Screening For Developmental Delay in The Setting of A Community Pediatric Clinic - A Prospective Assessment of Parent-Report Questionnaires

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Screening for Developmental Delay in the Setting of a Community Pediatric Clinic: A Prospective Assessment of Parent-Report Questionnaires David Rydz, Myriam Srour, Maryam Oskoui, Nancy Marget, Mitchell Shiller, Rena Birnbaum, Annette Majnemer and Michael I. Shevell Pediatrics 2006;118;e1178 DOI: 10.1542/peds.2006-0466


The online version of this article, along with updated information and services, is located on the World Wide Web at: http://pediatrics.aappublica t/118/4/e1178.full.html .full.html http://pediatrics.a

PEDIATRICS is the official journal of the American Academy of Pediatrics. A monthly publication, it has been published continuously since 1948. PEDIATRICS is owned, published, and trademarked by the American Academy of Pediatrics, 141 Northwest Point Boulevard, Elk Grove Village, Illinois, Illi nois, 60007. Copyright © 2006 by the American Academy of Pediatrics. All rights reserved. Print ISSN: 0031-4005. Online ISSN: 1098-4275.

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Screening for Developmental Delay in the Setting of  a Community Clinic:Questionnaires A Prospective Assessment of Pediatric Parent-Report David Da vid Ryd Rydz, z, MSca, My Myri riamSrou amSrour, r, MD MD,, CMa,b, Mar MaryamOskou yamOskoui, i, MD, CMa,b, Nan Nancy cy Mar Marget get,, MScc, Mit Mitche chell ll Shi Shille ller, r, MD,CMb, Rena Ren a Bir Birnba nbaum, um, MSc MSc,, OTd, An Annet nette te Maj Majnem nemer, er, PhD PhD,, OTa,d, Mi Mich chae aell I. Sh Shev evel ell, l, MD MD,, CMa,b

Departments of  a Neurology/Neurosurgery, bPediatrics, and  c Educational and Counseling Psychology and  d School of Physical and Occupational Therapy, McGill University, Montreal, Quebec, Canada  The authors have indicated they have have no financial relationships relevant relevant to this article to disclose.


OBJECTIVES. Our goal for this study was to prospectively test whether parent-com-

pleted questionnaires can be effectively used in the setting of a busy ambulatory pediatric clinic to accurately screen for developmental impairments. Specific ob jectives included (1) assessing the feasibility of using parent-report instruments in the set settin ting g of a com commun munity ity pediatri pediatricc cli clinic nic,, (2) eva evalua luati ting ng the acc accura uracy cy of 2 available screening tests (the Ages and Stages Questionnaire and Child Development Inventory), and (3) ascertaining if the pediatrician’s clinical judgment could  be used as a potential modifier. METHODS. Subjects were recruited from the patient population of a community clinic

providing primary ambulatory pediatric Subjects menta me ntall del delay ay or con concer cerns ns not noted ed wer were e care. contac con tacted ted at without the timeprevious of the their irdeveloproutin rou tine e 18-month 18-m onth-old -old visit. Those subjects subjects who agre agreed ed to part particip icipate ate were randomly assigned to 1 of 2 groups and completed either the Ages and Stages Questionnaire or Child Development Inventory. The child’s pediatrician also completed a brief questionn ques tionnaire aire regarding regarding his or her opinion of the child’s development development.. Those children for whom concerns were identified by either questionnaire underwent addition addi tional al deta detailed iled test testing ing by the Batt Battelle elle Development Development Inve Inventor ntory, y, the “gol “gold d standard” for the purposes of this study. An equal number of children scoring within wit hin the nor norms ms of the scr screen eening ing me measu asures res als also o und underw erwent ent tes testin ting g wit with h the Battelle Development Inventory. peds.2006-0466 doi:10.1542/peds.2006-0466 Key Words developmental screening, parent-report, questionnaires, infant Abbreviations AAP—American Academy of Pediatrics CCC—Children’s Care Clinic BDI—Battelle Developmental Inventory

ASQ—Ages and Stages Questionnaire CDI—Child Development Inventory Accepted for publication May 23, 2006 Address correspondence to Michael I. S hevell, MD, CM, Montreal Children’s Hospital, Room A-514, 2300 Tupper, Montreal, Quebec, Canada H3H 6P3. E-mail: [email protected] PEDIATRICS (ISSNNumbers:Print, 0031 PEDIATRICS 0031-4005 -4005;; Online, Onlin e, 10981098-4275 4275).). Copyr Copyright© ight© 2006by the AmericanAcademy canAcademy of Pediat Pediatrics rics

RESULTS. Of the 356 pare parents nts contacted, contacted, 317 paren parents ts (90%) agre agreed ed to participate. participate.

Most parents correctly completed the Ages and Stages Questionnaire (81%) and the Child Development Inventory (75%). Predictive values were calculated for the Ages and Stages Questionnaire and the Child Development Inventory (sensitivity: 0.67 and 0.50; specificity: 0.39 and 0.86; positive predictive value: 34% and 50%; negative predictive value: 71% and 86%, respectively). Incorporating the physi-


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cian’s opinion regarding the developmental status of the child chi ld did not improve improve the acc accura uracy cy of the scr screen eening ing questionnaires. CONCLUSIONS. Three important conclusions were reached:

(1) pare parent-c nt-compl ompleted eted quest questionna ionnaires ires can be feasi feasibly bly used in the setting of a pediatric clinic; (2) the pediatrician’s opinion had little effect in ameliorating the accuracy of either questionnaire; and (3) single-point accuracy rac y of the these se scr screen eening ing ins instru trumen ments ts in a com commu munit nity y setting did not meet the requisite standard for development screening tests as set by current recommendations. This study raises important questions about how developmental screening can be performed, and we recommend me nd add addit ition ional al res resear earch ch to elu elucid cidate ate a suc succes cessfu sfull screening procedure.

