Journal of Speech and Hearing Research, Volume 34, 1169-1179, October 1991
Very-LowBirthweight Children and Speech and Language Development Dorothy M. Aram Maureen Hack Suzanne Hawkins Department of Pediatrics Case Western Reserve University Rainbow Babies and Childrens Hospital Cleveland, OH
Barbara M. Weissman Department of Pediatrics Emory University, Atlanta, GA
Elaine Borawski-Clark Department of Pediatrics Case Western Reserve University Rainbow Babies and Childrens Hospital Cleveland, OH
Very low birthweight (VLBW) is often considered to be a risk factor for speech and language disorders, yet data are equivocal. The present study compared speech and language comprehension and production between 249 very-low-birthweight (VLBW
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Journal of Speech and Heanng Research
have not included comparison groups drawn from similar social class backgrounds. When such comparison groups are included, several studies (Eilers, Desai, Wilson, & Cunningham, 1986; Lloyd, 1984; Lloyd, Wheldall, & Perks, 1988) have demonstrated that the incidence of speech and language disorders is no higher among VLBW children than among their social class peers. Others have shown a relationship between social class and language performance. For example, Lewis and Bendersky (1989) reported that among VLBW children with and without intraventricular hemorrhage, language performance on the Sequenced Inventory of Communication Development (SICD) was highly correlated with social class as well as with medical complications. When the variance accounted for by these factors was removed, language differences were no longer present. Many earlier studies included an entire population of very-low-birthweight children in follow-up studies, not differentiating as to their neurological status (e.g., Rubin, Rosenblath, & Balow, 1973; Zarin-Ackerman, Lewis, & Driscoll, 1977); others have excluded children with frank neurological disorders including cerebral palsy, hydrocephalus, or sensory deficits (Lloyd, 1984; Pearce, Saunders, Creighton, & Sauve, 1988). Still others have compared outcome for children with and without specific neurological abnormalities such as intraventricular hemorrhage (e.g., Byers-Brown, Bendersky, & Chapman, 1986; Fitzhardinge & Ramsay, 1973; Lewis & Bendersky, 1989), typically reporting poorer speech and language performance among the neurologically impaired group. Age of the low-birthweight child at follow-up has ranged from infancy to 10 years of age, thus introducing a further factor potentially related to variable findings. Most studies with VLBW infants or toddlers have reported delayed language (e.g., Byers-Brown et al., 1986; Lewis & Bendersky, 1989; Vohr, Coll, & Oh, 1988); but school-age findings have been less conclusive (e.g., Eilers et al., 1986; Lloyd et al., 1988; Michelsson, Lindahl, Parre, & Helenius, 1984). At least one study reported improved language with increased age: Rickards et al. (1987) found that a cohort of 58 children with birthweights High school High school < High school Social class: Numbera 1-3 4 5 Note. VLBW corrected for preterm birth. aHollingshead two-factor. *Significant at p < .05. **Significant at p < .01. Randomly selected comparison group. A geographically based sample of normal-birthweight children born in 1977-1979 was randomly selected to serve as controls. They were selected from the total population of children born in the eastern part of Cuyahoga County (Cleveland) who were enrolled in public and Catholic diocese schools. The original plan was to select 10 children from each of 50 randomly selected schools assigned to six strata on the basis of racial composition (black versus other) and median family income (high, medium, low) of the school's catchment area. However, because of extensive busing of children in the city of Cleveland, the stratification scheme was used only for children enrolled in suburban and Catholic schools. A simple random sample of children attending public schools on the east side of Cleveland was used. A total of 643 children were thus randomly selected. Of these children, the families of 124 declined to participate, and 156 were either born out of Cuyahoga County or preterm. The control population thus included 363 normal-birthweight full-term children (refer to Table 2). Procedures The speech and language assessments were conducted by masters-level speech pathologists as part of an extensive follow-up program, requiring a full day (5-6 hours) of testing. A structured questionnaire was administered to the parent (usually the mother) concerning marital status, race, income, education, occupation, employment, family composition, and the child's prior health and developmental history. Hearing was assessed with pure-tone audiometric screening and impedance audiometry. Visual acuity was tested with Snellen letters. Several language measures (described below) were administered, and an extensive battery of cognitive, academic achievement, visual-perceptual, and fine motor assessments was conducted. Intelligence test results are summarized in Table 3. A neurological examination, includ-
