Recurrent Giant Phyllodes Tumour in a Young Female: A Case Report

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1 CASE REPORT J. Trnsl. Med. Res 2016;21(3): DOI: /jtmr Recurrent Gint Phyllodes Tumour in Young Femle: A Cse Report Mhim Koshriy 1, Ashish Shrm 1, Ajy Gehlot 1, Surhi Grg 1, M.C. Songr 1, Krn Peepre 2 Corresponding uthor: Mhim Koshriy, MS, FMAS, FIASGO Deprtment of Surgery, Gndhi Medicl College & Associted Hmidi Hospitl, Bhopl, Indi E-mil: 1 Deprtment of Surgery; 2 Deprtment of Nucler Medicine; Gndhi Medicl College & Associted Hmidi Hospitl, Bhopl, Indi ABSTRACT Phyllodes tumors (PTs) re uncommon neoplsms of the rest, constituting 0.3 to 0.9% of ll rest tumors in femles comprised of oth stroml nd epithelil elements. The term cystosrcom phyllode ws coined y Johnnes Muller, misleding term s tumors re rrely cystic nd the mjority follow enign clinicl course. The term gint phyllodes is used when the tumor size exceeds 10 cm in dimeter. WHO (World Helth Orgniztion) clssifiction hs identified three ctegories of phyllodes tumors of the rest (PT): enign, orderline nd mlignnt. The minsty of tretment of non-metsttic phyllodes tumors of the rest is complete surgicl resection with wide resection mrgins. Locl recurrence up to 50 % fter surgery hs een reported in Phyllodes tumors. We report cse of two episodes of recurrent phyllodes tumor in young femle. Key words: Phyllodes Tumors (PTs), recurrence, mstectomy INTRODUCTION Received: Accepted: Copyright Celsius Pulishing House The Phyllodes tumors (PTs) tumors of the rest re firoepithelil tumors which re rrely seen nd hve potentil for recurrence. Less thn 1 % of ll the rest tumors consist of Phyllodes tumors (1-2). The mjority of PTs occur in women etween the ge of 35 nd 55 (3), lthough there re reported cses in dolescents, s well s in elderly women (4). Phyllodes tumors re usully not considered initilly in clinicl dignosis, show slow or rpid growth pttern, nd re usully dignosed fter iopsy. These infrequent tumors re typiclly seen s firm, circulr, moile, well-defined nd pinless mss in the dimeter of 5 centimeters or more. The term gint phyllodes is used when the tumor size exceeds 10 cm in mximum dimeter (4). PTs with dimeters of 40 centimeters hve lso een reported in the literture (5). These lesions re difficult, if not impossile, to distinguish from firodenom on physicl exmintion or y rdiologic studies except when 210 Journl of Trnsltionl Medicine nd Reserch, 21 (3), 2016

2 Recurrent Gint Phyllodes Tumour in Young Femle: A Cse Report quite lrge. The tumors re chrcterized y comintion of hypercellulr strom nd cleft like or cystic spces lined y epithelium into which clssiclly projects the strom in lef-like fshion (6,7). PTs re distinguished from FA histopthologiclly y their cellulr pttern, hving incresed cellulr typicl chnges nd excessive stroml growth. WHO (World Helth Orgniztion) clssifiction hs identified three ctegories of phyllodes tumors of the rest (PT): enign, orderline nd mlignnt (8). Zurrid nd his co-uthors (9) reported tht 9%, 44% nd 33% of enign, orderline nd mlignnt PTs developed locl recurrence CASE REPORT A 32 yer old ldy presented to our deprtment with chief complint of lump in her right rest since 1 yer. Ptient hd history of similr lump in pst, 3 yers ck for which she ws operted upon, wide locl excision ws done nd histoptjology report ws consistent with phyllodes tumor, ut it ws followed y recurrence. Ptient ws gin tken for surgery 2 yers ck, this time iopsy ws indictive of Pericnliculr firodenom. Unfortuntely ptient met with second time recurrence nd ptient ws referred to us. Exmintion reveled lrge mss in the right rest mesuring 18 x 15 cms with loulted surfce. Few dilted veins were noticed on the skin surfce. The nipple ws pushed down inferolterlly. Two scr mrks of previous surgery were seen in inferomedil nd outer upper qudrnt (fig. 1, ). Contr lterl rest exmintion ws norml, nd there ws no lymphdenopthy on ilterl xillry exmintion. Mmmogrphy ws done which showed recurrence of phyllode like growth in right rest with incresed intrlesionl vsculrity. USG rest showed lrge multiloulted mss lesion completely replcing right rest with very less firoftty glndulr tissue, there ws no significnt xillry lymphdenopthy noted. USG domen ws found to e norml. SPECT CT-Thorx nd domen (fig. 2, ) showed mss lesion of 15X 12 cm with incresed trcer uptke, no evidence of xillry or pectorl lymph node enlrgement. There ws no evidence of liver metstsis. Bone scn (fig. 3) ws found to e norml. FNAC done from the lump confirmed the dignosis of phyllodes tumor. The ptient underwent surgicl mngement. A simple mstectomy ws performed with primry closure of the skin, without ny skin grft or flp reconstruction. She hd n uneventful post- opertive course Figure 1 - () Gross ppernce (nterior view) showing lrge mss in right rest involving whole of the rest. The nipple ws pushed down inferolterlly. Two scr mrks of previous surgery were seen in inferomedil nd outer upper qudrnt () Gross ppernce (lterl view) showing lrge mss in the right rest mesuring 18 X 15 cms with loulted surfce. Few dilted veins were lso noticed nd ws dischrged on 8 th postopertive dy. Cut section of mstectomy specimen reveled ovl grey white tissue mesuring 18 x 15 x 10 cms. Histologicl sections (fig. 6) reveled rest tissue showing slit like ducts with epithelil hyperplsi with cellulr spindle strom, no typi mitosis or necrosis ws seen in strom, no srcomtous res seen, picture ws suggestive of Benign Phyllodes tumor. Reconstructive rest surgery ws offered to the ptient ut the ptient refused surgery proly ecuse there ws history of two times recurrence. Ptient ws kept in regulr follow up nd fter 1 yer of surgery there ws no evidence of locl recurrence. DISCUSSION Tumors of mixed connective tissue nd epithelium constitute n importnt group of unusul primry rest tumors. Phyllodes tumors contin iphsic prolifertion of mmmry epithelium nd strom, the Journl of Trnsltionl Medicine nd Reserch, 21 (3),