a study from 1987 demonstrated that only 20% to 30% of developmentally impaired children were being identified tifie d befo before re schoo schooll age by prim primary ary care practitione practitioners rs 5 using developmental surveillance. For this reason, professional organizations advocate the use of standardized developmental screening tests.2 Developmental screening has been shown to improve the accuracy with which children chil dren’s ’s deve developm lopmenta entall dela delays ys are ident identified ified when compared comp ared with decis decisions ions based only on clin clinical ical judg6–8

lifelong benefits but also that developmental attainment is maximized when intervention is commenced earlier. Consequently, professional societies such as the American Academy of Pediatrics (AAP) advocate the identification cat ion of dev develo elopme pmenta ntally lly del delay ayed ed chi childr ldren en bef before ore 2 years of age.2 The most effective manner to identify children with developmental delay still remains elusive, although the need to find a practical screening method has been indicated in the literature as early as 1979. Shonkoff et al concluded conc luded that “mor “more e prec precise ise techn technique iquess for pedi pediatri atricc developmentall assessment and conclusive evaluations of developmenta specific interventions will have to be produced. . . . The current difficulty in defining criteria for optimal pediatric

ment. Indeed, the sensitivity and specificity of screening instruments are usually reported between 70% and 90%.9–12 Although the use of screening tests would improve the rate and accuracy of identification, a recent survey sur vey dem demons onstra trated ted tha thatt onl only y 23% of pri primar mary y car care e clinicians used a standardized screening tool, leading the authors to conclude: “Our findings do raise. . .the concerns that systems of care that foster the proper use of adequate detection methods in the primary care setting continue to be elusive.”13 A previous survey documented some of the obstacles faced by primary care practitioners, with time constraints being voiced by 82% of them.14 The lack of medical staff (48%) and the burden of cost (44%)) in moni (44% monitori toring ng devel developme opmental ntal delay, for whic which h they are not well compensated, were also listed as significant preventive factors.14 As a substitute to these time-intensive, pediatricianadmini adm iniste stered red scr screen eening ing tes tests, ts, a rol role e for sta standa ndardi rdized zed parent-c pare nt-compl ompleted eted ques questionn tionnaire airess can be cons consider idered. ed. Parent-completed questionnaires are as accurate as developmental screening instruments, because current research sear ch stro strongly ngly suppo supports rts the obse observat rvation ion that pare parents, nts, regardle rega rdless ss of diffe difference rencess in soci socioeco oeconomi nomicc stat status, us, geographical location, or parental well-being, can give accurate information about their child’s development. 15–21 Moreover More over,, pare parent-c nt-compl ompleted eted scre screenin ening g tool toolss are cost cost-effective in the short-term, can be completed over the teleph tel ephone one,, in the waiting waiting room, room, or by mai mail, l, and are 22 time-efficient. Realizing the advantages of parent-com-

management emphasizes the need for creative, methodologically sophisticated research in the area.”3 More recently, Sices et al4 stated, “The AAP . . . does not provide specific guidance on how a primary care physician is to perform developmental surveillance and screening. Research on how these guidelines can be best implemented in the context of primary care practice would help standardiz dar dize e and enhance enhance the value of the experien experience ce for 4 patients and families.” These authors call for the formulation of guidelines, which can be used by primary care practitioners in a time- and cost-efficient manner. Currently Curre ntly,, most prim primary ary care prac practiti titioners oners inco incorporporate developmental surveillance that relies heavily on a pediatrician’s intuition and clinical judgment. Although such a method might be accurate given enough time, training, supplementary resources, and expertise, studies have demonstrated that clinical judgment alone does not accurately detect developmental delay. For example,

pleted questionnaires, Regalado and Halfon wrote: “The available evidence suggests that assessment of developmental issues might benefit from the wider use of structured, validated approaches. . . . [S]ystemic assessment of parent’s concerns can play a role in identifying children with developmental problems, replacing or supplementing longer and more costly developmental screening assessments.” 23 Conse Consequent quently, ly, the use of paren parent-com t-com-pleted ple ted que questi stionn onnair aires es mig might ht be mor more e pra practi ctical cal in the  busy pediatric clinic. The literature concerning the topic of developmental screenin scre ening g with pare parent-c nt-compl ompleted eted ques questionn tionnaire airess has several shortfalls that limit the widespread applicability of the results. Previous studies have focused on specific cohorts that had already been flagged as being delayed or consisted of high-risk infants (very low birth weight and prematurity prematurity). ).24–26 Sam Sample ple sizes in the ma major jority ity of studies, which rely on specific cohorts, are small, and the


N ESTIMATED 12%  to 16% of the US pediatric pop-

ulation has a developmental disability.1 There has  been increasing pressure to identify these children at an earlier age, with the current focus being on infants.2 Part of the reason is that research has demonstrated not only that intervention programs are cost-effective and have

