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1.
J Craniofac Surg ; 35(4): e405-e407, 2024 Jun 01.
Article En | MEDLINE | ID: mdl-38666772

This retrospective cross-sectional study reviewed adult patients with operated cleft lip and/or palate (CL/P) and normal control, and performed comprehensive craniofacial and nasal morphological analyses based on lateral cephalometric radiographs. Pearson or Spearman correlation coefficient assessed intraclass correlation. Seven hundred fifty-seven operated patients with CL/P, and 165 noncleft normal controls were enrolled. Among the normal and CL/P groups, S-N-A angle registered positive correlations with nasal base prominence (S-N'-Sn, degrees). Upper facial height (N-ANS, mm) had positive correlations with nasal dorsum length (N'-Prn, mm) and nasal bone length (N-Na, mm). Although in patients with bilateral cleft lip and palate, there were moderate negative correlations ( r =-0.541, P <0.05) with soft tissue facial profile angle (FH-N'Pog', degree) and nasolabial angle (Cm-Sn-ULA, degree). Correlation exists between the morphology of jaw bones and external nose among patients with CL/P. Maxillary sagittal insufficiency is associated with concave nasal profile, and maxilla height is associated with nasal length.


Cephalometry , Cleft Lip , Cleft Palate , Nose , Humans , Cleft Lip/pathology , Cleft Lip/diagnostic imaging , Cleft Lip/surgery , Cleft Palate/pathology , Cleft Palate/diagnostic imaging , Cleft Palate/surgery , Retrospective Studies , Female , Male , Cross-Sectional Studies , Adult , Nose/diagnostic imaging , Nose/anatomy & histology , Nose/pathology , Case-Control Studies , Adolescent , Maxilla/diagnostic imaging , Maxilla/pathology
2.
Orthod Craniofac Res ; 27 Suppl 1: 80-89, 2024 Jun.
Article En | MEDLINE | ID: mdl-38305564

OBJECTIVE: The objective of this study is to measure the morphological changes of the nose and lip in patients with unilateral cleft lip and palate before and after cheiloplasty with primary rhinoplasty (primary correction) in conjunction with Korat-NAM usage. DESIGN: Longitudinal cohort study. SETTING: Cleft Center Maharat Nakhon Ratchasima Hospital, Nakhon Ratchasima, Thailand. SUBJECTS: Twenty-six patients with unilateral cleft lip and palate. INTERVENTIONS: Control group: only active obturator before primary correction. Experimental group: an active obturator and Korat-NAM I before primary correction. A customized endotracheal tube was retained in the nostril for 3 weeks before switching to Korat-NAM II for 1 year. MAIN OUTCOME MEASURES: Six measurements comprising nostril rim length, nostril height, nostril sill width, columella angle, vertical lip height, and horizontal lip length were measured from the patients' photographs. All measurements, except the columella angle, were reported as the cleft side/non-cleft side value ratio. Measurements were taken at the initial appointment, immediately before, 3 weeks after, and 1 year after primary correction. RESULTS: Nostril rim length ratio, nostril height ratio, nostril sill width ratio, columella angle on the cleft side, and vertical lip height ratio were improved using Korat-NAM before and 3 weeks after primary correction. Nostril rim length and height ratios were significantly better than the control group. CONCLUSIONS: Korat-NAM improved nose and lip morphology before primary correction. An overcorrection improved the nose and lip morphology on the cleft side. The nostril rim length and vertical lip height on the cleft side also improved with Korat-NAM II 1 year after primary correction.


Cleft Lip , Cleft Palate , Lip , Nose , Rhinoplasty , Humans , Cleft Lip/surgery , Cleft Lip/pathology , Cleft Palate/surgery , Cleft Palate/pathology , Nose/pathology , Rhinoplasty/methods , Male , Female , Lip/pathology , Longitudinal Studies , Nasoalveolar Molding , Palatal Obturators , Child , Infant
3.
J Plast Reconstr Aesthet Surg ; 89: 134-141, 2024 Feb.
Article En | MEDLINE | ID: mdl-38181634

