{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,10]],"date-time":"2026-10-10T00:21:01Z","timestamp":1791591661316,"version":"4.3.4"},"reference-count":18,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2024,6,28]],"date-time":"2024-06-28T00:00:00Z","timestamp":1719532800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2024,6,28]],"date-time":"2024-06-28T00:00:00Z","timestamp":1719532800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/501100007129","name":"Natural Science Foundation of Shandong Province","doi-asserted-by":"publisher","award":["ZR2020QH044"],"award-info":[{"award-number":["ZR2020QH044"]}],"id":[{"id":"10.13039\/501100007129","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100007129","name":"Natural Science Foundation of Shandong Province","doi-asserted-by":"publisher","award":["ZR2020MH272"],"award-info":[{"award-number":["ZR2020MH272"]}],"id":[{"id":"10.13039\/501100007129","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100007129","name":"Natural Science Foundation of Shandong Province","doi-asserted-by":"publisher","award":["ZR2020MH272"],"award-info":[{"award-number":["ZR2020MH272"]}],"id":[{"id":"10.13039\/501100007129","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["BMC Women's Health"],"abstract":"<jats:title>Abstract<\/jats:title><jats:sec>\n                <jats:title>Background<\/jats:title>\n                <jats:p>Peutz-Jeghers syndrome (PJS) is characterized by the presence of hamartomatous polyps in the gastrointestinal tract and mucocutaneous pigmentation on the lips, oral mucosa, nose, fingers, and toes. Synchronous mucinous metaplasia and neoplasia of the female genital tract (SMMN-FGT) refers to the occurrence of multifocal mucinous lesions in at least two sites, including the cervix, uterus, fallopian tubes, and ovaries, in the female genital tract. SMMN-FGT and PJS are rare diseases with a very low incidence, especially when occurring simultaneously.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Case presentation<\/jats:title>\n                <jats:p>We report a case in which a woman with a large mass on the left ovary underwent a gynecological surgery and was diagnosed with cervical gastric-type adenocarcinoma and mucinous lesions in the endometrium, bilateral fallopian tubes, and ovary, i.e., SMMN-FGT, by postoperative paraffin pathology. The patient sought medical attention for abdominal distension and enlargement. A gynecological ultrasound revealed a multilocular cystic mass in the pelvis, while serum tumor markers were within normal limits, with mildly elevated carbohydrate antigen 199 and carbohydrate antigen 125 levels. Cervical thin-prep cytology test result was negative. The patient had a family history of PJS with black spots on her skin and mucous membranes since the age of 8 years. She underwent multiple partial small bowel resections and gastrointestinal polypectomy owing to intestinal obstruction and intussusception. She underwent left adnexectomy, hysterectomy, right salpingectomy, greater omental resection, appendectomy and right ovary biopsy, and received six courses of adjuvant chemotherapy with Lopressor plus Carboplatin. Genetic testing revealed a heterozygous serine threonine kinase 11 germline mutation and there were no signs of recurrence during the 18-month follow-up period after treatment.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Conclusions<\/jats:title>\n                <jats:p>This is a rare case in which PJS was complicated by SMMN-FGT. Owing to its extreme rarity, there are no guidelines, but reported cases appear to indicate a poor prognosis. We retrospectively reviewed all cases of collisions between PJS and SMMN-FGT and explored the clinical features, pathological characteristics, diagnosis, treatment methods, and prognosis when the two diseases coexisted. The aim is to deepen the clinicians\u2019 understanding of this disease for early detection, diagnosis and treatment.<\/jats:p>\n              <\/jats:sec>","DOI":"10.1186\/s12905-024-03184-y","type":"journal-article","created":{"date-parts":[[2024,6,28]],"date-time":"2024-06-28T02:01:42Z","timestamp":1719540102000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":10,"title":["When synchronous mucinous metaplasia and neoplasia of the female genital tract and peutz-jeghers syndrome meet: a case report and literature reviews"],"prefix":"10.1186","volume":"24","author":[{"given":"Yue","family":"Zhou","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xinyi","family":"Wang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yang","family":"Li","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Weiru","family":"Zhang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xiaoxuan","family":"Xu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yingxin","family":"Pang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Peishu","family":"Liu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2024,6,28]]},"reference":[{"issue":"26","key":"3184_CR1","doi-asserted-by":"publisher","first-page":"1031","DOI":"10.1056\/NEJM194912292412601","volume":"241","author":"H Jeghers","year":"1949","unstructured":"Jeghers H, Mc KV, Katz KH. Generalized