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All VLBW (N = 249)
Controls (N = 363)
106.1 +4.8* 120 (48%) 31 (13%)* 1177 ±218 29.7 -+2
107.0 ±4.6* 171 (47%) 7 (2Yo)**
33.6 ±6.4 139 (56%)
34.5 +5.9 224 (62%)
69 (28%) 135 (55%) 43 (17%)
116 (32%) 185 (51%) 60 (17%)
77 (31%) 125 (51%) 45 (18%)
130 (36%) 160 (45%) 69 (19%)
ing an evaluation of the motor system, was peformed by a single pediatric neurologist. For the purpose of the analyses of this report, the children were classified as neurologically normal or abnormal (i.e., suffering from a major motor or sensory abnormality). Major neurosensory abnormalities at 8 years were identified in 24 children: 10 with spastic diplegia (2 of whom had severe visual impairments); 4 with hemiplegia; 5 with spastic quadriplegia; 3 with shunt-dependent hydrocephalus (1 of whom also had spastic diplegia); 1 with Tourette's Syndrome; and 1 with a severe hearing loss. Speech and language measures. Speech and language measures were selected to assess multiple aspects of comprehension and production. Only measures for which normative data were available were selected, to permit calculation of standard scores. An attempt was also made to select measures that permitted a range of performance at the 8-year age level and, wherever possible, were sensitive to subtle neurological abnormalities. Administration time and ease of scoring also were factors in test selection, given the extensiveness of the overall test battery and the large number of subjects involved. Three measures of language comprehension were administered. The Peabody Picture Vocabulary Test-Revised (PPVT-R) (Dunn & Dunn, 1981) was included as a measure of single-word vocabulary comprehension because it is a well-standardized measure, used extensively in previous studies of low-birthweight children. Standard scores with a mean of 100 and a standard deviation of 15 are reported. Part IV of the Token Test for Children (DiSimoni, 1974) assessed a child's ability to comprehend and hold in memory commands requiring retention of six bits of information (e.g., Touch the large green circle and the small yellow square). Part V of the Token Test for Children assessed comprehension of 21 commands varying in syntactic complexity. Raw scores representing the number correct from a total of 10 on Part IV and 21 on Part V are reported.
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TABLE 3. Summary of test results. All VLBW (N = 249) Measure
M
Language Comprehension PPVT-R (standard score) 90.57 7.01 Token IV (raw score) 15.61 Token V (raw score) Language Production 197.95 RAN Total (total seconds) 38.00 CELF (raw score) Speech 3.07 PAT (no. errors) Syllable rate (seconds for 10 8.30 repetitions) WISC-R Verbal IQ 95.68 Performance IQ 94.16 Full Scale IQ 94.58 *Significant at .05 using Bonferroni correction.
Ttest, VLBW-controls
SD
t
19.66 2.46 3.41
-2.05 -2.95 -2.96
76.77 10.90
p
Controls (N = 363)
Ttest, controlsIntact VLBW
Neurologlcally Intact VLBW (N = 225)
M
SD
t
p
M
SD
.040 .003* .003*
93.79 7.58 16.41
18.55 2.49 3.02
-1.24 -2.65 -2.63
.215 .008* .009*
91.86 7.06 15.72
17.83 2.43 3 18
4.66 -4.28
.008* .001*
172.91 41.54
41.18 8.68
3.53 -3.55
.001' .001*
187.80 38.81
53.78 9.64
7.86
2.90
.004*
1 53
3.45
1.67
.096
2.13
4.66
4.27
1 55
.123
7 78
3.52
.03
.974
7.79
3 10
17.90 17.70 18.10
-3.35 -4.44 -4.12
.001* 001' 2 SD discrepancy, only one mother each in the VLBW and the control group was less than 18 years of age at the time of the child's birth. When defined by a > 1 SD discrepancy, the mothers of 4 VLBW and 2 control children were younger than 18 years at the child's birth. Of the demographic factors, only race differed for the language-impaired group defined by > 1 but not > 2 SD discrepancy. Among the language-disordered VLBW children, 55% were white, 44% black, and 1% other, compared to the remainder of the VLBW children, where 62% were black and 38% were white. Given the multiple comparisons made and the absence of a race difference when language impairment was defined by > 2 SD, this finding is not robust and was not considered meaningful. Thus, none of the perinatal risk factors was found to be associated as predicted with specific language disorders, and the only demographic factor, race, associated with specific language impairment, appears to have a weak and possibly nonmeaningful association.
