Using Online Health Communication to Manage Chronic Sorrow: Mothers of Children with Rare Diseases Speak
2014; Elsevier BV; Volume: 30; Issue: 1 Linguagem: Inglês
10.1016/j.pedn.2014.09.013
ISSN1532-8449
Autores Tópico(s)Cystic Fibrosis Research Advances
Resumo•Only half of the mothers experienced chronic sorrow.•The other half experienced feelings of fright, anger or being overwhelmed that the researcher classified as "unpleasant feelings".•One overarching theme emerged in the interviews: online communication was essential to the mothers in managing chronic sorrow or unpleasant feelings.•Four essential themes and nine sub-themes that contributed to the development of the overarching theme. The four themes were connectedness, online triggers, empowerment, and seasons of online use.•Mothers of children with rare chronic diseases used online communication differently. Families affected by rare disease experience psychosocial reactions similar to families with prevalent chronic diseases. The ability to respond and manage the condition depends on psychosocial factors. This phenomenological study of 16 mothers of children with Alagille syndrome explored their lived experience in using online health communications to manage their chronic sorrow. Data consisted of semi-structured interviews analyzed using techniques described by van Manen. Analysis yielded four essential themes: connectedness, online triggers, empowerment, and seasons of online use contributed to online communication essential to a rare disease community. Findings suggest mothers need emotional support and help accessing appropriate online resources. Families affected by rare disease experience psychosocial reactions similar to families with prevalent chronic diseases. The ability to respond and manage the condition depends on psychosocial factors. This phenomenological study of 16 mothers of children with Alagille syndrome explored their lived experience in using online health communications to manage their chronic sorrow. Data consisted of semi-structured interviews analyzed using techniques described by van Manen. Analysis yielded four essential themes: connectedness, online triggers, empowerment, and seasons of online use contributed to online communication essential to a rare disease community. Findings suggest mothers need emotional support and help accessing appropriate online resources. RARE DISEASES POSE a significant and under-researched problem in health care. Almost 7000 rare diseases affect 25–30 million Americans, although each disease affects fewer than 200,000 Americans (National Institutes of Health, 2011National Institutes of Health Rare Diseases Network fact sheet.2011Google Scholar). Children comprise approximately half of those individuals affected with rare diseases (National Institutes of Health, 2012National Institutes of Health Rare Disease Day information fact sheet.2012Google Scholar). Rare diseases by their isolating nature make it difficult for parents of such children to find support (Dellve et al., 2006Dellve L. Samuelsson L. Tallborn A. Fasth A. Hallberg L.R. Stress and wellbeing among parents and children with rare diseases: A prospective intervention study.Journal of Advanced Nursing. 2006; 53: 392-402Crossref PubMed Scopus (146) Google Scholar).Families affected by rare diseases experience reactions similar to families with more prevalent chronic diseases, including shock, confusion, relief and grief/sadness (Hatton et al., 1995Hatton D.L. Canam C. Thorne S. Hughes A.M. Parents' perceptions of caring for an infant or toddler with diabetes.Journal of Advanced Nursing. 1995; 22: 569-577Crossref PubMed Scopus (90) Google Scholar). The isolating nature of rare disease and the unfamiliarity of many health care providers complicate the management of the condition and psychosocial experiences. Parents caring for a child with chronic illness generally seek and use social support from health professionals, family, friends and faith (Coffey, 2006Coffey J.S. Parenting a child with chronic illness: A metasynthesis.Pediatric Nursing. 2006; 32: 51-59PubMed Google Scholar, Croezen et al., 2012Croezen S. Picavet S.J. Haveman-Nies A. Verschuren W.M. De Groot L. van't Veer P. Do positive or negative experiences of social support relate to current and future health? Results from the Doetinchem cohort study.BMC Public Health. 2012; 12 (Retrieved from http://www.biomedcentral.com/1471-2458/12/65): 65https://doi.org/10.1186/1471-2458-12-65Crossref PubMed Scopus (56) Google Scholar, Wang et al., 2003Wang H.H. Wu S.Z. Liu Y.Y. Association between social support and health outcomes: A meta-analysis.Kaohsiung Journal of Medical Sciences. 