{"paper_id":"d76775e9-e2c4-4219-bad5-a87998bc1f8a","body_text":"Gynecol Surg (2007) 4: 191 –193\nDOI 10.1007/s10397-006-0252-7\nCASE REPORT\nSalvatore Lanzafame . Antonio Giuseppe Nicolosi .\nRosario Caltabiano\nBilateral massive osseous metaplasia in ovaries:\n“ovarian stones ”\nReceived: 14 July 2006 / Accepted: 22 August 2006 / Published online: 3 October 2006\n# Springer-V erlag 2006\nAbstract We report a case of bilateral ovarian ossifications\nwith images that mimic stone formations. A 65-year-old\nwoman presented with a 2-year history of pelvic pain.\nComputed tomographic scan and pelvic ultrasound identi-\nfied an enlarged uterus with two calcified lesions interpreted\nas leiomyomas. Surgical exploration revealed two ovaries\nwith a cystic appearance and stony hard areas. The cyst\ncontents consisted of chocolate-colored material. The\npathologic findings were compatible with benign bilateral\novarian endometriotic cysts with extensive ossification. The\npelvic pain resolved completely after the surgery. Though\nthe cause of this rare case remains unknown, recognition of\ncysts with a content of chocolate-colored material and\npigment-laden histiocytes allowed us to make the diagnosis\nof bilateral ovarian endometriotic cysts with extensive\nossification. Complete excision was the treatment of choice.\nKeywords Ovary . Ossification . Endometriotic cyst\nBilateral massive osseous metaplasia in ovaries:\n“ovarian stones ”\nIn the absence of an ovarian neoplasm, extensive ossifi-\ncation and calcification involving the whole ovary is an\nunusual occurrence. It may develop within periovarian\nadhesions or the walls of endometriotic cysts and rarely\nwithin otherwise normal ovaries [ 1]. We describe a rare\ncase of bilateral massive ossification in ovaries with a\ncystic appearance.\nCase report\nA 65-year-old woman, with a previous history of chole-\ncystectomy, presented with a 2-year history of pelvic pain\nleading to the preoperative diagnosis of two highly\ncalcified uterine masses interpreted as leiomyomas on\ncomputed tomographic (CT) scan (Fig. 1). Also pelvic\nultrasound confirmed an enlarged uterus with two calcified\nlesions. Laboratory investigations, including serum calci-\num levels, were in the range of normality. Unexpectedly,\nsurgical exploration revealed bilateral enlarged, ovoid, pale\nbrown ovaries, closely connected with the posterior surface\nof the uterus and looking like ovaries made of stones.\nTherefore, the patient underwent a hysterectomy and\nbilateral salpingo-oophorectomy. On gross examination\nthe left ovary measured 5.5×3.5×2 cm and the right ovary\nmeasured 2.5×2×1.3 cm. The cut surface revealed in both\novaries a cyst with stony hard areas, a diffusely gritty\ntexture, and a yellow surface (Fig. 2). The cyst contents\nconsisted of chocolate-colored material. Both ovaries,\nwhich required 2 days of decalcification, were entirely\nembedded for histologic examination. Contrary to the\npreoperative CT scan and pelvic ultrasound diagnosis, both\non gross and histological examination, the uterus and the\nfallopian tubes were unremarkable. Instead, histological\nexamination revealed in both ovaries a cyst without\nepithelial lining cells, but surrounded by dense fibrous\ntissue, a diffuse infiltration of pigment-laden histiocytes\n(Fig. 3), and extensive calcification with areas of meta-\nplastic ossification (Fig. 4). Osteoblasts and osteoclasts\nsurrounded the surface of the heterotopic bone. Haversian\ncanals were occasionally identified in the bony trabeculae.