Content

Issues

Research

Permissions

Community Content

Videos and Multimedia

IJMSC Awards

For Authors

Information for Authors

Manuscript Submission

Patient Consent Form

About IJMSC

Aim and Scope

Ethics

Editorial Board

Peer Review

Contact Us

Home

Advertising

IJMSC

1rem

Authors

Experiences of Word-Finding Difficulties in Multiple Sclerosis: A Qualitative Interview Study

 To access the article and evaluation online, go to 

 The target audience for this activity is physicians, advanced practice clinicians, nursing professionals, mental health professionals, social workers, and other health care providers involved in the management of patients with multiple sclerosis (MS).

Describe the 3 theories discussed and characterize their overlap with usual care in order to implement changes to improve health-related quality of life in children with MS.

Describe how recommendations derived from these theories may improve the health-related quality of life of children with MS and their parents by strengthening self-concept, hope, and knowledge.

In support of improving patient care, this activity has been planned and implemented by the Consortium of Multiple Sclerosis Centers (CMSC) and Intellisphere, LLC. The CMSC is jointly accredited by the Accreditation Council for Continuing Medical Education, the Accreditation Council for Pharmacy Education, and the American Nurses Credentialing Center to provide continuing education for the health care team.

This activity was planned by and for the healthcare team, and learners will receive .75 Interprofessional Continuing Education credit for learning and change.

 The CMSC designates this journal-based activity for a maximum of .75 

™. Physicians should claim only the credit commensurate with the extent of their participation in the activity.

 The CMSC designates this enduring material for .75 contact hour of nursing continuing professional development (none in the area of pharmacology).

 This activity is awarded .75 CE credits.

 As a Jointly Accredited Organization, the CMSC is approved to offer social work continuing education by the Association of Social Work Boards Approved Continuing Education program. Organizations, not individual courses, are approved under this program. State and provincial regulatory boards have the final authority to determine whether an individual course may be accepted for continuing education credit. The CMSC maintains responsibility for this course. Social workers completing this course receive .75 continuing education credits.

 It is the policy of the Consortium of Multiple Sclerosis Centers to mitigate all relevant financial disclosures from planners, faculty, and other persons that can affect the content of this CE activity. For this activity, all relevant disclosures have been mitigated.

, has served as physician planner for this activity. He has disclosed no relevant relationships. 

, has disclosed no relevant relationships.

Julia O’Mahony, PhD; Ruth Ann Marrie, MD, PhD; Audrey Laporte, PhD;

, have disclosed no relevant financial relationships.

, CMSC, and Intellisphere, LLC who are in a position to influence content have disclosed no relevant financial relationships. 

, CMSC continuing education director, has served as a planner and reviewer for this activity. She has disclosed no relevant financial relationships.

Release Date: January 1, 2023. Valid for Credit through: January 1, 2024.

(1) Review the continuing education information, including learning objectives and author disclosures.

(3) Complete the evaluation, which is available at 

Statements of Credit are awarded upon successful completion of the evaluation. There is no fee to participate in this activity.

 This educational activity may contain discussion of published and/or investigational uses of agents that are not approved by the FDA. The CMSC and Intellisphere, LLC do not recommend the use of any agent outside of the labeled indications. The opinions expressed in the educational activity are those of the faculty and do not necessarily represent the views of the CMSC or Intellisphere, LLC.

 Participants have an implied responsibility to use the newly acquired information to enhance patient outcomes and their own professional development. The information presented in this activity is not meant to serve as a guideline for patient management. Any medications, diagnostic procedures, or treatments discussed in this publication should not be used by clinicians or other health care professionals without first evaluating their patients’ conditions, considering possible contraindications or risks, reviewing any applicable manufacturer’s product information, and comparing any therapeutic approach with the recommendations of other authorities.

 Children with the chronic disease multiple sclerosis (MS) report lower health-related quality of life (HRQOL) compared with children who experience transient illness. The relationship between an MS diagnosis and the HRQOL of affected children is mediated by parental HRQOL. Interventions to improve the HRQOL of children with MS should, therefore, include parents of affected children.

We performed a configurative review for improvements in the HRQOL of children facing diseases similar to MS and their parents. We used the generated concepts to form theories. Next, we performed qualitative interviews with clinicians who care for children with MS to characterize overlap between the proposed theories and usual care. Finally, we generated recommendations for improving the HRQOL of children with MS and their parents.

