Lu et l. Journl of Medicl Cse Reports (2016) 10:221 DOI 10.1186/s13256-016-1018-0 CASE REPORT Bitril thromi resemling myxom regressed fter prolonged nticogultion in ptient with mitrl stenosis: cse report Open Access Hou Tee Lu 1,3*, Rusli Nordin 1, Norliz Othmn 2, Chun Ngok Choy 3, Ji Yen Km 3, Benjmin Cheng-Leng Leo 3, Gunsekrn Rmsmy 3 nd Teck Hw Goh 4 Astrct Bckground: Mny cses of crdic msses hve een reported in the literture, ut in this cse report we descried rre cse of itril crdic mss tht represented chllenge for dignosis nd therpy. The differentition etween crdic msses such s thromi, vegettions, myxoms nd other tumors is not lwys strightforwrd nd n exct dignosis is importnt ecuse of its distinct tretment strtegy. Trnsthorcic/ esophgel echocrdiogrphy nd crdic mgnetic resonnce ply n importnt role in estlishing the dignosis of crdic msses. However, no current noninvsive dignostic tool hs the ility to solutely dignose crdic msses; otining pthologicl specimen y surgicl resection of crdic msses is the only relile method to dignose crdic msses ccurtely. Our cse report is n exception in tht the finl dignosis ws ffirmed y empiricl nticogultion therpy sed on clinicl judgment nd noninvsive chrcteriztion of itril mss. Cse presenttion: We descried 54-yer-old Mly mn with severe mitrl stenosis nd tril firilltion who presented with itril mss. Trnsthorcic/esophgel echocrdiogrphy nd crdic mgnetic resonnce detected lrge, homogeneous right tril mss typicl of thromus, nd left tril mss dhering to intertril septum tht mimicked tril myxom. The risk fctors, morphology, loction, nd chrcteristics of the itril crdic mss indicted dignosis of thromi. However, our ptient declined surgery. As result, the nture of his crdic msses ws not specified y histology. Of note, his left tril mss ws completely regressed y long-term wrfrin, leving residul right tril mss. Thus, we ffirmed the most prole dignosis of crdic thromi. During the course of tretment, he hd n episode of non-ftl ischemic stroke most proly ecuse of thromoemolism. Conclusions: Noninvsive chrcteriztion of crdic mss is essentil in clrifying the dignosis nd directing tretment strtegy. Anticogultion is fesile tretment when the clinicl ssessment, risk fctors, nd imging findings indicte dignosis of thromi. After prolonged nticogultion therpy, complete resolution of itril thromi ws chievle in our cse. Keywords: Crdic mss, Thromus, Myxom, Atril firilltion, Mitrl stenosis, Echocrdiogrphy, Crdic mgnetic resonnce * Correspondence: luhoutee@gmil.com; lu.hou.tee@monsh.edu 1 Clinicl School Johor Bhru, Jeffrey Cheh School of Medicine nd Helth Sciences, Monsh University Mlysi, 8 Jln Msjid Au Bkr, 80100 Johor Bhru, Johor, Mlysi 3 Deprtment of Crdiology, Sultnh Aminh Hospitl, Jln Au Bkr, 80100 Johor Bhru, Johor, Mlysi Full list of uthor informtion is ville t the end of the rticle 2016 The Author(s). Open Access This rticle is distriuted under the terms of the Cretive Commons Attriution 4.0 Interntionl License (http://cretivecommons.org/licenses/y/4.0/), which permits unrestricted use, distriution, nd reproduction in ny medium, provided you give pproprite credit to the originl uthor(s) nd the source, provide link to the Cretive Commons license, nd indicte if chnges were mde. The Cretive Commons Pulic Domin Dediction wiver (http://cretivecommons.org/pulicdomin/zero/1.0/) pplies to the dt mde ville in this rticle, unless otherwise stted.