BACKGROUND: Bilateral cleft lip is a congenital defect often accompanied by secondary lip and nose deformity. The current classification system for secondary cleft lip deformity has limitations in guiding surgical planning. In this article, we report a method for secondary bilateral cleft lip classification that can guide surgery on the basis of the pathological anatomy of the columellar and upper lip. METHODS: Photographs of patients were retrospectively classified into four types on the basis of the ratio of columellar height to alar base width (CH/AW) and upper lip protrusion (UP) to lower lip, as follows: type I - with CH/AW ≥ 0.2 and UP ≥ 0; type II - with CH/AW ≥ 0.2 and UP <0; type III - with CH/AW < 0.2 and UP ≥0; type IV - with CH/AW < 0.2 and UP < 0. Surgical treatments and the change of the nasal profile were documented. RESULTS: A total of 105 patients from January 2008 to December 2018 were included in this study. The nasal profile was significantly improved in type III and IV patients with postoperative CH/AW values close to normal. The upper lip was distinctively retruded in type II and IV patients before treatment, and the postoperative view revealed improved upper lip protrusion with UP values close to normal. Ninety-eight patients reported satisfactory outcomes after treatment. CONCLUSIONS: The new classification method described provides key information regarding the deformity of different types of secondary bilateral cleft lip patients and provides clear guidance for surgical planning on the basis of the anatomical defect of each type.


Cleft Lip , Nose Diseases , Rhinoplasty , Adult , Humans , Cleft Lip/pathology , Rhinoplasty/methods , Retrospective Studies , Nose/abnormalities , Nose Diseases/surgery , Nasal Septum/surgery , Treatment Outcome
4.
Cleft Palate Craniofac J ; 61(2): 326-331, 2024 02.
Article En | MEDLINE | ID: mdl-38092680

OBJECTIVE: To evaluate the long-term outcomes of linear commissuroplasty and linear skin closure with a focus on commissural migration. DESIGN: Retrospective study. PATIENTS: Individuals who underwent transverse facial cleft repair at a single institution between 2004 and 2021. INTERVENTIONS: The disrupted orbicularis oris muscle was reoriented and sutured. A simple linear commissuroplasty technique was used, and the cheek skin was closed linearly without Z-plasty. MAIN OUTCOME MEASURES: The distances from Cupid's bow peak to the oral commissure were measured bilaterally, and the difference between the normal and cleft sides was obtained. Finally, its proportional value as a percentage of the total lip length was calculated from short- and long-term follow-up photographs. Cheek scarring and its effects on melolabial fold breakage were evaluated. RESULTS: Of the 18 patients who underwent transverse facial cleft repair, 12 were included in this study. The mean follow-up period based on medical photographs was 1773.5 days. The average proportional difference was 4.6%, demonstrating no observable commissural migration. There were no consistent trends in the direction of migration, either on the cleft or normal side. In patients with a transverse cleft crossing the melolabial fold, the folds appeared broken before and after the cleft repair surgery. CONCLUSIONS: No significant long-term commissural migration was observed after transverse facial cleft repair with simple linear commissuroplasty and linear skin closure. Deliberate positioning of the new oral commissure, proper myoplasty, and meticulous skin closure with minimal scar burden can be considered key procedures for successful transverse cleft repair.


Cleft Lip , Plastic Surgery Procedures , Humans , Infant , Retrospective Studies , Lip/surgery , Mouth Mucosa/surgery , Cleft Lip/surgery , Cleft Lip/pathology , Cicatrix/surgery
5.
Cleft Palate Craniofac J ; 61(1): 20-32, 2024 01.
Article En | MEDLINE | ID: mdl-35876322

Midface hypoplasia (MFH) is a long-term sequela of cleft lip and palate repair, and is poorly understood. No study has examined the aggregate data on sagittal growth restriction of the midface following repair of the lip, but not palate, in these patients.A systematic review of 3780 articles was performed. Twenty-four studies met inclusion criteria and 11 reported cephalometric measurements amenable to meta-analysis. Patients with Veau class I-III palatal clefts were included so long as they had undergone only lip repair. Groups were compared against both noncleft and unrepaired controls.Cephalometrics were reported for 326 patients (31.3% female). Noncleft controls had an average SNA angle of 81.25° ± 3.12°. The only patients demonstrating hypoplastic SNA angles were those with unilateral CLP with isolated lip repair (77.4° ± 4.22°). Patients with repaired CL had SNA angles similar to noncleft controls (81.4° ± 4.02°). Patients with unrepaired CLP and CL tended toward more protruding maxillae, with SNA angles of 83.3° ± 4.04° and 87.9° ± 3.11°, respectively. Notably, when comparing SNA angles between groups, patients with CLP with isolated lip repair had significantly more hypoplastic angles compared to those with repaired CL (P < .0001). Patients with CLP with isolated lip repair were also more hypoplastic than noncleft controls (P < .0001). In contrast, there was no significant difference between the SNA of patients with repaired CL and controls (P = .648).We found that cleft lip repair only appeared to contribute to MFH in the setting of concurrent cleft palate pathology, suggesting that scarring from lip repair itself is unlikely to be the predominant driver of MFH development. However, studies generally suffered from inadequate reporting of timing, technique, follow-up time, and cleft severity.