We theorize that the HRQOL of children with MS and their parents may be improved by strengthening self-concept, hope, and knowledge. Qualitative interviews with 7 clinicians who care for children with MS revealed no common psychosocial care protocol. The interviews did, however, reveal sources of psychosocial care that overlap with the proposed theories and barriers to optimizing such care.

Grounded in theory and clinically oriented practice, recommendations to improve the HRQOL of children with MS and their parents are to implement standardized screening, pool provider counseling strategies, create computer applications with psychosocial interventions, promote age-appropriate education resources, and secure positions for MS specialists.

We thank the clinicians who participated in the qualitative interviews and Dr Brenda Banwell for her assistance in identifying participants for the qualitative interviews.

 Dr O’Mahony receives research funding from the Multiple Sclerosis Society of Canada, the Multiple Sclerosis Scientific Foundation, and the Consortium of Multiple Sclerosis Centers. Dr Marrie receives research funding from the Canadian Institutes of Health Research, Research Manitoba, the Multiple Sclerosis Society of Canada, the Multiple Sclerosis Scientific Foundation, Crohn’s and Colitis Canada, the National Multiple Sclerosis Society, the Consortium of Multiple Sclerosis Centers, the Arthritis Society, and the US Department of Defense; she is a coinvestigator on studies receiving funding from Biogen Idec and Roche Canada. The other authors declare no conflicts of interest.

 This study was funded by the Multiple Sclerosis Scientific Research Foundation.

 This work was presented as a live poster at the virtual Americas Committee for Treatment and Research in Multiple Sclerosis meeting; February 25-27, 2021. It was also presented during the pediatric MS educational session at the Consortium of Multiple Sclerosis Centers Annual Meeting; October 25-28, 2021; Orlando, Florida.

Children with multiple sclerosis (MS) report lower health-related quality of life (HRQOL) compared with children who experience monophasic acquired demyelinating syndromes.

 Whereas HRQOL among adults with MS is associated with physical impairments, children with MS with low HRQOL mostly do not have functional physical impairments. Instead, parental HRQOL mediates the relationship between the diagnosis of MS and the children’s HRQOL, suggesting that interventions to improve the HRQOL of children with MS should include their parents.

 To date, research has focused primarily on diagnostic and treatment issues. No psychosocial care guidelines exist for improving the HRQOL of children with MS, and no studies have evaluated psychosocial interventions for children with MS and their parents.

When interviewed, children with MS report psychosocial challenges, including a more negative self-image after diagnosis,

 the loss of their hoped-for future, and fear of the unknown.

 Talking about the MS diagnosis with peers may aid the transition toward acceptance and psychosocial development; however, children have also reported strained peer relationships due to peer misinformation about pediatric MS, such as the need for a wheelchair.

 Children with MS report similar challenges when discussing their illness and associated needs with teachers.

Parents of children with MS also face psychosocial challenges. Parents can be anxious about their child’s ability to continue participating in day-to-day life

 and worry that the MS diagnosis is a death sentence.

 They can also be frustrated when they are unable to address skepticism from family and others about their child’s MS diagnosis due to their own poor understanding of the disease

 and when they are unprepared for MS sequelae.

 Parents describe using information searching as a coping mechanism; however, some parents despair when they uncover negative stories that create further uncertainty about how the disease will manifest in their lives.

This study aimed to (1) generate theories to optimize the HRQOL of children with MS and their parents based on a review of concepts attributed to improvements in HRQOL among families facing similar diseases and (2) characterize overlap between proposed theories and the current psychosocial care delivered to children with MS and their families through qualitative interviews with care providers.

We performed a configurative review, which is a systematic review that tries to interpret and understand the world by arranging concepts to develop theories.

 We identified concepts cited for improvements in HRQOL of children and parents facing diseases with stable physical statuses and unpredictable heterogeneous outcomes. Concepts were configured to generate theories (

Because the theories generated from this study are intended to inform the development of interventions for children with MS and their parents, we included studies that used the PedsQL measurement tools to detect changes in HRQOL.

 PedsQL questionnaires are validated for use among children with chronic diseases and their parents and are used widely.

 The literature search was conducted using the index of PedsQL publications made available via the instrument’s author on the PedsQL website (

). This index is believed to be comprehensive because to use the PedsQL tools, all publications must report to the author for inclusion in the index.

