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Intravenous immunoglobulin (IVIG) therapy, performed for a long-term period, has been demonstrated able to improve the clinical picture of MMN patients, ameliorating motor symptoms and\/or preventing disease progression. Treatment with subcutaneous immunoglobulin (SCIg) has been shown to be as effective as IVIG. However, previously published data showed that follow-up of MMN patients in treatment with SCIg lasted no more than 56\u00a0months. We report herein the results of a long-term SCIg treatment follow up (up to 96\u00a0months) in a group of 8 MMN patients (6\u00a0M; 2F), previously stabilized with IVIG therapy. Clinical follow-up included the administration of Medical Research Council (MRC) sum-score, the Overall Neuropathy Limitation Scale (ONLS) and the Life Quality Index questionnaire (LQI) at baseline and then every 6\u00a0months. Once converted to SCIg, patients\u2019 responsiveness was quite good. Strength and motor functions remained stable or even improved during this long-term follow-up with benefits on walking capability, resistance to physical efforts and ability in hand fine movements.<\/jats:p>","DOI":"10.1038\/s41598-021-88711-9","type":"journal-article","created":{"date-parts":[[2021,4,28]],"date-time":"2021-04-28T10:04:55Z","timestamp":1619604295000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":7,"title":["Long-term treatment with subcutaneous immunoglobulin in multifocal motor neuropathy"],"prefix":"10.1038","volume":"11","author":[{"given":"Luca","family":"Gentile","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Massimo","family":"Russo","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Carmelo","family":"Rodolico","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ilenia","family":"Arimatea","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Giuseppe","family":"Vita","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Antonio","family":"Toscano","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anna","family":"Mazzeo","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2021,4,28]]},"reference":[{"issue":"5","key":"88711_CR1","doi-asserted-by":"publisher","first-page":"559","DOI":"10.1097\/WCO.0000000000000605","volume":"31","author":"K Beadon","year":"2018","unstructured":"Beadon, K., Guimar\u00e3es-Costa, R. & L\u00e9ger, J. M. Multifocal motor neuropathy. Curr. Opin. Neurol. 31(5), 559\u2013564. https:\/\/doi.org\/10.1097\/WCO.0000000000000605 (2018).","journal-title":"Curr. Opin. Neurol."},{"issue":"6","key":"88711_CR2","doi-asserted-by":"publisher","first-page":"1","DOI":"10.2147\/DNND.S96258","volume":"22","author":"E Jovanovich","year":"2015","unstructured":"Jovanovich, E. & Karam, C. Human immune globulin infusion in the management of multifocal motor neuropathy. Degener. Neurol. Neuromuscul. Dis. 22(6), 1\u201312. https:\/\/doi.org\/10.2147\/DNND.S96258 (2015).","journal-title":"Degener. Neurol. Neuromuscul. Dis."},{"issue":"5","key":"88711_CR3","doi-asserted-by":"publisher","first-page":"397","DOI":"10.1007\/s40265-013-0029-z","volume":"73","author":"E Nobile-Orazio","year":"2013","unstructured":"Nobile-Orazio, E. & Gallia, F. Multifocal motor neuropathy: Current therapies and novel strategies. Drugs 73(5), 397\u2013406. https:\/\/doi.org\/10.1007\/s40265-013-0029-z (2013).","journal-title":"Drugs"},{"issue":"375","key":"88711_CR4","doi-asserted-by":"publisher","first-page":"190","DOI":"10.1016\/j.jns.2017.01.061","volume":"15","author":"A Kumar","year":"2017","unstructured":"Kumar, A., Patwa, H. S. & Nowak, R. J. Immunoglobulin therapy in the treatment of multifocal motor neuropathy. J. Neurol. Sci. 15(375), 190\u2013197. https:\/\/doi.org\/10.1016\/j.jns.2017.01.061 (2017).","journal-title":"J. Neurol. Sci."