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The effect of preoperative use of an orthopedic plate on articulatory function in children with cleft lip and palate.

OBJECTIVE: To evaluate the effect of preoperative use of an orthopedic plate (OP) on postoperative articulatory function in children with cleft lip and palate. SUBJECTS: The subjects had complete unilateral or bilateral cleft lip and palate and were scheduled for a one-stage palatoplasty. MAIN OUTCOME MEASURES: Tongue movements during sucking were analyzed by ultrasonography. Postoperative articulatory behavior was also assessed at 5 years 4 months of age. RESULTS: There was an excessive downward excursion of the rear portion of the tongue during sucking regardless of the use or nonuse of the OP. This indicated that infants with cleft palate could not create negative pressure in the oral cavity, even with the OP. However, the OP appeared effective for preventing irregular movements of the tongue during sucking. The proportion of subjects obtaining excellent articulation was significantly higher in the group using the OP until palatoplasty than in the group who did not continue using the OP. The proportion of subjects with disturbed articulatory function in the latter group was comparable with that in the control group, who never used the OP. CONCLUSIONS: Continuous use of the OP up to the time of palatoplasty appeared to be effective for the postoperative articulatory function in children with complete cleft lip and palate. Inhibiting irregular movements of the tongue, the OP might assist in preventing "palatalized articulation."

Articulation Disorders↗

Changes of vertical skeletal morphology in patients with complete unilateral and bilateral cleft lip and palate.

OBJECTIVE: The aim of this retrospective investigation was to qualify and quantify changes of the vertical skeletal morphology during puberty in cleft patients compared with an age-matched noncleft control. METHODS: Patients with unilateral cleft lip and palate (UCLP; n = 12) and bilateral cleft lip and palate (BCLP; n = 11) who fulfilled strict inclusion criteria according to the Hanover treatment protocol were evaluated by means of cephalometric analysis. Lateral cephalograms were analyzed for each cleft patient at age 10 and 15 and compared with corresponding data of a noncleft control (n = 20). RESULTS AND CONCLUSIONS: It was concluded that a vertical growth impairment of the maxilla is found in patients with cleft lip and palate. The maxilla of cleft patients shows a significant clockwise rotation, whereas the inclination of the mandible shows little difference. Consequently, there is a skeletal superposition of the jaws. The posterior height of the maxilla is significantly shorter in patients both with UCLP and BCLP at T(0) and T(1). An increase of the anterior height of the mandible is found in the cleft groups.

Adolescent↗

Three-dimensional nasal changes following nasoalveolar molding in patients with unilateral cleft lip and palate: geometric morphometrics.

OBJECTIVE: To evaluate three-dimensional changes in nasal morphology in patients with unilateral cleft lip and palate treated with presurgical nasoalveolar molding (NAM) to correct naso-labio-alveolar deformity. DESIGN: This was a prospective, longitudinal study. Digital stereophotogrammetry was used to capture three-dimensional facial images, and x, y, and z coordinates of 28 nasal landmarks were digitized. SAMPLE: Ten patients with unilateral cleft lip and palate. MAIN OUTCOME MEASURES: Nasal form changes between T1 (age: 28 +/- 2 days, pre-NAM) and T2 (age: 140 +/- 2 days, post-NAM), using conventional measurements and finite-element scaling analysis. RESULTS: Overall nasal changes were statistically different (p < .01), but no linear or curvilinear changes were found. Specifically, relative size increases were found on the noncleft side, involving the upper nose (30%), alar depth (20%), alar dome (30%), columella height (30%), and lateral wall of the nostril (17%). On the cleft side, the following showed a size increase: upper nose (8%), alar dome (5%), columella height (30%), and lateral wall of the nostril (30%). The cleft-side alar curvature, however, showed a large decrease in size (80%), but no changes on the noncleft side were found. Corresponding shape changes and angular changes were also found. CONCLUSIONS: Using NAM, bilateral nasal symmetry in patients with unilateral cleft lip and palate was improved before surgical repair. Furthermore, slight overcorrection of the alar dome on the cleft side using pressure exerted by the nasal stent is indicated to maintain the NAM result.