Cleft Lip , Cleft Palate , Humans , Female , Male , Cleft Lip/surgery , Cleft Lip/pathology , Cleft Palate/surgery , Cleft Palate/pathology , Face , Maxilla , Cephalometry/methods
6.
Ann Plast Surg ; 92(1): 60-67, 2024 Jan 01.
Article En | MEDLINE | ID: mdl-38117046

BACKGROUND: Several assessment systems of the cleft-related facial deformity have been reported in the medical literature. Assessments have been made from direct clinical evaluations, photographs, on-screen digital images, and 3-dimensional imaging. An evaluation method based on standardized photographic views is developed to evaluate the most common postoperative deformities and to detect the responsible factors for occurrence of these deformities and how to avoid them. MATERIALS AND METHODS: One hundred forty-five cleft lip cases (105 unilateral and 40 bilateral) were evaluated by using standard sheet and scoring system designed by Operation Smile Inc (Virginia Beach). The scoring system is based on photographic analysis of items including Cupid's bow, nasal symmetry, vermilion contour, white roll continuity, and scar quality. RESULTS: In the unilateral cleft cases, we found 0.4% excellent, 48.57% good, 38% fair, and 2.85% poor cases. For bilateral clef lip cases, we found 27.5% excellent, 47.5% good, 17.5% fair, and 7.5% poor outcomes. The most common postoperative deformities were nasal asymmetry, scar hypertrophy, deformed Cupid's bow, and vermilion contour asymmetry. CONCLUSIONS: This objective evaluation system can determine the common cleft lip nasal deformities with detection of the responsible factors. Principles that guide optimum surgical repair have been advocated to avoid the common postoperative deformities. Scar formation is an independent factor that must be managed early and separately to maintain surgical outcomes.


Cleft Lip , Plastic Surgery Procedures , Humans , Cleft Lip/diagnostic imaging , Cleft Lip/surgery , Cleft Lip/pathology , Cicatrix/surgery , Lip/surgery , Surgical Flaps/surgery
7.
Genes (Basel) ; 14(6)2023 06 10.
Article En | MEDLINE | ID: mdl-37372427

Ankyloblepharon-ectodermal defects-cleft lip/palate (AEC) syndrome is a rare autosomal dominant disorder. AEC is caused by mutations in the TP63 gene that encodes the tumor suppressor p63 protein, itself involved in the regulation of epidermal proliferation, development, and differentiation. We present here a typical AEC case of a four-year-old girl with extensive skin erosions and erythroderma of the scalp and the trunk, and to a lesser extent of the limbs, nail dystrophy on the fingers and toes, xerophthalmia, a high-arched palate, oligodontia, and hypohidrosis. Mutation analysis of the TP63 gene detected a de novo missense mutation in exon 14 (c.1799G>T; p.Gly600Val). We discuss the phenotype-genotype correlation by presenting the clinical features of AEC in the patient, and the effect of the detected mutation in p63 structure and function using protein structural modeling, in view of similar cases in the literature. We performed a molecular modeling study in order to link the effect on the protein structure level of the missense mutation G600V. We noted that the introduction of the bulkier Valine residue in place of the slim Glycine residue caused a significantly altered 3D conformational arrangement of that protein region, pushing away the adjacent antiparallel α helix. We propose that the introduced locally altered structure of the G600V mutant p63 has a significant functional effect on specific protein-protein interactions, thus affecting the clinical phenotype.


Cleft Lip , Cleft Palate , Ectodermal Dysplasia , Humans , Cleft Lip/genetics , Cleft Lip/pathology , Cleft Palate/diagnosis , Cleft Palate/genetics , Cleft Palate/pathology , Transcription Factors/genetics , Ectodermal Dysplasia/diagnosis , Ectodermal Dysplasia/genetics , Ectodermal Dysplasia/pathology , Tumor Suppressor Proteins/genetics , Genetic Association Studies
8.
J Craniofac Surg ; 34(5): 1435-1438, 2023.
Article En | MEDLINE | ID: mdl-37143197

