Developing preschool surveillance tools for adaptive functioning: Lessons for neuro-oncology

e u r o p e a n j o u r n a l o f p a e d i a t r i c n e u r o l o g y 1 4 ( 2 0 1 0 ) 3 6 8e3 7 9

Official Journal of the European Paediatric Neurology Society

Review article

Developing preschool surveillance tools for adaptivefunctioning: Lessons for neuro-oncology5

Michael E. Msall*

University of Chicago, Pritzker School of Medicine, Kennedy Research Center on Intellectual and Developmental Disabilities,

Section of Developmental and Behavioral Pediatrics, Comer Children’s Hospital at the University of Chicago,

LaRabida Children’s Hospital, USA

a r t i c l e i n f o

Article history:

Received 15 March 2010

Accepted 20 March 2010

Keywords:

Neuro-oncology

Outcomes

Functional skills

Adaptive skills

Neurodevelopmental

Preschool

5 Presented in part at the European Society* University of Chicago Section of Develop

USA. Tel.: þ1 773 702 3095; fax: þ1 773 702 0E-mail address: [email protected]

1090-3798/$ e see front matter ª 2010 Europdoi:10.1016/j.ejpn.2010.03.010

a b s t r a c t

Advances in neuro-oncology have resulted in dramatic increases in the survival of children

with pediatric brain tumors, including those with medulloblastoma, the most common

early childhood central nervous system (CNS) malignancy. Because of this increased

survival, there is heightened awareness and recognition that an understanding of the

impact of treatments and interventions on children’s development, learning, and adaptive

behaviors is essential for optimization of long-term outcomes. One of the major reasons for

studying these outcomes is that the developing nervous system has its unique vulnera-

bilities with respect to the primary tumor, its complications (hydrocephalus, increased

intracranial pressure, hazards of tumor resection), and the late effects of treatments on

children’s developmental, learning, and adaptive status. In addition, very young children

are especially vulnerable to the toxic effects of radiation and other therapies, highlighting

the importance of measuring emerging communication, coordination, and social adaptive

skills. Lastly, there is increased concern about the long-term consequences of chemo-

therapy on specific neural populations and neural connectivity that affect memory,

learning, and executive function.

The purpose of this article is to review ways of conceptualizing CNS adaptive functioning

in children diagnosed with brain tumors in early childhood. The international classification

of functioning (ICF) model will be used to describe the spectrum of health and develop-

mental outcomes of child neurodisability. An integrated strategy of surveillance for motor,

communicative, and adaptive skills that can be linked to neuropsychological assessments

will also be highlighted. This health, development, and functional surveillance framework

will help us better evaluate how our management impacts on child and family well-being

and how our interventions lessen severe multiple motor, communicative, and neuro-

behavioral morbidities.

ª 2010 European Paediatric Neurology Society. Published by Elsevier Ltd. All rights

reserved.

of Pediatric Neurology, Harrogate UK October 2,2010.mental and Behavioral Pediatrics, 950 East, 61st Street, SSC Room 207, Chicago, IL 60637,208.go.eduean Paediatric Neurology Society. Published by Elsevier Ltd. All rights reserved.

mailto:[email protected]

e u r o p e a n j o u r n a l o f p a e d i a t r i c n e u r o l o g y 1 4 ( 2 0 1 0 ) 3 6 8e3 7 9 369

Contents

1. Introduction . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 3691.1. The International Classification of Functioning (ICF) Model . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 3691.2. Measuring developmental status in children . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 3711.3. Assessing adaptive and functional skills in preschool children . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 3711.4. Developmental aspects of adaptive behavior across preschool, middle childhood, and young adulthood . . . . . 3741.5. Developmental and behavioral surveillance scales . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 374

1.5.1. Ages and stages questionnaire (ASQ) . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 3741.6. Motor and prelinguistic milestone scales . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 374

1.6.1. Brief infant toddler social emotional assessment (BITSEA) . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 3751.6.2. Strengths and difficulties questionnaire (SDQ) . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 375

1.7. Application of developmental and functional surveillance to children with medulloblastoma . . . . . . . . . . . . . . . 3751.8. What are the lessons? . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 3761.9. Cryosurgery for retinopathy of prematurity (CRYO-ROP) . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 377Acknowledgement . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 377References . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 377

1. Introduction

Until two decades ago, there were few long-term survivors of

childhood central nervous system (CNS) malignancies.1 The

current incidence of CNS tumors exceeds 3 per 100,000 per

year with 33% of these occurring in the preschool years.

