Feasibility and Preliminary Efficacy of an Internet Support Group for Parents of a Child with Neurofibromatosis Type 1: a Pilot Study
Abstract
This pilot study investigated the feasibility and preliminary efficacy of an Internet Support Group (ISG) for parents of children with NF1. Eligible parents were recruited by email and completed baseline questionnaires assessing social support, self-efficacy, depression, and anxiety. The ISG involved eight weekly 90-min chat sessions and a discussion forum open 24 h/day for 8 weeks. Follow-up measures were completed immediately post-intervention and 3 months later. Parents from 33 families (29 mothers, 4 fathers) completed baseline measures. Over half of parents (52 %) rated their child's disease severity as mild, 33 % moderate, and 15 % severe. Among 21 parents who completed post-intervention measures, ratings of perceived emotional (p = .0008) and informational (p = .0003) support increased. There were no significant changes in self-efficacy, depression, or anxiety (ps > .05). The mean satisfaction rating was moderately high (7.6/10; range 4–10). Some parents commented that the chat sessions were at inconvenient times, which may have limited participation. Preliminary evidence in this small sample of parents suggests that ISGs may be a feasible and potentially efficacious method of providing support to parents of children with NF1. Having multiple weekly chat sessions held at various days and times may improve accessibility and participation. Clinicians are encouraged to help parents access online support resources.
Introduction
Chronic illness in youth poses numerous challenges to the parents who care for them. Across various pediatric populations, psychological functioning in parents is compromised by the stress of having a child with special healthcare needs. For example, parents of children with cancer are more stressed than control groups and normative samples (Grootenhuis and Last 1997) both immediately after diagnosis (Patino-Fernandez et al. 2008) and for one or more years following diagnosis (Boman et al. 2003; Hoekstra-Weebers et al. 1999). Similar results have been found among parents of children with more long-term conditions such as heart disease (Arafa et al. 2008) and epilepsy (Chiou and Hsieh 2008). In addition, proportions of parents of children with chronic medical conditions display significant symptoms of depression (Iseri et al. 2006; Szabo et al. 2010; Wood et al. 2008) and/or anxiety (Kreutzer et al. 2009). In studies involving qualitative interviews of parents of children with chronic illness, social isolation, withdrawal, and changes in social roles are frequently reported (Klassen et al. 2011; Kratz et al. 2009; Rodriguez and King 2009).
Neurofibromatosis type 1 (NF1) is a rare, autosomal dominant condition caused by a mutation on chromosome 17 affecting approximately 1 in 3500 individuals (Tonsgard 2006). The clinical presentation varies considerably, within and between patients (Rieley et al. 2011), and includes plexiform neurofibromas (PNs), café au lait macules, optic pathway gliomas, short stature, Lisch nodules, scoliosis, malignant peripheral nerve sheath tumors (MPNSTs), as well as cognitive deficits, learning disabilities, and social-emotional problems. Moreover, several of these symptoms (e.g., PNs, scoliosis) can be disfiguring, often cause significant pain (Kim et al. 2009; Wolters et al. 2015), and negatively impact quality of life (Wolkenstein et al. 2009).
Few studies have examined the well-being of parents of children with NF1. Reiter-Purtill et al. (2008) found that greater neurological impairment in the child with NF1 was associated with lower perceived social support and greater maternal distress compared with mothers of children with less neurological impairment. More recently, Barke et al. (2016) conducted qualitative interviews with parents of a child with NF1. Participants noted difficulties in accessing social support and in finding accurate information about the disease. Other aspects of parental functioning in NF1 have not been given adequate attention in the literature to date.
Positive Impact of Social Support among Parents of Children with Chronic Illness
There is evidence that social support helps parents of children with chronic illness (Rearick et al. 2011). The rapid expansion in computer-mediated communication has fostered new opportunities for these parents to participate in peer-to-peer supportive interactions within an online network of similar others. Specifically, internet support groups (ISGs) allow for connections with peers any time of the day or night from the privacy of one's home (Hinton et al. 2010; Malik and Coulson 2008). They enable users to reach out to a large, diverse group of people, which is particularly beneficial for those who are isolated geographically or due to stigmatized conditions, or for those living with a rare disease. Also, ISGs allow for user anonymity and facilitate a sense of empowerment or improved self-efficacy (Barak et al. 2009; Malik and Coulson 2008; Shaw 2000).
