Anorectal melanoma is a rare malignant tumor with a poor prognosis. However, several studies have reported cases of long-term survival. In this report, we present a patient with anorectal melanoma who has survived for 9 years after endoscopic mucosal resection. An 85-year-old man was referred to our hospital for further examination and treatment of an anal tumor 2cm in size. Endoscopic ultrasonography
revealed that the depth of tumor invasion was confined to the submucosal layer. Endoscopic mucosal resection was performed, and the tumor was diagnosed as a malignant melanoma. The patient was followed without any additional treatment, which was per his wishes. Although melanoma recurred 4 times thereafter, endoscopic mucosal resection was performed for each recurrent lesion. Thus, he has been alive for 9 years since the first endoscopic mucosal resection without distant metastases.
If the depth of tumor invasion is shallow, endoscopic mucosal resection is a useful option among other therapeutic modalities.</p

Fujimoto, Tsuyoshi

Makino, Yasuhiro

Murakami, Ichiro

Ohta, Takeyuki

Tanaka, Shouichi

English

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Endoscopic Mucosal Resection of Primary Anorectal Malignant Melanoma:A Case Report

Okayama University Scientific Achievement Repository

Acta Medica OkayamaVolume 62, Issue 6 2008 Article 10DECEMBER 2008Endoscopic Mucosal Resection of PrimaryAnorectal Malignant Melanoma:A CaseReportShouichi Tanaka∗ Takeyuki Ohta† Tsuyoshi Fujimoto‡Yasuhiro Makino∗∗ Ichiro Murakami††∗Department of Gastroenterology, Iwakuni Clinical Center, National Hospital Organization,†Department of Gastroenterology, Iwakuni Clinical Center, National Hospital Organization,‡Department of Gastroenterology, Iwakuni Clinical Center, National Hospital Organization,∗∗Department of Gastroenterology, Iwakuni Clinical Center, National Hospital Organization,††Department of Pathology, Iwakuni Clinical Center, National Hospital Organization,Copyright c©1999 OKAYAMA UNIVERSITY MEDICAL SCHOOL. All rights reserved.Endoscopic Mucosal Resection of PrimaryAnorectal Malignant Melanoma:A CaseReport∗Shouichi Tanaka, Takeyuki Ohta, Tsuyoshi Fujimoto, Yasuhiro Makino, andIchiro MurakamiAbstractAnorectal melanoma is a rare malignant tumor with a poor prognosis. However, several stud-ies have reported cases of long-term survival. In this report, we present a patient with anorectalmelanoma who has survived for 9 years after endoscopic mucosal resection. An 85-year-old manwas referred to our hospital for further examination and treatment of an anal tumor 2cm in size.Endoscopic ultrasonography revealed that the depth of tumor invasion was confined to the sub-mucosal layer. Endoscopic mucosal resection was performed, and the tumor was diagnosed as amalignant melanoma. The patient was followed without any additional treatment, which was perhis wishes. Although melanoma recurred 4 times thereafter, endoscopic mucosal resection wasperformed for each recurrent lesion. Thus, he has been alive for 9 years since the first endoscopicmucosal resection without distant metastases. If the depth of tumor invasion is shallow, endoscopicmucosal resection is a useful option among other therapeutic modalities.KEYWORDS: malignant melanoma, anorectum, endoscopic mucosal resection (EMR), long-term survival∗Copyright c©2008 OKAYAMA UNIVERSITY MEDICAL SCHOOL. All rights reservedEndoscopic Mucosal Resection of Primary Anorectal  Malignant Melanoma: A Case ReportShouichi Tanakaa＊,  Takeyuki Ohtaa,  Tsuyoshi Fujimotoa,   Yasuhiro Makinoa,  and Ichiro MurakamibDepartments of aGastroenterology and bPathology,  Iwakuni Clinical Center,  National Hospital Organization,   Iwakuni,  Yamaguchi 740ﾝ0041,  JapanAnorectal melanoma is a rare malignant tumor with a poor prognosis.  However,  several studies have reported cases of long-term survival.  In this report,  we present a patient with anorectal melanoma who has survived for 9 years after endoscopic mucosal resection.  An 85ﾝyear-old man was referred to our hospital for further examination and treatment of an anal tumor 2cm in size.  Endoscopic ultraso-nography revealed that the depth of tumor invasion was conﬁned to the submucosal layer.  