{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,17]],"date-time":"2026-10-17T12:12:19Z","timestamp":1792239139690,"version":"4.4.0"},"reference-count":48,"publisher":"SAGE Publications","issue":"2","license":[{"start":{"date-parts":[[2023,6,13]],"date-time":"2023-06-13T00:00:00Z","timestamp":1686614400000},"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":["Int J Surg Pathol"],"published-print":{"date-parts":[[2024,4]]},"abstract":"<jats:p>\n                    Deficiency of fumarate hydratase (FH) protein expression in uterine corpus leiomyomas may be attributable to either germline or somatic mutations of the\n                    <jats:italic toggle=\"yes\">FH<\/jats:italic>\n                    gene, the former being definitional for the hereditary leiomyomatosis and renal cell cancer syndrome. The authors assess whether, using previously reported FH-associated morphologic features, FH protein-deficient uterine corpus leiomyomas associated with a pathogenic germline mutations of the\n                    <jats:italic toggle=\"yes\">FH<\/jats:italic>\n                    gene (group 1) are distinguishable from FH protein-deficient uterine corpus leiomyomas without such mutations (and whose FH protein loss is presumed to be attributable to somatic\/epigenetic inactivation or other unknown phenomena: group 2). Groups 1 and 2 were compared regarding a variety of clinicopathologic features, including 7 core \u201cFH-associated\u201d tumoral morphologic features: staghorn vasculature; alveolar-type edema; bizarre nuclei; chain-like tumor nuclei; hyaline cytoplasmic globules; prominent nucleoli, intranuclear inclusions, and perinucleolar halos; and prominent eosinophilic\/fibrillary cytoplasm. Among 2418 patients diagnosed with uterine corpus leiomyoma during the study period, FH-associated morphologic features were reported in 1.5% (37 patients), and FH immunohistochemistry was performed in 29 (1.19%). Fourteen (48.27%) of the 29 patients showed FH protein deficiency by immunohistochemistry. Twelve patients underwent germline testing, of which 8 (66.7%) were classified as group 1 and 4 (33.3%) as group 2. FH protein-deficient tumors were larger (10.44 vs 4.08\u2005cm,\n                    <jats:italic toggle=\"yes\">P<\/jats:italic>\n                    \u2009\u2009=\u2009\u20090.01) and associated with younger patients (42.05 vs 47.97,\n                    <jats:italic toggle=\"yes\">P<\/jats:italic>\n                    \u2009\u2009=\u2009\u20090.004) than 370 randomly selected uterine leiomyoma controls. Groups 1 and 2 showed no significant differences in patient age and tumor size. In group 1 tumors, the FH-associated morphologic features were generally present diffusely; all group 1 tumors displayed \u22655 FH-associated features, whereas all group 2 tumors displayed &lt;5 FH-associated features (means 6.5\u2009\u2009\u00b1\u2009\u20090.53 vs 3.5\u2009\u2009\u00b1\u2009\u20091.00,\n                    <jats:italic toggle=\"yes\">P<\/jats:italic>\n                    \u2009&lt;\u20090.001). Notably, eosinophilic\/fibrillary cytoplasm and alveolar-type edema were each significantly more prevalent in group 1 tumors than group 2 tumors (\n                    <jats:italic toggle=\"yes\">P<\/jats:italic>\n                    \u2009\u2009=\u2009\u20090.018 for both). No single morphologic feature was found to be completely sensitive and specific in making the distinction between group 1 and 2 tumors. Our findings suggest that groups 1 and 2 are unlikely to be morphologically distinguishable by individual morphologic features. Whether there is a combination of features that can reliably make this distinction is unclear and will require additional studies with larger cohorts.\n                  <\/jats:p>","DOI":"10.1177\/10668969231180285","type":"journal-article","created":{"date-parts":[[2023,6,14]],"date-time":"2023-06-14T02:30:54Z","timestamp":1686709854000},"page":"340-355","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":9,"title":["Fumarate Hydratase-Deficient Leiomyoma of the Uterine Corpus: Comparative Morphologic Analysis of Protein-Deficient Tumors With and Without Pathogenic Germline Fumarate Hydratase Gene Mutations"],"prefix":"10.1177","volume":"32","author":[{"given":"Wangpan","family":"Shi","sequence":"first","affiliation":[{"name":"University of California San Diego Health System"},{"name":"University of California San Diego School of Medicine"},{"name":"University of California San Diego School of 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