Research with parents of chronically ill children has found that ISG participation can lessen the impact of the child's disorder (Binford Hopf et al. 2013). Other positive outcomes include satisfaction with participation, improved parent-child relationships, and hopefulness (Baum 2004). While we are unaware of any studies of ISGs conducted with parents of children with NF1, a recent survey by our group indicated that considerable percentages of these parents were likely or very likely to use an ISG to find out about research studies (87 %), talk to other parents of children with NF1 (67 %), and get answers to questions about NF1 (50 %; Martin et al. 2014). Parents further indicated interest in a discussion forum to post messages for a health professional (67 %) and other parents of children with NF1 (67 %). There also was substantial interest in a chat room where they could talk to a health professional (67 %) and other parents (53 %) in real time.
Purpose of the Study
Several chat rooms and discussion forums for individuals with NF and their families exist, yet to our knowledge, none have been studied empirically and none are specifically geared toward parents of these children. Therefore, the aim of this pilot study was to assess the feasibility and preliminary efficacy of an ISG on perceptions of social support, as well as self-efficacy for managing the child's disease, depression, and anxiety among parents of a child with NF1.
Methods
Eligibility Criteria
Approval for this study was obtained from the Institutional Review Board of the National Cancer Institute (NCI). Eligible participants needed to be the primary caregiver of one or more children ages 0 to 25 with a confirmed diagnosis of NF1. They also must have reported living in the same home as the child most of the time, being at least 18 years old, having regular internet access, and being able to read and communicate in the English language. Individuals could participate if they were part of another NF internet forum, but they were excluded if they reported a plan to start a new ISG during the study period.
Measures
Instrumentation
Demographic Questionnaire
An 11-item Demographic Questionnaire developed by the researchers was administered to obtain information about the participants’ relationship to the child with NF1 (mother, father, or other relative/legal guardian), the parent's NF1 status, and whether participants had taken part in an ISG before. Participants also provided the name of a contact person in case concerns arose about the participant's welfare (e.g., if they expressed thoughts of self-harm during a chat). Participants completed one item from the NF Severity Scale (adapted from Ablon (1996) by rating their child's symptoms as mild, moderate, or severe. Parents with more than one child with NF1 were prompted to answer for each child.
Perceived Social Support
The NF1 Parent Support Scale, shown in Table 1, is a 12-item measure adapted with permission from the Diabetes Support Scale (DSS; Barrera et al. 2002) by changing the word “diabetes” to “NF1” and by changing items from the patient's to the parent's perspective (e.g., “my NF1 care” was changed to “my child's NF1 care”). This new measure, the NF Parent Support Scale (NFPSS), assesses social support that could be provided online, so it is appropriate to the context of an ISG. Scores are obtained on two subscales: Emotional Support (4 items) and Informational Support (8 items). Respondents rate how much they agree with statements such as “I could contact people who have a child with NF1 just to express some of the concerns I had about my child's illness,” using a time frame of the past 8 weeks. Answer options range from Strongly Disagree (1) to Strongly Agree (5). Total scores are the mean of the 12 items (possible range: 12–60).
| Item |
|---|
| 1. I could contact people who were interested in how well I was doing with my child's NF1 care. |
| 2. It was difficult for me to reach people to get advice about my child's NF1 care. |
| 3. It was convenient for me to reach people who could give me personal information about what they did to improve their health. |
| 4. It was hard for me to find people I could contact who personally understood what it's like to have a child with NF1. |
| 5. I could contact people who could give me good advice about managing my child's NF1. |
| 6. I found it difficult to contact people who could give me the facts about NF1. |
| 7. I could contact people who have a child with NF1 just to express some of the concerns I had about my own illness. |
| 8. When I wanted advice from someone who has a child with NF1, I found that I didn't have people I could contact. |
| 9. I had people I could contact to get information about understanding NF1 as an illness. |
| 10. I didn't really have people I could contact to express some of my personal feelings about my child's NF1. |
| 11. It was easy for me to find people who could make personal suggestions about what has worked for them in handling their child's NF1. |
| 12. I didn't have access to other people with a child with NF1 who had current information about how to manage NF1 symptoms. |
Self-Efficacy
The Stanford Chronic Disease Self-Efficacy Scales (SCDSES; Lorig et al. 1996) assess confidence in one's ability to perform disease-related care tasks in individuals with chronic illness. With the author's permission, a parent-report form was created. The subscales included in this study were: Getting Information About Disease (1 item), Communication with Physician (3 items), and Managing Disease in General (5 items), for a total of 9 items. Respondents rate their confidence in their ability to accomplish tasks or activities related to managing their child's disease on 1–10 Likert scales with anchors of “not at all confident” and “totally confident.” Responses on the Communication with Physician and Managing Disease in General scales are averaged. The SCDSES have been found reliable and valid with various chronic illness populations (Jerant et al. 2008; Lorig et al. 1996).
