{"paper_id":"3ae10335-7786-4e86-ba34-b3b0c327b234","body_text":"TurkiyeKlinikleriJMedSci2010;30(5)1732 \nterus-likeovarianmass(UOM)wasfirstdescribedbyCozzuttoin \n1981.1‘Endomyometrioma’,‘adenomyoma’and‘tumorofmulleri-\nantype’arerepresentativeofsomeoftheterms,reportedforUOM \nsince1981.Thislesionshowsacentralcavitylinedbyendometrialmuco-\nsasurroundedbythickmusclewall.Toourknowledge,11casesofUOM,\naged11-54yearsold,werereported .1-7 Itwasdocumentedthatfiveoutof\n11caseswerefollowedbecauseofinfertility,three hadrenalanomalies,\ntwohadbreastcancersandthreehadelevatedserumCA 125 levels(Table \n1).UOMwasalsofoundinsomeotherlocationssuchascervixuteri,broad \nUterus-LikeOvarianMasswith\nElevatedSerumLevelsof\nCA 19-9andCA 125:A CaseReportand\nReviewoftheLiterature \nAABBSS  TTRRAACCTT  Ute rus-li ke ova ri an mass is a ra re be nign en tity with con tro ver si al eti o pat ho ge ne sis. A \n37 -ye ar-old wo man, gra vi da 2, pa ra 2, pre sen ted with the comp la int of pa in in right lo wer ab do -\nmi nal qu ad rant. Physi cal eva lu a ti on re ve a led ten der ness in this are a and the ul tra so nog rap hic ex -\na mi na ti on sho wed a right ad ne xi al mass. Bi oc he mi cal blo od tests sho wed mi ni mal le u cocy to sis and \nele va ted se rum le vels of both CA 19-9: 117.79 mI U/mL (N: 0-37) and CA 125: 41.4 u/mL (N: 0-35).\nTo exc lu de a ma lig nancy, the spe ci men was exa mi ned with fro zen sec ti ons and was sub se qu ently \ndi ag no sed as en do met ri o sis. The re fo re, the sur gi cal pro ce du re was li mi ted to right sal pin go-oop -\nho rec tomy. The right ovary was 6 x 4 x 2.5 cm in si ze and the dis sec ted sur fa ce of ovary re semb led \na mi ni a tu re ute rus. His to lo gic sec ti ons as well as im mu no his toc he mistry (smo oth musc le ac tin, cal -\ndes mon and CD10) we re con sis tent with the en do met ri al mu co sal tis su e that sur ro un ded the bun -\nd les of smo oth musc le. \nKKeeyy  WWoorrddss::  En do met ri o sis; CA 19-9 an ti gen; ade nom yo ma \nÖÖZZEETT  Ute rus-ben ze ri ovar yan kit le et yo pa to ge ne zi tar tış ma lı, na dir gö rü len, be nign bir lez yon -\ndur. 37 ya şın da ka dın has ta, gra vi da 2, pa ra 2, sağ alt ab do mi nal kad ran ağ rı sı ile baş vur du. Fi zik mu -\na ye ne ile has sa si yet göz le nen ay nı böl ge de, ul tra son ile in ce le me de sağ ad nek si yel kit le sap tan dı.\nBi yo kim ya sal in ce le me ler de, mi ni mal lö ko si to zis ya nı sı ra se rum Ca19.9: 117.79 mI U/mL (N: 0-37)\nve Ca 125: 41.4 u/mL (N: 0-35) de ğer le ri yük sek li ği iz len di. Ma lig ni te dış lan ma sı için ol gu fro zen \nile in ce len di ve kit le en do met ri o zis ola rak de ğer len di ril di. Bu nun üze ri ne cer ra hi uy gu la ma sağ sal -\npin go o fe rek to mi ile sı nır lan dı rıl dı. Sağ over 6 x 4 x 2.5 cm bo yut la rın da olup ke si yü zü min ya tür\nbir ute rus gö rü nü mün dey di. His to lo jik ke sit le ri ya nı sı ra düz kas ak tin, kal des mon ve CD10’u içe -\nren im mün his to kim ya sal in ce le me le ri, düz kas de met le ri ile çev re len miş en do met ri yal mu ko za ile \nuyum lu idi. \nAAnnaahh  ttaarr  KKee  llii  mmee  lleerr::En do met ri yo zis; CA 19-9; ade nom yo ma  \nTTuurrkkiiyyee  KKlliinniikklleerrii  JJ  MMeedd  SSccii  22001100;;3300((55))::11773322--66\nCana n KELTEN, MD, a\nNagihan YALÇIN, MD, a\nŞermin ÇO BAN, MD, a\nErkan ALATAŞ, MD, b\nMetin AKBULUT, MD a\nDepartments of \naPathology \nbObstetrics and Gynecology, \nPamukkale