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. 2020 Aug 5;8:125. doi: 10.1186/s40478-020-01007-3

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© The Author(s) 2020

Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data.

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Fig. 2 — Characteristic muscle pathology in patients with high and low titers of Mi-2 auto-antibody. a-d Patient 4 with high Mi-2 titer. a Low and (b) high power images of H&E stained cryosections showed prominent perifascicular myofiber necrosis and regeneration. c Alkaline phosphatase stain highlighted frequent regenerating fibers (arrow heads) as well as strong perimysial connective tissue reactivity (arrows). d C5b-9 immunostain showed frequent necrotic fibers concentrated in the perifascicular regions (arrow heads) but might present at the center of fascicles. Viable but injured myofibers might show sarcolemmal C5b-9 reactivity (arrow heads). There was no significant capillary C5b-9 reactivity. Perimysium was outlined by black lines. “a” indicate perimysial artery. “v” indicate perimysial vein. e-h Patient 8 with low Mi-2 titer. e Low and (f) high power H&E showed prominent perifascicular atrophy and frequent internal nuclei, but few acutely necrotic fibers. g Alkaline phosphatase stain highlighted rare regenerating fibers (arrow heads) and patchy discontinuous perimysial connective tissue reactivity (arrows). h C5b-9 immunostain highlighted occasional necrotic fibers (arrow heads) but rather wide spread sarcolemmal C5b-9 reactivity in viable myofibers (arrow heads). There was no significant capillary C5b-9 reactivity