A case of long-term survival after surgical resection for solitary adrenal recurrence of esophageal squamous carcinoma

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1 Kny et l. Surgicl Cse Reports (2017) 3:61 DOI /s CASE REPORT Open Access A cse of long-term survivl fter surgicl resection for solitry drenl recurrence of esophgel squmous crcinom Nouhiko Kny 1,KzuhiroNom 1*,TsuyoshiOkd 1, Noki Med 1, Shunsuke Tne 1, Kzufumi Skurm 1,2, Ysuhiro Shirkw 1 nd Toshiyoshi Fujiwr 1 Astrct Bckground: Esophgel crcinoms re highly mlignnt tumors with high frequency of lymph node nd distnt orgn metstsis. Tretment for recurrent tumors is generlly decided on n individul sis. Although multidisciplinry tretments involving chemotherpy, surgicl resection, nd rdition re performed, the prognosis remins poor. Here, we report cse of prolonged recurrence-free survivl (38 months) fter esophgel crcinom surgery nd susequent lproscopic drenlectomy for right drenl metstsis. Cse presenttion: An 83-yer-old mn ws dignosed with type 3 esophgel squmous cell crcinom (T3N1M0, cstge IIIA, UICC-7), spreding from the lower thorcic esophgus to the dominl esophgus. He underwent thorcoscopic esophgectomy with two-field lymph node dissection followed y susternl gstric tue reconstruction. The finl dignosis ws modertely differentited squmous cell crcinom (T3N2M0, fstge IIIB). Adjuvnt chemotherpy ws not dministered ecuse of the dvnced ge nd postopertive condition of the ptient. Computed tomogrphy (CT) t 14 months postopertively showed mss with 2-cm dimeter t the right drenl glnd. Positron emission tomogrphy (PET)/CT reveled high fluorodeoxyglucose (FDG) uptke in the mss. It ws suspected tht the mss ws metsttic lesion secondry to the primry esophgel crcinom. No metstses to lymph nodes or other distnt orgns were identified. The ptient underwent lproscopic right drenlectomy. The histopthologicl exmintion reveled modertely differentited squmous cell crcinom, suggesting metstsis from the primry esophgel crcinom. He hs survived without recurrence for 38 months since lproscopic drenlectomy to remove the right drenl metststic mss fter the esophgel crcinom surgery. Conclusions: We descrie very elderly mle who survived lprodrenlectomy for right drenl metstsis following esophgel cncer surgery without recurrence for 38 months postopertively. Therefore, surgicl resection might e n option for solitry drenl recurrence. Keywords: Esophgectomy, Adrenl metstsis, Esophgel squmous cell crcinom Bckground Esophgel crcinoms re highly iologiclly mlignnt tumors ecuse the high frequency of lymph node nd distnt orgn metstsis [1]. Common ptterns of recurrence re metstsis to the lymph nodes, lungs, liver, one, rin, nd drenl glnds [2, 3]. Tretment for recurrent * Correspondence: knom@md.okym-u.c.jp 1 Deprtment of Gstroenterologicl Surgery, Okym University Grdute School of Medicine, Dentistry nd Phrmceuticl Sciences, Shikt-cho, Kit-ku, Okym , Jpn Full list of uthor informtion is ville t the end of the rticle lesions is generlly decided on n individul sis. Although multidisciplinry tretments re comined with chemotherpy, surgicl resection, nd rdition, the prognosis remins poor [4, 5]. The role of surgicl resection for metstsis from esophgel crcinom hs not een clrified, ut some reports hve recently descried the enefit of resection for oligometstsis from esophgel crcinom [6, 7]. The Author(s) Open Access This rticle is distriuted under the terms of the Cretive Commons Attriution 4.0 Interntionl License ( 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.