University \nFaculty of Medicine, Denizli \nGe liş Ta ri hi/ Re ce i ved: 10.10.2008 \nKa bul Ta ri hi/ Ac cep ted: 16.03.2009 \nThis case report was presented as a poster \nat 18 th National Pathology Congress \n(25-29 October, 2008, Antalya, TURKEY). \nYa zış ma Ad re si/ Cor res pon den ce: \nCanan KELTEN, MD \nPamukkale University \nSchool of Medicine, \nDepartment of Pathology, Denizli, \nTÜRKİYE/TURKEY \nckelten@pamukkale.edu.tr  \nCop yright © 2010 by Tür ki ye Kli nik le ri \nOLGU SUNUMU   \n\nTurkiye Klinikleri J Med Sci 2010;30(5) 1733 \nMedical Pathology Kelten et al\nli ga ment, pe ri to ne um, pel vic lymph no des, scro -\ntum, small bo wel, blad der and co nus me dul la ris.8-11 \nTwo hypot he ses ha ve be en pro po sed for the ori gin \nof UOM:1-In the pre sen ce of en do met ri o sis, thro -\nugh the me tap la si a of en do met ri al stro mal cells to \nthe smo oth musc le cells, 2- In the pre sen ce of the \nuro ge ni tal ano maly, the pro ba bi lity of be ing a part\nof con ge ni tal mal for ma ti on.1-5,12,13 \nCA SE RE PORT \nA 37-ye ar-old wo man pre sen ted with ab do mi nal\npa in. The pa ti ent had al ler gic ast hma. Physi cal eva -\nlu a ti on re ve a led ten der ness in the right lo wer ab -\ndo mi nal qu ad rant. The ul tra so nog rap hic \nexa mi na ti on sho wed a right ad ne xi al mass (6 cm)\nand no ot her gyne co lo gic or uri nary ab nor ma li ti es.\nOn ab do mi nal com pu ted to mog raphy (CT), li ver,\nsple en, gall blad der and bi la te ral kid neys we re nor-\nmal in si ze and lo ca ti on. The re were no sus pi ci o us\nlympha de no pathies in the ab do men or ret ro pe ri -\nto ne al are a. Du ring bi oc he mi cal la bo ra tory eva lu -\na ti on, mi ni mal le u cocy to sis (12.1 K/uL), ele va ted \nse rum CA 19-9: 117.79 mI U/mL (N: 0-37) and \nslightly ele va ted se rum CA 125: 41.4 u/mL (N: 0-\n35) le vels we re de tec ted. Blood count, TSH, f T3, f\nT4, CE A and AFP va lu es we re wit hin the nor mal\nran ges. To exc lu de a ma lig nancy, the spe ci men was\nexa mi ned with fro zen sec ti ons and di ag no sed as\nen do met ri o sis. The re fo re ,the sur gi cal pro ce du re \nwas li mi ted to right sal pin go-oop ho rec tomy. In tra -\no pe ra ti vely, the right ovary was ob ser ved in Do ug -\nlas sac ad he red to the left ovary and rec tal se ro sa.\nThe ute rus was of nor mal si ze and ap pe a ran ce. No \nfo ci sus pi ci o us for en do met ri o sis we re fo und in the \npel vic or ab do mi nal re gi on or in anot her or gan of\nge ni tal tract. The right ovary was 6 x 4 x 2.5 cm in \nsi ze and com po sed of two ad he rent no du les. One \nof the no du les re semb led a mi ni a tu re ute rus sin ce \nthe cut sec ti on had a ca vity with he morr ha gic ap -\npe a ran ce, which was sur ro un ded with the bund les\nof smo oth musc le (Fi gu re 1). Mic ros co pic eva lu a ti -\non re ve a led a ca vity li ned with co lum nar epit he li -\num as so ci a ted with en do met ri al stro ma and \nsur ro un ded by a thick mus cu lar wall (Fi gu re 2).\nCells with aty pi a such as ple o morp hic nuc le i, hy -\nperc hro ma si a, nuc le ar ve si cu la ti on, pro mi nent nu -\nc le o lus and eo si nop hi lic cytop lasm we re ob ser ved \nin the wi de are as of sur fa ce co lum nar epit he li um \n(Fi gu re 3). He mo si de rin-la den mac rop ha ges we re \npre sent in en do met ri al stro ma, which we re sur ro -\nun ded by thick walls of smo oth musc le bund les.