Journal of Pediatric Psychology, Vol. 25, No. 3, 2000, pp. 185-190
© 2000 Society of Pediatric Psychology
Brief Report: Cognitive and Psychomotor Development of Infants With Orofacial Clefts
1 University of Washington School of Medicine, 2 Children's Hospital & Regional Medical Center, 3 University of Washington School of Dentistry
All correspondence should be sent to Matthew L. Speltz, University of Washington, Box 359300-CL-08, Seattle Washington 98105. E-mail: mspeltz{at}u.washington.edu . Marya C. Endriga is now at California State University-Sacramento; Catherine L. Maris is now at the University of California-Berkeley.
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Abstract |
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Objective: To compare at ages 3, 12, and 24 months the cognitive and psychomotor development of 29 infants with cleft lip and palate (CLP), 28 infants with cleft palate only (CPO), and a demographically matched comparison (COMP) group of 69 infants; to examine predictors of cognitive status at age 24 months in the cleft group.
Methods: Infants were administered the Bayley Scales of Infant Development (BSID), mother-infant interactions were observed, and medical records were reviewed.
Results: CLP and CPO groups scored lower than the COMP group on the BSID, but did not differ from one another. Cleft group infants scored lower than COMP group infants on BSID items assessing nonverbal and expressive language skills. Quality of maternal interaction predicted the 2-year Mental Development Index (MDI) scores of infants with clefts.
Conclusions: Infants with clefts show relative deficits in cognitive and psychomotor development. Cognitive deficits are apparent in nonverbal as well as verbal areas of performance.
Key words: orofacial clefts; infants; cognition; psychomotor development.
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Introduction |
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School-age children with cleft lip and palate (CLP) and cleft palate only (CPO) typically score within the low-average range of intelligence, with verbal IQ usually lower than nonverbal IQ. Learning disabilities in reading and language are found in a third to half of these youngsters (Endriga & Kapp-Simon, 1999
). In one of the few longitudinal studies to date, school-age
children with CPO were at greater risk for persistent reading problems than
those with CLP (see Richman, Eliason, &
Lindgren, 1988); however, there is limited understanding of how
cleft diagnosis might specifically lead to differing outcomes in learning
abilities.
The origins and early course of these problems in language and cognition
rarely have been studied. It is reasonable to hypothesize that neurocognitive
deficits would appear early in life, given the interrelated embryological
development of the face and brain, as well as the many infancy risk factors
associated with clefts. These include difficult feedings and poor physical
growth, variable hearing acuity due to ear infections, effect of cleft palate
mechanics on vocalization and speech, cleft surgeries and hospitalizations,
atypical facial appearance, and difficult parent-infant relationships (see
Endriga & Kapp-Simon, 1999,
for a review of these risks).
In three studies, the well-researched Bayley Scales of Infant Development
(BSID) have been administered to infants with clefts. Starr, Chinsky, Canter,
and Meier (1977) found in
cross-sectional analyses that the BSID scores of infants with cleft lip, CLP,
or CPO did not differ from norms at ages 6 and 12 months, but did differ at 18
and 24 months; relative deficits were found on both BSID standard score
indices, the Mental Development Index (MDI) and Psychomotor Development Index
(PDI). In a subgroup of subjects followed longitudinally, Starr et al.
(1977) found no differences in
MDI or PDI scores over time. Kapp-Simon and Krueckeberg
(1996) found that 15% of their
cleft sample had MDI scores two or more standard deviations below the
normative mean (PDI scores were not examined). Unlike the findings of Starr et
al., the MDI scores of infants with clefts declined significantly from early
infancy to the beginning of the third year. Jocelyn, Penko, and Rode
(1996) administered the BSID at
age 12 and 24 months to infants with CLP and a noncleft comparison (COMP)
group matched for gender and relevant social variables (infants with CPO were
not included). Infants with CLP had lower MDI and PDI scores than the
comparison group at both time points.
Our study sought to extend this research by giving the BSID to infants with and without CLP or CPO at ages 3, 12, and 24 months. Three questions were addressed.
First, how do the BSID scores of infants with CLP and CPO compare to those of nonimpaired infants across the first 2 years of life? Given the infancy risk factors listed above and the results of the earlier studies, we expected infants in both cleft groups to show lower MDI and PDI scores than our demographically matched COMP group. No hypotheses regarding differences between the two cleft groups were tendered, nor did we hypothesize group differences in performance across time, given the inconsistency and paucity of previous data pertaining to these issues.
