Accepted Manuscript

Title: Implementation of Early Diagnosis and Intervention Guidelines for

Cerebral Palsy in a High-Risk Infant Follow-Up Clinic

Author: Rachel Byrne, Garey Noritz, Nathalie Maitre, NCH Early

Developmental Group

PII: S0887-8994(17)30782-8
DOI: http://dx.doi.org/doi: 10.1016/j.pediatrneurol.2017.08.002
Reference: PNU 9207

To appear in: Pediatric Neurology

Received date: 21-7-2017

Revised date: 6-8-2017
Accepted date: 7-8-2017

Please cite this article as: Rachel Byrne, Garey Noritz, Nathalie Maitre, NCH Early
Developmental Group, Implementation of Early Diagnosis and Intervention Guidelines for
Cerebral Palsy in a High-Risk Infant Follow-Up Clinic, Pediatric Neurology (2017),
http://dx.doi.org/doi: 10.1016/j.pediatrneurol.2017.08.002.

This is a PDF file of an unedited manuscript that has been accepted for publication. As a service
to our customers we are providing this early version of the manuscript. The manuscript will
undergo copyediting, typesetting, and review of the resulting proof before it is published in its
final form. Please note that during the production process errors may be discovered which could
affect the content, and all legal disclaimers that apply to the journal pertain.
Title:
Implementation of early diagnosis and intervention guidelines for cerebral
palsy in a high-risk infant follow-up clinic

Authors and affiliations:

Rachel Byrne1, Garey Noritz2 and Nathalie Maitre2,3, on behalf of the NCH Early
Developmental Group2.

Cerebral Palsy Foundation, New York, NY

1

2
Department of Pediatrics, Nationwide Children’s Hospital, Columbus, OH
3
Center for Perinatal Research at Nationwide Children’s Hospital, Columbus, OH

Correspondence:
Dr. Nathalie L. Maitre, Department of Pediatrics
Nationwide Children’s Hospital
700 Children’s Drive, WB6225, Columbus, Ohio 43205
Nathalie.maitre@nationwidechildrens.org
PH: 614-722-4559
FX: 614-722-4541

Manuscript Word Count: 2759

Abstract

Background: Cerebral palsy (CP) is the most common physical disability in childhood,
and is mostly diagnosed after 2 years of age. Delays in diagnosis can have negative long-
term consequences for children and parents. New guidelines for early diagnosis and
intervention for CP were recently published, after systematic review of the evidence by
international multidisciplinary experts aiming to decrease age at diagnosis. The current
study’s goal was to test the feasibility of implementing these guidelines in an American
clinical setting.

Methods: We designed a stepwise implementation process in a neonatal intensive care

follow-up clinic. Efficacy was tested by comparing 10-month pre/post-implementation
periods. Clinic visit types, CP diagnosis, provider competencies and perspectives, and
balancing measures were analyzed.

Page 1 of 30
Results: Changes to infrastructure, assessments, scheduling algorithms, documentation
and supports for therapist/providers in diagnosis or counseling were successfully
implemented. Number of 3-4 month screening visits increased (255 to 499, p<0.001);
mean age at diagnosis decreased (18 to 13 months, p<0.001). Clinic team awareness of
early CP diagnosis and interventions increased (p<0.001). There was no decrease in
family satisfaction with overall clinic function. Opportunities for improvements included
documentation for transitioning patients, generalizabilty across hospital clinics,
systematic identification of high-risk status during hospitalization and need for CP care
guidelines for infants under two.

Conclusions: We demonstrated for the first time in a US clinical setting the feasibility of
implementation of international early diagnosis and treatment guidelines for CP. This
process is adaptable to other settings and underscores the necessity of future research
on CP treatments in infancy.

Keywords: infant; cerebral palsy; early intervention; diagnosis

Background:

Worldwide, cerebral palsy is the most common physical disability in childhood1,2.

Cerebral palsy is a non-progressive disorder of movement originating from neural

lesions in the perinatal period3, and is associated with a wide range of common

comorbidities4. Many of these respond well to interventions in early childhood,

when brain plasticity is at its greatest5 and developmental trajectories can be

altered with maximal benefit into adulthood6. In large research networks,

investigators have successfully decreased the age at cerebral palsy diagnosis to

Page 2 of 30
19 months7. However, in most clinical settings the age for diagnosis of cerebral

palsy is on average 2 years or beyond8. Delays in diagnosis of cerebral palsy are

associated with worse long-term function and participation, parental

dissatisfaction, and higher rates of mental health conditions, including

depression4,9.

