In a 58-year-old male, upper digestive endoscopy revealed a protruding lesion in the esophagus on a medical examination. The patient was referred to the Department of Surgery in our hospital to undergo surgery. On the initial consultation, upper digestive endoscopy showed a smooth, soft, black purple, typeⅡprotruding lesion measuring approximately 25mmat 35 cm apart from the incisor. For diagnotic treatment and patient’s request, endoscopic mucosal resection (EMR)was performed. The resected specimen measured 25mm× 25 mm. The histological findings suggested cavernous hemangioma. To treat esophageal hemangioma, esohagectomy, tumor enucleation, or sclerotherapy has been performed. However, recently, thorough preoperative examination, such as endoscopic ultrasonography (EUS), has facilitated endoscopic resection, such as EMR

Fukui, Yasuo

Hayashi, Hiroshige

Hibino, Shingo

Ito, Susumu

Iwaki, Hiroshi

Kimura, Eriko

Kimura, Yoshitaka

Muguruma, Naoki

Okamoto, Koichi

Okamura, Seisuke

Okita, Yoshio

Onose, Yukiko

Ozaki, Yuji

Sato, Kei

Sawada, Seizo

Sogabe, Masahiro

Taniki, Toshikatsu

Yoshida, Takahiro

Tokushima University Institutional Repository

INTRODUCTIONAmong benign tumors of the esophagus, esophagealhemangioma is relatively rare(1). However, the recentwidespread use of endoscopy andmedical examinationhas increased the incidence of this tumor. To treatesophageal hemangioma, esophagectomy or tumorenucleation has been performed, but recently, thenumber of endoscopic therapy is increasing. Endoscopicmucosal resection (EMR) of esophageal heman-gioma is rare. In this study, we report a patient withesophageal hemangioma that was detected at themedical examination and could be resected byendoscopic mucosal resection (EMR), and reviewthe literature.CASE REPORTCASE REPORTA patient with esophageal hemangioma treated by endo-scopic mucosal resection : a case report and review of theliteratureMasahiro Sogabe,1,3 Toshikatsu Taniki5, YasuoFukui, 5 TakahiroYoshida,2 KoichiOkamoto,4Yoshio Okita,3 Hiroshige Hayashi6, Eriko Kimuara3, Yoshitaka Kimura3, Yukiko Onose3,Yuji Ozaki,3 Hiroshi Iwaki3, Kei Sato3, Shingo Hibino3, Seizo Sawada3, Naoki Muguruma1,Seisuke Okamura1, and Susumu Ito11Department of Digestive and Cardiovascular Medicine, and 2Department of Oncological and RegenerativeSurgery, Institute of Health Biosciences, The University of Tokushima Graduate School, Tokushima, Japan;3Department of Internal Medicine, Anan Central Hospital of The Medical Association, Tokushima, Japan;4Department of Gastroenterology, and 5Department of Surgery, Kochi Health Sciences Center, Kochi, Japan;and 6Fukuda Clinic of Internal Medicine Heart and Digestive, Kochi, JapanAbstract : In a 58-year-old male, upper digestive endoscopy revealed a protruding lesion in theesophagus on a medical examination. The patient was referred to the Department of Surgeryin our hospital to undergo surgery. On the initial consultation, upper digestive endoscopyshowed a smooth, soft, black purple, typeⅡprotruding lesion measuring approximately 25mm at35 cm apart from the incisor. For diagnotic treatment and patient’s request, endoscopicmucosal resection (EMR) was performed. The resected specimen measured 25 mm× 25 mm.The histological findings suggested cavernous hemangioma. To treat esophageal hemangioma,esohagectomy, tumor enucleation, or sclerotherapy has been performed. However, recently,thorough preoperative examination, such as endoscopic ultrasonography (EUS), has facilitatedendoscopic resection, such as EMR.J. Med. Invest. 