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Pediatric Neurology
j o u r n a l h o m e p a g e : w w w. e l s e v i e r. c o m / l o c a t e / p n u
Original Article
ABSTRACT
BACKGROUND: Cerebral palsy is the most common physical disability in childhood, and is mostly diagnosed after
age 2 years. Delays in diagnosis can have negative long-term consequences for children and parents. New guide-
lines for early cerebral palsy diagnosis and intervention were recently published, after systematic review of the
evidence by international multidisciplinary experts aiming to decrease age at diagnosis. The current study tested
the feasibility of implementing these guidelines in an American clinical setting. METHODS: We designed a step-
wise implementation process in a neonatal intensive care follow-up clinic. Efficacy was tested by comparing 10-
month pre- and post-implementation periods. Clinic visit types, cerebral palsy diagnosis, provider competencies
and perspectives, and balancing measures were analyzed. RESULTS: Changes to infrastructure, assessments, sched-
uling algorithms, documentation and supports in diagnosis or counseling were successfully implemented. Number
of three- to four-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 diagnosis and interventions increased (P < 0.001). There was
no decrease in family satisfaction with overall clinic function. Opportunities for improvements included documen-
tation for transitioning patients, generalizabilty across hospital clinics, systematic identification of high-risk status
during hospitalization, and need for cerebral palsy care guidelines for infants under age 2 years. CONCLUSIONS: We
demonstrated for the first time in a US clinical setting the feasibility of implementation of international early di-
agnosis and treatment guidelines for cerebral palsy. This process is adaptable to other settings and underscores
the necessity of future research on cerebral palsy treatments in infancy.
Keywords: infant, cerebral palsy, early intervention, diagnosis
Pediatr Neurol 2017;76:66–71
© 2017 The Authors. Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license
(http://creativecommons.org/licenses/by-nc-nd/4.0/).
0887-8994/$ — see front matter © 2017 The Authors. Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license
(http://creativecommons.org/licenses/by-nc-nd/4.0/).
https://doi.org/10.1016/j.pediatrneurol.2017.08.002
R. Byrne et al. / Pediatric Neurology 76 (2017) 66–71 67
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 three- to four-month visits. 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, a 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 nine 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 three- to four-
month visits 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. * Select examinations 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 as-
sessment, etc.). ** Neurological examination after 2-year visit includes Amiel-Tison or other. Bayley III, Bayley Scales of Infant and Toddler Development
Third Edition; CBCL, Child Behavior Checklist; CO, cerebral palsy; GMA, General Movements Assessment; HINE, Hammersmith Infant Neurological
Examination; TIMP, Test of Infant Motor Performance. (The color version of this figure is available in the online edition.)
and intervention for cerebral palsy,10 based on a systematic infant populations with common identifiable factors for ce-
of evidence for why and how cerebral palsy should be rec- rebral palsy: preterm infants and term infants with neonatal
ognized as early as possible. 11 The guidelines include encephalopathy. Many children with less common risk factors
recommendations for neuroimaging, neurological exami- for cerebral palsy also receive care in such programs due to
nations, and motor assessments for infants below five months medical complexity in the perinatal period. The goal of our
corrected age and between five and 24 months corrected age. study was therefore to test the effectiveness of an imple-
They define the concept of “high-risk for cerebral palsy” at mentation process for clinical use of the early diagnosis
three to four months, a category in which infants do not yet guidelines10 and apply them to a NICU follow-up clinic
receive a diagnosis, but due to a constellation of clinical factors, setting, where a large population of high-risk infants is rou-
neuroimaging findings and standardized assessments, have tinely monitored. We hypothesized that comparison of
a 95% probability of developing cerebral palsy in the fol- quantitative metrics for clinic visits, diagnoses, and provid-
lowing years. Best current evidence for counseling is based er training competency would demonstrate improvements
on published parent surveys and high-quality research. The before and after implementation. We complemented our
guidelines can be translated into clinical practice by adapt- study with balancing measures of parent satisfaction and
ing core elements to suit individual clinical settings illustrated qualitative data on provider perceptions.
in Fig 1 (setting is a neonatal intensive care unit (NICU) follow-
up program with applied examples of the guidelines). Methods
Although research programs leveraged the evidence base
for early detection of cerebral palsy,12-14 no clinical pro- We analyzed data under an exempt determination from the Nation-
grams implemented the guidelines in their entirety as wide Children’s Hospital Institutional Review Board (IRB17-00409) due
standard care. NICU follow-up programs routinely follow two to the use of unidentified data.