Second, do infants with clefts show relative deficits in only the verbal
aspects of cognitive problem solving? As cleft palate adversely affects the
mechanics of early vocalization and speech production as well as hearing
acuity, infants with either CLP or CPO might score poorly on the MDI because
they fail many of the language-related items. With age, there is increasing
emphasis in MDI items on speech and language skills and less emphasis on
nonverbal skills (e.g., form board completion, motor imitation tasks). This
might account for cleft group infants' declining MDI scores over time in the
Kapp-Simon and Krueckeberg
(1996) study. We therefore
compared the performance of the combined CLP and CPO groups (CLP +
CPO) with the COMP group at ages 12 and 24 months (when there is adequate
language) on three subsets of MDI items: receptive language, expressive
language, and nonverbal problem solving (see
Reznick, Corley, & Robinson,
1997, for details of item subsets and their development). Group
differences were expected only on the language item subsets.
And third, among infants with clefts, do maternal skills during interaction
with the infant predict infants' later cognitive development, after
controlling for infant medical variables? In normative and other medically
high-risk groups, quality of maternal interaction has been associated with
infants' cognitive development (e.g.,
Cohen, 1995) and mothers of
infants with CLP and CPO have shown less responsive interactions with their
infant than comparison group mothers (see
Endriga & Kapp-Simon,
1999). In regression analyses involving the CLP + CPO group, we
investigated the predictive significance of maternal interaction skills to MDI
scores after controlling for four infant medical variables that may also
influence intellectual development (cleft diagnosis, physical growth, age of
child when cleft palate surgery was performed, and hearing acuity).
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Participants
The participants were 126 infants and their mothers. Twenty-nine infants had CLP and 28 infants had CPO. Infants in the cleft groups were consecutive referrals to a cleft-craniofacial clinic at an urban children's hospital. Infants with additional birth defects, known genetic syndromes, or perinatal problems (e.g., preterm birth) were excluded from the study. The COMP group contained 69 healthy, typically developing infants and their mothers. COMP group infants were recruited by posters placed at pediatric clinics and community centers. COMP group participants were case-matched to cleft group infants for age (within a month), gender, ethnicity, maternal age (within a year), socioeconomic status (SES), parity, and parent marital status (it was necessary to match some COMP cases to the mean values of the CLP + CPO group). No significant group differences (CLP vs. CPO and CLP + CPO vs. COMP) were found on any background variable except gender. Consistent with population gender differences for these disorders, there were significantly more males in the CLP group (64%) than in the CPO group (30%),
2(1) = 6.8,
p <.01. This sample was predominantly middle-class, Caucasian (85%), and consisted mostly of two-parent families (84%). Between the first and last assessment point (3 and 24 months), 11% of the sample was lost to attrition (n = 14; 8 were COMP infants). Comparisons of nonparticipating and participating families at 24 months revealed no differences on matching variables.
Measures
Bayley Scales of Infant Development (BSID)
The MDI and PDI scores were used to quantify infants' developmental status
at all time points (Bayley,
1969).
Observational Measures of Maternal Interaction
The quality of maternal interaction while feeding the infant at age 3
months was assessed with the Nursing Child Assessment of Feeding Scale
(NCAFS). The Nursing Child Assessment of Teaching Scale (NCATS) was used to
assess maternal skills in a teaching task with the child at 12 months. Barnard
et al. (1989) report data
supporting the validity and reliability of the NCAFS and NCATS. For both
scales, the Parent Total score was used to index maternal interaction skill,
which is the sum of four subscales (Sensitivity to Cues, Response to Distress,
Social-Emotional Growth-Fostering, and Cognitive Growth-Fostering). In this
sample, alphas of.85 and.79 were obtained for the parent scales of the NCAFS
and NCATS, respectively. Trained coders watched videotapes of mother-infant
interaction. One coder observed the majority of cases, and three coders served
as reliability coders on a randomly selected subgroup of cases (about 50%).
Interrater reliabilities were established for each NCAFS and NCATS item. Mean
percent agreement across all items was 91.2% for the NCAFS (mean 
=.65)
and 86.2% for the NCATS (mean =.59).
Measures of Infant Medical Factors in the Cleft Group
Infants' height and weight were obtained from medical records.
Weight-for-height ratios were calculated and converted to z scores.
Medical clinic visits that included "ear infection" (otitis media)
on the physician's problem list were obtained from medical records and
tallied; this served as a proxy measure of hearing problems. The ages of
infants' cleft palate surgeries were obtained from medical records. Average
age at the initial cleft palate surgery was 12.7 months for the CLP group
(SD = 2.96 months, range = 7.4-18.8 months) and 12.3 months for the
CPO group (SD = 2.87 months, range = 5.1-19.6 months).
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Procedures |
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The first (3-month) assessment was scheduled to occur before any of the infants with CLP had lip repair, with all other infants "yoked" to this schedule; actual infant age was 2.9 months on average (SD = 0.6). The second and third assessments occurred within a month of infants' 12-month and 24-month birthdays, respectively. Infant testing and observations of mother-infant interaction for all visits took place in a clinic room with an observation window that concealed videotaping equipment. Procedures for obtaining parents' consent to participate in the study were approved by an institutional review board.
