Case Report
Kathryn C. Nesbit, PT DPT DSc 1‡, Laura Greiss Hess, PhD OTR/L2‡, Shivani Mahuvakar, BA3, Christine K. Fox, MD MAS4, Christine Mrakotsky, PhD 5‡*; on behalf of the Investigators of the AHA-Bugher Pediatric Hemorrhagic Stroke Research Center of Excellence
‡K.C.N, L.G.H., and C.M. contributed equally
1 Graduate Program in Physical Therapy, University of California San Francisco/San Francisco State University, San Francisco, California, USA
2 Department of Occupational Therapy, Dominican University of California, San Rafael, California, USA
3 Division of Pediatric Neurosurgery, University of California San Francisco, San Francisco, California, USA
4 Departments of Neurology and Pediatrics, University of California San Francisco, San Francisco, California, USA
5 Departments of Psychiatry and Neurology, Center for Neuropsychology, Boston Children’s Hospital, Harvard Medical School, Boston, Massachusetts, USA
Corresponding Author: Dr. Christine Mrakotsky, Boston Children’s Hospital, 300 Longwood Avenue, Boston, MA 02115, USA; christine.mrakotsky@childrens.harvard.edu
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Abstract
Hemorrhagic stroke accounts for half of incident strokes in children yet remains understudied. Clinical and research teams assessing recovery from pediatric hemorrhagic stroke are limited because commonly used outcome measures may not be meaningful to the care team, educators, and foremost the child and family. The current paper serves as a model for bringing together multiple disciplines for shared purposes of better assessing child recovery and development after hemorrhagic stroke over time, advocating for child- and family-centered care, and facilitating discourse in the community of those caring for children with pediatric hemorrhagic stroke. The purpose of this work is to describe our guiding principles and the process of our team’s development of a toolkit that emphasizes inclusion of the patient and family perspective of recovery in the home, school, and community contexts. We provide the toolkit and an example of specific outcome measures selected to address unique aims of a research project. The toolkit is intended to be a living platform for further evolution of outcome measures for pediatric hemorrhagic stroke. As such, we hope it will be a dynamic document that serves the ongoing and future clinical and research needs of the multidisciplinary professional teams providing holistic care after a pediatric hemorrhagic stroke.
Introduction
Stroke during childhood has the potential to cause permanent disabilities, resulting in new or emerging cognitive challenges, motor deficits, psychosocial impacts, and impairment in bodily functions.1 While outcomes after pediatric stroke are variable, resultant deficits often have a significant impact on a child’s overall function and can impede ability to perform age-appropriate activities of daily living and social participation. These disabilities may change school function, with an impact on learning and academic performance. An estimated one-third to one-half of children with stroke later require assistance in school and/or attend a specialized education program.2 A central focus in rehabilitation after childhood stroke is re-integration into the child’s natural environments (academic, social, family), and particularly return to school.3,4
The field of pediatric stroke research began in the early 2000’s and has benefitted from international and multidisciplinary collaboration.5,6 Historically, a majority of this international collaborative research has focused on ischemic rather than hemorrhagic stroke. Yet these stroke types differ in etiology, acute management, and approach to secondary stroke prevention. Although lessons learned from ischemic stroke research may inform studies of outcome in hemorrhagic stroke, there are unique characteristics of hemorrhagic stroke that need to be considered. For example, hemorrhagic stroke may result in higher burden of disability (neurological and psychological) as measured by Disability-Adjusted Life Years,7 and in higher mortality rates than ischemic stroke.8 However, study of outcomes of hemorrhagic stroke across domains and contexts, particularly in the long term, is limited. Care models for pediatric hemorrhagic stroke require a collaborative effort of multiple disciplines including: neurology; neurosurgery; neuroradiology; interventional radiology; physiatry and rehabilitation medicine; neuropsychology and psychology; occupational, physical, and speech/language therapy; and social work. A multidisciplinary review and discussion of pediatric stroke outcome measures with a focus on hemorrhagic stroke are important for the advancement of pediatric stroke care.