Although surgical techniques have developed, the incidence of secondary cleft lip deformities after the primary repair is still high. Asymmetry of Cupid's bow and philtrum is a common presentation and a technical challenge to reconstruct the upper lip. The authors introduce a technique to use the free grafts of the full-thickness scalp for the correction of scars and tissue deficiency in secondary unilateral cleft lip deformity. Thirty-seven patients with a prominent scar, tight upper lip, deformed Cupid's bow and philtrum, and irregular vermillion border were included in this study. The specific points of interest were assessed before and after surgery by independent examiners with both subjective and objective methods. All surgeries were successfully completed, and the assessment results were satisfactory. The rating scores of philtrum improvement was 1.6±0.4, the POSAS result of patient scales was 13.48±3.21, and the observer scale result was 11.98±3.88. The asymmetry of Cupid's bow was corrected ( P =0.004), the central tubercle of vermilion was more natural ( P =0.001), and the irregular vermillion border was improved ( P =0.015). The results presented significant differences before and after surgery. This method could be an optional treatment for repairing scars and tissue deficiency in secondary unilateral cleft lip deformity.


Cleft Lip , Plastic Surgery Procedures , Humans , Cleft Lip/surgery , Cleft Lip/pathology , Cicatrix/etiology , Cicatrix/surgery , Cicatrix/pathology , Scalp/surgery , Lip/surgery
9.
Front Cell Infect Microbiol ; 13: 1159455, 2023.
Article En | MEDLINE | ID: mdl-37143743

Orofacial cleft disorders, including cleft lip and/or palate (CL/P), are one of the most frequently-occurring congenital disorders worldwide. The health issues of patients with CL/P encompass far more than just their anatomic anomaly, as patients with CL/P are prone to having a high incidence of infectious diseases. While it has been previously established that the oral microbiome of patients with CL/P differs from that of unaffected patients, the exact nature of this variance, including the relevant bacterial species, has not been fully elucidated; likewise, examination of anatomic locations besides the cleft site has been neglected. Here, we intended to provide a comprehensive review to highlight the significant microbiota differences between CL/P patients and healthy subjects in various anatomic locations, including the teeth inside and adjacent to the cleft, oral cavity, nasal cavity, pharynx, and ear, as well as bodily fluids, secretions, and excretions. A number of bacterial and fungal species that have been proven to be pathogenic were found to be prevalently and/or specifically detected in CL/P patients, which can benefit the development of CL/P-specific microbiota management strategies.


Cleft Lip , Cleft Palate , Humans , Cleft Lip/diagnosis , Cleft Lip/epidemiology , Cleft Lip/pathology , Cleft Palate/diagnosis , Cleft Palate/epidemiology , Cleft Palate/pathology , Nasal Cavity
10.
Int. j. morphol ; 41(2): 343-348, abr. 2023.
Article Es | LILACS | ID: biblio-1440315

Las fisuras orofaciales representan un grupo heterogéneo de malformaciones congénitas que afectan a distintas estructuras de la cavidad oral y de la cara. Globalmente, los bebés con estos trastornos presentan una mayor morbilidad y mortalidad a lo largo de su vida en comparación con individuos no afectados. Por ello, los avances en la investigación biomédica resultan ineludibles. Así, el objetivo general de este trabajo fue llevar a cabo una revisión bibliográfica para analizar narrativamente los 10 principales estudios primarios sobre fisuras orofaciales llevados a cabo en España, publicados del 2018 hasta la actualidad. Según esto, a nivel institucional, destaca la Universidad Complutense de Madrid (UCM) con cuatro artículos publicados por el grupo de investigación UCM 920202. También sobresale la Universidad Rey Juan Carlos de Madrid, con tres artículos relacionados con diferentes aspectos de la personalidad y la calidad de vida de los pacientes fisurados, así como otras muchas variables cognitivo-emocionales. En relación con la Universidad de Valencia, encontramos dos artículos llevados a cabo en amplias muestras de pacientes con fisuras. Por último, en Barcelona resulta destacable un estudio observacional sobre problemas otorrinolaringológicos en pacientes operados de fisura palatina. En conclusión, si bien en los últimos años se han publicado varios artículos sobre distintos aspectos relacionados con las fisuras, aún queda mucho trabajo por hacer. España debería seguir potenciando proyectos con líneas de trabajo centradas en estas alteraciones del desarrollo craneofacial. Se necesitan estudios amplios, multicéntricos y colaborativos, para ahondar en los mecanismos etiológicos y, en última instancia, en las posibles herramientas para su prevención. Del mismo modo, se necesitan ayudas para dilucidar mejor las cuestiones relacionadas con los tratamientos en todas las dimensiones de la salud, preferentemente a partir de ensayos clínicos controlados aleatorizados, que faciliten la traslación de conocimientos y su accesibilidad universal dentro del sistema sanitario público español.