Adolescent↗

Changes in nasorespiratory function in association with maxillary distraction osteogenesis in subjects with cleft lip and palate.

OBJECTIVE: The current study aimed to determine how nasorespiratory function changes in association with maxillary distraction osteogenesis (DO). Furthermore, with regard to impaired nasorespiratory function, the possibility of a relationship between the cleft side and laterality and any effect of maxillary distraction osteogenesis was investigated. DESIGN: In this descriptive, prospective clinical report, subjective and objective data regarding nasorespiratory function before and after maxillary distraction osteogenesis were compared. SUBJECTS: Data from 13 subjects with cleft lip and palate were used. Subjects had a severe maxillary deficiency and underwent distraction osteogenesis using a rigid external device system. OUTCOME MEASURES: The subjective measure was the score on a questionnaire regarding nasorespiratory function using a visual analog scale. The objective measure was nasal resistance. RESULTS: The visual analog scale score for two items significantly decreased just after distraction osteogenesis. Nasal resistance also significantly decreased 1 year after distraction osteogenesis. Moreover, nasal resistance on the cleft side was significantly greater than that on the noncleft side just before and 1 year after distraction osteogenesis. There was a significant positive correlation between changes in the visual analog scale score and nasal resistance. CONCLUSIONS: These results suggest that nasorespiratory function changes in association with maxillary distraction osteogenesis in subjects with cleft lip and palate. Moreover, it appears that nasal obstruction on the cleft side does not change in subjects with unilateral cleft lip and palate.

Adolescent↗

Extraoral nasal molding in a newborn with unilateral cleft lip and palate: a case report.

OBJECTIVE: The purpose of this case report is to introduce an extraoral nasal molding appliance (ENMA) and treatment approach for presurgical nasoalveolar molding in newborns with unilateral cleft lip and palate. METHODS: A 15-day-old girl presented with complete unilateral cleft lip and palate. A circumferential headband supported the actual nasoalveolar molding device, which consisted of a nasal stent made from a 0.8-mm stainless steel helical spring. The spring was activated at 2-week intervals. DISCUSSION: The shape of the cartilaginous septum, alar cartilage tip, medial and lateral crus and alveolar segments were molded to resemble the normal shape of these structures. ENMA can be helpful in any patient with unilateral cleft lip and palate because it is easy to fabricate, practical to activate, and comfortable to wear and use.

Alveolar Process↗

Determination of facial symmetry in unilateral cleft lip and palate patients from three-dimensional data: technical report and assessment of measurement errors.

OBJECTIVE: To assess measurement errors of a novel technique for the three-dimensional determination of the degree of facial symmetry in patients suffering from unilateral cleft lip and palate malformations. DESIGN: Technical report, reliability study. SETTING: Cleft Lip and Palate Center of the University of Erlangen-Nuremberg, Erlangen, Germany. PATIENTS: The three-dimensional facial surface data of five 10-year-old unilateral cleft lip and palate patients were subjected to the analysis. Distances, angles, surface areas, and volumes were assessed twice. MAIN OUTCOME MEASURES: Calculations were made for method error, intraclass correlation coefficient, and repeatability of the measurements of distances, angles, surface areas, and volumes. RESULTS: The method errors were less than 1 mm for distances and less than 1.5 degrees for angles. The intraclass correlation coefficients showed values greater than .90 for all parameters. The repeatability values were comparable for cleft and noncleft sides. CONCLUSION: The small method errors, high intraclass correlation coefficients, and comparable repeatability values for cleft and noncleft sides reveal that the new technique is appropriate for clinical use.

Anthropometry↗

Dynamic presurgical nasal remodeling in patients with unilateral and bilateral cleft lip and palate: modification to the original technique.