Medulloblastoma represents the most common malignant

brain tumor in early childhood, accounting for 20% of all

primary CNS tumors in this age group.2 Its location in the

posterior fossa requires expertise in microsurgery, attention

to complications of hydrocephalus, recognition of the neural

circuitry of the cerebellum, and awareness of the cerebellar

mutism syndrome.3

A child historically has been considered to be standard risk

if they were older than 3 years and if residual tumor was less

than 1.5 cm2 following neurosurgery with no evidence of

metastatic disease. Although a recent review highlighted a 5-

year survival rate of 85% for those with standard risk, this

decreased to 70% for those considered high risk.4 However,

there have been disproportionately high rates of long-term

physical, cognitive, academic, behavioral, and social limita-

tions among the survivors.5e7 In particular, impairments of

memory, executive functioning, and information processing

speed are frequently found in school-age survivors and may

represent the neuropsychological basis of many of the

ongoing difficulties that children experience in cognitive skills

and academic achievement in reading and mathematics, as

well as adult health-related quality of life.8e12 The complex

factors underlying late effects have been the focus of recent

multicenter efforts.13e19 It is essential, especially in preschool

children, to have surveillance tools that can be used on several

occasions and longitudinally in order to better understand the

early factors contributing to long-term disability.

1.1. The International Classification of Functioning (ICF)Model

The International Classification of Functioning (ICF)

Disability and Health model conceptualizes health and

enablement outcomes in four domains: Body Structure,

Body Function, Activities, and Participation.20 This is

a multi-dimensional, multi-directional outcome model that

can help describe the impact of medical interventions on

organ structure, physiology, functioning, and participation

within family, school, and community roles. Body structures

are anatomical parts of the body, such as organs and limbs,

as well as structures of the nervous, sensory, endocrine, and

musculoskeletal systems. Body function refers to the phys-

iological functions of body systems (e.g., breathing, growth,

and digestion), as well as psychological functions (e.g.,

regulating behavior, attending, learning, problem solving,

remembering, and information processing). Activities are

tasks, including communicating, walking, dressing, toilet-

ing, bathing, reading, calculating, writing, interacting with

peers, and solving specific problems. Participation means

involvement in family and community life during and after

school, such as chores, family events, friendships, as well as

recreational, religious, civic, and club activities (e.g., scouts,

sports). The ICF Model also includes contextual factors in

a child’s life and highlights environmental and personal

factors. Environmental facilitators include factors such as

mentors, positive social attitudes, legal protections, and

barrier-free architecture. Environmental barriers include

negative attitudes of others, non-accessible transportation,

inadequate health insurance, and discriminatory practices.

Personal factors include age, gender, interests, and sense of

self-efficacy. Table 1 shows how the ICF model can be

applied to children with medulloblastoma at ages 2, 3, and 4

years.

The ICF framework illustrates how different components

of everyday life influence one another in non-linear ways. In

this model, one can highlight how the multimodal manage-

ment for children with medulloblastoma requires integration

of medical, educational, and community supports. Fig. 1

applies the ICF model to a 7-year-old who has survived

medulloblastoma for five years and has evidence of white

matter injury on cranial magnetic resonance imaging (MRI).

Among long-term survivors, this model increases our

emphasis on understanding developmental processes

Table 1 e International Classification of Functioning (ICF) Model: preschool children with medulloblastoma.

Dimension Definition Boy, 2 years Girl, 3 years Boy, 4 years

Body structures & body

functions

Organ structure/

function

Unsteady gait Central hypotonia,

speech delays, 40 db

hearing loss

Growth delays,

hyperactivity,

impulsivity, clumsy

Activity (functional)

strengths

Ability to perform

essential activities: feed,

dress, toilet, walk, talk

Walks holding onto rail,

drinks with sippy cups,

likes to pretend play

with trucks, says 10

words

Walks, sits on

potty, talks

in phrases unclear

to strangers

Learns songs, plays with

peers outdoors

Activity (functional)

limitations

Difficulty in performing

essential activities

Has difficulty with

crayons and pencils,

not speaking in phrases

Has difficulty running,

speaks more clearly and

is more social with

hearing aides.

Has difficulty with

coordination,

perception, and

attention.

Participation Involvement in

community roles

typical of peers

Plays in parallel with

peers.

Attends Montessori

preschool.

Attends YMCA

swimming lessons.

Participation restrictions Difficulty in assuming

roles typical of peers

Unable to attend

day care because

parents fearful he

will become ill.

Teachers forbid gym

because of fear that

she will fall.

Was expelled from

preschool because of

impulsive behaviors.

Contextual Factors:

Environmental

facilitators

Attitudinal, legal,

policy, and architectural

facilitators

Has home hospital

program with quality

child life, speech and

occupation

therapy services.

Loves songs with

gestures, learns many

things from watching

peers.

Parents know his

strengths and uses

positive behavior

management

interventions.

Contextual Factors:

Environmental barriers

Attitudinal, legal, policy,

and architectural

barriers

On waiting list for early

intervention.

Family lacks strategies

to promote preschool

experiences.

Small peer social skill

groups; family moves

frequently.