2003; 19: 345-351Abstract Full Text PDF PubMed Google Scholar). Online communication provides the critical mass of people needed for support and facilitates finding information about rare conditions. Women are more likely to search the Internet for health related information and use online support groups (Pew Internet and American Life Project, 2007Pew Internet and American Life Project How gender influences health searches.2007Google Scholar).This study explores the lived experience of mothers of children with Alagille syndrome (ALGS) in using online health communications to manage their chronic sorrow. It seeks to inform nurses about these issues so they can consider how mothers manage their daily experiences. ALGS is a complex disease (Spinner et al., 2001Spinner N.B. Colliton R.P. Crosnier C. Krantz I.D. Hadchouel M. Meunier-Rotival M. Jagged1mutations in Alagille syndrome.Human Mutation. 2001; 17: 18-33Crossref PubMed Scopus (183) Google Scholar) involving mutations at numerous different loci of the JAG-1 and NOTCH-2 genes on chromosome 20 and affecting approximately 1 in 70,000 live births. Almost all patients have a reduction in liver bile ducts (ALGS National Digestive Diseases Information Clearing House, 2009National Digestive Diseases Information Clearing House Alagille syndrome.2009Google Scholar) and congenital heart defects (McElhinney et al., 2002McElhinney D.B. Kranz I.D. Bason L. Piccoli D.A. Emerick K.M. Spinner N.B. et al.Analysis of cardiovascular phenotype and genotype–phenotype correlation in individuals with a JAG1 mutation and/or Alagille syndrome.Circulation. 2002; 106: 2567-2574Crossref PubMed Scopus (217) Google Scholar). The disease also can affect numerous other organs, including the vascular system, kidneys, and spine, as disease severity varies widely. No cure exists for ALGS, but common treatments target improving the heart function and reducing effects of impaired liver function, with liver transplants being necessary for many patients (National Digestive Diseases Information Clearing House, 2009National Digestive Diseases Information Clearing House Alagille syndrome.2009Google Scholar).Chronic SorrowMothers caring for a child with a chronic illness experience increased distress (Isaacs and Sewell, 2003Isaacs D. Sewell J.R. Children with chronic health conditions.Medical Journal of Australia. 2003; 1: 235-236Google Scholar), similar to grief reactions (Eakes, 1995Eakes G.G. Chronic sorrow: The lived experience of parents of chronically mentally ill individuals.Archives of Psychiatric Nursing. 1995; 9: 77-84Abstract Full Text PDF PubMed Scopus (56) Google Scholar, Vickers, 2005Vickers M.H. Bounded grief at work: Working and caring for children with chronic illnesses.Illness, Crisis and Loss. 2005; 13: 201-218Google Scholar). Unlike grieving mothers, they also face the recurring disparity between their expectations for their child and the constraints imposed by the disease (Eakes et al., 1998Eakes G.G. Burke M.L. Hainsworth M.A. Middle range theory of chronic sorrow.The Journal of Nursing Scholarship. 1998; 30: 179-184Crossref Google Scholar) and ongoing loss with periodic triggering events (Northington, 2000Northington L. Chronic sorrow in caregivers of school age children with sickle cell disease: A grounded theory approach.Issues in Comprehensive Pediatric Nursing. 2000; 23: 141-154Crossref PubMed Scopus (19) Google Scholar).Chronic sorrow theory addresses the ongoing disparity between hopes and reality. Chronic sorrow is cyclical sadness that is "pervasive, permanent, periodic and potentially progressive in nature" (Eakes et al., 1998Eakes G.G. Burke M.L. Hainsworth M.A. Middle range theory of chronic sorrow.The Journal of Nursing Scholarship. 1998; 30: 179-184Crossref Google Scholar). It is a normal response as parents adjust to life with a child with a rare disease. The chronic sorrow literature emphasizes the importance of coping and social support, since many health care providers do not regard chronic sorrow as a normal response (Bowes et al., 2009Bowes S. Lowes L. Warner J. Gregory J.W. Chronic sorrow in parents of children with type 1 diabetes.Journal of Advanced Nursing. 2009; 65: 992-1000https://doi.org/10.111/j.1365-2648.2009.04963.xCrossref PubMed Scopus (0) Google Scholar) and view the mother's optimism and advocacy as denial of reality (Bettle and Latimer, 2009Bettle A.M. Latimer M.A. Maternal coping and adaptation: A case study examination of chronic sorrow in caring for an adolescent with a progressive neurogenerative disease.Canadian Journal of Neuroscience Nursing. 2009; 31: 15-21PubMed Google Scholar).Maternal and Parental CopingResearchers define coping as adaptive/constructive behaviors that can be classified into three types: appraisal focused, problem focused, and emotion focused (Weiten et al., 2011Weiten W. Dunn D.S. Hammer E.Y. Psychology applied to modern life: Adjustments in the 21st century. Wadsworth, Belmont, CA2011Google Scholar). Mothers tend to use appraisal and problem focused strategies when caring for a chronically ill child (Coffey, 2006Coffey J.S. Parenting a child with chronic illness: A metasynthesis.Pediatric Nursing. 2006; 32: 51-59PubMed Google Scholar). The most helpful coping behavior is the problem focused strategy of seeking social support from others (Hodgkinson and Lester, 2002Hodgkinson R. Lester H. Stresses and coping strategies of mothers living with a child with cystic fibrosis: Implications for nursing professionals.Journal of Advanced Nursing. 