In response to these concerns, a large working group of international experts in

cerebral palsy-related disciplines and parent stakeholders published guidelines

for early detection and intervention for cerebral palsy10, based on a systematic of

evidence for why and how cerebral palsy should be recognized as early as

possible 11. The guidelines include recommendations for neuroimaging,

neurological exams and motor assessments for infants below 5 months corrected

age and between 5-24 months corrected age. They define the concept of “high-

risk for cerebral palsy” at 3-4 months, a category in which infants do not yet

receive a diagnosis, but due to a constellation of clinical factors, neuroimaging

findings and standardized assessments, have a 95% probability of developing

cerebral palsy in the following years. Best current evidence for counseling is

based on published parent surveys and high-quality research. The guidelines can

be translated into clinical practice by adapting core elements to suit individual

clinical settings illustrated in Figure 1 (setting is a neonatal intensive care unit

(NICU) follow-up program with applied examples of the guidelines).

Page 3 of 30
While research programs leveraged the evidence base for early detection of

cerebral palsy12-14, no clinical programs implemented the guidelines in their

entirety as standard care. NICU follow-up programs routinely follow two infant

populations with common identifiable factors for cerebral palsy: preterm infants

and term infants with neonatal encephalopathy. Many children with less common

risk factors for cerebral palsy also receive care in such programs due to medical

complexity in the perinatal period. The goal of our study was therefore to test the

effectiveness of an implementation process for clinical use of the early diagnosis

guidelines10 and apply them to a NICU follow-up clinic setting, where a large

population of high-risk infants are routinely monitored. We hypothesized that

comparison of quantitative metrics for clinic visits, diagnoses and provider

training competency would demonstrate improvements before and after

implementation. We complemented our study with balancing measures of parent

satisfaction and qualitative data on provider perceptions.

Methods:

The Nationwide Children’s Hospital Institutional Review Board under an exempt

determination IRB17-00409 due to the use of unidentified data.

Part 1-Baseline conditions:

Setting and population: The NICU follow-up clinic in a large referral tertiary care

center encompassing a network of nine NICUs including levels II, III and IV units

(a range from special care nurseries to intensive care to surgical referral

centers)15 and geographical region of approximately 200 square miles. Prior to

Page 4 of 30
the implementation of the guidelines, no specific early cerebral palsy program

existed. Suspicion of cerebral palsy prompted referral at discharge from the clinic

(2 years of age) to a developmental pediatrician or various specialty programs

outside the NICU follow-up program for further evaluation. The exception to this

pattern was for infants who received hypothermia, all of whom were followed by

the pediatric neurologist within the NICU follow-up program. The clinic saw an

average of 5000 high-risk visits per year, mostly referred from the NICU network,

but also from community providers. Referrals to the clinic were based on

established criteria (Supplemental Table 1).

Infrastructure: The electronic medical record was on an Epic platform, commonly

used by large United States health systems to access, organize, store and share

patient medical records. Therapist documentation of Bayley Scales of Infant and

Toddler Development Third ed.16 was standardized with flow sheets and

checklists. Another feature of Epic was the MyChart option, an online secure

portal, allowing parents to communicate with clinic providers. Pre-

implementation, documentation of assessments was heterogeneous, reflecting

personal choice and occasional practice in other clinics. Clinic scheduling

templates were based on therapist scheduling availability and medical

practitioners rarely had assigned patients.

Visit schedules, assessments and referral: Pre-implementation, schedules

followed very preterm infants at 3-4 months, 9-12 months and 22-26 months with

Page 5 of 30
interim visits based on specific therapist or dietitian identified needs (Figure 1).

Assessments were guided by participation in the Neonatal Research Network 17

as well as a strong evidence-based practice group in the therapy department. In

addition, specialized providers examined those patients with high neurological

complexity. Therapist assessments often determined referrals for therapy

services while medical providers made the referrals to subspecialty providers and

programs (audiology, ophthalmology, physical medicine, developmental

pediatrics, psychology, complex care, cerebral palsy) or for procedures based on

their clinical judgment and a developmental algorithm for the 2-year visit.

Providers: The NICU follow-up team was multidisciplinary and diverse.

Supplemental table 2 lists roles and responsibilities of each provider in the NICU

Follow-Up clinic pre- and post-implementation.

Research: Clinic personnel were involved in research through Neonatal

Research Network studies requiring follow-up of 2-year neurodevelopmental

outcomes as a measure of efficacy of NICU-based interventions. However,

interventional studies for infants were not yet integrated into the workflow of the

clinic, making it difficult to incorporate new and proven interventions for cerebral

palsy and to promote a culture of change into the setting.