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applicability of these results to the general pediatric population as a whole is uncertain.24–26 Studies, using other screening instruments as the gold standard, must also be analyzed skeptically, because validating a screening instrument with another parent-completed screening instrument can skew results.25 These studies do not offer guidel gui deline iness tha thatt pri prima mary ry car care e pra practi ctitio tioner nerss can use to screen for developmental delays.7,24–26 Part of the reason might be that these tests are not designed with an em-

typically occurs at 2–3 years).18 All subjects turning 18 months were contacted and eligible to participate unless there was an established significant developmental disability. abil ity. Recruitment Recruitment was term terminat inated ed after 101 asses assesssments were completed with the Battelle Development Inventor Inve ntory y (BDI (BDI)) beca because use of fundi funding ng cons constrai traints. nts. Also, parents completing the questionnaire needed to have a fourth-grade reading level and understanding of English,  because these questionnaires are presently available in

phasis on keeping the methods in line with the actual reality of the clinical setting. The majority of research has focused on validation or predictive value and not on actual methods or practicality. Indeed, Sonnander states that “few . . . studies focusing on evaluation of developmental screening programs conducted within a clinical setting sett ing were found found,” ,” wher whereas eas “emp “empiric irical al rese research arch into child development and the predictive value of developmental tests is extensive.”27 Thus, there is a lack of direct pragmatic research addressing the area of how a primary care practitioner should screen for developmental delay in a community. To create an efficient system for recognition of development opm ental al pro probl blems ems,, we defi defined ned 3 obj object ective ivess for our study: (1) to test whether questionnaires can be feasibly completed by the caregiver in the waiting room of a busy pediat ped iatric ric cli clinic nic;; (2) to exa exami mine ne the acc accura uracy cy of the these se parent-completed questionnaires when used in such a setting; and (3) to assess what role the practitioner plays in ameliorating the questionnaire’s accuracy. These aspects pec ts of par parent ent-co -compl mplete eted d scr screen eening ing tes tests ts nee need d to be examined systematically to provide a rational foundation to the formulation of a feasible, clear, and accurate screening protocol.

English only.


Subjects Subjects were recruited from the patient population of the Children’s Care Clinic (CCC). Located in Pierrefonds, Quebec, Queb ec, Canada, the CCC is an exclu exclusive sively ly pedi pediatri atrics cs group practice, incorporating 7 full-time pediatricians. It is community based, drawing from a suburban, largely middle midd le clas classs popul populatio ation n and prov providin iding g comp comprehen rehensive sive primary pediatric care to its clientele. Its patient demographic grap hic and appoi appointme ntment nt syst systems ems are full fully y comp computer uter-ized. Subj Su bjec ects ts we were re re recr crui uite ted d at th the e ti time me of th thei eirr 18 18-monthmon th-old old vis visit. it. Thi Thiss is a sta standa ndard rd rou routin tine e vis visit it tha thatt coincides with the administration of a number of vaccines. The 18-month timing was also chosen because it allows allo ws for asse assessme ssment nt of moto motor, r, langu language, age, social, and cognitive skills by standardized developmental screening instrume inst ruments. nts. Furth Furthermo ermore, re, succ successfu essfull iden identific tificatio ation n of delays at this point in time would represent a substantial improvement over what is currently achieved (ie, referral to pediatric subspecialists and rehabilitation services e1180

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Procedures The database at the CCC was scanned continually for subjec sub jects ts who wer were e tur turnin ning g 18 mon months ths of age in the forthcom fort hcoming ing 2 mont months. hs. The iden identifie tified d care caregive givers rs were sent a cover letter, signed by the pediatricians at the CCC indicati indi cating ng thei theirr suppo support, rt, that descr described ibed the stud study y and included incl uded stud study y conta contact ct info informat rmation. ion. A tele telephone phone call from a research assistant was made soon after receipt of the letter, during which any questions or concerns regarding the study were addressed and verbal consent or refusal was noted. If a parent refused participation, the reason for refusal was obtained and noted. All parents who refused were also asked to complete a simple demographic questionnaire over the telephone that gathered basi basicc demo demograp graphic hic infor informati mation on on empl employme oyment, nt, income, and education. If verbal consent was given, the subject’s appointment date was noted, and a dossier was placed pla ced in the chi child’ ld’ss fol folder der at the clinic. clinic. Eac Each h dos dossie sierr contained: 1. One of 2 selected parental-report parental-report measures (the Ages and Stages Questionnaire [ASQ] or Child Development Inventory [CDI]) depending on group assignment, which was performed using random-number tables and blocking to ensure equal distribution. 2. A sheet with the question question “Do you have any present developme devel opmental ntal conce concerns rns rega regarding rding your chil child d (yes (yes// no)?” If answering “yes,” the caregiver chose from a checklist to specify if this concern related to motor (gross and/or fine), language, social, or cognitive domains or to   1 domain; the caregiver was provided space to provide any additional comments. 3. A simp simple le demog demographi raphicc quest questionna ionnaire ire reque requestin sting g information on employment, education and income. 4. A consent form for participati participation. on. 5. A Likert-type questionnaire questionnaire regarding the ease of use of the questionnaire they had completed. Completed questionnaires were scored by a research assistant (D.R.) according to established procedures for each eac h me measu asure. re. The num number ber of inc incorr orrect ectly ly com comple pleted ted questionnaires for each measure was noted. Those children who fail failed ed the paren parent-com t-complet pleted ed ques question tionnaire naire (2 SDs on   1 doma domain) in) underwent BDI test testing ing (the

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gold standard for the purpose of this study), which was administ admi nistered ered appr approxim oximatel ately y 3 mont months hs afte afterr the init initial ial screen by an experienced pediatric occupational therapist who was blinded to both the screening instruments used use d and the res result ultss obt obtain ained. ed. The 3-m 3-mont onth h int interv erval al used in this study represents the 3-month referral wait typically experienced by families awaiting a more thorough developmental assessment, and at 21 months of age, the child falls in the middle of the 18- to 23-month

were performed by using SPSS 12.0 (SPSS Inc, Chicago, IL), and all data entered were verified for errors.