2002; 39: 377-383Crossref PubMed Scopus (59) Google Scholar). Four types of social support activities exist: emotional, informational, appraisal, and material/instrumental (Eastin and LaRose, 2005Eastin M.S. LaRose R. Alt.support: Modeling social support online.Computers in Human Behavior. 2005; 21: 977-992Crossref Scopus (102) Google Scholar). Parents of children with chronic illness identify the need for emotional, informational and material/instrumental support (Liu et al., 2010Liu C. Chao Y. Huang C. Wei F. Chien L. Effectiveness of applying empowerment strategies when establishing a support group for parents of preterm infants.Journal of Clinical Nursing. 2010; 19: 1729-1737https://doi.org/10.111/j1365-2702.2009.03082xCrossref PubMed Google Scholar).Online communication encompasses social support activities that differ based on parental needs. Parents of children with more common chronic health conditions use various forms of online communication to supplement information from physicians, develop knowledge and increase advocacy (Coulson and Greenwood, 2011Coulson N.S. Greenwood N. Families affected by childhood cancer: An analysis of the provision of social support within online support groups.Child: Care, Health and Development. 2011; 38: 870-877https://doi.org/10.1111/j.1365-2214.2011.0136xCrossref PubMed Google Scholar, Roche and Skinner, 2009Roche M.I. Skinner D. How parents search, interpret, and evaluate genetic information obtained from the Internet.Journal of Genetic Counseling. 2009; 18: 129-199Google Scholar). These parents do not readily turn to the Internet for health information, but preferred receiving information from their health provider (Gage and Panagaskis, 2011Gage E.A. Panagaskis C. The devil you know: Parents seeking information online for paediatric cancer.Sociology of Health and Illness. 2011; 34: 444-458https://doi.org/10.111/j.1467-9566.2011.01386.xCrossref PubMed Google Scholar, Nordfeldt et al., 2013Nordfeldt S. Angarne-Lindberg T. Nordwell M. Krevers B. Parents of children with type 1 diabetes—Their views on information and communication needs and Internet use. A qualitative study.Parents of Adolescents with Type 1 Diabetes. 2013; 8: 1-8https://doi.org/10.1371/journal.pone.0062096Crossref Scopus (37) Google Scholar). In contrast, emerging research suggests that parents of children with a rare chronic disease often turn to the Internet first to gain access to information regarding the disease and resources (Schaffer et al., 2008Schaffer R. Kuczynski K. Skinner D. Producing genetic knowledge and citizenship through the Internet: Mothers, pediatric genetics, and cybermedicine.Sociology of Health and Illness. 2008; 30: 145-159Crossref PubMed Scopus (91) Google Scholar, Skinner and Schaffer, 2006Skinner D. Schaffer R. Families and genetic diagnoses in the genomic and Internet age.Infants and Young Children. 2006; 19: 16-24Crossref Scopus (30) Google Scholar). We lack a significant research base that explains other uses of online communication by these parents.MethodologyThe researcher employed a phenomenological approach rooted in a hermeneutic orientation to study the experiences/feelings of mothers using online health communications. Hermeneutic phenomenology involves interpreting the "texts" of life and lived experiences (van Manen, 1990van Manen M. Researching the lived experience: Human science for action sensitive pedagogy. State University of New York Press, Albany, NY1990Google Scholar). Chronic sorrow theory aided in forming questions with regard to lived experiences of mothers using online communication and can best be described metaphorically as a "spotlight". "Theory is a spotlight, it illuminates what you see" (Maxwell, 2013Maxwell J.A. Qualitative research design: An interactive approach. Sage Publications, Los Angeles, CA2013Google Scholar). Furthermore, its use facilitated cohesion of the interview process.The researcher recruited a purposive sample of mothers of children diagnosed with ALGS using an informational flyer on Facebook and in the ALGS Alliance's newsletter. The researcher was known to the ALGS community because her daughter died from complications related to ALGS.The sample included 16 mothers, aged 31 to 53 years. Participants were White (81%; n = 13) or Black (19%; n = 3). Most were college educated (88%; n = 14), married (75%; n = 12), and employed outside the home (88%; n = 14). Fifteen participants resided across the United States; one participant was from Europe.Their children with ALGS ranged in age from 6 months to 17 years and had a variety of medical diagnoses associated with ALGS. The predominant diagnoses associated with ALGS, in addition to liver problems, included peripheral pulmonary stenosis and congenital heart disease. Five children had additional medical diagnoses not generally associated with ALGS. Five children were liver transplant recipients.Data Collection Procedures and InstrumentationThe researcher developed a two part interview guide. Part one gathered demographic information. Part two consisted of 10 open-ended questions related to the mother's experiences with online communication and her thoughts/feelings since the child's diagnosis, Given the centrality of chronic sorrow emerging from the literature, the researcher used some questions contained in the Chronic Sorrow Questionnaire–Caregiver Version (Burke et al., 1992Burke M.L. Hainsworth M.A. Eakes G.G. Lingren C.L. Current knowledge and research on chronic sorrow: A foundation for inquiry.Death Studies. 