Abstract
Objective
To evaluate the effect of a new care organization on multiple outcomes of transition success and its cost-effectiveness in patients with any endocrine or metabolic disease diagnosed during childhood and transferred to adult care.
Design
Non-randomized controlled trial in a French university hospital.
Methods
Patients transferred to adult care during the control period (04/2014–08/2016) and the intervention period (09/2016–06/2018) were included. The intervention is based on case management involving liaising with pediatric services, personalizing care pathways, and liaising with structures outside hospital (general practitioner, educational and social sector). The primary endpoint was the percentage of patients lost to follow-up at 24 months post transfer. Other outcomes were collected from medical files, consultation software, and questionnaires. A cost analysis was performed.
Results
Two hundred two patients were included (101 per period), the most represented pathologies were congenital and non-congenital hypopituitarism (respectively n = 34 (17%) and n = 45 (22%)) and thyroid diseases (n = 21, 10%). Patients were aged 22.5 in median at 24 months post transfer where 12 were lost to follow-up in the control group vs 9 with the intervention (P = 0.49). The percentage of honored consultation among those planned during 24 months was higher with intervention (P = 0.0065). Patient satisfaction, physician trust, and transfer delay did not differ between the groups. The incremental cost-effectiveness ratio was €179 per patient not lost to follow-up.
Conclusions
At 24 months post transfer, the rate of lost to follow-up did not differ significantly, but indicators of a steadier follow-up were increased and the intervention appeared to be cost-effective.
Introduction
Transition is defined by the Society for Adolescent Medicine as a ‘voluntary, planned process for adolescents and young adults with chronic illnesses, moving from a child-centered to an adult-centered health care system’ (1). In the literature, patients and families report low satisfaction of care and increased psychological distress during the transition and this period is associated with failure in care attendance that have various consequences on the health of young people (2). Adapted transition care are therefore recommended to ensure appropriate treatment and support for young patients during this important time (3).
Numerous programs that attempted to improve the transition of young people to adult medicine have been developed in recent years, particularly in pediatrics care. With the aim of improving the quality of transition in adult care, a new program of transition based on case management named ‘TransEND’ has been developed for young people with various endocrine and metabolic diseases in a French adult hospital (4). These patients are particularly in need of support to increase medical knowledge, practical skills and communication, and to reduce the risk of drop out (5). TransEND was developed based on patients’ needs and on what exists in the literature; however a little evidence-based data exist on the real effectiveness of transition intervention and still less on effectiveness of case management (6).
In a context where healthcare systems face mounting economic constraints, it is also important to assess whether those types of programs are cost-effective – that is, whether the added benefits that may be derived from them compared to usual care are worth the additional costs or, ideally, are associated with cost savings – so as to help in the decision-making process of whether to generalize them.
Our objective was to evaluate the effect of a new care organization based on case management compared to usual adult care on indicators of transition success (i.e. care attendance, physician trust, transfer delay, transition satisfaction, health-related quality of life) along with its cost-effectiveness in patients with any endocrine or metabolic disease diagnosed during childhood and transferred to adult care.
Subjects and methods
Study design and ethics
A non-randomized experimental study (NCT04277286) was conducted in a tertiary hospital (Centre Hospitalier Universitaire (CHU) de la Pitié Salpétrière) located in Paris, France. The study protocol was approved by the institutional review board Comité de Protection des Personnes Ile de France X (No ID RCB 2019-A02243-54). This study was conducted according to the guidelines laid down in the Declaration of Helsinki. Written information and consent were ensured for each participant before any research-related activities. The study is reported of following the Transparent Reporting of Evaluations with Nonrandomized Designs (TREND) guidelines (7) (Supplementary material A, see section on supplementary materials given at the end of this article).
Participants
All the patients addressed by hospital-based pediatricians in transition to adult care, aged less than 25 at transfer, and referred to care department of nutrition or endocrinology (metabolism, reproduction, thyroid, and endocrine tumors) were included in the evaluation. The patients were recruited consecutively starting from the implementation of TransEND (September 2016) and until reaching the number of subjects required in each group: the control period ranged from April 2014 to August 2016 and the intervention period from September 2016 to June 2018.