68 R. Byrne et al. / Pediatric Neurology 76 (2017) 66–71
Infrastructure
The electronic medical record was on an Epic platform, commonly
used by large US health systems to access, organize, store, and share
patient medical records. Therapist documentation of Bayley Scales of
Infant and Toddler Development Third Edition16 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 FIGURE 2.
heterogeneous, reflecting personal choice and occasional practice in other Schedule of visits prior (A) and after (B) implementation. The journey
clinics. Clinic scheduling templates were based on therapist schedul- in blue represents the schedule before guideline implementation, the
ing availability and medical practitioners rarely had assigned patients. red one after. The cognitive, motor, and language domains of the Bayley
Scales of Infant and Toddler Development Third edition are adminis-
Visit schedules, assessments, and referral tered. Standardized neurological examinations modified from the
Pre-implementation, schedules followed very preterm infants at three Amiel-Tison17 are performed at 22 to 26 months on research patients
to four months, nine to 12 months, and 22 to 26 months, with interim by medical providers trained and certified on an annual basis by a gold-
visits based on specific therapist- or dietitian-identified needs (Fig 2). standard per NRN procedures.18 Bayley III, Bayley Scales of Infant and
Assessments were guided by participation in the Neonatal Research Toddler Development Third Edition; CBCL, Child Behavior Checklist; GMA,
Network18 as well as a strong evidence-based practice group in the General Movements Assessment; HINE, Hammersmith Infant Neuro-
therapy department. In addition, specialized providers examined those logical Examination; NRN neuro, Neonatal Research Network neurological
patients with high neurological complexity. Therapist assessments often examination; OT, occupational therapist; PT, physical therapist; SLP,
determined referrals for therapy services, whereas medical providers speech and language pathologist; TIMP, Test of Infant Motor Perfor-
made the referrals to subspecialty providers and programs (audiology, mance. (The color version of this figure is available in the online edition.)
ophthalmology, physical medicine, developmental pediatrics, psychol-
ogy, complex care, cerebral palsy) or for procedures based on their clinical
judgment and a developmental algorithm for the two-year visit. was accomplished by Prechtl’s Trust certified trainers. Clinical compe-
tency in the Hammersmith Infant Neurological Examination (HINE) was
Providers a novel implementation process published recently.19 Training of medical
The NICU follow-up team was multidisciplinary and diverse. providers in scope of practice of various therapy disciplines, available
Supplemental Table S2 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 Re-
search Network studies requiring follow-up of two-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 incor-
porate new and proven interventions for cerebral palsy and to promote
a culture of change into the setting.
services, and best evidence for occupational, physical, speech and feeding TABLE 1.
therapies in infancy was accomplished by a series of self-directed modules Clinic Visit Numbers and Diagnosis Metrics for Implementation
designed by clinical therapy leaders. Dietitians utilized first World Health
7/14 to 5/15 7/16 to 5/17
Organization curves for infants,20 then the cerebral palsy-specific growth
curves21 based on Gross Motor Function Classification System levels. Doc- Total visits in clinic ≤12 2287 1999
months*
umentation in Epic was modified to match the new visit schedule, driven
0 to 2 months 1022 593
by providers practicing regularly in the clinic. Clinic templates were
3 to 4 month visits 255 499§
switched to a provider-based system, allowing continuity of medical care 5 to 12 month visits 1010 907
and neurological trajectories as well as longitudinal perspective from Visits ≤12 months in clinic 2 27
the team leaders who could request specific therapy assessments based with new cerebral palsy
on new knowledge gained in the modules. Visits before three months diagnosis
were scheduled for those with the greatest risk factors and complexi- Mean age at cerebral palsy 18 (SD 7) 13 (SD 4)||
ty. Research was integrated through additional workspace, training of diagnosis in months, range 11 to 32 range 4 to 29
coordinators, improved communication with nursing and social work patients still in clinic
Mean age at cerebral palsy 28 (SD 7) 16 (SD 7)
leaders, and visibility of rehabilitation study materials.22,23
diagnosis in months, any
Parent support was addressed through counseling with the ele- patients seen in clinic†
ments recommended in published literature and the guideline mandates: Total number of patients 70 175
“face-to-face, with both caregivers present if appropriate, private, honest, at NCH with a diagnosis
jargon-free, with empathic communication tailored to the family.”4 Iden- of cerebral palsy under 3
tification of strengths and goals was included, and questions were fostered years‡
by allowing the diagnosing provider to return at conclusion of the visit Abbreviations:
to reassess the emotional context and answer additional questions. NCH = Nationwide Children’s Hospital
MyChart use was explained and encouraged. Referral to services was SD = standard deviation
complemented by a set of written materials (therapist handouts and * Number of visits over a 10-month period in all NICU follow-up clinics with de-
parent educational booklet).24 velopmental testing.