Part 2-Implementation Process:

The process followed a predetermined timeline (Figure 3). The planning phase

involved the design of a clinical schedule based on the guidelines (Figure 1), also

respecting the primary mandate of the NICU follow-up program to follow all high-

risk infants, not only those with cerebral palsy, and to promote effective transition

Page 6 of 30
to existing services after leaving the program. The preparation phase initiated

provider culture change through a series of small moderated workgroups, each

charged with guiding individual aspects of optimal developmental surveillance for

all patients. Training in the General Movements Assessment (GMA) was

accomplished by Prechtl’s Trust certified trainers. Clinical competency in the

Hammersmith Infant Neurological Examination (HINE) was a novel

implementation process published recently18. Training of medical providers in

scope of practice of various therapy disciplines, available services and best

evidence for occupational, physical, speech and feeding therapies in infancy was

accomplished by a series of self-directed modules designed by clinical therapy

leaders. Dietitians utilized first World Health Organization curves for infants19,

then the cerebral palsy specific growth curves20 based on Gross Motor Function

Classification System levels. Documentation in Epic was modified to match the

new visit schedule, driven by providers practicing regularly in the clinic. Clinic

templates were switched to a provider-based system, allowing continuity of

medical care and neurological trajectories as well as longitudinal perspective

from the team leaders who could request specific therapy assessments based on

new knowledge gained in the modules. Visits before 3 months were scheduled

for those with the greatest risk factors and complexity. Research was integrated

through additional workspace, training of coordinators, improved communication

with nursing and social work leaders and visibility of rehabilitation study

materials21,22.

Page 7 of 30
Parent support was addressed through counseling with the elements

recommended in published literature and the guideline mandates: “face-to-face,

with both caregivers present if appropriate, private, honest, jargon-free, with

empathic communication tailored to the family”4. Identification of strengths and

goals was included and questions were fostered by allowing the diagnosing

provider to return at conclusion of the visit to reassess the emotional context and

answer additional questions. MyChart use was explained and encouraged.

Referral to services was complemented by a set of written materials (therapist

handouts and parent educational booklet)23.

Implementation occurred over one month, during which process improvement

guides were available to observe, troubleshoot, record challenges and provide

support if necessary. Written feedback was also solicited.

Consolidation involved further observation and support in areas identified as

challenges such as knowledge verification, training gaps, hand-off problems,

template and workflow, scheduling difficulties and referral difficulties.

Development was driven by group identification of new priorities for the next

cycle in clinical, operational and research domains during regular provider

meetings. Intra-hospital as well as community outreach were added as new

priorities.

3- Evaluation metrics

Quantitative metrics (Table 1) were obtained in the 10-month periods from July 1,

2014 to May 30, 2015 and July 1, 2016 to May 30, 2017 to prevent the possibility

Page 8 of 30
of crossover effects from early phases of implementation. Balancing measures

were obtained using standard anonymous feedback clinic questionnaires at the

end of each visit from any parent in the clinic, not only those of children with

cerebral palsy. The goal of these measures was to determine whether

implementation of the program negatively impacted parent satisfaction through

three questions about overall perception of clinic function. Provider perspectives

on cerebral palsy recognition and intervention for those involved in the care of

infants 12 months and under were obtained through a survey scored on a 5-item

ranked scale.

Results:

Clinic Metrics (Table 1)

The proportion of preterm infants seen in the clinic was not significantly different

between the two periods (85.7% vs. 76.6%, p = 0.9). The incidence of cerebral

palsy was unchanged pre- and post-implementation of the guidelines in the

hospital system18. The average age of diagnosis of cerebral palsy was decreased

from 18 months to 13 months of age18. Number of 3-4 month visits increased

compared to other types of visits (due to the decreased clinic load of new

patients and increased focus on 3-4 month visit), resulting in more children

having GMA and HINE assessments performed at this time point. The number of

children in the clinic diagnosed by 12 months increased, with the age at

diagnosis decreasing significantly. In addition, changes in the clinic were

Page 9 of 30
reflected by a decreased mean age at diagnosis in the overall population of

children with diagnoses of cerebral palsy at the hospital under the age of three.

Provider metrics

Provider metrics (Table 2) demonstrated significant improvement of awareness

of best practices around early diagnosis and early interventions for infants less

than 12 months. When providers were asked to describe what they had learned

post-implementation five major themes emerged: optimizing developmental

outcomes for children, improvement of support to parents, awareness of

assessment tools, effective rehabilitation interventions, and importance of early

detection. Balancing measures of parent satisfaction with clinic processes

remained high and unchanged (Supplemental Table 3).

Statistical analysis: Continuous variables were analyzed using two-tailed t-tests.

Distributions of visit types pre- vs. post-implementation were compared using

McNemar’s test with alpha=0.05. Scores on scales using ranked items were

analyzed using two-tailed Wilcoxon matched-pairs signed rank test, with with

alpha=0.05. All calculations were performed using SPSS software (IBM Corp.

