PMID- 33911162
OWN - NLM
STAT- MEDLINE
DCOM- 20211014
LR  - 20211014
IS  - 2045-2322 (Electronic)
IS  - 2045-2322 (Linking)
VI  - 11
IP  - 1
DP  - 2021 Apr 28
TI  - Long-term treatment with subcutaneous immunoglobulin in multifocal motor 
      neuropathy.
PG  - 9216
LID - 10.1038/s41598-021-88711-9 [doi]
LID - 9216
AB  - Multifocal motor neuropathy (MMN) is a rare disease with a prevalence of less 
      than 1 per 100,000 people. 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 months. We report 
      herein the results of a long-term SCIg treatment follow up (up to 96 months) in a 
      group of 8 MMN patients (6 M; 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 months. Once converted to 
      SCIg, patients' 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.
FAU - Gentile, Luca
AU  - Gentile L
AD  - Unit of Neurology and Neuromuscular Diseases, Department of Clinical and 
      Experimental Medicine, University of Messina, Messina, Italy. 
      lucagentile84@yahoo.it.
AD  - UOC Neurologia e Malattie Neuromuscolari, Policlinico G. Martino, via Consolare 
      Valeria 1, 98124, Messina, ME, Italy. lucagentile84@yahoo.it.
FAU - Russo, Massimo
AU  - Russo M
AD  - Unit of Neurology and Neuromuscular Diseases, Department of Clinical and 
      Experimental Medicine, University of Messina, Messina, Italy.
FAU - Rodolico, Carmelo
AU  - Rodolico C
AD  - Unit of Neurology and Neuromuscular Diseases, Department of Clinical and 
      Experimental Medicine, University of Messina, Messina, Italy.
FAU - Arimatea, Ilenia
AU  - Arimatea I
AD  - Unit of Neurology and Neuromuscular Diseases, Department of Clinical and 
      Experimental Medicine, University of Messina, Messina, Italy.
FAU - Vita, Giuseppe
AU  - Vita G
AD  - Unit of Neurology and Neuromuscular Diseases, Department of Clinical and 
      Experimental Medicine, University of Messina, Messina, Italy.
FAU - Toscano, Antonio
AU  - Toscano A
AD  - Unit of Neurology and Neuromuscular Diseases, Department of Clinical and 
      Experimental Medicine, University of Messina, Messina, Italy.
FAU - Mazzeo, Anna
AU  - Mazzeo A
AD  - Unit of Neurology and Neuromuscular Diseases, Department of Clinical and 
      Experimental Medicine, University of Messina, Messina, Italy.
LA  - eng
PT  - Journal Article
DEP - 20210428
PL  - England
TA  - Sci Rep
JT  - Scientific reports
JID - 101563288
RN  - 0 (Immunoglobulins, Intravenous)
SB  - IM
MH  - Adult
MH  - Aged
MH  - Aged, 80 and over
MH  - Female
MH  - Follow-Up Studies
MH  - Humans
MH  - Immunoglobulins, Intravenous/*administration & dosage
MH  - Infusions, Subcutaneous
MH  - Male
MH  - Middle Aged
MH  - Motor Neuron Disease/*drug therapy/pathology
MH  - Polyneuropathies/*drug therapy/pathology
MH  - Prognosis
MH  - Quality of Life
MH  - Retrospective Studies
PMC - PMC8080704
COIS- 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.
EDAT- 2021/04/30 06:00
MHDA- 2021/10/15 06:00
PMCR- 2021/04/28
CRDT- 2021/04/29 06:10
PHST- 2021/02/22 00:00 [received]
PHST- 2021/04/16 00:00 [accepted]
PHST- 2021/04/29 06:10 [entrez]
PHST- 2021/04/30 06:00 [pubmed]
PHST- 2021/10/15 06:00 [medline]
PHST- 2021/04/28 00:00 [pmc-release]
AID - 10.1038/s41598-021-88711-9 [pii]
AID - 88711 [pii]
AID - 10.1038/s41598-021-88711-9 [doi]
PST - epublish
SO  - Sci Rep. 2021 Apr 28;11(1):9216. doi: 10.1038/s41598-021-88711-9.