A single author (J.O’M.) performed a 2-stage literature screening process. She applied an open code to each article at each screening stage to capture the primary reason for inclusion or exclusion. This search strategy aligns with the methods of configurative reviews, which are meant to identify sufficient cases to explore patterns and are not meant to be exhaustive.

We excluded articles during the first stage if (1) the primary objective was methodological (eg, validation of the PedsQL); (2) they involved evaluation of interventions that would preclude attribution of changes in HRQOL to improved management of psychosocial challenges; (3) the patient populations were palliative and faced psychological complexities associated with imminent death; (4) they did not evaluate interventions, mediators, buffers, or moderators; or (5) they evaluated diseases in which pain was a predominant symptom as pain is an uncommon symptom among children with MS and negatively impacts HRQOL.

 Cross-disease comparisons of HRQOL were limited to intrastudy rather than interstudy comparisons to control for external factors that might contribute to differences in HRQOL across populations.

The second round of screening appraised whether each article provided evidence for a psychosocial intervention or mediator/moderator/buffer. We recorded all concepts cited as being associated with the HRQOL of children or their parents, the strength of evidence provided by the study, and the applicability to the present study. We excluded articles that did not provide evidence of a psychosocial intervention on HRQOL.

We recorded all concepts cited in the included articles associated with improved HRQOL of children or parents. Axial coding was performed to identify relationships among the concepts. The information about the concepts provided in the included articles informed the configuration of 3 theoretical models (Appendix S1).

One of us (J.O’M.) then performed qualitative interviews to characterize how the theories generated from the configurative review align with current clinical practice. Interviews and accompanying analyses were guided by the principles of grounded theory to allow for exploration and in-depth understanding of clinicians’ perspectives.

 The University of Toronto Research Ethics Board approved this study.

To capture standards of psychosocial care across different avenues of care, we invited 3 groups of health care providers to participate in qualitative interviews: pediatric MS specialists, adult MS specialists who treat children with MS, and pediatric non-MS specialists who treat children with MS. Drs Banwell and Marrie are principal investigators of the Canadian Pediatric Demyelinating Disease Network and identified the providers who were invited to undergo interviews.

Interviews were directed by a topic guide (

), conducted in person or via telephone, recorded, and transcribed verbatim by one of us (J.O’M.). Data collection continued until categories were fully developed and saturation was reached. One of us (J.O’M.) presented a verbal summary with visual adjuncts of our previous empirical findings

 and an overview of the proposed theories so that clinicians could discuss how the theories generated from the configurative review aligned with their usual clinical care. Participants did not have access to the empirical findings or theories before the interviews.

Interviews were analyzed inductively using qualitative data analysis software (NVivo; QSR International).

 Initial codes were developed from line-by-line coding that examined experiences, actions, and assumptions.

 Open and focused coding was used to develop categories and understand their relationships. We adhered to standards for performing and reporting qualitative research.

The interviewer (J.O’M.) maintained a reflexive standpoint throughout the study to account for her potential to influence the research process and co-construct data with interviewees.

 A reflexive journal was used to record her beliefs, actions, and observations that might have influenced data generation and analysis. She considered how her positionality and professional knowledge may have shaped her interaction with the interviewees.

Sixty-seven of the 1677 articles listed on the PedsQL index on March 4, 2019, cleared the first round of screening, and 36 articles cleared the second round. Characteristics of the included articles are summarized in 

The concepts identified as being associated with improved HRQOL of children or parents were configured into self-concept, hope, and disease-specific knowledge theories.

Of the 36 included studies, 8 described interventions that facilitated the child’s or parent’s formation of a new self-schema that was not jeopardized by the child’s disease (

). Self-schema—beliefs about one’s self that inform one’s overall self-image—relate to various aspects of the current and future self, including personality, skills, abilities, occupation, hobbies, and physical characteristics.

 Each person forms self-schema based on their environment and external cues. Self-concept is the summation of the self-image and the ideal self.

 Discordance between the self-image and the ideal self can cause low self-esteem and emotional dysfunction, observed among children with MS and their parents.

 A person may experience reduced HRQOL when they are unable to fulfill their current self-schema or are unsure of their ability to fulfill their future self-schema, as may be the case for children with MS and their parents.