SUMMARY: Orofacial clefts represent a heterogeneous group of congenital malformations affecting different structures of the oral cavity and face. Overall, infants with these disorders have a higher lifetime morbidity and mortality compared to unaffected individuals. Therefore, advances in biomedical research are unavoidable. Thus, the overall objective of this work was to conduct a literature review to narratively analyse the 10 main primary studies on orofacial clefts carried out in Spain, published from 2018 to date. According to this review, at an institutional level, the Complutense University of Madrid (UCM) is notable with 4 articles published by the UCM 920202 research group. The Rey Juan Carlos University of Madrid also stands out, with three papers related to different aspects of the personality and quality of life of cleft patients, as well as many other cognitive-emotional variables. In relation to the University of Valencia, we found two studies carried out on large samples of cleft patients. Finally, in Barcelona, an observational study on otorhinolaryngological problems in cleft palate patients is noteworthy. In conclusion, although several studies have been published in recent years on different aspects related to clefts, there is still much work to be done. Spain should craniofacial development. Large, multicenter and collaborative studies are needed to delve deeper into the aetiological mechanisms and, ultimately, into the possible tools for their prevention. Similarly, support is needed to better elucidate questions related to treatments in all dimensions of health, preferably randomised controlled clinical trials, which facilitate the transfer of knowledge and its universal accessibility within the Spanish public health system.


Humans , Cleft Lip/pathology , Cleft Palate/pathology , Spain
11.
Plast Reconstr Surg ; 152(1): 126e-133e, 2023 07 01.
Article En | MEDLINE | ID: mdl-36728477

BACKGROUND: Modern cleft lip surgery aims to restore symmetry and create a level, normal-appearing Cupid's bow. However, families' concerns often center on the degree of scarring. The authors hypothesized that the lip angle would be less influential than scar severity in layperson ratings. METHODS: The authors received institutional review board approval for modifying patients' postoperative photographs to create systematic variations displaying different levels of scarring and lip angle. Each child's resulting composite images were presented in pairs to internet raters using Amazon Mechanical Turk. Users selected the simulated postoperative result they felt to be most normal. The Bradley-Terry model was used to determine raters' preferences between different levels of scarring and lip angle. RESULTS: Four children with primary unilateral cleft lip repair had their postoperative photographs modified (mean age, 1.4 years; mean follow-up, 1.0 years). Twelve-hundred crowdsourced pairwise ratings were collected for each patient (4800 combined ratings). For all four children, raters preferred images with more severe scarring than those with a greater lip angle, suggesting uneven lip angle has a more negative effect on perceived appearance. CONCLUSIONS: Online crowdsourcing postoperative lip angle had a significantly greater influence on ratings of normal appearance than does the severity of scarring. Although patients may ask about scars more often, clinically, this study suggests perceptions of a cleft lip repair result may be more likely influenced by the angle of the cleft lip repair.


Cleft Lip , Crowdsourcing , Plastic Surgery Procedures , Child , Humans , Infant , Cleft Lip/surgery , Cleft Lip/pathology , Cicatrix/surgery , Lip/surgery
12.
Cleft Palate Craniofac J ; 60(5): 621-626, 2023 05.
Article En | MEDLINE | ID: mdl-34967231

Chromosome 17 duplication is correlated with an increased risk of developmental delay, birth defects, and intellectual disability. Here, we reported a female patient with trisomy 17 on the whole short arm with bilateral complete cleft lip and palate (BCLP). This study will review the surgical strategies to reconstruct the protruding premaxillary segment, cleft lip, and palate in trisomy 17p patient.The patient had heterozygous pathogenic duplication of chromosomal region chr17:526-18777088 on almost the entire short arm of chromosome 17. Beside the commonly found features of trisomy 17p, the patient also presented with BCLP with a prominent premaxillary portion. Premaxillary setback surgery was first performed concomitantly with cheiloplasty. The ostectomy was performed posterior to the vomero-premaxillary suture (VPS). The premaxilla was firmly adhered to the lateral segment and the viability of philtral flap was not compromised. Two-flap palatoplasty with modified intravelar veloplasty (IVV) was performed 4 months after.Successful positioning of the premaxilla segment, satisfactory lip aesthetics, and vital palatal flap was obtained from premaxillary setback, primary cheiloplasty, and subsequent palatoplasty in our trisomy 17p patient presenting with BLCP. Postoperative premaxillary stability and patency of the philtral and palatal flap were achieved. Longer follow-up is needed to evaluate the long-term effects of our surgical techniques on inhibition of midfacial growth. However, the benefits that the patient received from the surgery in improving feeding capacity and facial appearance early in life outweigh the cost of possible maxillary retrusion.


