres soaked in murine purified Wnt9b protein were microsurgically implanted at the midface lambdoidal junction. Embryos were cultured in a 37°C modified whole-embryo culture system.Targeted release of Wnt9b resulted in augmented Wnt expression at the lambdoidal junction. Microsurgical implantation of Wnt9b-soaked microspheres resulted in cleft correction in 27.1 percent of the Pbx-deficient embryos. The difference in the ratio of the areas of clefting between implanted and nonimplanted embryos was significant (p < 0.05).Ex utero correction of cleft lip with or without cleft palate in the authors' murine model by means of microsurgical intervention and targeted delivery of Wnt proteins is an innovative and promising strategy. Although further refinement and optimization of this technique will be required to improve efficacy, the authors believe that this approach will open new avenues toward unconventional prenatal interventions for patients with cleft lip with or without cleft palate, and provide future approaches for prenatal repair of other congenital head and neck disorders.
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