Reproductive history of mothers of children with solitary, nonsyndromic cleft lip and/or palate.

The reproductive history was studied to evaluate if the three types of solitary, nonsyndromic clefts: cleft lip (CL), cleft lip and palate (CLP), and cleft palate (CP) are homogeneous entities. Occurrence of fetal loss, maternal health, and drug consumption of the mother during the pregnancy were compared in cases involving three types of clefts. Data was gathered from 87 children with clefts, 55 males and 32 females. Spontaneous abortions and vaginal bleeding were found to occur significantly more often in the (older) mother of a CLP child. This suggests that the factors involved in the etiology of CLP differ from the factors involved in CL and CP. Therefore, grouping of data of the three types of clefts in studies on the etiology should be avoided.     

Habilitation of patients with clefts: parent and child ratings of satisfaction with appearance and speech.

This study examined ratings regarding satisfaction with facial appearance and speech performance from 495 parent-child pairs. Data were obtained from school-aged children (5-18 years old) and their parents using standardized independent interviews. Results revealed that 54 percent of the children with cleft lip (CL) or cleft lip and palate (CLP) were very pleased with their appearance, and 62 percent of the cleft palate or CLP subjects were very pleased with speech. Low, but statistically significant correlations exist between the female subjects and their parents in satisfaction with appearance. Although older cleft lip and/or palate patients reported increased satisfaction with speech, no age differences in patient satisfaction with appearance were observed in subjects with CL/CLP. Parents of females expressed more concern about their daughters' appearance than parents of males, while parents of males were more concerned about speech. Implications for craniofacial habilitation teams and research suggestions are discussed.     

A comparative study of prespeech vocalizations in two groups of toddlers with cleft palate and a noncleft group.

OBJECTIVE: This study examined the prelinguistic contoid (consonant-like) inventories of 14 children with unilateral cleft lip and palate (C-UCLP) at 13 months of age. The children had received primary veloplasty at 7 months of age and closure of the hard palate was performed at 3-5 years. The results of this investigation were compared to results previously reported for 19 children with cleft palate and 19 noncleft children at the age of 13 months. The children with clefts in that study received a two-stage palatal surgery. This surgical procedure was formerly used at our center and included closure of the lip and hard palate at 3 months of age and soft palate closure at 22 months of age. DESIGN: Retrospective study. SETTING: The participants were videorecorded in their homes during play with their mothers. The videotapes were transcribed independently by three trained speech pathologists. PATIENTS: Fourteen consecutive patients born with C-UCLP and no known mental retardation or associated syndromes served as subjects. RESULTS: The children who received delayed closure of the hard palate demonstrated a significantly richer variety of contoids in their prespeech vocalizations than the cleft children in the comparison group. Both groups of subjects with clefts had significantly fewer plosives in their contoid inventory than the noncleft group, and there was no difference regarding place of articulation between the group that received delayed closure of the hard palate and the noncleft group.     

Comparative study between children with and without cleft lip and cleft palate, part 2: electromyographic analysis.

OBJECTIVE: This study was conducted to compare electromyographic (EMG) activity of superior orbicularis oris muscle between children with repaired cleft lip and cleft palate and children without clefts. METHODS: This study included 28 children with mixed dentition. They were divided into two groups. The study group included 14 children with repaired unilateral cleft lip and cleft palate, ranging in age from 6 to 12 years, who presented clinically with a short upper lip, abnormal lip seal, and inhibition of sagittal development of the midface as assessed radiographically. The control group included 14 children without clefts ranging in age from 8 to 11 years. All had normal lip seal, nasal breathing, and a clinically normal body posture. DESIGN: Bipolar surface electrodes were used for EMG recordings of resting level activity and during swallowing of saliva, speech, and chewing and swallowing of an apple. RESULTS AND CONCLUSIONS: A significantly higher level of activity at rest and during swallowing of saliva was observed in the cleft lip and cleft palate group. Similar activity during speech and chewing and swallowing of an apple was observed in both groups. The higher level of activity at rest and during swallowing of saliva in children with cleft lip and cleft palate seems to suggest that upon higher functional demands their activity increases less than in children without clefts. From a clinical point of view, if increased EMG activity at rest and during swallowing of saliva reflects increased force on the maxilla, then our findings may corroborate Bardach's findings (1990) that surgical treatment of cleft lip has an iatrogenic effect on facial growth, although the lack of significant correlation between the cephalometric data and EMG findings in the present study.     