\nFrom the block of this tis su e, im mu no his toc he mi -\ncal sta i nings we re app li ed for CD 10 (56C6 clo ne,\n1:60 di lu ti on, Ne o mar ker Lab vi si on), smo oth mus-\nc le ac tin (1A4 clo ne, 1:100 di lu ti on, Bi o ge nex) and \ncal des mon (h-CD clo ne, 1/100 di lu ti on, Bi o ge nex)\nCase Year, Author Age Urogenital System Anomaly Breast Cancer CA 125 Other \n1 1981 , Cozutto C 32 Unilateral renal agenesis -- -\n2 1985 , Peublitz-Peredo et al. 1 8 Double excretory system, double ureter, bifid pelvis -- -\n3 1991 , Rahilly et al. 38 -- - Endometrioid cancer in \nboth ovary and endometrium \n4 1994 , Noel et al. 49 -- - Cytogenetic anomaly (+), \ncongenital anomaly (-) \n5 1997 , Mitra et al. 34 Not known -- -\n6 1998 , Pai-Sanjay et al. 39 -+ + -\n7 1998 , Pai-Sanjay et al. 43 -- + -\n8 1998 , Pai-Sanjay et al. 38 -+ - -\n9 2005 , Shutter 1 1 Resected right pelvic kidney -- -\n10 2007 , Gurel et al. 54 -- - Uterus unicornis \n11 2007 , Zannoni et al. 33 -- + Histologic atypia \n1 2 2008 , Present 37 -- + Elevated CA 19-9 and \nhistologic atypia \nTABLE 1: Clinicopathologic features in patients with uterus-like ovarian masses .\n\nTurkiye Klinikleri J Med Sci 2010;30(5)1734 \nKelten ve ark. Tıbbi Patoloji\nwith use of a stan dard la be led strep ta vi din bi o tin \npe ro xi da se met hod. Po si ti ve sta i ning with CD10 \n(con sis tent with en do met ri al stro ma), smo oth mus-\nc le ac tin and cal des mon (con sis ting with smo oth \nmusc le tis su e) con fir med the his to lo gi cal di ag no -\nsis. In one po le of this no du le, the bund les of smo -\noth musc les we re in ter ming ling with the spind le \ncells of the ova ri an stro ma of the ot her no du le,\ncom po sed of ova ri an stro ma with fol li cu lar cysts\nand he morr ha gic cor pus lu te um. No ab nor ma lity \nwas fo und in the sec ti ons of right tu bal tis su es.The \npa ti ent had be en fol lo wed for two ye ars du e to in -\nfer ti lity. Me anw hi le, the only preg nancy ac hi e ved \nthro ugh in vit ro fer ti li za ti on was di ag no sed as\n‘bligh ted ovum’, thus fi nally cu ret ta ge was app li ed \nto the pa ti ent. The re has be en no preg nancy sin ce \nthen.\nDI S CUS SI ON \nThe eti o pat ho ge ne sis of UOM is con tro ver si al.\nThe re are two ma in hypot he ses. Coz zut to sug gests\nthat me tap la si a of ova ri an stro ma in the back gro -\nund of en do met ri o sis may exp la in the ca u se of the \nle si on.1This hypot he sis has be en sup por ted by se -\nve ral ot her ca ses.3,4,13 On elec tron mic ros co pic eva l-\nu a ti on, Coz zut to ob ser ved myo fib rob lasts as well\nas smo oth musc le cells,and con se qu ently ,he sug -\nges ted that myo fib rob lasts co uld be a tran si ti o nal\ncell du ring the me tap las tic chan ges. Sup por ting this\nvi ew, Scully sug gested that pres su re of an ex pan -\nding en do met ri o tic cyst co uld trig ger a me tap la si a \nthro ugh smo oth musc le cells in the cells sur ro un -\nding the cyst.14 H e al so in di ca ted that smo oth mus-\nc le fi bers could be se en in so me of ot her ova ri an \nle si ons such as le i om yo ma, mu ci no us cysta de no ma \nand ova ri an stro mal hyperp la si a . In ca ses with ab -\nsen ce of de mons trab le en do met ri al mu co sa du e to \nbleeding or pres su re, dif fe ren ti a ti on from ova ri an \nstro mal hyperp la si a and le i om yo ma sho uld be con -\nsi dered. Alt ho ugh the pe ri od which trig gers the \nme tap