The Positive Impact of Journaling on Adolescents with Cystic Fibrosis
Sean Kow MD1, Brenda Rieger MD1,
Kimberly Morse LCSW1, Thomas Keens
MD1, Susan Wu MD1
1
Children’s Hospital Los Angeles, Los Angeles, CA
This work was supported by the Cystic Fibrosis Foundation [grant
CMHCO14-15].
keywords: cystic fibrosis, pediatric, adolescent, narrative therapy,
journaling
Correspondence:
Sean Kow, MD
Children’s Hospital Los Angeles. 4650 Sunset Blvd MS #76, Los Angeles,
CA 90027.
skow@chla.usc.edu
https://orcid.org/0000-0002-6748-7808
tele: (323) 669-2113
fax: (323) 720-5666
Abstract
Background
Individuals with cystic fibrosis (CF) often have psychological
difficulties on top of their medically complex care, such as anxiety,
depression, and medical mistrust. These have been shown to be associated
with worse adherence, pulmonary function test results, and other health
outcomes. In this pilot trial, we implemented a journaling program based
on narrative therapy methodology to improve mental and physical health
outcomes for individuals with CF.
Methods
Eight adolescents aged 12-17 with a confirmed diagnosis of CF followed
in a single center cystic fibrosis clinic were emailed weekly journaling
prompts that explored topics like: treatment adherence, feeling
different with CF, anxiety, depression, and interpersonal relationships.
Subjects were emailed surveys about their experience with the writing
assignment, and baseline health data was collected from the electronic
medical records.
Results
The average score for the Pediatric Symptom Checklist (PSC-17) decreased
by 5.5 points, and the post-study average (mean 23.5, SD 12.2) fell to
less than 28, which is the cutoff for screening positive for behavioral
or emotional problems. Participants reported the study was enjoyable and
had improvement in feelings of anxiety/depression. 100% of participants
responded “Strongly Agree” to the statement “I recommend other people
with CF to write about the topics from this study”.
Conclusions
The journaling intervention for individuals with CF was feasible and
well received. Initial results show improvement in PSC-17 and other
wellbeing measures. Further studies are needed to evaluate the impact of
journaling on mental health and disease outcomes.
Background
Individuals with cystic fibrosis often face many psychological stresses
on top of their medically complex care.1 These can
include depression, anxiety, feelings of isolation, risk-taking
behavior, and difficulty navigating relationships.2 In
one international study, adolescents with cystic fibrosis had depression
rates from 5-19%, and anxiety rate of 22%.3Adolescents with cystic fibrosis also are saddled with medically and
emotionally taxing management plans. This can put them at risk for
issues regarding medication adherence, treatment compliance, time
management, and poor relationships with their medical
team.4-6 These psychological and mental health issues
in patients with cystic fibrosis have been shown to be associated with
worse adherence, worse pulmonary function, increased hospitalization and
healthcare costs, and decreased health related quality of
life.3
A potential avenue to help address some of these issues is through
narrative medicine therapy. Narrative therapy is a journaling practice
that encourages objectification of the problem, making it possible to
experience an identity that is separate from the problem.7 Journaling and writing to explore psychologically
distressing issues have been shown to increase understanding of
self-awareness and understanding as well as provide the foundations for
potential positive cognitive change.8 Expressive
writing also has the potential benefits of promoting cognitive
processing, creating adaptive internal schemas, and reducing
physiological stress resulting from inhibition of negative emotions.9 There has been prior research done in adult
populations regarding expressive writing and chronic disease management
that have shown positive results in mental health, daily functioning,
baseline disease control, and other health measures .10-13 Specifically, there have been narrative medicine
and expressive writing interventions in patients with breast cancer and
coronary artery disease that have shown improvements in physical and
psychological health.14-16 Notably, data regarding
this intervention is not as well studied in pediatric population, and
even less so in those with chronic illnesses. Therefore, we developed a
feasibility trial that applied theories from narrative therapy to
structure a journaling therapy for adolescents with cystic fibrosis with
the goal of improving their mental and physical health outcomes.
