Publisher DOI: | 10.1016/j.eclinm.2023.102260 | Title: | Effect of the addition of a mental health specialist for evaluation of undiagnosed patients in centres for rare diseases (ZSE-DUO): a prospective, controlled trial with a two-phase cohort design | Language: | English | Authors: | Hebestreit, Helge Lapstich, Anne-Marie Brandstetter, Lilly Krauth, Christian Deckert, Jürgen Haas, Kirsten Pfister, Lisa Witt, Stefanie Schippers, Christopher Dieris-Hirche, Jan Maisch, Tim Tüscher, Oliver Bârlescu, Lavinia Berger, Alexandra Berneburg, Mark Britz, Vanessa Deibele, Anna Graeßner, Holm Gündel, Harald Heuft, Gereon Lücke, Thomas Mundlos, Christine Quitmann, Julia ![]() Rutsch, Frank Schubert, Katharina Schulz, Jörg Bernhard Schweiger, Susann Zeidler, Cornelia Zeltner, Lena de Zwaan, Martina |
metadata.local.contributorCorporate.other: | ZSE-DUO Working Group | Keywords: | Diagnostic services; Medically unexplained symptoms; Mentalhealth; Patient care team; Rare diseases | Issue Date: | 6-Oct-2023 | Publisher: | Elsevier | Journal or Series Name: | EClinicalMedicine | Volume: | 65 | Abstract: | Background: People with complex symptomatology but unclear diagnosis presenting to a centre for rare diseases (CRD) may present with mental (co-)morbidity. We hypothesised that combining an expert in somatic medicine with a mental health specialist working in tandem will improve the diagnostic outcome. Methods: Patients aged 12 years and older who presented to one of the 11 participating German CRDs with an unknown diagnosis were recruited into this prospective cohort trial with a two-phase cohort design. From October 1, 2018 to September 30, 2019, participants were allocated to standard care (SC, N = 684), and from October 1, 2019 to January 31, 2021 to innovative care (IC, N = 695). The cohorts consisted mainly of adult participants with only a minority of children included (N = 67). IC included the involvement of a mental health specialist in all aspects of care (e.g., assessing medical records, clinic visits, telehealth care, and case conferences). Clinicaltrials.gov identifier: NCT03563677. Findings: The proportion of patients with diagnoses established within 12 months after the first visit to the CRD explaining the entire symptomatology (primary outcome) was 19% (N = 131 of 672) in the SC and 42% (N = 286 of 686) in the IC cohort (OR adjusted for centre effects 3.45 [95% CrI: 1.99-5.65]). The difference was mainly due to a higher prevalence of mental disorders and non-rare somatic diseases in the IC cohort. The median time to explaining diagnoses was one month shorter with IC (95% CrI: 1-2), and significantly more patients could be referred to local regular care in the IC (27.5%; N = 181 of 659) compared to the SC (12.3%; N = 81 of 658) cohort (OR adjusted for centre effects 2.70 [95% CrI: 2.02-3.60]). At 12-month follow-up, patient satisfaction with care was significantly higher in the IC compared to the SC cohort, while quality of life was not different between cohorts. Interpretation: Our findings suggested that including a mental health specialist in the entire evaluation process of CRDs for undiagnosed adolescents and adults should become an integral part of the assessment of individuals with a suspected rare disease. Funding: The study was funded by the Global Innovation Fund from the Joint Federal Committee in Germany (Innovationsfonds des Gemeinsamen Bundesausschusses), grant number 01NVF17031. |
URI: | https://hdl.handle.net/20.500.12738/17195 | ISSN: | 2589-5370 | Review status: | This version was peer reviewed (peer review) | Institute: | Department Soziale Arbeit Fakultät Wirtschaft und Soziales |
Type: | Article | Additional note: | article number: 102260 |
Appears in Collections: | Publications without full text |
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