Prosthetic Valve Dysfunction 35 Years after Mitral Valve Replacement with a Starr-Edwards Caged-disc Valve

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CASE REPORT Prosthetic Vlve Dysfunction 35 Yers fter Mitrl Vlve Replcement with Strr-Edwrds Cged-disc Vlve Tetsuro Yokokw 1, Tkhiro Ohr 1, Seiji Tkshio 1, Mri Skmoto 1,YukoWd 1, Kenji Nkmur 1, Hiroyuki Tkhm 1, Mkoto Amki 1, Tkuy Hsegw 1, Ysuo Sugno 1, Hideki Knzki 1, Stoshi Ysud 1,HisoOgw 1, Tomoyuki Fujit 2, Junjiro Koyshi 2, Yoko Okmoto 3, Tk-ki Mtsuym 3, Htsue Ishishi-Ued 3 nd Toshihis Anzi 1 Astrct A 49-yer-old mn ws dmitted to our hospitl with chief complint of dyspne. He hd history of mitrl vlve replcement (MVR) with Strr-Edwrds (SE) cged-disc vlve t the ge of 14. Echocrdiogrphy reveled elevted trns-vlvulr pressure grdient of the mitrl prosthetic vlve with neither disk motion normlity nor norml structure. Ctheteriztion confirmed n elevtion of the men distolic grdient of the mitrl vlve to 12.3 mmhg. Re-MVR ws performed, nd norml tissue ttched to the cge of the vlve nd proliferting eneth the vlve ws oserved. Histologic exmintion reveled them s firinous tissue nd mild pnnus prolifertion, respectively. This rre cse report focuses on long-term follow-up nd the compliction of SE cged-disc vlve. A SE cged-disc vlve my ecome stenotic, only detected with trns-vlvulr pressure grdient without ny disk motion normlity or norml structure during prolonged follow-up period. Key words: mitrl vlve replcement, prosthetic vlve dysfunction, Strr-Edwrds cged-disc vlve, pnnus, firinous tissue (Intern Med 55: 479-483, 2016) (DOI: 10.2169/internlmedicine.55.5466) Introduction The Strr-Edwrds (SE) cged-disc vlve, model 6520, ws developed in the erly 1970s for mitrl vlve replcement (MVR). The SE cged-disc vlve ws used for only short period of time efore eing discontinued universlly. Some ptients lived with the SE cged-disc vlve for long time. The very long-term (over 30 yers) outcome of the SE cged-disc vlve hs een reported to e ssocited with structurl deteriortion in few ptients (1). We witnessed cse of SE cged-disc vlve dysfunction due to firinous tissue ttched to the cge nd mild pnnus formtion 35 yers fter MVR, in which the ntomicl dignosis ws preopertively difficult to mke. Cse Report A 49-yer-old mn ws dmitted to our hospitl complining of dyspne. He hd history of cererl infrction due to infective endocrditis, nd MVR with SE cgeddisc vlve model 6520 for mitrl regurgittion nd infective endocrditis in our hospitl t 14 yers of ge. Twelve yers fter the MVR procedure, proxysml tril firilltion occurred. At 31 yers of ge, the ptient underwent ctheter ltion for tril flutter. Thirty-five yers fter the MVR, when the ptient ws 49 yers old, he suffered from leg edem nd dyspne on effort. Trnsthorcic echocrdiogrphy reveled n elevted men trns-vlvulr pressure grdient of 10.8 mmhg, with Deprtment of Crdiovsculr Medicine, Ntionl Cererl nd Crdiovsculr Center, Jpn, Deprtment of Crdiovsculr Surgery, Ntionl Cererl nd Crdiovsculr Center, Jpn nd Deprtment of Pthology, Ntionl Cererl nd Crdiovsculr Center, Jpn Received for puliction Mrch 27, 2015; Accepted for puliction June 1, 2015 Correspondence to Dr. Tkhiro Ohr, tkohr@ncvc.go.jp 479

