Patient and carer involvement in the formulation of research questions: findings from the Italian guideline on palliative care in adults with glioma.

BACKGROUND: In 2017, the European Association for Neuro-Oncology (EANO) published the guideline for palliative care (PC) in adults with glioma. The Italian Society of Neurology (SIN), the Italian Association for Neuro-Oncology (AINO), and the Italian Society for Palliative Care (SICP) joined forces to update and adapt this guideline to the Italian context and aimed to involve patients and carers in the formulation of the clinical questions. METHODS: During semi-structured interviews with glioma patients and focus group meetings (FGMs) with family carers of deceased patients, participants rated the importance of a set of pre-specified intervention topics, shared their experience, and suggested additional topics. Interviews and FGMs were audio-recorded, transcribed, coded, and analyzed (framework and content analysis). RESULTS: We held 20 interviews and five FGMs (28 carers). Both parties considered the pre-specified topics as important, chiefly information/communication, psychological support, symptoms management, and rehabilitation. Patients aired the impact of focal neurological and cognitive deficits. Carers reported difficulties in dealing with patient's behavior and personality changes and appreciated the preservation of patient's functioning via rehabilitation. Both affirmed the importance of a dedicated healthcare path and patient's involvement in the decision-making process. Carers expressed the need to be educated and supported in their caregiving role. CONCLUSIONS: Interviews and FGMs were well informative and emotionally challenging. Both parties confirmed the importance of the pre-specified topics, and carers suggested one additional topic: education/support to caregivers. Our findings strengthen the importance of a comprehensive care approach and of addressing the needs of both patients and their family carers.

Evaluation of a program for training psychologists in an acceptance and commitment therapy resilience intervention for people with multiple sclerosis: a single-arm longitudinal design with a nested qualitative study.

PURPOSE: This single-arm longitudinal study evaluated the effectiveness of a program for training psychologists in delivering an acceptance and commitment therapy-based program (REsilience and Activities for every DaY; READY) for people with multiple sclerosis (PwMS). MATERIALS AND METHODS: The training encompassed three phases: (1) training workshop; (2) READY participation; (3) READY delivery to PwMS. Self-report data were collected immediately before the workshop, before and after participation in READY, and at 3- and 15-month follow-ups. RESULTS: Forty psychologists successfully completed the training. The training was effective in fostering the acquisition of knowledge and skills for effective delivery of READY to PwMS. Participants improved over the course of training in resilience, positive affect, wellbeing, psychological flexibility, and associated processes. These improvements peaked during the participation in READY phase and continued to accrue at a slower rate three months later. Psychological flexibility mediated the improvements in resilience, positive affect, and wellbeing. Qualitative data confirmed the personal, professional, and multiple sclerosis (MS) psychologist community level positive training impacts. CONCLUSIONS: The training fostered positive professional and personal development in trainees and consolidated the integration of READY into a frontline service for PwMS. To date, more than 50 READY groups for PwMS have been conducted in Italy.Implications for rehabilitationTraining psychologists in delivering an acceptance and commitment therapy (ACT)-based resilience intervention for people with multiple sclerosis (MS) is associated with positive personal and professional impacts for the trainees.The training program strengthened the sense of community among members of the professional network who attended as trainees.In ACT training, psychological flexibility plays a key role in improving resilience, positive affect, and wellbeing in trainees, and is therefore an important intervention target.ACT training for practitioners fosters the integration of ACT-based interventions into frontline services.

Conversion to secondary progressive multiple sclerosis: Multistakeholder experiences and needs in Italy.

