Predisposing Factors to Formation of Cleft Lip and Palate: Inherited Craniofacial Skeletal Morphology

2004 ◽  
Vol 41 (6) ◽  
pp. 613-621 ◽  
Author(s):  
Evangelia Chatzistavrou ◽  
R. Bruce Ross ◽  
Bryan D. Tompson ◽  
Malcom C. Johnston

Objective To identify inherited craniofacial morphologic features in individuals at high risk for cleft of the lip, cleft palate, or both. Subjects Twins without cleft from 33 pairs of monozygotic twins discordant for cleft lip, cleft palate, or both were studied. There were 17 males and 16 females of Caucasian origin, ranging from 3 to 18 years (15 with cleft lip and palate [CLP], 10 with cleft lip [CL], and 8 with cleft palate [CP]), collected from five craniofacial centers (United States and Canada). Design The twin without cleft (noncleft) from each set was compared with an age- and sex-matched control individual from the Burlington Growth Centre, Toronto, Ontario. Posteroanterior and lateral cephalograms were traced and digitized using a computer custom analysis. Descriptive statistics, Student's t tests, and analysis of variance were used to test 40 variables in a pilot study comparing the noncleft twin groups with the controls. Preliminary analysis permitted pooling of the CLP and CL groups (n = 25). To minimize false-positive significance, only 14 variables (from the maxillofacial area) were tested. Results Using the raw probabilities, eight variables showed significant differences between the pooled noncleft CLP and CL (CL[P]) twin group and the controls. However, when the level of significance was adjusted, only four (nasal width [p < .01], cranial base length [p < .05], cranial base width/length ratio [p < .001], and maxillary width/length ratio [p < .05]) were significantly different. No significant differences were confirmed between the noncleft CP twin group and the controls. Conclusions The noncleft member of a discordant monozygotic pair has a number of facial characteristics that differ from the general population. These may predispose to the formation of a cleft lip or palate and may result from a deficiency or distortion of the mesenchyme that forms the craniofacial structures.

2014 ◽  
Vol 51 (6) ◽  
pp. 658-664 ◽  
Author(s):  
Qinghua Shao ◽  
Zhengxi Chen ◽  
Yang Yang ◽  
Zhenqi Chen

Objective To evaluate the effects of lip repair on maxillofacial development of patients with unilateral cleft lip with or without cleft palate. Design Retrospective. Patients A total of 75 patients were recruited, including 38 surgical patients with complete unilateral cleft lip and alveolus and 37 patients with complete unilateral cleft lip and palate who had lip but not palate repair. As controls, 38 patients with no cleft were selected. All subjects were divided according to two growth stages: before the pubertal peak (GS1) and after the pubertal peak (GS2) Interventions Lateral cephalograms of all subjects were obtained. Main Outcome Measures Cephalograms were analyzed and compared in the study and control groups. Results The patients with unilateral cleft lip and palate in both GS1 and GS2 demonstrated an almost normal maxillary and mandibular growth with retroclined maxillary incisors. The patients with unilateral cleft lip and palate showed a shorter length of maxilla, a more clockwise-rotated mandible, and retroclined maxillary incisors. Conclusions There was an almost normal maxillary and mandibular growth but retroclined maxillary incisors in patients with cleft lip with or without cleft palate who had received lip repair only, indicating that lip repair may not have a negative impact on the maxillofacial development and influences only the inclination of the maxillary incisors. The shorter anterior-posterior maxillary length and larger gonial angle in patients with unilateral cleft lip and palate compared with those in patients with unilateral cleft lip and alveolus suggest that these variations in maxillary and mandibular growth may be a consequence of the cleft itself.