Released 2013. IBM SPSS Statistics for MacIntosh, Version 22.0. Armonk, NY:

IBM Corp.)

Discussion:

This study represents the first successful attempt in the United States to

implement the international guidelines for early detection and intervention for

Page 10 of 30
cerebral palsy. Average age at diagnosis decreased in the clinic program with an

increase in provider knowledge-base. Overall, parents were not dissatisfied by

the process changes and qualitative reports indicated satisfaction from providers

and patients on the new model.

The NICU follow-up program presented a unique opportunity for success in

implementation due to the strong institution-wide culture of commitment to

excellence in clinical care, research and quality improvement. Institutional

support at the operational level allowed business managers and clinical leaders

to direct the change in vision. For example, the therapist group already had a

funded evidence-based practice group. The commitment of the NICU follow-up

program team to their high-risk population made a change in practice relatively

easy, as it did not represent a culture change.

In the consolidation phase, several immediate opportunities for process

improvement were identified. In particular, as previously reported18, various

providers had different comfort levels with giving the diagnosis of high-risk for or

of cerebral palsy. Establishing confidence in administering the HINE or reading

GMAs was sometimes difficult. For all these issues, a team-based support

system was established so that providers could always rely on a more advanced

or experienced provider for assistance. For diagnosis, if the provider did not feel

comfortable or assessed that the family was not ready (emotional state, lack of

support system, other parent not present, child acutely ill at visit) to receive a

diagnosis, they explained that findings of an assessment were not typical. They

then scheduled a return visit with a neurodevelopmental-trained specialist in the

Page 11 of 30
clinic. Similarly, for the GMA, therapists with less experience or without

certification recorded the GMA on a secure tablet, explain that results would be

reviewed by an experienced provider and communicated as soon as available.

Experienced providers were available either in person or remotely. Provider

confidence in neurological exams improved 18. On average, providers currently

perform 5-6 HINE exams per day, leading to increasing clinical competency and

confidence. A year after the initial training program, a repeat observation of

clinical exams prevented drift and clarified questions. When trained providers left

and new providers were incorporated, a systematic training process and team-

support system helped overcome this new challenge.

The documentation implementation was overall a success, with streamlined

notes and flow sheets routinely used for the GMA and HINE. However, a noted

failure was lack of compliance with an additional documentation form used in the

Comprehensive Cerebral Palsy program for older children. The goal of filling out

this flow sheet in addition to regular Epic documentation was to provide data for

the Learn from Every Patient project24 and facilitate transitions. However, the

added burden of repetitive documentation proved unsustainable and use of the

forms was paused while new processes were developed to capture the needed

information without duplication of effort. A streamlined referral process was

devised with the indication of “transition to the cerebral palsy program”, placed at

30-33 months to ensure continuity between the early and later cerebral palsy

programs.

Page 12 of 30
The success of the program in early identification prompted troubleshooting

questions currently not addressed in the care of cerebral palsy patients.

Partnering with physical and occupational therapists, orthopedic surgeons and

orthotists specializing in the care of very young children and infants allowed a

reframing of expectations and ordering patterns for upper and lower extremity

orthotics. Evidence-base for truncal supports is also being reviewed and

guidelines for prescription in the clinic are still in development. Another area of

critical importance to be addressed was hip surveillance in infants 12 months and

under, in circumstances where the severity of cerebral palsy is uncertain or

unstable at best25 (see Supplemental Table 4).

The development phase highlighted opportunities for future improvements. Early

identification in the NICU of all infants at-risk for cerebral palsy in addition to

those already meeting criteria for follow-up became a priority. Patients referred to

the NICU for reasons that are not commonly associated with neurological insults

are neither routinely screened using neuroimaging or referred to the follow-up

program (surgical intervention, routine preterm care from 32-37 weeks,

respiratory distress in term newborns, hyperbilirubinemia or sepsis evaluations).

Routine use of the GMA prior to discharge from the NICU can help screen for a

infants meeting the “infant-attributable risk” pathway of the international

guidelines10. Results of a one-year experience using the GMA in the NICU for

this purpose are forthcoming.

Page 13 of 30
In addition, several other clinics at the hospital that follow children at high risk for

cerebral palsy have not implemented the guidelines. Many of these children are

referred when pediatricians identify the need for a specialist after the age of 3,

while others were never cared for in a NICU. Opportunities and willingness

currently exist to adapt the guidelines to these settings to promote more

consistent early care for high-risk infants. Community outreach and education to

support pediatric providers through education materials and care coordination is

ongoing.