The control group consisted of patients ‘not exposed’ to TransEND. At the time of their arrival from the pediatric services in adult care, these patients did not benefit from any adaptation in care compared to an adult patient. Before TransEND implementation, patients were ‘addressed’ by their hospital pediatrician with or without a liaising letter, with or without a specific deadline for making the first appointment, and with or without a transmission of their pediatric file. No follow-up was carried out to accompany the making of the first appointment or that of follow-up care.
Intervention
In the group of patients ‘exposed’ to TransEND, that is, those who transferred from pediatrics after August 2016, patients benefited from coordinated care described in a precedent article (4). It was led by a case manager identified as key contact and based on three steps which involved (1) liaising with pediatric services and patient to facilitate his or her first visit in adult care (transfer of the file, calling the patient, and file presentation in a transition staff); (2) defining the care pathway in adult services in relation with the case manager and new doctors, based on the (repeated) personalized needs assessments of each patient, and involving the different available resources and professionals (medical, paramedical, and social); (3) liaising with structures outside the hospital (general practitioner (GP), educational and social sector) to improve the long-term follow-up of patients and to enhance large-scale coordination of the project of life (Supplementary material B).
Outcome measures
Evaluation indicators were selected based on an international consensus (8).
The primary endpoint was the percentage of patients lost to follow-up at 24 months post transfer; it included three components and was defined as (1) no medical consultation in our care department between 12 and 24 months post transfer, (2) no evidence of follow-up in another care department at 24 months post transfer, and (3) no honored consultation after 24 months post transfer in our care department.
The secondary endpoints include engagement in care (the number of honored consultation/total planned consultation) between the time of the transfer and 24 months post transfer, the transfer period defined as the number of months between the first medical consultation in adult services and the last medical consultation in pediatric services, and subjective outcomes (physician trust score assessed by the Wake Forest questionnaire express from 0 to 20 (9), transition satisfaction (on a visual scale from 0 to 10), subjective outcomes, health-related quality of life measured by the 12-Item Short-Form Health Survey (SF-12) and its two summary scores, the Physical (PCS-12) and the Mental Component Summary (MCS-12)). The percentage of patients lost to follow-up at 36 months post transfer was described for exploratory purposes.
Data collection
The objective data were extracted from patients’ medical files and the consultation software of the hospital (data completed in routine care) until December 2020. The subjective data were collected using a two-page paper anonymous self-questionnaire questionnaire sent by post at 36 months ± 6 months post transfer to the patient’s home between May 2019 and December 2020. It included Wake Forest questionnaire, SF-12, care consumption, and ad hoc questions (Supplementary material C).
Economic evaluation
Direct medical costs used in this evaluation included consultations (derived from the hospital software), unplanned hospital admissions and emergency department visits (derived from patients’ medical files and the self-questionnaire), and at-home emergency consultations with a GP (derived from the self-questionnaire), along with the cost of the TransEND program for the exposed group. The latter was derived from the annual salary of the case manager as well as fixed one-time costs spent on developing the program when it was first implemented (e.g. equipment, personnel time, etc.). As there was no data available on how much time the case manager spent with each specific patient, the intervention cost per patient was obtained by dividing total costs by the number of patients followed by the case manager on an average in a given year. Direct medical costs were calculated from an all-payer perspective (statutory health insurance, complementary health insurances, and patients) with a time horizon of 24 months. No discount rate was applied as the questionnaire only asked about the number and type of care consumption, not when it had taken place.
An economic evaluation was performed to put into perspective those costs with the positive outcomes for patients (quality of life and better follow-up).
A cost–utility analysis was carried out. Utility scores were derived from a generic quality of life questionnaire, the SF-12 using the Medical Outcome Study Short Form 6 Dimensions (SF-6D) and Brazier’s algorithm (10, 11, 12) and converted into quality-adjusted life years (QALY). QALYs are a measure that adjust life years spent with a disease on the quality of the patient’s life during that time, with 1 QALY representing 1 year spent in perfect health. Patients who are not in perfect health have QALY values lower than 1 (which can be negative when their health state is deemed worse than death). As only one measure was available in both groups, we assumed that patients remained in the same health state over time.