\nMany corpora albicantia were also evident in the ovarian\nstroma (Fig. 5). The ovarian surface was characterized by\nfibrous tissue and inflammatory cells that justified the\nclose connection with the posterior surface of the uterus.\nIn conclusion the morphological aspect was highly\nsuggestive, but not conclusive, for the diagnosis of\nbilateral ovarian endometriotic cysts with extensive ossi-\nfication. The pelvic pain resolved completely after the\nsurgery.\nS. Lanzafame ( *) . R. Caltabiano\nDepartment G.F. Ingrassia, Section of Anatomic Pathology,\nUniversity of Catania,\nSanta Sofia 87 Street,\n95123 Catania, Italy\ne-mail: lanzafas@unict.it\nFax: +39-095-3782023\nA. G. Nicolosi\nU.O. Casa di Cura Basile, Oncologic Surgery,\nCatania, Italy\n\nDiscussion\nFocal calcifications are quite common in neoplastic and\nnon-neoplastic diseases of the ovary. They are usually\nassociated with serous tumor, mucinous tumor, dermoid\ntumor, fibroma, thecoma, and gonadoblastoma. Other\novarian non-neoplastic diseases such as torsion, infarction,\nand hypoplasia may be associated with ovarian calcifica-\ntion [ 2]. On the other hand, massive ovarian calcification,\ndiscernible at gross examination, is rare. From the review\nof the literature we found only one case of extensive,\nbilateral, multifocal calcification of the ovarian stroma with\nno apparent cause [ 3]. Microscopic examination showed,\nin fact, numerous spherical, laminated, calcific foci without\naccompanying cells. In cases like this one, occasional\nneoplastic cells must be sought in order to exclude tumors\nsuch as a serous borderline or malignant tumor with\nconfluent psammoma bodies [ 4] and a gonadoblastoma\nreplaced by laminated calcified masses, but with evidence\nof abnormal gonadal development. Also ossification of an\novary is extremely rare and usually associated with ovarian\ncystic teratoma [ 5], mucinous cystadenoma [ 5], papillary\nserous cystadenocarcinoma [ 6], endometrioid adenocarci-\nnoma [ 7], thecoma [ 8], and endometriosis [ 9]. In fact, a\ncommon manifestation of ovarian endometriosis is cysti-\nfication resulting in an endometriotic cyst. In many\ncircumstances the epithelial lining becomes attenuated\nand recognition of an endometrial cyst may only be\npossible if a rim of subjacent endometrial stroma persists.\nCommonly in old endometrial cysts, the lining of the\nendometrial epithelium and stroma is totally lost and\nreplaced by granulation tissue, dense fibrous tissue, and\nvariable amounts of pigment-laden histiocytes. In those\ncases, ossification and calcification may be observed. In\nour case, recognition of bilateral cysts without epithelial\nlining cells, but with a content of chocolate-colored\nmaterial, surrounded by dense fibrous tissue, a diffuse\ninfiltration of pigment-laden histiocytes, and extensive\ncalcification with areas of metaplastic bone, suggested the\ndiagnosis of bilateral ovarian endometriotic cysts with\nextensive ossification. In the literature there is also a case of\nosseous metaplasia in a benign ovarian cyst in association\nwith a complex urogenital malformation. In this case,\nhistological examination revealed a tubal cyst with chronic\nsalpingitis and a simple follicular ovarian cyst, in the wall\nof which osseous metaplasia was noted [ 10]. All those\ncases raise the question about the pathogenesis of osseous\nmetaplasia in neoplastic and non-neoplastic diseases of the\novary. V arious hypotheses have been suggested for this\nphenomenon; in some circumstances, such as torsion and\ninfarction of the ovary, it appears to be an unusual reaction\nFig. 1 Two highly calcified masses interpreted as uterine leiomyo-\nmas on