Finally, this single site experience in implementation processes prompted interest

at the national level for implementation of the international guidelines. The

Cerebral Palsy Foundation therefore initiated the creation of a structured

implementation network across 6 major United States sites in July 201726 to be

expanded over three years in a widening US-specific network. Knowledge

translation, toolkits, training and infrastructure will be provided to all participants,

many derived from this single site experience.

Conclusions

This work demonstrates the first successful attempt at implementing the

international early diagnosis and treatment guidelines for cerebral palsy in a

clinical setting in the United States. The process feasibly lends itself to

adaptation to other settings. The current experience highlights the need for new

clinical care guidelines and novel intervention research for infants with cerebral

palsy diagnosed under 2 years.

Page 14 of 30
Competing interests

All the authors declare that they have no competing interests. This research did

not receive any specific grant from funding agencies in the public, commercial, or

not-for-profit sectors.

Authors' contributions

NM conceptualized the implementation process and the research study. RB

contributed to the research design and wrote the first draft of the manuscript. GN

contributed to the implementation process and draft of the manuscript. All

authors in the NCH Early Developmental Group designed and participated in

components of the implementation process and contributed to the manuscript. All

authors read and approved the final manuscript.

Acknowledgements

We would like to thank all our patients and their families. In addition, we would

like to thank Ms. Rebecca Lam and Ms. Olena Chorna for their help in preparing

this manuscript and obtaining all regulatory materials.

NCH Early Developmental Group includes: Rukhshana Ahmed, Erin Anderson,

Nancy Batterson, Lawrence Baylis, Brooke Bernstein, Teresa Borghese, Regina

Bray, Chelsea Britton, Marie Cain, Ilene Crabtree, Caitlin Cummings, Kelli Dilver,

Jaclyn Gray, Jennifer Haase, Andala Hardy, Megan Harrison, Alyssa Huddleston,

Cherie Isler, Cheryl Kokensparger, Beth Kosch, Joanna Kinner, Jessica Lewis,

Ryan Nicoll, Brianne Palay, Audrey Shifflett, Laurel Slaughter, Michelle Smith,

Page 15 of 30
Annie Temple, Kristy Theller, Lindsey Tobias, Jill Tonneman, Elizabeth Wagner,

Kylie Weber.

References

1. Boyle CA, Boulet S, Schieve LA, et al. Trends in the prevalence of

developmental disabilities in US children, 1997-2008. Pediatrics.
2011;127(6):1034-1042.
2. Christensen D, Van Naarden Braun K, Doernberg NS, et al. Prevalence of
cerebral palsy, co-occurring autism spectrum disorders, and motor
functioning - Autism and Developmental Disabilities Monitoring Network,
USA, 2008. Dev Med Child Neurol. 2014;56(1):59-65.
3. Rosenbaum P, Paneth N, Leviton A, et al. A report: the definition and
classification of cerebral palsy April 2006. Dev Med Child Neurol Suppl.
2007;109:8-14.
4. Novak I, Hines M, Goldsmith S, Barclay R. Clinical prognostic messages
from a systematic review on cerebral palsy. Pediatrics.
2012;130(5):e1285-1312.
5. Johnston MV, Ishida A, Ishida WN, Matsushita HB, Nishimura A, Tsuji M.
Plasticity and Injury in the Developing Brain. Brain & development.
2009;31(1):1-10.
6. Heckman JJ. Schools, Skills, and Synapses. Econ Inq. 2008;46(3):289.
7. Morgan C, Novak I, Dale RC, Guzzetta A, Badawi N. Single blind
randomised controlled trial of GAME (Goals - Activity - Motor Enrichment)
in infants at high risk of cerebral palsy. Res Dev Disabil. 2016;55:256-267.
8. Hubermann L, Boychuck Z, Shevell M, Majnemer A. Age at Referral of
Children for Initial Diagnosis of Cerebral Palsy and Rehabilitation: Current
Practices. J Child Neurol. 2016;31(3):364-369.
9. Baird G, McConachie H, Scrutton D. Parents' perceptions of disclosure of
the diagnosis of cerebral palsy. Arch Dis Child. 2000;83(6):475-480.
10. Novak I, Morgan C, Adde L, et al. Early, Accurate Diagnosis and Early
Intervention in Cerebral Palsy: Advances in Diagnosis and Treatment.
JAMA Pediatr. 2017.
11. Early Identification and Early Neurorehabilitation in cerebral palsy: 2014
Summit Report. Available from:
https://impact.cerebralpalsy.org.au/activities/research-summits/2014-
summit/. Accessed 1 Jul 2017
12. George JM, Boyd RN, Colditz PB, et al. PPREMO: a prospective cohort
study of preterm infant brain structure and function to predict
neurodevelopmental outcome. BMC Pediatr. 2015;15:123.
13. Morgan C, Novak I, Dale RC, Badawi N. Optimising motor learning in
infants at high risk of cerebral palsy: a pilot study. BMC Pediatr.
2015;15:30.