Methods and Procedures
The IRB approved study was divided into three phases – the screening
phase, intervention phase, and follow up phase. [Figure 1] In the
screening phase, a maximum of eight participants were recruited using
verbal and written means of communication. Inclusion criteria included
participants who have diagnosed cystic fibrosis, participants who were
followed at the research institutions’ CF Center, age 12-17 at the start
of the screening phase, participants are able to read and write in
English at an age appropriate level, participants have the ability to
receive and send assignments electronically, including but not limited
to access to the internet and access to compatible devices (computer,
smart phone, etc.), and ability to have follow up for the duration of
the study intervention phase. Exclusion criteria included age
< 12 or > 17 years at start of screening phase,
inability to read or write English proficiently to allow participation
in the writing intervention, and lack of means to receive and send
assignments electronically. Participants all received their care at a
Children’s Hospital Los Angeles, a quaternary free standing children’s
hospital with a Cystic Fibrosis Center. We selected a maximum of eight
participants because the study was a trial to assess the feasibility of
the intervention and the qualitative feedback from participants. The
social worker of the CF clinic was responsible for screening,
identifying, and recruiting participants, including the consent and
assent process. Guardians filled out paper consents and minors filled
out paper assents. Information regarding the study was sent to
participants and their guardians electronically. The screening phase
concluded once eight participants were enrolled or at the end of two
months since the start of the screening phase.
The intervention phase started one week after the conclusion of the
screening phase. Baseline health data was collected from the
participants via the electronic medical record (EMR) as well the CF data
registry PortCF. Baseline information included gender and
ethnicity/race, age, weight, height, BMI, FEV1 % of predicted, number
of hospitalizations, number of respiratory infections in the last year,
and previous positive sputum culture species. Study data were collected
and managed using REDCap electronic data capture tools hosted at
Children’s Hospital Los Angeles. REDCap (Research Electronic Data
Capture) is a secure, web-based software platform designed to support
data capture for research studies, providing 1) an intuitive interface
for validated data capture; 2) audit trails for tracking data
manipulation and export procedures; 3) automated export procedures for
seamless data downloads to common statistical packages; and 4)
procedures for data integration and interoperability with external
sources. 17,18
Every week, a survey from REDCap was automatically distributed via email
to all participants. All surveys and data were stored in REDCap under
password, and only investigators had access to the data. These surveys
included a pre-study survey, eight weekly prompts, and a post-study
survey. The pre-study survey was created by the research team and
included questions regarding familiarity with narrative therapy, health
related quality of life, and the Pediatric Symptom Checklist. The
Pediatric Symptom Checklist is a validated psychosocial screen used to
evaluate and recognize cognitive, emotional, and behavioral
problems.19 Weekly prompts covered topics that
included: introduction to narrative therapy, living with cystic
fibrosis, treatment adherence, feeling different with cystic fibrosis,
anxiety, limitations, depression, and interpersonal relationships. To
encourage participation, if the participant did not want to answer the
topic, they were able to state that, offer an explanation why, and had
space to write about whatever they wanted instead of the topic sent to
them. Recurring questions were provided at the end of weekly surveys
regarding how much time was spent writing that week’s topic, how well
the participants liked that week’s topic, and how well the participants
felt they managed their CF that week. Post-study survey included the
same questions as the pre-study survey as well as questions regarding
how well the participants liked the study and other feedback.
As some topics could be foreseen to trigger negative emotions, responses
were reviewed weekly by the investigators for any content that could
indicate that the participants were in immediate danger from
psychological distress, such as suicidal or homicidal ideation. We
created a standard of procedures (SOP) for this in conjunction with the
cystic fibrosis social worker. If such content was found, the
investigators would contact participants through the CF clinic social
worker during normal business hours, the on-call social worker on off
hours, or national/local authorities. The surveys also had information
regarding who to contact if negative feelings were induced by
participating. We also let participants know what day of the week the
results would be reviewed. Upon completion of one survey, the subsequent
survey was distributed automatically at the start of the next week. If a
participant did not turn on the survey by the one week deadline,
automated reminders were sent, and participants did not receive
additional surveys until the previous week’s survey was completed. The
intervention phase concluded once all participants had attended a
routine quarterly clinic visit after completion of all eight weekly
assignments or at the conclusion of six months, whichever came earlier.
