Type of Facial Cleft, Associated Congenital Malformations, and Parents' Ratings of School and Conduct Problems

Transcription

1 Type of Facial Cleft, Associated Congenital Malformations, and Parents' Ratings of School and Conduct Problems JoYcE M. ToBiasEN, PH.D. JANET LEVY, PH.D. Mary A. CARPENTER, PH.D. JOHN M. HIEBERT, M.D. Parents' ratings of conduct and school problems were evaluated in 79 subjects with palatal clefts. Subjects were divided into four groups consisting of: (1) unilateral complete clefts of the lip and palate (UC Group); (2) unilateral complete clefts of the lip and palate with associated congenital malformations (UC-C Group); (3) cleft palate only (CPO Group); and (4) cleft palate only with associated congenital malformations (CPO-C Group). Comparisons between groups suggested that subjects with associated congenital malformations, whether or not they had UC or CPO, tended to have more serious school and conduct problems when compared to children with clefts and no associated congenital malformations. The association of congenital malformations with a cleft lip and palate increased the risk of reported conduct problems at home and behavioral and learning problems at school. Children with facial clefts of all types are at higher risk than children without facial clefts for academic problems (Richman and Eliason, 1982). The sources of these problems are complex and not well understood. Children with clefts are a heterogenous group who vary in many ways including: the severity of the visible facial deformity, type of cleft, and whether or not the child has associated congenital malformations (c. f. Richman and Eliason, 1982). Children with clefts of the palate only do not perform as well as children with clefts of the lip and palate on individual tests of intelligence (McWilliams and Mathews, 1979), language association tasks, auditory short term memory tasks, and tests of reading comprehension (Richman and Eliason, 1984). The presence of associated con- The authors are affiliated with the University of Kansas, College of Health Sciences and Hospital, Kansas City, KS. Dr. Tobiasen is Assistant Professor in the Department of Pediatrics and is affiliated with the Sutherland Institute for Facial Rehabilitation. Dr. Levy is Biostatistician at the R.L. Smith Research Center. Dr. Carpenter is Associate Professor in the Department of Speech and Hearing and is also affiliated with the Sutherland Institute. Dr. Hiebert is Professor and Chairman of the Section of Plastic Surgery in the Department of Surgery and is affiliated with the Sutherland Institute. This research was supported in part by the R.L. Smith Research Center, NIH Grant H002528, and the Children's Deformities Program Endowment Fund genital malformations in addition to the cleft has been associated with lower performance on intelligence tests (McWilliams and Mathews, 1979). These findings, while preliminary suggest that there is a continuum of reproductive casualty that exists within the clinical cleft lip and palate group that is in part defined by the type of cleft and the presence or absence of associated congenital malformations and is manifested by demonstrable differences in cognitive abilities. The purpose of this study was to extend these findings and to examine if type of cleft and presence or absence of associated congenital malformations were also related to children's conduct problems and progress at school as rated by parents. It was reasoned that if children with clefts of the palate only and children with clefts and associated congenital malformations are at greater risk for cognitive development problems than children with clefts of the lip and palate and no associated congenital malformations, they may also be at greater risk for school and conduct problems. Overview METHODS Four groups of children (CPO, CPO-C, UC,

2 210 Cleft Palate Journal, July 1987, Vol. 24 No. 3 and UC-C) were compared with each other on parents' ratings of children's frequency of problem behaviors and parents' ratings of whether or not those behaviors were a problem to them. The cleft groups were also compared with each other on parents' ratings of six school problems. Subjects The subjects were 79 parents of children with clefts. The children ranged in age from 2 to 12 years of age; 20 were female with CPO, 15 were male with CPO, 19 were female with UC, 25 were male with UC. None of the children had other identified genetic syndromes or significant hearing loss (mean loss of worst ear at 500, 1,000, 1,500 cps less than 30 db) at the time these data werecollected. Approximately 82 percent of the families were intact. Most of the families were relatively well educated with fathers' average number of years in school equal to However, the average income was relatively low, with 62 percent of families earning less than or equal to $20,000 per year. Parents completed the questionnaires during a regularly scheduled follow-up visit at a cleft palate clinic. A description of the congenital malformations found in this sample is given in Table 1. Thirtyseven subjects had unilateral complete clefts of the lip and palate with no other recorded congenital abnormalities. This group represented 84.1 percent of the UC