Midline Cervical Cleft: Case Report and Current Understanding.

Congenital midline cervical cleft (CMCC) is an extraordinarily rare midline lesion that is present at birth. The lesion can cause micrognathia and cervical contracture via underlying traction on the mandible. It is essential to recognize the condition and excise the lesion within 1 year of diagnosis to mitigate long term cosmetic and functional problems. We report a 10-month-old male infant with CMCC diagnosed immediately after birth. Ultrasound with color doppler imaging confirmed the anatomic characteristics of the lesion. The lesion was surgically excised and closed in layers, with no postoperative complications and good functional and cosmetic results. The pathognomonic presentation, etiology, differential diagnosis, and optimal surgical approach are also discussed.

Facial growth parameters in Down syndrome: Review of the literature and forensic application for missing persons age progression.

PURPOSE: Individuals with Down syndrome (DS) show growth trajectories which deviate from standard ones due to variations in the growth of facial structures. Studying the effect of aging on the faces of DS individuals is necessary to obtain an accurate result through age progression, a technique based on the study of physiognomic features and used in cases of missing persons. Here we present scientific publications that delve into the rhythms of aging and morphological characteristics of facial features in DS individuals to enable appropriate age progression in cases of missing DS individuals. RESULTS: The scientific literature considered in this review studies the growth of soft tissue and bone substrate by comparing standard growth values with those measured through anthropometric measurements of individuals with DS. Growth trajectories are described by considering morphological trends both by comparing standard values with those found in individuals with DS and by observing individual physiognomic traits. CONCLUSIONS: When a young individual with DS goes missing, the realization of an age progression requires knowledge of the aging dynamics peculiar to the DS face. Therefore, physical, cognitive, and clinical factors must be considered. Delayed physical development and early aging, such as the onset of puberty and weight gain, have an important impact on the realization of age progression. In fact, depending on the life period to be considered, the effects of aging must be calibrated based on the knowledge gathered from scientific research.

A Pilot Study on the Relationship between Obstructive Sleep Apnoea-Hypopnea Syndrome, Neurodevelopment, and Ricketts' Cephalometry.

Background: The aim of this research is to achieve the early detection of facial characteristics in patients diagnosed with neurodevelopmental deficits and obstructive sleep apnoea-hypopnea syndrome (OSAHS) through the analysis of the VERT index and Ricketts' cephalometry to minimise the neurocognitive morbidity associated with these disorders. Methods: This clinical study was conducted on 44 patients aged 4 to 15 years. Participants completed an initial questionnaire about sleep disturbances, followed by a polysomnography, a radiographic study, and an oral examination. Results: The maximum variability in the data was obtained in the mandibular plane angle, where we observed that the measurement was higher in patients diagnosed with OSAHS. The relative and normalised indices of facial depth and the mandibular plane showed variations between patients with a clinical picture compatible with OSAHS and the control group without pathology. Conclusions: Our findings indicate that Ricketts' VERT index by itself is unable to provide evidence of a diagnosis compatible with OSAHS in patients with early neurodevelopmental deficits, but, after analysing the results obtained, we observed that for the cephalometric measurements of the mandibular plane angle and facial depth relative to the patient's age, there are sufficiently strong variations for creating a solid method of differential diagnosis, thus preventing complications at the neurocognitive level.

Sella turcica and facial bones: Morphological integration in the human fetal cranium.

OBJECTIVES: The cranial base plays a significant role in facial growth, and closer analyses of the morphological relationship between these two regions are needed to understand the morphogenesis of the face. Here, we aimed to study morphological integration between the sella turcica (ST) and facial bones during the fetal period using geometric morphometrics. MATERIALS AND METHODS: Magnetic resonance images of 47 human fetuses in the Kyoto Collection, with crown-rump lengths of 29.8-225 mm, were included in this study. Anatomical homologous landmarks and semilandmarks were registered on the facial bones and the midsagittal contour of the ST, respectively. The shape variations in the craniofacial skeleton and the ST were statistically investigated by reducing dimensionality using principal component analysis (PCA). Subsequently, the morphological integration between the facial bones and ST was investigated using two-block partial least squares (2B-PLS) analysis. RESULTS: PCA showed that small specimens represented the concave facial profile, including the mandibular protrusion and maxillary retrusion. The 2B-PLS showed a strong integration (RV coefficient = 0.523, r = .79, p < .01) between the facial bones and ST. The curvature of the anterior wall of the ST was highly associated with immature facial morphology characterized by a concave profile. CONCLUSION: The strong integration between the two regions suggested that the anterior ST may be associated with facial morphology. This result quantitatively confirms previous studies reporting ST deformities in facial anomalies and induces further research using postnatal subjects.