53 : 177-182, February, 2006Keywords : esophageal hemangioma, endoscopic mucosal resectionReceived for publication August 22, 2005 ; accepted November22, 2005.Address correspondence and reprint requests toMasahiro Sogabe,M.D., Department of Internal Medicine, Anan Central Hospitalof The Medicical Association, 2 Kawahara, Takarada-cho, Anan,Tokushima 774-0045, Japan and Fax : +81-88-423-6773.The Journal of Medical Investigation Vol. 53 2006１７７The patient was a 58-year-old male. There wasno complaint. The family history was not contributory.Concerning previous history, he underwent surgeryfor rectal cancer at the age of 50 years. In September2002, upper digestive endoscopy in a periodic medicalexamination revealed a protruding lesion in theesophagus. The patient was referred to the Departmentof Surgery in our hospital to undergo surgery. Forpreoperative endoscopy, the patient was referredto the Department of Internal Medicine. Concerningphysical examination on admission, height and bodyweightwere 162 cmand54kg, respectively. The physicalstatus was moderate, and nutritionwas good. Anemiawas not observed in the palpebral conjunctivae,and jaundice was not observed in the bulbar con-junctivae. There were no abnormal physical findingsin the thoracic or abdominal regions. Palpation didnot reveal any abnormalities in the superficiallymph nodes. There was no edema of the lowerthigh.Concerning blood biochemistry (Table 1), therewere no abnormalities in peripheral blood/bloodbiochemistry/blood coagulation test findings ortumor/viral markers.Esophagography (Fig. 1) showed an oval shadowdefect measuring approximately 22 mm×8mmwith asmooth surface at the anterior wall of the lower thoracicesophagus.Upper digestive endoscopy (Fig. 2) revealed asmooth, soft, black purple, type protruding lesionmeasuring approximately 25 mm at the anterior wallapproximately 35 cm from the incisor.On endoscopic ultrasonography (EUS)(Fig. 3),the main location of the lesion was visualized as aTable 1. Laboratory data on admissionHematological testWBC 8450/mm3RBC 440×104/mm3Hb 14.3 g/dlHt 41.6％Plt 31.9×104/mm3Urinary analysisProtein ( -)augar ( -)occult blood ( -)Serological testCRP 0.1 g/mdlBlood chemistryT-Bil 0.4 mg/dlGOT 30 IU/LGPT 26 IU/LLDH 330 IU/LALP 183 IU/Lγ-GTP 49 IU/LT-Cho 129 mg/dlT.P. 7.4 g/dlAlb 4.3 g/dlChE 142 IU/LBun 11 mg/dlCRNN 0.7 mg/dlNa 138 mEq/LK 4.2 mEq/LCl 98 mEq/LFBS 98 mEg/dlCoagulation testPT 10.9 secPT 96.4%APTT 31.4 secB.T. 2 : 00 minTumor markersSCC 0.5 ng/mlCYFRA 2.9 ng/mlViral markersRPR ( -)TPHA ( -)HBs-Ag ( -)HCV Ab ( -)Fig. 3．EUS showing hypoechoic～isoechoic mass, which islocated in the mucosal and submucosal layer.Fig.1．Esophagography showing a filling defect, measuring 22× 8 mm in size on the lower esophagus.Fig. 2．Endoscopic findings. (a) A showing black purple, type IIprotruding lesion with a smooth surface. (b) The lesion is soft.M. Sogabe, et al. Esophageal hemangioma treated by EMR１７８hypoechoic to isoechoic tumor involving mucosato the submucosal layer, and the muscularis proprialayer was intact. There was no continuity between thetumor and low echoic bands such as the blood vessels.Contrast-enhanced computed tomography (CT)of the thoracic region (Fig. 4) revealed a tumormeasuring approximately 8 to 10mmat an area adjacentto the anterior wall of the thoracic esophagus.After informed consent which progress observationor treatment was chosen was obtained, EMR (Fig. 5)was performed for diagnotic treatment and patient’srequest on October 30. Endoscopic esophageal mucosalresection(EEMR)-tube method was used as EMRmethod. Resectionwas successfulwithout complicationssuch as hemorrhage and perforation.The resected specimenmeasuring 25mm×25mm(Fig. 6). The soft surface was blue white to blue purple.The histopathological findings (Fig. 7) showedformation of a vascular lumen with irregular dilatationjust below the lamina muscularis mucosa, suggestingcavernous hemangioma.After EMR, there were no complications. Thepatient was discharged 1 week after EMR. Threemonths after EMR, upper digestive endoscopy did notreveal stenosis of the esophagus, residual hemangioma,or relapse.DISCUSSIONThe incidence of benign tumors of the esophagusis relatively low. Moersch & Harrington et al.