Purpose, Goals and Objectives
The purpose of this work is to describe our multidisciplinary team’s process of generating a toolkit of outcome measures that highlight the child and family perspectives of recovery from hemorrhagic stroke over time in real-life contexts, including home and school. Reliable and valid outcome measures specific to hemorrhagic stroke are lacking. While the few outcome measures available and validated for ischemic stroke have the potential to capture some of the unique aspects of hemorrhagic stroke recovery as well, none are validated for patient centered or patient reported outcomes in either population. We initially aimed to develop a relatively narrow battery of outcome measures to address research questions for a specific pediatric hemorrhagic stroke study. During that process, we found that few outcome measures were validated for or used in pediatric hemorrhagic stroke research. Those that were available were focused on disability ratings that did not capture how well or poorly children interacted within their family and community. Assessment of child and family centered outcomes is, however, an important component of care management for multidisciplinary teams.
Patient and Family Centered Outcome Measures
The World Health Organization defines outcome as a “change in the health of an individual, group of people, or population that is attributable to an intervention or series of interventions.”9 After a change in health, outcome measures are used to assess current level of function, monitor changes in the condition over time, and prognosticate future level of function.10 Outcome assessment provides critical data to track recovery and development across patients and over time. According to the framework of the International Classification of Functioning, Disability and Health, outcome measures may be focused on impairments, activity limitations, and/or participation restrictions.11 Each of these domains of outcome assessment may include specific areas such as cognitive (intellectual, language, visual-perceptual, executive functions), social-emotional, motor, adaptive functioning, as well as quality of life. In developing our toolkit, we first sought to include a breadth of outcome measures that could address each of these domains and measure recovery within each domain.
Outcome measures are meaningful to patients, families and clinicians when they relate to a child’s real-life contexts such as home, school, or community. Therefore, outcome assessments conducted in research and clinical settings alone may not be relevant or applicable to recovery in real-life contexts. Examples of child- or family-informed and contextually anchored outcome measures include the Participation and Environment Measure for Children and Youth (PEM-CY)12, and the Behavior Rating Inventory of Executive Functions (BRIEF/BRIEF2) 13,14, which includes versions for child, parents and teachers as informants. We considered inclusion of patient-or family-centric outcome measures informed by the child, parents, and teacher perspectives as essential components of the toolkit to improve clinical care and ongoing research.
Pediatric Stroke Outcome Measures- Current Knowledge and Gaps
Pediatric stroke rehabilitation should focus on recovery that optimizes health and uses integrative multidisciplinary approaches.3,15,16 The outcomes measures should thus be clinically meaningful to a multidisciplinary stroke care team (medical, behavioral health, rehabilitation), educators, and foremost the child and family.
A systematic review by Yale et al.17 examined pediatric stroke outcomes. After analysis of 116 studies that utilized 95 unique outcome measures, the authors noted the most frequently used outcome measures focused on global disability or motor impairments and served a specific medical purpose. Their synthesis of the characteristics of these 95 measures revealed a paucity of stroke outcome measures focusing on behavioral and cognitive function, patient and family perspectives, real-life contexts (e.g., home, school, community), and quality of life. In addition, the authors found a lack of reliable and valid outcome measures specific to children with hemorrhagic stroke and infrequent use of outcome measures to monitor recovery over time. This systematic review provides a description of characteristics and shortcomings of outcome measures in current use; however, it does not include recommendations for specific instruments in pediatric stroke care.
In another recent review, Felling et al.18 suggested a roadmap for outcome assessment and management during the different phases (acute, subacute, chronic) after pediatric stroke. The authors identified critical information to be gathered at each stage. In the acute phase, indicators of global function are recommended. During the subacute phase, additional screening in motor, language, cognitive, behavior and emotional domains should be completed. During the chronic phase, comprehensive neuropsychological evaluations repeated over time are recommended.18 This review provides general guidance about types of outcomes at different stages after pediatric stroke, although did not recommend specific outcome measures other than the Pediatric Stroke Outcome Measure (PSOM). While the PSOM is a useful rating scale for clinicians, it cannot be easily used to describe outcomes to children or families.