},{"issue":"1","key":"88711_CR5","doi-asserted-by":"publisher","first-page":"48","DOI":"10.1038\/nrneurol.2011.175","volume":"8","author":"L Vlam","year":"2011","unstructured":"Vlam, L. et al. Multifocal motor neuropathy: Diagnosis, pathogenesis and treatment strategies. Nat. Rev. Neurol. 8(1), 48\u201358. https:\/\/doi.org\/10.1038\/nrneurol.2011.175 (2011).","journal-title":"Nat. Rev. Neurol."},{"issue":"4","key":"88711_CR6","doi-asserted-by":"publisher","first-page":"295","DOI":"10.1111\/j.1529-8027.2010.00290.x","volume":"15","author":"Joint Task Force of the EFNS and the PNS","year":"2010","unstructured":"Joint Task Force of the EFNS and the PNS. European Federation of Neurological Societies\/Peripheral Nerve Society guideline on management of multifocal motor neuropathy. Report of a joint task force of the European Federation of Neurological Societies and the Peripheral Nerve Society-first revision. J. Peripher. Nerv. Syst. 15(4), 295\u2013301. https:\/\/doi.org\/10.1111\/j.1529-8027.2010.00290.x (2010).","journal-title":"J. Peripher. Nerv. Syst."},{"issue":"8","key":"88711_CR7","doi-asserted-by":"publisher","first-page":"S21","DOI":"10.12968\/bjon.2012.21.Sup8.S21","volume":"21","author":"ME Braine","year":"2012","unstructured":"Braine, M. E. & Woodall, A. A comparison between intravenous and subcutmaneous immunogobulin. Br. J. Nurs. 21(8), S21\u2013S22. https:\/\/doi.org\/10.12968\/bjon.2012.21.Sup8.S21 (2012) (S24\u2013S27).","journal-title":"Br. J. Nurs."},{"issue":"4","key":"88711_CR8","doi-asserted-by":"publisher","first-page":"610","DOI":"10.1002\/mus.25967","volume":"57","author":"I Christiansen","year":"2018","unstructured":"Christiansen, I., Markvardsen, L. H. & Jakobsen, J. Comparisons in fluctuation of muscle strength and function in patients with immune-mediated neuropathy treated with intravenous versus subcutaneous immunoglobulin. Muscle Nerve. 57(4), 610\u2013614. https:\/\/doi.org\/10.1002\/mus.25967 (2018).","journal-title":"Muscle Nerve."},{"issue":"10","key":"88711_CR9","doi-asserted-by":"publisher","first-page":"2443","DOI":"10.1111\/trf.13727","volume":"56","author":"LH Markvardsen","year":"2016","unstructured":"Markvardsen, L. H., Christiansen, I. & Jakobsen, J. Improvement of hemoglobin levels after a switch from intravenous to subcutaneous administration of immunoglobulin in chronic inflammatory demyelinating polyneuropathy and multifocal motor neuropathy. Transfusion 56(10), 2443\u20132448. https:\/\/doi.org\/10.1111\/trf.13727 (2016).","journal-title":"Transfusion"},{"issue":"1","key":"88711_CR10","doi-asserted-by":"publisher","first-page":"14","DOI":"10.1177\/1756285614563056","volume":"8","author":"RD Hadden","year":"2015","unstructured":"Hadden, R. D. & Marreno, F. Switch from intravenous to subcutaneous immunoglobulin in CIDP and MMN: Improved tolerability and patient satisfaction. Ther. Adv. Neurol. Disord. 8(1), 14\u201319. https:\/\/doi.org\/10.1177\/1756285614563056 (2015).","journal-title":"Ther. Adv. Neurol. Disord."},{"key":"88711_CR11","doi-asserted-by":"publisher","first-page":"19","DOI":"10.1016\/j.jns.2017.04.039","volume":"378","author":"LH Markvardsen","year":"2017","unstructured":"Markvardsen, L. H. & Harbo, T. 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A smooth transition protocol for patients with multifocal motor neuropathy going from intravenous to subcutaneous immunoglobulin therapy: An open-label proof-of-concept study. J. Peripher. Nerv. Syst. 16(2), 92\u201397. https:\/\/doi.org\/10.1111\/j.1529-8027.2011.00330.x (2011).","journal-title":"J. Peripher. Nerv. Syst."},{"issue":"11","key":"88711_CR16","doi-asserted-by":"publisher","first-page":"2159","DOI":"10.1007\/s00415-014-7444-2","volume":"261","author":"D Cocito","year":"2014","unstructured":"Cocito, D. et al. SCIg and chronic dysimmune neuropathies Italian Network. Subcutaneous immunoglobulin in CIDP and MMN: A short-term nationwide study. J. Neurol. 