We theorize that the HRQOL of children with MS and their parents may be improved by ensuring they form self-schemas that are not threatened by MS (

). We propose that this can be achieved by changing the environment, changing the external cues, and reframing ideal-self expectations. We further theorize that parents and children enable formation or reframing of self-schema in one another through environmental changes and external cues.

The Snyder hope theory is grounded in the observation that humans are constantly thinking about how to reach their goals and that their emotions reflect that perceived capacity; a high-hope person has enduring positive emotions with a sense of affective zest, whereas a low-hope person has negative emotions with a sense of affective lethargy.

 Thus, hope is determined by whether there are workable routes (ie, pathways) to desired goals and the person’s willingness to use the identified routes to reach desired goals (ie, agency).

 Concepts associated with the Snyder hope theory were cited by 14 of the studies as related to improved HRQOL among children and parents (

We theorize that children with MS and their parents may report improved HRQOL if they can achieve agency and establish pathways to achieve their goals even when the goals are unrelated to the child’s MS (

The Mishel uncertainty in illness theory provides a framework to understand how disease-specific knowledge may improve HRQOL by reducing uncertainty. Uncertainty in the context of illness occurs when those affected are unable to understand how the disease will impact their lives.

 The HRQOL may improve when affected individuals are able to detect patterns in the disease course and clearly understand how it may impact their lives.

Disease-specific knowledge may also improve HRQOL through strengthened social connections by providing families with the vocabulary to respond to questions or skepticism and communicate their experiences, fears, and needs.

 For families choosing not to disclose the child’s MS diagnosis, disease-specific knowledge may allow them to communicate what is likely central to their lives without violating their child’s privacy. Disease-specific knowledge was associated with improved HRQOL in 14 of the included articles.

We theorize that disease-specific knowledge may improve HRQOL among children with MS and their parents by reducing perceived uncertainty (

) and by providing families with the knowledge necessary to communicate their experiences to others.

Of 14 providers invited to undergo qualitative interviews, 13 (93%) agreed to participate and 1 did not respond. Saturation was reached on completion of interviews with 7 participants. Characteristics of the 7 participants are presented in 

. No differences were observed between in-person and telephone interviews. Interviews adhered to the topic guide, with flexibility for development of themes.

Participating clinicians reported that they detect low HRQOL through red flags, conversations with families, responses to questions about mood during clinical assessments, and families reaching out to them. Red flags cited by providers included nonadherence to treatments, depressive symptoms, comments about substantial frustration with the limitations of their illness, moodiness, irritability, and absence from school. One clinician who treats adults uses a questionnaire to screen for depression among people with MS. None of the participants used measurement tools to ascertain HRQOL as part of usual care of their patients or their patients’ parents.

Qualitative interviews revealed no common protocol for providing psychosocial care to children with MS and their parents but did identify 3 sources of psychosocial support that overlap with the proposed theories: provider counseling, standardized programs, and departmental referrals to social work, neuropsychology, and psychiatry.

Providers saw their own counsel as a form of psychosocial support delivered as part of routine clinical care or in response to parent outreach. They described standard pieces of information (herein, “talking points”) that they conveyed to each child with MS and their parents. These talking points were formed by negative experiences that they observed among families facing pediatric MS. For instance, a pediatric neurologist observed family distress due to misinformation about the likelihood of wheelchair use that the family received from a referral center at the time of diagnosis. Thereafter, he made sure to identify and expel myths during his first encounter with children with MS. “I make a point in the first visit to tell them, and I’m cautious about how I say it, but I say that my goal is that, in 10 years, you’re dancing at your wedding, you’re having kids. My goal is to keep you as you are now in the future. I can’t promise it, obviously, but my goal for you is to not be in a wheelchair” (provider 5).

Providers also described talking points that prepared families for what to expect during the disease course. “I usually tell them that there are very few emergencies in MS and so, oftentimes, things can wait until the morning without necessity of them going to the ER. I think what often relieves a lot of them, too, is the fact that they’re not going to have to rush to the ER at midnight because their child is experiencing numbness” (provider 5).

Other talking points covered the distinction between progressive and relapsing disease; the disproportionate number of people with primary progressive MS on the internet whose experiences are unique to those with progressive disease; the low prevalence of physical deficits among children with MS; concrete analogies to explain MS pathobiology; and the importance of including the child when deciding to whom to disclose the child’s diagnosis.

