The study was approved by the ethics committee of the « Université de Bretagne Occidentale » Faculty of Medicine, Brest. The participants had to sign a written informed consent to participate in the study. The Family Physicians involved provided a verbal consent.
The recruitment was handled by 31 FPs in western Brittany, France, who agreed to participate in the study after being contacted at their office. All were chosen from the Clinical Teacher list of Brest University.
All patients met the EGPRN definition of multimorbidity. 96 patients were included by 19 FPs between July and December 2014. 12 other FPs enrolled 35 new patients from September to November 2015 to increase the study’s power.
Inclusion criteria were: having a chronic disease, with at least one other disease (acute or chronic) or a biopsychosocial factor (associated or not) or a somatic risk factor.
Biopsychosocial risk, including all psychological risk factors, psychosocial risk factors, lifestyle, demographic data (age, gender), psychological distress, socio-demographic characteristics, aging, beliefs and expectations of patients, physiology and pathophysiology were taken into account.
Each patient was monitored over time and had to sign an informed consent.
Exclusion criteria: patients not meeting the criteria of the multimorbidity definition, patients living in a nursing home, patients who were unable to follow the study over time, patients under legal protection, and patients whose condition was already life-threateningor whose life expectancy was estimated at less than three months.
Each FP had to follow a plan: at the first stage, the multimorbid patient was given full information on the study by the FP and asked to give his/her consent.
Then, the FPs had to fill in a questionnaire to explore the potential decompensation risk factors within themes and subthemes of multimorbidity. (Table 1)
The questionnaire was designed by the research group with reference to the definition of multimorbidity. There were 52 questions, covering every item of the definition and based on a clinical and anamnestic approach by the FP. First, there were 7 questions about the patient’s diseases. Then, there were 8 questions to evaluate the somatic risks factors. Questions 16 to 19 focused on the psychosocial factors, social network and coping strategies (defined as: diet and lifestyle, planning, instrumental support, social support, expression of feelings, positive reinstatement, acceptance, denying, blaming, sense of humor, religion, leisure activities, substance use). From Question 20 to Question 37, the FPs were asked about burden of diseases and health care consumption. There were then 4 questions about prevention vaccination and screening. The last questions pondered the core competencies of the FP and his/her relationship with his/her patient.
The questionnaire is fully available (Appendix 1) as it was given to the FPs (in the French language).
From the first pilot study, some irrelevant variables were deleted for different reasons:
- Chronic condition redundancy with chronic disease or psychological risk factor)
- Cost of care (impossible to estimate given the time and resources dedicated to the study)
- Disability (disability / impairment, qualitiy of life and health outcomes (consequences rather than the characteristics of the multimorbidity)
- frailty (absence of consensual definition, criterion assessed by study, methodologically impossible to assess at the beginning of the study)
- physiology (too broad a notion, impossible to evaluate)
- disease and assessment (present in the theoretical definition but missing from the coding book and not found in the transcripts)
- demography and aging (redundant with sociodemographic characteristics)
Somatic risks were evaluated as cardiovascular risk factor, risk of falling factors (calculated with the CETAF score) (25), an assessment of hygiene, nutrition and physical activity at the discretion of the FP.
This questionnaire was accepted by the scientific committee of the research team and tested with FPs and medical students.
Thanks to the comments made during the first pilot study, a revised questionnaire was used for those patients included from September to November 2015. The question order was actually rearranged to ease and shorten the time needed to complete it. No question could be asked before the previous one had been answered in order to avoid omitting data. The FP was only asked question number 40 if he/she had answered « yes » to organized or individual screening in question number 39 to avoid errors in completion identifed in previous studies.
Data were saved using Microsoft Excel and computed by the online survey software EVA-LANDGO ®.
24 months after inclusion, FPs were contacted by email or phone (INSERER APPENDIX 5) to collect patient status information. Two types of status were defined after a consensus had been reached in a peer group gathering on multimorbidity made up of physicians, residential students and researchers in family practice: « decompensation » (D) and « Nothing to Report » (NTR). Decompensation was understood as the occurrence of hospitalization of at least 7 days duration, or death, during the 24 months of follow-up, as the mean duration for hospitalization in the European Union is 6.7 days (26).
Groups labelled « frail » or « not frail » in the feasibility study were changed at the six-month follow-up as confusion between « frail » and « frailty » might occur and this would include a definition which was not consensual (27).
Data cleaning was performed to harmonize data for analysis. The ICD 10 was used to standardize the mentioned chronic diseases. 102 chronic diseases were reported.
Missing data were spotted during descriptive statistics analysis. They were replaced by the median value to be incorporated into the statistical analysis.
Each modification and the reasons for it were compiled in the « dictionary » available on demand from the corresponding author.
A description of the population was the first step. Both types of status « decompensation » and « nothing to Report » were compared using a bi-dimensional analysis for each variable.
Quantitative variables were compared using a Fischer’s exact test or a chi–2 test with an alpha level set at 5 %. Qualitative variables were compared using a Student’s test when it followed a normal distribution to compare the means of the two groups from a normal population, and a Shapiro-Wilk’s test when it did not follow a normal distribution, to compare the medians of the two groups.
Patients with the same characteristics. regardless of status « Decompensation » or « Nothing to Report », were grouped together using a multidimensional analysis. Non-discriminating and non-descriptive variables were removed. Then, a clustering represented in a dendrogram and a multiple correspondence analysis (MCA) were performedto identify discriminating variables for each group and the resulting information was combined using the technique of hierarchical clustering on principal components (HCPC).
The second step comprised a statistical analysis of the follow-up. A logistic regression was used for the six and nine-month follow-up as the dependent variable was binary (D versus NTR), regardless of follow-up time. A Cox model was chosen to complete the analysis from the twelve-month study, making it possible to apply different durations of follow-up time to support each patient.The aim was to find the best subgroup of variables for predicting and explaining the patient’s status at 24 months.
At first, the overall survival of the two populations for each variable were compared using a non-parametric estimator by Kaplan-Meier, with a Wilcoxon test of alpha-risk at 5%. Then, the team estimated the survival function using semi-parametric models. A Hazard Ratio (HR) was first obtained by a univariate analysis using Cox’s Model. Then, an adjusted HR was obtained with a multivariate analysis using Cox’s model, representing the association between a variable and the decompensation risk factor.