3 Mhim Koshriy et l Figure 2 - () SPECT CT- (Trnsxil view) showed mss lesion of 15X 12 cm with incresed trcer uptke, no evidence of xillry or pectorl lymph node enlrgement. There ws no evidence of liver metstsis. () SPECT CT- (Coronl View) Figure 3 - Bone Scn- No norml uptke in skeleton reflects norml one scn ltter determines the mlignncy. Initilly clled cystosrcom phylloides, misleding description s tumors re rrely cystic nd the mjority follow enign clinicl course. With incresing cellulrity, invsive mrgin nd srcomtous ppernce, these tumors my e clssified s mlignnt phyllodes Figure 4 - () Intr-op picture showing dissection of tumor from chest wll. (). Intr-op picture showing seprtion of tumor from chest wll 212 Journl of Trnsltionl Medicine nd Reserch, 21 (3), 2016

4 Recurrent Gint Phyllodes Tumour in Young Femle: A Cse Report Figure 5 - Showing primry skin closure tumors. Histologiclly, these tumors re similr to firodenoms, ut the whorled strom forms lrger clefts lined y epithelium tht resemle clusters of lef like structures (10). The mjority of PTs occur in women etween the ge of 35 nd 55 (3). Ptients usully presents with complints of firm round mcroloulted mss which is pinless in nture. Lrge lesions my e ssocited with dilted veins visile over the skin, which my e stretched nd ttenuted. Rre findings which hve een reported re Nipple retrction (4, 11), skin ulcertion invsion of the chest wll (4, 12, 13), nd loody nipple dischrge (13). Axillry lymph nodes re usully not involved however rectionry inflmmtory lymph nodes re seen in out 20% cses (14) Surgery remins the minsty of tretment however the extent of surgicl pproch historiclly hs een controversil, nd continues to evolve. Complete surgicl resection with wide resection mrgins (t lest 1 cm) should e performed, except for lesions lrger thn 10 cm in which totl mstectomy is recommended. If the initil surgery fils to chieve negtive mrgin, it is recommended tht the ptient should undergo reexcision to otin wider mrgin in order to prevent locl recurrence. Mstectomy is dvocted for lrge tumors for microscopic mrgin of one centimeter without significnt deformtion of the rest, or recurrent tumors despite dequte mrgins (16). Axillry dissection is not routinely recommend s metstsis to the lymph nodes occur in less thn 5% of ptients Mstectomy ws the preferred tretment of choice in our cse s the tumor ws two times recurrent hving very lrge size covering lmost whole of the rest. Mstectomy hs een the most commonly performed surgery for orderline nd mlignnt PTs Figure 6 - () Resected specimen (gross ppernce) showing lrge mss with nodulr surfce nd dilted veins. () Cut section showing multiloulted surfce Figure 7 - Histopthologicl picture showing slit like ducts with epithelil hyperplsi with cellulr spindle strom, no typi mitosis or necrosis seen in strom, suggestive of enign phyllodes tumor Journl of Trnsltionl Medicine nd Reserch, 21 (3),