Talking points about the MS disease course tell affected children and their parents about how MS might affect the ability to fulfill their self-schema and achieve their goals; they align with the theories of self-concept and hope. Talking points that convey information about how families should respond to signs of MS align with the patterned information component of the knowledge theory. Talking points that explain MS pathobiology align with the communication component of the knowledge theory.

Of the interviewed clinicians, 2 offer standardized educational programs. The first offers a 45- to 60-minute session with a trained nurse who covers MS risk factors, diagnostic criteria, identifying relapses and what to do when they occur, prognosis, common symptom management, therapies, wellness, health maintenance, and resources available through the clinic. These sessions are tailored to the individuals with MS, but family members are welcome to attend. The other standardized program is a peer-to-peer match program for children and parents.

These standardized programs align with all the proposed theories. They provide families with an understanding of how the disease might affect their ability to fulfill their self-schema and achieve their goals, thereby aligning with the self-concept and hope theories. They also align with the knowledge theory by providing families with information about possible manifestations of the disease and information to enhance communication.

Referrals to social workers, clinical or health psychologists, and psychiatrists were consistent with the theories on self-concept and hope. Providers with specialized MS training may provide families with information about how the disease might affect their self-schema or pathways to achieve their goals, and specialists without MS-specific training may provide general reinforcement of self-schema and goal setting that may strengthen feelings of hope. External resources are also available, such as the National Multiple Sclerosis Society’s MS Navigator program, which connects people with MS to mental health providers.

The qualitative interviews with the clinicians made it clear that providers offer a wealth of psychosocial resources to children with MS and their families that overlap with the proposed theories, but there are also barriers to optimizing these resources.

Most participants used unsystematic and time-consuming strategies to detect low HRQOL. Psychosocial care that aligns with the proposed theories cannot be implemented if the need for such care is unknown.

 We recommend standardized screening programs to detect low HRQOL among children with MS and their parents. Questionnaires are a straightforward way to identify clinically relevant concerns that may be missed during clinical examinations. Learning health systems are poised to enable families to record their HRQOL before clinical encounters and then join HRQOL and clinical data for clinicians to visualize and export findings to research registries. Patients have reported valuing learning health systems for the individualized information they provide.

 Clinically meaningful cutoff values are not known for all HRQOL questionnaires, but individual questions can serve as clear beacons for clinician concern, including a response of “almost always” to statements on the PedsQL parent report such as “It is hard for me to tell doctors and nurses how I feel,” “I worry about my child’s future,” “I worry about the side effects of my child’s medication/medical treatments,” and “I worry about how others will react to my child’s condition.”

Confidential questionnaires may also increase the chance of identifying concerns. Individuals may be more forthcoming about mental health concerns through written communication, and confidentiality offers children and parents the opportunity to report concerns that they may not feel comfortable raising in front of one another. Clinicians in this study sensed that parents were hesitant to raise concerns in front of their child for fear of making them worry or feel like a burden.

Three participants relayed experiences of misinformation communicated to children with MS and their parents by clinicians, often at referral centers, that are obvious sources of psychosocial distress for families.

“I’ve had experience with several patients who are told things at outside institutions that are sometimes very shocking in terms of what their life is going to be like. I mean I had kids who have been told, or at least tell me they’ve been told, that they were going to die at an early age, that they’ll be in a wheelchair, that they could never get pregnant. It’s pretty shocking” (provider 5).

Providers also noted that much of their counseling occurs as second nature, especially for those who care for thousands of patients, and would require further evaluation to understand.

“Some of these things we do without necessarily being conscious of why we do them. So, it’s trying to ask somebody why you felt like this person needed that thing. So some of us will say, well ‘I just felt it, knew, and I’ve seen it before,’ but you really want to be able to decompile that so anybody on your team can actually identify that individual and do it and you’re likely not to get there unless you actually have people who are consciously aware of what the issues are and then adopt some framework to work within it. And so, coming from different backgrounds, different members of the team probably frame these issues in a different way and adopt different interventions and some people probably don’t have as conscious a framework. So it’s probably worthwhile even as a starting point to help people understand what these frameworks are” (provider 3).

 In the interviews, clinicians cited talking points as valuable components of psychosocial care; this overlaps with the proposed theories. We recommend that a set of talking points be made available so that all families are provided with accurate information to mitigate psychosocial distress. The International Pediatric Multiple Sclerosis Study Group is a global network of clinicians and researchers whose mission is to improve the care of children with MS by promoting clinical initiatives, education, and research

 and would, therefore, be well-suited to compile and distribute these talking points.