Fig. 1 e Application of the International Classification of Functioning (ICF) Model to a 7-year-old boy with medulloblastoma

and white matter injury.


Table

2e

Presc

hoolco

gnitiveandadaptiveass

ess

ments.

Bayley3

scales

Mullensc

ales

ofearly

learn

ing

Battelle

developm

ental

inventory

2

Griffi

thsm

ental

developm

ental

scales2

Adaptivebehavior

ass

essm

entsy

stem

,se

condedition

Vinelandadaptive

beh

aviorsc

ales,

seco

ndedition

Age ra

nge

1e42month

s0e68month

s0e8years

0e8years

0e5years

0e18þ

years

Domains

Motor,

Language,

Cognitive,

Social-emotional,

Adaptive

Gro

ssmotor,

Finemotor,

Expressivelanguage,

Rece

ptivelanguage,

Visualrece

ption

Motor,

Communication,

Cognitive,

Personal-so

cial,

Adaptive

Loco

motor,

Eyeandhandco

ord

ination,

Hearingandsp

eech

,

Perform

ance

Practicalreaso

ning,

Personal-so

cial

Practical

Conce

ptu

al

Social

Motorsk

ills

Communication

Socializa

tion

Dailyliving


underlying motor, communicative, and adaptive skills which

promote independence, social participation, and learning in

middle childhood and adolescence.

1.2. Measuring developmental status in children

Beginning with Arnold Gesell and Edgar Doll, there has been

a strong tradition of examining the developmental trajectories

of children’s motor, communicative, cognitive, social-

emotional, and adaptive skills.21,22 A variety of preschool

assessment tools have been devised for these purposes and

are highlighted in Table 2.23e25

These assessments are multi-dimensional and include

a variety of manipulation, problem solving, communication,

and exploration activities. These tools have been used

among children receiving neonatal intensive care, cardiac

surgery, or genetic metabolic interventions, and during early

childhood to assess and manage early neurodevelopmental

disorders.

However, it is critically important to realize that summary

scores from these assessments cannot be used to make

statements that a child is free of neurodevelopmental

impairments. Specifically, a Bayley 3 Cognitive Standard Score

of 90 at age 2 years does not guarantee that children will

master complex executive function, memory, and academic

tasks at school.26 Similarly, a Bayley 3Motor Standard Score of

65 at age 3 years does not preclude a child running, drawing,

and riding a two wheel bicycle at age 6 years. However, chil-

dren performing in communicative and nonverbal problem

solving domains with scores of 2e2.5 standard deviations

below the mean have higher rates of complex learning chal-

lenges and high probabilities of following trajectories of

ongoing intellectual disability.27

There are two additional problems with these assessment

tools. First, they do not fully capture how a child requires

progressively less one-to-one supervision and thereby make

the transition to greater independence at home and small

group learning in other settings. Second, these assessments

also require the evaluator to be skilled in establishing rapport

and using the appropriate items flexibly to capture children’s

current developmental processes. In order to bridge this gap,

the concept of functional assessment of adaptive skills is

a critical component of assessment of children with evolving

neurodevelopmental disabilities.21e25

1.3. Assessing adaptive and functional skills inpreschool children

Early childhood is a period of dynamic growth, learning, and

acquisition of basic adaptive skills. These criterion referenced

tasks that occur daily include feeding oneself, maintaining

hygiene (by washing hands, brushing teeth, and bathing),

changing a variety of clothes, and controlling bowel and

bladder. Motor daily activities include changing positions,

maintaining postural control, and self mobility indoors and

outdoors, as well as negotiating stairs and uneven surfaces.

Activities during preschool years include learning to under-

stand words, concepts, and directions, communicate basic

needs through sentences and conversations, take turns, and

negotiate social interactions. These self-care, mobility, and

Table 3 e Functional, behavioral, and heath-related quality of life assessments.

WIDEA-FSWarner InitialDevelopmental

Evaluation of Adaptiveand Functional Skills

BITSEABrief Infant ToddlerSocial Emotional

Assessment

PEDI-CAPediatric

Evaluation of DisabilityInventory Caregiver

Assistance

CHQ-PF 28Child

Health QuestionnaireParent Form 28

PedsQL 4Pediatric Qualityof Life Inventory,Fourth Edition

SDQStrengths andDifficulties

Questionniare

Age range 0e24 months 1e3 years 0e8 years For Parents 2e18 years 3e10 years

Domains Self-care

Mobility

Communication

Social skills

Internalizing and

externalizing problems,

dysregulation, and social

competencies

Motor

Self-care

Social skills

Caregiver assistance

Physical

Social emotional

Self-esteem

Health

Bodily pain

Mental health

Behavior

Parental impact

Family activity and

cohesion

Physical, emotional,

social, and school

functioning

Overall stress, emotional problems,

behavioral difficulties, attention

and hyperactive disorders, kind

and helpful, social skills, family

impact.

