OBJECTIVE: To evaluate the agreement between parental reporting of development of children born very preterm using the Parents’ Evaluation of Developmental Status (PEDS®) questionnaire and professional assessment by a paediatric developmental team in the detection of sensorineural disability.
METHODS: A cross-sectional cohort study of 362 children born in Queensland with a birthweight < or = 1250 g, who were surviving at 2 and 4 years of age corrected for prematurity, was conducted. Parents completed the PEDS® questionnaire prior to their child receiving a neurodevelopmental assessment. The level of agreement for sensorineural disability between the neurodevelopmental assessment and the parents' score on the PEDS® questionnaire was measured using the kappa statistic, and screening test characteristics were calculated. Logistic regression was used to investigate factors that might affect agreement.
RESULTS: Two hundred and eighty-three (78%) of the eligible children were located and contacted. Of these, 216 (76%) agreed to participate in the study (110, 2-year-olds; 106, 4-year-olds). Agreement between the two forms of rating sensorineural disability (developmental quotient > -2SD (standard deviation), cerebral palsy, bilateral blindness and deafness requiring aids) for the 4-year age group children was fair (kappa = 0.27, P = 0.001). PEDS® accurately identified 69% (11 of 16) of disabled children and 72% (65 of 90) non-disabled children. The test characteristics for these children were similar to Glascoe’s norming sample with a PPV 31% (95% CI: 14-48%), specificity 72% (95% CI: 62-81%), but lower sensitivity 69% (95% CI: 62-81%) and higher false-negative rate 31% (95% CI: 11, 59). Agreement for the 2-year age group was fair with poor test characteristics. Other comparisons for both age groups (PEDS® A and B compared using a disability status with DQ > -1SD) showed poor agreement and test characteristics. Gestation age < 27 weeks and maternal education at or below grade 10 in the 2-year age group were the only factors independently affecting agreement.
CONCLUSIONS: The agreement between parental evaluation of sensorineural disability status using PEDS® and paediatrics developmental assessment in children born very preterm at 4-years corrected age for prematurity compares favourably with Glascoe’s norming sample. The lower agreement seen in the 2-year age group limits the utility of PEDS® to be used as a screen for disability at an age when early intervention may be useful. The PEDS® questionnaire is designed and normed for the general paediatric population, and it is not clear if parents of children born very preterm may have interpreted the PEDS® questionnaire in relation to their satisfaction with their child’s developmental progress rather than their child’s functional ability.
Comment from Dr. Glascoe: The authors were gracious enough to share their data. Problems in the administration of PEDS® were numerous: a) many children were previously diagnosed and receiving intervention, but were not excluded from the study or elevated to Path A (per PEDS® Brief Guide directions). In this study, 6 were previously diagnosed and 4 of the 6 were already in services; b) the authors did not re-administer PEDS® by interview if nothing was written on the Response Form (ahem)! This problem occurred with 13% of cases (N = 19/144), and; c) clinician/researchers did not add their own concerns from observations or physical exam, i.e., over-ride PEDS® scoring if clinical judgment indicated a problem.The consequences for reported sensitivity/specificity.
Had PEDS® been scored correctly, 30 out of 34 children with a diagnosis of CP or intellectual deficits ( > 1sd below average), would have received Path A or B results, i.e., sensitivity = 88%. Parents whose children did not have a diagnosis also had high levels of worry (often about language, or described features of ASD and so forth). So, specificity was understandably low (59/163 = 36%).
Bottom line, researchers need to read the manuals and adhere to the directions for each test under study–both screening and diagnostic tools. Although this recommendation sounds axiomatic, it needs reiterating!
Below is a letter to the editor about this paper. In general, children with previously identified disabilities do not need screening and should either be excluded from validation studies using PEDS® or automatically elevated to Path A when a developmental diagnosis is described. Parents, if satisfied with services, may not raise concerns, understandably:
RE: PARENTS’ EVALUATION OF DEVELOPMENTAL
The article by Drs Pritchard, Colditz and Beller entitled, ‘Parents’ Evaluation of Developmental Status’ (PEDS®) in children born with a birthweight of 1250 g or less (J. Paediatr. Child Health 2005; 41: 191–6) involved use of a screening and surveillance tool I authored, known informally as PEDS®.1 I congratulate the authors on a potentially helpful study and offer several suggestions for explaining their findings and planning additional analyses:
- The authors attempted to view whether PEDS® identified children with cerebral palsy. Although this is valuable, PEDS® is designed, in contrast, to identify children eligible for special education.
