Is the process of delivery of an individually tailored lifestyle intervention associated with improvements in LDL cholesterol and multiple lifestyle behaviours in people with Familial Hypercholesterolemia?

Participants of the PRO-FIT trial were recruited from the national cascade screening program of the Foundation for the Identification of Persons with Inherited Hypercholesterolemia (StOEH). Within this program, the StOEH actively approaches first and second degree relatives of index patients (that is, clinically diagnosed FH patients with a known mutation) about their potential risk by mail. A genetic field worker telephones and, if the family member agrees to participate, makes an appointment for testing at home. If the results of DNA analysis are positive, first and second degree relatives are approached and offered testing, and so on. No further counseling is given within the screening program.[28] Within the PRO-FIT project, individuals were invited who were diagnosed with FH by StOEH from January 1^st 2007 to April 15^th 2009, no longer than 2 years before the start of the project. Access to internet, sufficient fluency in Dutch, residency < 150 km radius from Amsterdam, age 18–70 and LDL-C > 75^th percentile were eligibility criteria. People were invited by postal mail and telephoned in case of no response. When people decided to participate, the study procedure was explained by telephone. After randomised allocation to the intervention group, participants were encouraged to visit a weblink referring to the project website, on which they could log on to a personal PRO-FIT*advice account. This account gave access to six tailored advice modules on smoking, physical activity, saturated fat intake, fruit intake, vegetables intake and compliance to statin therapy. Each module required the completion of a screening questionnaire. Subsequently, on-screen personalised feedback was tailored to personal performance level (current lifestyle behaviour), awareness of one’s own performance, as well as personal motivation to change, outcome expectations, attitude and self-efficacy. Personalised feedback to compliance to statin therapy was tailored to knowledge and personal beliefs about (the effect of) statin therapy, potential side effects of the prescribed drug and current compliance. After finishing a module, participants were encouraged to make action plans to change behaviour (except for the advice module on compliance to statin therapy). Thereafter, in a face-to-face session, the participant and the personal coach together further established the level of the participant’s knowledge/awareness about FH and cardiovascular risk factors, according to the assessment(s) and advice(s) within the participant’s personal PRO-FIT*advice account. Ambivalence and barriers related to the recommended behaviour changes were explored in a face-to-face session based on MI techniques. Further, the participant was encouraged to plan five additional counsellor-initiated booster telephone calls, according to their need for additional counselling, intended to support the participant’s behavioural changes and to provide further brief MI to encourage the planned behavioural changes. The two personal coaches had lifestyle coaching and nursing/ teaching backgrounds and had received an additional 3-day MI workshop, incorporating both introductive lessons and practical training sessions with professional actors. A schematic overview of the intervention can be found in Figure 1, including the assumed efficacy pathways of the intervention (associations A-D). A more detailed description of the PRO-FIT intervention can be found elsewhere [7]. The ethical principles of the Helsinki Declaration were followed and the PRO-FIT project was approved by the Medical Ethics Committee of the VU University Medical Centre (reference number: NL23932.029.08). All participants gave written informed consent.

The RE-AIM evaluation framework conceptualised the evaluation of the translatability of an intervention and included reach, efficacy, adoption, implementation and maintenance [29]. Linnan and Steckler also included implementation among their key components of a process evaluation including context, reach, dose delivered, dose received, fidelity, implementation and recruitment [3]. Both authors agree on reach, implementation, including dose and fidelity as important factors for process evaluations. Generally, reach is defined as the number of people of the target population taking part in the project and their representativeness with regard to the target population. Dose is either defined as ‘dose delivered’, i.e. the number of components of the intervention delivered, or as ‘dose received’, i.e. the extent to which the participants used the components of the intervention as intended. Fidelity is defined as the extent to which the intervention was implemented as intended.