Participants were given compensation for participation from residual
funds from the CF Foundation (CFF) Mental Health Coordinator Grant
[grant CMHCO14-15]. Participants were offered their choice of an
incentive item (meditation board, mindfulness journal) at the completion
of the fourth weekly prompt, and a choice of gift card ($25 to
Footlocker or iTunes) at the completion of all eight weekly prompts.
The follow up phase lasted up to three months from the end of the
intervention phase. Additional health data was collected to compare to
the baseline health data collected at the beginning of the intervention
phase. These data included weight, height, BMI, FEV1 % of predicted,
number of hospitalizations and infections in the last three months, and
previous positive sputum culture species. We also obtained written
feedback on the participants’ experiences in the study.
Analysis
Data analysis was done via Microsoft Excel. Due to this being a pilot
study for feasibility, data analysis for statistical power or
significance was not pursued. Qualitative analysis was not pursued for
this pilot study. Sample size calculation was done for an independent
t-test using data from our initial study results in order to gain
insight into future numbers needed for statistical
significance.20
Results
A total of eight participants were recruited for the study. Our patient
population included participants with varying degrees of mental health
history and disease control. [Table 1] Three participants were
actively receiving mental health services, two had a history using
mental health services but were not currently. One participant was
healthy enough to have every other quarterly visit be telehealth and
only required PFT monitoring twice a year per the CF clinic policies.
One participant frequently had inpatient admission for CF exacerbations.
All but one participant had a history of multiple positive sputum
cultures. Not exclusively, six participants had positive sputum cultures
for MSSA, one with MRSA, six with pseudomonas, two with aspergillus, one
with mycobacteria, and one with achromobacter.
In response to the question “Are you limited in any way in any
activities because of any impairment or health problem?”, before
completion of the study, two of the four participants who completed the
study responded “Yes”, one responded “Don’t Know/Not Sure” and one
responded “No”. After completion of the study, two of the four same
participants responded “No”, and two responded “Don’t Know/Not
Sure”. When asked “What other topics do you wish you were able to
write about?”, participants responded, “What our relationships or
friendships are like with CF” and “More positive things about my CF
about how you can have a healthy outlook on it or how there may be some
things that CF patients have an advantage in”. When asked “What things
did you not like, or would you change about the study?”, participants
responded that they felt a time pressure to complete the prompts within
one week and two participants (50%) specifically mentioned wanting to
write about more positive topics during the study. When asked “What
things did you like, or would you keep the same about the study?”,
participants responded “the week we have to turn it in”, the topics
and their variety, and the different options of prompts within each
prompt. 75% of participants responded “Yes” when asked if they would
like their writings to be shared with other people with CF. 75% of
participants responded “Yes” when asked if they would like to read
about writings from other people with CF.
Additional participant feedback included: “I enjoyed participating in
this study because I felt as though I had a safe space to talk about my
troubles and obstacles I faced with my CF [along] with my
accomplishments and any positive things that have happened with my CF”,
“I liked participating in the journaling study since it helped me to
think about my CF in a different light. Usually, I avoid thinking and
talking about my CF or thinking of the things that make me different
from everyone else. This study helped me to realize that I am not the
only one with CF who may have those experiences,” and “I liked it
because it helped me explore and understand.”