Concurrent

validity

Capute Scales

Chronological Age

CBCL 1.5e5 Y ASQ

eSEBayley 3

VABS2

Battelle

Wee FIM

VABS I

Disorder severity Distinguishes between

healthy children

and children with

special needs

CBCL

Disability

samples

Children in Early

intervention, or

children

with special health care

needs

Children at risk

because of social,

biomedical,

or community factors.

Children with brain

injury,

cerebral palsy, spinal

cord injury,

and juvenile arthritis

Children

with epilepsy,

cerenbral

palsy,

ADHD, very low birth

weight,

juvenile arthritis, asthma

Cerebral palsy,

asthma,

oncology, or arthritis

Children with and without

developmental, behavioral,

or special health care needs

CBCL¼Child Behavioral Checklist/ASQSE¼Ages and Stages Questionnaires e Social Emotional Scale.

european

journalofpaedia

tric

neurology

14

(2010)368e379

372


communicative skills will become the cornerstone of a child’s

knowledge of routines, consistent problem solving skills, and

progressive independence of adult caregivers.

There is, however, an order in which childrenmaster these

tasks. This timing reflects the interaction of neurological,

developmental, and social experiences. Basic indoor mobility,

self-feeding with fingers, using a spoon, and communicating

in words, sounds, and gestures are present in 18-month-olds

following typical developmental trajectories. By the age of two

and a half years, most children are able to walk a distance of

approximately one neighborhood block outdoors, manage

stairs, communicate in short sentences, remove items of

clothing, and indicate to parents that they need to be changed

when they have been incontinent. By age four years, children

can put on articles of clothing (shoes, socks, jackets, pants),

wash and dry their hands without adult help, tell a story, and

sing songs. Thus, well before kindergarten entry there are

basic indicators of children’s independence in mobility, self-

care, communication, and social skills.

In childhood there are several instruments with multi-

center norms currently available (Table 3).

The Pediatric Evaluation of Disability Inventory (PEDI)

consists of 197 functional skills in mobility, self-care, and

social cognition, as well as a 20-item caregiver assistance and

environmental modification (CA-EM) scale. The PEDI has been

translated into several European languages and has criterion

validity with the Vineland Adaptive Behavior Scale, second

edition (VABS II) and the pediatric functional independence

measure (Wee FIM�). The PEDI-CA/EM involves a 10-min

structured interview. Through this assessment, one is able to

describe a child’s independence in basic activities or the

environmental modifications that support task completion. In

this latter category, alternative assistance modalities of

learning and participation can increase the child’s ability to

access a switch or to understand sequences of information

and indicate a response.

The Functional Independence Measure for Children (Wee

FIM�) is an evaluative measure of basic functional skills that

consists of 18 items encompassing three subscales: self-care,

mobility, andsocial cognition.Thereareeight items for self-care

activitiesofdaily living,five items formobility, andfive items for

social-cognitive functioning. The latter domain includes

understanding verbal and nonverbal communication, use of

language and gestures, social interaction, play, and memory of

routines. The Wee FIM� has been used in children ages 2e7

years old including children with extreme prematurity,

encephalopathy, traumatic brain injury, and genetic disability.

The Vineland Adaptive Behavior Scales, second edition

(VABS II) involves survey interviews for assessing adaptive

behaviors. For children younger than 7 years, the domains

include communication (receptive, expressive, and written

language), daily living skills, (self-care/personal), socialization

(interpersonal relations and play), and motor skills (gross and

fine). The VABS II has been used in children with preschool

developmental disabilities, including autistic spectrum

disorders, cerebral palsy, intellectual disability, and traumatic

brain injury. Used with children older than the chronological

and developmental age of 7 years, the VABS II adaptive

composite consists of 99 communication, 109 daily living,

and 99 socialization items. For children older than 5 years,

there is also a maladaptive behavior domain that includes

internalizing, externalizing, and stereotypical and peculiar

behaviors.

Within the construct of health-related quality of life, there

are explicit indicators of physical, developmental, and

behavioral functioning.28e30 Of these, two deserve comment.

The Child Health Questionnaire (CHQ) measure physical

functioning, role and social limitations, general health

perception, bodily pain, self-esteem, parental impact of time

and emotions, mental health, general behavior, family activ-

ities, family cohesion, and change in health among children

aged five years or more.31 There are three parent-completed

forms: two comprised of 28 items and one of 50 items. In

addition, a 87-item form is available for self reporting by

children 10 years and older. The strength of the CHQ is that it

measures physical, behavioral, and developmental status in

middle childhood and adolescence, as well as a child’s school

and social functioning and family impact. Its use in multi-

center projects is facilitated by inclusion of a uniform parental

interview as well as a checklist for identifying ongoing special

health care need conditions such as anxiety, asthma, inat-

tention, intellectual disability, autism, epilepsy, hearing or

visual impairments, learning problems, speech problems, and

sleep disorders.31

For children under five years of age, the Infant Toddler

Quality of Life Scale (ITQOL) builds on the domains of the

CHQ. It is available in English, Dutch, and Spanish and

consists of 97 items. Ratings are transformed to a scale from

0 (worst health) to 100 (best health).32 Domains include

overall health; amount of limitation in eating, sleeping,

grasping, and playing; satisfaction with physical, motor,

language, and cognitive development; eating, feeding, and

sleeping habits and overall temperament. Additional

domains include bodily pain or discomfort and the extent to

which pain and discomfort interferes with normal activities.