Depression
The PROMIS Depression Scale – Short Form
The PROMIS Depression Scale – Short Form (Pilkonis et al. 2011) is an 8-item measure used with individuals 18 years and older. Respondents rate how frequently in the past week they experienced symptoms such as feeling depressed, worthless, and unhappy. Scoring is on a 0 (never) to 4 (always) format. Responses are summed so total scores can range from 0 to 32. Raw scores are converted to T-scores; scores ≥1 SD above the mean (≥60) are clinically significant. This tool has demonstrated reliability and validity in the general U.S. population and in various clinical and community samples (Cella et al. 2010; Olino et al. 2013; Pilkonis et al. 2014).
Anxiety
The PROMIS Anxiety Scale – Short Form (Pilkonis et al. 2011) is an 8-item measure used with individuals ages 18 years and older. Respondents rate how frequently in the past week they have experienced symptoms such as feeling tense and overwhelmed by worries. Item choices range from 0 (never) to 4 (always), raw scores are converted to T-scores, and higher scores suggest worse symptomatology. Preliminary reliability and validity data for this measure are acceptable (Pilkonis et al. 2011; Stone et al. 2015).
Post-Study Questionnaire
The Post-Study Questionnaire, developed by the researchers, includes 16 questions about participants’ experience during the study. Items ask about the best and worst aspects of the chat room and discussion forum and study satisfaction. This measure includes ten 5-point Likert scale items, two questions assessing study satisfaction on 0–10 scales, and four open-ended questions.
Procedures
Recruitment emails were sent to two groups of potential participants. First, parents were emailed if they had a child enrolled on a protocol for NF1 at the NCI. Second, the Children's Tumor Foundation (CTF) maintains a registry of over 6000 individuals with NF1, many of whom are minors whose parents registered them. Among parents who indicated a willingness to be contacted about research studies, a random sample was sent the recruitment email. The emails described the study briefly, and included a link to a webpage hosted by CTF that included more details and allowed interested people to sign the consent document online. In total, 466 recruitment emails were sent: 86 were sent to individuals at the NCI, and 380 were sent through the CTF registry. Groups of emails were sent each week until a minimum of thirty participants had registered.
The informed consent process was conducted online, following published ethical guidelines and best-practice recommendations (Buchanan and Zimmer 2012; Vayena et al. 2013). Participants read the consent document and were required to click an “I agree” button at each section in order to proceed. The consent also required participants to agree to guidelines governing use of the chat room and discussion forum (for example, not giving medical advice, being respectful, and refraining from using inappropriate or offensive language). Users were informed that a health professional would monitor communications in the chat room to promote safety and to correct any misinformation. A link to these guidelines remained on the website for the study duration.
After consenting, individuals were directed to an explanation of how to navigate the site and were asked to complete baseline questionnaires online, which were expected to take about 20 min. When a participant completed the baseline questionnaires, an automated email was sent to the Principal Investigator (PI). The PI reviewed questionnaire responses within 48 h. Participants were informed that if they obtained a T-score of ≥70 on the depression measure, indicating severe depression, an investigator would contact them to assess the appropriateness of their participation and, if necessary, make a referral for mental health services. No participants met this cut-off.
There were two components to the ISG: a chat room and a discussion forum, both housed by the study website for 8 weeks. Chat rooms are typically open for scheduled periods of time when users can communicate in real time. For this study, the chat room was open during one scheduled 90-min evening session each week. During these sessions, users could discuss any topics of interest, related or unrelated to NF1. Emails reminding people about the start date and time of the next chat session were sent on the day of the chat. One monitor reviewed the chat posts in real time each week to ensure compliance with the guidelines; the monitors included the PI, who is a licensed psychologist, or a graduate level psychology research assistant supervised by the PI.