Type of oral cleft and mothers' perceptions of care, health status, and outcomes for preadolescent children.

OBJECTIVE: To evaluate the outcomes of care for children by type of oral cleft. DESIGN: Data were collected through structured telephone interviews during 2003 in Iowa with mothers of 2- to 12-year-old children with oral clefts. Interviews with mothers of children with clubfoot and statewide data on Iowa children were used for comparison. PARTICIPANTS: Participants included mothers of children in Iowa born between 1990 and 2000 with nonsyndromic oral clefts. Children were identified by the statewide Iowa Registry for Congenital and Inherited Disorders. MAIN OUTCOME MEASURES: Rating of cleft care, severity of condition, health status, esthetic outcome, speech, and school performance were evaluated by type of oral cleft. RESULTS: Children with cleft lip and palate were most likely to have their clefts rated as very severe. Children with palatal involvement were reported to have a lower health status and were almost twice as likely to be identified as having a special health care need compared with either children with cleft lip or children statewide. Children with cleft lip had more esthetic concerns; children with palatal involvement had the most speech concerns. CONCLUSIONS: Although mothers generally believed their children had received high-quality care, ratings of the children's current health status and outcomes of care varied significantly by type of cleft (cleft lip, cleft palate, and cleft lip and palate). Differences observed in this population-based study support the proposition that cleft type should be considered when examining outcomes of care for children with oral clefts.     

Left-sided predominance of hypodontia irrespective of cleft sidedness in a French population

Objective : Individuals with oral clefts exhibit considerably more dental anomalies than do individuals without clefts. Our aim was to evaluate the prevalence of tooth agenesis in a sample composed of 124 children (81 boys and 43 girls, mean age 12.5?years) with clefts registered with the Cleft Palate Center in Strasbourg (France). Design : Cleft types and dental agenesis were assessed by clinical and radiographic examination. Cleft types were divided into four groups according to the clinical extent of the cleft (cleft lip only [CL, 12.9%], cleft lip and alveolus [CLA, 4%], cleft lip and palate [CLP, 49.2%], and cleft palate only [CP, 33.9%]). Results : Of the subjects 63% had evidence of hypodontia: maxillary lateral incisors (54%) and upper and lower premolars (32%) were the most common missing teeth. The number of dental ageneses associated with CP (54%) and CLP (79%) was significantly higher than that with CL (33%). All these anomalies were found in proportionately higher frequencies as the severity of the cleft increased, and we found left side predominance for hypodontia (p < .01) irrespective of cleft sidedness. Conclusions : Both right-sided and left-sided clefts were more frequently correlated with left-sided dental agenesis. This left-sided prevalence suggests that common signaling malfunctions might be involved, both in dental development anomalies and cleft genesis.     

Asymmetry of left versus right unilateral cleft impairments: an experimental study of face perception.