In addition, a cost-effectiveness analysis was also conducted, the effectiveness criterion being patients lost to follow-up.
Incremental cost-effectiveness ratios (ICER) were then calculated to assess the extra cost that must be spent to gain an additional unit of effectiveness with TransEND compared to usual care. It is done by dividing the difference in mean costs between the two groups by the difference in mean effectiveness (in QALY and in patients lost to follow-up).
Uncertainty was assessed with a probabilistic sensitivity analysis using bootstrapping methods, which created 1000 random resamples of patients in both groups and allowed for a re-calculation of the ICERs in each new sample. The ICERs resulting from the bootstrap were represented graphically on a cost-effectiveness plane to compare four possible situations: (1) TransEND is more effective and less costly than usual care and is therefore adopted; (2) TransEND is less effective and more costly and is therefore rejected; (3) TransEND is more effective and more costly and it is up to decision-makers to decide whether they are willing to pay the extra cost; and (4) TransEND is less effective but also less costly, and decision-makers must then decide whether they are willing to part with some effectiveness to save costs. Those results were used to build cost-effectiveness acceptability curves showing the probability that TransEND would be cost-effective for different willingness-to-pay by the decision-maker.
Sample size calculation
Based on an observed percentage of 18% of patients lost to follow-up in patients with congenital adrenal hyperplasia transitioning out of the TransEND organisation in our center (13) and an expected percentage of 5% of patients lost to follow-up in patients of the TransEND group, a total of 188 subjects – or 94 per group – was required to demonstrate a significant difference on the primary endpoint (alpha 5%, power 80%, two-tailed test, calculated with epiR package 0.9-96 method). To mitigate the risk of patient opposition to research, 202 patients were invited to participate.
Statistical methods
The quantitative variables were described by their median (first quartile (Q1) − third quartile (Q3)), minimum, and maximum. Qualitative variables were described by counts and percentages. Intergroups comparison was assessed using Student’s t-test for continuous and chi-squared tests for categorical variables. Data on care attendance were censored at 24 months post transfer. According to the intention-to-treat principle, data were analyzed for all included participants regardless of compliance to TransEND organisation. No participant was excluded from the analysis. All statistical tests were tested bilaterally, with a significance level set at 5% using SAS software © 2002–2012 by SAS Institute Inc. (Cary, NC, USA), version 9.4.
Results
Between May 2019 and December 2020, 202 patients were invited to participate in the study; no patient objected to the use of their data for research, but 100 did not complete the study questionnaire for secondary outcomes assessments (see flow chart, Fig. 1). Baseline comparison between participants who completed the questionnaire and those who did not showed no difference on sex, disease, transition delay, age at last pediatric consultation, number of consultations during the 24 months post transfer, or engagement rate (data not shown). Characteristics of patients are described in Table 1.
Flowchart of the study.
Citation: European Journal of Endocrinology 186, 3; 10.1530/EJE-21-1127
Flowchart of the study.
Citation: European Journal of Endocrinology 186, 3; 10.1530/EJE-21-1127
Flowchart of the study.
Citation: European Journal of Endocrinology 186, 3; 10.1530/EJE-21-1127
Baseline characteristics of the participants by group. Data are presented as n (%) or as median (Q1, Q3).
| Total | Control | TransEND | |
|---|---|---|---|
| n | 202 | 101 | 101 |
| Sex | |||
| Female | 104 (51%) | 58 (57%) | 46 (46%) |
| Male | 98 (49%) | 43 (43%) | 55 (54%) |
| Diseases | |||
| Non-congenital hypopituitarism | 45 (22.3%) | 22 (21.8%) | 23 (22.7%) |
| Congenital hypopituitarism | 34 (16.8%) | 17 (16.8%) | 17 (16.8%) |
| Thyroid diseases | 21 (10.4%) | 13 (12.9%) | 8 (7.9%) |
| Disorders of pubertal development | 18 (8.9%) | 4 (4%) | 14 (14%) |
| Congenital adrenal hyperplasia | 15 (7.4%) | 7 (6.9%) | 8 (7.9%) |
| Turner syndrome | 13 (6.4%) | 7 (6.9%) | 6 (5.9%) |
| Obesity | 11 (5.5%) | 6 (5.9%) | 5 (5%) |
| GH deficiency | 2 (1%) | 2 (2%) | 0 (0%) |
| Other conditions† | 43 (21.3%) | 23 (22.8%) | 20 (19.8%) |
| Age at last pediatric consultation | 18.8 (18.0, 19.9) | 18.6 (18.0, 20)* | 18.9 (18.1, 19.9) |
| Age at first adult medical consultation | 19.5 (18.6, 20.7) | 19.5 (18.5, 20.9) | 19.4 (18.6, 20.5) |
*Three missing data; †rare diseases, pituitary benign tumor, patients at risk following disease or treatment.