Craniocervical posture in patients with skeletal malocclusion and its correlation with craniofacial morphology during different growth periods.

The association between craniocervical posture and craniofacial structures in the various sagittal skeletal malocclusion during different growth stages has been the focus of intense interest in fields of orthodontics, but it has not been conclusively demonstrated. Thus, this study aimed to investigate the association between craniofacial morphology and craniocervical posture in patients with sagittal skeletal malocclusion during different growth periods. A total of 150 from a large pool of cephalograms qualified for the inclusion and exclusion were evaluated and classified into three groups according to the Cervical Vertebral Maturation (CVM) by examining the morphological modifications of the second through fourth cervical vertebrae, each group consisted of 50 cephalograms. In each growth period, for the comparison of head and cervical posture differences among various skeletal classes, the radiographs were further subdivided into skeletal Class I (0° < ANB < 5°, n = 16), skeletal Class II (ANB ≥ 5°, n = 18), and skeletal Class III (0° ≤ ANB, n = 16) on the basis of their ANB angle. There was no significant difference in gender (P > 0.05). Some variables were found to be significant during pubertal growth and later in patients with sagittal skeletal malocclusion (P < 0.05). Most indicators describing craniocervical posture were largest in skeletal Class II and smallest in skeletal Class III during the peak growth periods and later. Cervical inclination variables were greater in skeletal Class III than in skeletal Class II. Variables of craniofacial morphology and craniocervical posture are more correlated during the pubertal growth period and later in patients with sagittal skeletal malocclusion. A tendency is an indication of the close interrelationship that a more extended head was in skeletal Class II while a flexed head was in skeletal Class III. Nevertheless, with the considerations of some limitations involved in this study, further longitudinal studies with large samples are required to elucidate the relationship clearly.

The Influence of Sidedness in Unilateral Cleft Lip and Palate on Mid Facial Growth at Five Years of Age.

OBJECTIVE: To determine whether facial growth at five years is different for children with a left versus right sided cleft lip and palate. DESIGN: Retrospective cohort study. SETTING: Seven UK regional cleft centres. PATIENTS: Patients born between 2000-2014 with a complete unilateral cleft lip and palate (UCLP). MAIN OUTCOMES MEASURE: 5-Year-Old's Index scores. RESULTS: 378 children were included. 256 (68%) had a left sided UCLP and 122 (32%) had a right sided UCLP. 5-Year-Old's index scores ranged from 1 (good) to 5 (poor). There was a higher proportion of patients getting good scores (1 and 2) in left UCLP (43%) compared to right UCLP (37%) but there was weak evidence for a difference (Adjusted summary odds ratio 1.27, 95% CI 0.87 to 1.87; P = .22). CONCLUSIONS: Whilst maxillary growth may be different for left versus right sided UCLP, definitive analysis requires older growth indices and arch forms.

Variability associated with maxillary infrazygomatic crest and palatal bone width, height, and angulation in subjects with different vertical facial growth types: a retrospective cone-beam computed tomography study.

OBJECTIVES: To assess the infrazygomatic crest (IZC) and palatal bone width, height, and angulation in patients with different vertical facial growth types as potential miniscrew insertion sites. MATERIALS AND METHODS: In this retrospective cone-beam computed tomography study, 162 subjects (81 males and 81 females, mean age 16.05 ± 0.65 years) were included. They were divided into three groups (hypodivergent, normodivergent, and hyperdivergent) based on the Frankfort mandibular plane angle. Ten buccal bone measurements were made at two different coronal sections: maxillary first molar mesiobuccal and distobuccal roots (bilaterally). Six palatal bone measurements were made on a sagittal section at the maxillary central incisors (bilaterally). A total of 32 measurements per subject were considered in the study. RESULTS: No significant difference was observed for the IZC (width and angle) at the maxillary first molar mesiobuccal root. A comparison of normodivergent and hyperdivergent groups for buccal width at the distobuccal root of the first molar showed significant differences. Palatal bone thickness at the level of 2 mm distal to the apex of the central incisor was significantly higher for the hyperdivergent group (10.43 mm) compared with the normodivergent (7.58 mm) and hypodivergent groups (7.83 mm). CONCLUSIONS: Hyperdivergent subjects tend to present a longer and deeper IZC and increased palatal bone thickness compared with other groups. The recommended insertion angle for the IZC mini-implant at 3 mm from the alveolar crest should be between 75.5° and 77°.