la si a cannot be cle arly de ter mi ned, the re is\nso me evi den ce to sup port the ro le of ova ri an hor-\nmo nes. Bre ast can cers we re de ter mi ned in two out\nFI GU RE 1: Gross pho to of the sal pin go-oop ho rec tomy: right-top is the ute rus-li ke \nova ri an m ass, con ta i ning a ca vity sur ro un ded with the thick wall of musc les and \nright-bot tom is the ova ri an stro ma with a he morr ha gic cyst and fol li cu lar cysts. \nFI GU RE 2: Thick myom etrium -like smooth mu scle comp onent surrounds en -\ndom etrial mucosa (H&E, original magnification x40). \nFI GU RE 3: En do met ri al co lum nar cells with aty pi a, (a) Stra ti fi ca ti on, tuf ting, ple o -\nmorp hic nuc le i, nuc le ar ve si cu la ti on, pro mi nent nuc le o lus and eo si nop hi lic cytop -\nlasm we re ob ser ved (H&E, ori gi nal mag ni fi ca ti on x40), (b) in set shows si mi lar \naty pi a (H&E, ori gi nal mag ni fi ca ti on x40). \n\nTurkiye Klinikleri J Med Sci 2010;30(5) 1735 \nMedical Pathology Kelten et al\nof three ca ses re por ted by Pa i et al.4En do met ri o id \ncar ci no ma was iden ti fi ed in both ovary and ute rus\nas well as en do met ri o sis in the ca se of Ra hilly et\nal.13 Pe ter son et al. re por ted a ca se with precocious\npuberty and this po ints to the ro le of es tro gen in \nfor ma ti on of UOM.9\nThe ot her hypot he sis pro po sed by Ro sen is\nthat this le si on may be a part of uro ge ni tal con ge -\nni tal mal for ma ti on as a fu si on de fect of Mul le ri an \nduct or a re al par ti al dup li ca ti on of Mul le ri an sys-\ntem.12 The de tec ti on of re nal age ne sis in the ca se of\nCoz zut to ret ros pec ti vely and the pre sen ce of uri-\nnary ano maly in two ot her ca ses al so sup port this\nhypothesis.2,5 The hypot he sis of me tap la si a se ems\nmo re li kely in the ca ses wit ho ut any ute rus and/or\nre nal con ge ni tal ano maly.3,4,13 \nIt is known that the ca ses with en do met ri o sis\nsho uld be eva lu a ted ca re fully by the pat ho lo gists\nin terms of the pre sen ce of cyto lo gic aty pi a. Epit -\nhe li al ab nor ma li ti es (hyperp la si a or aty pi a) ha ve \nbe en con si de red as a step in the prog res si on to \nma lig nancy.15,16 En do met ri o id car ci no mas and cle -\nar cell car ci no masare the two most com mon tu -\nmors de ve lo ped in ova ri an en do met ri o sis.15,16 \nRa rely, va ri o us tu mors in the back gro und of ova ri -\nan en do met ri o sis ha ve be en re por ted such asova r-\ni an se ro us cysta de no ma of low ma lig nant po ten ti al,\nbe nign and ma lig nant mu ci no us tu mors, squ a mo us\ncell car ci no ma , gra nu lo sa cell tu mor, en do met ri o -\nid stro mal sar co ma, ma lig nant me so der mal mi xed \ntu mor and ade no sar co ma .16,17 Aty pi cal cells disp lay \neo si nop hi lic cytop lasm, lar ge hyperc hro ma tic or\npa le ple o morp hic nuc le i, in cre a sed nuc le ar to cy -\ntop las mic ra ti o, cel lu lar crow ding, stra ti fi ca ti on or\ntuf ting. Zan no ni et al. re por ted the first ca se of ova -\nri an en dom yo met ri o ma with his to lo gic aty pi a (hy -\nperc hro ma si a, ple o morp hic nuc le i, eo si nop hi lic \ncytop lasm) and fo ci of mu ci no us me tap la si a \non ca vity li ned by co lum nar epit he li um.18 In this\nstudy ,aut hors ha ve men ti o ned that the si mul ta ne -\no us pre sen ce of en do met ri al epit he li um with his to -\nlo gic aty pi a and me tap la si a sup por ted the \nme tap