Choroid Plexus Volume in Multiple Sclerosis vs Neuromyelitis Optica Spectrum Disorder: A Retrospective, Cross-sectional Analysis

Jannis Müller; Tim Sinnecker; Maria Janina Wendebourg; Regina Schläger; Jens Kuhle; Sabine Schädelin; Pascal Benkert; Tobias Derfuss; Philippe Cattin; Christoph Jud; Florian Spiess; Michael Amann; Therese Lincke; Muhamed Barakovic; Alessandro Cagol; Charidimos Tsagkas; Katrin Parmar; Anne-Katrin Pröbstel; Sophia Reimann; Susanna Asseyer; Ankelien Duchow; Alexander Brandt; Klemens Ruprecht; Nouchine Hadjikhani; Shoko Fukumoto; Mitsuru Watanabe; Katsuhisa Masaki; Takuya Matsushita; Noriko Isobe; Jun-Ichi Kira; Ludwig Kappos; Jens Würfel; Cristina Granziera; Friedemann Paul; Özgür Yaldizli

doi:10.1212/NXI.0000000000001147

Choroid Plexus Volume in Multiple Sclerosis vs Neuromyelitis Optica Spectrum Disorder: A Retrospective, Cross-sectional Analysis

Neurol Neuroimmunol Neuroinflamm. 2022 Feb 25;9(3):e1147. doi: 10.1212/NXI.0000000000001147. Print 2022 May.

Authors

Jannis Müller¹, Tim Sinnecker¹, Maria Janina Wendebourg¹, Regina Schläger¹, Jens Kuhle¹, Sabine Schädelin¹, Pascal Benkert¹, Tobias Derfuss¹, Philippe Cattin¹, Christoph Jud¹, Florian Spiess¹, Michael Amann¹, Therese Lincke¹, Muhamed Barakovic¹, Alessandro Cagol¹, Charidimos Tsagkas¹, Katrin Parmar¹, Anne-Katrin Pröbstel¹, Sophia Reimann¹, Susanna Asseyer¹, Ankelien Duchow¹, Alexander Brandt¹, Klemens Ruprecht¹, Nouchine Hadjikhani¹, Shoko Fukumoto¹, Mitsuru Watanabe¹, Katsuhisa Masaki¹, Takuya Matsushita¹, Noriko Isobe¹, Jun-Ichi Kira¹, Ludwig Kappos¹, Jens Würfel¹, Cristina Granziera¹, Friedemann Paul¹, Özgür Yaldizli²