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Results |
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Question 1: How Do the BSID Scores of CLP, CPO and COMP Group Infants Compare Over Time?
MDI and PDI scores (see Figure 1) were analyzed with repeated measures analyses of variance (ANOVAs). Alpha was.01 for these analyses (with Bonferonni correction). Diagnosis was the between-subjects factor (CLP, CPO, COMP) and time was the within-subjects factor (3, 12, and 24 months). There was a main effect of diagnosis for MDI scores, F(2, 108) = 6.39, p <.005. Between-subjects contrasts showed that the CLP group had lower MDI scores across time than the COMP group, F(1, 108) = 11.70, p <.001. No other main effects or interaction were found for MDI scores.
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Analysis of PDI scores indicated a main effect for diagnosis, F(2, 107) = 5.95, p <.005. The CPO group received significantly lower PDI scores than the COMP group, F(1, 107) = 10.56, p <.005. A main effect of time was also found, F(2, 106) = 26.79, p <.001. Within-subjects contrasts revealed differences among all three time points, Ft1t2(1, 107) = 54.08, p <.001; Ft2t3(1, 107) = 6.94, p <.01; Ft1t3(1, 107) = 21.93, p <.001. Trend analysis of PDI scores showed significant linear and quadratic trends across time, F(1, 107) = 21.92, p <.001, and F(1, 107) = 39.35, p <.001, respectively.
Question 2: Do infants With CLP and CPO Show Only Language Deficits
on the MDI?
We combined CLP and CPO groups for these analyses because both share the
presence of cleft palate. Contrary to our hypothesis, CLP + CPO group infants
at 12 months scored lower than COMP group infants on nonverbal MDI
items, t (114) = -2.31, p <.05; no group differences were
found on either language item subset at 12 months. At 24 months, CLP + CPO
group infants scored lower than COMP group infants on the nonverbal MDI item
subset, t (87) = -2.26, p <.05, and the expressive
language MDI item subset, t (93) = -3.26, p <.01.
Question 3: Do Maternal Interactions With CLP + CPO Group Infants
Predict MDI Scores?
We conducted separate regressions to test a 3-month and 12-month model
predicting the MDI scores of infants with CLP + CPO at 24 months. Both models
tested the hypothesis that maternal interaction would contribute to the
prediction of 24 month MDI scores, after controlling for infant medical
variables. Missing values for predictor variables were estimated with group
means. Low intercorrelations among predictor variables precluded
multicollinearity problems.
Age 3 Months. Three infant variables were entered together at step 1: cleft diagnosis (CLP vs. CPO), weight-for-height z scores at 3 months, and number of clinic visits for ear infections from birth to 3 months. Step 2 entered the NCAFS maternal interaction feeding score. At step 1, with only the infant variables entered, R2 was 0.19, F(3, 53) = 4.3, p <.01. Addition of the maternal NCAFS score to the equation in step 2 produced a significant R2 increment from 0.19 to 0.28, Fchange(1, 52) = 6.5, p <.02. The final model containing all variables offered a significant prediction of 24-month MDI scores, F(4, 56) = 5.2, p <.001. Two variables contributed significantly to this model: weight-for-height (standardized ß =.30; t = 2.5, p <.02) and maternal feeding scores (standardized ß =.30; t = 2.6, p <.02).
Age 12 Months. Four infant variables were entered together at step 1: cleft diagnosis, weight-for-height at 12 months, number of ear infection visits from 3 to 12 months, and child's age when cleft palate surgery was performed. Step 2 entered the NCATS (teaching) maternal interaction score. At step 1, with only the infant variables entered, R2 was 0.17, F(4, 52) = 2.6, p <.05. Addition of the NCATS maternal score to the equation in step 2 produced a significant R2 increment from 0.17 to 0.29, Fchange(1, 51) = 8.9, p <.01. The final model containing all variables offered significant prediction of 24-month MDI scores, F(4, 56) = 5.2, p <.001. Only the maternal NCATS scores contributed significantly to the final model (standardized ß =.40; t = 3.0, p <.01).
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Discussion |
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To our knowledge, this is the first study to examine the MDI and PDI scores of infants with CLP and CPO, in relation to a matched group of typical infants with equivalent social and demographic characteristics. Mean scores of the two cleft groups were consistently lower than those of COMP group infants at all time points, with the exception of PDI scores for the CLP group at 3 months. Infants with CLP had significantly lower MDI scores in relation to COMP group infants, while those with CPO had significantly lower PDI scores. However, as Figure 1 indicates, both cleft groups showed similar MDI and PDI patterns in relation to the COMP group across time, and these groups did not differ significantly from one another on either the MDI or PDI at any time point. Infants with CLP showed a decline in MDI scores at 24 months, but this was not a statistically significant trend. However, given the relatively small number of participants in each of the cleft groups, there is high probability of Type II error, necessitating a replication of these findings. Given the pattern of our results, future studies should administer the PDI as well as the MDI to allow for examination of diagnostic group differences in both areas.
