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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 designResearch in context

Authors :
Helge Hebestreit
Anne-Marie Lapstich
Lilly Brandstetter
Christian Krauth
Jürgen Deckert
Kirsten Haas
Lisa Pfister
Stefanie Witt
Christopher Schippers
Jan Dieris-Hirche
Tim Maisch
Oliver Tüscher
Lavinia Bârlescu
Alexandra Berger
Mark Berneburg
Vanessa Britz
Anna Deibele
Holm Graeßner
Harald Gündel
Gereon Heuft
Thomas Lücke
Christine Mundlos
Julia Quitmann
Frank Rutsch
Katharina Schubert
Jörg Bernhard Schulz
Susann Schweiger
Cornelia Zeidler
Lena Zeltner
Martina de Zwaan
Federica Akkaya
Christine Babka
Lisa Bannert
Anja Bärsch-Michelmann
Leonie Böhm
Folke Brinkmann
Monika Bullinger
Holger Cario
Moritz de Greck
Klaus-Michael Debatin
Katrin Dillmann-Jehn
Jutta Eymann
Julia Frisch
Anja Glode
Vega Gödecke
Corinna Grasemann
Eva Grauer
Astrid Haas
Lea Haisch
Isabell
Heinrich
Melissa Held
Julia Hennermann
Stephan Herpertz
Anne Herrmann-Werner
Julian Hett
Peter Heuschmann
Bettina Hilbig
Laura Holthöfer
Christiane Imhof
Florian Junne
Jan Kassubek
Kevin-Thomas Koschitzki
Heike Krassort
Birgit Kropff
Julia Kuhn
Philipp Latzko
Thomas Loew
Albert C. Ludolph
Torsten Meyer
Isabell Meyer dos Santos
Klaus Mohnike
Martina Monninger
Martin Mücke
Susanne Müller
Thomas Musacchio
Margret Nießen
Mariel Nöhre
Stephan Ott
Andrea Petermann-Meyer
Christina Pfeifer-Duck
Lea-Sophie Piduhn
Carina Rampp
Olaf Rieß
Kristina Schaubert
Annika Schmidt
Simone Schneider
Ludger Schoels
Martina Schwalba
Udo Selig
Alexandra Sroka
Toni Steinbüchel
Sebastian Stösser
Steffi Suchant
Kathrin Ungethüm
Matthias Vogel
Daniela Volk
Christoph Vollmuth
Solange Volnov
Thomas O.F. Wagner
Sabrina Walter
Bodo Warrings
Kamil Zajt
Karola Zenker
David Zhang
Stephan Zipfel
Lavinia Aurelia Bârlescu
Julia Hannah Quitmann
Jörg B. Schulz
Lena Margarete Zeltner
Source :
EClinicalMedicine, Vol 65, Iss , Pp 102260- (2023)
Publication Year :
2023
Publisher :
Elsevier, 2023.

Abstract

Summary: 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.

Details

Language :
English
ISSN :
25895370
Volume :
65
Issue :
102260-
Database :
Directory of Open Access Journals
Journal :
EClinicalMedicine
Publication Type :
Academic Journal
Accession number :
edsdoj.7104fbd316dd40be90da99c4bb8c26c0
Document Type :
article
Full Text :
https://doi.org/10.1016/j.eclinm.2023.102260