{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,27]],"date-time":"2026-06-27T14:10:56Z","timestamp":1782569456722,"version":"3.54.5"},"reference-count":18,"publisher":"SAGE Publications","issue":"1","license":[{"start":{"date-parts":[[2022,12,26]],"date-time":"2022-12-26T00:00:00Z","timestamp":1672012800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["Pediatr Dev Pathol"],"published-print":{"date-parts":[[2023,1]]},"abstract":"<jats:sec>\n                    <jats:title>Background:<\/jats:title>\n                    <jats:p>Pathogenic mutations in the smooth muscle myosin heavy chain gene, MYH11, cause megacystis megacolon intestinal hypoperistalsis syndrome and other forms of chronic intestinal pseudo-obstruction. Evaluation of intestinal tissues from affected patients is often performed before mutational analysis, but the pathological findings of MYH11-variant visceral myopathy have not been well defined.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods:<\/jats:title>\n                    <jats:p>Light microscopic, immunohistochemical, and ultrastructural findings from multiple intestinal samples from 2 patients with MYH11-variant visceral myopathy were reviewed, including MYH11-specific immunohistochemistry. The findings were compared with intestinal samples from patients with gamma-smooth muscle actin ( ACTG2)-variant visceral myopathy and non-pseudo-obstruction controls.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results:<\/jats:title>\n                    <jats:p>Apart from non-specific changes (e.g., muscle hypertrophy and distension-related muscularis propria necrosis), no alterations were identified by routine histopathological evaluation or electron microscopy. Immunohistochemistry with antibodies against a battery of smooth muscle proteins, including MYH11, revealed indistinguishable patterns of immunoreactivity in the muscularis propria of both patients and controls.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions:<\/jats:title>\n                    <jats:p>Myopathic morphological or immunohistochemical changes may not be present in intestinal specimens from patients with MYH11-variant visceral myopathy. Molecular genetic studies should be considered for patients with chronic intestinal pseudo-obstruction and normal or non-specific pathology findings.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1177\/10935266221128133","type":"journal-article","created":{"date-parts":[[2022,12,26]],"date-time":"2022-12-26T06:59:29Z","timestamp":1672037969000},"page":"39-51","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":5,"title":["Histopathological, Ultrastructural, and Immunohistochemical Findings in\n                    <i>MYH11<\/i>\n                    -Variant Visceral Myopathy"],"prefix":"10.1177","volume":"26","author":[{"given":"Raj P.","family":"Kapur","sequence":"first","affiliation":[{"name":"Department of Laboratories, Seattle Children\u2019s Hospital, Seattle, WA, USA"},{"name":"Department of Laboratory Medicine and Pathology, University of Washington, Seattle, WA, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2022,12,26]]},"reference":[{"key":"bibr1-10935266221128133","doi-asserted-by":"publisher","DOI":"10.1152\/ajpgi.00066.2021"},{"key":"bibr2-10935266221128133","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.1320460110"},{"key":"bibr3-10935266221128133","doi-asserted-by":"publisher","DOI":"10.1152\/ajpcell.00131.2007"},{"key":"bibr4-10935266221128133","doi-asserted-by":"publisher","DOI":"10.1152\/physrev.00023.2003"},{"key":"bibr5-10935266221128133","doi-asserted-by":"publisher","DOI":"10.1038\/ejhg.2014.256"},{"key":"bibr6-10935266221128133","doi-asserted-by":"publisher","DOI":"10.1111\/cge.13617"},{"key":"bibr7-10935266221128133","doi-asserted-by":"publisher","DOI":"10.1002\/humu.23986"},{"key":"bibr8-10935266221128133","doi-asserted-by":"publisher","DOI":"10.1038\/s10038-019-0651-z"},{"key":"bibr9-10935266221128133","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.38647"},{"key":"bibr10-10935266221128133","doi-asserted-by":"publisher","DOI":"10.1111\/cge.13557"},{"key":"bibr11-10935266221128133","doi-asserted-by":"publisher","DOI":"10.1177\/10935266221107449"},{"key":"bibr12-10935266221128133","doi-asserted-by":"publisher","DOI":"10.1053\/gast.2001.26320"},{"key":"bibr13-10935266221128133","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3468(89)80564-0"},{"key":"bibr14-10935266221128133","doi-asserted-by":"publisher","DOI":"10.1053\/j.sempedsurg.2012.07.005"},{"key":"bibr15-10935266221128133","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3468(83)80275-9"},{"key":"bibr16-10935266221128133","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3468(96)90058-5"},{"key":"bibr17-10935266221128133","doi-asserted-by":"publisher","DOI":"10.1097\/MEG.0b013e3282efa514"},{"key":"bibr18-10935266221128133","doi-asserted-by":"publisher","DOI":"10.1146\/annurev-genom-091212-153408"}],"container-title":["Pediatric and Developmental Pathology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/10935266221128133","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/full-xml\/10.1177\/10935266221128133","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/10935266221128133","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,5,2]],"date-time":"2026-05-02T04:09:11Z","timestamp":1777694951000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.sagepub.com\/doi\/10.1177\/10935266221128133"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,12,26]]},"references-count":18,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2023,1]]}},"alternative-id":["10.1177\/10935266221128133"],"URL":"https:\/\/doi.org\/10.1177\/10935266221128133","relation":{},"ISSN":["1093-5266","1615-5742"],"issn-type":[{"value":"1093-5266","type":"print"},{"value":"1615-5742","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022,12,26]]}}}