Lu et l. Journl of Medicl Cse Reports (2016) 10:221 Pge 2 of 5 Bckground The differentition etween crdic msses such s thromi, vegettion, myxoms, nd other tumors is not lwys strightforwrd nd n exct dignosis is importnt ecuse of its distinct tretment strtegy [1]. Trnsthorcic echocrdiogrphy (TTE) nd trnsesophgel echocrdiogrphy (TEE) ply n importnt role in estlishing dignosis of crdic msses [2]. Crdic mgnetic resonnce (CMR) offers potentil dvntges nd is complementry to echocrdiogrphy in the evlution of crdic msses [3, 4]. However, no current noninvsive dignostic tool hs the ility to solutely dignose crdic msses; otining the pthologicl specimen y surgicl resection of crdic msses is the only relile method to dignose crdic msses ccurtely [5 7]. Cse presenttion In Jnury 2011, 54-yer-old Mly mn ws referred to our hospitl for evlution of plpittion. On exmintion, his pulse rte ws 85/minute with irregulr rhythm nd his lood pressure ws 120/80 mmhg. He ws ferile. A loud first hert sound nd soft middistolic rumling murmur (grde 2/6 degrees) were uscultted t mitrl re with no signs of hert filure. Electrocrdiogrphy reveled tril firilltion (AF). The lortory results showed norml hemogloin, white cells, nd pltelet counts. Renl test, liver function test, nd other lortory test results were unremrkle. His urine nlysis ws lso norml. TTE nd TEE reveled severe mitrl stenosis (MS). His mitrl vlve ws thickened nd modertely clcified, nd his nterior mitrl vlve leflet ws dooming during distole suggestive of rheumtic origin. His mitrl vlve re, which ws estimted y two-dimensionl plnimetry nd pressure hlf-time method, ws 0.92 cm 2 nd 0.91 cm 2, respectively. His pek nd men mitrl vlve grdients were 14 nd 9 mmhg, respectively. His left trium () nd right trium () were dilted. In his, we found lrge, moile, homogeneous round mss mesuring 40 35 mm. A lrge mss with similr echogenicity to his mss ws found dhering to intertril septum nd protruding into the left ventricle with crdic motion (Fig. 1). We could not identify ny stlk ttched to the mss y TTE nd TEE exmintions. In ddition, there were no identifile msses in the left tril ppendge. Spontneous echocrdiogrphic contrst (SEC) ws oserved in his nd left ventricle. Mild tricuspid regurgittion nd mild pulmonry hypertension were identified with pek pulmonry systolic pressure of 26 mmhg (estimtion sed on pek tricuspid regurgittion jet velocity of 210 cm/second nd n estimted right tril pressure of 5 mmhg). His inferior ven cv ws not dilted nd collpsile with respirtion, nd there ws no thromus. His ejection frction ws estimted s 45 %. Ptients with severe MS nd AF re t high risk of developing intrcrdic thromi. Bsed on the findings y TTE, TEE, nd the presence of risk fctors (AF), his mss ws likely to e thromus s descried erlier. Similr fetures were found in mss nd it ws thought to e thromus s well. However, the chrcteristic of mss dhering to tril septum mimics tril myxom, which is the most common enign crdic tumor [1]. The shpe, moility, nd loction of the mss mde it difficult to rule out tril myxom with solute certinty. Therefore, our ptient underwent CMR for dditionl noninvsive chrcteriztion of the itril mss. CMR showed gint right tril mss mesuring 5.3 3.2 3.9 cm nd lrge left tril lesion mesuring 5.0 2.4 5.1 cm dhering to intertril septum (Fig. 2). Both lesions were intermedite in signl on cine CMR, nd not enhnced during erly nd delyed enhncement CMR. The morphology, loction, nd vsculr chrcteristics mde thromi the most likely dignosis. The CMR result ws in greement with TTE nd TEE findings. Bsed on his history, physicl exmintions nd imging findings, we recommended thromectomy nd mitrl vlve replcement. Despite medicl dvice, our ptient declined surgery. We empiriclly initited heprin nd wrfrin dministered intrvenously to prevent thromoemolism. His interntionl normlized rtio (INR) ws trgeted t 2.5 to 3.5. Bisoprolol ws prescried for AF rte control. However, his clinicl course ws complicted y his non-dherence to wrfrin. His INR ws sutherpeutic (medin=1.8). After 2 yers of wrfrin (prtil complince), seril TTE showed no regression of nd msses. After 3 yers of uneventful follow-up, he experienced n episode of trnsient ischemic ttck in Ferury 2014. Susequently, in Decemer 2014, he