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Figure 1. Distributions and chi-square results for performance grouped by standard deviation intervals.
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Aram et al.: Very-Low-Birthweight Children
1177
TABLE 4. Specific language Impairment discrepancy between WISC-R PIQ and language measures. 1 standard deviation PIQ-Language measure
2 standard deviations PIQ-Language measure
VLBW (N = 249)
Controls (N = 363)
VLBW (N = 249)
Controls (N = 363)
Category
#
%
#
%
#
%
#
%
Not discrepant Discrepant & other problemsa SLI
99 66 84
39.8 26.5 33.7
144 53 116
39.7 14.6 45.7
180 33 36
72.3 13.3 14.5
270 25 68
74.4 7.9 18.7
Note. WISC-R = Wechsler Intelligence Scale for Children-Revised; PIQ = performance intelligence quotient; VLBW = very-low-birthweight children; SLI = specific language impairment. aOther problems = PIQ < 85, impaired hearing, and/or neurological deficit.
domly selected from comparable geographic areas and measures of social class status did not differ between the groups. Differences, while statistically significant, are small and the significance achieved is in part attributable to the large numbers of children included in this study. Nonetheless, the findings here are consistent with earlier reports of statistically significant differences on language measures between VLBW children and full-term children. Despite the presence of statistically significant mean differences between VLBW and control children, VLBW children's scores were not more negatively skewed than were controls' scores. The standard deviation distributions demonstrate that the performance of VLBW children is very similar to that of full-term children from comparable social backgrounds. When children with major neurologic abnor-
Discussion The above findings once again present evidence that in comparison to normal-birthweight controls, 8-year-old children whose birthweight was < 1500 grams do present statistically significant poorer mean language comprehension, language production, and speech abilities. When children with frank neurological deficits are excluded, variability among VLBW children's performance is lessened and mean differences are reduced; however, differences remain significant on four of seven measures. Thus, it appears that these mean language differences are not simply attributable to a small number of children with major neurological deficits. Social class differences also cannot explain these significant differences, given the facts that control subjects were ran-
TABLE 5. Perinatal and demographic factors of VLBW children with and without specific language disorders defined as >1 and >2 SD discrepancy. Group >1 SD discrepancy Factor Perinatal Type of delivery Presentation at delivery Multiple birth Birthwt. small vs. ave. for gest. age - Pre-eclampsia/eclampsia + Premature rupture of membranes • Antepartum hemorrhage Apgar 1 or a > 2 SD criterion. In addition, when an array of perinatal and demographic factors including young maternal age were examined to determine whether any were associated with the presence of a specific language disorder, differences were negligible and not in the direction expected for suggesting a possible causal relationship. Language deficits accompanied by low IQs, hearing impairments, and/or neurological deficits, however, were more common among VLBW than control children. These findings have several implications for speech-language pathologists. First, the present study provides little support for the assumption that VLBW and concomitant medical problems are risk factors for SLI. Furthermore, no perinatal risk factors differentiated as predicted between VLBW children with or without SLI. Although VLBW children do not present a higher incidence of SLI as defined here, almost twice as many VLBW as control children do have language deficits accompanied by low IQs, hearing impairments and/or major neurological deficits. Therefore, although VLBW children do not appear to be at greater risk for SLI, they are at higher risk for more pervasive developmental problems that include language. This is reflected in the proportion of children reported by their parents to have received speech and language therapy: 27.5% of the VLBW children, compared to 13.9% of the control children. These data relative to the receipt of therapy, however, must be interpreted with caution for several reasons. First, the VLBW children were a clinically defined group receiving regular medical and developmental follow-up, through which speech and language problems were identified and appropriate referrals for service initiated. Comparable assessments were not routinely available to the controls. Secondly, whether or not a child actually receives speech and language therapy is dependent upon many factors (e.g., availability of services, ability of parents to follow through with recommendations), only one of which is the presence of a speech and language problem. Although comparable proportions of parents of VLBW and control children reported having had concerns during development about their child's speech and language (28.3% and 22.2% respectively), of those whose parents had concerns, 53.5% of the VLBW children in comparison to 38.2% of the controls were reported to have received speech and language evaluations. Further implications of the present findings come from the very high number of children identified in both the VLBW and control groups as having SLI or language deficits accompanied by other developmental problems. On the one hand,