BDI  The BDI,28 a psychometrically sound developmental assessme ses sment nt for children children from bir birth th to 8 yea years, rs, is wid widely ely used in studies evaluating developmental delay and in US-ma US -manda ndated ted ear early ly int interv ervent ention ion pro progra grams. ms. The BDI gathers information, via a structured test format, to include interviews with the caregiver and direct observation of the child. Items are assigned an age level at which 75% of the norming population was able to perform. It is composed of 341 items that evaluate 5 different domains: main s: perso personal/ nal/soci social, al, adapt adaptive, ive, moto motor, r, comm communic unicaation,, and cogni tion cognitive tive,, with each doma domain in furth further er subd subdiivided vi ded int into o sub subdom domain ains. s. Ite Items ms are sco scored red as typ typic icall ally y signifyi sign ifying ng full fully y devel developed oped skil skills, ls, some sometime timess signi signifying fying emerging abilities, or rarely signifying absent skills and are ar e sc scor ored ed at va valu lues es of 2, 1, or 0, re resp spec ecti tive vely ly.. Th The e authors of the test recommend the test for many functions including general screening, detailed assessment of children who have previously been identified as having

marking scheme of the BDI, givi marking giving ng the most accu accurate rate estimati esti mation on of his or her real abil abilitie itiess accor according ding to the parameters of this measure. To inv invest estiga igate te fal falsese-neg negati ative ve est estima imates tes,, an equ equal al number num ber of chi childr ldren en sco scorin ring g wit within hin the nor norms ms of the screening questionnaires were selected as controls and also als o und underw erwent ent BDI tes testin ting. g. Con Contro trols ls wer were e the nex nextt chi child ld of the same gender participating in the study who had scored normally on the parent-completed screening instrument stru ment.. A fals false-neg e-negativ ative e esti estimate mate for this approach was also obtained in a likewise manner. In this fashion, the control group was selected in an unbiased manner. Because of constraints posed by funding and time available from the occupational therapist, only 101 children underwent BDI testing, after which ongoing study recruitment was terminated. Although this is not as favorable abl e as tes testin ting g all the par partic ticipa ipants nts with the BDI, 101 complete comp leted d BDIs represents represents a larg large e enoug enough h samp sample le to be a good indicator of the accuracy of the ASQ and CDI. The pediatrician who provided care to the child was  blinded to the actual questionnaire used and the results obtained but was asked as part of the study to answer a question identical to the one given to the parent regarding any possible concerns about the child’s development. This question was answered at the time of the subject’s visi vi sitt by si simp mply ly ch chec ecki king ng th the e ap appr prop opri riat ate e re resp spon onse se  box(es). The results of this simple questionnaire were used in the analysis to establish what role the pediatrician could play in influencing the accuracy of the ASQ

 ASQ The AS ASQs Qs29 are 19 pare parent-r nt-report eport quest questionna ionnaires ires that span the age range of 4 to 60 months. Questionnaire points include 4, 6, 8, 10, 12, 14, 16, 18, 20, 22, 24, 27, 30,, 33 30 33,, 36 36,, 42 42,, 48 48,, 54 54,, an and d 60 mo mont nths hs of ag age. e. Ea Each ch questionnaire is composed of 3 sections: a brief set of demographic items; 30 questions about the infant’s or child’ chi ld’ss dev develo elopme pment nt ass assess essing ing 5 dif differ ferent ent dom domain ainss equally (communication, gross motor, fine motor, prob-

and CDI. Parents who had agreed over the telephone to participate ipa te but did not ret return urn the que questi stionn onnair aire e wer were e con con-tacted by telephone and reminded to return the questionnaire. If the questionnaire had not been returned 3 months after it was due, the parents were sent a demographic questionnaire to assess whether any parameters different diffe rentiate iated d thes these e pare parents nts from thos those e who actua actually lly complete comp leted d the quest questionna ionnaire. ire. Families Families with whom we had no personal contact by telephone were not included in the study. Throughout this study, data obtained were stored and manag ma naged ed on sec secure ure com comput puters ers wit within hin the Mon Montre treal al Children’s Hospital Division of Pediatric Neurology with separa sep aratio tion n of sub subjec jectt ide identi ntifier fier var variab iables les.. Eth Ethica icall approval for conduct of the study was obtained from the Montreal Children’s Hospital–McGill University Health Centre Research Ethics Board. Data storage and analysis

lem-solving, lem-solv ing, and pers personal/ onal/soci social); al); and 7 open open-ende -ended d questions eliciting parental concerns. The choice of responses for each item is “yes,” “sometimes,” or “not yet,” which are scored as 10, 5, or 0, respectively. The test is graded grad ed acco accordin rding g to the domain test tested ed and comp compared ared with an empirically derived screening cutoff score and takes 10 to 15 minutes to complete and 5 minutes to score. Although the ASQ states that the questionnaire can  be used in-clinic, the ASQ is meant for mail-out purposes to be completed at home at the specific age intervals listed above. Because of this, the instructions ask the parents to attempt every activity with their child and, if the child is noncompliant, to try at a later time. Unfortunate tun ately, ly, this is not feasible feasible in the context context of a bus busy y waiting wait ing room room.. Thus, these inst instruct ructions ions were abri abridged, dged, and parents were asked to base their responses responses on thei theirr observations and previous experiences with their child.

developmental delay, or identifying strengths and weaknesses of normal or developmentally impaired children. The manual states that the assessment may take up to 2 hours to administer with children 3 years of age.

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The ASQ manual does state that completion of the question ti onna nair ire e in th the e wa wait itin ing g ro room om is th the e le leas astt ac accu cura rate te method, because the parent has a limited time to complete the activities, and the appointment might not fall at an age at which the best results are achieved.

