Maintenance Intravenous Immunoglobulin Treatment for Multiple Sclerosis Coexisting with Ehlers-Danlos Syndrome and Muir-
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CASE REPORT
Maintenance Intravenous Immunoglobulin Treatment for Multiple Sclerosis Coexisting with Ehlers-Danlos Syndrome and Muir-Torre Syndrome: A Case Study Srishti Gupta . Mohnish Suri . Cris S. Constantinescu
Received: July 9, 2020 Ó The Author(s) 2020
ABSTRACT The therapeutic options for disease modification in relapsing-remitting multiple sclerosis (RRMS) have expanded remarkably in the last 15 years. Although intravenous immunoglobulins (IVIg) have shown some therapeutic effects in multiple sclerosis, reducing global supplies, restriction of treatment to essential indications and availability of effective alternative treatments for MS currently exclude IVIg from being an accepted therapy for MS, other than for some exceptional considerations. We report the case of a female patient with RRMS who was diagnosed with Ehlers-Danlos syndrome (EDS) and Muir-Torre syndrome (MTS) Digital Features To view digital features for this article go to https://doi.org/10.6084/m9.figshare.12759788. S. Gupta (&) C. S. Constantinescu (&) Department of Neurology, Queen’s Medical Centre Campus, Nottingham University Hospitals NHS Trust, Derby Road, Nottingham, UK e-mail: [email protected] C. S. Constantinescu e-mail: [email protected] M. Suri Clinical Genetics Service, City Hospital Campus, Nottingham University Hospitals NHS Trust, Nottingham, UK C. S. Constantinescu Division of Clinical Neuroscience, Section of Clinical Neurology, University of Nottingham, Nottingham, UK
soon after the diagnosis of active RRMS was made. The coexisting conditions precluded the use of available disease-modifying treatments. She benefited from monthly and then bi-monthly IVIg, with a single mild relapse over 10 years. Discontinuation of IVIg due to reduced availability with a brief aborted course of subcutaneous PEGylated interferon-beta was followed by significant relapses. Five months after the first ocrelizumab infusion, she developed caecal cancer requiring colectomy. Reinstitution of IVIg is contemplated.
Keywords: Ehlers-Danlos syndrome; Muir-Torre syndrome; Multiple sclerosis
IVIg;
Key Summary Points A complex case of RRMS with EDS and MTS-probable association/trigger/ unmasking IVIg has been found beneficial as an immunomodulatory and potential cancer immunoprophylaxis agent Unmet need of treatment options in this rare case—IVIg kept MS and cancer in remission Despite the global shortage of IVIg, it should be considered for clinically indicated cases
Neurol Ther
INTRODUCTION The landscape for disease-modifying treatments for multiple sclerosis (MS), in particular for the relapsing-remitting forms, has changed dramatically over the last two decades. All current treatments target the immune system by reducing pro-inflammatory responses, blocking lymphocyte migration, providing non-specific immunosuppression or depleting selected immune cell populations. Despite the evidence of a potential therapeutic benefit, intravenous immunoglobulin (IVIg) is currently only very rarely used in MS, where it is reserve
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