1992; 16: 231-245https://doi.org/10.1080/07481189208252572Crossref Scopus (40) Google Scholar) in part 2. Two content experts reviewed the interview guide to address possible researcher bias, a validity threat often identified in qualitative research (Maxwell, 2013Maxwell J.A. Qualitative research design: An interactive approach. Sage Publications, Los Angeles, CA2013Google Scholar).Mothers participated in one audio-recorded interview. Fifteen were interviewed by telephone, while one used Skype. The researcher transcribed interviews verbatim, reflected upon her beliefs, preconceptions, theories, motives, interview experiences, and personal biases, and documented her reflections in a journal after each interview. The researcher addressed concerns of potential biases/beliefs through "decentering", an approach advocated by Munhall, 2012Munhall P.L. Nursing research: A qualitative perspective. Jones and Bartlett Learning, Sudbury, MA2012Google Scholar, that creates an awareness of beliefs, values and ideas and constructs an "unknowing" environment to properly interpret the lived experience. The researcher's personal experience with ALGS required her to be acutely aware of her personal biases.Data AnalysisThe researcher uploaded transcriptions into the Web application Dedoose Version 4.5, 2013Dedoose Version 4.5 Web application for managing, analyzing, and presenting qualitative and mixed method research data. SocioCultural Research Consultants, LLC, Los Angeles, CA2013Google Scholar to organize and code the data. Following van Manen, 1990van Manen M. Researching the lived experience: Human science for action sensitive pedagogy. State University of New York Press, Albany, NY1990Google Scholar, the researcher engaged in "hermeneutic phenomenological reflection" (p. 77) by reflecting on and analyzing the lived experiences of the mothers. Next, she conducted thematic analysis via reflection on essential themes in relation to the lived experiences within the context of four life world existentials: "lived space, lived body, lived time and lived human relation" (p. 101). The researcher then incorporated Munhall, 2012Munhall P.L. Nursing research: A qualitative perspective. Jones and Bartlett Learning, Sudbury, MA2012Google Scholar approach by using decentering to aid in phenomenological thinking and returning to the participants for validation of the descriptions of the phenomenon. The researcher also used the reflective journal discussed above during data analysis to enhance the description of the phenomena. These techniques proved critical for the researcher due to her experiences with ALGS.Lincoln and Guba, 1985Lincoln Y.S. Guba E.G. Naturalistic inquiry. Sage Publications, Newbury Park1985Crossref Google Scholar naturalistic perspective was used to establish trustworthiness of data based on the elements of credibility, transferability, dependability, and confirmability. After data analysis, the researcher presented a summary of essential themes, interpretations and findings to one mother who experienced chronic sorrow and one mother who experienced "unpleasant feelings" (but not chronic sorrow). These mothers verified the researcher's representation of their lived experience.ResultsSurprisingly, only half of the mothers experienced chronic sorrow. The other half experienced feelings of fright, anger or being overwhelmed that the researcher classified as "unpleasant feelings". Differences in maternal support networks appeared to explain the different lived experiences. Mothers experiencing chronic sorrow had strong and consistent support from three groups: the online community, family/friends, and health care providers, while mothers experiencing unpleasant feelings lacked support from at least one of these groups. Lee experienced both feelings over time.Initially… I was angry at the doctor as well because they had tested me for Down syndrome, [and] the typically things they test women for when they're pregnant. I just could not understand for the life of me how come they had not tested me for ALGS. It was not until I went to genetic testing, realized that it was rare… your OB/GYN doesn't test for those things.Lee's main support came from her mother at that time. She lacked online support or support from others in a similar situation. However, the feelings of anger were not