Primary criterion
At 24 months post transfer, 21 patients were considered as lost to follow-up (Fig. 2), there were 12 in the control group and 9 in the TransEND group (P = 0.49). The only component of lost to follow-up definition which statistically differs between groups was the absence of consultation between 12 and 24 months post transfer (41% in control group vs 28% in TransEND group; P = 0.04). In patients not considered as lost to follow-up because they received care in another center , 19 (9.5%) reported a follow-up in another hospital, 8 (4%) in a specialist private practice, and 5 (2.5%) by a GP. Those followed up by a GP had congenital hypopituitarism (n = 2), Turner syndrome (n = 1), hypothalamic lipoma (n = 1), and acquired hypogonadism (n = 1).
Description of the three components of lost to follow-up primary criterion.
Citation: European Journal of Endocrinology 186, 3; 10.1530/EJE-21-1127
Description of the three components of lost to follow-up primary criterion.
Citation: European Journal of Endocrinology 186, 3; 10.1530/EJE-21-1127
Description of the three components of lost to follow-up primary criterion.
Citation: European Journal of Endocrinology 186, 3; 10.1530/EJE-21-1127
Patients lost to follow-up were mostly male (n = 13, 62%), aged 18.3 (17.1, 19.3) at the last pediatric consultation, and had a transfer delay between pediatric and adult care of 6.5 months (2.5, 13.6). Of these five (24%) had congenital hypopituitarism, four (19%) had non-congenital hypopituitarism, and three (14%) had disorders of pubertal development.
Secondary criteria
The results of secondary outcomes are presented in Table 2. Engagement in care between transfer and 24 months was significantly higher in TransEND group (fewer non-honored consultations); in patients who completed the questionnaire, perceived attentiveness by adult care professionals to their parents was significantly higher in TransEND group.
Secondary outcome assessments. Data are presented as median (Q1, Q3) or as n (%).
| Control | TransEND | P | Missing data | |
|---|---|---|---|---|
| n | 101 | 101 | ||
| Care attendance | ||||
| Engagement in care (in %) | 70 (50, 89) | 80 (67, 100) | 0.01 | 0/0 |
| Transition delay (in months) | 6.3 (4.4, 11.5) | 6.0 (4.4, 9.1) | 0.15 | 3/0 |
| Subjective outcomes | ||||
| Transition satisfaction | 8 (8, 10) | 9 (9, 10) | 0.41 | 88/73 |
| Physician trust (Wake Forest score) | 11 (9, 13) | 10 (8, 12) | 0.06 | 47/61 |
| Mental component score (SF-12) | 47 (40, 52) | 49.5 (40, 53) | 0.39 | 51/67 |
| Physical component score (SF-12) | 51 (44, 56) | 52 (44, 56) | 0.81 | 51/67 |
| Feeling of rupture during transition (yes) | 11 (20%) | 7 (20%) | 0.97 | 47/66 |
| Perceived continuity of care during transition (yes) | 43 (77%) | 31 (86%) | 0.27 | 45/65 |
| Need to contact the pediatrician after transfer (yes) | 13 (24%) | 3 (9%) | 0.08 | 47/68 |
| Perceived attentiveness by adult care professionals to the patient (yes) | 53 (90%) | 37 (97%) | 0.16 | 50/63 |
| Perceived attentiveness by adult care professionals to the parents (yes) | 42 (82%) | 37 (97%) | 0.03 | 50/63 |
Bold text indicates statitically significant (P<0.05) difference.