Discussion forums are structured so that users can post questions or comments, or respond to comments from others, at any time of the day or night. In the study discussion forum, the PI posted one question per week for participants to discuss. Participants also could start their own topic threads. The PI monitored the discussion forum at least daily, excluding weekends. The PI occasionally responded to participants’ posts to try to keep the thread active, and answered questions posed to the health professional within 48 h. More complex medical questions were referred to the nurse-practitioner for responses. Topics were based on the survey results described previously (Martin et al. 2014; see Table 2).
| Week in ISG | Moderator question |
|---|---|
| Week 1 | There are a lot of different symptoms of NF1. What are the most difficult symptoms your child has had to deal with? |
| Week 2 | Do people treat your child differently because of his or her NF1? If so, how? |
| Week 3 | Does your child have pain? If so, how do you help him or her cope? Does it get in the way of his or her schoolwork or other activities? |
| Week 4 | What has been your experience with research treatment protocols? For people who have a child who has enrolled on a treatment protocol, how did you find out about the study? Do you feel like your child benefitted? Would you do it again? |
| Week 5 | Do you spend a lot of time worrying about your child's NF1? How do you cope with having a child with a chronic illness? How does it affect your child's life? How does it affect your life? |
| Week 6 | Does anyone have a child who has been diagnosed with a learning disability or who has cognitive problems? How about ADHD (Attention Deficit Hyperactivity Disorder)? How does it affect his or her school or work? Did he/she need special services in school? |
| Week 7 | There are treatment studies out there, but some people are worried about the side effects these treatments might cause. Do you think it would be hard to weigh the advantages and disadvantages of enrolling your child on a treatment study? |
| Week 8 | Are there any positive aspects of your child having NF1? What are they? |
Post-Study Evaluations
At the end of week eight (Time 2), participants were emailed a link to the follow-up questionnaires, which included all baseline questionnaires except the Demographic Questionnaire, as well as the Post-Study Questionnaire. Participants who did not complete the questionnaires within 1 week were emailed a reminder. Three months after the website closed (Time 3), participants were emailed a link to the same questionnaires to complete once more.
Data Analyses
The primary endpoint for this study was change in perceived social support (mean score on the NF1 Parent Support Scale, our primary outcome measure) from baseline to immediately post-intervention. Secondary outcome measures assessed self-efficacy, anxiety, and depression. Power calculations were based on a 1-sided .025 level t-test. Twenty-one patients were necessary with 85 % power to detect a 1-point change at each time point. However, we enrolled 33 participants in order to maximize the likelihood of interactive communication in the ISG. We compared baseline data from the completers (baseline and Time 2 measures) versus non-completers (baseline only) using independent t-tests. We also examined changes in secondary outcome measures from baseline to 8 weeks, and planned to assess changes in primary and secondary outcome measures from baseline to 3 months post-intervention; however, the small number of participants who completed Time 3 measures did not allow for the latter. Pearson correlations were conducted to assess relationships between study satisfaction and primary and secondary outcome measures, as well as pre- to post-intervention difference scores.
Results
Participant Characteristics
Thirty-three participants completed the online consent and baseline measures (29 mothers, 4 fathers; see Table 3). One parent had two children with NF1, and all others had one. No parents reported having NF1, while two said that their NF1 status was unknown. The age of the child's diagnosis ranged from less than 1 year to 9 years (mode = <1 year). The child's age at the time of parental participation ranged from 2 to 23 years. Parent ratings of their child's NF1 severity were mild in 52 % of cases, moderate in 33 %, and severe in 15 %. Over half (59 %) of participants had never used an ISG previously to connect with other parents of children with NF1. There were no significant mean score differences on any of the baseline measures between those who completed Time 2 measures (n = 21) and those who did not provide data at Time 2 (n = 12; ps > .05).