DOI: 10.2169/internlmedicine.55.5466 c d Figure 1. Two-dimensionl trnsthorcic (, ) nd trnsesophgel (c, d) echocrdiogrphy. : Trnsthorcic echocrdiogrphy did not indicte either ny significnt motion or structurl normlity of the disc. : A Doppler continuous flow reveled n elevted men trns-vlvulr pressure grdient of 10.8mmHg. The pek velocity ws 2.0m/s nd the pressure hlf time ws 353msec. c, d: Two-dimensionl trnsesophgel echocrdiogrphy did not revel ny normlities tht would cuse mitrl dysfunction (c: distole, d: systole). Figure 2. Cinerdiogrphy of the mitrl prosthetic vlve showing no restrictive motion of the disc. : Distolic phse. : Systolic phse. neither disc motion normlity nor significnt norml structure (Fig. 1). Electrocrdiogrphy displyed tril firilltion. Crdic ctheteriztion showed men pressure difference etween the pulmonry cpillry wedge pressure nd left ventriculr pressure of 12.3 mmhg. The men pulmonry rtery pressure ws 24 mmhg nd crdic index ws 2.26 L/min/m 2. Cinerdiogrphy of the mitrl vlve indicted tht the disc could still move (Fig. 2). Trnsesophgel echocrdiogrphy did not find relevnt structurl normlity regrding the mitrl vlve (Fig. 1, 3). Although we did not find ny structurl normlity tht would explin the dysfunction, we dignosed the ptient s hving symptomtic prosthetic mitrl vlve stenosis of the SE cged-disc vlve. He underwent MVR with St. Jude Medicl mechnicl vlve nd mze procedure. Intropertive oservtion showed mild pnnus formtion on the tril side of the prosthetic vlve nd norml tissue ttching to the cge of 480

DOI: 10.2169/internlmedicine.55.5466 Figure 3. Three-dimensionl trnsesophgel echocrdiogrphy (3D TEE). 3D TEE did not show ny norml structures tht could cuse mitrl vlve dysfunction. : Atril spect. : Ventriculr spect. 5mm Figure 4. Mcroscopic findings of the removed Strr-Edwrds cged-disc. : Pnnus formtion round the prosthetic vlve nnulus on the tril side. Note the mild extension of the firinous tissue over the ring (yellow rrows). The lower prt of the pnnus in this figure ws removed from the vlve during the opertion, which ws comprle mount to the remining tissues. : Ventriculr side of the frme nd disc. Firinous soft tissue ws ttched to the frme (yellow rrows). ventriculr side (Fig. 4). After the opertion, the ptient s symptoms improved. Echocrdiogrphy showed the men pressure grdient of the mitrl vlve to e 4 mmhg. He ws dischrged 11 dys fter the opertion. A pthologic exmintion of the removed vlve reveled mild extension of proliferted firinous tissue with clcifiction, or pnnus, over the mitrl nnulus, s well s some soft firinous sustnce which ws ttched to the frme of the cge (Fig. 5). Discussion In this pper, we reported mitrl vlve stenosis of the SE cged-disc vlve 35 yers fter the disc ws replced due to n norml soft firinous sustnce which ws ttched to the cge of the vlve nd mild pnnus formtion on the tril spect. To the est of our knowledge, this is the first report descriing prosthetic vlve filure with this kind of mechnism. The SE cged-disc vlve is low-profile prosthetic vlve developed to overcome the limittions of SE cged-ll vlve. It egn to e used in the 1970s, especilly for ptients whose ventriculr chmers were smll. This vlve s use ws discontinued fter short period of time ecuse it wore out firly quickly nd cused thromosis due to lood strem turulence due to its structure (2). However, some ptients hve survived with this vlve. The long-term outcomes of the SE cged-disc vlve hd not een fully investigted. Aoygi et l. reported four ptients who underwent MVR with the SE cged-disc vlve over 30 yers go (1). The report descried frcture, wer, nd crcks of the SE cged-disc vlve over 30 yers fter implnttion. Pnnus overgrowth fter the implnttion of SE cged-disc vlve hs lso een previously reported (3). There hve lso een few reports regrding the hemodynmic evlution of SE cged-disk vlve without vlve dysfunction, nd the norml men mitrl distolic grdient of the SE cged-disc vlve ws reported to rnge from 4.0 to 481