2 Kny et l. Surgicl Cse Reports (2017) 3:61 Pge 2 of 5 Fig. 1 Resected specimen of esophgel crcinom. Gross ppernce shows type 3 tumor, pproximtely 4.5-cm long in LtAe. Hemtoxylin nd eosin stining shows modertely differentited squmous cell crcinom Here, we report cse of 38-month recurrence-free survivl fter lproscopic drenlectomy for right drenl metstsis fter esophgel crcinom surgery. Cse presenttion An 83-yer-old mn ws dignosed with type 3 esophgel squmous cell crcinom (LtAe, T3N1M0, cstge IIIA, UICC-7) mesuring 6 cm in dimeter. Computed tomogrphy (CT) nd 8F-fluorodeoxyglucose (FDG) positron emission tomogrphy (PET)/CT showed no metstsis to distnt orgns, wheres metsttic lymph nodes were present t the lesser curvture of the stomch. The ptient underwent thorcoscopic esophgectomy with two-field lymph node dissection, lproscopic susternl gstric tue reconstruction (Fig. 1). The finl dignosis ws modertely differentited squmous cell crcinom (T3N2M0, stge IIIB). Three lymph nodes t the lesser curvture of the stomch were dignosed s contining metsttic squmous cell crcinom. The ptient suffered from dirrhe fter the opertion. Adjuvnt chemotherpy ws not dministered ecuse of the ptient s dvncing ge nd postopertive condition. After tretment, follow-up ws conducted on n outptient sis once every 3 months. Follow-up included physicl exmintion nd lortory tests, including those for tumor mrkers. Alternting 6-month periods of upper gstrointestinl endoscopy, contrst-enhnced CT, nd FDG-PET/CT were performed. CT t 14 months postopertively showed smll growing mss, which hd enlrged to 2 cm in dimeter in the right drenl glnd (Fig. 2) nd PET/CT reveled high FDG uptke t the mss (mximum stndrdized uptke vlue (SUV), 7.67). The physicl exmintion t tht time showed no normlities. Lortory tests showed tht concentrtions of squmous cell crcinom (SCC) ntigen nd crcinoemryonic ntigen (CEA) hd incresed to 12.3 ng/ml (norml rnge, ng/ml) nd ng/ml (norml rnge, ng/ml), lthough these concentrtions hd een norml just fter the primry opertion (Fig. 3). The mss ws suspected to e metstsis from the primry esophgel crcinom. No metstses to lymph nodes or other distnt orgns were pprent. The ptient ws dmitted to our hospitl for surgery nd underwent lproscopic right drenlectomy from the left lterl decuitus position. A port for endoscopy ws present where Fig. 2 Imges from CT nd FDG-PET/CT. A mss with 2-cm dimeter in the right drenl glnd. PET/CT shows high ccumultion of FDG (stndrdized uptke vlue mx, 7.67) in the right drenl mss. Arevitions: CT computed tomogrphy, PET positron emission tomogrphy, FDG 8F-fluorodeoxyglucose

3 Kny et l. Surgicl Cse Reports (2017) 3:61 Pge 3 of 5 Primy ope. Second ope. 35 SCC (ng/ml) CEA (ng/ml) Fig. 3 Clinicl chnges in concentrtions of tumor mrkers, crcinoemryonic ntigen (lue line) nd squmous cell crcinom (red line). Ope opertion the right rcus costlis nd externl mrginl dominis muscle crossed nd the other three ports were long the sucostl lines. If there hd een severe intr-dominl dhesion, then the lproscopic pproch would hve een chnged to the retroperitonel pproch. Though there ws inflmmtory dhesion etween the inferior ven cv nd dipose tissue round the tumor, it ws possile to exfolite them. This second opertion ended without ny prolems ecuse the right drenl glnd ws ntomiclly wy from the gstric tue. The opertive time ws 127 min. The volume of lood loss ws 30 ml. The histopthologicl exmintion reveled modertely differentited squmous cell crcinom, strongly suggesting the presence of metstsis from the primry esophgel cncer (Fig. 4). Lortory tests fter this second opertion showed decresed concentrtions of SCC ntigen nd CEA to 1 nd 4.4 ng/ml, respectively. Thus fr, no postopertive recurrences hve occurred s of 38 months fter drenlectomy. Conclusions Distnt recurrence fter curtive esophgel crcinom surgery is one of the most difficult complictions to tret. Recently, multidisciplinry therpy hs een performed for recurrent esophgel crcinom. However, the prognosis of the ptients with recurrence is very poor, with reported survivl of 4 7 months [8, 9]. The enefit of surgicl resection for ptients with recurrence remins controversil. Some reports indicted tht lymphdenectomy or chemordiotherpy might improve survivl in ptients with lymph node recurrence in the neck or medistinum fter curtive resection [4, 5]. Ptients with lung metstsis reportedly show reltively good prognosis, with medin survivl of 9.8 months [8].Wheres, ptients with liver metstsis reportedly exhiit poor prognosis ecuse of the high frequency of multiple metstses [6]. The initil recurrent site could thus contriute to the prognostic heterogeneity of the ptients with recurrent esophgel crcinom. Most tumors rising in the drenl glnd re enign denoms [10], nd mlignnt tumors of the drenl glnd re quite rre. The frequency of non-drenl crcinom metstses to the drenl glnd is of pproximtely % [10]. Common crcinoms ssocited with metstses to the drenl glnds re lung, gstrointestinl, rest, kidney crcinom, nd melnom. Adrenl metstsis is generlly identified on CT, PET, or MRI, during postopertive follow-up [10, 11]. Therpy for drenl metstsis is decided on n individul sis depending on the primry crcinom. The chrcteristics of drenl metstsis on imging studies include n irregulr shpe, inhomogeneous nture, high vsculrity on contrst-enhnced CT, nd elevted SUV on PET[11].Inourcse,thedrenlmsswsfoundon follow-up CT nd FDG-PET/CT in the sence of other primry lesions. Tumor mrkers such s SCC nd CEA were lso elevted. Additionlly, FDG uptke ws high Fig. 4 Resected specimen of the right drenl mss. Gross ppernce shows hrd, solid mss pproximtely 2.0 cm in dimeter. Hemtoxylin eosin stining shows modertely differentited squmous cell crcinom