Positive and negative behaviors include frequency of irrita-

bility or upset, fussiness, unresponsiveness, playfulness and

alertness. A key domain includes the child’s perceptions of

current, past and future behavior; current, past, and future

physical health; and perceptions of changes in health over

the past year. Other domains of the ITQOL are parent-

focused concepts about the amount of concern regarding

their child’s physical and emotional well-being, regulatory

behavior, learning abilities, temperament, behavior, and

social skills. Additional domains include limitations experi-

enced by parent due to child’s eating or sleeping habits,

physical and emotional well-being, learning abilities,

temperament, behavior, social skills, and family’s ability to

get along and supporting each other.

The 97-item format of the ITQOL precludes its frequent

use. However, the domains can be used to elicit those

treatment areas impacting on child’s current health, devel-

opmental and behavioral management especially with

respect to pain management, feeding, sleep, and behavioral

adaptability.

The Pediatric Quality of Life Inventory, 4th edition (PedsQL

4�) was designed to measure health-related quality of life in

children.33 For preschoolers, there are parent proxy-reports

for ages 2e5 and ages 5e7. The generic core scale consists of 23

items covering physical functioning (8 items), emotional


functioning (5 items), and school functioning (5 items). This

instrument distinguishes between healthy children and chil-

dren with special needs. It has been translated into multiple

languages, including Spanish. For both pediatric oncology and

pediatric brain tumors, there are disease-specific models

available.34

Lastly, there is the Child Health Impairments, Functioning,

Supports, and Participation (CHI-FSP) survey that was devel-

oped by Hogan and Msall to capture key questions from

childhood population survey methodologies.21 The CHI-FSP

survey has domains that include descriptions of medical

impairments, activity limitations, functional strengths and

limitations, and ratings of physical, mental, and develop-

mental health status.22,35 In addition, this assessment enables

parents to rate their own physical andmental health status. It

is in a two page format that can be quickly administered as

a structured phone interview or as a checklist questionnaire

and does not require separate forms for rating parents and

child well-being and family impact.

1.4. Developmental aspects of adaptive behavior acrosspreschool, middle childhood, and young adulthood

In defining adaptive behavior for situations where there may

be intellectual or cognitive developmental disability, three

dimensions have been proposed: conceptual skills, social

skills, and practical skills.36 Conceptual skills include

communication (expressive and receptive language skills),

functional academics (basic reading, writing, math), self-

direction (management of time, using a schedule), and health

and safety awareness (communicating sickness or injury,

following safety rules). Social skills include interacting and

cooperatingwith others and use of leisure time. Practical skills

include self-care (eating, toileting, dressing, hygiene), home

living (clothing care, food preparation, housekeeping),

community use (traveling within the community, using the

library), health and safety (communicating sickness or injury,

following safety rules), and work (job-related skills). Depend-

ing on the age of the child at the time of the assessment and

the competencies comprising criteria for age appropriate

adaptive behaviors differs. Self-care, communication, social,

safety skills are given more importance in younger children.

For adolescents and adults, the focus shifts to home living,

community use, andwork skills. Although there is not a linear

relationship between these dimensions, children who are

communicative, able to demonstrate functional academic

skills, and independently interact with peers without

disabilities have a better chance of achieving independent

living.

One of the important lessons from developmental

surveillance is the dynamic changes that occur in childhood.

This necessitates the use ofmulti-dimensional tools over time

linked to neuropsychological evaluations at key ages. This can

be achieved with phone-based interviews at pre-defined

intervals using developmental questionnaires that include

motor, communicative, self-care, cognitive and social-

emotional domains and define current adaptive status.

Explicit criteria for consistent task accomplishment are

specified. The components of this system will be described in

the next section.

1.5. Developmental and behavioral surveillance scales

1.5.1. Ages and stages questionnaire (ASQ)The Ages and Stages Questionnaire (ASQ), 3rd edition, was

designed to elicit key indicators of development from parents

of children aged 1 month to 5.5 years.37 In pediatric primary

care, there are specific questionnaires for infancy (2, 4, 6, and 8

months of age), toddler (9, 10, 12, 14, 16, 18, 20, 22, 24, 27, 30,

and 33 months of age), and preschooler (36, 42, 48, 54, and 60

months). Each age group has 30 explicit developmental items

encompassing Gross Motor, Fine Motor, Problem Solving, and

Personal-Social Domain.