1994 ◽  
Vol 31 (6) ◽  
pp. 452-460 ◽  
Author(s):  
Mohammad Mazaheri ◽  
Athanasios E. Athanasiou ◽  
Ross E. Long

This investigation compares the patterns of velopharyngeal growth in cleft lip and/or palate patients. Those who had velopharyngeal competence and acceptable speech are compared with those who presented with velopharyngeal incompetence requiring pharyngeal flap surgery or prosthesis later. Lateral cephalograms of 30 cleft palate only (CPO), 35 unilateral cleft lip and palate (UCLP), and 20 bilateral cleft lip and palate (BCLP) children of the Lancaster Cleft Palate Clinic were studied. These records were taken at 6 month intervals during the first 2 postnatal years and annually thereafter up to 6 years of age. Soft tissue landmark points in the velopharyngeal region were digitized. Length and thickness of the soft palate and height and depth of the nasopharynx were measured. Evaluation of the growth curves of these four cephalometric variables indicated only two significant differences between children who later required pharyngeal flap surgery and those who did not. These differences were found in the growth in length of the soft palate of the CPO group and in the growth in depth of the nasopharynx of the BCLP group. Based on the present cephalometric data, it is Impossible to predict at an early age those cleft lip and/or palate patients who will later require pharyngeal flaps.


1993 ◽  
Vol 30 (2) ◽  
pp. 175-181 ◽  
Author(s):  
Akira Suzuki ◽  
Yo Mukai ◽  
Masamichi Ohishi ◽  
Yasuko Miyanoshita ◽  
Hideo Tashiro

Relationships between the width of the palatal cleft measured at paIatopIasty and the craniofacial morphology or the occlusal conditions present at approximately 4 years of age were studied in 25 cleft palate (CP) and 39 complete unilateral cleft lip and palate (UCLP) subjects treated at the Dental Clinic of Kyushu University. Posteroanterior cephalograms and dental casts showed that the width of the palatal cleft was significantly correlated with wider upper facial width and posterior dental arch width in UCLP, but not in CP subjects. Cleft width was not significantly correlated with the buccolingual occlusal relationship in either subject type. The anterior occlusal relationship in UCLP was not as good as in CP subjects. On lateral cephalograms, the width of the palatal cleft was significantly correlated with vertical hypoplasia of the upper face in UCLP, but not in CP subjects. The cleft palate width appears to be related to the lateral displacement and the retardation of the downward and forward growth of the nasomaxillary complex in UCLP subjects.


1996 ◽  
Vol 33 (2) ◽  
pp. 96-103 ◽  
Author(s):  
Tuula Laatikainen ◽  
Reijo Ranta ◽  
Rolf Nordström

The craniofacial morphology of 11 pairs of monozygotic (MZ) and 28 pairs of dizygotic (DZ) Finnish twins, discordant or concordant for cleft of the lip (CL), unilateral cleft lip and palate (UCLP), or cleft palate only (CP) were investigated by means of lateral cephalometric radiographs. The results were compared to those of age-, sex-, and cleft-type matched single-birth cleft subjects, and also with normative data from the Nordic population. The co-twins with no cleft lip or palate (NONC) showed only slightly more obtuse gonial and steeper mandibular angles compared to normative data. Twins with CL, UCLP or CP had a more retrusive mandible, a wider cranial base and mandibular angle, and a wider angle between the maxilla and mandible than did the single-birth cleft subjects. Comparison of the noncleft twin group with the CL, UCLP, and CP twin groups for the CL twins showed no significant differences. For the UCLP twins, a more retrusive and down- and backward rotation of both jaws, a wider gonial angle, and a wider cranial base angle was seen. The CP twins had their maxillae slightly retrusive, the down- and backward rotation of both jaws was apparent, and the gonial angle was more obtuse. A comparison between the noncleft MZ and noncleft CDZ twins showed no significant differences. The MZ CP twins had a more retrusive mandible and more down- and backward rotation of both jaws than did DZ CP twins. It thus can be suggested that twinning itself does not seem to have an effect on maxillofacial morphology, but the features of the mandibular structure, the cranial base angulation, and the inclination of the jaws are at least partly genetically induced.