4 Kny et l. Surgicl Cse Reports (2017) 3:61 Pge 4 of 5 Tle 1 Eight ptients with surgicl resection of drenl metstsis from esophgel crcinom Yer Author Age Sex Loction of the EC Histology fstge Loction of the AM Size of AM (cm) Intervl from the EC Prognosis Chemotherpy 1992 Shimd 59 Mle MtLt SCC III Right 6 6 cm 4 months 18 months live Unknown 1997 Yoshizumi 56 Mle MtLt SCC IV Left cm 0 22 months live Ht 67 Mle MtLt SCC III Left cm 8 months 14 months live 2004 Ngno 57 Mle MtLt SCC II Left cm 3 months unknown Unknown 2006 MM.Cho 70 Mle LtAe SCC III Left 5 4 cm 8 months 42 months live Sito 71 Mle Mt Adeno III Right cm 22 months 71 months live O Sullivn KE 50 Mle AeG Adeno II Left Unknown 48 months 48 months live Our cse 83 Mle LtAe SCC III Right 2 cm 14 months 36 months live SCC squmous cell crcinom, Adeno denocrcinom, AM drenl metstsis, EC esophgel crcinom despite the reltively smll size. Metstsis from esophgel crcinom ws therefore suspected. Adrenl metstsis in ptients with esophgel crcinom is very rre [12]. We found only eight cses etween 1995 nd 2015 (Tle 1) [13 18]. The sex rtio showed cler mle predominnce (mle:femle, 8:0). Medin ge t primry dignosis ws 63 yers (rnge, yers). The drenl metstsis ws right-sided in three ptients nd left-sided in five. The medin intervl from the primry opertion to discovery of metstsis ws 8 months. Ptients who underwent drenlectomy survived for medin of 36 months (rnge, months), with ll chieving reltively long survivl fter drenlectomy. Thus, in our very elderly ptient, lproscopic drenlectomy ws selected. In generl, chemotherpy is chosen for distnt recurrence of esophgel crcinom ecuse of the oncologicl systemic sttus. Chemotherpy such s S-1 ws considered for this ptient. However, chemotherpy ws not recommended for our ptient ecuse of his dvnced ge nd concern out the side effects such s dirrhe. Thus, with sufficiently informed consent, surgicl resection represents n option for solitry drenl metstsis, even for those with distnt recurrence. Best prctice therpy including surgicl resection nd rdition therpy s well s chemotherpy is thus importnt to consider for ptients with drenl metstsis recurrence of esophgel crcinom. In prticulr, surgicl resection might e more importnt for solitry drenl metstsis thn for other distnt metstses. We report cse of prolonged recurrence-free survivl (38 months) fter lprodrenlectomy for right drenl metstsis following esophgel cncer surgery in very elderly mle. Surgicl resection might e n option for solitry drenl recurrence. Arevitions CEA: Crcinoemryonic ntigen; CT: Computed tomogrphy; FDG: 8F-fluorodeoxyglucose; PET: Positron emission tomogrphy; SCC: Squmous cell crcinom; SUV: Stndrdized uptke vlue Funding The uthors declre tht this study ws not funded externlly. Authors contriutions NK performed the literture review nd wrote the mnuscript. YS, KN, KS, ST, nd NM were involved in the clinicl mngement of the ptient. TO prticipted in literture review. TF, YS, nd KN revised the mnuscript. All uthors were involved in the mngement of the ptient. All uthors red nd pproved the finl mnuscript. Competing interests The uthors declre tht they hve no competing interests. Consent for puliction The consent to pulish is otined from this ptient. Pulisher s Note Springer Nture remins neutrl with regrd to jurisdictionl clims in pulished mps nd institutionl ffilitions. Author detils 1 Deprtment of Gstroenterologicl Surgery, Okym University Grdute School of Medicine, Dentistry nd Phrmceuticl Sciences, Shikt-cho, Kit-ku, Okym , Jpn. 2 Deprtment of Surgery, Shigei Medicl Reserch Institute, Okym, Jpn. Received: 2 Ferury 2017 Accepted: 1 My 2017 References 1. Lerut T, De Leyn P, Coosemns W, Vn Remdonck D, Scheys I, LeSffre E. Surgicl strtegies in esophgel crcinom with emphsis on rdicl lymphdenectomy. Ann Surg. 1992;216: Kto H. Clssifiction of recurrent esophgel cncer fter rdicl esophgectomy with two- or three-field lymphdenectomy. Anticncer Res. 2010;210: Ate E, DeMeester SR, Zehetner J, Oezcelik A, Ayzi S, Costles J, Bnki F, Liphm JC, Hgen JA, DeMeester TR. Recurrence fter esophgectomy for denocrcinom: defining optiml follow-up intervls nd testing. J Am Coll Surg. 2010;210: Nkmur T, Ot M, Nrumiy K, Sto T, Ohki T, Ymmoto M, Mitsuhshi N. Multimodl tretment for lymph node recurrence of esophgel crcinom fter curtive resection. Ann Surg Oncol. 2008;15: Kuniski C, Mkino H, Tkgw R, Ymmoto N, Ngno Y, Fujii S, Kosk T, Ono HA, Otsuk Y, Akiym H, Ichikw Y, Shimd H. Surgicl outcomes in esophgel cncer ptients with tumor recurrence fter curtive esophgectomy. J Gstrointest Surg. 2008;12: Huddy JR, Thoms RL, Worthington TR, Krnji ND. Liver metstses from esophgel crcinom: is there role for surgicl resection? Dis Esophgus. 2015;28:483 7.