The clinician interviews also highlighted a wealth of MS-specific clinical acumen that underlies psychosocial care and overlaps with the proposed theories, but participants noted that there are no frameworks for such counseling. We recommend study, production, and then distribution of psychosocial care frameworks for MS providers. Neurologists participating in postgraduate training in MS clinics would be well-suited to conduct qualitative studies including interviews with MS specialists to gain a better understanding of how they deliver care; these studies could be funded by national and international MS societies.

Limited Availability of Mental Health Professionals  

Providers were confident in their ability to provide psychosocial support and issue referrals to mental health professionals; however, they also reported time constraints and the limited availability of mental health professionals as barriers to optimizing these resources.

 The articles included in the configurative review revealed that psychosocial intervention computer applications can have meaningful impacts on HRQOL.

 Applications allow for broad access and do not require clinician time.

 It sounds as though a lot of the models that I’m proposing align with your beliefs and observations, but I’m not sure that they’re being implemented.

 No, I’m sure they’re not. I’m sure they’re not and they can’t ever be. The clinicians don’t have time so we have got to figure out some way of imparting it. Maybe an app. Maybe some sort of internet communication. It’s time. It’s time now.

We, therefore, recommend the use of computer applications with psychosocial interventions that align with the proposed theories. Importantly, any psychosocial intervention computer application available to people with MS should include a 24/7 crisis hotline number and a caveat that one should go to an emergency department when experiencing severe distress, psychopathology, and/or suicidal ideation.

This study highlights the importance of disease-specific knowledge and the existing paucity of educational materials, particularly age-appropriate resources, for families facing pediatric-onset MS. With more than 1863 existing academic publications about pediatric-onset MS, and the data from longitudinal observational studies in the past 15 years, data to form the content of such resources are readily available.

 Providers noted the importance of information, with a clinician stating, “Information is one of the things that people need to figure out their pathway” (provider 3). Information is a core component of the knowledge theory. We recommend that age-appropriate educational materials be compiled and made centrally available. Educational materials are priorities for national MS societies; this effort would, therefore, be likely to garner their support.

This study revealed that MS specialists are valuable sources of and gateways to psychosocial care that overlaps with the proposed theories. It is, therefore, imperative to address the projected shortage of MS specialists anticipated to occur in the next 2 decades.

 A participant described the potential consequences of a shortage to the psychosocial health of children with MS who do not have access to pediatric MS clinics. “We have children and adults in the same clinic, which may be prone to increased emotional distress because they see adults in wheelchairs and using canes and elderly individuals because it’s a general neurology clinic that deals with strokes and you know people with severe epilepsy wearing helmets, so it’s an environment that can be potentially overwhelming for kids. Ideally there would be enough kids or space where one could have them be in a more pediatric-centered environment” (provider 3).

 We recommend implementation of strategies to encourage postgraduate medical residents, medical students, and health psychologists to pursue specialized training in MS (eg, fellowships) to facilitate ongoing delivery of psychosocial care that aligns with the proposed theories. Studies suggest that more neurology residents are interested in pursuing MS fellowships than there are funded positions available; increasing the number of funded MS fellowships may, therefore, help secure MS specialists.

 In addition, securing funding for research grants for MS specialists may incentivize trainees to pursue an MS subspecialty.

This study generated 3 theories about interventions that may improve the HRQOL of children with MS and their parents. Qualitative interviews with clinicians characterized the existing psychosocial care that overlaps with the proposed theories and the barriers to the delivery of that care. The present findings allowed us to make 5 recommendations to optimize the HRQOL of children with MS and their parents that are grounded in theory and clinical practice.

The recommendations align with the conclusions of previous studies. Pooling provider talking points echoes previous calls to reassure families that MS during childhood is not fatal.

 Computer applications address previous concerns about the inadequate availability of mental health services for youth with MS and their parents.

 Standardized screening for low HRQOL is consistent with parents reporting that they would welcome screening and referrals for their own mental health from their child’s care providers.

 Increased educational resources aligns with previous appeals for improved educational resources for families facing pediatric-onset MS.

This study addressed the psychosocial needs of a specific subgroup: children with MS who face minimal physical impairments. Due to earlier diagnosis and the rapidly improving MS treatment landscape, more children live with MS without physical deficits and may benefit from this study. The present findings, however, are not necessarily generalizable to people who face persistent physical deficits, pain, or fatigue, which are known to affect HRQOL.