OBJECTIVE: Previous psychosocial studies of adults born with cleft lip and palate have provided circumstantial evidence that surgically repaired right-sided unilateral clefts may be more disfiguring than left-sided clefts. The present study asked if such asymmetries are physiognomic asymmetries or arise "in the eye of the beholder," representing perceptual processes in face recognition. DESIGN: Color slides of 160 children (6 years of age) and young teenagers (16 years of age) were rated by subjects for perceived disfigurement. Sixty of the subjects had unilateral complete cleft lip and palate (30 had a right-sided cleft and 30 had a left-sided cleft), 60 had unilateral cleft lip/alveolus (30 right-sided and 30 left-sided clefts), 32 children had bilateral cleft lip and palate, and 8 children had cleft palate only. Faces were shown in normal and in mirror-reversed versions; the order in which faces were shown was randomized, as were other stimulus factors such as cleft type, age, and gender. SETTING: The study was conducted as a classroom-type experiment at the Vision Laboratory, Department of Psychology, Oslo, Norway. PARTICIPANTS: Thirty-seven students of psychology at the University of Oslo, who were ignorant of the purpose of the study, acted as subjects. MAIN OUTCOME MEASURE: Subjects rated perceived disfigurement using a visual analog scale. RESULTS: Modest but highly consistent hemifacial asymmetries in judged disfigurement were found, with left-sided unilateral clefts rated as less disfiguring than right-sided unilateral clefts. Unilateral clefts were judged as being less disfiguring than the bilateral clefts, and cleft lip/alveolus was judged as being less disfiguring than cleft lip and palate. The patterns of facial judgments were almost identical in the normal and reversed-slides conditions. CONCLUSIONS: Asymmetries between left- and right-sided clefts reside in physiognomic factors rather than in hemispheric asymmetries controlling the perceptual process of face judgment.     

Assessment of patients' level of satisfaction with cleft treatment using the Cleft Evaluation Profile.

OBJECTIVES: Determination of the psychosocial status and assessment of the level of satisfaction in Malaysian cleft palate patients and their parents. DESIGN: Cross-sectional study. PARTICIPANTS AND METHODS: Sixty cleft lip and palate patients (12 to 17 years of age) from Hospital Universiti Sains Malaysia and their parents were selected. The questionnaires used were the Child Interview Schedule, the Parents Interview Schedule, and the Cleft Evaluation Profile (CEP), administered via individual interviews. RESULTS: Patients were teased because of their clefts and felt their self-confidence was affected by the cleft condition. They were frequently teased about cleft-related features such as speech, teeth, and lip appearance. Parents also reported that their children were being teased because of their clefts and that their children's self-confidence was affected by the clefts. Both showed a significant level of satisfaction with the treatment provided by the cleft team. There was no significant difference between the responses of the patients and their parents. The features that were found to be most important for the patients and their parents, in decreasing order of priority, were teeth, nose, lips, and speech. CONCLUSIONS: Cleft lip and/or palate patients were teased because of their clefts, and it affected their self-confidence. The Cleft Evaluation Profile is a reliable and useful tool to assess patients' level of satisfaction with treatment received for cleft lip and/or palate and can identify the types of cleft-related features that are most important for the patients.     

Sucking performance of babies with cleft conditions.

OBJECTIVE: To describe the sucking performance of bottle-fed babies with cleft conditions. PARTICIPANTS: Forty 2-week-old-babies with cleft lip (CL; n = 8), cleft palate (CP; n = 22), and cleft lip and palate (CLP; n = 10) were examined. METHODS: Suction, compression, and other sucking parameters were measured during bottle-feeding and compared to determine if they varied with cleft condition or feeding ability. RESULTS: All babies with CL and one with CLP demonstrated suction. Thirteen of 22 babies with CP demonstrated suction but only three maintained regular pressure changes over time. Between-group differences in the amplitude of suction and compression were associated with cleft condition. Cleft lip participants demonstrated the greatest amplitude of suction followed by those with CP and CLP. Cleft lip and CP participants generated similar amplitudes of compression. This was greater than their counterparts with CLP. Good feeders (n = 15) generated high levels of suction, while satisfactory (n = 15) and poor feeders (n = 10) did not generate any during bottle-feeding. CONCLUSION: Between-group differences in intra-oral pressures were confirmed when babies were examined by cleft condition. Babies with smaller clefts (i.e., CL or minor soft palate clefts) were more likely to generate normal levels of suction and compression compared to their counterparts with larger clefts. Since good feeders were more likely to have smaller clefts it was not surprising that they demonstrated higher suction pressures than babies with satisfactory or poor feeding ability. Compression values were not significantly different across the feeding ability groups. These data may inform feeding management strategies.     

Conversational skills of children with cleft lip and palate: a replication and extension.