Comparison of mesiodistal angulations of premolars and molars in anterior open bite subjects with different sagittal malocclusions: A retrospective study.

INTRODUCTION: Mesiodistal angulation of premolars and molars can be altered by forces of open bite malocclusion. The aim of this study was to compare the mesiodistal angulations of the posterior teeth in class I, II, and III individuals with anterior open bite (AOB) versus individuals with harmonious occlusion. METHODS: This comparative cross-sectional study used 299 lateral head radiographs of individuals with permanent dentition. There were 4 groups (harmonious occlusion [n=89], Class I open bite [OB] [n=75], Class II OB [n=66], and Class III OB [n=69]). Premolar (1UPM, 2UPM) and molar (1UM, 2UM) angulations were measured relative to the occlusal plane and the palatal or mandibular plane by a trained and calibrated evaluator. ANOVA and Scheffe tests were used for statistical analyses (P<0.05). RESULTS: The mesial angulation of the upper premolars showed greater angulation of between approximately 2° and 5° in the OB groups compared to the harmonious occlusion group (P<0.05). Only in the Class II OB group did the first and second upper molars show distal angulation in relation to the palatal plane (1UM 81.85°±5.42°; 2UM 75.32±7.4°) (P<0.05). The Class III OB group presented the greatest distal angulations of the lower premolars and molars (between 3° to 5° of difference, P<0.05) in relation to those of the harmonious occlusion group. CONCLUSIONS: The upper first premolars in all the AOB groups and the lower second premolars in the Class II OB group had greater mesioangulation. Additionally, the upper molars of the Class II OB group and the lower molars of the Class III OB group showed distoangulation compared with the molars in the group with harmonious occlusion.

Buccal Fat Pad in Primary and Secondary Cleft Palate Repair: A Systematic Review of the Literature.

OBJECTIVE: To review the evidence supporting the use of buccal fat pad (BFP) in primary and secondary cleft palate repair and its short- and long- term clinical outcomes. DESIGN: Systematic review conducted by 2 independent reviewers following PRISMA guidelines. SETTING: NONE PARTICIPANTS: Articles were identified from three databases (Pubmed/Medline, Embase and Web of Science). Search terms included "cleft palate", "palatoplasty", "palate repair", "buccal fat pad". INTERVENTIONS: Use of BFP in primary and secondary cleft palatoplasty. MAIN OUTCOME MEASURES: Primary outcomes were immediate postoperative complications, postoperative fistula, and maxillary growth. Secondary outcomes were palatal length, speech, and donor site morbidity. RESULTS: Ninety-one reports were retrieved after excluding duplicates. Twenty-three studies were included (13 case series and 10 comparative studies). Overall level of evidence was low. Randomized and non-randomized studies had a high risk of bias. In primary palatoplasty, BFP was more frequently used filling lateral relaxing incisions(57.4%), or in the hard-soft palate junction and covering mucosal defects(30.1%). In these patients, post operative fistula incidence was 2.8%. Two studies found wider transverse maxillary dimensions after BFP use. No higher incidence of bleeding, infection, dehiscence, or flap necrosis was reported. In secondary palatoplasty, no recurrent fistulas were reported for patients undergoing BFP for fistula repair. CONCLUSIONS: BFP appears to be associated with a favorable impact in fistula prevention and management, as well as in transverse maxillary growth. However, there is a high heterogeneity among studies, high risk of bias and overall low quality of evidence. More high-quality research with long-term follow-up is warranted.

Longitudinal Three-Dimensional Stereophotogrammetric Growth Analysis in Infants with Unilateral Cleft Lip and Palate from 3 to 12 Months of Age.