(1)reported these tumors in 44 (0.6%) of 7,459 autopsycases. In addition, esophageal hemangioma wasobserved in only 3 patients (0.04%). In Japan, 83patients with esophageal hemangioma, includingour patient, have been reported in the literature(agendas were excluded because the details wereunclear). In 20 of these patients, endoscopic resectionwas performed (Table 2)(2-18). Procedures includedpolypectomy in 16 patients and EMR in 4 patients(11,15,16), as performed in our patient. There were16 males and 4 females, with a mean age of 62.4±11.4 years. Complaints consisted of hematemesis(hemorrhage of the digestive tract) in 1patient anddysphasia in 9 patients. In 7 patients, esophagealhemangioma was asymptomatic, as demonstratedFig. 4．Chest CT revealing a tumor at the anterior wall of thethoracic esophagus.Fig. 5．Endoscopic mucosal resection. (a) The tumor is snaredat the base which is injected with hypertonic saline and epi-nephrine solution. (b) Resection site without bleeding and per-foration.Fig. 6．The resected specimen measuring 25× 25 mm is bluewhite to blue purple.Fig. 7．A photomicrograph of the resected tumor showing formationof a vascular lumen with irregular dilation immediately below the laminamuscularis mucosaThe Journal of Medical Investigation Vol. 53 February 2006 １７９in the present patient. Locations consisted of thethoracic upper esophagus in 5 patients, thoracicmiddleesophagus in 5 patients, thoracic lower esophagusin 9 patients, and cervical esophagus in 1 patient.The macroscopic type was typein 3 patients,typein 10, and typein 7. The tumor size rangedfrom 5 to 30 mm. Histologically, cavernous hemangiomawas suggested in 7 patients, and capillary hemangiomain 13 patients.In diagnosing the presence of esophageal he-mangioma, esophagography facilitates visualizationin most patients. However, recent improvement inendoscopy has facilitated presence diagnosis. Onesophagography, most lesions are visualized assubmucosal tumors ; however, the characteristicsof esophageal hemangioma remain to be clarified.For definitive diagnosis, endoscopy is needed, andblue white to red purple soft lesions with a submu-cosal tumor-like morphologymay be readily diagnosed.Histological diagnosis by biopsy is useful;however,hemorrhage may occur after biopsy. Palchick et al.(19) and Hanel et al.(20) have reported that therewas no hemorrhage. Recently, several studies classifiedhemangioma by itsmorphology and color, and indicatedendoscopic polypectomy (2, 13). Sekimata et al.(13)classified patients with hemangioma into 5 groups,and reported that endoscopic polypectomy was notindicated for type/ lesions (Groups 1 and 2),considering the risks of morphology-associatedincomplete resection and postoperative hemorrhagefrom the residual stump of hemangioma. Recentstudies have reported qualitative diagnosis bymagneticresonance imaging (MRI) and EUS (10, 15, 16).The present patient was assigned to Group 1 accordingto the classification described by Sekimata et al.