OBJECTIVE: To present technical modifications to the original presurgical nasal remodeling appliance introduced in 1991. The purpose of the modifications is to improve the cleft nasal deformity before unilateral and bilateral cleft lip repair. METHOD: The principle behind this technique, known as dynamic presurgical nasal remodeling (DPNR), is the use of the force generated during suction and swallowing. A conventional intraoral plate is built with a nasal extension added to the labial vestibular flange. The nasal extension was modified and consists of three components. The palatal plate is left loose in the mouth to generate a discontinuous but controlled impact directed to the affected nasal structures during suction and swallowing. The principle aim of the DPNR technique in unilateral cases is to improve the deformation of nasal structures by straightening the columella, elevating the nasal tip, and remodeling the depressed cleft side alar cartilages. In bilateral cases, the aims are to elongate the columella and to obtain nasal tip projection. CONCLUSIONS: The modifications introduced in the appliance enhance the original DPNR technique and are effective in ameliorating the initial cleft nasal deformity. This facilitates primary surgical cleft lip and nose correction and improves surgical outcomes in patients with complete unilateral and bilateral cleft lip and palate.

Biomechanical Phenomena↗

Hard palate repair timing and facial growth in cleft lip and palate: a systematic review.

OBJECTIVE: To evaluate the effect of timing of hard palate repair on facial growth in patients with cleft lip and palate, with special reference to cranial base, maxilla, mandible, jaw relation, and incisor relation. DESIGN: A systematic review. METHODS: The search strategy was based on the key words "facial growth," "cleft lip palate," and "timing of (hard) palate repair." Case reports, case-series, and studies with no control or comparison group in the sample were excluded. RESULTS: Fifteen studies met the selection criteria. All the studies were retrospective and nonrandomized. Five studies used cephalometry and casts, seven used cephalometry, and three used casts. Methodological deficiencies and heterogeneity of the studies prevented major conclusions. CONCLUSION: The review highlights the importance of further research. Prospective well-designed, controlled studies, especially targeting long-term results, are required to elucidate the effect of timing of hard palate repair on facial growth in patients with cleft lip and palate.

Cleft Lip↗

Reliability of linear measurements on a virtual bilateral cleft lip and palate model.

OBJECTIVE: To assess the reliability and validity of measurements performed on three-dimensional virtual models of neonatal bilateral cleft lip and palate patients, compared with measurements performed on plaster cast models. MATERIALS AND METHODS: Ten high-quality plaster cast models of bilateral cleft lip and palate patients were scanned with an LDI-scanner to obtain a three-dimensional virtual model. Linear measurements were performed on the plaster cast models using a digital caliper and also on the three-dimensional virtual model using Viscam RP version 2.1 software. The measurements were performed by two observers on two occasions. RESULTS: Intraclass correlations ranging from .81 to .96 were found for all measurements except the measurement between the constructed reference point pr and reference point i (intraclass correlation = .40). A post hoc procedure in which top-view screen prints of the three-dimensional virtual model were used to perform the measurement between reference points pr and i demonstrated an intraclass coefficient of .90. CONCLUSIONS: Three-dimensional virtual models obtained by laser scanning neonatal cast models of bilateral cleft lip and palate patients can be used reliably and validly to perform linear measurements between existing reference points on the surface of the model using Viscam RP version 2.1 software. Measurements between reference points constructed outside the surface of the model cannot be validly performed on the three-dimensional virtual model with the software used in this study. For these measurements, top-view screen prints of the three-dimensional virtual model can be used.

Cephalometry↗

Hard palate repair timing and facial growth in unilateral cleft lip and palate: a longitudinal study.