261(11), 2159\u20132164. https:\/\/doi.org\/10.1007\/s00415-014-7444-2 (2014).","journal-title":"J. Neurol."},{"issue":"7","key":"88711_CR17","doi-asserted-by":"publisher","first-page":"791","DOI":"10.1136\/jnnp-2014-310280","volume":"87","author":"D Cocito","year":"2016","unstructured":"Cocito, D. et al. Subcutaneous immunoglobulin in CIDP and MMN: A different long-term clinical response?. J. Neurol. Neurosurg. Psychiatry. 87(7), 791\u2013793. https:\/\/doi.org\/10.1136\/jnnp-2014-310280 (2016).","journal-title":"J. Neurol. Neurosurg. Psychiatry."},{"key":"88711_CR18","doi-asserted-by":"crossref","unstructured":"Katzberg, H., Rasutis, V. & Bril, V. Subcutaneous immunoglobulin (IgPRO20) for maintenance treatment in patients with multifocal motor neuropathy (P7.094). Neurology 84(Suppl 14) (2015).","DOI":"10.1212\/WNL.84.14_supplement.P7.094"},{"key":"88711_CR19","doi-asserted-by":"publisher","first-page":"1299","DOI":"10.3389\/fimmu.2018.01299","volume":"9","author":"Y Guo","year":"2018","unstructured":"Guo, Y., Tian, X., Wang, X. & Xiao, Z. Adverse effects of immunoglobulin therapy. Front. Immunol. 9, 1299. https:\/\/doi.org\/10.3389\/fimmu.2018.01299 (2018).","journal-title":"Front. Immunol."}],"container-title":["Scientific Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.nature.com\/articles\/s41598-021-88711-9.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/www.nature.com\/articles\/s41598-021-88711-9","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/www.nature.com\/articles\/s41598-021-88711-9.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,8,29]],"date-time":"2024-08-29T17:30:33Z","timestamp":1724952633000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.nature.com\/articles\/s41598-021-88711-9"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,4,28]]},"references-count":19,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2021,12]]}},"alternative-id":["88711"],"URL":"https:\/\/doi.org\/10.1038\/s41598-021-88711-9","relation":{},"ISSN":["2045-2322"],"issn-type":[{"value":"2045-2322","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,4,28]]},"assertion":[{"value":"22 February 2021","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"16 April 2021","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"28 April 2021","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"Luca Gentile is sub-investigator in clinical trials of Alnylam, Ionis, Takeda. He also reports trav-el grants from Kedrion and CSL Behring to attend scientific meeting and acknowledge speaker fee and consulting honoraria from Pfizer. Massimo Russo acknowledges receiving speaker fee and consulting honoraria from Akcea and Alnylam and a travel grant from Pfizer. Anna Mazzeo is Principal Investigator in clinical trials of Alnylam and Ionis, sub-investigator in clinical trials of Alnylam, Ionis, Takeda. She also re-ports travel grants from Kedrion and CSL Behring to attend scientific meeting and acknowledge speaker fee and consulting honoraria from Alnylam, Akcea and Pfizer. Giuseppe Vita is Princi-pal Investigator in clinical trials of Roche, Sarepta, Santhera, Italfarmaco, Biogen, Avexis, Al-nylam, Ionis, Wave. He is also consulent of Advisory Board for Roche, Avexis, Alnylam, Akcea and he acknowledges speaker fee and consulting honoraria from Alnylam, Akcea and Pfizer. Antonio Toscano is Principal Investigator in clinical trials of Takeda and Genzyme. He acknowledges speaker fee and consulting honoraria from Kedrion, CSL Behring and Genzyme. Carmelo Rodolico and Ilenia Arimatea declare no competing interests.","order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"9216"}}