2003 ◽  
Vol 40 (5) ◽  
pp. 453-459 ◽  
Author(s):  
Mary Hardin-Jones ◽  
Kathy L. Chapman ◽  
Julie Schulte

Objective The purpose of the present investigation was to examine the impact of cleft type on early sound development in children with cleft palate (with or without cleft lip). Participants The participants included 53 babies with unrepaired cleft palate aged 8 to 10 months. Thirty-five babies had cleft lip and palate (CLP) and 18 had cleft palate only (CPO). Main Outcome Measures Spontaneous vocalizations of the CLP and CPO groups were compared to determine whether differences were evident in canonical babbling, size of consonant inventory as well as place and manner of consonant production, and frequency of vocalization. Results Student's t tests revealed no significant differences between the groups in canonical babbling, size of consonant inventory, place and manner of consonant production, or frequency of vocalization. However, there was a trend for babies in the CPO group to produce fewer compensatory stop consonants and more anterior place features. In addition, a larger percentage of children in the CPO group had entered the canonical babbling stage at the time of this study. Conclusions In general, the findings of this study suggested that cleft type does not appear to play a significant role in early consonant development for babies with cleft palate. Additional study using a larger CPO group is recommended to further investigate the trend toward better performance noted in this group.


1997 ◽  
Vol 34 (5) ◽  
pp. 405-409 ◽  
Author(s):  
Takeshi Wada ◽  
Koichi Satoh ◽  
Takashi Tachimura ◽  
Unai Tatsuta

Objective: This study was a comparison of the cephalometric growth characteristics of the nasopharyngeal structures between UCLP and noncleft controls. Method: Eighty patients with complete unilateral cleft lip and palate (UCLP group) and 82 noncleft controls (NCC group) were assigned to four develop mental stages (i.e., stage 1, at 4 years; stage 2, at 8 years; stage 3, at 12 years; and stage 4, at 17 years of age). Measurements on the anteroposterior and the vertical dimensions were derived from reference lines and points of nasopharyngeal structures on the lateral cephalograms. Results: The results showed that there were no growth differences between the two groups at any stages in the regions of cranial base and cervical vertebrae, and that growth of the posterior maxilla in the UCLP group was significantly less at any stage in both A-P and vertical dimensions than in the NCC groups. As well, the nasopharyngeal triangle (Ho-At-PMP) in the groups showed almost parallel increase with stage, though with short vertical dimension in the UCLP group, and the soft palate length in the UCLP group was significantly less at stages 2, 3, and 4 compared to that in the NCC group. The adequate ratio (soft palate length/pharyngeal depth) in the UCLP group tended to decrease and was significantly less at stage 4 compared to that in the NCC group. Conclusions: These results indicate that the growth of the cranial base and the upper cervical vertebrae is independent of the effect of clefts or of surgeries on clefts, and that the growth inhibition at the posterior maxilla results in morphologic disharmony of upper nasopharyngeal structures. This could be a potential factor for the reappearance of velopharyngeal incompetence at a later age.


2009 ◽  
Vol 46 (5) ◽  
pp. 468-476 ◽  
Author(s):  
Da-wei Lu ◽  
Bing Shi ◽  
Huai-qing Chen ◽  
Yu Li ◽  
Tian Meng ◽  
...  