hd n episode of non-ftl ischemic stroke mnifested y right hemipresis (motor power reduced to 3/5) most proly ecuse of thromoemolism. We counseled him with regrd to the dnger of the thromoemolism nd re-emphsized the need for tretment complince. Fortuntely, he resumed wrfrin nd did not demonstrte further thromoemolic events. In Mrch 2016 (5 yers fter the detection of itril msses), follow-up TTE documented complete regression of the mss nd ner-complete resolution of the mss (Fig. 3). Thus, the most prole dignosis of crdic thromi ws ffirmed. He remins well on medicl tretment. Discussion Mny cses of crdic msses hve een reported in the literture, ut in this cse report we descried rre cse of itril crdic mss tht represented chllenge for dignosis nd therpy. In our ptient, the chrcteristic of left
Lu et l. Journl of Medicl Cse Reports (2016) 10:221 Pge 3 of 5 c d Fig. 1 Imges otined y trnsthorcic echocrdiogrphy nd trnsesophgel echocrdiogrphy., Trnsthorcic echocrdiogrphy imges. c, d Trnsesophgel echocrdiogrphy imges. A lrge left tril mss (rrows) dhering to intertril septum in prsternl long xis () nd picl four chmer view (, d) nd gint right tril mss (notched rrows) ws seen in picl four chmer () nd icvl view (c). left trium, left ventricle, right trium, right ventricle tril mss dhering to the tril septum posed dignostic chllenge in differentiting etween thromus nd myxom, the most commonly reported crdic msses. The following discussion will review cses of itril msses in ptients with MS reported etween 2008 nd 2016 (Tle 1). First, 40-yer-old womn with rheumtic MS ws found to hve itril thromi mimicking myxom, nd she underwent successful thromectomy nd vlve replcement [8]. Second, 77-yer-old mn with AF, severe MS nd hert filure presented with dyspne. TEE nd TTE reveled itril thromi confirmed y pthologicl exmintion following thromectomy nd mitrl vlve replcement [9]. Third, 58-yer-old womn presented with cute lim ischemi; she ws found to hve moile itril thromi, AF, nd MS nd underwent successful emolectomy, thromectomy, nd mitrl vlve replcement [10]. The fetures of itril thromi, MS, nd AF were common to ll three ptients. In ddition, cse of itril myxom with mild MS presented with cererl ischemi ws successfully treted with thromolytic therpy dministered intrvenously nd surgicl resection [11]. However, numerous cses of crdic mss with or without mitrl vlve disese with dignostic difficulties hve een reported erlier. Overll, crdic thromi were frequently reported [5, 7, 10, 12, 13]. In some instnces crdic thromi mimicked tril myxom [5, 7]. By contrst, tril myxom cn simulte thromus in the setting of MS [14]. Of interest, thromus could form on top of myxom. In one cse report of ptient with MS, the left tril mss showed fetures of thromus Fig. 2 Crdic mgnetic resonnce. Cine four chmer view: white lood grdient echo () nd lck lood imging () on erly gdolinium imges showed low signl floting lesion in the right trium nd left tril lesion dhering to the intertril septum (white tringles). left trium, left ventricle, right trium, right ventricle
Lu et l. Journl of Medicl Cse Reports (2016) 10:221 Pge 4 of 5 Fig. 3 Outcome fter long-term nticogultion therpy. The left tril thromus is completely resolved s viewed from prsternl long xis () nd picl four chmer view () in the left trium. A residul right tril thromus (notched rrow) is still visile in the right trium (). left trium, left ventricle, right trium, right ventricle chrcterized y echocrdiogrphy nd CMR. However, histopthologicl evlution of the left tril mss removed during surgery reveled mssive thromus formed on top of very smll pre-existing left tril myxom [12]. To complicte mtters further, tril thromus my hve stlk [6] or neovsculriztion [13] mimicking tril myxom, potentilly leding to dely in nticogultion therpy. In nother cse report, itril intrcrdic msses were detected y three-dimensionl TEE in n 80-yer-old womn with hert filure, mitrl vlve repir, dul chmer permnent pcemker implnttion, nd AF. Although direct pthologicl specimens were not otined, the reduction in the size of oth msses fter intensive nticogultion tretment rises the strong possiility tht oth msses were thromi [15]. This cse report highlighted the fcts tht nticogultion is fesile tretment in