Notably, we saw improvement in almost all areas in mental health
questions from the pre and post survey. There was a decrease in the
number of days participants self-reported their physical and mental
health as poor and days that this interfered with their daily
activities. There was also a decrease in the reported days participants
felt pain, sad, anxious, not getting enough sleep. [Table 2, Table
3] Pediatric Symptom Checklist scores also decreased from a mean of 29
to 23.5. There was a decrease in BMI percentile and FEV1 percent of
predicted for all study participants, regardless of completion of study
[Table 4]. However, it must be noted that the study was not powered
to analyze these data for statistical significance.
Discussion
Our study was initially designed as a feasibility pilot study for
initiating journaling therapy in individuals with cystic fibrosis. In
designing the study, the investigators had previous experience with
journaling and narrative therapy and wanted to apply this knowledge to
individuals with cystic fibrosis. After discussing the study design with
social workers and psychiatrists who were knowledgeable about narrative
therapy, as well as the CF clinic social worker and providers, we were
able to design a pilot study to assess feasibility of such an
intervention. Based on the CF clinic social worker’s experience and the
baseline GAD7 and PHQ9 scores that are routinely collected in the
clinic, the social worker also recognized that this population would
benefit from and be amenable to this intervention. Therefore, we wanted
to include journaling topics that targeted major issues that face
individuals with cystic fibrosis, including those recognized by the
staff in the CF clinic as well as those reviewed in literature. Overall,
we wanted to highlight the importance of the multidisciplinary
collaboration necessary to design a study that benefits and aligns with
the goals of our specific population with cystic fibrosis.
Unfortunately, no individual with cystic fibrosis were directly involved
in the study design. Due to this fact, we included specific survey
questions and a section for open feedback and comments to elicit
participants’ opinions about the study design.
We were very pleased with the level of participation in our study. We
limited the maximum participants to eight, but there was at least one
other individual who met inclusion criteria who expressed interest in
the study after we had completed recruitment. Two participants did not
complete any weekly prompts after the initial pre-study survey. One
participant completed up to the 3rd weekly prompt, and
one participant completed up to the 4th weekly prompt.
Three participants stopped writing upon starting the school year due to
concerns of the world load of completing the study on top of regular
schoolwork. One participant who only completed up to the fourth weekly
prompt was hospitalized during the study for a prolonged period.
Participation in the study was promising and above our expectations
since the other four participants (50%) completed the entirety of the
study.
When comparing pre-study and post-study data for those four participants
who completed the entire study, we found improvement in many areas. The
average score for the Pediatric Symptom Checklist decreased by 5.5
points, and more importantly the post-study average fell to less than
28, which is the cutoff for children ages 6-16 for screening positive
for significant behavioral or emotional problems. There was a decrease
in averages for items related to internalizing problems, indicative of
the success of narrative medicine theory. Interestingly, the average
Pediatric Symptom Checklist score for all participants is lower than
that for participants who completed the study, suggestive that healthier
individuals chose not to continue the study.
The study also helped with participant exposure to narrative therapy and
journaling. There were improvements in average scores for Table 2 items
1 and 2, which ask about the participants familiarity and comfort with
narrative therapy. These results show we achieved our goal of increasing
familiarity and comfort level of our patient population towards using
journaling/narrative therapy to process emotions. There was a decrease
in responses for item 4 indicating a lower desire to plan to
journal/write, which is inverse to what we would expect after
participating in our study. Perhaps the question was worded poorly to
assess participants’ current action of writing versus their planning to
write more. Conversely, this may also indicate that participation in our
study was associated with a decreased desire to plan to write more,
pointing out that our study was either too time consuming, causes
participant distress, or had other reasons to dissuade them to continue
writing.
Participant feedback was important for this pilot study. 100% of
participants reported “I recommend other people with CF to write about
the topics from this study” and “this study helped me express my
feelings”. 75% of respondents replied “Strongly Agree” with Table 3
items 1, 4, 6, and 7, which shows that the study not only was enjoyable
and likely feasible to expand beyond the pilot study, but also provided
subjective improvement in participants’ feelings of anxiety/depression
and perception that providers understood their patients more though
these writings.