BACKGROUND: Conversion to secondary progressive multiple sclerosis (SPMS) is associated with a relatively poor prognosis, and SPMS is responsible for the majority of the social and economic costs associated with MS. Managing the Transition to SPMS (ManTra) is a mixed methods project conducted in Italy and Germany aimed to set up a user-led resource to empower and improve the quality of life of newly diagnosed SPMS patients. AIMS: To assess the experiences and the needs of Italian people who recently converted to SPMS, patient significant others (SOs), neurologists and other health professionals (HPs). METHODS: We conducted 15 personal semistructured interviews (PSIs) with SPMS patients who transitioned up to five years, and three focus group meetings (FGMs), one of SPMS SOs, one of neurologists, and one of other HPs. Participants were purposely selected from the three geographic areas of Italy, and varied in terms of gender, education and (for patients) disease severity. PSIs and FGMs were audiorecorded, transcribed and analyzed by two researchers using the framework analysis. RESULTS: One hundred sub-categories were identified, grouped into 13 categories and four themes: 'awareness of the transition', 'communication of the transition', 'dealing with symptoms worsening', and 'needs'. The major unmet needs were collected in four dimensions 'organization and management; 'empowerment training'; 'information'; and 'policies'. CONCLUSIONS: Two are the main findings: first, the widespread lack of awareness around the transition; second, the need to improve the quality of the care pathway in the Italian context. It was particularly stressed the need for a holistic and multidisciplinary approach (with patients and SOs as members of the team), the development of an ad hoc plan of follow up visits with easy access to MS specialists' consultation/treatment; specialized training for each stakeholders; more information on SPMS, daily management and changes at policy level.

Ascorbic acid in Charcot-Marie-Tooth disease type 1A (CMT-TRIAAL and CMT-TRAUK): a double-blind randomised trial.

BACKGROUND: Ascorbic acid reduced the severity of neuropathy in transgenic mice overexpressing peripheral myelin protein 22 (PMP22), a model of Charcot-Marie-Tooth disease type 1A (CMT1A) associated with the PMP22 duplication. However, in three 1-year trials, ascorbic acid had no benefit in human beings. We did a multicentre 2-year trial to test the efficacy and tolerability of ascorbic acid in patients with CMT1A. METHODS: Adult patients (aged 18-70 years) with symptomatic CMT1A were enrolled from nine centres in Italy and the UK, and were randomly assigned (1:1 ratio) to receive 1·5 g/day oral ascorbic acid or matching placebo for 24 months. The randomisation sequence was computer generated by block randomisation, stratified by centre and disease severity, and patients were allocated to treatment by telephone. The primary outcome was change in the CMT neuropathy score (CMTNS) at 24 months. Secondary outcomes were timed 10 m walk test, nine-hole peg test, overall neuropathy limitations scale, distal maximal voluntary isometric contraction, visual analogue scales for pain and fatigue, 36-item short-form questionnaire, and electrophysiological measurements. Patients, treating physicians, and physicians assessing outcome measures were masked to treatment allocation. Analysis of the primary outcome was done on all randomised patients who received at least one dose of study drug. This study is registered, numbers ISRCTN61074476 (CMT-TRAUK) and EudraCT 2006-000032-27 (CMT-TRIAAL). FINDINGS: We enrolled and randomly assigned 277 patients, of whom six (four assigned to receive ascorbic acid) withdrew consent before receiving treatment; 138 receiving ascorbic acid and 133 receiving placebo were eligible for analysis. Treatment was well tolerated: 241 of 271 patients (89% in each group) completed the study; 20 patients (nine receiving ascorbic acid) dropped out because of adverse events. Mean CMTNS at baseline with missing data imputed was 14·7 (SD 4·8) in the ascorbic acid group and 13·9 (4·2) in the placebo group. Mean worsening of CMTNS was 0·2 (SD 2·8, 95% CI -0·3 to 0·7) in the ascorbic acid group and 0·2 (2·7, -0·2 to 0·7) in the placebo group (mean difference 0·0, 95% CI -0·6 to 0·7; p=0·93). We recorded no differences between the groups for the secondary outcomes at 24 months. 21 serious adverse events occurred in 20 patients, eight in the ascorbic acid group and 13 in the placebo group. INTERPRETATION: Ascorbic acid supplementation had no significant effect on neuropathy compared with placebo after 2 years, suggesting that no evidence is available to support treatment with ascorbic acid in adults with CMT1A. FUNDING: Telethon-UILDM and AIFA (Italian Medicines Agency) for CMT-TRIAAL, and Muscular Dystrophy Campaign for CMT-TRAUK.