Literature DB >> 33308940

Sporadic late-onset nemaline myopathy with monoclonal gammopathy of undetermined significance: Report of four patients.

Ali Asghar Okhovat1, Yalda Nilipour2, Reza Boostani3, Fahimeh Vahabizad4, Safa Najmi5, Shahriar Nafissi6, Farzad Fatehi7.   

Abstract

Sporadic late-onset nemaline myopathy (SLONM) is a rare, acquired muscle disease presenting with subacute progression in adulthood. It can be accompanied by a monoclonal gammopathy of undetermined significance (MGUS). We describe clinical and histopathological findings of four SLONM patients with MGUS. In all patients, nemaline rod, inter-myofibrillary network disruption, atrophic changes, peripheral basophilic discoloration, vacuole without rim, and cytoplasmic body without inflammation were seen. Three out of four patients were treated with prednisolone in combination with IVIG monthly and had an appropriate response to the treatment. The optimal first-line treatment remains unclear in SLONM-MGUS, although corticosteroids plus IVIg is associated with favorable clinical response. These treatment modalities might be used as an optional treatment before autologous stem cell transplantation; however, further studies with a higher number of patients are required.
Copyright © 2020 Elsevier B.V. All rights reserved.

Entities:  

Keywords:  Adult; Intravenous immunoglobulins; Monoclonal gammopathy of undetermined significance; Nemaline myopathy; Prednisolone; Therapy

Year:  2020        PMID: 33308940     DOI: 10.1016/j.nmd.2020.11.004

Source DB:  PubMed          Journal:  Neuromuscul Disord        ISSN: 0960-8966            Impact factor:   4.296


  3 in total

1.  Clinicopathologic Profiles of Sporadic Late-Onset Nemaline Myopathy: Practical Importance of Anti-α-Actinin Immunostaining.

Authors:  Bing Zhao; Tingjun Dai; Dandan Zhao; Xiaotian Ma; Cuiping Zhao; Ling Li; Yuan Sun; Yongqing Zhang; Yaping Yan; Jian-Qiang Lu; Fuchen Liu; Chuanzhu Yan
Journal:  Neurol Neuroimmunol Neuroinflamm       Date:  2022-05-17

2.  Rituximab as a treatment for human immunodeficiency virus-associated nemaline myopathy: What does the literature have to tell us?

Authors:  Jucier Gonçalves Júnior; Samuel Katsuyuki Shinjo
Journal:  World J Clin Cases       Date:  2022-02-06       Impact factor: 1.337

3.  Inflammatory features in sporadic late-onset nemaline myopathy are independent from monoclonal gammopathy.

Authors:  Jantima Tanboon; Akinori Uruha; Yukie Arahata; Carsten Dittmayer; Leonille Schweizer; Hans-Hilmar Goebel; Ichizo Nishino; Werner Stenzel
Journal:  Brain Pathol       Date:  2021-05       Impact factor: 6.508

  3 in total

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