This longitudinal study aimed to evaluate facial growth and soft tissue changes in infants with complete unilateral cleft lip, alveolus, and palate (CUCLAP) at ages 3, 9, and 12 months. Using 3D images of 22 CUCLAP infants, average faces and distance maps for the entire face and specific regions were created. Color-coded maps highlighted more significant soft tissue changes from 3 to 9 months than from 9 to 12 months. The first interval showed substantial growth in the entire face, particularly in the forehead, eyes, lower lip, chin, and cheeks (p < 0.001), while the second interval exhibited no significant growth. This study provides insights into facial soft tissue growth in CUCLAP infants during critical developmental stages, emphasizing substantial improvements between 3 and 9 months, mainly in the chin, lower lip, and forehead. However, uneven growth occurred in the upper lip, philtrum, and nostrils throughout both intervals, with an overall decline in growth from 9 to 12 months. These findings underscore the dynamic nature of soft tissue growth in CUCLAP patients, highlighting the need to consider these patterns in treatment planning. Future research should explore the underlying factors and develop customized treatment interventions for enhanced facial aesthetics and function in this population.

"Strategic Application of Anatomical Subunit Approximation Technique for Correction of Complete Unilateral Cleft lip".

OBJECTIVE: To discuss advantages, disadvantages and strategical application of anatomical subunit approximation technique in complete unilateral cleft lip repair. DESIGN: Analysis of consecutive 28 cases. SETTING: A single surgeon experience at a university hospital. PATIENTS: Among 77 patients who underwent cleft lip repair between May 5, 2019 and June 30, 2021, 28 patients with complete unilateral cleft lip who received cheiloplasty by author's technique. MAIN OUTCOME MEASURES: Surgical outcomes were assessed by frontal view medical photographs obtained postoperatively at follow-up visits. RESULTS: The average length difference between cleft and noncleft sides was <10% for transverse lip length and philtral height. Nostril sill height demonstrated about twenty percent of difference with much variable results. Although the one sample t-test revealed that differences exist between the cleft and noncleft side, they were within an acceptable range. CONCLUSIONS: Understanding the strengths and limitations of anatomical subunit approximation technique enabled strategic application in complete unilateral cleft lip repair. Satisfactory outcomes were achieved not only regarding the scar placement but also regarding the symmetry of the lips and augmentation of nasal sill.

Evaluating diagnostic performance of three cephalometric vertical parameters.

Objectives: The goal of this research was to see how reliable and valid three selected parameters from different analysis were in detecting the vertical skeletal pattern. Methods: There were 94 cephalometric x-rays used in all. The vertical skeletal pattern was assessed using the mandibular plane angle by Steiner, Frankfort mandibular angle by Tweed, and facial axis angle by McNamara. According to the diagnostic results of the majority of the measures, the samples were classified as normo-divergent, hypodivergent, or hyperdivergent. Kappa statistics, positive predictive value, and sensitivity were employed to verify the validity and reliability of the analyses. Results: A statistically significant weak negative correlation was found between Frankfort mandibular angle and facial axis angle in female group (p value < 0.01). A good agreement was found between mandibular plane angle with final diagnosis (K = 0.726). The mandibular plane angle showed highest sensitivity and positive predictive value in hypodivergent group (0.939, 0.816) and normo-divergent group (0.795, 0.833), respectively. Conclusion: For determining the facial vertical growth pattern the most accurate indicators were identified to be mandibular plane angle (SN-GoGn) and Frankfort mandibular angle.

Comparison between Two Methods of Skeletal Growth Evaluation: Cervical Vertebrae Maturations and Middle Phalanx Maturation.