Page 16 of 30
14. Spittle AJ, Lee KJ, Spencer-Smith M, Lorefice LE, Anderson PJ, Doyle
LW. Accuracy of Two Motor Assessments during the First Year of Life in
Preterm Infants for Predicting Motor Outcome at Preschool Age. PLoS
One. 2015;10(5):e0125854.
15. Barfield WD, Papile LA, Baley JE, Benitz W, Cummings J, Carlo WA,
Kumar P, Polin RA, Tan RC, Wang KS, Watterberg KL. Levels of neonatal
care. Pediatrics. 2012 Sep 1;130(3):587-97.
16. Bayley N. Bayley Scales of Infant and Toddler Development, Third Edition
(Bayley III). In. San Antonio (TX): Pearson Clinical; 2006.
17. Neonatal Research Network (NRN). Background and Overview of the
Follow-up Program. Springer International Publishing; 2016. Available
from: https://neonatal.rti.org/about/fu_background.cfm. Accessed 1 Jul
2017
18. Maitre NL, Chorna O, Romeo DM, Guzzetta A. Implementation of the
Hammersmith Infant Neurological Examination in a High-Risk Infant
Follow-Up Program. Pediatr Neurol. 2016;65:31-38.
19. Borghi E, de Onis M, Garza C, et al. Construction of the World Health
Organization child growth standards: selection of methods for attained
growth curves. Stat Med. 2006;25(2):247-265.
20. Brooks J, Day S, Shavelle R, Strauss D. Low weight, morbidity, and
mortality in children with cerebral palsy: new clinical growth charts.
Pediatrics. 2011;128(2):e299-307.
21. Chorna O, Heathcock J, Key A, et al. Early childhood constraint therapy
for sensory/motor impairment in cerebral palsy: a randomised clinical trial
protocol. BMJ Open. 2015;5(12):e010212.
22. J. H. Daily and Weekly Rehabilitation Delivery for Young Children With
Cerebral Palsy (DRIVE); 2017. Available
from: https://clinicaltrials.gov/ct2/show/NCT02857933. Accessed 1
Jul 2017.
23. Cerebral Palsy Now. From Diagnosis to Understanding. Greenville, SC
29604; 2015. Available from: https://cpnowfoundation.org/wp/wp-
content/uploads/2015/11/cerebral palsy-ToolKit.pdf. Accessed 1 Jul 2017.
24. Lowes LP, Noritz GH, Newmeyer A, Embi PJ, Yin H, Smoyer WE. 'Learn
From Every Patient': implementation and early results of a learning health
system. Dev Med Child Neurol. 2017;59(2):183-191.
25. Hagglund G, Lauge-Pedersen H, Wagner P. Characteristics of children
with hip displacement in cerebral palsy. BMC Musculoskelet Disord.
2007;8:101.
26. Cerebral Palsy Foundation: http://yourcpf.org/early-recognition. Accessed
1 Jul 2017.
27. Amiel-Tison C. Update of the Amiel-Tison neurologic assessment for the
term neonate or at 40 weeks corrected age. Pediatr Neurol.
2002;27(3):196-212.

Page 17 of 30
Figure 1: Translation of international guidelines into a clinical practice
algorithm in a neonatal intensive care unit follow-up program.
Infants referred due to newborn-attributable risks (e.g. neuroimaging findings
consistent with neonatal encephalopathy) most frequently start surveillance
according to the guidelines at the 3-4 month visit. If they are still hospitalized
(e.g. extremely preterm infant with white matter injury and lung disease), the
guideline mandated surveillance happens at the bedside with a HINE, TIMP and
a GMA. Infants referred due to infant-attributable risks start at closest point on
the pathway. For example, an infant referred to the program at 9 months by their
pediatrician due to inability to sit would start with a HINE and a Bayley Scales of
Infant and Toddler Development. If an MRI has not yet been performed by the 3-
4 month visit when neither sedation nor anesthesia is required, it is deferred until
12 months when anesthesia is no longer required by this particular setting’s
radiology protocols. During the program, infants are referred to services and
subspecialists according to needs and established clinical algorithms.

HINE: Hammersmith Infant Neurological Examination

GMA: General Movements Assessment
TIMP: Test of Infant Motor Performance
Bayley III: Bayley Scales of Infant and Toddler Development Third edition
CBCL: Child Behavior Checklist

*select exams target developmental progression and represent best feasible

evidence for specific concern (e.g. Gross Motor Function Measure-66 at 18
months and 30 months when infant walking to begin establishing trajectory,
Infant Toddler Sensory Profile for sensory processing concerns, Quality of Upper
Extremity Skills Test for arm evaluation in hemiplegia, Receptive Expressive
Language Test-3 for speech assessment, etc.)
** Neurological exam after 2-year visit includes Amiel-Tison or other.