The reading level of each questionnaire range from 4th to

6th grade with illustrations and wording that clearly describe

a developmental task. Questionnaires are available in English

and Spanish. Two DVDs are available which encompass

scoring, referral, and materials that can be used during home

visits. Onlinemanagement software is also available for single

or multicenter use, as well as an ASQ Family Access site.

Overall scoring of ASQ has been based on 18,000 question-

naires by parents of children ages 1e66months. The cutoff for

referrals is ASQ score of more than two standard deviations

below thenorm (DQ< 70).Overall sensitivity and specificity for

Battelle Developmental Inventory domain being less than the

standard score of 75 was 83e89% across infants, toddlers, and

preschool questionnaires. Specificity exceeded 85% for ages

2e12 months, 27e36 months, and 42e60 months. Specificity

was 78% at 14e24 months reflecting the dynamic changes in

language and social behaviors occurring in this period.

Age equivalents and developmental quotients have been

set up for each item in each domain. Testeretest reliability

and criterion validity are psychometrically sound. In addition,

activities that support the child’s development are available

as handouts for parents to implement at home.

The strength of ASQ is that it systematically captures the

diversity of children’s developmental competencies at

frequent intervals. The assessment includes identification of

red flags and can be integrated with referrals for family

supports for those with children between 0e3 years and 3e5

years of age. The ASQ has the advantage of updating historical

milestones described by Gesell and colleagues as well as

updating the broad dimensions that are currently required in

early childhood programs. In addition, it has promoted formal

partnerships between parents and health and developmental

professionals and has allowed for sequential home, primary

care, and preschool community care developmental

observations.

1.6. Motor and prelinguistic milestone scales

In addition to the ASQ, there are useful indicators of emerging

gross motor and prelinguistic developmental competencies

that have been described by Capute and colleagues.38 The

motor milestones have been standardized during primary

care visits and operationally describe postural control through

independent walking.39 Normative samples have included

both term and preterm cohorts. They can serve as an indicator

for more complex neuromotor assessments such as the Pea-

body Developmental Motor Scales or more recently developed


gross motor, manual abilities, or functional mobility

scales.40e43

The prelinguistic milestones are part of the Clinical

Linguistic andAuditoryMilestone Scale (CLAMS) of the Capute

Scales.44 They provide a series of sequential indicators that

can generate a communicative quotient. These develop-

mental questionnaires that determine which motor and

communicative milestones are consistently achieved thus

allow classification of developmental status as typical e i.e.

normal e (DQ> 84), some delay (DQ 71e84), and significant

delay (DQ< 71). This same classification can also be applied to

the child’s overall adaptive status. Most importantly, both the

CLAMS and the Motor Quotient can help establish premorbid

developmental status and have the capacity to detect regres-

sion of previously acquired motor and communicative

competencies.

1.6.1. Brief infant toddler social emotional assessment(BITSEA)The Brief Infant Toddler Social Emotional Assessment (BIT-

SEA) is a 34-item questionnaire that addresses social,

emotional, and behavioral problems as well as competen-

cies.45 Problem behaviors are categorized as either part of

typical normal development (aggression, sadness, and fear) or

as occurring with excessive frequency and/or intensity, or as

absent when they would be developmentally appropriate.46

Deviant behaviors include repetitive behaviors, self injurious

behaviors, and developmentally inappropriate behaviors.

In the BITSEA, overall problem behaviors are scored as

externalizing problems, internalizing problems, problems of

dysregulation, and atypical behaviors. Behavioral competen-

cies include attention, reflecting the child’s determination to

complete complex tasks such as a 5 piece puzzle, pro-social

peer relations, empathy, imitation/play skills, and social

readiness. The parent form takes about 5e7 min to complete

or 7e10 min to be administered with structured interview.

The BITSEA includes items that capture some of the indicators

of autistic spectrum disorders and clinically significant

behavior stressor that require discussions to insure appro-

priate management and intervention strategies.

1.6.2. Strengths and difficulties questionnaire (SDQ)The Strengths and Difficulties Questionnaire (SDQ) was

initially developed as a health screening tool for psychiatric

‘case-ness’ applicable in the general population of children. It

has also been validated in children with hemiplegia and can

be used sequentially to detect stressors and behavioral diffi-

culties in the setting of chronic illness. It measures attention,

social, anxiety, conduct problems and, importantly, pro-social

behavior among children aged 3e16 years.47 This last score

adds valuable information on ‘difficulties’ and also increases

its acceptability to users. In addition to scoring the behavioral

difficulties themselves, the SDQ provides a separate score for

the impact of those difficulties on the child, family and social

life and, particularly in preschoolers, can serve as a bridge to

more complex behavioral inventories such as the Behavior

Assessment System for Children, Second Edition (BASC 2),

Achenbach System of Empirically Based Assessment (ASEBA),

or Conner’s Rating Scales.48

1.7. Application of developmental and functionalsurveillance to children with medulloblastoma

With increased survival of children with medulloblastoma,

there is increased recognition that in young children surveil-

lance for developmental and adaptive status, linked to

a schema of neuropsychological evaluations of core cognitive,

memory, executive function, and behavior, is required.