Care consumption and costs
The total number of consultations in the center was 426 in the TransEND group vs 409 in the control group (median per patient = 4 in both groups). There was no unplanned admission or emergency consultations in the control group, while in the TransEND group, two patients were admitted, four had emergency department (ED) visits, and one had a General Practitioner (GP) emergency visit at home.
The salary of the case manager was €42 000 annually and a one-shot budget of €203 000 – assumed to be amortized over 10 years – was invested to develop TransEND. Based on an average of 146 new patients followed each year and a mean follow-up of a little under 2 years (4), the intervention cost of TransEND was €427 for 2 years (€213.5 annually). However, at the moment patients do not ‘leave’ TransEND after 2 years, although some will need it less and less as time goes by. Assuming a longer follow-up (5 years), the cost of TransEND was €256 (€128 annually).
Economic evaluation
Mean utilities at 2 years were 1.439 (s.d. = 0.198) in the control group vs 1.487 (s.d. = 0.230) in the TransEND group, while mean total costs were €114 (s.d. = 58) vs €652 (s.d. = 801), leading to an ICER of €11 208 per QALY gained and €179 per patient not lost to follow-up. Under the assumption of a 5-year follow-up, the ICER was €7646 per QALY and €122 per additional patient not lost to follow-up.
The sensitivity analysis found little uncertainty surrounding the results: TransEND was more effective and more costly than usual care in 82.2 and 64.4% of cases in the cost–utility (Fig. 3A) and cost-effectiveness analyses (Fig. 3B), respectively. There was a 78% probability that TransEND would be cost-effective if the decision-maker was willing to pay €50 000 per QALY (Supplementary material D).
Sensitivity analysis of the economic evaluation at 2 years using bootstrap methods. (A) Cost–utility analysis; (B) cost-effectiveness analysis. X axis: Effectiveness difference between TransEND and usual care, in quality-adjusted life years (A) or patients not lost to follow-up (B) Y axis: Cost difference between TransEND and usual care, in euros. A full color version of this figure is available at https://doi.org/10.1530/EJE-21-1127.
Citation: European Journal of Endocrinology 186, 3; 10.1530/EJE-21-1127
Sensitivity analysis of the economic evaluation at 2 years using bootstrap methods. (A) Cost–utility analysis; (B) cost-effectiveness analysis. X axis: Effectiveness difference between TransEND and usual care, in quality-adjusted life years (A) or patients not lost to follow-up (B) Y axis: Cost difference between TransEND and usual care, in euros. A full color version of this figure is available at https://doi.org/10.1530/EJE-21-1127.
Citation: European Journal of Endocrinology 186, 3; 10.1530/EJE-21-1127
Sensitivity analysis of the economic evaluation at 2 years using bootstrap methods. (A) Cost–utility analysis; (B) cost-effectiveness analysis. X axis: Effectiveness difference between TransEND and usual care, in quality-adjusted life years (A) or patients not lost to follow-up (B) Y axis: Cost difference between TransEND and usual care, in euros. A full color version of this figure is available at https://doi.org/10.1530/EJE-21-1127.
Citation: European Journal of Endocrinology 186, 3; 10.1530/EJE-21-1127
Follow-up after 24 months
At the time of data analysis, patients in the control group had been in adult care for a median duration of 60.6 months (Q1, Q3 = 56.6, 65.9) and patients in the TransEND group for 45.6 months (Q1, Q3 = 41.5, 48.2). For exploratory purposes, we analyzed the lost to follow-up between 24 and 36 months post transfer. According to the same definition as the primary criterion, percentage of patients lost to follow-up did not significantly differ between control and TransEND groups (respectively 21 (21%) and 26 (26%), P = 0.41). They differed neither on component 1 (no medical consultation in our center between 24 and 36 months post transfer P = 0.7) nor on component 2 (no evidence of follow-up in another center at 24 months post transfer P = 0.7) but significantly more patients in the TransEND group had no honored consultation after 36 months post transfer in our center (P = 0.01) which is directly correlated with the patient follow-up time which is less than the control group.