| Characteristic | n | % |
|---|---|---|
| Female gender (parent) | 29 | 88 |
| Parent NF1 diagnosis | ||
| Yes | 0 | 0 |
| No | 31 | 94 |
| Unknown | 2 | 6 |
| Past ISG use for NF1 | ||
| Never | 19 | 58 |
| Yes, in the past | 7 | 21 |
| Yes, currently | 7 | 21 |
| Age of child diagnosis* | ||
| Less than 1 year | 20 | 59 |
| 1–5 years | 11 | 32 |
| 6–10 years | 3 | 9 |
| 11+ years | 0 | 0 |
| Age of child currently* | ||
| 1–5 years | 9 | 26 |
| 6–10 years | 6 | 18 |
| 11–15 years | 9 | 26 |
| 16–20 years | 6 | 18 |
| 21–25 years | 4 | 12 |
| NF1 disease severity* | ||
| Mild | 17 | 50 |
| Moderate | 12 | 35 |
| Severe | 5 | 15 |
- *One parent had two children with NF1 so the total adds up to 34
Use of Chat Room and Discussion Forum
Twenty parents (61 %) posted in either the chat room and/or discussion forum. In the chat sessions, the mean number of posts over the 8 weeks was 138 per session (range 35–262). The mean number of participants across the eight chat sessions was 5.9 (range 3–10 per session). Regarding the discussion forum, in addition to the eight posts (one per week) from the monitor to generate discussion, 11 other topic threads were started by participants. These topics included medical issues (e.g., pain, MRI scans), social-emotional challenges (e.g., behavior problems, making friends), and research (e.g., investigational treatments). There were 91 forum posts by parents over the 8-week ISG. While it was not possible to track who logged in without posting a comment, there were 487 views in the forum, collectively. There were no instances of the guidelines being violated in the chat room or discussion forum. Baseline scores on social support, self-efficacy, depression, and anxiety, as well as disease severity, were unrelated to whether a participant posted in the chat room or discussion forum (ps > .05). Also, the child's age and disease severity were unrelated to study satisfaction or ratings of the helpfulness of the ISG (ps > .05).
Baseline Functioning on Primary and Secondary Outcome Measures
As shown in Table 4, the baseline mean NF Social Support score was 2.85 ± 0.75 on the 1 to 5 scale (higher scores indicate more perceived support). An examination of item responses shows that 67 % of parents answered Agree/Strongly Agree to the statement: “It was hard for me to find people I could contact who personally understood what it's like to have a child with NF1.” Sixty-one percent answered Agree/Strongly Agree to the statement: “I didn't really have people I could contact to express some of my personal feelings about my child's NF.”
Regarding secondary measures, parents’ self-efficacy scores were fairly high on the 1–10 Likert scale of the SCDSES (mean Total = 7.15 ± 1.56), as well as on the Communication with Physician (M = 7.8 ± 2.29) and Managing Disease in General (M = 7.2 ± 1.83) subscales. On the 1-item Getting Information About Disease subscale, the mean score was much lower (4.95 ± 2.46). Finally, mean scores on the PROMIS Depression and Anxiety scales were within normal limits. No baseline depression scores were in the clinically severe range (≥70).
| Measure | Response range | Baseline | Time 2 | t | p |
|---|---|---|---|---|---|
| NFSS* | 1–5 | 2.85 (0.75) | 3.85 (0.70) | -4.45 | 0.0003 |
| SCDSES | 1–10 | 7.15 (1.56) | 7.05 (1.80) | -0.30 | 0.76 |
| PROMIS depression | 0–4 | 51.50 (5.40) | 50.43 (7.10) | -0.35 | 0.72 |
| PROMIS anxiety | 0–4 | 53.71 (4.50) | 55.10 (4.60) | 0.64 | 0.52 |
- NFSS NF Support Scale, SCDSES Stanford Chronic Disease Self-Efficacy Scales, PROMIS Patient-reported Outcomes Measurement Information Systems. For the PROMIS scales, raw scores are converted to T-scores, and are compared to a mean of 50 and standard deviation of 10. *n = 20 for this measure as one participant did not complete this form
Pre to Post-ISG Differences
On the primary outcome measure (NFSS), parents’ mean social support scores significantly increased from baseline to post-ISG participation (t = 4.45, p = .0003; see Table 4). Analysis of subscale score differences indicated that perceptions of both emotional (t = 3.99, p = .0008) and informational (t = 4.48, p = .0003) support increased.
On the secondary measures of self-efficacy, depression, and anxiety, no significant pre- to post-intervention differences emerged (all ps > .05). When only participants who had posted in the chat room or discussion forum (n = 16) were included in the analyses, the pattern of results remained the same: the only significant pre- to post-ISG differences among this subgroup were on the NFSS (t = 4.32, p = .0007). Since only 13 participants completed Time 3 measures, insufficient power prevented repeated measures analysis of these scores.
Further Analyses of Time 2 Variables (n = 21).