† Includes patients either discharged from clinic before a diagnosis was made
Implementation occurred over one month, during which process im-
or patients referred to other clinics where diagnosis was subsequently made.
provement guides were available to observe, troubleshoot, record ‡ Does not reflect all patients with cerebral palsy at NCH, only those under age
challenges, and provide support if necessary. Written feedback was also
3 years. Incidence unchanged between periods.
solicited. § McNemar test, P < 0.001.
Consolidation involved further observation and support in areas iden- ||
t Test, P < 0.001.
tified 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 pri-
orities for the next cycle in clinical, operational, and research domains
during regular provider meetings. Intra-hospital and community out-
reach were added as new priorities. point. The number of children in the clinic diagnosed by 12
months increased, with the age at diagnosis decreasing sig-
nificantly. In addition, changes in the clinic were reflected
Part three—evaluation metrics
by a decreased mean age at diagnosis in the overall popu-
lation of children with diagnoses of cerebral palsy at the
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
hospital under age 3 years.
prevent the possibility of crossover effects from early phases of imple-
mentation. Balancing measures were obtained using standard anonymous
feedback clinic questionnaires at the end of each visit from any parent
Provider metrics
in the clinic, not only those of children with cerebral palsy. The goal of
these measures was to determine whether implementation of the Provider metrics (Table 2) demonstrated significant im-
program negatively impacted parent satisfaction through three ques- provement of awareness of best practices around early
tions about overall perception of clinic function. Provider perspectives diagnosis and early interventions for infants less than 12
on cerebral palsy recognition and intervention for those involved in the months. When providers were asked to describe what they
care of infants 12 months and under were obtained through a survey
had learned post-implementation, five major themes
scored on a five-item ranked scale.
emerged: optimizing developmental outcomes for chil-
dren, improvement of support to parents, awareness of
Results assessment tools, effective rehabilitation interventions, and
importance of early detection. Balancing measures of parent
Clinic metrics
satisfaction with clinic processes remained high and un-
changed (Supplemental Table S3).
The proportion of preterm infants seen in the clinic was
not significantly different between the two periods (85.7%
versus 76.6%, P = 0.9). The incidence of cerebral palsy was Statistical analysis
unchanged pre- and post-implementation of the guide- Continuous variables were analyzed using two-tailed t
lines in the hospital system.19 The average age of diagnosis tests. Distributions of visit types pre-implementation versus
of cerebral palsy was decreased from age 18 months to 13 post-implementation were compared using McNemar test
months19 (Table 1). Number of three- to four-month visits with alpha = 0.05. Scores on scales using ranked items were
increased compared with other types of visits (due to the analyzed using two-tailed Wilcoxon matched-pairs signed-
decreased clinic load of new patients and increased focus rank test, with with alpha = 0.05. All calculations were
on three- to four-month visits), resulting in more children performed using SPSS software (IBM Corp. Released 2013,
having GMA and HINE assessments performed at this time IBM SPSS Statistics for MacIntosh, Version 22.0, Armonk, NY)
70 R. Byrne et al. / Pediatric Neurology 76 (2017) 66–71
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