patients. Only 7 subjects (15.9 percent) with unilateral clefts also had other congenital abnormalities. There were 35 subjects who had clefts of the palate only. Of these, 21 (60 percent) had no other congenital malformations. Fourteen of the 35 subjects (40 percent) with isolated palatal clefts also had other congenital defects. Associated congenital malformations were more common in children with CPO than in children with UC (x[1] = 5.80; p< 0.02). These findings with regard to the relative proportion of children with congenital malformations in the CPO and UC groups are similar to those reported by McWilliams and Mathews (1979). They reported that 34 percent of the CPO children had associated congenital malformations, and only 14 percent of the UC children did. The samples used in this study and in McWilliams and Mathews (1979) are too small to make meaningful generalizations about the relative proportions of children with congenital malformations in different cleft groups. However, findings across the two studies are consistent and support the conclusion that there is a greater incidence of congenital malformations in CPO children. Measures Eyberg Child Behavior Inventory (ECBI). The ECBI (Robinson, Eyberg, and Ross, 1980) is a comprehensive, behaviorally specific parent rating scale for the assessment of behavioral problems in children. It was standardized on a sample of parents of normal children. Robinson and her colleagues (1980) conceptualized behavior problems (e.g., distractibility, fighting, attention seeking) as normal and present in most children. Children with conduct disorders were described as respresenting cess behavior that deviates from the social norm with a frequency or an intensity that the child's environment deems too high'' (Robinson et al, 1980, p 22). Items on the ECBI describe chil- TABLE 1 Description of Congenital Malformations Cleft Palate Group Unilateral Cleft Group (No. = 35) (No. = 44) No. % No. % - Cleft deformity only Pierre Robin syndrome Without other defects (2) 0 With other defects (5) 0 Other Anomalies Neuromotor/developmental delay (1) (3) Eye Pulmonary hypertension (1) (1) (0) (0) Heart (3) (3) Kidney (1) (0) Minor anomalies (2) (1) Skeletal (0) (1) Neurological (0) (1)

3 Tobiasen et al, PARENTS' RATINGS OF SCHOOL AND CONDUCT PROBLEMS 211 dren's social aggressiveness with peers, parents, and siblings. The ECBI has two scales: the Problem Scale, which is answered as either ""No" or ''Yes", is used to identify the specific behaviors that parents rate as problems; the Intensity Scale consists of a 7-point rating by parents regarding how frequently their child actually exhibits a given problem behavior. Robinson et al (1980) reported that the two scales have a moderate positive correlation. However, parents may note that a specific misconduct occurs very frequently, but that it is not a problem for them. Conversely, an infrequently occurring misbehavior may be rated as a problem by some parents. School Problems. Parents of children attending the cleft palate clinic were asked to rate whether children never, sometimes, or usually had any of the following six problems in school: following instructions, talking out of turn, slow learner, short attention span, not finishing work, and getting along with other children. This information has been useful in the clinic for assessing children's progress in school. RESULTS Intensity of Conduct Problems The mean intensities of each behavior by cleft type and presence or absence of congenital malformations were compared using analysis of variance. The mean intensities for all items and groups are given in Table 2. Subjects with missing values on the ECBI were eliminated from the analyses. This reduced the number of subjects from 79 to 65. Separate analyses of variance were conducted on each item in a 2 % 2 design with cleft type (CPO and UC) and presence or absence of associated malformations as the independent variables. There are eight statistically significant main effects for congenital malformations. That is, children with associated congenital malformations in addition to the cleft, in either the UC or CPO groups, are rated by their parents as more likely to dawdle at mealtime (F [1,61] = 6.56; p < 0.02), be disobedient (F [1,61] = 5.37; p < 0.03), sass adults (F [1,61] = 4.25; p < 0.05), hit parents TABLE 2 Mean Intensities of Conduct Behaviors* CPO CPO-C UC UC-C (No. = 18) (No. = 11) (No. = 29) (No. = 7) Behaviors X SD X SD X SD = X SD. 1. Dawdles getting dressed Dawdles at mealtime , Poor table manners Refuses to eat Refuses to do chores Slow getting to bed Refuses to go to bed Disobeys house rules Disobedient Defiant Argumentative Angry Temper tantrums Sasses adults Whines Cries easily Yells or screams Hits parents Destroys toys , Careless with toys Steals Lies Teases children Verbally fights peers Verbally fights siblings Physically fights friends Physically fights siblings Seeks attention Interrupts Easily distracted , Short attention span Fails to finish tasks Difficult entertaining self Difficulty concentrating Overactive or restless Wets bed * Ratings for intensity are based on a 7-point scale. The higher the mean intensity, the greater the attributed frequency of the conduct problem.