Guided by Saunders and colleagues, a process evaluation plan was developed in order to monitor and document the implementation of an intervention [30]. In this plan, the evaluated intervention was described in detail, including its specific strategies as well as what would be entailed in a complete and acceptable delivery of the intervention. Consequently, a list of potential process evaluation questions and measures was made and answered by using self-formulated methods (see Table 1).

In order to assess the number of people with FH that took part in the project, as well as how representative the participants in the intervention group were for the study population and non-participants (people who did not respond to the invitation to participate, or people who chose not to participate), the StOEH client database, as well as the PRO-FIT client database were consulted. Differences between participants and non-participants in main characteristics (age, gender, and LDL-C levels) were explored.

The number of participants who had received a PRO-FIT*advice log on account, a face-to-face counselling session and subsequent telephone booster calls (dose delivered), was assessed by logs that were kept by the coaches and stored in the project database. We aimed at a 100% delivery of the intervention components and delivery of one to five telephone booster calls. The way participants used the PRO-FIT*advice log on account (dose received) was assessed by exploring participants’ log on behaviour (% of participants that logged on), as well as participants’ actions on the PRO-FIT* advice account (number of modules finished,% of participants that had made online action plans) by means of log on rates and website use data.

Whether face-to-face counselling sessions were implemented as planned according to MI guidelines (i.e. MI fidelity) was assessed by two MI experts, following the Motivational Interviewing Treatment Integrity code (MITI 3.1.1.) [31]. The MI experts were attached to the Foundation Centre for Motivation and Change (Hilversum, the Netherlands), that works in cooperation with the International Motivational Interviewing Network of Trainers (Virginia, US; http://​www.​motivationalinte​rviewing.​org), and were trained in coding fidelity using the MITI 3.1.1. For this assessment, a random sample of 20 audio taped counselling sessions (10 sessions of each lifestyle coach; approximately 10% of all sessions) was drawn. A verbatim transcript [32] of each drawn session was evaluated and resulted in two scores: a global score and behaviour counts. The global score captured an overall impression of the conversation on a 5-point Likert scale for the following 5 dimensions: Evocation, Collaboration, Autonomy/Support, Direction and Empathy. In addition, the behaviour counts capture specific behaviours of the lifestyle coach, such as the number of open/closed questions, simple/complex reflections, MI (non)adherent utterances and provision of information. We aimed for 100% of the counselling sessions to be provided according to MI. Counselling sessions were considered MI if the following conditions were met: average of global scores ≥ 3.5, reflection to question ratio is in favour of reflection, >50% open questions, >40% complex reflections and >90% MI-adherent utterances. The total scores were weighed for the number of counselling sessions conducted by each coach.

The level of physical activity was measured by the Short QUestionnaire to ASsess Health-enhancing physical activity (SQUASH) and was expressed as minutes of moderate to vigorous physical activity performed per week [33]. Saturated fat, fruit and vegetables intake were measured by the short Dutch questionnaire on total and saturated fat intake and on fruit and vegetable intake. From this questionnaire, a score for saturated fat intake, ranging from 0 (lowest) to 80 (highest) fat points was computed, as well as servings of fruit and grams of vegetables per day [34‐36]. Smoking behaviour was assessed by a self-reported measure, resulting in a score of 0 (non-smoker) or 1 (smoker) [37]. The five-item Medication Adherence Report Scale (MARS-5) was used to measure self-reported compliance to statin therapy. Scores on five items were combined to a total score ranging from 5 (lowest) to 25 (highest). Participants with a score of 25 were categorised as compliant to statin therapy, others (score < 25) as non-compliant [38].

At baseline and 12-month follow-up, the participants’ LDL-C was assessed at the participant’s home with fasting finger stick samples analysed on a Cholestech LDX desktop analyser (Cholestech, Hayward, USA). This portable analyser is capable of providing a lipid profile in approximately 5 minutes. The reproducibility and precision of lipids measurement using the LDX analyser are within the guidelines of the National Cholesterol Education Program (NCEP) [39, 40]. The Cholestech LDX analyser has been validated for point-of-care lipid measurements in clinical practice [41].