Affiliations

¹ From the Neurology Clinic and Policlinic (J.M., T.S., M.J.W., R.S., J.K., S.S., P.B., T.D., M.B., A.C., C.T., K.P., A.-K.P., S.R., L.K., C.G., O.Y.), Departments of Head, Spine and Neuromedicine, MS Center and Research Center for Clinical Neuroimmunology and Neuroscience Basel (RC2NB), Clinical Research and Biomedical Engineering, University Hospital Basel and University of Basel; Translational Imaging in Neurology (ThINk) Basel (J.M., S.S., M.B., A.C., C.T., K.P., C.G., O.Y.), Department of Biomedical Engineering, University Hospital Basel and University of Basel, Switzerland; Department of Biomedical Engineering (P.C., C.J., F.S., J.W., C.G.), University of Basel, Allschwil, Switzerland; Medical Imaging Analysis Center AG (M.A., J.W.); Section of Neuroradiology (T.L.), Clinic for Radiology & Nuclear Medicine, University Hospital Basel and University of Basel, Switzerland; Reha Rheinfelden (K.P.), Rheinfelden, Switzerland; NeuroCure Clinical Research Center and Experimental and Clinical Research Center (S.A., A.D., A.B., J.W., F.P.), Max Delbrueck Center for Molecular Medicine and Charité-Universitätsmedizin Berlin, Germany; Department of Neurology (A.B.), University of California, Irvine; Department of Neurology (K.R.), Charité-Universitätsmedizin Berlin, corporate member of Freie Universität Berlin and Humboldt-Universität zu Berlin, Germany; Martinos Center for Biomedical Imaging (N.H.), Massachusetts General Hospital and Harvard Medical School, Boston; Gillberg Neuropsychiatry Center (N.H.), Sahlgrenska Academy, University of Gothenburg, Sweden; Department of Neurology (S.F., M.W., K.M., T.M., N.I., J.-I.K.), Neurological Institute, Graduate School of Medical Sciences, Kyushu University, Fukuoka, Japan; Department of Neurology (J.-I.K.), Brain and Nerve Center, Fukuoka Central Hospital, International University of Health and Welfare, Fukuoka; and Translational Neuroscience Center (J.-I.K.), Graduate School of Medicine, and School of Pharmacy at Fukuoka, International University of Health and Welfare, Okawa, Japan.
² From the Neurology Clinic and Policlinic (J.M., T.S., M.J.W., R.S., J.K., S.S., P.B., T.D., M.B., A.C., C.T., K.P., A.-K.P., S.R., L.K., C.G., O.Y.), Departments of Head, Spine and Neuromedicine, MS Center and Research Center for Clinical Neuroimmunology and Neuroscience Basel (RC2NB), Clinical Research and Biomedical Engineering, University Hospital Basel and University of Basel; Translational Imaging in Neurology (ThINk) Basel (J.M., S.S., M.B., A.C., C.T., K.P., C.G., O.Y.), Department of Biomedical Engineering, University Hospital Basel and University of Basel, Switzerland; Department of Biomedical Engineering (P.C., C.J., F.S., J.W., C.G.), University of Basel, Allschwil, Switzerland; Medical Imaging Analysis Center AG (M.A., J.W.); Section of Neuroradiology (T.L.), Clinic for Radiology & Nuclear Medicine, University Hospital Basel and University of Basel, Switzerland; Reha Rheinfelden (K.P.), Rheinfelden, Switzerland; NeuroCure Clinical Research Center and Experimental and Clinical Research Center (S.A., A.D., A.B., J.W., F.P.), Max Delbrueck Center for Molecular Medicine and Charité-Universitätsmedizin Berlin, Germany; Department of Neurology (A.B.), University of California, Irvine; Department of Neurology (K.R.), Charité-Universitätsmedizin Berlin, corporate member of Freie Universität Berlin and Humboldt-Universität zu Berlin, Germany; Martinos Center for Biomedical Imaging (N.H.), Massachusetts General Hospital and Harvard Medical School, Boston; Gillberg Neuropsychiatry Center (N.H.), Sahlgrenska Academy, University of Gothenburg, Sweden; Department of Neurology (S.F., M.W., K.M., T.M., N.I., J.-I.K.), Neurological Institute, Graduate School of Medical Sciences, Kyushu University, Fukuoka, Japan; Department of Neurology (J.-I.K.), Brain and Nerve Center, Fukuoka Central Hospital, International University of Health and Welfare, Fukuoka; and Translational Neuroscience Center (J.-I.K.), Graduate School of Medicine, and School of Pharmacy at Fukuoka, International University of Health and Welfare, Okawa, Japan. [email protected].

Abstract

Background and objectives: The choroid plexus has been shown to play a crucial role in CNS inflammation. Previous studies found larger choroid plexus in multiple sclerosis (MS) compared with healthy controls. However, it is not clear whether the choroid plexus is similarly involved in MS and in neuromyelitis optica spectrum disorder (NMOSD). Thus, the aim of this study was to compare the choroid plexus volume in MS and NMOSD.

Methods: In this retrospective, cross-sectional study, patients were included by convenience sampling from 4 international MS centers. The choroid plexus of the lateral ventricles was segmented fully automatically on T1-weighted MRI sequences using a deep learning algorithm (Multi-Dimensional Gated Recurrent Units). Uni- and multivariable linear models were applied to investigate associations between the choroid plexus volume, clinically meaningful disease characteristics, and MRI parameters.

Results: We studied 180 patients with MS and 98 patients with NMOSD. In total, 94 healthy individuals and 47 patients with migraine served as controls. The choroid plexus volume was larger in MS (median 1,690 µL, interquartile range [IQR] 648 µL) than in NMOSD (median 1,403 µL, IQR 510 µL), healthy individuals (median 1,533 µL, IQR 570 µL), and patients with migraine (median 1,404 µL, IQR 524 µL; all p < 0.001), whereas there was no difference between NMOSD, migraine, and healthy controls. This was also true when adjusted for age, sex, and the intracranial volume. In contrast to NMOSD, the choroid plexus volume in MS was associated with the number of T2-weighted lesions in a linear model adjusted for age, sex, total intracranial volume, disease duration, relapses in the year before MRI, disease course, Expanded Disability Status Scale score, disease-modifying treatment, and treatment duration (beta 4.4; 95% CI 0.78-8.1; p = 0.018).

Discussion: This study supports an involvement of the choroid plexus in MS in contrast to NMOSD and provides clues to better understand the respective pathogenesis.

MeSH terms

Choroid Plexus / diagnostic imaging
Choroid Plexus / pathology
Cross-Sectional Studies
Humans
Migraine Disorders*
Multiple Sclerosis* / diagnostic imaging
Multiple Sclerosis* / pathology
Neuromyelitis Optica* / diagnostic imaging
Neuromyelitis Optica* / pathology
Retrospective Studies