CDI  The CDI30 is useful for testing children whose ages range from birth to 6 years. It consists of 300 items: 270 items composed of yes/no statements about the child’s development opm ent and 30 it items ems tha thatt eva evalua luate te var variou iouss pot potent ential ial sensory, physical, motor, language, and behavioral problems. These items are grouped into 8 subscales: social, self-help, gross motor, fine motor, expressive language, compre com prehen hensio sion, n, let letter ters, s, and num number bers. s. Eac Each h sca scale le is scored sco red by tal tallyi lying ng the “yes” “yes” ans answer wers; s; a chi child ld who receives a score that is 1.5 SDs below the mean is graded as  borderline, whereas a child scoring   2 SDs below the mean is grad graded ed as delay delayed. ed. The pare parent nt needs 10 to 15 minutes to complete this screening instrument and   5 minutes to score it. The CDI was ref reform ormatt atted ed for the pur purpos poses es of thi thiss study. stu dy. In its origina originall for form, m, the CDI is ans answer wered ed on a separa sep arate te ma marki rking ng she sheet et suc such h tha thatt the que questi stions ons are found in one booklet and the answers are written in another. To make the process easier for parents, the CDI was retyped and presented in a format such that the parent can immediately read the question and then answer the item in the same booklet. RESULTS

Descriptive Data Descriptive Over the recruitment period we received the names of 532 pos possib sible le sub subjec jects ts fro from m the database database at the CCC.

From this list, 84 children were excluded because (1) the children were no longer being followed at the clinic (60 children), (2) the children had moved (12 children), (3) the parents did not speak or read English (8 children), (4) the children had an established developmental delay (2 chi childr ldren) en),, (5) the stu study dy ter termi minat nated ed bef before ore the 18month appointment had been booked (1 child), or (6) the child had passed away (1 child). Of the 448 remaining children, we were unable to contact by telephone 92 of them; therefore, they also were not included in the study. We successfully contacted the remaining 356 children, and of these, 317 families (90%) agreed to participate over the telephone and 39 families (10%) refused. Finally, of the 317 who had agreed over the telephone to participate at the forthcoming clinic visit, 5 returned the questi que stionn onnair aire e inc incomp omplet lete e and 64 did not ret return urn the questionnaire at all despite regular subsequent prompting as des descri cribed bed.. Tho Those se who had ori origin ginall ally y agr agreed eed to participate over the telephone but never returned the question ques tionnaire naire were labe labeled led “fals “false e part particip icipants ants.” .” Thus, the total number that completed the questionnaire was 248.. Tab 248 Table le 1 sho shows ws the demogra demographi phicc dat data a of the these se 3 groups: true participants, false participants, and refusals. The average age of the children who participated in the study was 18.4     0.64 months. The data for gestational ages and gender of the children are summarized in Table 2.

Performan Perfor mance ce on Scr Screen eening ing Too Tools ls Of the 134 ASQ ASQss tha thatt wer were e ret return urned ed com comple pleted ted,, 53 (40%) children failed at least one domain. The domain that the infant failed most often on the questionnaire was communication (46), followed by the problem solving, (7) gross motor, (4) social, (4) and fine motor (3)

TABLE 1   Demo Demograph graphic ic Data for 3 Popu Populatio lations ns Characteristics

True Participants, n

Responding parent Mother Father Other Education, last year of school completed, mother/father High school incomplete High school CEGEP/college University Graduate school Working mother   Yes Combined income for the household, $   0–19 000 20 000–39 999 40 000–59 999 60 000–79 999 80 000

False Participants,


223 (90) 19 (8) 6 (2)   N   243 3 (1 (1)/2(1) 18 (7)/28 (12) 80 (33)/76 (32) 116 (47)/102 (42) 26 (11)/33 (14) N   248 159 (64) N   228 6 (3) 14 (7) 23 (10) 45 (19) 140 (61)






NA NA NA NA N   17 2 (1 (12)/0 (0 (0) 3 (18)/2 (12) 4 (24)/3 (18) 7 (4 (41)/11 (6 (65) 1 (6)/1 (6) N   17 10 (59) N   12 0 (0) 1 (8) 2 (17)





NA NA NA N   23 1 (4)/1 (4) 3 (13)/3 (13) 4 (17)/8 (35) 10 (44)/10 (44) 5 (22)/1 (4) N   23 13 (57) N   21 2 (10) 0 (0) 8 (37)

1 (8) 8 (67)

2 (10) 9 (43)

 The 3 populations were true participants, false participants (those who agreed to participate over the telephone but never returned the questionn aire), and refusals. refu sals. NA indic ates not appli cable; CEGEP, CEGEP , colle`ge d’enseignemen d’ enseignemen t ge´ne ´ne´ra l et professionnel. professio nnel.


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TABLE 2   Desc Descripti riptive ve Data for 3 Popu Population lationss True Participants

False Participants


ASQ and the CDI being negligible. Table 3 summarizes these data.

