Pre-Transition Consultations for Adolescents With Chronic Conditions in a General Pediatric Hospital: Lessons Learned

Hélène Mellerio (  helene.mellerio@insem.fr ) APHP: Assistance Publique Hopitaux de Paris https://orcid.org/0000-0001-6586-8677 Agnès Dumas Institut national de la santé et de la recherche médicale: INSERM Corinne Alberti Institut national de la santé et de la recherche médicale: INSERM Sophie Guilmin-Crépon APHP: Assistance Publique Hopitaux de Paris Margaux Gastaldi APHP: Assistance Publique Hopitaux de Paris Loïc Passini APHP: Assistance Publique Hopitaux de Paris Céline Gabarro Institut national de la santé et de la recherche médicale: INSERM Maxime Morsa Paris 13 University: Universite Sorbonne Paris Nord Enora le Roux Institut national de la santé et de la recherche médicale: INSERM Paul Jacquin APHP: Assistance Publique Hopitaux de Paris


Introduction
Since the rst de nition of transition from pediatric to adult healthcare in adolescents with chronic condition (CC) [1], various guidelines have been published [2][3][4][5], based on growing evidence of the relevance of a structured and personalized healthcare transition process [6][7][8][9][10]. Health self-e cacy is associated with better long-term wellbeing, participation, satisfaction with services and condition-speci c measures [11]. Key part of the learning process, aspects related to psychosocial development, including how to approach the self-concept, social life and the future with the illness, should also be considered [12]. However, the most effective ways to evaluate and promote adolescents' con dence in managing their CC remain unclear. Despite their relevance [2], the readiness questionnaires focus on skills and knowledge without exploring in depth the adolescent's global approach to adulthood with a CC. Indeed, transition of care is one of many changes from childhood to adulthood [13], that presupposes the need to adopt a developmentally appropriate approach to healthcare [11].
Developing personalized and structured transition support nevertheless remains a challenge in many clinical units, because of time or funding restrictions or limited interactions with adult healthcare providers (HCPs) [9,14,15].
To pool resources and provide more opportunities for care units without any transition program, the dedicated transition unit AD'venir was created in 2017 at the Robert Debré pediatric hospital (Paris, France). AD'venir aims to deliver patient-centered care to all adolescents with a CC, to enhance their quality-of-life through sustained high-quality healthcare and ongoing social participation in adulthood. To this end, we implemented individual pre-transition consultations (PTCs) to assess adolescents' readiness and tailor their preparation to transition.
Here, we report the clinically relevant features of the PTC.

Materials And Methods
This research was mainly based on qualitative methods, and included quantitative methods.

Population
PTCs were proposed by the referring HCPs (RHCPs) to adolescents (15-25 years), with any CC or disability managed at the R.Debré hospital, due to be transferred to adult healthcare within the following 2 years. Adolescents were informed about the research through an information letter. We recruited the rst adolescents attending a PTC from May 2017 (launch of AD'venir), including participants until data saturation achievement, meaning that patients' discourses had become redundant and no new theme would arise with supplemental inclusions [16]. We also recruited each adolescent's RHCP.

Content of the PTC
The PTC is a special medical appointment during transition preparation (Table 1). During almost 1 hour, a transition pediatrician (TP) receives each adolescent alone to explore chronologically their perceived experience and view of their CC: past, present, future. It includes: an interview structured according to the HEADSS checklist [17]; the Treatment Burden Questionnaire (TBQ, 15 items)[18] and the Good2Go questionnaire (25 items) [19]; and a physical examination. At the end, the TP and the adolescent each highlight the points requiring further transition support. This debrie ng serves as a basis for the PTC report.