OBJECTIVE: To replicate and to extend a previous study examining the conversational skills of children with cleft lip and palate. PARTICIPANTS: Thirty-four children (33 to 44 months) participated: 17 children with cleft lip and palate and 17 noncleft children. METHODS: The children were observed during an interaction with caregivers in their homes. Samples of caregiver-child interactions were coded as assertive or responsive, for type of conversational act, and for discourse level categories. Profiles of conversational activity were determined for each child based on the coding. Correlations were performed to examine the relationship between assertiveness and speech variables (articulation and resonance) for the children with cleft lip and palate. RESULTS: Group comparisons revealed that the children with cleft lip and palate produced fewer assertive utterances, were less likely to respond adequately to comments by caregivers, and produced more topic maintaining and fewer topic extending utterances than did their noncleft peers during conversational interactions. Examination of individual child data indicated that 35% of the children with cleft lip and palate exhibited conversational profiles characterized by either low assertiveness or low responsiveness. Finally, a significant positive correlation was noted between conversational assertiveness and speech production skills. CONCLUSION: These findings suggested that the children with cleft lip and palate were less conversationally assertive than their noncleft peers. Further, there appeared to be a relationship between speech production skills and conversational skills, suggesting that poor speech may be impacting language performance for these children.     

Prevalence of nonsyndromic oral clefts in a reference hospital in the state of Minas Gerais, Brazil, between 2000-2005

The aim of the present study was to analyze the prevalence of nonsyndromic oral clefts in children receiving treatment at the Center for the Rehabilitation of Craniofacial Anomalies, José do Rosário Vellano University, Alfenas, MG, Brazil. All the data for the epidemiological study was retrieved from the files of 126 pediatric patients with oral clefts without any additional malformation, who came to the center for treatment between 2000 and 2005. A predominance of clefts was observed in Caucasians, and the ratio of male to female was 1.3. Males were 2.57 times more affected by cleft lip and palate (CLP) than females. CLP with a prevalence of 39.68% and isolated cleft lip (CL) with a prevalence of 38.09% were the most common anomalies, followed by isolated cleft palate (CP; 22.23%). Complete and unilateral CLP (26.19%) presented the highest prevalence, followed by incomplete and unilateral CL (23.81%). The present study presents the experience of a reference hospital in the state of Minas Gerais; however, the real prevalence of oral clefts in Brazil is still unknown. Our findings differ from those of a few previous Brazilian reports because they suggest similar prevalences of CLP and CL, and a higher prevalence of CLP in Caucasian males.     

Parent and child ratings of satisfaction with speech and facial appearance in Flemish pre-pubescent boys and girls with unilateral cleft lip and palate

The purpose of this controlled study is to determine satisfaction with speech and facial appearance in Flemish pre-pubescent children with unilateral cleft lip and palate. Forty-three subjects with unilateral cleft lip and palate and 43 age and gender matched controls participated in this study. The Cleft Evaluation Profile was used to assess the perceived satisfaction for individual features related to cleft care. Both the cleft palate subjects and their parents were satisfied with the speech and facial appearance. The Pearson χ(2) test revealed significant difference between the cleft palate and the control group regarding hearing, nasal aesthetics and function, and the appearance of the lip. An in depth analysis of well specified speech characteristics revealed that children with clefts and their parents significantly more often reported the presence of an articulation, voice and resonance disorder and experienced /s/ /r/ /t/ and /d/ as the most difficult consonants. To what extent the incorporation of specific motor oriented oral speech techniques regarding the realisation of specific consonants, attention to vocal and ear care, and the recommendation of secondary velopharyngeal surgery, with the incorporation of primary correction of the cleft nose deformity simultaneously with primary lip closure, will aid these patients are future research subjects.     

Comparative study between children with and without cleft lip and cleft palate, part 1: cephalometric analysis.