Cleft Lip , Cleft Palate , Humans , Female , Cleft Lip/genetics , Cleft Lip/surgery , Cleft Lip/pathology , Cleft Palate/genetics , Cleft Palate/surgery , Cleft Palate/pathology , Chromosomes, Human, Pair 17 , Maxilla/abnormalities , Esthetics, Dental , Osteotomy
13.
Cleft Palate Craniofac J ; 60(10): 1337-1341, 2023 10.
Article En | MEDLINE | ID: mdl-35702006

Benign salivary gland tumors are rarely found in children and adolescents compared with adults. Pleomorphic adenomas (PAs), the most common benign salivary gland tumor, account for only 1% of all head and neck lesions and fewer than 5% of all salivary gland tumors in individuals under the age of 16 years. The data on palatal PA in the first 2 decades of life is confined to published case reports and case series. To date, there has never been a report of palatal PA in a patient with cleft lip and palate. Here we describe an adolescent female with bilateral cleft lip and palate with PA of the hard and soft palate who underwent wide local excision and reconstruction with a buccal fat pad and buccal myo-mucosal flap.


Adenoma, Pleomorphic , Cleft Lip , Cleft Palate , Salivary Gland Neoplasms , Adult , Child , Adolescent , Humans , Female , Adenoma, Pleomorphic/diagnostic imaging , Adenoma, Pleomorphic/surgery , Adenoma, Pleomorphic/pathology , Cleft Lip/surgery , Cleft Lip/pathology , Cleft Palate/surgery , Cleft Palate/pathology , Salivary Gland Neoplasms/diagnostic imaging , Salivary Gland Neoplasms/surgery , Salivary Gland Neoplasms/pathology , Palate, Soft
14.
Angle Orthod ; 92(6): 764-772, 2022 11 01.
Article En | MEDLINE | ID: mdl-36279255

OBJECTIVES: To evaluate inflammatory mediator levels and periodontal changes following distraction osteogenesis (DO) in patients with cleft lip and palate (CLP) using mid-maxillary distraction (MMD). MATERIALS AND METHODS: A total of 20 healthy patients with CLP with Class III malocclusion were included. Segmental forward advancement of the anterior maxilla from the second premolars on both sides using DO was performed. A custom-made, tooth-borne distractor connecting buccal molar segments to the anterior maxilla was used for 7 days with 0.5-mm distraction for the first 2 days and then increased to 1 mm daily until overcorrection. Crevicular interleukin IL-1ß and tumor necrosis factor TNF-α levels were measured during distraction. Periodontal clinical parameters and indices were recorded at baseline and 3 and 6 months postoperatively. Soft tissue healing was evaluated histologically at 2 and 4 weeks after distraction. RESULTS: The periodontal parameters remained stable during the follow-up periods. Insignificant increases in the level of inflammatory cytokines compared with the control were observed. Histological findings revealed mild inflammatory and structural changes in the gingiva immediately after distraction, whereas regeneration was noticed after 4 weeks. CONCLUSIONS: MMD was an effective technique in treating patients with CLP, leading to new bone and soft tissue formation without significant detrimental effect on the periodontium of the adjacent teeth.


Cleft Lip , Cleft Palate , Osteogenesis, Distraction , Humans , Cephalometry/methods , Cleft Lip/surgery , Cleft Lip/pathology , Cleft Palate/surgery , Cleft Palate/pathology , Inflammation Mediators , Maxilla/pathology , Osteogenesis, Distraction/adverse effects , Osteotomy, Le Fort/methods , Treatment Outcome , Tumor Necrosis Factor-alpha
15.
J Tissue Eng Regen Med ; 16(8): 707-717, 2022 08.
Article En | MEDLINE | ID: mdl-35524474