OBJECTIVE: To investigate whether timing of hard palate repair had a significant effect on facial growth in patients with unilateral cleft lip and palate (UCLP). DESIGN: Retrospective longitudinal study. SETTING: Sri Lankan Cleft Lip and Palate Project. PATIENTS: A total of 104 patients with nonsyndromic UCLP who had hard palate repair by age 13 years, with their 290 cephalometric radiographs taken after lip and palate repair. MAIN OUTCOME MEASURES: Clinical notes were used to record surgical treatment histories. Cephalometry was used to determine facial morphology and growth rate. RESULTS: Timing of hard palate repair had a significant effect on the length and protrusion of the alveolar maxilla (PMP-A and SNA, respectively) and the anteroposterior alveolar jaw relation (ANB) at age 20 years but not on their growth rates. CONCLUSION: Timing of hard palate repair significantly affects the growth of the maxilla in patients with UCLP. Late hard palate repair has a smaller adverse effect than does early hard palate repair on the growth of the maxilla. This timing effect primarily affects the anteroposterior development of the maxillary dentoalveolus and is attributed to the development being undisturbed before closure of the hard palate.

Adolescent↗

The relationship between nasal airway size and nasal-oral breathing in cleft lip and palate.

Clefts of the lip and palate generally result in reduced size of the nasal airway. Procedures such as the placement of a pharyngeal flap tend to further compromise nasal breathing. The purpose of this study was to determine how size of the nasal airway affects the mode of breathing in adults with cleft lip and/or palate. A heterogeneous population of 50 adult subjects with cleft lip and/or palate was studied. Nineteen of the subjects had pharyngeal flaps. Respiratory inductive plethysmography was used in combination with an integrating pneumotachograph to measure percent nasal breathing. Pressure-flow studies were used to estimate nasal airway size. The data revealed that a majority of subjects had an airway size of less than 0.4 cm2, which constitutes impairment. Mean cross-sectional area for all subjects was 0.38 cm2 +/- 0.20 SD. Seventy percent of the subjects studied were oral breathers to some extent. A Spearman rank correlation coefficient of 0.725 (p less than 0.0001) indicated that oral-nasal breathing mode was related to airway size. Airway size in the subgroup with pharyngeal flaps was even smaller (0.31 cm2), while percent nasal breathing was lower. Mouthbreathing was observed in all subjects whose airway size was less than 0.38 cm2.

Adolescent↗

Congenital heart anomalies in patients with clefts of the lip and/or palate.

The nature and severity of congenital heart disease in 78 patients who presented with clefts of the lip and/or palate is reported. The prevalence of bilateral cleft lip and palate in patients with heart lesions was much higher than in cleft patients with normal hearts. Cardiac defects were predominantly conotruncal. Tetralogy of Fallot was present in 24 percent of patients; the prevalence of transposition, atrioventricular septal defect, and truncus arteriosus was also disproportionately high. Patients with conotruncal defects had a greater prevalence of either unilateral or bilateral cleft lip and palate. Most patients with congenital heart disease and clefting had additional abnormalities. Anomalies of other systems were found to be present in 87 percent of patients.

Abnormalities, Multiple↗

Anterior paraseptal cartilage development in normal and cleft lip and palate human fetal specimens.

As part of an ongoing study of age-related variation in fetal cleft lip and palate nasal capsule morphology, anterior paraseptal cartilage development was examined histologically in a sample of nine complete cleft lip and palate human fetuses, and 20 without clefts ranging in age from 8 to 21 weeks. A computer reconstruction technique was used to quantify lengths, volumes, and generate growth curves from various regression equations. Anterior paraseptal cartilage length was seen to change in a sigmoidal fashion while volume changes were best described by a logarithmic curve for both groups, with the steepest increases from 15 to 21 weeks. Cleft specimens exhibited significantly different growth rates (line slopes) for both length and volume dimensions compared with the normal specimens. The larger, for age, cartilages were associated with an enlarged, inferior border of the nasal septal cartilage and vomer in the cleft sample. These results are consonant with previous findings in this cleft fetal sample of other enlarged midline structures (i.e., nasal septal cartilage and vomer). Results suggest that growth abnormalities in one or all of these structures may be yet another mechanism for early nasal airway impingement and reduction in cleft lip and palate neonates.

Case-Control Studies↗

Prevalence and severity of malocclusion in children with cleft lip and/or palate in Hong Kong.