las tic the ory of UOM. They ha ve sug ges ted \nthe term ‘en dom yo met ri o ma with aty pi as’ for the -\nse ca ses. Si mi larly, our ca se showed epit he li al tuf t-\ning with ple o morp hic nuc le i, hyperc hro ma si a,\nnuc le ar ve si cu la ti on, pro mi nent nuc le o lus and eo -\nsi nop hi lic cytop lasm . \nH igh se rum le vels of both CA 19-9 and CA 125 \nha ve be en re por ted in so me be nign le si ons such as\nle i om yo ma, en do met ri o sis, te ra to ma of the fal lo pi -\nan tu be and tu ba-ova ri an abs ces ses.19-22 In en do -\nmet ri o sis, se rum le vel of CA 125 and CA 19-9 is\nex pec ted to be no mo re than 100 IU/ml and 1000 \nIU/ml, res pec ti vely. Ho we ver, very high se rum le -\nvels of CA 125 and CA 19-9 (CA 125: 9537 IU/ml,\nCA 19-9: 15.653 IU/ml) was re cor ded in a pa ti ent\nwith rup tu red ova ri an en do met ri o ma.20 The mec h -\na nisms that may ele va te se rum le vels of CA 125 and \nCA 19-9 in en do met ri o sis are not yet cle arly un -\nders to od; ho we ver, the dif fu si on of cyst flu id thro -\nugh the pe ri to neal sur fa ce in to the cir cu la ti on is\ncon si de red in so me ca ses.20   \nWe be li e ve that UOM was de ve lo ped du e to \nthe me tap la si a of ova ri an en do met ri o ma in our pa -\nti ent. First, no uro ge ni tal ab nor ma lities were found \non ra di o lo gic eva lu a ti ons. Se cond, high se rum le v -\nels of CA 19-9 and CA 125 we re as so ci a ted with the \npre sen ce of ova ri an en do met ri o sis. Third, the pre s-\nen ce of his to lo gic aty pi a in co lum nar epit he li um of\nen do met ri al mu co sa sup ports the me tap las tic the -\nory.  \nAAcckk  nnooww  lleedd  ggee  mmeenntt\nWe wo uld li ke to thank Ha ti ce Al tun for her as sis tan ce \nin edi ting Eng lish language .\n\nTurkiye Klinikleri J Med Sci 2010;30(5)1736 \nKelten ve ark. Tıbbi Patoloji\n1. Cozzutto C. Uterus-like mass replacing ovary: \nreport of a new entity. Arch Pathol Lab Med \n1981;105(10):508-11. \n2. Pueblitz-Peredo S, Luévano-Flores E, Rincón- \nTaracena R, Ochoa-Carrillo FJ. Uteruslike \nmass of the ovary: endomyometriosis or con -\ngenital malformation? A case with a discus -\nsion of histogenesis. Arch Pathol Lab Med \n1985;109(4):361-4. \n3. Noel JC, Hustin J, Simon P, Verhest A. \nUterus-like mass of the ovary. J Obstet Gy -\nnecol 1994;14(33):209-11. \n4. Pai SA, Desai SB, Borges AM. Uteruslike \nmasses of the ovary associated with breast \ncancer and raised serum CA 125. Am J Surg \nPathol  1998;22(3):333-7. \n5. Shutter J. Uterus-like ovarian mass present -\ning near menarche. Int J Gynecol Pathol \n2005;24(4):382-4. \n6. Gurel D, Tuna B, Yorukoglu K. Uterus-like \nmass of the ovary. Turkish J  Pathol \n2007;23(2):103-6. \n7. Mitra S, Nicol A, Scott GI. Uterus-like mass of \nthe ovary. J Obstet Gynaecol 1997;17(1):94-5. \n8. Ahmed AA, Swan RW, Owen A, Kraus FT, \nPatrick F. Uterus-like mass arising in the broad \nligament: a metaplasia or mullerian duct \nanomaly? Int J Gynecol Pathol  1997;16(3): \n279-81. \n9. Peterson CJ, Strickler JG, Gonzalez R, \nDehner LP. Uterus-like mass of the small in -\ntestine. Heterotopia or monodermal teratoma? \nAm J Surg Pathol  1990;14(4):390-4. \n10. Fukunaga M. Uterus-like mass in the uterine \ncervix: superficial cervical endometriosis with \nflorid smooth muscle metaplasia? Virchows \nArch  2001;438(3):302-5. \n11. Redman R, Wilkinson EJ, Massoll NA.  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