Discussion
The evaluation of the TransEND program is one of the largest and most diverse of the comparative studies existing on transition. We did not demonstrate statistical significant difference in the percentage of patients lost to follow-up at 24 months post transfer between TransEND and control group; however, we observed an absolute difference of three patients lost to follow-up between the two groups, which is clinically relevant. A lack of statistical power may explain the lack of significance; indeed, our sample size calculation was based on an assumption of 18% of patients lost to follow-up in the control group and 5% in the TransEND group; the difference observed was smaller and therefore required a much larger number of patients to ensure the statistical demonstration of this difference. However, we showed that in the TransEND group, more patients had at least one consultation between 12 and 24 months post transfer and the engagement rate in medical consultation was higher which is an interesting indicator of precoce integration in adult care. We also found that while costs were higher in the TransEND group, the incremental cost-effectiveness ratio was €11 208 per QALY gained, with good robustness in the sensitivity analysis, making it highly probable that TransEND was cost-effective and therefore a good use of ressources.
For reasons of reduction of the biases inherent in the before/after comparative studies (ref), we chose to compare two very close periods around TransEND launch. There was no substantial change in the organization of care, the composition of the care team, and the training of caregivers during the study period meaning that the differences observed between the two groups are only related to the implementation of TransEND. However, the observed lower number of lost-to-follow-up patients than expected in the control group can be explained by the fact that the team was more and more aware about the transition issue. The coming TransEND launch involved service meetings on this subject and file preparation to argue TransEND utility to hospital heads and funders several months before the recruitment of a case manager, which may partly explain a smaller difference than expected. Moreover, before the launch of TransEND and at the arrival of the case manager, the patients leaving pediatrics were not known to the adult service before their first consultation, so a certain number of patients who dropped out from just after pediatrics could have not been taken into account in the control group.
In international literature, few completed comparative studies exist but those that published transition programs evaluation on the same population also show moderate results: a young person’s clinic for patients with congenital adrenal hyperplasia showed no difference in engagement with adult services in those who transitioned through the program or not. However, this adult care program did not make the link with pediatrics and many patients were lost to follow-up between pediatrics and adult medicine (14), which reinforces the relevance of this function for the case manager in TransEND. A multi- center randomized controlled trial of transition support program for patients with type 1 diabetes showed no sustained benefits (clinic attendance, satisfaction with care, and diabetes-related distress) after 12 months of the intervention completion (15). This raises the question about the relevance of the outcomes that we use today to evaluate these programs. A diversification of the outcomes, involving more patient-centered outcomes, must be carried out to better study the positive effects of transition (16).
Strengths and limitations
We carried out a large comparative study; however, this could not be randomized for reasons of acceptability. However, selection bias was avoided as all patients meeting the criteria were included and compared periods were really closed. Regarding measurement bias, the primary endpoint was based on a trio of indicators collected in usual care independently of research, prospectively and which is objective. Although the profiles of the respondents and non-respondents to the questionnaires (secondary endpoints) were similar, it cannot be ruled out that bias exist, that should moderate their interpretation.
The design was adapted to complex interventions and respected the recommendations of the evaluation of these by measuring the ‘triple aim’: indicators of importance for the patient, for the care system, and costs (17). The measurement of the latter is rare in the evaluation of transitional interventions (6) which is nevertheless important for evaluating the sustainability of programs after research. There are of course limits associated with the economic evaluation, due to the small number of patients who answered the questionnaire, which likely led to the unexpected finding of more unplanned admissions and emergency visits in the TransEND group. In addition, some costs could not be taken into account, such as consultations in an ambulatory care setting outside our hospital. As more patients in the control group were followed elsewhere, it is therefore likely that the cost difference is smaller, increasing the probability that TransEND would be cost-effective. Finally, another limit is that treatment compliance and clinical morbidity indicators were not studied because of the diversity of conditions in the included population and that quality of life could only be assessed once due to the design of the study and so its evolution in both groups over time was not incorporated in the QALY calculation. This likely led to a conservative estimation of the ICER, as one could assume more improvement of quality of life with TransEND.