computed tomographic scan\nFig. 2 On gross examination\nboth ovaries revealed a cyst with\nstony hard areas, a diffusely\ngritty texture, and a yellow\nsurface\nFig. 3 Diffuse infiltration of pigment-laden histiocytes (E.E., ×10)\n192\n\nto tissue damage and repair. The overgrowth of coexisting\ncoalesced psammomatous calcifications is another plau-\nsible explanation, but not all diseases of the ovary have this\ntype of calcification [ 11]. The histogenesis of psammoma\nbodies is also not very clear. Some authors believe they\nmay arise from spontaneous or induced necrosis of the\ntissue; for others they are related to secretion of a family of\ngrowth factors, called bone morphogenetic proteins. These\nfactors may induce osseous metaplasia of multipotential\nstromal stem cells with psammoma body and bone\nformation in ovarian cancer [ 7].\nIn conclusion, our case of bilateral ovarian ossifications\nis interesting because even if a correlation with neoplastic\ndiseases does not exist, also non-neoplastic diseases of the\novary are difficult to identify because we could not find any\nepithelial lining cells in the luminal cysts. Nevertheless,\nrecognition of cysts with a content of chocolate-colored\nmaterial and pigment-laden histiocytes allowed us to make\nthe diagnosis of bilateral ovarian endometriotic cysts with\nextensive ossification. The calcification was not related to\nhypercalcemia because serum calcium levels were in the\nrange of normality. Also a diagnosis of uterine leiomyomas\nwas made based on the computed tomographic scan and\npelvic ultrasound, and only surgical intervention with gross\nand histological examination supplied the right diagnosis.\nSurgical intervention is the treatment of choice in cases of\nadnexal masses presenting with extensive calcification and/\nor ossification.\nReferences\n1. Su WH, Wang PH, Chang SP (2002) Ovarian stone. A case\nreport. J Reprod Med 47:329 –331\n2. Okada S, Ohaki Y , Inoue K, Kawamura T, Hayashi T, Kato T,\nKumazaki T (2005) Calcifications in mucinous and serous\ncystic ovarian tumors. J Nippon Med Sch 72:29 –33\n3. Clement PB, Cooney TP (1992) Idiopathic multifocal calcifi-\ncation of the ovarian stroma. Arch Pathol Lab Med 116:\n204–205\n4. Vimplis S, Williamson KM, Chaudry Z, Nuuns D (2006)\nPsammocarcinoma of the ovary: a case report and review of the\nliterature. Gynecol Surg 3:55 –57\n5. Zahn CM, Kendall BS (2001) Heterotopic bone in the ovary\nassociated with a mucinous cystadenoma. Mil Med 166:\n915–917\n6. Bosscher J, Barnhill D, O ’Connor D, Doering D, Nash J, Park\nR (1990) Osseous metaplasia in ovarian papillary serous\ncystadenocarcinoma. Gynecol Oncol 39:228 –231\n7. Mukonoweshuro P , Oriowolo A (2005) Stromal osseous\nmetaplasia in a low-grade ovarian adenocarcinoma. Gynecol\nOncol 99:222 –224\n8. Morizane M, Ohara N, Mori T, Murao S (2003) Ossifying\nluteinized thecoma of the ovary. Arch Gynecol Obstet\n267:167–169\n9. Badawy SZ, Kasello DJ, Powers C, Elia G, Wojtowycz AR\n(1995) Supernumerary ovary with an endometrioma and\nosseous metaplasia: a case report. Am J Obstet Gynecol 173:\n1623–1624\n10. Godbole P , Outram A, Sebire N (2005) Osseous metaplasia in a\nbenign ovarian cyst in association with cloacal anomaly. J Clin\nPathol 58:334 –335\n11. Barua R, Cox LW (1982) Occurrence of bone in serous\ncystadenocarcinoma of the ovary. Aust N Z J Obstet Gynaecol\n22:183–186\nFig. 5 Coexistence of corpus albicans ( right) and calcification ( left)\n(E.E., ×10)\nFig. 4 Extensive calcification with areas of metaplastic ossification\n(E.E., ×10)\n193","source_license":"CC0","license_restricted":false}