(13) However, pretreatment EUS facilitated confirmingthe main location of the lesion and the absence ofcontinuity with blood vessels ; therefore, EMR couldbe performed, considering that hemorrhage may notoccur.To treat esophageal hemangioma, esophagectomyor tumor enucleation has been performed. Recently,endoscopic sclerotherapy (21, 22) and laser therapy(23) have been reported. The number of patientsundergoing EMR or endoscopic polypectomy is in-creasing. Esophageal hemangioma frequently developsin the middle/lower esophagus. For surgery, tho-racotomy may be performed, which is stressful.Furthermore, endoscopic sclerotherapy may causehemorrhage on puncture, the side effects of infusedagents, and complications. Endoscopic resection is lessinvasive than this sclerotherapy, and advances ofendoscopic treatments and instruments may reducethe risk of serious complications. In the future, thisprocedure may be indicated for a larger number ofpatients. However, evaluation by various preoperativeTable 2. Characteristics of the reported cases of endoscopic resection for the treatment of esophageal hemangioma in JapanInvestigator (Year) Age Sex Chiefcomplain Location Form Size(mm)PathologicalfindingsVariety ofendscopictherapy1 Kawahara2 Oguchi（２）3 Kurashita4 Yoshioka(3)5 Arai(4)6 Kubo(5)7 Sato(6)8 Onozawa(7)9 Ohkubo(8)10 Inui(9)11 D.Cantero(10)12 Yoshikane(11)13 Sekimata(12)14 Uchida(13)15 Seki(14)16 Suematsu（１５）17 Araki(16)18 Tominaga(17)19 Our case20 Yamamoto(18)(1983)(1984)(1985)(1985)(1986)(1986)(1986)(1988)(1989)(1990)(1994)(1995)(1996)(1997)(1997)(1999)(1999)(2000)(2003)(2004)６０８２４１５７５６７６５０８２５５７２７２４９６６７０５１６９６０５９５８４４FMMMFMMMMMMMMFFMMMMMdysphagiadysphagia(-)hematemesisdysphagiadysphagia(-)dysphagiaabdominal paindysphagiachest pain(-)dysphagiadysphagiadysphagialoss of BW(-)(-)(-)(-)MtLtLtCeLtUtLtLtUtMtMtLtMtUtLtUtUtMtLtMtIVIIIIIIIIIIVIVIVIIIIIIIVIVIIIVIIIIIIIIIIIIIIIIIII9×8×718×11×86×5×524×23×1822×13×722×12×95×3×318×11×818×15×146×5×420×12×710×9×630×20×158×7×630×2015×715×1530256×5cap HAcap HAcap HAcav HAcav HAcap HAcap HAcap HAcap HAcap HAcap HAcav HAcap HAcap HAcav HAcap HAcav HAcav HAcav HAcap HAPolypectomyPolypectomyPolypectomyPolypectomyPolypectomyPolypectomyPolypectomyPolypectomyPolypectomyPolypectomyPolypectomyEMRPolypectomyPolypectomyPolypectomyEMREMRPolypectomyEMRPolypectomycap HA : capillary hemangioma, cav HA : cavernous hemangioma, BW : body weight, Ce : cervical esophagusUt : upper thoracic esophagus, Mt : middle thoracic esophagus, Lt : lower thoracic esophagusM. Sogabe, et al. Esophageal hemangioma treated by EMR１８０examinations, and close management are needed,as complications, such as hemorrhage and perforation,may occur.ACKNOWLEDGMENTSWe thank Satoshi Numoto (Departments of Pa-thology, Kochi Health Sciences Center) for patho-logical diagnosis.REFERENCES1. Moersch HJ, Harrington SW : Benign tumorof the. Ann Otol Rhnol Laryngol 53 : 800-817,19942. Oguchi S, Nabeya K, Onozawa K, Kobayashi Y,Suzuki N : A case of esophageal hemangiomapolypectomised endoscopically. GastroenterolEndosc 29 : 96-100, 19873. Yoshioka Y, Sakai K, Hamanaka Y, Hirata S,Higashino S, Hai M, Ohsugi H, Itoh S, Koizumi H,Maekawa N, So H, Ueno T, Kobayashi Y:Threecases of the esophageal hemangioma. Gastro-enterological Surgery 8 : 370-377, 19854. Arai K, Suzuki S, Tanio N, Fukushima M,Araida O, Hataya K, Kawamura K, Koizumi K,Koike T, Ishii J, Sugiyama Y, Suzuki : A case ofesophageal hemangioma. Progress of DigestiveEndoscopy 29 : 190-194, 19865. Kudo M, Hirasa M, Takakuwa H, Ibuki Y,Hujimi K, Miyamura M, Tomita S, Komori E,Todou A, Shirane H : A case of esophagealhemangioma excised by endoscopic polypectomy.Gastroenterol Endosc 28 : 318-323, 19866. Sato K, Asagi S, Koseki Y : A case of endo-scopically polypectomized capillary hemangiomaof the esophagus. Progress ofDigestive Endoscopy28 : 215-218, 19867. Onozawa K, Nabeya K, Motojima T, Kaku C,Kawaguchi T, Oguchi S, Arai Y : EndoscopicalPolypectomy for Esophageal Submucosal Tumors.J Jpn Bronchoesophagol Soc 39(in Japanese) :374-378, 19888. Ohkubo K, Nakano K, Seto K, Yamamoto S,Uno J, Fujisawa Y : A case of esophageal he-mangioma polypectomised endoscopically. EhimeIgaku 8(in Japanese) : 511-515, 19899. InuiM,Horie E : A case of esophagealhemangioma.