Differences in age, gender, and LDL-C levels between participant and non-participant characteristics were checked with linear and logistic regression analyses for each variable separately. Associations between intervention dose and lifestyle behaviours and LDL-C (associations A-D) were explored with linear (for physical activity, fat/fruit and vegetables intake and LDL-C levels) and logistic (for smoking and compliance to statin therapy) regression analysis with the following independent variables: logged on at PRO-FIT*advice and advice module completed (yes/no) (association A), face-to-face counselling received (yes/no) (association B), number of telephone booster calls (association C), and the complete intervention-package (at least one PRO-FIT*advice module, face-to-face counselling and at least one telephone booster call) received (yes/no) (association D). The post-test scores of the dependent variables were regressed to the baseline measures. Effect parameters (regression coefficient (beta) or odd’s ratio (OR)) either indicated a positive association if LDL-C/lifestyle behaviours improved when regressed to the intervention dose, or a negative association if vice versa. An association was considered as significant if p < 0.05.

During the six months of recruitment for the PRO-FIT project, nearly 6200 people in the Netherlands were screened by StOEH, of whom an averaged 35% actually did have FH [42]. Invitation brochures were send to 986 people who were screened by StOEH and who were positively diagnosed with FH. Of those, 340 (34%) responded and agreed to participate. This number included 23 family members of invited people who spontaneously responded and met the eligibility criteria. Reasons for not participating were mainly a lack of interest and time, and reporting to ‘already have a healthy lifestyle’. The participants did not differ from the non-participants (those who did not respond to the invitation and those who refused to participate; N = 623) in age (beta:0.23; 95% CI:-1.85-2.31) and gender (OR:0.89; 95% CI:0.68-1.16), but did with regard to LDL-C levels (beta:-0.35; 95% CI:-0.63- - 0.07) (see Table 1). The majority (57%) of the study sample was female, middle-aged (mean age = 45.3 years) , and had elevated (≥2.5 mmol/l) LDL-C levels. No significant baseline differences between intervention and control group were found. During the PRO-FIT project, five participants in the intervention group dropped out (i.e. their participation was discontinued with a given reason). Their reasons for discontinuation were no motivation (n = 1), no interest (n = 2), death (n = 1), and health constraints (n = 1).

An account to use the online PRO-FIT*advice, was provided to 172 (95%) of the 181 participants in the intervention group (see Table 2). The remaining 5% (9 participants) explicitly reported to have no interest in using PRO-FIT*advice and therefore, received no log on information. Subsequently, nearly all participants (99%) in the intervention group were visited by the lifestyle coach. Furthermore, on average of 4.2 telephone booster calls per respondent were conducted. The main reasons for not receiving subsequent booster calls was no perceived need for additional counselling because respondents regarded the lifestyle as healthy.

Of the 172 participants in the intervention group who had received a log on account, 85 (49%) actually logged on to PRO-FIT*advice, and completed at least one of the six advice modules. The most popular module, based on completion rates, was physical activity (41%), followed by fruit intake (37%), fat intake (35%), vegetable intake (34%), smoking (14%) and compliance to statin therapy (26%). Nearly one third (31%) completed at least one module and made an action plan online. Although revisiting the website was not so explicitly encouraged, 7% did. The complete intervention-package as intended, requiring log on at PRO-FIT*advice, the completion of at least one module, face-to-face counselling and at least one received telephone booster call, was delivered to 47% of the intervention group.

Eighty-five percent of the face-to-face counselling sessions were performed by coach 1, and 15% by coach 2. In Table 3, the extent to which MI was applied during the face-to-face counselling sessions by the two coaches is shown. The global scores and behavioural counts indicate that none of the sessions was implemented according to MI guidelines. Significant differences in counselling performance between the two coaches were found for using (complex) reflections, the number of MI adherent statements, the reflection to question ratio, directiveness and showing empathy.