{"paper_id":"6699f723-c05f-45af-8024-74984ebb5182","body_text":"Gynecol Surg (2006) 3: 213 –214\nDOI 10.1007/s10397-006-0186-0\nCASE REPORT\nJ. C. Knudsen . P. Lundorff\nComplex hyperplastic endometrium in a peritoneal leiomyoma\nfollowing a CISH hysterectomy\nReceived: 27 June 2005 / Accepted: 23 December 2005 / Published online: 30 May 2006\n# Springer-V erlag Berlin / Heidelberg 2006\nAbstract We describe a case of a patient presenting with\nan abdominal tumor 4 years after a classical intrafascial\nserrated-edged macro-morcellated hysterectomy. The\ntumor was removed surgically and proved microscopically\nto be a peritoneal leiomyoma containing complex hyper-\nplastic endometria. To our knowledge, this has never been\ndescribed before. In addition, the pathogenesis of this rare\ndisease is discussed.\nKeywords Peritoneal leiomyomatosis . Hormone\nreplacement therapy . Uterine fibroids\nIntroduction\nLeiomyomatosis peritonealis disseminata is a rare disease.\nIn the past, more than 100 cases have been described [ 1, 2].\nIn the vast majority, these tumors occurred in women who\nwere pregnant or taking oral contraceptives or hormone\nreplacement therapy. The tumor is thought to arise from\nMueller’s epithelium, which is distributed throughout the\nsubperitoneal mesenchyme. Proliferation of the epithelium\nmay be stimulated by estrogen in predisposed women\n[1, 3, 4]. The disease mimics peritoneal malignancy but is\ngenerally benign. In rare cases (fewer than 10%), malignant\nleiomyosarcomas do occur [1–4]. Fewer than 10 cases have\nbeen described among postmenopausal women [ 3–6].\nMany patients, but not all, have uterine leiomyomas as\nwell. To our knowledge, this is the first case describing a\nperitoneal myoma containing a complex hyperplastic\nendometrium.\nCase report\nA 48-year-old woman was admitted by her general\npractitioner for a growing abdominal tumor. The patient\nwas para 2 with normal deliveries. Four years earlier, she\nhad undergone laparoscopic hysterectomy ad modum\nclassical intrafascial serrated-edged macro-morcellated\n(SEMM) hysterectomy (CISH) because of menorrhagia\nand uterine leiomyomas [ 7, 8]. CISH is a synthesis of three\nwell-established and widely used procedures:\n1) Supracervical amputation of the uterus\n2) Conization of the cervix\n3) Laparoscopy\nThe operation was done at our department and was\nvideotaped, confirming that no part of the uterus except the\ncollum was left in the abdomen (see Fig. 1). The\ntransformation zone and endometrium of the collum uteri\nwere removed. The remaining collum was left in situ. After\nthe operation, the patient had hormone replacement therapy\nconsisting of Femanest 2 mg daily and V agifem every 2nd\nday. The patient had been well since the operation, but after\nan intended weight loss of 10 kg, she felt a tumor in her\nlower abdomen.\nClinical and ultrasonic examinations revealed a large\ntumor in the lower part of the abdomen and in the pelvic\ncavity. Blood samples were normal except for a CA-125\nlevel of 78 kIU/l. At laparotomy a large multilobular, soft,\nsmooth tumor was found attached to adhesions around both\nadnexae and the pouch of Douglas. The adnexae, liver,\nomentum, and intestines were normal. Tumorectomy,\nappendectomy, and bilateral salpingo-oophorectomy were\nperformed. At no time during the operation was the\nremaining collum uteri touched. The tumor seemed\nmacroscopically to arise from somewhere near the right\nadnexa. Macroscopic examination revealed a 15×15×16-\ncm tumor built up from many large and small nodules\nmeasuring from 1 to 8 cm in diameter. Microscopic\nexamination showed a leiomyoma with almost no mitotic\nactivity and no cellular polymorphy but containing a very\nhyperplastic endometrium without atypia (see Fig. 2). The\nJ. C. Knudsen . P . Lundorff (*)\nDepartment of Obstetrics and Gynaecology,\nViborg County Hospital,\n8800 Viborg, Denmark\ne-mail: jc942726@hotmail.com\nTel.: +45-892-72727\n\npostoperative period was uneventful, and the patient was\ndischarged 4 days after the operation and has since been\nwell.\nDiscussion\nThe most likely explanation for the origin of this tumor is a\ncase of peritoneal leiomyomatosis. The pathogenesis of\nperitoneal leiomyomatosis is thought to be multipotential\nsubcoelomic mesenchymal cells that proliferate in response\nto estrogens. Proliferation into myoblasts, myofibroblasts,\nfibroblasts, and decidua-like cells has been described [ 9].\nAnother explanation of the development of this tumor\ncould be spillage of endometrial tissue during the CISH\nwith regrowth of myoblasts, thus simulating regeneration\nof a uterus with endometrial tissue. This explanation has\nnever been described previously and is only speculative.\nWe present a case with a peritoneal leiomyoma consisting\nof myoblasts, fibromyoblasts, and a complex hyperplastic\nendometrium without atypia. To our knowledge, this has\nnever been reported before.\nReferences\n1. Bekkers RLM, Willemsen WNP , Schijf CPT, Massuger LFAG,\nBulten J, Mercus JMWM (1999) Leiomyomatosis peritonealis\ndisseminata: does malignant transformation occur? A literature\nreview. Gynecol Oncol 75:158 –163\n2. Trabelsi S, Mrad K, Driss M, Ben Rhomdhane K (2001)\nDisseminated peritoneal leiomyomatosis: apropos of a case\nwith immunohistochemical study. Gynecol Obstet Fertil\n29:692–695\n3. Heinig J, Neff A, Cirkel U, Klockenbusch W (2003) Recurrent\nleiomyomatosis peritonealis disseminata after hysterectomy and\nbilateral salpingo-oophorectomy during combined hormone\nreplacement therapy. Eur J Obstet Gynecol Reprod Biol\n111:216–218\n4. Sharma P , Chaturvedi KU, Gupta R, Nigam S (2004)\nLeiomyomatosis peritonealis disseminata with malignant\nchange in a post-menopausal woman. Gynecol Oncol\n95:742–745\n5. Strinié T, Kuzmié-Prusac I, Eterovié D, Jakié J, Sékanec M\n(2000) Leiomyomatosis peritonealis disseminata in a post-\nmenopausal woman. Arch Gynecol Obstet 264:97 –98\n6. Rejab KE, Aradi AN, Datta BN (2000) Postmenopausal\nleiomyomatosis peritonealis disseminata. Int J Gynaecol Obstet\n68:271–272\n7. Semm K, Lehmann-Willenbrock E, Mettler L (1995) Laparo-\nscopic and other intrafascial hysterectomy techniques or\nmucosal ablation –a choice for maximum organ conservation.\nDiagn Ther Endosc 2:61 –70\n8. Vietz PF, Ahn TS (1994) A new approach to hysterectomy\nwithout colpotomy: pelviscopic intrafascial hysterectomy. Am\nJ Obstet Gynecol 170:609\n9. Morizaki A, Hayashi H, Ishikawa M (1999) Leiomyomatosis\nperitonealis disseminata with malignant transformation. Int\nJ Gynaecol Obstet 66:43 –45\nFig. 1 Classical hysterectomy technique\nFig. 2 Classical intrafascial serrated-edged macro-morcellated\nhysterectomy\n214","source_license":"CC0","license_restricted":false}