4 212 Cleft Palate Journal, July 1987, Vol. 24 No. 3 (F [1,61] = 4.87; p < 0.04), be distracted (F [1,61] = 4.54; p < 0.04), display a short attention span (F [1,61] = 8.34; p < 0.006), have difficulty concentrating (F [1,61] = 9.66; p < 0.004), and to whine (F [1,61] = 4.00; p < 0.05). An examination of the average intensity of each group (see Table 2) shows that the mean differences between the CPO and CPO-C groups on items are somewhat smaller than the mean differences between UC and UC-C groups. The interactions or the differences between these differences are statistically significant on three items; whining (F [1,61] = 11.19; p < 0.002); interruping (F [1,61] = 8.45; p < 0.006); and attention span (F [1,61] = 8.66; p < 0.005). These effects suggest that children with congenital malformations in addition to UC may be more likely to have conduct problems of a certain type, than CPO-C children when each group is compared to a comparable cleft group without congenital malformations. However, age may also account for these findings in that UC children (X = 7.69; SD = 3.76) were on the average more than 1 year older than UC-C children (X = 6.43; SD = 2.44) while CPO and CPO-C were closer in age (X = 6.71; SD 3.76; X = 7.29; SD = 3.38) for CPO and CPO-C, respectively. While the differences in age are not statistically significant, (F [1,61] = 2.13; NS) they may have influenced parents' ratings of problem behaviors. It has been reported that younger children display conduct problems more frequently than older children (Robinson et al, 1980). There was only one item, "tease other children'' in which children with congenital malformations were rated as less of a problem than children without congenital malformations (F [1,61] = 648; p < 0.02). There were no other statistically significant effects in this set of analyses. In summary, these data hint that there may be some tendency for children who have clefts and associated congenital malformations to more frequently display behavior related to noncompliance with parental discipline and conduct problems related to difficulty in the completion TABLE 3 Mean Percent of Parents Endorsing Conduct as Problems * p < t p < 0.05 p < 0.01 No Other Anomalies Some Other Anomalies Behaviors (Mean %) (Mean %) 1. Dawdles getting dressed Dawdles at mealtime * 3. Poor table manners Refuses to eat Refuses to do chores Slow getting to bed Refuses to go to bed Disobeys house rules Disobedient Defiant Argumentative Angry Temper tantrums Sasses adults % 15. Whines Cries easily Yells or screams Hits parents Destroys toys Careless with toys Steals Lies Teases children Verbally fights peers Verbally fights siblings Physically fights friends Physically fights siblings Seeks attention Interrupts Easily distracted Short attention span Fails to finish tasks Difficult entertaining self Difficulty concentrating Overactive or restless Wets bed

5 Tobiasen et al, PARENTS' RATINGS OF SCHOOL AND CONDUCT PROBLEMS 213 of tasks than children who have clefts and no evidence of associated congenital malformations. Behaviors that Parenis Rate as Problems Parents rated which behaviors, specifically, were a problem for them. Parents rated an average of 3.8 behaviors as problems (SD = 4.7; range = 0-18). Parents expressed considerable variability in which behaviors they considered to be a problem. Parents do not statistically differ by cleft type (F [1,61] = 0.004; NS), or by presence of other malformations (F [1,61] = 2.25; NS) in the average number of behaviors they rate as problems (CPO, X = 3.28; CPO- C, X = 4.83; UC, X = 2.95; UC-C, X = 5.71). Item analyses were conducted on the mean percent of parents by cleft group that endorsed a specific behavior as a problem. The data for the cleft groups with associated congenital malformations versus the group with no associated congenital malformations are presented in Table 3. Chi square analyses reveals that on 6 of the 36 behaviors, significantly more parents with children who have clefts and other malformations endorse the items as "problems'' than do parents of children with clefts and no associated malformations. These behaviors are: dawdles at mealtime (Item 2) (x2[1] = 12.51; p < ), disobedient (Item 9)(x2[1] = 4.71; p < 0.03), temper tantrums (Item 13) (x2[1] = 6.56; p < 0.02), sasses adults (Item 14)(x2[1] = 8.22; p < 0.005), short attention span (Item < 4.72; p < 0.03), wets bed (Item 36)(x*[1] = 3.72; p < 0.05). Eighteen other conduct problems were more often endorsed as problems by parents of children with clefts and associated malformations than by parents of children with clefts and no other associated malformations. These differences were not statistically different