OBJECTIVE: This study was conducted to compare craniofacial relationships, position, and curvature of the cervical spine between children with cleft lip and cleft palate who had been operated on and children without clefts. METHOD: This study was performed in 28 children with mixed dentition. They were divided into two groups. The study group included 14 children with unilateral operated cleft lip and cleft palate, ranging in age from 6 to 12 years, who clinically presented with a short upper lip, abnormal lip seal, and inhibition of sagittal development of the midface that was radiographically assessed. The control group included 14 children without clefts, ranging in age from 8 to 11 years. All of them had normal lip seal, nasal breathing, and a clinically normal body posture. DESIGN: A lateral craniocervical radiograph in a self-balanced natural head position in an erect posture, and without using a head holder, was taken for each child of both groups, with the mandible in maximum intercuspation and lips in habitual posture. The true vertical was marked on all the films. Specific angular and linear dimensions were used to assess the craniocervical relationships, as were the position of the cervical spine, its curvature, or both. RESULTS AND CONCLUSIONS: The study group presented a significant increase in the extension of the head on the neck, forward position of the cervical spine, and a decrease in the curvature of the cervical spine in comparison with the children without clefts. These results are more relevant considering that the study group also presented higher significant values of lower facial height than children without clefts.     

Preferential associations between oral clefts and other major congenital anomalies

Objectives: To identify preferential associations between oral clefts (CL = cleft lip only, CLP = cleft lip with cleft palate, CP = cleft palate) and nonoral cleft anomalies, to interpret them on clinical grounds, and, based on the patterns of associated defects, to establish whether CL and CLP are different conditions. Design And Settings: Included were 1416 cleft cases (CL = 131, CLP = 565, CP = 720), among 8304 live- and stillborn infants with multiple congenital anomalies, from 6,559,028 births reported to the International Clearinghouse for Birth Defects Surveillance and Research by 15 registries between 1994 and 2004. Rates of associated anomalies were established, and multinomial logistic regressions applied to identify significant associations. Results: Positive associations with clefts were observed for only a few defects, among which anencephaly, encephaloceles, club feet, and ear anomalies were the most outstanding. Anomalies negatively associated with clefts included congenital heart defects, VATER complex (vertebral defects, imperforate anus, tracheoesophageal fistula, and radial and renal dysplasia), and spina bifida. Conclusion: The strong association between all types of clefts and anencephaly seems to be attributable to cases with disruptions; the association between CP and club feet seems to be attributable to conditions with fetal akinesia. Some negative associations may depend on methodologic factors, while others, such as clefts with VATER components or clefts with spina bifida, may depend on biological factors. The different patterns of defects associated with CL and CLP, indicating different underlying mechanisms, suggest that CL and CLP reflect more than just variable degrees of severity, and that distinct pathways might be involved.     

Self-concept of early primary school age children with visible or invisible defects

This study compares self-concept scores on the Primary Self-Concept Inventory (PSCI) of 58 7-year-old children. The experimental groups included the three following subgroups: those with visible defects (cleft lip), mixed visible and invisible defects (cleft lip and palate), and invisible defects (cleft palate). The control consisted of first grade students with no physical defects. Significant differences between the subjects with clefts and controls were found. Children with cleft lip and palate (visible and invisible defects) demonstrated the lowest self-concept scores. These test results suggest that early primary school age children experience significant stigma. Therefore, early evaluation of psychosocial stress factors for children with clefts is suggested. School personnel can contribute to addressing self-concept concerns of children with defects. Suggestions for further research are presented.     

Different cleft conditions, facial appearance, and speech: relationship to psychological variables.

OBJECTIVE: The purpose of this investigation was to study the adjustment and learning characteristics of children with different types of clefts. The hypotheses were that there may be different relationships among cleft variables (speech and appearance) according to the cleft types. DESIGN: The study compared three cleft groups on behavior rating, anxiety scales, depression scales, and self-perception (analyses of variance) and examined the influence of facial and speech ratings on self-perception (multiple regression analyses). SETTING: All patients were treated at a university hospital cleft palate clinic. PATIENTS: Sixty-five children aged 8 years to 17 years were selected based on nonsyndromic cleft (unilateral cleft lip and palate [ULP], bilateral cleft lip and palate [BLP], and cleft palate only [CPO]) and no significant neurological condition or hearing loss. RESULTS: The findings indicated children with CPO showed greater problems with parent- and teacher-reported depression, anxiety, and learning related to speech than children with ULP or BLP. The later two groups showed fewer problems and a greater relationship of problem to facial appearance. The children with ULP self-reported lower levels of depression than the other two groups. CONCLUSIONS: Children with cleft show relatively good overall adjustment, but some problems appear related to speech and facial appearance. Subgroups may need to be studied separately.     