Endoscopic mucosal resection was performed,  and the tumor was diagnosed as a malignant melanoma.  The patient was followed without any additional treatment,  which was per his wishes.  Although melanoma recurred 4 times thereafter,  endoscopic mucosal resection was performed for each recurrent lesion.  Thus,  he has been alive for 9 years since the ﬁrst endoscopic mucosal resection without distant metas-tases.  If the depth of tumor invasion is shallow,  endoscopic mucosal resection is a useful option among other therapeutic modalities.Key words: malignant melanoma,  anorectum,  endoscopic mucosal resection (EMR),  long-term survivaln the gastrointestinal (GI) tract,  the anorectum is a site at which malignant melanoma arises frequently.  Anorectal malignant melanoma has been shown to result in worse prognoses than cutaneous melanoma [1].  However,  several studies [2ﾝ4] have reported cases of long-term survival when treatment consisted only of local surgical excision.  We herein present the case of a patient with anorectal malignant melanoma treated by endoscopic mucosal resection (EMR) alone.  He has survived for 9 years without distant metastases.Case Report　 An 85ﾝyear-old man visited an outside hospital with a chief complaint of anal bleeding in July 1999.  He underwent colonoscopy and an anal tumor was found.  He was admitted to our hospital for further examination and treatment.  His past medical history was signiﬁcant and included sigmoidectomy in 1986 for sigmoid colon cancer.  Physical examination and rou-tine blood tests revealed no major abnormalities.  Barium enema showed a nodular tumor about 2cm in size in the rectum adjacent to the anal verge (Fig.  1A).  Colonoscopic examination revealed a dark and nodular polypoid lesion about 2cm in size,  which was 1cm above the anal verge (Fig.  1B).  The tumor had a smooth surface,  and there was no evidence of erosion or ulceration.  A 20-MHz ultrasound catheter probe IActa Med.  Okayama,  2008Vol.  62,  No.  6,  pp.  421ﾝ424CopyrightⒸ 2008 by Okayama University Medical School.Case Report http ://escholarship.lib.okayama-u.ac.jp/amo/Received June 9, 2008 ; accepted August 22, 2008. ＊Corresponding author. Phone : ＋81ﾝ827ﾝ31ﾝ7121; Fax : ＋81ﾝ827ﾝ31ﾝ7059E-mail : tanaka@iwakuni-nh.go.jp (S. Tanaka)1Tanaka et al.: Endoscopic Mucosal Resection of Primary Anorectal Malignant MelanProduced by The Berkeley Electronic Press, 2008revealed a hypoechoic mass,  and indicated that the lesion was nearly conﬁned to the submucosal layer (Fig.  1C).  Whole body computed tomography and upper GI endoscopy did not reveal any other abnor-malities.　 The endoscopic biopsy specimen revealed a round cell tumor,  but a deﬁnitive diagnosis was not made.  Therefore,  EMR was performed to not only control the bleeding,  but also to make a deﬁnitive diagnosis.  The resection specimen,  measuring 22×18mm,  showed a black solid tumor that was covered with normal mucosa in a cut surface view (Fig.  2A).  Microscopic ﬁndings revealed diﬀuse inﬁltration of round or spindle-shaped tumor cells that contained melanin in the submucosal layer (Fig.  2B).  Immuno-histochemically,  tumor cells were positive for Human Melanin Black-45 (HMB-45) (Fig.  2C).  The ﬁnal diagnosis was a primary anorectal malignant melanoma 422 Acta Med.  Okayama　Vol. 62, No. 6   Tanaka et al.Ａ Ｂ ＣFig. 1　 A,  Barium enema image showing a nodular tumor about 2cm in size in the rectum adjacent to the anal verge (arrows); B,  Colonoscopic view of lesion,  demonstrating a dark and nodular polypoid lesion about 2cm in size at 1cm above the anal verge; C,  EUS image showing the lesion as a hypoechoic mass,  indicating it is almost conﬁned to the submucosal layer.  Arrows indicate the submucosal layer.Ａ Ｂ ＣFig. 2　 A,  A cut surface view of the resection specimen,  measuring 22×18mm,  showing a black solid tumor covered with normal mucosa; B,  Photomicrograph of resection specimen showing diﬀuse inﬁltration of round or spindle-shaped tumor cells containing melanin (arrows) in the submucosal layer (Hematoxylin and Eosin staining,×300); C,  Immunohistochemically,  tumor cells were positive for HMB-45 (HMB-45 staining,×400).2Acta Medica Okayama, Vol. 62 [2008], Iss. 6, Art. 10http://escholarship.lib.okayama-u.ac.jp/amo/vol62/iss6/10based on these histopathological features.  