To implement this project, a pediatric nurse practitioner and

a neurodevelopmental specialist trained in multidisciplinary

assessments established a surveillance system for structured

phone interviews with families.49 This protocol was based on

previousneurodevelopmentaloutcomeassessmentsof children

who survived extreme prematurity, underwent surgery for

repairs of congenital cardiac malformations, were enrolled in

randomized trials to prevent retinal detachment following reti-

nopathy of prematurity, had experienced white matter injury,

traumatic brain injury, or were part of a genetic registry (Rett

Syndrome, Down Syndrome).21,50

Overall, 76 childrenwithmedulloblastomaunder the age of

36 months were enrolled as part of an investigation of devel-

opmental and functional outcomes following post-operative

chemotherapy and local conformal radiation in infants with

medulloblastoma (Pediatric Oncology Group Protocol P9934,

David Ashley, PI).49,51

The overall survival to the age of six years was approxi-

mately 70%. The event-free survival was 50% at four years.

The goal was tomaintain an ongoing relationship between the

investigators and the family and to measure the child’s

performance over time across developmental and adaptive

domains. The instruments included the ASQ and the Pediatric

Independence Measure (WEE FIM�) which provides explicit

measures of functioning in self-care, mobility, and social-

cognitive domains.52 We defined functional developmental

status as globally normal when developmental quotients (DQ)

were greater than 70 or more, evolving disability in func-

tioningwhen DQwas<70, and severe disability in functioning

when the DQwas<55.53 In addition,we established the child’s

baseline motor and communicative developmental compe-

tencies prior to diagnosis of medulloblastoma, using the

Motor Quotient and CLAMS.38,39

Overall, the phone interview followed the schedule in

Table 4 for eliciting health, developmental, and adaptive

status in preschool children.

This interview involved approximately 30 min of phone

time. We coupled this protocol with neuropsychological

testing, which included the Bayley Scales of Infant Develop-

ment (2nd edition) for children younger than 3 years old,

Wechsler Preschool and Primary Scale of Intelligence Testing

3 if the child was between 3 and 6 years. In addition, ongoing

long-term assessments in middle childhood and teen years

are in process and will include executive function, memory,

health-related quality of life, and educational achievement in

reading and mathematics.

Compared to baseline developmental competencies, there

was a significant decline in the percentage of children with

motor and cognitive functional skill DQ scores greater than 70

with the majority of infants showing low (‘delayed’) develop-

mental quotients when assessed after diagnosis and surgery

Table 4 e Core health, developmental, adaptive, and family well-being surveillance.

Time Child healthand familywell-being

Ages and stagesquestionnaires

Adaptivefunctionalassessments

Gross motorand clinical linguisticauditory milestones

Baseline/Registration X X X X

Post-surgery 1/Pre-chemotherapy X X X X

Post-surgery 2/Pre-radiation X X X X

Post-diagnosis (12 mos) X X X X




5e10 min 10 min 10 min 5 min


and prior to adjuvant chemotherapy and radiation therapy

(Fig. 2). This suggests that surgery and/or the tumor may have

more impact on development than previously recognized.49,51

Although there was continued developmental progression

and improved gross motor and cognitive functioning over

time, the children’s performance remained impaired

compared to typically developing children, less than 5% of

whom have motor or cognitive functional DQ less than 70.

Although the majority of children during their first year

after diagnosis have some functional limitations across

mobility, self-care and cognitive domains, most of these are

not severe, but are mild to moderate. Importantly, more than

70% children are classified as globally normal (DQ> 70) and

only 10% of the cohort have severe functional limitations

(DQ< 55) two years after diagnosis (Fig. 3).

1.8. What are the lessons?

First, telephone interview is a powerful method that can

capture both developmental and functional status that can be

linked to both face-to-face interviews and a standard protocol

for periodic family feedback at intervals. Secondly, this

surveillance system allows us a multi-dimensional view of

day-to-day management and can describe how the child is

typically performing activities that are essential to early

childhood well-being. We have tools that help to describe

developmental processes, adaptive skills, and social-

emotional competencies in language that are common to all

Fig. 2 e Percentage of children with medulloblastoma prior

to 36 months of age and WEE-FIM functional

developmental quotients greater than 70 at four time

points.

stages of multidisciplinary management. An additional

strength of this study was the linkage of these telephone

assessments to periodic neuropsychological assessments. In

this regard, the developmental trajectories of developmental

and adaptive consequences will help us to understand the

pathways of risk and resilience for learning and academic

progress. A centrally administered phone-based interview

measuring key indicators of health development and func-

tioning can serve as a form of quality assurance and audit of

outcomes against pre-defined standards. Our initial use of

these surveillance strategies at regular intervals also

improved the families’ willingness to participate in neuro-

psychological assessments.