-Endoscopic Polypectomy-Gastroenterol Endosc32 : 554-562, 199010. D Cantero, Yoshida T, Suzumi M, Tada M,Okita K:Esophageal hemangioma:Endoscopicdiagnosis and treatment. Endoscopy 26 : 250-253, 199411. Yoshikane H, Suzuki T, Yoshioka N, Ogawa Y,Ochi T, Hasegawa N:Hemangioma of theesophagus : endosonographic imaging andendoscopic resection. Endoscopy 27 : 267-269,199512. Sekimata T, Usuki H, Okada S, Ishimura K,Yatida S, Wakabayashi H, Wakabayashi H,Maeba T, Utida Y : A case of esophageal he-mangioma polypectomised endoscopically.Gastroenterol Endosc 41 : 1083-1089, 199913. Uchida N, Nagamachi Y, Takenoshita S,Hosouchi Y, Nakamura J, Fujita K : A case ofesophageal hemangioma. J Jpn Surg Assoc 58:1018-1022, 199714. Seki E, Gonda H, Fujii Y, Katsuura Y, Daibo M,Sakai Y, Okada T, Ouchi K, Iida A, Sakurai H,Tomiki Y, Urabe M, Sakakibara N : A case ofhemangioma of esophagus safely resectedwith adetachable snear. Progress of DigestiveEndoscopy 50 : 240-241, 199715. Suematsu M, Takeshita S, Oda Y, Kiyozumi T,Akashi R, SagaraK : A case of esophagealangiomaresected endoscopically. Endoscopia Digestiva11 : 948-952, 199916. Araki K, Ohno S, Egashira A, SaekiH,KawaguchiH,Ikeda Y, Kitamura K, Sugimachi K : Esopha-geal hemangioma : a case report and reviewof the literature. Hepatogastroenterology 46 :3148-3154, 199917. TominagaK, ArakawaT,AndoK,UmedaS,ShibaM,Suzuki N, Watanabe T, Takaishi O, Fujiwara Y,Uchida T, Fukuda T, Higuchi K, Kuroki T :Oesophageal cavernous haemangioma diagnosedhistologically, not by endoscopic procedures.J Gastroenterology and Hepatol 15 : 215-219,200018. Yamamoto S, Okazaki K:A case of esopahagealhemangioma. Clinical Gastroenterology 19 :1443-1435, 200419. Palchick BA, Alpert MA, Holmes RA, Tully RJ,Wilson RC : Esophageal Hemangioma ; Diagnosiswith Computed Tomography and RadionuclideAngiography. Southern Med J 76 : 1582-1584,198320. Hanel K, Talley NA, Hunt DR : Hemangiomaof the esophagus ; An unusual case of uppergastrointestinal bleeding. Dig Dis Sci 26 : 257-263, 1981The Journal of Medical Investigation Vol. 53 February 2006 １８１21. Nagata-Narumiya T, Nagai Y, Kashiwagi H,Hama M, Takifuji K, Tanimura H : Endoscopicsclerotherapy for esophageal hemangioma.Gastrointest Endosc 52 : 285-287, 200022. Aoki T, Okagawa K, Nishioka K, Miyata H,Ukei T, Miyauchi K, Terashima T, Kaneko T,Mizunoya S : Successful treatment of an esophagealhemangioma by endoscopic injection scle-rotherapy : report of a case. Surg Today 27 :450-452, 199723. Shigemitsu K, Naomoto Y, Yamatsuji T, Ono K,Aoki H, Haisa M, Tanaka N : Esophageal he-mangioma successfully treated by fulgurationusing potassium titanyl phosphate/yttriumaluminum garnet (KTP/YAG) laser : a casereport. Dis Esophagus 13 : 161-164, 2000M. Sogabe, et al. Esophageal hemangioma treated by EMR１８２

A patient with esophageal hemangioma treated by endoscopic mucosal resection : a case report and review of the literature

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A patient with esophageal hemangioma treated by endoscopic mucosal resection : a case report and review of the literature

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