but are consistent with the other findings that these parents may experience more conduct problems with their children. Four of the items that are more frequently rated as problems are also rated as more frequently occurring on the intensity scale: dawdles at mealtime, disobedient, sasses adults, and short attention span. There were no statistical differences on items for percent endorsement as a problem by cleft type and for the interaction of cleft type by presence or absence of other congenital malformations. Parents' Ratings of School Problems Parents rated six school behaviors on whether they were a problem; almost never, sometimes, and usually. Only data from children aged 4 to 12 and attending school were analyzed. This reduced the number of subjects to 56 (CPO = 15; CPO-C = 8; UC = 27; UC-C = 6). Chi square analyses were conducted on each of these items by cleft type and presence or absence of other malformations, after regrouping responses into two categories: ''almost never'' and ''sometimes or usually." The frequencies of endorsements for school problems are given in Table 4. Chi square analyses reveal that on 4 of the 6 behaviors, significantly more parents with children who have clefts and other malformations endorse the items as "problems'' than do parents of children with clefts and no associated malformations. These behaviors are: following instructions (x2[1] = 8.09; p < 0.005), slow learner (x2[1] = 8.84; p < 0.003), short attention span (x2[1] = 9.98; p < 0.002), and not finishing work (x2[1] = 3.73; p < 0.05). These findings suggest that children with clefts and other associated congenital malformations may be at increased risk for problems in school. There were no other statistical effects associated with school problems. TABLE 4 Mean Percent of Parents Endorsing School Behaviors as Problems *p < 0.01 tp < 0.05 Parents Endorsing School Behavior as Problems (%) No Related Related Malformation Malformations School Behaviors (No. = 42) (No. = 14) Following instructions * Talking out of turn Slow learner " Short attention span * Not finishing work Getting along with others

6 214 Cleft Palate Journal, July 1987, Vol. 24 No. 3 DISCUSSION AND CONCLUSIONS The findings of this investigation moderately support the contention that children with facial clefts and associated congenital malformations may be at higher risk for conduct problems at home and difficulties in school than children with clefts and no associated congenital malformations. Other investigators have reported findings that suggest that children with associated congenital malformations may be a special risk group within the facial cleft population. Lewis (1961) found no difference in IQ by type of cleft in 548 children, but did find lower IQs in those with other congenital malformations. McWilliams and Mathews (1979) found that cleft children with other anomalies tended to have lower IQs. They also reported that the CPO-C and UC- C groups in their study had more articulation errors than the CPO and UC groups. The possibility of speech deficits in children with clefts and associated congenital malformations was investigated in post hoc analyses in the present study. A retrospective analysis was conducted of speech evaluations of study patients made when they attended the cleft lip and palate clinic. These evaluations were part of a routine follow-up by the three speech language pathologists associated with the clinic. The speech measure abstracted from the records was the general scale used to gauge the overall impact of the child's expressive communication skill on the listener. The measure included considerations of fluency, phonation, resonance, articulation, and some aspects of expressive language. The ratings were scaled as follows: (1) no deviance (difference) noted; (2) deviant, but minimal and nondistracting (no interference with communication); (3) distracting, but no effect on intelligibility; (4) mild effect on intelligibility; (5) moderate effect on intelligibility; (6) severe effect on intelligibility. The mean speech ratings between groups were analyzed in a 2 X 2 analysis of variance (cleft type x presence or absence of associated malformations). There was a statistically significant main effect for associated malformations (F [1,61] = 9.14; p < 0.005). There were no other statistically significant effects. The means (X) and standard deviations for groups were: CPO, X = 2.93, SD = 1.39; UC, X = 2.42, SD = 1.50; CPO-C, X = 4.88, SD = 1.13; UC-C, X = 3.50, SD = Children with clefts and associated malformations had significantly worse speech ratings in the clinic. These findings support the suggestion that children who have facial clefts