In terms of health benefits from the study, we only collected BMI
percentile and FEV1 as surrogate objective markers for health. Although
the sample size was small and analysis was confounded by missing FEV1
data for two participants, we did find that FEV1 percent of predicted
decreased less for those who completed the study compared to those who
did not. BMI percentile change was more negative for those who completed
the study as well. Of note, those who completed the study started at a
higher BMI percentile, averaging > 85, and so increased
negative change could indicate improvement from overweight status as
well. A properly powered study would be helpful to elucidate these
changes in health measures. It is important to note that although
participants did write about negative emotions, the SOP never needed to
be activated and no mental health intervention was necessitated,
indicating that our intervention for this group of participants was able
to explore sensitive topics without increasing risk for
suicidal/homicidal ideation or other reportable events.
Participants also provided feedback regarding their experience at the
end of the study. In designing the study, the prompts being too negative
was one of the team’s focuses. We had wanted to phrase the topics in a
way to discuss resiliency in the face of adversity. However, these
responses indicate that we were not successful in our efforts. In future
endeavors, we hope to focus more on the phrasing of the topics, as well
as introducing topics focused solely on positive strength without
comparing to hardship. One participant liked the weekly deadline, and
another participant did not and felt like the deadline caused too much
pressure instead of allowing “my own time” to write. Based on the
completion rate of our pilot study and mixed feedback about the timing,
we felt that a weekly deadline was appropriate for this type of
intervention. This intervention was also completed during the start of a
new school year, and so it is likely a weekly deadline is not too
burdensome for our patient population. 75% of participants who finished
the study indicated they would like their writings to be shared with
other people with CF, and they would like to read about writings from
other people with CF as well. We were glad to see these results, since
we had specifically wanted these interventions to be written so that
they can be recorded in the hopes of providing valuable material to be
shared with other individuals with CF. Although the writings of the
participants from this pilot study will not be shared, future
interventions can have publishing of results as an option, in hopes that
sharing and reading other relatable stories can help individuals with CF
and other complex medical problems. We hope that our study can be an
example and gateway to more journaling and dissemination of writings to
help patients with medical complexity address the topics included in our
study, such as isolation, depression, anxiety, and interpersonal
relationships.
One limitation of our study is that our participants may not be the most
representative of all individuals with CF, since the average FEV1
percent of predicted was 92.1, with low average hospitalizations in a
year, and also majority being non-white Hispanic or Latino and female.
However, these demographics may indicate a subpopulation that is more
open to journaling or would be more receptive to other interventions
focused on mental health. In the future, timing of the study initiation
can be optimized to increase participation, such as adjusting for the
school year and timing of exams or holiday breaks, and to expand
participant numbers for sufficient statistical power. We hope that this
study design can be utilized and tailored to other patient populations
that suffer from chronic conditions that can be emotionally taxing
(i.e., type 1 diabetes mellitus, sickle cell disease, and oncologic
conditions). Using the Pediatric Symptom Checklist data, we calculated
that a properly powered study will require 128 participants in each
study group, assuming a 50% attrition rate. We hope future properly
powered studies will be able to gather more statistically significant
data regarding this intervention.
Conclusion
This feasibility pilot study to implement a journaling intervention for
individuals with CF was successful. Our expected level of participation
and completion of the study was exceeded. Additionally, initial results
are promising for improvement of general wellness as measured by the
Pediatric Symptom Checklist, as well as participant subjective reports
of benefit from the study. Overall, we hope this study design can be
expanded to more participants in a formal study, as well as be adapted
to include other populations with medical complexity who can benefit
from journaling and improvement in wellness. Our study was specific to
our patient population, and required a multidisciplinary approach
towards designing, screening and recruiting, and choosing of journaling
prompts. Adaptation to other populations would require equal specificity
for the benefits of journaling and narrative therapy theories to be
realized.
Acknowledgements
The Cystic Fibrosis Foundation
Sarah Voyer, Social Worker
Truc Nguyen, IMPACT Program Coordinator
Amanda Daigle, IMPACT Program Coordinator
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