 Strategies to maximize the HRQOL of people with MS who face such challenges warrant further investigation.

The configurative review benefited from systematic methods to limit bias and maximize the potential for reproducibility. The present findings, however, may have been influenced by publication bias from the disproportionate publication of studies with statistically significant results.

 Publication bias may have led to an overestimate of the potential for the proposed theories to positively impact HRQOL. Future studies should account for this potential bias. We made a conscious effort to limit the terminology and concepts of the theories to those cited within the articles included in the configurative review to maximize reproducibility. For example, models of resilience overlap with the hope theory and concepts such as identity, liminality, and cognitive dissonance overlap with the self-concept theory; however, these concepts did not appear in the configurative review articles and, therefore, were not cited in the proposed models. Spiritual and religious beliefs were not cited in the included articles; however, they have been cited elsewhere by children with MS and their parents and may warrant further exploration.

 To overcome the limitation of having a single reviewer, each stage of article adjudication and inclusion were fully documented.

The recommendations generated from this review address psychosocial challenges previously raised by families facing pediatric-onset MS and substantiate the conclusions of previous studies. The resources needed to implement the recommendations and improve the HRQOL of children with MS and their parents are readily available.

Addressing Health-Related Quality of Life Among Children With Multiple Sclerosis

From the Frances Payne Bolton School of Nursing, Case Western Reserve University, Cleveland, OH, USA (SLD, MP). 

Sara L. Douglas, PhD, Frances Payne Bolton School of Nursing, Case Western Reserve University, 10900 Euclid Ave, Cleveland, OH 44104, USA; email: SLD4@case.edu.

Caregivers of persons with multiple sclerosis (MS) report high levels of distress. The National Comprehensive Cancer Network Distress Thermometer (DT) is used extensively with patients with cancer and their caregivers but has not been tested in nononcology caregivers. The purpose of this study was to examine the psychometric properties and clinical utility of the barometer portion of the DT in caregivers of persons with MS.

A secondary analysis was performed of data from a randomized trial comparing the effectiveness of 2 interventions aimed at reducing psychological outcomes associated with caregiving. The DT and the 4-item Patient-Reported Outcomes Measurement Information System Anxiety and Depression scales, which were administered at baseline, were used for all analyses. Construct validity (known groups) and convergent validity (interscale correlations) were evaluated. Receiver operating characteristic curve analysis was used to evaluate clinical diagnostic test evaluation.

 The DT had good construct validity supported by strong correlations for known-groups analyses and good convergent validity (

 = 0.70–0.72). The DT also demonstrated good discrimination for anxiety (area under the curve [AUC] = 0.83) and depression (AUC = 0.80). The optimal screening cut point on the DT was 4 for anxiety and 5 for depression.

The barometer portion of the DT demonstrates good psychometric properties and clinical utility in caregivers of persons with MS. This is the first examination of the DT in MS care partners.

 The authors declare no conflicts of interest.

 This study was supported by the Patient-Centered Outcomes Research Institute (grant MS-1610-37015-Enhancement).

Psychological distress has been conceptualized as a “state of emotional suffering characterized by symptoms of anxiety and depression,”

 and research has demonstrated its effect not only on a patient’s quality of life but also on their adherence to treatment, satisfaction with care, and survival.

 Distress and negative emotional outcomes have also been documented in caregivers. Rates of distress and psychiatric disorders for caregivers have ranged from 15% to 60%,

 and caregiver distress has been shown to contribute to the delivery of lower-quality patient care and to poorer quality of life for the patient.

 Therefore, caregiver distress has been shown to impact not only the caregiver but also the patient.

To date, the most common tool used to measure distress in patients with cancer has been the National Comprehensive Cancer Network Distress Thermometer (DT).

 It has good psychometric properties and clinical utility in oncology care

 and consists of a visual analog scale (resembling a thermometer) that ranges from 0 (no distress) to 10 (extreme distress).

 There is also a list of 42 problems representing 5 different categories (practical, family, emotional, spiritual, physical) where individuals identify sources of distress from the list of problems. In the clinical setting, the single-item thermometer is often used to identify individuals who display elevated distress (score >4) and who should be referred for additional evaluation. The DT has high acceptability among patients, caregivers, and clinicians, primarily because of its brevity.

