Table 2.
Results from the process analysis organized as per process-evaluation components, related process measures, and process variables
| Process components | Findings |
|---|---|
| Study population | 1. Recruitment and selection rate |
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1a. Number of eligible persons in screened population Fifty-five patients from the Department of Clinical Genetics of Radboud University Medical Center and all contributors to the Dutch Noonan Syndrome Foundation were contacted by letter or email with information regarding the training. This information was also displayed on the websites of the Centre of Excellence for Neuropsychiatry of the Vincent van Gogh Institute for Psychiatry and the Dutch Noonan Syndrome Foundation and presented in newsletters and at meetings of the Dutch Noonan Syndrome Foundation. In total, approximately 150 patients were approached, of whom a maximum 25% were thought to be eligible, because children and adolescents aged <16 years, adult patients without social cognitive complaints, or patients who were not interested in this intervention were not eligible for participation. | |
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1b. Number of participants from sample of eligible persons Initially, 15 patients were interested in participating in the training. Five patients were excluded due to age (<16 years) or low verbal intelligence. Four other patients were eligible for inclusion but did not (yet) participate, because they could not combine the training with current work or study obligations or because they did not respond to invitations. Given the small number of patients who volunteered, it was not possible to allocate patients randomly to a treatment group and a waiting-list control group. Therefore, control participants were actively recruited at meetings of the Dutch Noonan Syndrome Foundation, and five patients were included in the control group. The control group did not follow the training at a later point in time. One patient of the control group subsequently participated in the training. No pre-or posttest information was obtained from this participant after the training because they had already been included in the control group and repeated neuropsychological assessment after participation in the training would have affected the results. The training was provided in two treatment groups (n=4 and n=3), which started consecutively. In sum, 11 patients were included in this study: six in the treatment group and five in the control group. | |
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1c. Number of participants vs aimed-at number The aim was to include 20 patients (ten in the treatment group and ten in the control group). The recruitment objective was not reached. | |
| 2. Barriers and facilitators in recruitment and selection | |
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2a. Difference in baseline characteristics between nonparticipating and participating eligible persons No information was available regarding differences between participating and nonparticipating eligible patients. There were no significant differences in demographics or baseline variables between the control group and treatment group, although this could reflect a power problem due to the small study sample size. Because the groups could not be allocated randomly, a selection bias might be present, as patients with more severe social cognitive deficits may be more inclined to participate in the treatment group. | |
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2b. Motivation of nonparticipating and participating eligible persons Some patients who were initially interested in the training could not make the required time investment in combination with current work or study activities. There also seemed to be some hesitation for participation in group training. Possibly, the nature of the (social) cognitive problems of patients with NS could provide an explanation for these findings. | |
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2c. Experience with recruitment and selection Although there appeared to be much interest for the social cognitive training, the number of patients that volunteered in the feasibility study was lower than expected. | |
| 3. Number of participants completing follow-up vs number started | |
| 3a. All participants completed the neuropsychological measures at pre- and posttest. However, not all self-report and proxy questionnaires were returned. | |
| 4. Barriers to and facilitators of follow-up | |
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4a. Reasons for dropout and motivation for continued participation There were no dropouts. All participants in the treatment group completed the training and wanted to improve their social cognitive functioning. Patients in both groups valued the importance of this study to increase the treatment options for patients with NS and were driven to contribute. | |
| Complex intervention | 1. Quality of delivery of the interventional components |
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1a. Part of each component and complete intervention delivered by instructors All three phases of the intervention (perception, interpretation, reaction) were delivered successfully. The number and difficulty of exercises could be adjusted flexibly to the level of social cognitive functioning of the group or to individual participants. | |
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1b. Satisfaction with delivery All phases appeared to be feasible. It was observed that participants were inclined to spend a lot of time on sharing their experiences at the expense of practicing social cognitive exercises. A possible explanation for this observation could be that patients with NS seldom met other patients and enjoyed exchanging experiences. However, it could also reflect avoidance of (possibly distressing) exercises or be the result of verbosity, due to executive difficulties. The time spent on psychoeducation could be shortened in favor of more (group) exercises. The training initially incorporated one proxy session without participants and one evaluation session with proxies and participants together. After evaluation of the first training group, both proxy sessions were provided in the presence of the participants. | |
| 2. Barriers and facilitators for delivery of interventional components | |
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2a. Reasons for diverging from or applying (planned) components The three phases of the training were all delivered, although the exercises were slightly adjusted to the level of social cognitive functioning of the group. Despite differences in intellectual capacities and education level, participants had comparable training aims and found many similarities between one another’s social cognitive difficulties. | |
| 3. Adherence to interventional components | |
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3a. Number of sessions followed Three participants (50%) followed all sessions. One participant missed one-and-a-half sessions due to problems with public transport. Two participants missed two sessions, because of work-related activities or health issues. When sessions were missed, participants received the information and exercises of that week by email, studied them independently, and had the opportunity to ask questions via email or telephone. On average, less than one session was missed (mean 9.08±1.02 sessions). Not all training partners attended both proxy sessions, due to the required time investment, work obligations, or cognitive difficulties. | |
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3b. Intervention components (partly) followed The participants received all three training phases. | |
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3c. Compliance with individual recommendations Individual recommendations were followed up. | |