Most importantly, this strategy also allowed us to receive

ongoing feedback regarding child and family well-being, and

the long-term impact of treatments on the children and their

families. We can explicitly assess not only families’ basic

needs (e.g., food, housing, employment) but also community,

medical, and educational supports as well as caregiver phys-

ical and behavioral health. This will help us to better under-

stand the appropriateness or otherwise of early, aggressive,

multimodal treatments, as well as ways to reduce the risk of

their late effects that sometimes include multiple motor,

communicative, and adaptive functional limitations.

The methodology highlighted in this article has been

applied to children receiving other forms of translational

technologies who are at highest risk for neurodevelopmental

disabilities. These cohorts include extremely low birthweight

infants, infants undergoing neuroprotection after neonatal

encephalopathy, children with early onset of genetic meta-

bolic storage diseases receiving enzyme replacements, stem

Fig. 3 e Percentage of children with medulloblastoma prior

to 36 months of age and WEE-FIM functional

developmental quotients greater than 70 and less than 50

at five points over the first two years from diagnosis.


cell transplantation, or chaperone interventions, and children

receiving cardiac surgery.

To illustrate lessons learned from this approach we will

describe a series of adaptive, educational, and health-related

quality of life outcomes in the multicenter for Cryosurgery for

Retinopathy of Prematurity (CRYO-ROP).

1.9. Cryosurgery for retinopathy of prematurity(CRYO-ROP)

Childrenwith Retinopathy of Prematurity (ROP) are among the

sickest, tiniest, and most fragile and have been either born

very low birthweight (1001e1499 g) or extremely low birth-

weight (<1000 g). Developmental and functional surveillance

strategies were implemented in a large cohort exceeding 1000

children who had birthweight of less than 1250 g and were

enrolled in the NIH sponsored Multicenter Randomized Trial

of Cryosurgery for Retinopathy of Prematurity (CRYO-ROP).

Approximately 88% of the subjects were followed across 25

centers over the first five and a half years of life.54 Functional

status declined and severe disability inmobility, self-care, and

communicative functioning increased as the severity of ROP

increased. At kindergarten entry at 5.5 years of age, functional

limitations in childrenwithout ROPwere infrequent, affecting

motor, self-care, continence and communicative/cognitive

domains in 4%, 7%, 4%, and 8% respectively. By contrast,

functional limitations at the same age for those with severe

ROP and unfavorable vision were very frequent, affecting

these same domains in 43%, 78%, 51%, and 67% respectively.

Multiple logistic regression analysis revealed that favorable

visual status and favorable neurological score at two years of

age predicted overall adaptive functional status at 5.5 years.

At eight years, 255 survivors (88%) that had themost severe

ROP were examined with respect to developmental and

educational outcomes.55 Of those with severe ROP and unfa-

vorable vision, special education placement occurred in 63%,

and performance at below average level for their year group in

48%. Less than 20% of this group, compared to 50% of those

with favorable vision, were competent in reading, mathe-

matics, and handwriting. Neuro-sensory and functional

status at kindergarten entry as well as higher socioeconomic

rates predicted the child’s need for special education services

and challenges keeping up with academic skills of peers.

Additional studies by the CRYO-ROP Cooperative Group at

age 10 years involved parental assessment with the Health

Utility Index (HUI) as ameasure of health-related quality of life

(HRQOL).Theproportionof sightedchildrenwith limitations in

four or more HUI attributes of mobility, speech, dexterity,

cognition, emotion, pain, or hearing was 6.4% compared with

47% in children with blindness or low visual acuity.55

Thus, this study demonstrated that the complex interrela-

tionships between neuro-sensory status, adaptive skills at

kindergartenentry, educational status inmiddlechildhood,and

adolescent HRQOL can be elucidated by measuring functional

status among children at highest risk for cerebral palsy (20%),

developmental delay or developmental disability (35%), and

visual disability (62%). A similar strategy could be applied to

measure outcomes among children with CNS malignancies as

the best of translational and clinical sciences is applied to

improve long-term outcomes. This will help us to identify and

understanddevelopmentalandadaptiveskill trajectoriesand to

use them in order to optimize adolescent and adult success.56

Acknowledgement

This article was supported in part by the Grant Healthcare

Foundation’s “Passport to Health” grant and Leadership

Education in Neurodevelopmental Disabilities (LEND) grant

(T73MC). The funding source had no role in the preparation,

review, or approval of this manuscript.

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Developing preschool surveillance tools for adaptive functioning: Lessons for neuro-oncology