regressing thromi when surgery is reltively contrindicted in n older ptient with comoridities. TEE is superior to TTE in delineting nd chrcterizing crdic msses [2]. CMR provides high sptil resolution imges, improves tissue chrcteriztion nd is complementry to echocrdiogrphy in the ssessment of crdic msses [3]. Common CMR sequences re cine imge, T1-weighted nd T2-weighted spin echo, contrst first pss perfusion, nd stndrd dely enhncement. However, tumors nd chronic orgnized thromi cnnot e distinguished from one nother using the morphology, motility, nd enhncement ptterns y CMR. A pttern of hyperintensity/isointensity (compred with norml myocrdium) with short T1, nd hypointensity with long T1, ws very frequent in thromi, rre in tumors, nd hd the highest ccurcy for the differentition of oth entities [4]. In ddition to conventionl imging studies, the ssessment of vsculrity either y myocrdil perfusion contrst echocrdiogrphy [16] or crdic ctheteriztion my ssist in the differentition of thromi nd other type of crdic tumors. Nevertheless, in few instnces, the finl dignosis of crdic msses cn only e mde y otining pthologicl specimen fter surgicl resection of crdic msses [5 7, 12]. For our ptient, the nture of itril mss ws not specified y histology ecuse he declined surgery. The presence of MS, AF, SEC nd dilted indicted dignosis of thromi. Our cse is unusul ecuse the finl dignosis ws ffirmed y empiricl nticogultion sed on clinicl judgment nd noninvsive chrcteriztion of itril mss. A reported cse of left tril thromus with stlk showed tht tril of nticogultion ws eneficil in regressing the crdic mss prticulrly when the differentil dignosis ws difficult nd thromus ws possiility [6]. There were lso cses tht reported successful regressions of the thromus with nticogultion without the need for surgicl thromectomy [15, 17, 18]. Regression of thromus using wrfrin hs een studied in ptients with MS nd left tril thromus detected prior to percutneous trnsvenous mitrl commissurotomy. Among 219 ptients following 6 months of wrfrin (INR 2 to 3) therpy, 24.2 % of ptients were found to hve complete resolution of thromus nd 75.8 % of ptients were found to hve prtil resolution of thromus, nd higher INR (t lest 2.5) predicted thromus resolution [19]. For our ptient, the regression of his crdic msses ws not oserved in the short term fter tretment with Tle 1 Bitril mss with mitrl stenosis Author Yer Age/Sex Presenttion Atril firilltion Dignosis Tretment Irhim et l. [11] 2008 51/mle Right hemipresis Myxom Alteplse dministered intrvenously + tumor excision Tsdemir et l. [10] 2008 58/femle Left foot pin + Thromi Thromectomy + MV surgery Tsuokw et l. [9] 2010 77/mle Dyspne + Thromi Thromectomy + MV surgery Khnn et l. [8] 2015 40/femle Dyspne + Thromi Thromectomy + MV surgery MV mitrl vlve, (+) presence, ( ) sence
Lu et l. Journl of Medicl Cse Reports (2016) 10:221 Pge 5 of 5 wrfrin, csting dout on the true identity of the itril mss. We suspect the min contriuting fctor towrds the lck of thromi regression ws indequte nticogultion owing to his lck of complince. For this reson, complince to nticogultion therpy ws pivotl in thromi regression, nd it took 5 yers to visulize the resolution of itril thromi. Although thorough ttempt hd een mde, we could not convince our ptient to gree to surgery. We elieve tht erly mitrl vlve surgery nd thromectomy is eneficil in preventing thromoemolism. Conclusions Noninvsive chrcteriztion of crdic mss is essentil in clrifying the dignosis nd directing tretment strtegy. Anticogultion is fesile tretment when the clinicl ssessment, risk fctors, nd imging findings indicte dignosis of thromi. After prolonged nticogultion therpy, complete resolution of itril thromi ws chievle in our cse. Arevitions AF, tril firilltion; CMR, crdic mgnetic resonnce; INR, interntionl normlized rtio;, left trium; MS, mitrl stenosis;, right trium; SEC, spontneous echocrdiogrphic contrst; TEE, trnsesophgel echocrdiogrphy; TTE, trnsthorcic echocrdiogrphy Acknowledgements Not pplicle. Funding Self-funded. Avilility of dt nd supporting mterils Not pplicle. Authors contriutions HTL is the first uthor who treted the ptient, orgnized the investigtions, nd otined ptient consent. HTL nd RN wrote the first drft of mnuscript. CNC, THG, nd NO were involved in cquisition nd interprettion of imges. BCL, JYK, nd GR treted the ptient nd provided comments on the mnuscript. All uthors red nd pproved the finl mnuscript. References 1. Bruce CJ. Crdic tumours: dignosis nd mngement. Hert. 2011;97:151 60. 