Conclusion
Case management involving liaising with pediatric services, personalizing care pathways, liaising with structures outside hospital is suitable for the management of patients with endocrine or metabolic diseases in transition. At 24 months post transfer, the rate of lost to follow-up does not differ significantly but indicators of a steadier follow-up are increased. In addition, such intervention is likely to be cost-effective. The evaluation continuation is justified to assess the purpose of TransEND to improve the long-term follow-up of patients.
Supplementary materials
This is linked to the online version of the paper at https://doi.org/10.1530/EJE-21-1127.
Declaration of interest
The authors declare that there is no conflict of interest that could be perceived as prejudicing the impartiality of the research reported.
Funding
The transition program (dedicated room and case-manager funding) was supported by the ‘Hôpitaux de Paris – Hôpitaux de France’ Foundation (‘Transition program’ Grant, 2015). The realisation and analysis of the cohort did not receive any specific grant from any funding agency in the public, commercial or not-for-profit sector.
Author contribution statement
E L and P T wrote the protocol and designed the evaluation, P T, F M, S M, S B, M P developed and conducted the TRANSEND Program, E L, P T, I T, F M, S M, H M developed the evaluation tools, I T ensured the data collection, A T managed and analysed the data, M M designed and analysed the cost evaluation, E L, A T, M M wrote the first version of the article, E L, A T, M M, I T, F M, S M, S B, H M, M P, P T interpreted the data and revised the protocol. All the co-authors approved the final version of the article, and all agree to be accountable for all aspects of the work.
Acknowledgements
The authors would like to thank all the nurses, social workers, dieticians, psychologists, and medical secretaries from the departments involved in the TransEND cohort for participating in the elaboration of the program. The TransEND group includes all those doctors who refer their patients to the program and/or follow them in adult care: Prs and Drs. Afenjar, Aigrain, Amoura, Amouyal, Amsellem Jager, An, Andreelli, Azar, Bachelot, Belien Pallet, Beltrand, Bibal, Bidet, Bismuth, Blanc, Bodemer, Bougneres, Bourron, Bouvattier, Brauner, Brioude, Bruckert, Brugieres, Brune, Brunel, Burggraeve, Cabrol, Cacoub, Carel, Carlier, Carreau, Cassuto, Cessans, Chakhtoura, Cheikhelard, Chevignard, Ciangura, Consoli, Couderc, Coupaye, Courtillot, Da Costa, Dabbas, Dalla Vale, Dassa, De Beaufort, De Kerdanet, Delanoe, Delcroix, Diene, Dierick-Gallet, Donadieu, Dormoy, Doummar, Doz, Dubern, Dufour, Duranteau, Esteva, Fayech, Fiot, Flandrin, Flechtner, Fresneau, Gajdos, Gallo, Gaspar, Gelwane, Ghander, Giabicani, Gonzalez, Grill, Guilmin-Crepon, Grouthier, Hartemann, Haye, Heide, Houang, Ibrahim, Idbaih, Istanbullu, Jacqueminet, Jacquin, Jeandidier, Jeannin, Karsenty, Kenigsberg, Kipnis, Le Bastard, Le Poullenec, Leenhardt, Lefevre, Loison, Lorenzini, Lucidarme, Ly, Manh, Martinerie, Masson, Meneret, Mignot, Milcent, Mircher, Mochel, Moretti, Mosbah, Mourre, Moutafoff-Borie, Muller, Nasser, Navarro, Pasqualini, Paulsen, Pauwels, Personnier, Phan, Piquard Mercier, Porquet-Bordes, Puget, Ribeiro Parenti, Ricour, Rigaux, Robert, Robin, Rougeoreille Cretier, Samara Boustani, Sarnacki, Sauvion, Semeraro, Simon, Storey, Thalassinos, Tounian, Trang, Tubiana-Rufi, Villanueva, Whalen, Zaarour, Zenaty.
Within this group, we particularly thank F Menesguen, M Popelier, M Halbron, P Faucher, G Pinto, J Léger, C Poitou. Finally, the authors would also like to thank the University of Sheffield for granting them the license to use their algorithm to convert the SF-12 into utilities.
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