5 Kny et l. Surgicl Cse Reports (2017) 3:61 Pge 5 of 5 7. Chen F, Sto K, Ski H, Miyhr R, Bndo T, Okuo K, Hirt T, Dte H. Pulmonry resection for metstsis from esophgel crcinom. Interct Crdiovsc Thorc Surg. 2008;7: Mriette C, Blon JM, Piessen G, Fre S, Vn Seuningen I, Trioulet JP. Pttern of recurrence following complete resection of esophgel crcinom nd fctors predictive of recurrent disese. Cncer. 2003;97: Ichikw H, Kosugi S, Knd T, Ishikw T, Yjim K, Akzw K, Suzuki T, Wki T. Prognostic significnce of initil recurrence site in hemtogenous recurrence of esophgel squmous cell crcinom. Heptogstroenterology. 2014;61: Bolnd GW, Blke MA, Hollkere NS, Hhn PF. PET/CT for the chrcteriztion of drenl msses in ptients with cncer: qulittive versus quntittive ccurcy in 150 consecutive ptients. AJR Am J Roentgenol. 2009;192: Okd M, Shimono T, Komey Y, Ando R, Kgw Y, Ktsue T, Kuwr M, Ygyu Y, Kumno S, Imok I, Tsuchiy N, Ashikg R, Hosono M, Murkmi T. Adrenl msses: The vlue of dditionl fluorodeoxyglucose-positron emission tomogrphy/computed tomogrphy (FDG-PET/CT) in differentiting etween enign nd mlignnt lesions. Ann Nucl Med. 2009;23: Young WF. Clinicl prctice. The incidentlly discovered drenl mss. N Engl J Med. 2007;356: Ninomiy I, Okmoto K, Tsukd T, Kinoshit J, Oym K, Fushid S, Osugi H, Oht T. Recurrence ptterns nd risk fctors following thorcoscopic esophgectomy with rdicl lymph node dissection for thorcic esophgel squmous cell crcinom. Mol Clin Oncol. 2016;4: Ht S, Amno S, Skuri K, Fujii H, Pku E, Kno H, Oid N, Miyke H, Fukuzw M, Sugitni M. Resection of drenl metstsis from esophgel cncer: cse report. Jpn J Gstroenterol Surg. 2000;33: Sito H, Shuto K, Ot T, Tom T, Ohir G, Ntsume T, Uesto M, Akutsu Y, Kono T, Mtsur H. A cse of long-term survivl fter resection for postopertive solitry drenl metstsis from esophgel denocrcinom. Gn to Kgku Ryoho. 2010;37: Cho MM, Koyshi K, Aoki T, Nishiok K, Yoshid K, Htno N, Hirose H, Moon JH, Mtsumoto T, Uemur Y, Kneko T, Okgw K. Surgicl resection of solitry drenl metstsis from esophgel crcinom following esophgectomy. Dis Esophgus. 2007;20: Yoshizumi Y, Moriski Y, Koike H, Shit H, Yngw R, Sugiur Y, Tnk S. Successful comined resection of crcinom of the esophgus nd drenl metstsis: report of cse. Surg Tody. 1997;27: Shimd Y, Immur M, Knd Y. Resection of drenl metstsis from esophgel crcinom fter esophgectomy without recurrence for one nd hlf yer: cse report. Arch Jpn Chir. 1992;61:86. Sumit your mnuscript to journl nd enefit from: 7 Convenient online sumission 7 Rigorous peer review 7 Immedite puliction on cceptnce 7 Open ccess: rticles freely ville online 7 High visiility within the field 7 Retining the copyright to your rticle Sumit your next mnuscript t 7 springeropen.com

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