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3d. Homework adherence All participants did homework exercises, although some needed additional guidance or support. The number and nature of the homework exercises slightly differed each week. Some patients viewed the number of exercises as too many. Also, there seemed to be a tendency to avoid exercises with training partners. Some proxies were thus less involved in the training than intended. Adequate involvement of a training partner seemed to be supportive and beneficial for the participant. | |
| 4. Barriers and facilitators for adherence to interventional components | |
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4a. Motivation for (lack of) attendance and compliance Patients appeared motivated to attend the training. They valued the importance of the training and seemed to enjoy the sessions. | |
| 5. Experience of participants and instructors with interventional components | |
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5a. Perceived benefit All participants reported that they benefited from the training. They mentioned, eg, an increased awareness of social cognitive difficulties (related to NS) and the acquisition of useful strategies to improve social cognitive functioning in daily life. In addition to the content of the training, participants seemed to benefit from recognition of one another’s experiences and the support from their group members. For some participants, this training was the first time they had come in contact with other patients with NS. Patients also mentioned that the training was intensive and demanding with regard to emotional load and time investment required. Two participants suggested more time and elaboration, eg, on social rules and social interaction. Furthermore, it was suggested by one participant that it be stated more clearly that the homework exercises could be time-consuming and to provide a contact person who patients could reach when they felt worse or relapsed. Three of the training partners who evaluated the training posttest felt that the participants benefited from the training. The training partners judged the proxy sessions as sufficient in frequency, although one partner mentioned that more proxy sessions were preferred. For the three other patients, no evaluative information was provided by their training partners. On average, general satisfaction with the training, evaluated by five participants in the treatment group, was judged as 8.9 (ranging from 0 to 10). Satisfaction with the therapeutic relationship 7.8, satisfaction with aims and topics of training 7.6, and satisfaction with approach and methods 8.4. | |
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5b. Strong and weak aspects of the interventional components (structure and content) and the total intervention Strong aspects of the training concerned its (small) group-based format, in which patients learned from each other’s experiences, increased their awareness of social cognitive difficulties, and were able to practice learned strategies. The training stimulated three participants for additional psychotherapy to continue practicing with the training topics. For two patients, information and elements from the training were incorporated into already-existing coaching or counseling trajectories. In addition, patients used the psychoeducation provided during the training to inform other people (eg, healthcare professionals or family members) or in cases of relapse. Every 6 months, participants were invited to follow up evaluation-group sessions at meetings of the Dutch Noonan Syndrome Foundation. If patients were not able to participate in this session, individual (telephone) contact was offered. Besides the small sample, a weak aspect of the training may be the slight imbalance between time spent on psychoeducation and discussion of social cognitive complaints and the amount of practice time. More concise and practical psychoeducation in combination with additional exercises may be helpful. Also, some training partners were less involved in the training than intended. | |
| Evaluation data | 1. Outcome measure: coverage of interventional components |
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1a. Average number of outcomes per component Primary outcome measures covered all three phases of social cognition that were addressed in the training: perception (Emotion Recognition Task and alexithymia questionnaire), interpretation (social perception task, theory-of-mind task, and alexithymia questionnaire), and reaction (questionnaire of social behavioral problems and alexithymia). The five secondary outcome measures concerned nonsocial cognition (speed of information processing, inhibition, and planning), psychopathology ([social] anxiety and mood problems), quality of life, and self-efficacy. | |
| 2. Completeness of data collection | |
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2a. Number and characteristics of missing data Neuropsychological test data were complete. With regard to the questionnaires, for one patient in the treatment group the BVAQ and DEX-SC were missing posttest, while the SIB was missing both pre- and posttest in this individual. For one control participant the BVAQ, DEX-SC, and SIB were missing. One individual in the treatment group missed one page of the questionnaire book, which was replaced with the pretest scores. In addition, there were nine incidental missing data points (five in the treatment group and four in the control group): six at pretest and three at posttest. Most missing data were on the BVAQ, SIB, and DEX-SC. If it was not possible to recontact participants, incidental missing data points were replaced with the score at pre- or posttest (depending on which score was missing) or with the average score on other items of that questionnaire. When two answers were given, the average score was calculated. | |
| 3. Barriers to and facilitators of data collection | |
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3a. Feasibility of outcome measures The test battery lasted approximately 2 hours but was not considered too long by participants. Questionnaires were filled out mostly at home, which possibly led to a lower response rate. Three participants in the treatment group reported trouble with some of the questionnaires, and one received help from a friend. One proxy had difficulty filling out the questionnaires and thus did not complete the posttest. | |
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3b. Reasons data were missing Reasons for attrition in returning the questionnaires at posttest were difficulty with completing the questionnaires, lack of supervision while filling out the questionnaires, psychosocial problems, and unfortunate practical complications with mail-order services. | |
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3c. Reasons participants were excluded from analysis There were no participants excluded from analyses for reasons other than missing data. With regard to heterogeneity in patient characteristics in this sample, outliers could be present. However, for the purpose of this feasibility study, all data were included. | |
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4. Comparison of qualitative and quantitative effectiveness data The perceived benefit by patients and training partners was not reflected by the quantitative data. Finding no significant improvement on primary or secondary outcome measures may be the result of a power problem due to the small sample. Furthermore, it is possible that some tests in the current battery were not able to detect differences adequately between pre- and posttest. |
Abbreviations: BVAQ, Bermond–Vorst Alexithymia Questionnaire; DEX-SC, Dysexecutive Questionnaire – social conventions; NS, Noonan syndrome; SIB, Scale for Interpersonal Behavior.