Psychometri Psych ometricc Value Valuess

Child gender,  n  (%) Male Female Age Mean  SD, mo Range, mo Gestational age, mean  SD, wk

132 (53) 116 (47)

34 (53) 30 (47) 30

22 (56) 17 (44)

18.4  .64 17.03–20.47 38.6  2.0

NA NA 39.0  2.7

NA NA 38.5  2.5

Preterm (36 wk gestation),  n  (%)  Term (36 wk gestation),  n (%)

20 (8) 218 (9 (92)

3 (5) 56 (9 (95)

2 (6) 33 (9 (94)

NA indicates not applicable.

domains. Of the 53 children who failed the screening tool,, 41 cont tool continued inued study parti participa cipation tion and under underwent went BDI assessment (ie, 12 dropped out). Conversely, of the 114 children who were assessed by the CDI, 11 failed (failure rate: 9%). Expressive language was again the most likely domain to be failed (5), followed by the gross motor, (5) fine motor, (1) self-help (1), and language comprehension (1 failure) domains. Of the 11 children who failed the CDI, 8 continued study parti par ticip cipati ation on and und undert ertook ook BDI ass assess essme ment nt (ie (ie,, 3 dropped out). In total, 101 BDI assessments were performed. Twenty-nine children received a failing score. Similarly, the domain that was failed most often was communication (17), followed by the gross motor (11), cognition (6), personalpers onal-soci social al (3), and adapt adaptive ive (1) doma domains. ins. Of the 101 BDIs performed, 49 were children who had previously failed one of the questionnaires, whereas 52 infants were control subjects. Of the 101 BDI tests performed, 41 failed ASQs and 8 failed CDIs were assessed with the BDI, and the rema remainin ining g 52 BDI asse assessme ssments nts were performed on control subjects.

Ease of Quest Questionna ionnaire ire Compl Completion etion Closer examination was given to the rates of completion and the parents’ opinion of the questionnaires to assess feasibility and ease of use. Overall, the CDI was more likely to be either returned late (27% for CDI vs 22% for ASQ) or not returned at all (23% for CDI vs 17% for ASQ). This difference was to be expected, because the CDI contains more items to answer and, thus, is more labor intensive. Overall, 81% of the ASQs were returned completed, and 75% of the CDIs were returned completed. The Like Likert-t rt-type ype questionnair questionnaire e was used to asse assess ss the parent’s opinion of the questionnaire questionnaires. s. The options (very easy,, easy easy easy,, neut neutral, ral, difficult, difficult, and very diffic difficult) ult) were given give n the nume numerica ricall valu values es 1 throu through gh 5, resp respecti ectively vely.. The ASQ received a mean value of 1.5      0.6, whereas the CDI received a mean value of 1.6  0.7. Therefore, the majority of the parents ranked the questionnaires as either very easy or easy, with the difference between the

Psychometric Values of the Screening Questionnaires Using the BDI as the gold standard, we assessed the ASQ and the CDI for their psychometric properties: sensitivity, spec specifici ificity, ty, posit positive ive predi predictiv ctive e valu value, e, and nega negative tive predictive value. Not one of these questionnaires proved to be an ideal screening instrument. The ASQ had moderate sensitivity (0.67) but poor specificity (0.39). Conversely, the CDI had poor sensitivity (0.50) but excellent specificity (0.86). Table 4 summarizes the predictive values for the ASQ, CDI, and pediatrician’s questionnaire. Psychometric Values of Incorporating the Pediatrician’s Opinion With the Parent-Completed Questionnaires (ASQ and CDI) The physician’s opinion was incorporated with the parent-completed questionnaires to determine if this could  be used as a potential modifier. As a result, those children (1) who had failed the parent-completed questionnaire and (2) for whom concern was listed by the pediatrician were grouped into a new category as those who had failed both screening instruments. Likewise, those (1) who had passed the parent-completed questionnaire and (2) for whom no concern was listed by the pedia-

TABLE 3   Asse Assessme ssment nt of the Ques Questionn tionnaires aires’’ Feas Feasibili ibility ty ASQ   No. of questionnaires distributed No. (%) completed No. returned late No. (%) of questionnaires not returned No. (%) of questionnaires incomplete Parent’s opinion of questionnaire ( N   1; mean: 1.5  .6), n  (%) Very easy Easy Neutral Difficult Very difficult CDI   N  No. of questionnaires distributed No. (%) completed No. returned late No. (%) of questionnaires not returned No. (%) of questionnaires incomplete Parent’s opinion of questionnaire ( N   112; mean: 1.6  .7), n  (%) Very easy Easy Neutral Difficult Very difficult

183 165 134 (81) 37 28 (1 (17) 3 (2 (2)

75 (57) 50 (38) 6 (5) 1 (1) 0 171 152 114 (75) 41 36 (2 (23) 2 (2 (2) 54 (48) 49 (44) 8 (7) 1 (1) 0

Completion rate and the parental ranking of the ASQ and the CDI. (To collect data on the parent’s opinion of the questionnaire, the question “Did you find this questionnaire easy to complete? Place a check mark in the box that corresponds to your answer” was asked. The number and types of responses are listed.)

PEDIATRICS Volume 118, Number 4, October 2006

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TABLE 4   Psychomet Psychometric ric Value Valuess (Sen (Sensitiv sitivity, ity, Spec Specificit ificity, y, Posi Positive tive Predictiv Pred ictive e Value Value,, and Nega Negative tive Pred Predictiv ictive e Value Value)) for the ASQ AS Q an and d CD CDII BDI


Fai aill

Pas asss

ASQ Fail Pass  Total

14 7 21

27 17 44

41 24 65

CDFail I




4 8

24 28

28 36

Pass  Total









Feasibility The participation rate was encouraging. The majority of 0.50




PV indicates positive predictive value; PV , negative predictive value.