Figure 2: Schedule of visits prior (A) and after (B) implementation

The journey in blue represents the schedule before guideline implementation, the
red one after. The cognitive, motor and language domains of the Bayley Scales
of Infant and Toddler Development Third edition are administered. Standardized
neurological exams modified from the Amiel Tison27 are performed at 22-26
months on research patients by medical providers trained and certified on an
annual basis by a gold-standard per NRN procedures17.
TIMP: Test of Infant Motor Performance
NRN neuro: Neonatal Research Network neurological exam
Bayley III: Bayley Scales of Infant and Toddler Development Third edition
CBCL: Child Behavior Checklist
HINE: Hammersmith Infant Neurological Examination
GMA: General Movements Assessment

Page 18 of 30
Figure 3: Implementation timeline and phases
The initial 3 phases of the implementation process occurred over the course of
12 months. A consolidation phase with reassessment of strengths and
weaknesses offered the opportunity of a new development phase, planning for
priorities of the next implementation cycle.

Page 19 of 30
Table 1: Clinic visit numbers and diagnosis metrics for implementation
07/14-05/15 07/16-05/17
Total visits in clinic ≤12
2287 1999
months*
0-2 months 1022 593

3-4 month visits 255 4991

5-12 month visits 1010 907

Visits ≤12 m onths in clinic w ith

2 27
new cerebral palsy diagnosis

Mean age at cerebral palsy

18 (SD 7) 13 (SD 4)2
diagnosis in months, patients
range 11-32 range 4-29
still in clinic

Mean age at cerebral palsy

diagnosis in months, any
patients seen in clinic** 28 (SD 7) 16 (SD 7)

Total number of patients at

NCH with a diagnosis of
cerebral palsy under 3
years*** 70 175
SD: Standard Deviation
* number of visits over a 10-month period in all NICU Follow-Up clinics with
developmental testing
** includes patients either discharged from clinic before a diagnosis was made or
patients referred to other clinics where diagnosis was subsequently made.
*** does not reflect all patient with cerebral palsy at NCH, only those under three.
Incidence unchanged between periods.
1
Mc Nemar’s test, p<0.001
2
T-test, p< 0.001

Page 20 of 30
Table 2: Survey results for provider awareness
Median scores and p value for two-tailed Wilcoxon matched-pairs signed rank
test significance values are presented below
N 2014 2017 P value
How aware that cerebral palsy could 26 1 3 <0.001
be frequently diagnosed by 12
months
How aware of research interventions 22 0 3 <0.001
targeted to infants ≤12 m onths w ith
cerebral palsy
Therapist and Provider: awareness of 11 0 4 <0.001
GMA and HINE use
Nurse: awareness of sleep 5 0 2 0.05
importance for infants with cerebral
palsy
Dietitian: awareness of cerebral palsy 3 1 3 0.04
specific nutrition
Ranked items scale was 0= not at all; 1= very little; 2=somewhat; 3=very much;
4= always;

Page 21 of 30
Supplemental Table 1: Referral Criteria
Criteria Timing
Durable medical equipment: 2-4 weeks after discharge
G-Tube/feeding tube, tracheostomy, oxygen
Multiple congenital abnormalities (≥3 system s) and 3-4 months corrected age
no nutrition concerns
Multiple medication needs (≥ 3 m edications) 2-4 weeks after discharge
and no nutrition concerns
Multiple feeding issues (i.e.: persistent emesis with 2-4 weeks after discharge
Failure to thrive requiring multiple changes and 2-4 weeks after discharge
admissions
Birth at less than 27 0/7 weeks 2-4 weeks after discharge
Congenital abnormalities of the central nervous 3-4 months corrected age
system
Birth at less than less than1500 grams or less than 3-4 months corrected age
32 weeks
Neonatal encephalopathy not meeting hypothermia 3-4 months corrected age
criteria
Intraventricular hemorrhage Grades 3-4 or deep 3-4 months corrected age
medullary venous thrombosis or periventricular
leukomalacia
Congenital heart defects that are not single ventricle 3-4 months corrected age
defects
Extracorporeal membrane oxygenation 3-4 months corrected age
Neonatal encephalopathy requiring hypothermia 4 weeks after discharge
Seizures 4 weeks after discharge
Treated neonatal abstinence syndrome 1-2 weeks after discharge
Intrauterine growth restriction 2-4 weeks after discharge
Modular components in formula 2-4 weeks after discharge
High calorie formula >26K 2-4 weeks after discharge
Feeding allergy 2-4 weeks after discharge