and associated congenital malformations may be at greater risk for certain behavioral and developmental problems than children with clefts and no associated malformations. The speech findings are limited as they are basedon a clinical measure of speech with undocumented reliability. Other studies have reported relationships between behavior andthe incidence of congenital anomalies not including facial clefts. In particular, the incidence of minor physical anomalies in otherwise healthy infants and children has recently been related to various behavioral and developmental problems. For example, a higher number of minor physical anomalies (e.g., low seated ears, epicanthus, and third toe longer than second toe) has been related to hyperactivity in children 2% years old (Waldrop et al, 1968), and elementary school boys (Halverson and Victor, 1976; Quinn and Rapoport, 1974). Other investigators have reported finding a relationship between the number of minor physical anomalies and infant irritability (Quinn et al, 1977), temperament problems in children 2 years old (Burg et al, 1978, 1980), and school failure in first graders (Rosenberg and Weller, 1973). The presence of minor physical anomalies is presumably related to congenital insult(s) occurring within the first 4 months of fetal life (Smith, 1970). It has been suggested that the factors that produce the minor anomalies also contribute to the development of central nervous system abnormalities responsible for behavioral or developmental problems (Steg and Rapoport, 1978; Waldrop et al, 1978). The findings in the present investigation, while preliminary, do support the suggestion that children with facial clefts and associated congenital malformations may be at risk for behavioral and developmental problems. There are several notable weaknesses in the research design that limit this interpretation. The number of parent participants is relatively small, and the number of subjects in the different cleft groups varied. The available sample did not allow for statistical comparisons by sex or age. These data are based on parents' self reports and most likely reflect errors in recall and personal bias. Nevertheless, the findings indicate that much more research is needed of factors such as associated congenital malformations that may contribute to behavioral and developmental risk within the facial cleft patient population. Acknowledgement. We would like to express our appreciation to Ralph Shelton for his careful review and suggestions. REFERENCES Burg C, Hart D, QUInN P, RaPOPORT J. Newborn minor physical anomalies and prediction of infant behavior. Autism Child Schizophrenia 1978; 8:427.

7 _ Tobiasen et al, PARENTS' RATINGS OF SCHOOL AND CONDUCT PROBLEMS 215 BurG C, RAPOPORT J, BARTLEY LS, QUINN P, TimmMINS P. Newborn minor physical anomalies and problem behavior at age three. Am J Psychiatry 1980; 137, HALvERsSON CF, VICTORJB. Minor physical anomalies and problem behavior in elementary school children. Child Dev 1976; 47:281. LEwISs RA. A survey of the intelligence of cleft palate children in Ontario. Cleft Palate Bull 1961; 11:83. McWi1u1ams BJ, MaTHEws HP.. A comparison of intelligence and social maturity in children with unilateral complete clefts and those with isolated cleft palate. Cleft palate J 1979; 16:363. QUINN P, RaporoRrT J. Minor physical anomalies and neurologic status in hyperactive boys. Pediatrics 1974; 53: 742. QUINN P, RENFIELD M, BuURG C, RapPoPORT J. Minor physical anomalies: A newborn screening and 1-year followup. Am Acad Child Psych 1977; 16:662. RICHMAN LC, Eu1asoN M. Psychological characteristics of children with cleft lip and palate: Intellectual, achievement, behavioral, and personality variables. Cleft Palate J 1982; 19:249. RICHMAN LC, ELIAsoN M. Type of reading disability related to cleft type and neuropsychological patterns. Cleft Palate J 1984; 21:1. ROBINSON E, E BERG S, Ross AW. The standardization of an inventory of child conduct problem behaviors. Clin Child Psych 1980; 9:22. RosENBERG JB, WELLER GM. Minor physical anomalies and academic performance in young school children. Dev Med Child Neurol 1973; 15:131. SmITH D. Recognizable patterns of human malformation. Philadelphia: Saunders WB, STEG JP, Rapoport J. Minor physical anomalies in normal, neurotic, learning disabled, and severely distrubed children. J Autism Child Schizophrenia 1975; 5:299. WALDROP MF, BELL RQ, MCLAUGHLIN B, HALYERsON CF. Newborn minor physical anomalies predict short attention span, peer aggression, and impulsivity at age 3. Science 1978; 199:563. WaALproP M, PEDERSON FA, BELL RQ. Minor physical anomalies and behavior in preschool children. Child Dev 1968; 39:391.