Objective: To evaluate the parental craniofacial morphology in Chinese patients with sporadic nonsyndromic cleft lip with or without palate. Methods: A total of 98 parental pairs of nonsyndromic unilateral incomplete cleft lip children, 207 parental pairs of nonsyndromic complete cleft lip and palate children, and 206 normal persons from Sichuan University were involved in this study. A conventional cephalometric analysis was used to measure angles, linear distances, and their ratios. Two-sample Student's t tests and a multivariate discriminant analysis were applied to the data. Results: Data indicate that the unaffected parents of nonsyndromic cleft lip children had on average significantly more acute cranial base angle (Angle N-S-Ba) and larger nasal width (NC-NC′) (p < .01). The healthy parents of nonsyndromic cleft lip and palate children consistently displayed a more acute cranial base angle (Angle N-S-Ba), shorter palatal length (A- PNS) and maxillary length (PNS-ANS), a more obtuse gonial angle (Angle Me-Go-Ar), and a larger y-axis length (S-Gn) and nasal width (NC-NC′) (p < .01). Conclusions: All these results indicate that the healthy parents of patients with nonsyndromic cleft lip with or without palate show distinct characteristics in craniofacial morphology. These parental craniofacial features are more obvious in patients with cleft lip with palate than those with cleft lip only. In general, the characteristics seem to be more distinct in the fathers than in the mothers of cleft patients.


2005 ◽  
Vol 42 (5) ◽  
pp. 490-494 ◽  
Author(s):  
Jörg A. Lisson ◽  
Ilka Hanke ◽  
Joachim Tränkmann

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 T0 and T1. An increase of the anterior height of the mandible is found in the cleft groups.


2008 ◽  
Vol 45 (3) ◽  
pp. 272-277 ◽  
Author(s):  
Piotr Fudalej ◽  
Barbara Obloj ◽  
Zofia Dudkiewicz ◽  
Maria Hortis-Dzierzbicka

Objective: To assess mandibular structure and spatial position following one-stage simultaneous repair of the unilateral cleft lip and palate. Design: Forty boys and 17 girls with complete unilateral cleft lip and palate who underwent one-stage simultaneous repair of the cleft by the same surgeon at the age of 9.23 months (standard deviation  =  1.74) were selected. Lateral cephalograms taken at the age of approximately 10 years were analyzed and were compared with a sex- and age-matched control group that consisted of individuals with Angle Class I, no crossbite, positive overbite <5 mm, mild crowding (Incisor Irregularity Index <3.5 mm), and harmonious facial build. Results: No intergroup differences were demonstrated regarding structure of the cranial base. The mandible was found to be retruded and at a larger inclination to the cranial base as compared with controls. Both total mandibular length (ArGn) and length of the mandibular body were larger in the control group, at <2 mm. Height of the ramus and gonial angle were similar in both groups. Intergender comparison showed few significant differences in control subjects only (SN, SGo, and NMe variables). Conclusions: The mandible, following a one-stage simultaneous repair of cleft, was found to be retrusive, and the length of mandibular body was <2 mm shorter than that of the controls.


1998 ◽  
Vol 35 (2) ◽  
pp. 111-119 ◽  
Author(s):  
Omar Gabriel Da Silva Filho ◽  
Rita De Cássia Moura Carvalho Lauris ◽  
Leopoldino Capelozza Filho ◽  
Gunvor Semb

Objective This report is a retrospective study that compares the craniofacial morphology of adult subjects with unoperated bilateral complete cleft lip and palate (BCLP) with that of a noncleft group. Methods The study was performed on standardized lateral cephalograms obtained at the Hospital for Research and Rehabilitation of Cleft Lip and Palate, University of São Paulo, Brazil. The research group consisted of 28 subjects (20 males, 8 females) with unoperated BCLP, ranging in age from 15 to 41 years. The control group was matched to the cleft group with regard to gender and age. The findings were analyzed on the basis of the two-way analysis of variance (ANOVA) for cleft and gender. Results The most striking difference between the groups was the extremely prominent premaxilla in the cleft group that gave the BCLP face a very convex profile. The mandible exhibited a vertical growth pattern that resulted in a steep mandibular plane, an obtuse gonial angle and a long lower face height. The posterior face height was reduced. The cranial base dimensions were smaller, but there was no difference in cranial base angulation. Conclusions These findings confirm that in subjects with unoperated BCLP, the initial characteristics of the cleft malformation persist during growth.


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