Tumor cells were not seen in the resected margin.  However,  both lymphatic and venous invasion were noted.  The patient was followed without any additional treatment,  because the patient and his family refused additional treatment.　 Local recurrence was conﬁrmed at the same site 1 year and 10 months after EMR.  The recurrent tumor,  3×4cm in size,  was larger than the ﬁrst tumor.  EMR was performed for the recurrent tumor because the patient and his family refused other options.  The resection specimen was histopathologically similar to the ﬁrst tumor.　 Moreover,  3 years and 2 months after the ﬁrst EMR,  melanoma recurred at the site adjacent to the ﬁrst tumor.  This tumor was 1.5cm in size,  and EMR was performed.  To date there have been 2 more local recurrences,  but EMR was performed for each lesion.  The patient remains well without distant metastases.Discussion　 The GI tract is the third most common site of occurrence of malignant melanoma,  after the skin and eyes [3].  In the GI tract,  the anorectum is a site at which malignant melanoma arises frequently,  because of the presence of abundant melanocytes in the mucosa of the anal canal [5].  However,  anorectal malignant melanoma is a rare disease,  since melanoma accounts for less than 1ｵ of primary anorectal malignancies [6].　 Anorectal malignant melanoma is thought to have a poor prognosis because it is often misdiagnosed for hemorrhoids,  which causes a delay in making the cor-rect diagnosis [1,  3].  Overall,  the 5ﾝyear survival rate is reported to be 10ｵ or less even if radical surgery (abdominoperineal resection) and chemother-apy are performed [6].　 However,  several studies have reported cases of long-term survival.  For example,  Malik et al.  [3] reported the case of a patient with a 5.75-mm thick anorectal melanoma who survived for 10 years after wide local excision.  Whooley et al.  [7] reported a 5ﾝyear survivor after abdominoperineal resection and chemotherapy for a 2.5-mm thick anorectal melanoma.  It has been shown that an important determinant of prognosis for any primary melanoma is the tumor thickness.  Speciﬁcally,  “thin” anorectal melanomas (＜1mm) have a more favorable prognosis than thicker anorectal melanomas [8,  9].  However,  there are cases of long-term survival in spite of the presence of thick lesions (＞4mm) or lesions of intermediate thick-ness (1ﾝ4mm).  With regard to cutaneous melanomas,  Clark et al.  [10] reported that the invasive radial growth phase of melanoma is indolent and incapable of metastasis.  Histologically,  it is characterized by scat-tered foci of papillary dermal invasion that are only comprised of scattered cells or small clusters,  and with only rare mitotic ﬁgures.  Whooley et al.  [7] proposed that these histological ﬁndings might also be related to the prognosis for anorectal melanoma.  In the present case,  it is possible that this tumor pos-sessed the histological feature of being incapable of metastasis in addition to a small volume of tumor cells.　 In Japan,  approximately 15 cases of survival longer than 5 years have been reported [11,  12].  According to an analysis of these long-term survivors,  the condi-tions for longer survival were a tumor diameter of less than 5cm and a tumor depth of invasion conﬁned to the muscularis propria.　 EMR for primary malignant melanoma in the GI tract has been performed in only 5 cases [2,  13ﾝ15] including ours.  These cases are summarized in Table 1.  Relatively long-term survival was achieved in most cases.  Among these cases,  only our case involved no additional treatment after EMR.　 In the present case,  local recurrence has occurred four times so far.  EMR was performed for each recurrent lesion.  The patient is alive with no evidence of distant metastases even though 9 years have passed since the ﬁrst EMR.  Endoscopic ultrasonography (EUS) for the ﬁrst lesion conﬁrmed that tumor inva-sion was conﬁned to the submucosa.  Examination of the specimen resected by EMR conﬁrmed the negative margins.  Therefore,  this case is thought to have met the conditions for long-term survival.  However,  in the future,  close follow-up for local recurrence including distant metastases is necessary.　 