In oncology, the DT is recommended as a quick screening measure to identify individuals with psychological distress who may present with symptoms of depression and/or anxiety.

 In addition, its use in the clinical setting is recommended because patients and caregivers do not stigmatize it like standard anxiety and depression tools.

 Finally, the DT is recommended because of the numerous validation studies (using depression and anxiety as reference measures)

 that have yielded cut points at which to make referrals for further psychological assessments.

Although the DT has been used with some populations of patients without cancer, information about its psychometric properties and clinical utility with these populations is minimal.

 In a recent scoping review of the clinical utility of the DT for those without cancer,

 of the 53 studies identified, 37 included patients, 14 included parents of children with various illnesses, and only 2 included caregivers who were not parents of children with illness. Given the recognition of the important role that caregivers play in the support and care of all patients—especially those with long trajectories of care—there is a need to identify reliable and valid tools that can quickly identify elevated caregiver distress—especially in caregivers of nonchildren.

One such group is caregivers of persons with multiple sclerosis (MS); MS is a chronic demyelinating neurologic disorder that is often progressive in nature and results in problems with mobility and cognition as well as a variety of other physical and emotional outcomes.

 An estimated 46% to 58% of persons with MS receive informal, unpaid care not routinely provided through the established health care system over the course of their illness.

 Caregivers of persons with MS are reported to have low health-related quality of life and high levels of anxiety (68%), depression (44%), and distress (51%).

 As screening of cancer caregivers for distress has begun to be incorporated into outpatient clinics, MS caregivers should be screened as well, given the prevalence of anxiety, depression, and distress identified in this population, and health care providers should make referrals for further evaluation and treatment if needed.

 The first step in this process is to determine the psychometric properties and clinical utility of the DT for use in this caregiver population.

Baseline (prerandomization) data used for psychometric testing came from a randomized trial comparing the efficacy of 2 interventions aimed at reducing distress in caregivers of persons with MS.

 The study was conducted from April 2021 to February 2022 and included a national sample of MS caregivers. A caregiver was defined as someone (family or friend) who provides any type of support (eg, physical, emotional, or administrative support, such as paying bills) to the person with MS, who is not a professional caregiver, and who is not paid for their efforts.

 Convenience sampling was applied using social media and direct email to persons with MS who had participated in a previous study. Inclusion criteria for caregivers were as follows: (1) an adult family member or friend (≥18 years old) of a person with MS, (2) self-identified as an unpaid caregiver for a person with MS, (3) access to the internet, and (4) capable of providing informed consent in English. After initial screening, electronic informed consent was obtained using REDCap (Research Electronic Data Capture).

 Approval was obtained from Case Western Reserve’s institutional review board before data collection.

Distress was measured using the barometer portion of the DT.

 The barometer tool is a single-item, self-report measure of psychological distress and has excellent psychometric properties for both patients with cancer

 Instructions for completion of the barometer tool defines distress as an “unpleasant experience of a mental, physical, social, or spiritual nature which can affect the way one thinks, feels, or acts.”

 Instructions also state that distress may make it harder to cope with disease, its symptoms, or its treatment.

 Individuals are asked to rate their distress in the past 7 days on an 11-point visual analog scale ranging from 0 (no distress) to 10 (extreme distress), with higher scores indicating higher levels of distress.

Receiver operating characteristic (ROC) curves have demonstrated that the DT has good diagnostic utility for cancer caregivers relative to anxiety and depression reference measures.

 Area under the curve (AUC) analyses indicate that the DT has good overall discrimination (AUC = 0.85 for the Patient-Reported Outcomes Measurement Information System [PROMIS] Anxiety scale and AUC = 0.81 for the PROMIS Depression scale),

 and ROC curves also indicate that using a cut point of 4/5 maximizes sensitivity (86.2% Hospital Anxiety and Depression Scale [HADS]-Anxiety; 88.2% HADS-Depression) and specificity (71.2% HADS-Anxiety; 67.6% HADS-Depression).

 Scores of 4 or greater represent clinically elevated levels of distress for caregivers of those with cancer, and the DT is deemed appropriate to use as a clinical screening tool for patients with cancer and their caregivers.

The PROMIS Anxiety and PROMIS Depression short-version instruments

Charlotta Saldert, PhD

Articles

Latest Updated Articles