Data analysis
The qualitative approach was developed to gain insight into the conduct of the PTC, because they are relevant methods to understand how health services work and to provide detailed understandings of intervention functioning on a small scale [21]. PTCs were audio-recorded, transcribed by a professional, and contextualized by a form stating the schedule and the adolescent's psychological condition. A twostep qualitative analysis was performed. As a rst step, two parallel analyses consisting in an inductive identi cation of themes [22] were performed by two AD'venir TPs (HM, PJ) and two sociologists (AD, CG), respectively. Emerging themes and the analytical framework were discussed together to present a report to a dedicated multidisciplinary working group, including the two sociologists, the four AD'venir TPs, one researcher in public health (ELR) and one psychologist (MM). In a second step, the working group held regular meetings to discuss different views on the analysis of transcriptions and reach a consensus on the key results to be developed in the present paper. Two years after the PTC, an independent male HCP (LP), trained in adolescent medicine, conducted semi-structured phone interviews to explore adolescents' perceived support and transition progress. Data are reported according to the COREQ checklist [23].
The quantitative methods consisted in: 1-an analysis of responses given to the TBQ and the Good2Go (continuous variables expressed as median (quartiles) or mean (standard deviation, SD) as appropriate; categorical variables as frequencies (percentages)); 2-an anonymous questionnaire sent to RHCP 6 months after the PTC, to assess the perceived bene ts of the PTC (Google-forms®, 8 items).

Ethics
The R.Debré Ethics Committee approved the protocol (No.2016/268), subject to written approval being obtained from the heads of all hospital units. All data were anonymized.

Results
Twenty-seven adolescents were included ( ; "When I received the email, it was written 'parents if possible but not mandatory', I didn't even think of asking my mother." Clinical features of the PTC Past Adolescents were rst asked to describe their CC, from the start and up to the present. Most could say the exact name, but some had great di culty in naming their CC and mentioned their treatment or medical device (central catheter, insulin, etc.), or the symptom that revealed the CC (e.g. "I did not eat", "I kept vomiting"). Six adolescents, four with cystic brosis (CF) used periphrases such as "the same disease as Gregory Lemarchal" (a famous French singer who died from CF aged 24). One 18-year-old girl never said 'cystic brosis' for fear of being stigmatized: "I don't want people to say 'Oh, she's going to die soon…'".
The terms "digestive ostomy" and "incontinence" were also avoided. Sometimes, di culties re ected a lack of knowledge or scienti c terminology, especially in cases of cognitive impairment: "When I say 'nephrotic syndrome' I don't see what that's got to do with the kidneys." (18-year-old girl, early-childhoodonset nephropathy).
Two boys did not know the names of their CC, respectively laparoschisis and VACTERL syndrome, despite a history of multiple surgical interventions and, in one case, incontinence. The absence of an explicit link between the CC and the tests could also be confusing, such as a hand X-ray for growth retardation exploration in a girl with chronic diarrhea syndrome.

Present
Concerning TBQ, laboratory tests, diet, doctor appointments and administrative tasks emerged as impacting daily life (Fig. 1, Table 2). If laboratory tests were often synonymous with pain or stress, the loss of time and the constraints of appointments underlined the differences from healthy peers. Other items were scored low, even when treatment was constraining. Only 3 adolescents had a low TBQ global score. No effect of age, age at diagnosis, gender or type of CC was observed on global score (Fig. 2). Treatment observance was most often good, but talking about treatment frequently led to sharing their distress at living with a CC:"I don't like the thought of having to go on being treated, I just feel I want to be left alone" (19-year-old boy, VACTERL syndrome).
All adolescents complained of the limitations experienced in social life, notably diet, tiredness or having to renounce physical activity (Table 3). Intimate concerns were shared increasingly easily as the PTC progressed, sometimes only after 45 minutes. Even some refused the genital examination, the physical examination provided an opportunity to address their often-impaired body image and to go further in talking about intimate concerns (Table 3). Mostly, adolescents were willing to discuss their sexual concerns. However, as for tobacco, drugs and alcohol, Good2Go showed that a minority talked about sexuality with HCP. One 19-year-old boy with a penile malformation multiplied his sexual partners out of a need to be reassured, but he did not discuss this with his surgeon. This taboo made them feel quite ashamed. A boy (23-year-old, urogenital malformation) who was about to get married, declared: "Is it OK if I ask you?[...] I'm a bit paranoid about…not having children, you see, and stuff like that". Many were poorly informed about impact of CC on fertility or course of pregnancy. Parental presence during appointments with their RHCP was cited as a barrier.