intestinal polyposis and melanin spots of the oral mucosa, lips and digits; a syndrome of diagnostic significance. N Engl J Med. 1949;241(26):1031\u20136.","journal-title":"N Engl J Med"},{"issue":"6","key":"3184_CR2","doi-asserted-by":"publisher","first-page":"101042831770513","DOI":"10.1177\/1010428317705131","volume":"39","author":"HY Chen","year":"2017","unstructured":"Chen HY, Jin XW, Li BR, Zhu M, Li J, Mao GP, Zhang YF, Ning SB. Cancer risk in patients with Peutz-Jeghers syndrome: a retrospective cohort study of 336 cases. Tumour Biol. 2017;39(6):1010428317705131.","journal-title":"Tumour Biol"},{"issue":"2","key":"3184_CR3","doi-asserted-by":"publisher","first-page":"184","DOI":"10.1111\/j.1365-2559.2008.03202.x","volume":"54","author":"Y Mikami","year":"2009","unstructured":"Mikami Y, Kiyokawa T, Sasajima Y, Teramoto N, Wakasa T, Wakasa K, Hata S. Reappraisal of synchronous and multifocal mucinous lesions of the female genital tract: a close association with gastric metaplasia. Histopathology. 2009;54(2):184\u201391.","journal-title":"Histopathology"},{"issue":"1 Suppl","key":"3184_CR4","first-page":"s15","volume":"50","author":"J Costa","year":"1977","unstructured":"Costa J. Peutz-Jeghers syndrome: case presentation. Obstet Gynecol. 1977;50(1 Suppl):s15\u20137.","journal-title":"Obstet Gynecol"},{"issue":"6","key":"3184_CR5","first-page":"353","volume":"29","author":"G Berger","year":"1981","unstructured":"Berger G, Frappart L, Berger F, Seffert P, Serain F, Lamerant P, Feroldi J. [Sex cord tumor with annular tubules, mucinous metaplasia of tubal epithelium, cystic and mucinous hyperplasia of the endocervix, and the Peutz-Jeghers syndrome (author\u2019s transl)]. 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Am J Surg Pathol. 2021;45(8):1061\u201374.","journal-title":"Am J Surg Pathol"},{"issue":"03","key":"3184_CR12","first-page":"402","volume":"49","author":"\u674e\u4f73\u4f73 \u9648\u9ed8","year":"2022","unstructured":"\u9648\u9ed8 \u674e\u4f73\u4f73. \u9676\u7965, \u6731\u8fb0\u742a, \u8463\u620c\u6656, \u5c27\u826f\u6e05, \u674e\u52e4: \u5973\u6027\u751f\u6b96\u9053\u540c\u671f\u53d1\u751f\u7684\u9ecf\u6db2\u6027\u4e0a\u76ae\u5316\u751f\u548c\u80bf\u762414\u4f8b\u5206\u6790. \u590d\u65e6\u5b66\u62a5(\u533b\u5b66\u7248). 2022;49(03):402\u201310.","journal-title":"\u590d\u65e6\u5b66\u62a5(\u533b\u5b66\u7248)"},{"issue":"1","key":"3184_CR13","doi-asserted-by":"publisher","first-page":"92","DOI":"10.1177\/10668969221095264","volume":"31","author":"M Bronte Anaut","year":"2023","unstructured":"Bronte Anaut M, Arredondo Montero J, Fern\u00e1ndez Seara MP, Guarch Troyas R. Gastric-phenotype Mucinous Carcinoma of the fallopian tube with secondary ovarian involvement in a woman with Peutz-Jeghers syndrome: a Case Report. Int J Surg Pathol. 2023;31(1):92\u20137.","journal-title":"Int J Surg Pathol"},{"issue":"1","key":"3184_CR14","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1097\/PAP.0000000000000216","volume":"26","author":"EC Pirog","year":"2019","unstructured":"Pirog EC, Park KJ, Kiyokawa T, Zhang X, Chen W, Jenkins D, Quint W. Gastric-type adenocarcinoma of the Cervix: Tumor with wide range of histologic appearances. Adv Anat Pathol. 2019;26(1):1\u201312.","journal-title":"Adv Anat Pathol"},{"issue":"11","key":"3184_CR15","doi-asserted-by":"publisher","first-page":"1449","DOI":"10.1097\/PAS.0000000000000532","volume":"39","author":"YS Karamurzin","year":"2015","unstructured":"Karamurzin YS, Kiyokawa T, Parkash V, Jotwani AR, Patel P, Pike MC, Soslow RA, Park KJ. 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Am J Gastroenterol. 2015;110(2):223\u201362. quiz 263.","journal-title":"Am J Gastroenterol"}],"container-title":["BMC Women's Health"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s12905-024-03184-y.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1186\/s12905-024-03184-y\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s12905-024-03184-y.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,6,28]],"date-time":"2024-06-28T02:02:11Z","timestamp":1719540131000},"score":1,"resource":{"primary":{"URL":"https:\/\/bmcwomenshealth.biomedcentral.com\/articles\/10.1186\/s12905-024-03184-y"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,6,28]]},"references-count":18,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2024,12]]}},"alternative-id":["3184"],"URL":"https:\/\/doi.org\/10.1186\/s12905-024-03184-y","relation":{},"ISSN":["1472-6874"],"issn-type":[{"value":"1472-6874","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,6,28]]},"assertion":[{"value":"2 February 2024","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"5 June 2024","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"28 June 2024","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"This study was approved by the ethics committee of Qilu hospital, Shandong University with an ethical approval number of KYLL-202308-015.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"This study has obtained written informed consent to the publish from the patient.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare no competing interests.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"375"}}