tricia trician n wer were e gro groupe uped d int into o ano anothe therr cat catego egory ry as tho those se scoring normally on both tests. Children who did not fit the criteri criteria a for either either of the these se 2 gro groups ups (ie, they had passed pass ed the quest questionna ionnaire ire and faile failed d the pedi pediatri atrician cian’s ’s questionnaire or vice versa) were not included in the calculat calc ulation. ion. The 2 group groupss (one representing representing children who had pass passed ed both questionnai questionnaires res and the other representin rese nting g the chil children dren who had faile failed d both questionquestionnaires) were compared with the results of the BDI to obtain predictive values. Incorporating the pediatrician’s opinio opi nion n wit with h the res result ultss of the que questi stionn onnair aire e did not improve the predictive accuracy of either questionnaire substantially. Table 5 summarizes the results of comparing the combined result of the pediatrician’s opinion and the screening tool with the BDI. To be com compre prehen hensiv sive, e, dif differ ferent ent ana analy lyses ses,, suc such h as moving the BDI cutoff to 1 SD below the mean, removing failed failed com commun munica icatio tion n dom domain ainss on the ASQ as a potential confounder, and redefining a failing score for the ASQ as 2 SDs below mean on   2 domains, were attempted. None of these posthoc manipulations had a  beneficial effect on predictive values, and the results are not presented herein. DISCUSSION

To determine the usefulness of parent-completed questionnaires in a practical setting, 3 issues were examined: TABLE 5   Psycho Psychomet metric ric Val ValuesWith uesWith theAddi theAdditio tion n of the Pedia Pe diatri tricia cian’sOpini n’sOpinion on for theASQ andCDI BDI

Ped and ASQ Fail Pass  Total Ped and CDI Fail Pass  Total


Fail Fa il

Pass Pa ss

9 6 15

6 16 16 22













1155 22 36




3 5

23 23 26

26 31

Ped indicates pediatrician’s opinion; PV , positive predictive value; PV , negative predictive value.


  RYDZ et al

(1) the feasibility of using parent-completed questionnaires in the waiting room; (2) the role of the pediatrician’s impression in the screening process; and (3) the accuracy of 2 parent-completed screening measures (the ASQ and CDI). Each of these issues can be addressed separately in the context of our results.

parents (ASQ: 95%; CDI: 92%) found the questionnaire either eith er easy or very easy to comp complete lete.. The completion completion rate was also high for both questionnai questionnaires. res. Observed Observed completion was lower for the CDI (75%) than the ASQ (81%), which was to be expected because the CDI is longer and more detailed. More than 75% of the contacted population was assessed with a standardized screening tool, representing an improved rate over what is currently being achieved with practitioner-administered tests. (Only 23% of primary care physicians are regularly using a standardized screening tool.)13 Most important to note is that these question ques tionnaire nairess were administere administered d in the clin clinic ic at the time of the patient’s appointment and did not need the assistance of either office staff or the physician. Because these tests are also cost-effective, the 3 most frequently mentioned complaints associated with a screening test administered by health care professionals (lack of time, lack of staff, and cost) were overcome. It is also important to note that the overall completion rate may have been decreased by the additional burden placed on the parent from the 4 additional forms placed in the dossier to be completed. Nonetheless, simple endeavors can be conceptualized to increase the completion rate with active endorsement and encouragement  by the primary care physician and clinic staff. Thus, it seemss feasi seem feasible ble for paren parents ts to comp complete lete developmental developmental screen scr eening ing que questi stionn onnair aires es in the wai waiti ting ng roo room m whi while le waiting to be seen by their child’s physician.

Pediatric Pediat rician ian’s ’s Opi Opinio nion n as a Pote Potenti ntial al Mod Modifie ifierr The ped pediat iatric rician ian’s ’s opi opinio nion n did not im impro prove ve the pre predic dicti tive ve values valu es of the quest questionn ionnaires aires used. The pedi pediatri atrician cian’s ’s opinion had good specificity but poor sensitivity; thus, the use of their clinical judgment tended to underidentify deve developm lopmenta entall impa impairme irments. nts. Inde Indeed, ed, the pedia pediatritrician’s questionnaire and the CDI had very similar properties. It is dis discon concer certin ting g tha thatt com combin bining ing the res result ultss of a standardized screening tool with the pediatrician’s opinion could not produce enhanced and ultimately acceptable sensitivity and specificity values. Perhaps this indicates the lack of proper health care training in regards to childhood development. Indeed, the literature has emphasized that there is a gap between the knowledge and skills required in providing developmental services and the limited training that many clinicians receive in this

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area. This is a problem that seems to have persisted over decade dec ades. s. In 197 1979 9 Sho Shonko nkoff ff et al lam lament ented ed tha thatt “fo “four ur fifths of physicians . . . viewed their formal training in thiss are thi area a as ina inadeq dequat uate. e. . . . [A [A]lm ]lmost ost 2/3 did not fee feell that practical experience was an adequate substitute for formal form al trai training ning in deve developm lopmental ental asse assessme ssment nt skil skills.” ls.”3 This reality still persists   20 years later. A 2000 study performed by the AAP found that 64% of primary care physicians reported inadequate training in developmen-

can incorporate this particular strength within a screening procedure. Moreover, we found that a “one-shot” screenin scre ening g prot protocol ocol might not be suffic sufficient ient to iden identify tify 31 developmental delay accurately. Indeed, the AAP and other researchers have come to the same conclusion. For example, Darrah et al32 concluded that screening should involve multiple time points and multiple domains before referrals are made. A similar statement was issued  by the AAP Committee on Children With Disabilit Disabilities. ies.2


tal assessment. As a result, an improvement in screening acc accura uracy cy doe doess not jus justt nec necess essita itate te mo more re acc accura urate te developmental screening tests but also a joint effort from different facets of the professional, public, and research communit comm unity y advo advocati cating ng for more more-thor -thorough ough trai training ning in childhood development and impairments.