DOI: 10.2169/internlmedicine.55.5466 clc Fi c d Figure 5. Histopthologicl findings of pnnus nd firinous sustnce. : Immunostining for osteopontin. Clcifiction is immunohistochemiclly positive for osteopontin (Leic, New Cstle, UK, dilution 1:500). : Msson s Trichrome stining. c, d: Hemtoxylin nd Eosin stining. clc: clcifiction, fi: firin. : Grnulr clcified clusters re in the collgenous fier of the pnnus. : Firinous tissue is lso ttched to the surfce of the pnnus. c: Firinous sustnce ttched to the cge. d: High mgnifiction of Fig. 5c. Mcrophges (mrked y rrows) were oserved with the firin components. 7.9 mmhg (4-6). In our cse, the men mitrl distolic grdient ws 12.3 mmhg, significntly higher thn the norml rnge. The pre-opertive exmintion did not detect the ntomicl cuse of the elevted trns-vlvulr pressure. Intropertive oservtion showed pnnus formtion nd firinous sustnce surrounding the prosthetic vlve. There re no other findings to cuse vlve dysfunction, so we find it resonle to ssume tht those fctors cused prosthetic mitrl vlve stenosis. Evluting prosthetic mechnicl vlve vi echocrdiogrphy is difficult. No motion normlities of the disc ppered on fluoroscopy. The soft firinous sustnce on the cge nd pnnus might hve disrupted the lood flow through the prosthetic mitrl vlve without interfering with the disc s movement. Persistent mechnicl stress y foreign oject nd exposure to synthetic mteril cn cuse the humn ody to rect to the item with mcrophge infiltrtion. In turn, this might hve cused pnnus formtion nd firinous sustnce ttchment long fter the MVR procedure. We herein reported cse of mitrl vlve stenosis due to firinous sustnce ttched to the cge nd pnnnus eneth the prosthetic vlve. This sitution occurred 35 yers fter MVR with SE cged-disc vlve. As result, the condition ws difficult to dignose preopertively. Ptients who hve SE cged-disc vlve need creful long-term followup to identify ny prosthetic vlve dysfunction. If ptient with cged vlve showed n elevted trns-vlvulr grdient nd no relevnt structurl normlity, the possiility of firinous ttchment to the cge of the vlve nd pnnus formtion should e suspected. The uthors stte tht they hve no Conflict of Interest (COI). References 1. Aoygi S, Tym K, Okzki T, et l. Structurl vlve deteriortion in strr-edwrds mitrl cged-disk vlve prosthesis. Circ J 77: 105-108, 2013. 2. Skmoto Y, Hshimoto K. Development of prosthetic vlves. The Journl of Adult Diseses 44: 859-864, 2014. 3. Aoygi S, Fukung S, Tym E, Aring K, Od T, Kosug T. 482

DOI: 10.2169/internlmedicine.55.5466 Long-term survivors fter vlve replcement with Strr-Edwrds mitrl disk vlve prosthesis. Artif Orgns 30: 484-487, 2006. 4. Brown JW, Myerowitz PD, Cnn MS, Colvin SB, McIntosh CL, Morrow AG. Clinicl nd hemodynmic comprisons of Ky- Shiley, Strr-Edwrds No. 6520, nd Reis-Hncock porcine xenogrft mitrl vlves. Surgery 76: 983-991, 1974. 5. Nkno S, Mori T, Ohym C, et l. [Lte results of mitrl vlve replcement with Strr-Edwrds low profile disc vlve]. Kyou Gek (The Jpnese Journl of Thorcic Surgery) 31: 440-445, 1978 (in Jpnese). 6. Kremu EL, Kloster FE, Griswold HE, Strr A. Hemodynmic evlution of Strr-Edwrds model 6520 mitrl-tricuspid disc prosthesis. Circultion 178 (Suppl 2): 145-146, 1972. 2016 The Jpnese Society of Internl Medicine http://www.nik.or.jp/imonline/index.html 483