2. Mnning WJ, Weintru RM, Wksmonski CA, et l. Accurcy of trnsesophgel echocrdiogrphy for identifying left tril thromi: prospective, intropertive study. Ann Intern Med. 1995;123(11):817 22. 3. Altch MI, Squire SW, Kudithipudi V, et l. Crdic MRI is complementry to echocrdiogrphy in the ssessment of crdic msses. Echocrdiogrphy. 2007;24:286 300. 4. Pzos-López P, Pozo E, Siqueir ME, et l. Vlue of CMR for the differentil dignosis of crdic msses. JACC Crdiovs Imging. 2014;7(9):896 905. 5. Kodli S, Ymrozik J, Biedermn RWW. Left tril thromus msquerding s myxom in ptient with mitrl stenosis: cse report. Echocrdiogrphy. 2010;27:98 101. 6. Jng KH, Shin DH, Lee CK, et l. Left tril mss with stlk: thromus or myxom? J Crdiovsc Ultrsound. 2010;18(4):154 6. 7. Dhwn S, Tk T. Left tril mss. Thromus mimicking myxom. Echocrdiogrphy. 2004;21(7):621 3. 8. Khnn SN, Pul M, Shrm V, et l. A cse of itril mss mimicking myxom with rheumtic mitrl stenosis with regurgittion of moderte severity. J Ind Coll Crdio. 2015;5(4):338 40. 9. Tsuokw K, Murmoto A, Trui T, et l. Unusul itril thromus in mitrl stenosis. J Echocrdiogr. 2010;8(4):135 6. 10. Tsdemir K, Srli B, Ky MG, et l. Moile itril thromus in ptient with mitrl stenosis under heprin infusion. Inter CrdioVs Thor Surg. 2008; 7:667 9. 11. Irhim M, Iliescu C, Sfi HJ, et l. Bitril myxom nd cererl ischemi successfully treted with intrvenous thromolytic therpy nd surgicl resection. Tex Hert Inst J. 2008;35(2):193 5. 12. Deluigi CC, Meinhrdt G, Ursulescu A, et l. Noninvsive chrcteriztion of left tril mss. Circultion. 2006;113:19 20. 13. Acet H, Duygu H, Erts F, et l. A cse of moile gint left tril thromus which vsculrized with coronry rteries in severe mitrl vlve stenosis. Crdiovsc Revsc Med. 2010;11:137 8. 14. Mhdhoui A, Bouroui H, Amine MM, et l. The trnsesophgel echocrdiogrphic dignosis of left tril myxom simulting left tril thromus in the setting of mitrl stenosis. Echocrdiogrphy. 2004;21(4):333 6. 15. Immur K, Tkeuchi M, Hruki N, et l. Simultneous visuliztion of two intrcrdic msses in oth tri on 3D trnsesophgel echocrdiogrphy. Circultion J. 2011;75(4):986 8. 16. Kirkptrick JN, Wong T, Bednrz JE, et l. Differentil dignosis of crdic msses using contrst echocrdiogrphic perfusion imging. J Am Coll Crdiol. 2004;43:1412 9. 17. Khn H, Chuey S, Hill A, et l. Lrge left tril thromus fter mitrl vlve replcement: how to tret? Asin Crdiovsc Thorc Ann. 2012;20:457 9. 18. Al-Bezem R, Kwn T, Clrk LT. Regression of lrge tril thromi nd coronry neovsculriztions with conventionl nticogultion in mitrl stenosis, cse report. Angiology. 1999;50(10):859 63. 19. Silruks S, Thinkhmrop B, Kitchooskun S, et l. Resolution of left tril thromus fter 6 months of nticogultion in cndidtes for percutneous trnsvenous mitrl commissurotomy. Ann Int Med. 2004;140:101 5. Competing interests The uthors declre tht they hve no competing interests. Consent for puliction Written informed consent ws otined from the ptient for puliction of this cse report nd ccompnying imges. A copy of the written consent is ville for review y the Editor-in-Chief of this journl. Author detils 1 Clinicl School Johor Bhru, Jeffrey Cheh School of Medicine nd Helth Sciences, Monsh University Mlysi, 8 Jln Msjid Au Bkr, 80100 Johor Bhru, Johor, Mlysi. 2 Deprtment of Rdiology, Sultnh Aminh Hospitl, Jln Au Bkr, 80100 Johor Bhru, Johor, Mlysi. 3 Deprtment of Crdiology, Sultnh Aminh Hospitl, Jln Au Bkr, 80100 Johor Bhru, Johor, Mlysi. 4 Deprtment of Crdiology, Penng Generl Hospitl, Jln Residensi, 10990 Georgetown, Pulu Pinng, Mlysi. Received: 27 June 2016 Accepted: 28 July 2016 Sumit your next mnuscript to BioMed Centrl nd we will help you t every step: We ccept pre-sumission inquiries Our selector tool helps you to find the most relevnt journl We provide round the clock customer support Convenient online sumission Thorough peer review Inclusion in PuMed nd ll mjor indexing services Mximum visiility for your reserch Sumit your mnuscript t www.iomedcentrl.com/sumit