In a related review, Feldman et al.19 described the quality and feasibility of most frequently used pediatric stroke outcome measures in seven domains (global performance, motor, cognitive, language, behavioral/emotional, and adaptive function, as well as quality of life) and proposed a toolkit of measures that ranked high in quality of psychometric properties in each domain. However, the authors did not review whether the outcome measures included family/child perspectives or daily life contexts such as home, school and community. Krivitzky et al.20 reviewed recent advances in neuropsychological outcomes and interventions for pediatric stroke, with a focus on cognitive and socio-emotional outcomes. While the authors did not make recommendations for specific measures, they noted the need for more in-depth study of socio-economic and familial factors such as parental stress and resiliency as contributors to stroke recovery and outcome.
Together these recent reviews did not specifically address outcomes in pediatric hemorrhagic stroke, or address shortcomings in the current use of the outcome measures available. In particular, they did not prioritize patient and family perspectives or real-life context measures. Therefore, our team aimed to fill these gaps of prior studies and devise a comprehensive toolkit of pediatric stroke outcome measures for consideration in clinical and research applications.
The current paper serves as a model for bringing together multiple disciplines for the shared purpose of assessing a child’s recovery after hemorrhagic stroke over time, across real-life contexts, and advocating for child- and family-centered care. This model contributes to facilitating discourse in the community of those caring for children with pediatric hemorrhagic stroke. We aligned with guidelines that recommend a multidisciplinary team approach for assessment and monitoring of key domains of child development after pediatric stroke and other brain injuries.16, 21-23 We describe the process of developing a toolkit for pediatric hemorrhagic stroke outcome measures. The toolkit is intended to promote a multi-method, multi-informant approach of outcomes assessment that includes the child’s and family’s perspectives, and utilizes observations from different social contexts. This approach has been widely used in behavioral research and practice.24 We expect the toolkit to evolve with continued revision and input; therefore, this paper is intended to focus on the process of how the toolkit was developed and how it can continue to be useful for client-family care, clinical and research purposes. We intend this to facilitate discourse in the community of those caring for children with pediatric hemorrhagic stroke.
Method
Outcome Measures Task Force
An Outcome Measures Task Force was formed from the American Heart Association (AHA) Bugher Center of Excellence in Hemorrhagic Stroke research team. Our team drew together expertise from a total of four pediatric stroke centers and universities in the U.S. and consisted of 9 members, including 2 stroke neurologists, a neuroradiologist, a stroke neuropsychologist, a physical therapist, an occupational therapist, and 3 clinical research assistants. Each member has expertise in the areas of pediatric stroke, pediatric outcome measurement and child & family centered care. The purpose of the Outcome Measures Task Force was to identify outcome measures in a broad scope of domains that include assessment of home/school/community function through a patient-and family-perspective.
Toolkit Design and Process
The Outcome Measures Task Force developed the toolkit through an organized, iterative, and collaborative process of bi-monthly, multi-site video conference remote meetings and in-person discussions. Our team communicated frequently between meetings via email and generated shared folders of working documents and resources. Consensus building was grounded on respect for interprofessional collaboration, diverse perspectives, shared values, and a common vision for promoting best practices in pediatric stroke care. As new information arose, we returned to our initial goals and continually revised our work. As we gathered results from the outcome measures gathered for other projects in our research grant, we re-assessed our work. This process began in the Fall of 2020 and is ongoing. Our guiding principles focused on a holistic approach with the patient and family as primary stakeholders. Although we did not gather input form patients and families directly during the design process of the toolkit, part of the future directions for our research includes a qualitative study of patients’/families’ perspectives.
Other fields have documented team process approaches to establish toolkits to address clinical or research gaps.25,26 We used these models to address the shortcomings in the current literature. We began by first identifying a broad set of pediatric hemorrhagic stroke outcome measures and then creating a toolkit for clinicians and researchers that describes the pertinent characteristics of each measure. Our team’s process was focused on the unique considerations of recovery from hemorrhagic stroke in partnership with children and families.
An overarching goal was to develop a broader resource of commonly used pediatric stroke outcome measures consistent with patient- and family-centered needs that could be used for both research and clinical purposes.
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Pediatric Hemorrhagic Stroke Outcomes Toolkit