This study has several limitations. First, the ASQ was used as an in-clinic questionnaire; although the manual states that it can be used in this fashion, this screening instrume inst rument nt was orig original inally ly cons construc tructed ted as a take take-hom -home e questionnaire so that the parent could attempt to address dre ss the items items ove overr sev severa erall day dayss wit with h the child. child. By limiting the time to 15 minutes, with the parent relying on pas pastt exp experi erienc ences es to ans answer wer the que questi stions ons,, the accuracy of the instrument could have been somewhat compromised. Second, only 36 subjects underwent both the CDI and BDI. This limits the ability to evaluate the accuracy of the CDI, in particular with respect to sensitivit ti vity, y, bec becaus ause e the there re wer were e few sub subjec jects ts who act actual ually ly failed the CDI. Thus, a larger sample size may be needed

Accura Accu racy cy of th the e AS ASQ Q an and d CD CDII The psychometric properties of the ASQ and CDI were surprising, because several studies for both the ASQ and the CDI have advocated their accuracy in screening infants for developmental delay. A factor that may have reduced the accuracy of the ASQ and CDI in our study was the time difference between screening the child and performing the full assessment. Unlike other projects in which the gold standard was applied immediately after the screening instrument, our study imposed a 3-month waiting period representing the “real-life” delay typically experienc exper ienced ed betw between een scre screenin ening g and refer referral. ral. Prev Previous ious studies have demonstrated that 3 months represents a time interval interval with within in whic which h an infan infant’s t’s devel developme opmental ntal 32 status sta tus may cha change nge.. Da Darra rrah h et al fou found nd tha that, t, whe when n assessing fine motor skills, gross motor skills, and communication, the majority of children tested serially had 40 to 60 percentile fluctuations in performance. Changes of th this is ma magn gnit itud ude e to took ok pl plac ace e at le leas astt on once ce wi with thin in 5 testing points (ie, 9, 11, 13, 16, and 21 months of age). Cons Co nseq eque uent ntly ly,, a wa wait it of 3 mo mont nths hs co coul uld d re resu sult lt in a different developmental estimate. Because of the insta-

 before drawing clearer conclusions regarding the CDI’s performance as a screening tool. Finally, the community in which we were conducting the study was predominantly nant ly middle class with a high school education education and some postsecondary attendance. Our ability to generalize the results to the general population, particularly to disadvantaged populations, is limited. This study does lay the foundation for future studies on scr screen eening ing.. Fir First, st, and mos mostt imp import ortant ant,, the there re is the possibility of assessing the benefits of 2 serial screening testss at different points in tim test time. e. Becau Because se the developmental trajectory, especially at younger ages, is subject to lar large ge var variab iabil ility ity,, a sec second ond scr screen eening ing eff effort ort (ei (eithe therr completed in-clinic or sent by mail) has the potential to

 bility and discontinuity of a child’s development, what might have been identified as a transient weakness at 18 months mon ths cou could ld hav have e res resolv olved ed it itsel selff by 21 mon months ths.. The developmental domain of communication also seems to  be too variable at the 18-month age to provide a reliable indicato indi catorr betw between een norm normalit ality y and abno abnormal rmality. ity. At this age, the “nor “normal” mal” differences differences in the actual times that communication milestones are reached by children are too variable,2 which demonstrates the importance of the time of actual assessment and the advantage of regular, ongoing multiple screening efforts. Overall, Over all, neit neither her test provi provided ded acce acceptab ptable le scre screening ening properties at 18 months of age; therefore, a screening protocol suitable to the setting of a clinic cannot consist solely of either instrument applied at one point in time. However, our results demonstrate that the CDI identified children with normal development with high accuracy (ie, high negative predictive value). Future studies

improve the the accuracy of the initial identification process. To decrease amount of time used to score the questionnair tion naire, e, appli applicati cation on of comp computer uter software coul could d be implemen impl emented ted (ie, ques questionn tionnaire aire comp complete leted d and scor scored ed on a hand-held device). This would reinforce the practicality of these types of screening instruments by decreasing the effort required from office staff and personnel. Finally, a study conducted on a more representative popula pop ulatio tion n inc incorp orpora oratin ting g a lar larger ger sam sample ple siz size e wou would ld clarify issues of applicability, feasibility, and yield. With additional research, the knowledge gap between available screening tests and developing a practical protocol for their application can be bridged. Such efforts would have consi considerab derable le clin clinical ical impl implicat ications ions with resp respect ect to systematically enhancing the capability for earlier identification of children with developmental delays, which should shou ld exped expedite ite inte interven rvention tion and, theor theoretic etically ally,, opti opti-mize eventual outcome. PEDIATRICS Volume 118, Number 4, October 2006

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Dr Shevell is grateful for the support of the Montreal Children’s Hospital foundation during the writing of this manus ma nuscri cript. pt. Thi Thiss pro projec jectt was sup suppor ported ted by a cl clini inical cal resear res earch ch pro projec jectt gra grant nt fro from m the Mon Montre treal al Chi Childr ldren’ en’ss Hospital Research Institute.


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Screening for Developmental Delay in the Setting of a Community Pediatric Clinic: A Prospective Assessment of Parent-Report Questionnaires David Rydz, Myriam Srour, Maryam Oskoui, Nancy Marget, Mitchell Shiller, Rena Birnbaum, Annette Majnemer and Michael I. Shevell Pediatrics 2006;118;e1178 DOI: 10.1542/peds.2006-0466 Updated Information & Services

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PEDIATRICS is the official journal of the American Academy of Pediatrics. A monthly publication, it has been published continuously since 1948. PEDIATRICS is owned, published, and trademarked by the American Academy of Pediatrics, 141 Northwest Point Boulevard, Elk  Grove Village, Illinois, 60007. Copyright © 2006 by the American Academy of Pediatrics. All rights reserved. Print ISSN: 0031-4005. Online ISSN: 1098-4275.

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