On the 5-point Likert scale questions on the Post-Study Questionnaire, similar, high levels of perceived support were indicated from the chat room (M = 4.0 ± .86, range 3–5) and discussion forum (M = 4.2 ± .70, range 3–5). Similarly, the chat sessions and discussion forum were rated as helpful by 67 % and 86 % of the parents, respectively. The mean study satisfaction rating was moderately high (7.6 on a 1–10 scale; range 4–10). Moreover, 95 % of participants were likely or very likely to recommend an ISG to other parents of children with NF1. Ninety-five percent of participants reported that the questions posed by the moderator were helpful. Pre- to post-ISG improvements in primary and secondary outcome scores were unrelated to overall study satisfaction or the likelihood of recommending an ISG to others (ps > .05). However, a nonsignificant trend suggested that more improved anxiety scores from baseline to Time 2 tended to relate to higher perceptions of being supported in the chat room (r = .41, p = .06).
Open-Ended Comments
Eighteen participants provided responses to open-ended questions from the Post-Study Questionnaire; comments were largely positive and provide useful input for guiding future ISG development. When asked about the most helpful aspects of the study, parents commented mostly on the emotional support received from connecting with similar others. For example, one mother noted that “expressing in a ‘safe place’ the fears I have for my child's development and the guilt associated with that [was helpful]. I found that I was certainly not alone and I felt supported and encouraged.” Another parent remarked on the informational support she received, saying, “I felt for one of the first times since my daughter's diagnosis that I had a place to go for real information.” Some parents commented that the chats were at inconvenient times and should have been more frequent; this factor likely limited participation, particularly among those in other time zones. Others suggested saving chat transcripts for later viewing for those who could not attend.
Discussion
To our knowledge, this pilot study is the first to suggest that parents of children with NF1 may benefit from ISG participation. Importantly, parents felt that they were supported emotionally and in terms of the information they received from other participants. This is encouraging, and indicates that parents of children with NF1 can benefit from these forums. Indeed, our baseline results suggest that many parents perceived a lack of a support network surrounding their child's disease, indicating a real need for supportive interventions such as ISGs.
The parents who did not post in either the chat room or discussion forum showed similar increases in social support scores than those who were more actively involved in the ISG. Research on those who view posts but do not post themselves suggests that this “lurking” behavior is quite common in online groups. Factors such as personality traits and time availability are potential reasons for lurking (Amichai-Hamburger et al. 2016). While lurkers do not obtain all the benefits of more active participants, they do tend to improve (similarly to the posters) in certain factors such as self-efficacy, feeling more informed, and increased optimism and self-esteem (Mo and Coulson 2010; van Uden-Kraan et al. 2008).
Our preliminary results did not find any significant changes in self-efficacy, depression, or anxiety. This is not too surprising, since these domains were not directly targeted by the intervention, which was aimed at increasing social support. Other online forums have found improvements in self-efficacy when there was a specific component geared toward “parent empowerment” (Swallow et al. 2014). While several studies have found decreased depressive or anxious symptoms after ISG participation, others have not (Clifford and Minnes 2013; Crisp et al. 2014; Hoybye et al. 2010). One possible reason for a lack of improvement in our secondary measures is that the parents were doing fairly well at baseline and therefore there was little room for improvement. Baseline mean scores on the depression and anxiety scales were solidly within normal limits. Another possibility is that our ISG was not long enough for people to benefit more in these domains. Griffiths et al. (2012) found no changes 3 months post-ISG among a community sample, but improvements were seen 6 and 12 months after the intervention. An ISG investigation for parents of children with autism found no differences in depression or anxiety between the intervention and control groups, or over time (Clifford and Minnes 2013). These authors speculated that low chat participation and the relatively brief intervention may have contributed to the lack of significant findings; their intervention was similar in length to that of the current study (eight 1-h sessions). A longer intervention with more participants might lead to improvements in additional domains. Nonetheless, the identified improvements in our primary outcome, social support, speak to the value of the intervention.
The perceived helpfulness of the ISG was irrespective of the child's disease severity, suggesting that even parents with minimally impacted children can benefit from communicating with similar others. It was surprising, however, that no parents reported having NF themselves. Perhaps parents who have NF1 have been dealing with the disease for so long that they do not feel the need to seek out support from others, but this hypothesis warrants investigation.
Given the high study satisfaction ratings, the number of posts, and the low cost and personnel required, an ISG for parents of children with NF1 seems to be a feasible intervention. Open-ended responses suggest that having more than one weekly chat session at varied times may have enabled more users to participate and thus brought about more chat activity.