Future
Most were aware of the need to transfer. However, transfer was accepted with passivity as a natural process towards maturity, but with sadness at leaving the hospital they were used to and "their" RHCPs who were "allowed to know everything". Some were afraid of being forgotten. Some even refused to think about it, even when they felt ready to transfer: "Maybe I'm ready? But I don't want to." (17-year-old girl, Crohn's disease). None cited the non-physician HCP. Some were not ready and comfortable with being dependent on their parents but con dent they would be able to look after themselves when the time came: "I know that when I have to look after myself, I'll be able to." (16-year-old girl, in ammatory bowel disease).
In contrast, some pointed out the pediatricians' inability to "speak frankly" or to deal with "adult" issues, such as vaginal mycoses in an 18-year-old girl with CF. The adolescents rarely had comprehensive information about transfer (timing, doctor/hospital to be transferred to). Some had been involved in the choice of their future HCP (several girls preferred a female doctor) or in transfer planning ("after my baccalaureate exam"). Representations of adult healthcare were globally poor, with a similarity to pediatric units.
Previous discussion facilitated completion of the Good2Go questionnaire. Scores were globally consistent with the TP's impression of transition readiness, without correlation with age at diagnosis of the CC, knowing the CC's exact name, having information about transfer, educational level or socioeconomic background. Scores of the 3 domains revealed intrapersonal heterogeneity (Fig. 2). Older adolescents had slightly higher scores in domains 1 (health advocacy) and 3 (practical skills), whereas gender did not seem to in uence scores (Fig. 2). Non-scored items identi ed the mother as the main resource person, rather than siblings (one boy mentioned his brother, with CF too) or the father.
RHCPs' perceived bene ts of the PTC Thirteen RHCPs (13/27) completed the questionnaire. All were satis ed with the PTCs and the PTC reports. RHCPs reviewed their practices with regard to talking about transition or the long-term outcomes for adolescents with CC (n = 12), supporting the parents (n = 10) or involving adolescents during appointments (n = 9). RHCPs noted behavioral improvements in adolescents and their parents in terms of looking ahead to transfer (n = 13) or more generally to the future (n = 12), self-management skills (n = 12), or participation during appointments (n = 11).
Adolescents' perceived bene ts of the PTC All but one adolescents accepted the phone interview 2 years after PTC. None was lost of follow-up.
Transfer in adults' care had occurred for 19, of whom 18 were satis ed and trusted their new HCP. One girl (19 years, Crohn's disease) returned in pediatric healthcare; during the PTC, the Good2Go indicated high scores of transition readiness, but transfer in adults' care was mainly perceived as an opportunity to leave the fusional relationship with their mother. All others adolescents (n = 7) were now informed about the transfer planning.
Concerning experience of PTC, some participants had appreciated this opportunity to have information about transition, how will it proceed, and to prepare adequately for the transfer. PTC was also a place to talk about social issues like insurance or pregnancy, and to share about their intimate experience of the CC: "It allows me to have more information about the hospital, what was going to happen; I was maybe less anxious […]. It helped me to have a deeper understanding about my CC; I used to think it was a pulmonary problem, diabetes, and that's all." (18-year-old boy, CF).