Muscular dysplasia is the key factor in influencing surgical outcomes in patients with cleft lip/palate. In this research, we attempted to evaluate a new acellular dermal matrix (ADM) as a substitute for reconstruction of the orbicularis oris muscle with growth factors such as Insulin-Like Growth Factor I (IGF-I), vascular endothelial growth factor (VEGF) in a rabbit model. 30 male New Zealand Rabbits (2-3 m, 1700-2000 g) were divided into four groups as follows; a group in which the orbicularis oris muscle of the upper lip was implanted with ADM, a group in which the orbicularis oris muscle of the upper lip was implanted with ADM + IGF-I + VEGF, a group in which the upper lip was operated without implantation of an ADM scaffold, and a normal upper lip for comparison. Macroscopic observation, histological evaluation, and immunohistochemistry were employed to evaluate levels of the muscle regeneration, vascularization, and inflammation at 1, 2, 4, 6, and 12 weeks after the operation. All wounds healed well without infection, immune rejection and so on. Histological evaluation showed that ADM was totally degraded and replaced by connective tissue. The area in which the ADM scaffold was coated with growth factors show a significant increase in the formation of new myofibers after injury, and the vascularization improved compared to the control group and the normal group. In regard to the degrees of inflammation, there were no notable differences among the groups. In conclusion, Our study indicated that ADM grafts combined with IGF-I and VEGF have potential advantages in alleviating muscular dysplasia in cleft lip treatment.


Acellular Dermis , Cleft Lip , Cleft Palate , Animals , Cleft Lip/pathology , Cleft Lip/surgery , Cleft Palate/pathology , Facial Muscles/pathology , Facial Muscles/surgery , Inflammation/pathology , Insulin-Like Growth Factor I/pharmacology , Male , Rabbits , Vascular Endothelial Growth Factor A
16.
Development ; 149(9)2022 05 01.
Article En | MEDLINE | ID: mdl-35420127

The etiology of cleft lip with or without cleft palate (CL/P), a common congenital birth defect, is complex, with genetic and epigenetic, as well as environmental, contributing factors. Recent studies suggest that fetal development is affected by maternal conditions through microRNAs (miRNAs), a group of short noncoding RNAs. Here, we show that miR-129-5p and miR-340-5p suppress cell proliferation in both primary mouse embryonic palatal mesenchymal cells and O9-1 cells, a neural crest cell line, through the regulation of Sox5 and Trp53 by miR-129-5p, and the regulation of Chd7, Fign and Tgfbr1 by miR-340-5p. Notably, miR-340-5p, but not miR-129-5p, was upregulated following all-trans retinoic acid (atRA; tretinoin) administration, and a miR-340-5p inhibitor rescued the cleft palate (CP) phenotype in 47% of atRA-induced CP mice. We have previously reported that a miR-124-3p inhibitor can also partially rescue the CP phenotype in atRA-induced CP mouse model. In this study, we found that a cocktail of miR-124-3p and miR-340-5p inhibitors rescued atRA-induced CP with almost complete penetrance. Taken together, our results suggest that normalization of pathological miRNA expression can be a preventive intervention for CP.


Cleft Lip , Cleft Palate , MicroRNAs , Animals , Cell Proliferation/genetics , Cleft Lip/chemically induced , Cleft Lip/genetics , Cleft Lip/pathology , Cleft Palate/chemically induced , Cleft Palate/genetics , Cleft Palate/pathology , Mice , MicroRNAs/metabolism , Tretinoin/pharmacology
17.
J Craniofac Surg ; 33(3): 827-829, 2022 May 01.
Article En | MEDLINE | ID: mdl-35034086

ABSTRACT: Maxillary hypoplasia is common in patients with cleft lip and palate (CL/P), and its etiology is incompletely understood. The purpose of this study is to evaluate facial suture patency in patients with CL/P and maxillary hypoplasia. The authors hypothesize that patients with CL/P will demonstrate higher rates of premature midfacial suture fusion in comparison to unaffected controls. Skeletally mature patients with CL/P and midface hypoplasia were identified, along with a cohort of unaffected age- and sex-matched controls. High-resolution facial computed tomography scans were evaluated for the presence of facial suture fusion. Utilizing a previously published suture fusion grading scale, the facial sutures were classified as open, partially open, closed, or pathologically absent. Thirty-one CL/P patients with midface hypoplasia were identified, with age and sex-matched controls. The frequency of intermaxillary suture fusion did not differ between patients with CL/P and unaffected controls (P  > 0.05.) Pathologic absence of the midpalatal suture was more commonly present in patients with CL/ P and midface hypoplasia in comparison to unaffected controls (P < 0.05.) The role of midfacial sutures in the development of midfacial hypoplasia seen in CLP has not previously been studied or described. Our data show that the midpalatal suture is frequently pathologically absent in patients with CL/P and maxillary hypoplasia. The authors did not identify statistically significant differences in other midfacial sutures between patients with CL/P and controls, leading us to conclude that midfacial sutures may not play a key role in the development of midfacial hypoplasia.