permanent. Lee slowly developed support networks in online communities, with other similarly-situated mothers, and with medical professionals. Lee states, "Now at times I feel more sad." The evolution of Lee's feelings shows how impairment in component(s) of support inhibits the experience of chronic sorrow.Mothers who experienced chronic sorrow coped with different feelings than mothers who experienced unpleasant feelings, but the same communication themes emerged from the data connected to both sets of mothers. One overarching theme emerged in the interviews: online communication was essential to the mothers in managing chronic sorrow or unpleasant feelings. Mothers' experiences were overwhelming, scary, and lonely, with little information or support from health care providers immediately after diagnosis. Mothers learned new ways of building relationships/connections, in addition to coping with their child's illness. They shared their joy about meeting others and establishing a "personal connection" online, however most preferred in-person meetings with other parents. Kate who reported a chronic sorrow experience stated, "I wish there was somebody closer I could get together with, talk and share our experiences, but I am very fortunate for the online part of it." Clare, a mother who reported an unpleasant feelings experience described online communication was "incredibly important" yet she wished there was a better way to communicate because "you are sharing so much and learn things you don't need to know". However, Clare ultimately shares because of the need "to know that other people are going through similar things." The overwhelming need to share experiences with others in similar situations necessitated the use of online communication for these mothers, as an imperfect substitute for in person support.Table 1 shows the four essential themes and nine sub-themes that contributed to the development of the overarching theme. The four themes were connectedness, online triggers, empowerment, and seasons of online use.Table 1Essential themes and sub-themes.Essential themesSub-themesConnectednessThere is hopeBeing part of the communityOnline triggersWe could lose our childOverload via social mediaMedical pieceEmpowermentDeciding the best place for informationWe're the expertsSeasons of online communicationUsing online communication moreUsing online communication less Open table in a new tab ConnectednessIsolation is predominant in the rare disease community, so mothers viewed connections to an online community for support as critical, "like a lifeline for a lot of us". Numerous comments reflected this sentiment: "Community is incredibly important to know that other people are going through similar things." "It's such a rare disease there is nobody local, nobody around me, at least ever heard of it. They are always like 'What the heck is that?'" "The personal connection with other parents and the personal experiences, that's what gets you through. [Online communication] was just key in me feeling connected and not being alone in it." The mothers made social comparisons, as do parents with chronically ill children:Your child always looks a little different than everyone else's child or they don't progress at the same rate and I guess that sort of solidarity is really important because it make it feel like you're not, not something so terribly wrong with you child. When you look at them compared to other Alagille children, they're doing fine, they're doing just what Alagille kids do instead of looking at the world at large and other's kids and how they progress.Mothers indicated they used online communication for multiple purposes. They often began searching using a search engine like Google and eventually became able to access information, increase knowledge and empowerment, advocate for their child, and establish relationships with others going through a similar experience.Lee started with message boards and transitioned to social media. She captured a global view of connectedness mothers experience through the evolution of online communication:the Internet [social media] give you more of a personal touch with individuals, you can always chat with them, 'Hey how's your kid?' and they can ask you…the Internet has been a wealth of knowledge, resources, information and connectionThe Internet enabled mothers to communicate, seek support and information, and feel connected with other mothers. Currently, mothers use social media, specifically Facebook and Google, most frequently to feel connected and to obtain information regarding ALGS.Online TriggersOnline communication can trigger chronic sorrow and unpleasant feelings. Common triggers include: negative information (especially early deaths), extraneous information overload, and the "medical piece". Mothers seeking information and mothers heavily involved in social media experienced the negative triggers.All mothers experienced triggers when learning of children who died from complications of ALGS, mostly on Facebook posts and YouTube, when seeking other information or support. Some mothers wondered why they subjected themselves to Facebook groups related to ALGS:Sometimes a child will die and I'm in tears every time. Every time a little angel dies I'm distraught because I think of the parents and what they must be feeling. I've been, wondering if I should be on that Facebook group at all. Every time that [a child's death] happened I wonder what I am doing here?One mother looking for videos of older children shared, "Don't YouTube Alagille, then you see the kids that have passed away and then it makes you sad.