Oral clefts: a retrospective study of prevalence and predisposal factors in the State of Mexico

The purpose of this study was to up-date the records concerning oral clefts (OCs) encountered at the Child Hospital of the Maternal Infantile Institute of the State of México, and to examine the association of predisposing factors. A retrospective study of the medical records of patients generated over a 5-year period was carried out. A total of 835 files were reviewed, representing 504 boys and 331 girls. The studied variables were the type of oral clefts and predisposing factors. Kendal correlations at P < or = 0.05 and chi-squared at P < or = 0.05 were used to find any associations between variables. The distribution of oral cleft was: cleft lip and palate (CLP) 70%, cleft palate (CP) 21%, cleft lip (CL) 8%, separate cleft lip and cleft palate 1%. The sex ratios were 1.7 for CLP, 1.7 for CL, and 1 for CP. Municipalities with ethnic groups as well as industrial, agricultural and pottery activities showed a high rate of prevalence. Although there was no significant association with birth weight, familial history of clefting, consanguinity, medication usage during pregnancy, or paternal occupational risk, the results suggested that the most severe clefts were proportionally related to these factors. A significant association with maternal and paternal age, abortion rate, and parity was found. Additionally, maternal agricultural activities suggested that pesticide exposure might be a factor.     

Language input of mothers interacting with their young children with cleft lip and palate

Maternal language addressed to 1-, 2-, and 3-year-old children with cleft lip and palate was studied. Videotaped interactions were obtained from 23 mother-child dyads (13 mother-cleft lip and palate child dyads, and 13 mother noncleft child dyads) during free play. Results indicated more similarities than differences in maternal language characteristics for the two groups of mothers.     

The effect of early insertion of grommets on the development of speech in children with cleft palates

The aim of this study was to find out whether simultaneous primary palatoplasty and myringotomy was advantageous in the management of otitis media with effusion in children with cleft lip and palate and whether this combined operation resulted in improvement in the early development of speech. Assessments of hearing and speech were made in 50 consecutive patients with cleft lip and palate who had the combined operation (the cleft group). The results were compared with those of an age- and sex-matched control group. The incidence of otitis media with effusion in the cleft group was 24%, compared with 14% in the control group. Assessment of speech using the cleft audit protocol for speech showed that 90% of patients with clefts had speech-intelligibility ratings that were either normal or showed only minor defects, compared with 98% of controls. Among patients with clefts, there was no evidence of cleft-type characteristics of speech in 58%.     

Seasonal variation and regional distribution of cleft lip and palate in zambia

Objective: To assess variations in seasonality and regional distribution of orofacial clefts in babies born in Zambia. Design: A retrospective chart review was done using records of all cleft procedures performed by the only plastic surgeon in Zambia (G.J.). Delivery data from the University Teaching Hospital (UTH) were also examined to estimate the birth prevalence of orofacial clefts (55,108 live births between 2001 and 2005). Patients: All cleft patients operated in Zambia from 2000 to 2006 (413 patients). Results: A low birth prevalence of clefts (1/4239 live births) was found using UTH delivery data. Surgical data showed no difference for the frequency of one gender over another overall (M:F ratio is 1.04; p = .70). More bilateral clefts occurred in cleft lip and palate (CLP) patients than in cleft lip (CL) patients (p < .01), and more unilateral left-sided clefts occurred in CL than in CLP patients (p = .03). The data reflected seasonal variation in month of birth of cleft lip with or without cleft palate (CL+/-P) patients (p < .01), with a peak in April and May and more births in March through August (57.2%) than in September through February (42.8%). There was regional variation in cleft births among the nine Zambian provinces (p < .01). Conclusions: This study shows seasonal variation in clefts that may be explained, at least in part, by environmental factors affecting the development of CL+/-P. Access to treatment is likely the major determinant of regional disparity in clefts. These results provide a basis for further epidemiological studies of orofacial clefts in Zambia.