Aim: Growth measurement has always been essential to identify the best time to employ orthopedic or orthodontic appliances. Optimal timing for orthodontic treatment is strictly linked to the identification of periods of craniofacial growth when treatment is more effective.The aim of this study was to compare two different methods, middle phalanx maturation (MPM) and cervical vertebrae maturation (CVM), used to evaluate the stage of facial growth. Materials and methods: The research data was collected from July 2018 to April 2019 at the Dental Clinic of the San Gerardo Hospital in Monza. The study included a sample of 98 patients-46 males and 52 females. For each patient, a latero-lateral teleradiography of the skull and an X-ray on the middle finger of the right hand were obtained.The statistical analysis of the comparison of the stages of skeletal maturation obtained by the MPM and CVM methods was performed using the correlation coefficient for ranks of Spearman. Results: A descriptive statistical analysis of the entire sample of 98 patients was performed (mean age of 12.2 years and median of 12.2 years). The average age of females in every single stage of MPM was significantly lower than the average age of males. Of the total sample, 87 patients (88.8%) showed complete agreement between the two methods. Conclusion: The results obtained from the statistical analysis of this study allowed us to confirm a satisfactory agreement between the two methods.The intermediate phalanx method is a valid and alternative indicator to CVM for the identification of the puberty growth peak. We can, therefore, consider the MPM method a valid indicator of skeletal maturity. How to cite this article: Mirabelli L, Bianco E, Pigato G, et al. Comparison between Two Methods of Skeletal Growth Evaluation: Cervical Vertebrae Maturations and Middle Phalanx Maturation. Int J Clin Pediatr Dent 2023;16(2):327-332.

A Unique Case Report of Meningeal Hamartoma Within Alveolar Cleft.

This case report demonstrates a rare finding of a pediatric patient with Morning Glory anomaly and Moyamoya Disease with a palatal meningeal hamartoma discovered as a mass within a previously repaired incomplete cleft of the alveolus. Oral meningeal hamartomas are exceedingly rare with only two palatal cases described and none within a cleft palate or alveolus. These findings prompt a review of oral hamartomas with meningeal subclassification. Further discussion describes the relationship of the proposed origins of meningeal hamartomas within the setting of cleft palate development.

Do nasal and nasopharyngeal obstruction and their medical-surgical deobstruction significantly influence facial divergence? A concise review of major clinical studies with meta-analysis / L'obstruction du nez et du nasopharynx et leur désobstruction médico-chirurgicale influencent-elles significativement la divergence faciale ? Une revue concise des études cliniques majeures avec méta-analyse

Introduction: The term « adenoid facies ¼ suggests a causal relationship between nasopharyngeal obstruction and facial hyperdivergence in growing subjects. The strength of this association is controversial and few « quantified ¼ values exist. Materials and methods: A rapid electronic search was conducted on PubMed and Embase to find the main cephalometric studies involving patients with nasal/nasopharyngeal obstruction compared to a control sample. A meta-analysis was carried out to quantify the effect of obstruction (1) and intervention to relieve the obstruction (2) on mandibular divergence (SN/Pmand angle), maxillo-mandibular divergence (PP/Pmand angle), inclination of the occlusal plane (SN/Poccl) and the gonial angle (ArGoMe). Results: Qualitatively, the studies' bias level ranged from moderate to high. Results were concordant about the significant effect of the obstruction on facial divergence (1) with an increase in SN/Pmand (+3.6° on average, +4.1° in children <6 years), PP/Pmand (+5.4° on average, +7.7° <6 years), ArGoMe (+3.3°) and SN/Pocc (+1.9°). Surgical interventions to remove the respiratory obstacle in children (2) generally did not normalize the direction of growth, with the exception, with a very low level of evidence, of adenoidectomies/adeno-tonsillectomies, performed at an age less than 6-8 years. Conclusion: Early detection of respiratory obstacles and postural abnormalities associated with oral breathing appears to be decisive in order to hope for management at a young age and normalization of the direction of growth. However, the effects on mandibular divergence remain limited, requiring caution, and cannot be considered a surgical indication.