Cleft Lip , Cleft Palate , Micrognathism , Child , Cleft Lip/diagnostic imaging , Cleft Lip/pathology , Cleft Lip/surgery , Cleft Palate/diagnostic imaging , Cleft Palate/pathology , Cleft Palate/surgery , Face/pathology , Humans , Maxilla/surgery , Sutures
18.
Plast Reconstr Surg ; 149(2): 248e-253e, 2022 Feb 01.
Article En | MEDLINE | ID: mdl-35077422

BACKGROUND: Left clefts occur twice as frequently as right ones. The sidedness has been suggested to influence certain outcomes. Some surgeons consider a right cleft more challenging to repair. This is often attributed to their reduced prevalence. The authors question whether this may be caused by morphologic differences. The authors' hypothesis is that there are anthropometric differences between left and right complete cleft lips. METHODS: Patients with complete unilateral cleft lip, with or without cleft palate, operated on at the age of 3 to 6 months, between 2000 and 2018, by a single surgeon, were included. Eight standardized anthropometric measurements of the cleft lip, collected just before cleft lip repair, compare lip and vermillion dimensions and ratios between left and right clefts. RESULTS: One hundred thirty-nine left and 80 right unilateral cleft lips were compared. A significant difference was found between left and right clefts for cleft-side to non-cleft-side ratios comparing the lateral lip element vertical heights and vermillion heights. CONCLUSIONS: Patients with right cleft lips have a greater degree of lateral lip element hypoplasia, demonstrating greater deficiencies of lateral lip element vertical height and vermillion height when compared to patients with left clefts. This has clinical implications for preoperative assessment, choice of surgical technique, and postoperative and long-term outcomes.


Cleft Lip/pathology , Cleft Palate/pathology , Body Weights and Measures , Cleft Lip/surgery , Cleft Palate/surgery , Female , Humans , Infant , Male , Preoperative Period , Retrospective Studies
19.
Pesqui. bras. odontopediatria clín. integr ; 22: e210085, 2022. tab, graf
Article En | LILACS, BBO | ID: biblio-1386809

Abstract Objective: To assess the effect of the three types of unilateral cleft lip (UCL) [cleft lip only, cleft lip and alveolus, and cleft lip alveolus and palate] on the outcome of the repair. Material and Methods: This study was a case series of effect of types of UCL on the outcome of the repair. Fifteen subjects each were in three UCL phenotypes groups. Evaluation of the scar, lip, and nose was done qualitatively by both parents/guardians and professionals using a modified form of the criteria described by Christofides et al. (2006). Results: In the assessment of the surgical scar, the parents found a difference between the three types of cleft in terms of texture, shape, and width of the scar and presence of columella deviation. The professional assessors, however, only found the three types of cleft to be different in the presence of alar flattening. Conclusion: Differences truly exist in the outcome of surgical repair of the three types of unilateral cleft lip, especially in the aesthetics of the nose and in the width and shape of the residual lip scar. Thus, it is important to consider this in the assessment of UCL repair because putting the subtypes together might have a negative impact on the assessment.


Humans , Male , Female , Infant, Newborn , Infant , Surgical Procedures, Operative , Congenital Abnormalities/pathology , Cleft Lip/surgery , Cleft Palate/surgery , Chi-Square Distribution , Prospective Studies , Cleft Lip/pathology
20.
Sci Rep ; 11(1): 23609, 2021 12 08.
Article En | MEDLINE | ID: mdl-34880325

There are very few nationwide studies discussing the height, weight, head circumference, and dental screening of children with cleft lip with or without palate (CL/P), with most reports on this subject based on a 1900s cohort. This study aimed to characterize CL/P children in the 2000s in terms of height, weight, head circumference, and dental screening. This nationwide population-based study evaluated the National Health Insurance Service-Infants and Children's Health Screening (NHIS-INCHS), specifically the height, weight, and head circumference of millions of children. Dental screening data, including the status of each tooth and comprehensive dental judgment, were also evaluated. Syndromic and nonsyndromic CL/P children had lower height, weight, and head circumference than no CL/P children until the age of 66-71 months. Children with cleft palate only or both cleft lip and palate showed similar results. Regarding dental screening, the primary teeth of CL/P children erupted later and fell out faster than no CL/P children. Dental caries was also more common in CL/P children. Children with CL/P had inferior general growth, regardless of palatoplasty surgery. More aggressive dental treatment was required for CL/P children due to the instability of primary teeth and tendency for caries.


Cleft Lip/pathology , Cleft Palate/pathology , Growth , Anthropometry , Case-Control Studies , Child , Child, Preschool , Cohort Studies , Female , Humans , Male , Republic of Korea
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