The prevalence and severity of malocclusion among children with cleft lip and palate in Hong Kong has never been assessed or documented. A group of 20 children in the primary or early mixed dentition stages, with clefts of the lip and/or palate were assessed using the Occlusal Index. Results of this preliminary study showed that 92.3 percent of the male and 71.5 percent of the female subjects had severe malocclusion, which implied that comprehensive orthodontic treatment at a later stage would be needed. More than half (69.2% of the male, and 57.1% of the female subjects) had syndrome F, defined as mesial molar relationship, overjet, overbite, posterior crossbite, midline diastema, and midline deviation (Summers, 1966). Therefore malocclusion problems were shown to manifest early in patients with cleft lip and/or palate, and further that it is possible to assess malocclusion prevalence and severity in early stages of dental development.

Child↗

Maxillary lateral incisors of subjects with cleft lip and/or palate: Part 2.

Maxillary lateral incisors on the alveolar cleft were investigated in 431 cleft children registered in the Department of Orthodontics, Kyushu University Dental Hospital. The majority of primary maxillary lateral incisors were located on the distal side of the alveolar cleft in both unilateral cleft lip and alveolus (UCLA) and unilateral cleft lip and palate (UCLP) subjects. Permanent teeth in UCLA tend to be located distally, but in UCLP they tend to be congenitally absent (p less than .01). The majority of primary teeth had normal shapes; the majority of permanent teeth were of intermediate type or were missing congenitally. One third of the UCLA and one half of the UCLP subjects who had primary maxillary lateral incisors were not followed by permanent replacements. The location of the majority of permanent maxillary lateral incisors tallied with that of the primary ones except in four UCLA, ten UCLP, and two bilateral cleft lip and palate (BCLP) subjects. Four UCLA and ten UCLP subjects who had primary lateral incisors on the distal side were followed by their permanent successors on the mesial side. Three UCLP and one BCLP subjects had permanent maxillary lateral incisors even though they had no temporary predecessors.

Adolescent↗

Correction of the bilateral cleft lip nasal deformity: evolution of a surgical concept.

The origin of the nasal deformity of a bilateral complete cleft lip is both primary (deformation/malformation) and secondary (postoperative distortion). This is an interim report of a personal evolution from staged correction of the bilateral cleft nasal deformity to synchronous repair of the nose and the lip and premaxillary-maxillary clefts. The anatomic concept is that, because of the malpositioned alar cartilages, the columella only appears to be short in an infant with bilateral cleft lip. The technical stratagems to model the nose are: (1) alignment of the premaxilla and (2) anatomic placement of the alar cartilages with sculpturing of the overlying soft tissue.

Cleft Lip↗

Median cleft of the lip: its significance and surgical repair.

Median cleft lip is a midline vertical cleft through the upper lip in the absence of a prolabial remnant. This may occur as a sporadic event or be part of an inherited sequence of anomalies. A failure of formation or fusion of the medial nasal prominences derived from the frontonasal prominence is ultimately responsible for this aberration. Two categories of dysplasia are associated: (1) frontonasal deformity associated with hypotelorism and (2) median facial cleft syndrome associated with hypertelorism. A patient presents with median cleft lip, mild bifid nose, and hypertelorism. Following surgical reconstruction, a good result is achieved. The embryology, implications for associated abnormalities, and surgical technique for treating these cases are discussed.

Cleft Lip↗

Medieval example of cleft lip and palate from St. Gregory's Priory, Canterbury.

An archaeologically retrieved skeleton from medieval Canterbury possibly of the late eleventh or twelfth century, displays clear evidence of cleft lip and palate. A case of cleft palate dating from the seventh century, is known from an Anglo-Saxon cemetery at Burwell. This is the first evidence for both cleft lip and palate in British archaeological material. The individual had survived into adulthood. Apart from an odontome, there was no osseous evidence of any other abnormalities. Artistic evidence of cleft lip dates to the fourth century B.C. and surgical intervention (A.D. 390) is known from China.

Adult↗