Introduction: Le terme « faciès adénoïdien ¼ suggère une relation de causalité entre l'obstruction nasopharyngée et l'hyperdivergence chez le sujet en croissance. La force de cette association est controversée et peu de valeurs « chiffrées ¼ existent. Matériels et méthodes: Une recherche électronique rapide a été menée sur PubMed et Embase pour retrouver les principales études céphalométriques impliquant des patients avec obstruction nasale/nasopharyngée comparés à une population témoin. Une métanalyse a été réalisée pour quantifier l'effet de l'obstruction (1) et de la désobstruction (2) sur la divergence mandibulaire (angle SN/Pmand), la divergence maxillo-mandibulaire (angle PP/Pmand), l'inclinaison du plan occlusal (SN/Poccl) et l'angle goniaque (ArGoMe). Résultats: Qualitativement, le niveau de biais des études allait de modéré à élevé. Les résultats étaient concordants sur l'effet significatif de l'obstruction sur la divergence faciale (1) avec une augmentation de SN/Pmand (+3,6° en moyenne, +4,1° chez les enfants < 6 ans), PP/Pmand (+5,4° en moyenne, +7,7° < 6 ans), ArGoMe (+3,3°) et SN/Pocc (+1,9°). Les interventions chirurgicales pour lever l'obstacle respiratoire chez l'enfant (2) ne permettaient généralement pas une normalisation de la direction de croissance, à l'exception, avec un très faible niveau de preuve, des adénoïdectomies/adéno-amygdalectomies, réalisées à un âge inférieur à 6-8 ans. Conclusion: Le dépistage précoce des obstacles respiratoires et des anomalies posturales associées à la ventilation orale apparaît déterminant pour espérer une prise en charge en jeune âge et une normalisation de la direction de croissance. Les effets sur la divergence mandibulaire restent cependant limités, imposant la prudence et ne constituant pas une indication chirurgicale.

Functional cleft palate surgery.

Cleft lip and palate (CLP) as a dislocation malformation confronts parents with a malformation of their child that could not be more central and visible: the face. In addition to the stigmatizing appearance, however, in cases of a CLP, food intake, physiological breathing, speech and hearing are also affected. In this paper, the principles of morphofunctional surgical reconstruction of the cleft palate are presented. With the closure of the palate, and restoration of the anatomy, a situation is achieved enabling nasal respiration, normal or near normal speech without nasality, improved ventilation of the middle ear, normal oral functions with coordinated interaction of the tongue with the hard and soft palate important for the oral and pharyngeal phases of feeding. With the establishment of physiological function, in the early phases of the infant and toddler, these activities initiate essential growth stimulation, leading to normalisation of facial and cranial growth. If these functional considerations are disregarded during primary closure, lifelong impairment of one or more of the abovementioned processes often follows. In many cases, despite secondary surgery and revision, it might not be possible to correct and achieve the best possible outcomes, especially if critical stages of development and growth have been missed or there has been significant tissue loss due to resection of existing tissue while primary surgery. This paper describes functional surgical methods and reviews long term, over many decades, results of children with cleft palate.

In Utero Exposure to Maternal Electronic Nicotine Delivery System use Demonstrate Alterations to Craniofacial Development.

OBJECTIVE: Develop a model for the study of Electronic Nicotine Device (ENDS) exposure on craniofacial development. DESIGN: Experimental preclinical design followed as pregnant murine dams were randomized and exposed to filtered air exposure, carrier exposure consisting of 50% volume of propylene glycol and vegetable glycine (ENDS Carrier) respectively, or carrier exposure with 20 mg/ml of nicotine added to the liquid vaporizer (ENDS carrier with nicotine). SETTING: Preclinical murine model exposure using the SciReq exposure system. PARTICIPANTS: C57BL6 adult 8 week old female pregnant mice and exposed in utero litters. INTERVENTIONS: Exposure to control filtered air, ENDS carrier or ENDS carrier with nicotine added throughout gestation at 1 puff/minute, 4 h/day, five days a week. MAIN OUTCOME MEASURES: Cephalometric measures of post-natal day 15 pups born as exposed litters. RESULTS: Data suggests alterations to several facial morphology parameters in the developing offspring, suggesting electronic nicotine device systems may alter facial growth if used during pregnancy. CONCLUSIONS: Future research should concentrate on varied formulations and exposure regimens of ENDS to determine timing windows of exposures and ENDS formulations that may be harmful to craniofacial development.

Denosumab Therapy in Cherubism.

Cherubism is a rare disorder characterized by proliferative fibro-osseous lesions that result in bilateral bony hyperplasia of the face. Management varies based on symptom severity and includes longitudinal follow-up, pharmacotherapy, and/or surgical debulking. Off-label treatment with denosumab, a human monoclonal antibody that binds RANKL and inhibits osteoclast function to reduce bone resorption, can be beneficial in suppressing the proliferation of bone to minimize the need for surgery and to control postoperative reproliferation. Close follow-up is needed to maintain appropriate electrolyte levels. The present case demonstrates the achievement of symptomatic control with denosumab in a child with severe refractory cherubism.