- In the USA, cerebral palsy alone is not sufficient for placement in special classes. Rather, children qualify under the category of physical impairment only if they show motor disabilities in addition to deficits in intelligence, adaptive behaviour and/or academic/preacademic skills, that is, their difficulties must interfere with school success or its likelihood. As a consequence, parental concerns about communication and cognition have stronger predictive value than do gross motor concerns. If the criteria for special education placement are different in Australia, then Drs Pritchard et al. are encouraged to consider whether a different constellation of parental concerns as elicited by PEDSwould perform more effectively.
- If detection of cerebral palsy with or without other developmental deficits is, in fact, a goal for Australian children, it might be wise to evaluate whether parents of children with cerebral palsy describe motor development in a way that could be discerned from parents with motor concerns but whose children are developing in a typical fashion. For example, statements such as, ‘my four month old is very strong and can stand for hours’; ‘she holds her body in an odd way’; ‘she keeps her legs stretched out and crossed all the time’; ‘he’s like a rag doll’, all might be clues to the presence of spasticity, scissoring, persistence of primitive reflexes, hypotonicity etc. Drs Pritchard et al. are encouraged to make use of their rich dataset to consider secondary, qualitative analyses of the concerns of parents whose children have cerebral palsy.
- It may be unnecessary to point out that screening tests do not need to be deployed with children with previously identified disabilities. In the US validation studies, 25% of families whose children were already enrolled in special education, did not raise concerns on PEDS®. Drs Pritchard et al. speculate sagely that satisfaction with services may reduce or eliminate the parental concerns. It is also likely that parents whose children have known disabilities come to view their child’s development in a relativistic and incremental manner – comparing current progress, however slow, only to past performance – and not, as most parents do, by comparing their child to other children. This would make for an interesting longitudinal study and one that is also encouraged. Some evidence for marked early differences in the perspective of parents whose children have known disabilities is found in a study in which parents of 2-year-old children were asked to predict adult outcome. Parents of non-disabled children consistently predicted quite exalted futures, a phenomenon the researchers dubbed, ‘Presidential syndrome’. In contrast, parents of children with cerebral palsy and/or mental retardation, simply predicted their child would become an average, normal adult.
- Although both sets of predictions may be unlikely, they do suggest early and significant differences in the views of parents whose children have disabilities.
- Finally, PEDS® is not only a screening test, it is also a surveillance tool. As such, it calls for, when making referral decisions, provider input including results of a physical exam, medical history, observations and/or knowledge of the family. The PEDS® Brief Guide to Scoring and Administration states, ‘If parents have no concerns or nonpredictive concerns but clinical judgment suggests the presence of a problem, follow Path A or B’ (the two planks of the PEDS® algorithm denoting increased risk and need for additional assessment (p. 6)). Had the authors followed this recommendation, their sensitivity findings would have been substantially higher. I am happy to assist the authors with additional analyses of their and in formulating hypotheses for future research.
- Glascoe FP. Parents’ Evaluation of Developmental Status (PEDS®). Nashville, TN: Ellsworth & Vandermeer Press, 1997.
- Glascoe FP. Collaborating with Parents: Using Parents’ Evaluation of Developmental Status to Detect and Address Developmental and Behavioral Problems. Nashville, TN: Ellsworth & Vandermeer Press, 1998;
- l.Shapiro DM, Ostroff JS, Howe GW. Parents’ beliefs about the severity and permanence of their child’s handicap. In: Proceedings of the 19th Annual Gatlinburg Conference on Research and Theory in Mental Retardation and Developmental Disabilities; 13–15 March 1986, Gatlinburg, TN.
Frances Page Glascoe
Department of Pediatrics,
Nashville, TN, USA
Pritchard And Letter (PDF)