Page 22 of 30
Supplemental Table 2: Providers roles and responsibilities in 2014 vs. 2016
vs. development phase
Provider 2014 2016 New

Occupational Identification of GMA

and physical current therapy administration
therapist services Specialized
TIMP and Bayley III standardized
administration assessments:
Referrals to state early ITSP,
intervention services QUEST,
Referrals to therapy AIMS, GMFM
services 66
Clinical therapy
evaluations

Advanced Attend 3/4 of all HINE cerebral palsy

nurse patient visits administration web-based
practitioners/ Practice in the NICU Referrals for education
general or the NICU follow-up therapy materials
pediatricians clinic for a minimum of services
2 years Counseling
Administer the on diagnosis
standardized research cerebral palsy
exam for the neonatal booklet
research network at
the 2-year visit
Referrals to
subspecialty services
Diagnostic
surveillance (imaging,
labs)

Developmental Care for complex cerebral palsy cerebral palsy

Pediatrician patients at or after two booklet web-based
years of age education
Provide neurological materials
assessment and
partnered with speech
therapist for
assessments
Spasticity medication
management
Orthotics prescription
Diagnostic

Page 23 of 30
 surveillance (imaging,
labs)

Pediatric See infants from 0-2 cerebral palsy

Neurologist years of age who have web-based
received hypothermia education
from neonatal materials
encephalopathy
Partner with nurse
practioners or
pediatricians
Counseling on
neuroimaging,
neurological exam,
seizure medication
management,
diagnosis
Diagnostic
surveillance (imaging,
labs)

Nurses Acquire vitals and Assess Sleep and pain

growth data parental education
Perform a thorough awareness
preliminary intake with and comfort
a review of systems level with of
and parental cerebral palsy
developmental and diagnosis
medical concerns
Assess current sleep,
feeding and nutrition
routines, pain (chronic
or acute), medication
use, and family social
and emotional
stressors

Dietitians Track growth using Use cerebral Targeted

World Health palsy growth supplementation
Organization charts charts with
for weight, height, GMFCS
head circumference levels
and weight-for-height
adjusting for
prematurity as needed

Page 24 of 30
 Recommend plan
adjustments for
over/underweight,
specialized dietary
needs

Social Perform social, Coordination GMA with

Workers emotional, community of care from cramped
needs assessments the NICU for synchronized in
Screen for mental higher-risk the NICU meets
health problems families high-risk
Coordinate services category,
with state programs regardless of
for infants and parents imaging
Provide resources as
needed and available

Speech Not available at 12 REEL3, Neonatal Oral

Language months or below McArthur Motor
Pathologist inventory Assessment
AAC Scale
assessment
Clinical
Feeding
evaluation
Infant/Toddler Sensory Profile (ITSP); Quality of Upper Extremity Skills Test
(QUEST); Alberta Infant Motor Scale (AIMS); Receptive Expressive Emergent
Language (REEL3); Augmentative and Alternative Communication (AAC); Gross
Motor Function Classification System (GMFCS)

Page 25 of 30
Supplemental Table 3: Balancing measures - Family satisfaction with
general clinic function
Period How well you were Healthcare Overall
kept informed worker’s concern satisfaction
about delays for your questions with your visit
and worries
regarding your
child’s care
Pre Good/Very Good 398 412 416
Rating
Total Question 415 419 426
Responses
Satisfaction Score 96% 98% 98%
Post Good/Very Good 302 199 324
Rating
Total Question 312 327 329
Responses
Satisfaction Score 97% 100% 98%

Page 26 of 30
Supplemental Table 4: Hip surveillance in infants with cerebral palsy 12
months and under at NCH
These represent the consensus statements of pediatric orthopedic surgeons, the
Comprehensive Cerebral Palsy Program and the NICU Follow-Up Program
Consideration 1 exposure of developing infants to unnecessary radiation is
unacceptable
Consideration 2 Hip surveillance in infants with cerebral palsy is critical to later
health and function

Consensus The first set of hip X-Rays will be ordered at 12 months

guidelines corrected age in children with a diagnosis of cerebral palsy.
Hip X-rays will only be ordered at 6-9 months corrected age if
one of the following conditions exists:
1- an adductor angle of 80 degrees or less;
2- scissoring of the lower extremities on vertical
suspension;
3- lower extremity permanent contractures
4- receiving baclofen for treatment of severe spasticity of
the lower extremities.

Page 27 of 30
Figure 1.

Page 28 of 30
Figure 2.

Page 29 of 30
Figure 3.

Page 30 of 30