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these findings evidence the potential enormity of the need for speech and language services for children with a history of VLBW or from poor social backgrounds. On the other hand, that 45% of the full-term children would be defined as presenting SLI using a PIQ-language discrepancy of > 1 SD raises the question of the validity of these criteria for defining SLI. Even a criterion of a > 2 SD discrepancy identified more than 18% of the full-term group as having a SLI. This figure far exceeds any estimates of the incidence of developmental speech and language disorders which, for this age population, generally do not exceed more than 3-5% (Leske, 1981). We are unaware of other studies that have applied discrepancy criteria for specific language disorders to randomly selected groups of children who have not been previously identified as language impaired. Speech and language diagnosticians may need to rethink the appropriateness of discrepancy criteria for defining SLI and to validate criteria among other randomly selected groups of children. Several other factors related to this study's findings deserve comment. Because the study assessed children's skills at one point in time only, the variable effect of age at outcome was not examined in this study. VLBW infants and toddlers often are reported to present deficits greater than those comparison groups (e.g., Byers-Brown et al., 1986; Vohr et al., 1988), suggesting that differences in language of VLBW children may be more pronounced at younger ages. At least one study (Rickards et al., 1987) supports this hypothesis. It may be that VLBW contributes to a language delay that is more apparent in the earlier years and lessens over time. In addition, the present study did not attempt to account for the potential effects of any intervention program upon the 8-year speech and language performance of children in either the VLBW or control group. It may be that these children's 8-year status is the result of effective early intervention, although this possibility is unlikely given the well-documented longstanding nature of many developmental speech and language disorders (e.g., Aram, Ekelman, & Nation, 1984). The possibility of a selection bias among controls agreeing to participate in this study also may be present. Although controls were randomly selected from designated geographic areas, 124 families declined to participate. Those control families that chose to participate may have had an increased level of concern about their child's development and, thus an over-represented proportion of speech and language deficits. A cautionary note is also in order regarding the limitations in the language measures used. All of the measures were at the word or sentence level, with no assessment of discourse in either conversation or narration, and no measures of more abstract language abilities. Measures of reading, spelling, and written language also were not reported. Had these aspects of language and language-based academic skills been studied, it is possible that more subtle and/or residual language deficits may have been identified. It may be that by 8 years of age, aural language abilities have normalized but more complex aspects of connected discourse or secondary language systems, including reading and writing, evidence continued deficits. In conclusion, the present study suggests that at 8 years of age, specific language impairment, as defined and measured
Aram et al.: Very-Low-Blrthweight Children
here, is not more frequent among VLBW children. However, more VLBW than control children do present language deficits accompanied by more generalized developmental problems including reduced IQ, hearing loss, and/or major neurological abnormalities. The findings reported here also evidence that mean statistically significant differences between VLBW and control children are not simply the result of inclusion of children with major neurological deficits or the absence of appropriate comparisons to peers from similar social backgrounds. Nonetheless, statistically significant differences do not translate to clinically significant differences as defined by a greater occurrence of more extreme scores or a higher incidence of specific language impairment. Finally, the exceedingly high incidence of comparison children who would be defined as presenting specific language disorders were the criteria used here applied to this randomly selected group of children points to the need for speechlanguage diagnosticians to rethink the appropriateness of use of discrepancy criterion for definition of developmental language impairment.
Acknowledgments This work was supported by NIH Grant RO1 HD20057, the Grant Foundation (1985-1990), and the Robert Wood Johnson Foundation (1975-1978). We appreciate the support of Blanche Caron, Margot Bongiovani, Patricia Webb, and Sharon Cohen in this work.
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