5 Mhim Koshriy et l with locl recurrence rtes of 5% nd 12%; respectively (15,16,17). Locl recurrence is usully seen within the first few yers of surgery nd histologiclly resemles the originl tumor. Occsionlly, recurrent tumors show incresed cellulrity nd more ggressive histologicl fetures thn the originl lesion. In most ptients, locl recurrence is isolted nd is not ssocited with the development of distnt metstsis (18). Importnt risk fctors predicting recurrence is positive mrgin, young ge t presenttion, lrge tumor size, histologicl type. Other prmeters such s stroml hypercellulrity, stroml typi, stroml overgrowth, nd mitosis, were significntly ssocited with locl recurrence (19). The most importnt risk fctor for recurrence is the resection done within 1-2 cm negtive surgicl mrgins (20). It is importnt tht these fctors e identified nd pproprite surgicl procedure e selected to ensure cler mrgins. Rdiotherpy hs role in reducing locl recurrence in orderline nd mlignnt phylloides ut hs no impct on over ll survivl nd disese free survivl (21). CONCLUSION Phyllodes tumor re rre tumors of rest ut re notorious for locl recurrence. Wide locl excision is n cceptle method of tretment ut for recurrent nd lrge tumors (>10 cm) simple mstectomy is preferred for complete tumor excision. Most importnt fctor to prevent locl recurrence is to keep sufficient mrgin of helthy tissue. It is necessry tht the ptients e kept in follow up s there is risk of locl recurrence nd distnt metstsis. REFERENCES 1. Azzoprdi JG. Srcom in the rest. In Benningron J (ed). Prolems in Brest Pthology. Mjor Prolems in Pthology. Vol 11, Phildelphi: WB Sunders Co; p Rowell MD, Perry RR, Hsiu JG, Brrnco SC. Phlyllodes tumors. Am J Surg Mr;165(3): Slvdori B, Cusumno F, Del-Bo R, Delledonne V, Grssi M, Rovini D, et l. Surgicl tretment of phyllodes tumors of the rest. Cncer Jun 15;63(12): Reinfuss M, Mituś J, Dud K, Stelmch A, Ryś J, Smolk K. The tretment nd prognosis of ptients with phyllodes tumour of the rest: n nlysis of 170 cses. Cncer Mr 1;77(5): Ling MI, Rmswmy B, Ptterson CC, McKelvey MT, Gordillo G, Nuovo GJ, et l. Gint rest tumors: Surgicl mn gement of phyllodes tumors, potentil for reconstructive sur gery nd review of literture. World J Surg Oncol. 2008;11: Pietrusuzk M, Brnes L. Cystosrcom phyllodes: clinicopthologic nlysis of 42 cses. Cncer My;41(5): Mofft CJ, Pinder SE, Dixon AR, Elston CW, Blmey RW, Ellis IO. Phyllodes tumours of the rest: clinicopthologicl review of thirty-two cses. Histopthology Sep;27(3): World Helth Orgniztion. Histologicl typing of rest tumors. Tumori Jun 30;68(3): Zurrid S, Brtoli C, Glimerti V, Squiccirini P, Delledonne V, Veronesi P. Which therpy for unexpected phyllode tumour of the rest? Eur J Cncer. 1992;28(2-3): Kelly K. Hunt, Mrjorie C. Green, nd Thoms A. Buchholz. Diseses of the rest. In Siston textook of surgery, Nineteenth ed USA: Elsevier-Sunders; p Deodhr SD, Joshi S, Khuchndni S. Cystosrcom phyllodes. J Postgrd Med Apr;35(2): Dyer NH, Bridger JE, Tylor RS. Cystosrcom phylloides. Br J Surg My;53(5): Chen WH, Cheng SP, Tzen CY, Yng TL, Jeng KS, Liu CL, et l. Surgicl tretment of phyllodes tumors of the rest: retrospective review of 172 cses. J Surg Oncol Sep 1;91(3): Rowell MD, Perry RR, Hsiu JG, Brrnco SC. Phyllodes tumors. Am J Surg Mr;165(3): Treves N, Sunderlnd DA. Cystosrcom phyllodes of the rest: mlignnt nd enign tumor; clinicopthologicl study of seventyseven cses. Cncer Nov;4(6): Asoglu O, Mustf M, Blnchrd K, Grnt CS, Reynolds C, Ch SC, et l. Risk fctors for recurrence nd deth fter primry surgicl tretment of mlignnt phyllodes tumors. Ann Surg Oncol Nov; 11(11): Brth RJ Jr. Histologic fetures predict locl recurrence fter rest conserving therpy of phyllodes tumors. Brest Cncer Res Tret Oct;57(3): Ahmd Z, Koshriy M, Shukl S, Vtti V, Diwn A. A rre cse of recurrent mlignnt phyllodes tumor of the rest in young nulliprous womn. Clin Cncer Investig J. 2014;3: Wei J, Tn YT, Ci YC, Yun ZY, Yng D, Wng SS, et l. Predictive fctors for the locl recurrence nd distnt metstsis of phyllodes tumors of the rest: retrospective nlysis of 192 cses t single center. Chin J Cncer Oct;33(10): doi: / cjc Epu 2014 Aug Lenhrd MS, Khlert S, Himsl I, Ditsch N, Untch M, Buerfeind I. Phyllodes tumour of the rest: clinicl follow-up of 33 cses of this rre disese. Eur J Ostet Gynecol Reprod Biol Jun;138(2): Epu 2007 Sep Zeng S, Zhng X, Yng D, Wng X, Ren G. Effects of djuvnt rdiotherpy on orderline nd mlignnt phyllodes tumors: systemtic review nd met nlysis. Mol Clin Oncol My;3(3): Epu 2015 Fe Journl of Trnsltionl Medicine nd Reserch, 21 (3), 2016

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