Long-term survival is expected,  even if less inva-sive surgery such as EMR is performed,  in cases of anorectal melanoma in which a shallow depth of inva-sion is conﬁrmed by EUS.  Thus,  the staging system and selection of therapeutic modalities should be based on tumor size and depth of invasion.423EMR of Anorectal MelanomaDecember 20083Tanaka et al.: Endoscopic Mucosal Resection of Primary Anorectal Malignant MelanProduced by The Berkeley Electronic Press, 2008References 1. Wong JH,  Cagle LA,  Storm FK and Morton DL: Natural history of surgically treated mucosal melanoma.  Am J Surg (1987) 154: 54ﾝ57. 2. Nishina T,  Kunieda K,  Miya K,  Saji S,  Shimokawa K,  Katada M and Watanabe K: Two cases of anorectal malignant melanoma showing prolonged survival after local additional excision followed by biopsy and abdomino-perineal resection followed by incisional biopsy.  Jpn J Gastroenterol Surg (2001) 34: 292ﾝ296 (in Japanese). 3. Malik A,  Hull TL and Milsom J: Long-term survivor of anorectal melanoma: report of a case.  Dis Colon Rectum (2002) 45: 1412ﾝ1417. 4. Bullard KM,  Tuttle TM,  Rothenberger DA,  Madoﬀ RD,  Baxter NN,  Finne CO and Spencer MP: Surgical therapy for anorectal mela-noma.  J Am Coll Surg (2003) 196: 206ﾝ211. 5. Alazmi WM,  Nehme OS,  Regalado JJ and Rogers AI: Primary gastric melanoma presenting as a nonhealing ulcer.  Gastrointest Endosc (2003) 57: 431ﾝ433. 6. Weinstock MA: Epidemiology and prognosis of anorectal mela-noma.  Gastroenterology (1993) 104: 174ﾝ178. 7. Whooley BP,  Shaw P,  Astrow AB,  Toth IR and Wallack MK:Long-term survival after locally aggressive anorectal melanoma.  Am Surg (1998) 64: 245ﾝ251. 8. Wanebo HJ,  Woodruﬀ JM,  Farr GH and Quan SH: Anorectal mel-anoma.  Cancer (1981) 47: 1891ﾝ1900. 9. Angeras U,  Jonsson N and Jonsson PE: Primary anorectal malig-nant melanoma.  J Surg Oncol (1983) 22: 261ﾝ264.10. Clark WH Jr,  Elder DE,  Guerry Ⅳ D,  Braitman LE,  Trock BJ,  Schultz D,  Synnestvedt M and Halpern AC: Model predicting sur-vival in stage I melanoma based on tumor progression.  J Natl Cancer Inst (1989) 81: 1893ﾝ1904.11. Hara H,  Asano M,  Asai S,  Kato Y,  Furukawa S and Ando H: A case report of prolonged survival of anorectal melanoma.  Jpn J Gastroenterol Surg (1992) 25: 2046ﾝ2049 (in Japanese).12. Shimada K,  Chubachi S,  Ichinohe F,  Satoh M,  Sugawara K,  Rokkaku Y,  Aono G,  Kanno M,  Tsuchiya A and Ohara T: A long-surviving patients with concurrent malignant melanoma of the ano-rectal region and early gastric cancer-a case report and review of the Japanese literature-.  Jpn J Cancer Clin (1996) 42: 1109ﾝ1119 (in Japanese).13. Herman J,  Duda M,  Lovecek M and Svach I: Primary malignant melanoma of the esophagus treated by endoscopic ablation and interferon therapy.  Dis Esophagus (2001) 14: 239ﾝ240.14. Fukui R,  Hata F,  Yasoshima T,  Honma T,  Nomura H,  Sasaki K,  Nishimori H,  Sasaki K,  Kutomi G,  Ohno K,  Oikawa I,  Sasaki K,  Tarumi K,  Ishiyama Y,  Kobayashi K,  Yamamoto N and Hirata K:Malignant melanoma of the anorectum: report of four cases.  Surg Today (2002) 32: 555ﾝ558.15. Hirose T,  Idee Y,  Hanashi T,  Yoshida M,  Kato H,  Monma K,  Funada N and Koike M: Malignant melanoma of the esophagus,  report of a case.  Stomach and Intestine (2002) 37: 1361ﾝ1365 (in Japanese).424 Acta Med.  Okayama　Vol. 62, No. 6   Tanaka et al.Table 1　 Reported cases in which EMR was performed for primary malignant melanoma in the GI tractNo. Authors(year)Age/Sex Location Size(mm)Depth AdditionaltreatmentPrognosis1 Hermanet al. [13](2001)55/M Esophagus 50 － IFN§ therapy 25 months2 Nishinaet al. [2](2001)76/M Anorectum 10 M† Local excision 8 years3 Fukuiet al. [14](2002)54/F Anorectum 6 － APR¶＋chemotherapyMore than6 years4 Hiroseet al. [15](2002)57/M Esophagus 33 SM‡ Irradiation 12 months5 Ourcase(2008)85/M Anorectum 22×18 SM　 None 9 years†M,  invasion conﬁned to the mucosal layer;‡SM,  invasion conﬁned to the submucosal layer;§IFN,  interferon;¶APR,  abdominoperineal resection.4Acta Medica Okayama, Vol. 62 [2008], Iss. 6, Art. 10http://escholarship.lib.okayama-u.ac.jp/amo/vol62/iss6/10

Endoscopic Mucosal Resection of Primary Anorectal Malignant Melanoma:A Case Report

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