Average BSID scores were relatively high in this study, ranging from 97 to
107 for the cleft groups, and 103 to 114 for the COMP group. This likely
reflects the lower-than-average social risk status of this sample
(primarily middle-class, two-parent, stable families). This may also explain
why we did not find a decline in MDI scores for the cleft groups, as did
Kapp-Simon and Krueckeberg
(1996), who studied an
inner-city sample of families with higher social risks.
We found significant quadratic change in PDI scores across time. As evident in Figure 1, all groups showed diminished performance at 12 months relative to the BSID norms. This finding likely reflects study-specific testing factors at this particular age (e.g., examiner test administration or scoring of ambulatory skills).
Analyses of specific item subsets did not support the hypothesis that the relatively poor MDI performance of infants with clefts in this and previous studies is due primarily or only to speech and language delays or deficits. At both 12 and 24 months, the combined CPO + CLP group had lower scores on nonverbal MDI items than the COMP group, indicating relatively poor performance on tasks that do not require expressive language skills and minimal or no receptive language.
The regression analyses indicated that quality of maternal interactions in
the first year contributed to the prediction of 24-month cognitive status of
infants with clefts, after accounting for infant medical variables. At 12
months, mothers' teaching interaction skills alone accounted for nearly a
third of the variance in their infants' MDI scores a year later. It is
possible that infant variables would have contributed more to the prediction
of cognitive outcome if direct measures of hearing acuity had been used
instead of clinic visits (i.e., the frequency of clinic visits may reflect
factors other than infant health status); this issue needs to be studied
further with direct measures of hearing acuity. Nevertheless, these findings
suggest that interventions that enhance the relationship "match"
between mothers and infants during feeding and teaching situations (e.g.,
Erickson, Korfmacher, & Egeland,
1992) may have positive impact on the cognitive outcomes of
infants with clefts. At the 3-month assessment, infant physical growth also
contributed to the prediction of MDI scores, suggesting that interventions
aimed at dyads containing smaller-than-average infants and less responsive
mothers may be especially promising. The assessment tools used in this study
to observe and code mother-infant interactions (NCAFS and NCATS) are easily
applied in a clinical setting.
One limitation of our study is the confounding of gender and cleft diagnosis. Post-hoc analyses of gender within the cleft groups did not reveal differences or trends, but these comparisons were limited by very small numbers of the underrepresented gender. Future studies will need to recruit a sufficient number of cleft group infants to ensure relatively balanced gender representation, a strategy that will likely require multicenter projects.
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Acknowledgments |
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This research was supported by NICHD grant R01-HD25987-09. We thank Kris Barnes, Lynne Foss, Meg Hatlan, Angela Notari, and Jacquie Stock for their coding of the NCATS data; and Sterling Clarren and Wendy Mouradian for the referral of patients to this research project. We also thank the families who have participated in this research.
Received November 15, 1998; revision received March 11, 1999; accepted June 8, 1999
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References |
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Barnard, K. E., Hammond, M. A., Booth, C. L., Bee, H. L., Mitchell, S. K., & Spieker, S. J. (1989). Measurement and meaning of parent child interaction. In F. Morrison, F. Lord, & D. Keating (Eds.), Applied developmental psychology (vol. 3, pp. 39-79). San Diego: Academic Press.
Bayley, N. (1969). Bayley Scales of Infant Development. New York: Psychological Corporation.
Cohen, S. E. (1995). Biosocial factors in early infancy as predictors of competence in adolescents who were born prematurely. Journal of Developmental and Behavioral Pediatrics, 16, 36-41.[ISI][Medline]
Endriga, M., & Kapp-Simon, K. A. (1999). Psychological issues in craniofacial care: State of the art. Cleft Palate-Craniofacial Journal, 36, 3-11.
Erickson, M. F., Korfmacher, J., & Egeland, B. R. (1992). Attachments past and present: Implications for therapeutic intervention with mother-infant dyads. Special issue: Developmental approaches to prevention and intervention. Development and Psychopathology, 4, 495-507.
Jocelyn, L. J., Penko, M. A., & Rode, H. L.
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Kapp-Simon, K. A., & Krueckeberg, S. M. (1996). Mental development in infants with cleft lip and/or palate. Paper presented at the meeting of the American Cleft Palate-Craniofacial Association, Tampa, FL.
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Richman, L. C., Eliason, M. J., & Lindgren, S. D. (1988). Reading disability in children with cleft lip and/or palate. Cleft Palate-Craniofacial Journal, 25, 21-25.
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