Almost all parents (95 %) thought that having health professional involvement (i.e., weekly questions posted by the moderator) was helpful. While a moderate amount of time was required by the website developer to set up the site initially, very little time was required once the system was in place. Aside from the 90 min required to monitor the weekly chat sessions, approximately 30 to 60 min per week were spent reviewing discussion forum posts, responding to questions directed to health professionals, and/or clarifying points of confusion that had been posted. Only three comments or questions were posed by parents that required a response from a health professional; thus, managing the site was not considered burdensome. However, having multiple moderators was helpful in terms of keeping each professionals’ time commitment reasonable.
Study Limitations and Research Recommendations
Appropriate caution should be used in the interpretation of our results, given the preliminary nature of this uncontrolled pilot study. Future research using a randomized controlled trial design will be able to make more definitive statements about the effectiveness of ISG interventions for parents of children with NF1.
As mentioned previously, several factors may have contributed to a lack of significant findings on the secondary outcome variables. First, we had a relatively low response rate from the original number of people who received the recruitment email. Having additional chat sessions at various times may have led to more participation, and potentially more impact on self-efficacy and mood. In addition, future ISGs could target self-efficacy in particular, such as by posting questions to the group about how confident they are in communicating with their child's physician, for example.
The fact that only parents without NF1 responded to our invitation to participate limits the generalizability of the findings. It may be that parents with NF1 have already been “hooked in” to other supportive resources as a result of their own diagnosis, and did not feel the need to access additional support. Further, as stated earlier, levels of baseline depression and anxiety among participants were within the normal range; perhaps those parents who received the recruitment email and who were struggling emotionally did not view the ISG as a resource that would be helpful or did not feel comfortable with the idea of sharing their struggles with people they did not know. Indeed, these parents may have been accessing support from alternative sources (e.g., in-person counseling). Future efforts should explore how to reach parents who have NF1 and need emotional support. Moreover, studies with larger samples would allow for an examination of mediating variables that may help identify participant characteristics (such as parent or child age and gender, or parent emotional well-being) associated with the effectiveness of the intervention.
Another fruitful research direction would be to explore parent and child variables that may relate to outcomes. In our small sample, child age was unrelated to parent level of study satisfaction or the helpfulness of the intervention. We certainly had more mothers than fathers participate, but because of our very small number of fathers in the study (n = 4), we could not analyze parent gender as a potential moderator of helpfulness. Factors such as whether a parent is already connected with similar others (e.g., through in-person support groups or NF community resources) and their knowledge about NF1 may also predict level of interest in ISG participation. Larger studies may be helpful in answering these questions.
Finally, it would be helpful to examine ISG effectiveness for patients with NF1 across various age groups, including adolescents, who are frequent users of online social forums (Madden et al. 2013). Studies of online supportive interventions for youth with other chronic conditions support their effectiveness in reducing social isolation and increasing goal attainment (Letourneau et al. 2012; Raghavendra et al. 2013).
Practice Implications
Clinicians are encouraged to help parents of children with NF1 access online support resources. This is especially important for parents who are not able to come to in-person groups. Interested health professionals also should explore opportunities for becoming involved in an ISG, as the experience can be both rewarding and educational.
Conclusions
Preliminary evidence in our small sample of participants suggests that ISGs may be a feasible and effective means of connecting and providing support to parents of children with NF1 who are dealing with a rare and complex genetic condition. A forum where parents can receive both emotional and informational support from similar others and from a health professional is a feasible model. Having multiple weekly chat sessions held at various days and times likely would improve accessibility for geographically diverse individuals. Given the vastly increasing reach of social media, providing online interventions to caregivers is an efficient and inexpensive method of delivering assistance to those who need it most.
Acknowledgments
The authors appreciate the participation of the parents who took part in this study. We also thank the Children's Tumor Foundation for providing the online infrastructure for the internet support group. This research was supported by the Intramural Research Program of the National Institutes of Health, National Cancer Institute. In addition, this project was funded in whole or in part with federal funds from the National Cancer Institute, National Institutes of Health, under Contract No. HHSN261200800001E.
Compliance with Ethical Standards
Conflict of Interest
S. Martin, M.C. Roderick, R. Lockridge, M.A. Toledo-Tamula, A. Baldwin, P. Knight, P. Wolters declare that they have no conflict of interest.
Human Studies and Informed Consent
All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.
Informed consent was obtained from all individual participants included in the study.
Animal Studies
No animal studies were carried out by the authors for this article.