… You wanna see what kind of future they're gonna have and you see all the sad stuff too."Child loss reminded them of the fragility of their children and the unpredictability of ALGS. Lee described a child she followed on Facebook who seemed healthy, but died suddenly:Last year we lost one and I cried. I was at work and I actually had to leave work it bothered me so bad. For one, I had no idea that anything was going on and so it just, just made me cry because it was just like sudden and why?Most mothers avoided reading blogs about ALGS. They perceived blogs as generally containing negative information about children in poor health, "I try really not to [view blogs] that much because sometimes they sound so dire. I kinda don't want put myself in that kind of mind set like 'oh this is a possibility'. Alagille affects every child differently."Facebook "friends" can cause extraneous information overload. Mothers expressed the challenges associated with supporting other parents with whom they may share little in common.I know them because I am friends with them on Facebook. They post tons of stuff that is of no pertinence to me and so then that gets like almost overload for me. But, I don't want to defriend them because I know them; they have a child with ALGS and you know somewhere you can support each other too. It's like I know too many people. I know of them, but I don't really KNOW them.The final common trigger related to online searches for medical information. Sometimes when mothers found information regarding ALGS or a specific procedure, the information triggered sadness or unpleasant feelings. Kate reports, "I did read this one article and I remember reading about the survival rate age and that really kinda scared me."EmpowermentMothers recognized the value of information with statements such as "online sources for making yourself literate—it's a must; if you're gonna advocate for your child" and "I wanted information." The knowledge, information and resulting empowerment helped in providing a sense of relief and some level of control. Online communications provided immediate emotional support and empathy mothers needed to feel empowered and capable.Many mothers wrestled with the best places to get information. "It's hard to tell who's really the go-to people; where really is the best place to engage". Oftentimes, health care providers could not provide them with information or provided unhelpful/inaccurate information. "We had a hard time getting information from doctors."[Physician] was very technical and he was also very certain about the path of Alagilles and what it entails. He was like [my son] was going to have a feeding tube, and he was going to have this issue and he is going to have that issue. You know the path of Alagille is different for each child. He was kinda setting up those expectations for us and that WASN'T necessarily our path.Sometimes health care providers withheld information, complicating the mothers' online searches and their decision making ability. Mothers experienced frustration because of the professionals' lack of assistance.The worst feeling is when I have been managed by a doctor or a nurse; where they decide that they are going to give me a limited amount of information instead of all the information. I think from the medical community, for me that's the worst possible feeling. We can take it, we can understand it.Mothers developed high levels of health literacy due to the complexity involved in managing ALGS, including interactions with specialists, interpreting diagnoses, understanding medical instructions/recommendations, and engaging in complex conversations with providers. Mothers spoke of the determination and tenacity necessary to insure meaningful interactions with providers. "I remember reading the medical articles and then having to look up every other word because I didn't know what they were".Although mothers acquired more knowledge and confidence in managing their child's disease, providers often remained uninterested in collaborating or partnering with them in managing their child's disease. "There have been a few times when I have been discounted; like my opinions and thoughts were discounted because I don't have a medical degree."Seasons of Online CommunicationMothers described "seasons", a term coined by Faith. She no
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