Parental Experience in the Single Ventricle Interstage Utilizing a mHealth Innovation: A Comparative Case Study

Background Nearly 2,000 infants with single ventricle cardiac disease are born each year in
the United States, and have near certain mortality without surgical intervention. The heart
with this congenital disease can never be converted to a four-chambered heart but rather, a
palliative three-staged surgical strategy has been designed to maintain both circulation to
the body and lungs without a pump. After an initial neonatal surgery, most infants are
discharged home and return for a second surgery around six months of life. The time occurring
between surgeries is known as the interstage period. This interstage period is very high risk
and high demand time for parents, and there is a mortality rate during the interstage ranging
from 2-20%, often from unknown reasons.

Hypoplastic left heart syndrome (HLHS) is a specific type of single ventricle disease that is
one of the most complex forms of congenital heart disease. Publications have focused on
improving the care of infants with HLHS using a Quality Improvement Model with the National
Pediatric Cardiology Quality Improvement Collaborative (NPCQIC) through reduced variation,
best practices, and transparency. The concept of interstage home monitoring to reduce
mortality also was primarily focused on infants with only HLHS until recently when the focus
was made onto comprehensive care from birth to 1 year of age. Hemodynamic data such as oxygen
saturation, heart rate, intake, and overall status from a parental perspective were used to
promote communication and attempts of early recognition of deterioration in this high-risk
population. A strong set of qualitative studies have come from Rempel et al. in Canada on a
cross-section of mainly white married parents of infants with HLHS and their parenting
experience. Parenting under Pressure and the Facets of parenting a child with HLHS were
studies which used the grounded theory approach and provided substantial additions to our
knowledge of the interstage HLHS parenting experience. The basis of much of the care for all
types of single ventricle cardiac disease at home is found from the care of HLHS and applied
to infants with all types of single ventricle anatomy.

Most hospitals with pediatric cardiac surgery programs have interstage home monitoring
programs for high risk infants with single ventricle heart disease to provide care and
support for infants with all types of single ventricle cardiac disease. Education for
caregivers of infants with single ventricle disease include guidelines to call health care
providers (HCPs) in cases of hemodynamic interstage concerns, and symptom management
questions, based on home monitoring of oxygen saturation and weight gain. There are
variations in the staffing structure between pediatric hospitals but the programs often
include care coordination and education by a nurse or advanced practice nurse communicating
with parents in a reactive model waiting for data to be reported.

mHealth is defined through the National Institutes of Health (NIH) as the use of mobile and
wireless devices to improve health outcomes, service, and research. The use of applications
is widespread in adult care with estimated 40,000 health-care applications available. Some
studies in pediatric medical homes have shown association of mHealth with adherence and
self-monitoring to improved outcomes. mHealth innovations like the Cardiac High Acuity
Monitoring Program (CHAMP) and other applications work most effectively when caregivers and
interstage programs work together to improve care and are a focus of future research from the
National Institute of Nursing Research. Much of the recent interstage literature focuses on
mortality and weight measures with little focus on expanding types of outcomes to the
parental and interstage teams' perspectives and phenomena like infant development.

Recent developments in technology can provide a proactive means for data transfer for home
symptom monitoring so the interstage nursing team can review trends and provide proactive
monitoring for infants in the interstage. Since 2012, two pilot studies have been published
illustrating varying types of mHealth for the interstage with outcomes of improved somatic
growth and an improved system of interstage data review but did not show significant
mortality improvements. An innovative mHealth technology, CHAMP, was begun in 2014 for
interstage home monitoring symptom management and found improved interstage mortality
outcomes compared with historical controls. Research gaps remain in the role of adherence in
this population to the mHealth technology and the parental experience of utilizing these
innovative methods for communication with the clinical team.

Rationale The purpose of this study is to provide a rich description of the parental
perspectives of utilizing a mHealth application for data transfer during the single ventricle
interstage monitoring period for infants with congenital heart disease. The use of the case
study design in healthcare has been encouraged by Robert Yin because it adapts to the
complexity of interactions in a system that may be rapidly changing. Yin's qualitative case
research "design propositions", presented below for this study, are applied to the literature
and the PI's professional experience to focus the scope of the study and improve feasibility
of completion of this qualitative case study approach research study. Propositions in this
type of qualitative design are similar to hypothesis in quantitative designs to help
formulate themes emerging from the data. The use of propositions may help with contributions
for the body of knowledge after generalizing case study analysis, especially in cases of
critical or extreme cases. Comparative analysis of the theoretical target (non-adherent
parents) and rival cases (adherent parents) within the case study are improved with study
propositions to guide the organization of data.

Study Propositions:

1. The parental perspectives of the interstage period will influence the use of a home
monitoring mHealth technology

2. Parental perception of non-adherence of CHAMP and communication to the clinical team
will match the actual non-adherence patterns of the mHealth innovation

3. The use of Cardiac High Acuity Monitoring Program (CHAMP) application from the parental
perspective will benefit future improvements in this technology

4. Parents of infants with single ventricle will report the interstage report as a
stressful time

Research Design

Methods. The proposed research study uses a comparative case study methodology, utilizing
multiple sources of data to deeply examine 8-10 "cases," each of which is defined as
parent(s) of a child (< 5 years of age) with single ventricle disease who used the mHealth
application. One group of cases (4-5) will have used the mHealth application less than
recommended (non-adherent to the home monitoring recommendations) or declined to use the
application and returned it before the end of the interstage period. The other group of
comparison cases (4-5 cases) will have used the application as recommended by the clinical
team (adherent) and continued doing so through the interstage period.

Yin defines various types of cases, including, explanatory or causal case studies,
descriptive case studies, and exploratory case studies. The cases in this study are
exploratory in that they focus on "what" can be learned from these cases of using a novel
mHealth innovation and the CHAMP care team as a unit. Across the U.S., nearly 2,000 infants
are born with single ventricle cardiac disease each year but the actual experience at home is
difficult to comprehend. The research design is a multiple case study design with embedded
units of non-adherent and adherent cases of parental use of CHAMP within the larger case
group through the CHAMP Clinic at Children's Mercy Kansas City.

Data sources will include semi-structured interviews with parents that include observations
of simulated home monitoring equipment and processes that had been previously used including
location, time of day, and frequency.

Inclusion Criteria:

1. Adult over 18 years of age parent(s) of children with single ventricle cardiac disease

2. Parent(s) of children were discharge home in the interstage period from December 2013
to May 2017 that have completed the interstage period with Stage II Glenn cardiac
surgery as of June 8th, 2017.

3. Parents(s) of children who were followed in the CHAMP clinic at Children's Mercy.

4. Parent(s) of CHAMP children who had the CHAMP mHealth application (CHAMP App)
available for transfer of home monitoring data during the interstage

Exclusion Criteria:

1. Non-English speaking parent(s)

2. Parent(s) of children who were not discharged interstage