Discussion
The PTC relies on a holistic approach to care delivery for adolescents with CC and adopts most of the elements of the transition theoretical framework [24,25], inspired by the Chronic Care Model [26]: youthcentered and strength-based focus, emphasis on self-determination and self-management, acknowledgment of individual differences and complexities, and recognition of vulnerabilities, cultural beliefs and socioeconomic disparities. This model of dedicated consultations, assessing transition readiness and identifying tailored and negotiated actions for transition preparation, irrespective of the adolescent's CC, is a rst in France and, to our knowledge, very innovative internationally. The PTC is implemented by experts in adolescent medicine occupying an independent but complementary position with regard to the RHCP. One experience in neurodevelopmental conditions bears some similarities [27], but our research underlines the invariable points shared by all CC and highlights the relevance of a disease-neutral intervention.
First, the PTC offers adolescents an appreciated opportunity to step back, review and speak freely about their history with CC, how they have arrived at their present point and how they see themselves in the future. This helps them analyze their strengths and barriers to transition success [7]: CC self-management skills, feeling of self-e cacy, trust in adult healthcare and perceived social support. The PTC report gives the RHCP a comprehensive transition planning tool to tailor the transition preparation with a deeper overview of the day-to-day challenges facing the adolescent, such as fatigue, rarely explored despite its impact on their social life [28][29][30][31].
Second, the PTC is a place of training, positioning the adolescent as a direct interlocutor of a new practitioner, without their parents, and RHCPs pointed out a bene cial change in the adolescent-RHCP dynamic. Thus, it develops their self-advocacy, making them express their point of view, sometimes for the rst time, concerning all relevant life issues, from CC self-management to intimate issues. In this exercise, putting their CC into words was sometimes di cult because of an insu cient knowledge, a discrepancy between their own representation and medical physiopathology [32] or coping strategies such as avoiding words perceived as too crude. While this was not an obstacle to self-management skills acquisition, using the exact wording of the CC was encouraged to empower adolescents in an adult healthcare setting. Adolescents were also given crucial information about health needs and functioning in adult care, to help them to take responsibility for their health and enhance their level of con dence in their future health services, both of which facilitate access to healthcare [11].
Finally, the RHCPs' high satisfaction rate of and changes introduced in their clinical practices following PTCs, as well as the favorable 2-years outcomes of participants show that implementing a dedicated consultation helped to develop a transition policy within the hospital and to enhance transition support of adolescents.
Our methodology enabled us to analyze the implementation and functioning of this new service, in particular in terms of participants' responses to CPT, and to adapt the PTC modalities in real time [21]. Because adolescents often minimized the treatment burden, the TBQ was abandoned to only assess the most burdensome topics (tests, appointments, diet). Thus, to involve parents [4,5,15,19] bearing in mind their organizational di culties, we now offer them individual interviews with the AD'venir coordinator or a psychologist and speci c information on-line (https://advenir-robertdebre.aphp.fr/). Raising adolescents' awareness of their own situation, the Good2Go is now completed at the very end of the PTC to give a more accurate indication of what to work before transfer [2]. However, the failure of transfer for one adolescent reminds that the predictive value of transition readiness questionnaires remains unclear in terms of transition success [19,33]. Thus, we found no correlation between Good2Go scores and level of information regarding the transition or social context, which are nevertheless both key-points of transition success. Therefore transition readiness questionnaires must be seen as a complement to a global interview. Inversely, not assessed by Good2Go, lack of autonomy in daily life tasks was not considered su cient to defer the transfer. Indeed, most adolescents become truly independent years later and holistic life-skills are not associated with transition outcome [11]. This reinforces our choice of the Good2Go among others validated questionnaires [19,33].
Although recording might be intrusive, this research offers TPs a unique opportunity for self-re exive and collective work on clinical practice. The external and non-prescriber TP's position facilitates a nonjudgmental attitude and avoids focusing on compliance and inducing a feeling of 'good' or 'bad' patient.
Our results also emphasize two arguments in favor of choosing physician rather than non-physician HCPs to deliver PTCs. First, PTCs require medical knowledge in multiple elds to understand the context of the CC. Second, the physical examination helps to build a bond of trust necessary to address intimate concerns [34]. However, as adults [35], adolescents feel embarrassed to talk with HCPs about sexuality or risky behaviors, which are nevertheless frequent in adolescents with CC [36,37]. This emphasizes the need to develop adolescent medicine in pediatric settings [38].
The main strength is the multidisciplinary approach, crossing the viewpoints of both clinicians and researchers to allow a ner adjustment of the intervention. Well-accepted and less time-and costconsuming than qualitative interviews, audio-recordings allowed a real-life analysis. Finally, including a panel of CC responded to a need to enlarge the recruitment in transition studies [10]. Concerning weaknesses, including the rst adolescents attending a PTC may introduce a recruitment bias, leading to more positive results [39].

Conclusion
Thanks to the study, the re ned content of the PTC offers a comprehensive overview of transition and is now used in routine clinical practice in our hospital. Implementing the PTC represents an achievable prerequisite to improve other important aspects of transition preparation  Participants' responses to the Treatment Burden Questionnaire (n=27)