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      Slow-growing amelanotic malignant melanoma of the esophagus with long survival: a case report and review of the literature

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          Abstract

          Background and study aims  We report a case of amelanotic malignant melanoma of the esophagus (AMME), an exceedingly rare disease. A 77-year-old Japanese woman presented to our hospital with features suggestive of a middle esophageal submucosal tumor, which was diagnosed pathologically as AMME. The patient underwent thoracoscopic resection of the esophagus and laparoscopic gastric tube reconstruction. Three years after surgery, computed tomography showed no recurrence. Generally, the prognosis of malignant melanoma of the esophagus is very poor. However, our patient had no recurrence, and is alive 3 years after surgery and 5 years after the tumor was first detected.

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          Primary malignant melanoma of the esophagus: a clinicopathologic study of a case with comprehensive literature review.

          Michele Bisceglia, Francesco Perri, Antonio Tucci … (2011)
          Primary malignant melanoma originating in the digestive tract is particularly rare, mainly affecting the anorectum and oral cavity. Primary malignant melanoma of the esophagus has been the subject mostly of case reports. This tumor has a dismal prognosis with a frequency estimated to be approximately 0.1% to 0.2% of all esophageal malignancies. According to the review by Volpin et al of November 2002, 238 cases of primary malignant melanoma of the esophagus have been published up to early 2001. We present an additional case of primary malignant melanoma of the esophagus of the amelanotic variant in a 69-year-old man. The patient was preoperatively investigated by esophageal endoscopy and endoscopic ultrasound. The surgically resected tumor specimen was examined histologically and supplemented by immunohistochemical and ultrastructural analysis. Intra-abdominal relapse occurred after 8 months at the site of surgery, necessitating repeat resection. The patient died of advanced intra-abdominal disease 14 months after the primary diagnosis. A comprehensive computerized (PubMed/Medline) review of the world literature was also carried out and 99 additional cases (after the review by Volpin et al) were found, 9 of them from the 1990s which escaped previous tabulations, and 90 from the years 2000 to 2010, amounting to a grand total of 337 ever published.
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            Primary amelanotic melanoma of the esophagus.

            A Nadales, R Valdez, M. Abbruzzese … (2006)
            Primary melanoma of the esophagus (PME) is an uncommon malignancy with less than 250 cases reported in the literature. Amelanotic PME is exceedingly rare and accounts for 10-25% of melanomas of the esophagus. A 59-year-old male with a history of mild dysphagia, heartburn, moderate anorexia and weight loss for 1 month is described. Barium swallow examination and videogastroscopy showed a polypoid, ulcerated mass located 30-38 cm from the incisors. No skin or eye melanoma lesions were found. Five biopsy samples were obtained. Histological examinations revealed proliferation of large, loosely cohesive cells of variable shapes and prominent central nucleoli in the deep mucosa. Immunohistochemical findings included positive vimentin, protein S-100, Melan A, and HMB-45, and negative AE1/AE3, CD 17, and desmin. A total transhiatal esophagectomy with high cervical esophagogastric anastomosis was performed. Peritumoral lymph nodes revealed malignant invasion. A diagnosis of primary amelanotic melanoma of the esophagus was made. Fourteen months after diagnosis the patient developed disseminated PME.
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              Amelanotic malignant melanoma of the esophagus: case report and review of the literature.

              T Toyoda, S Sakuramachi, M. Matsuda … (1990)
              A case of amelanotic malignant melanoma of the esophagus in a 76-year-old woman is reported. A whitish polypoid tumor, measuring 3 x 2 x 2.7 cm, surrounded by black pigmented mucosa, was detected in the middle intrathoracic esophagus. The tumor showed a lobulated surface lined by squamous cell layer, and had epithelioid and polyhedral cells forming alveolar clusters. Melanin pigments or stainability for the dihydroxyphenylalanine (DOPA) reaction were only observed in a few tumor cells. Junctional changes and mucosal melanosis, however, were found freely in the mucosa around the tumor. Many tumor cells showed a strongly positive immunohistochemical reaction for neuron specific enolase (NSE) and S100 protein. The patient died of widespread metastases six months after surgery. Further, a review of 106 reported cases of primary esophageal malignant melanoma, including 29 autopsies, was made; the melanomas were found to include 10 of amelanotic type, eight of which had been misdiagnosed at biopsy. Junctional changes could be found in the mucosa over or around the tumor, in four cases, and mucosal melanosis in one. Lymph node metastasis was the most frequently observed development at autopsy regardless of whether the tumor was amelanotic or melanotic. For correct diagnoses of melanomas of the amelanotic type, peripheral mucosal findings, such as junctional changes or melanosis, should be helpful; and, in order to obtain a good prognosis, a careful resection of the regional lymph nodes could prove valuable.
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                Author and article information

                Journal
                Journal ID (nlm-ta): Endosc Int Open
                Journal ID (iso-abbrev): Endosc Int Open
                Journal ID (doi): 10.1055/s-00025476
                Title: Endoscopy International Open
                Publisher: © Georg Thieme Verlag KG (Stuttgart · New York )
                ISSN (Print): 2364-3722
                ISSN (Electronic): 2196-9736
                Publication date (Print): November 2017
                Publication date (Electronic): 27 October 2017
                Volume: 5
                Issue: 11
                Pages: E1076-E1080
                Affiliations
                [1 ]Department of Endoscopy, Aichi Cancer Center Hospital, Aichi, Japan
                [2 ]Department of Gastroenterology, Aichi Cancer Center Hospital, Aichi, Japan
                [3 ]Department of Gastroenterological Surgery, Aichi Cancer Center Hospital, Aichi, Japan
                [4 ]Department of Pathology and Molecular Diagnosis, Aichi Cancer Center Hospital, Aichi, Japan
                Author notes
                Corresponding author Yutaka Hirayama Department of Endoscopy Aichi Cancer Center Hospital 1-1 Kanokoden, Chikusa-kuNagoya, Aichi 464-8681, Japan+81-52-764-2963 yhirayama@ 123456aichi-cc.jp
                Article
                DOI: 10.1055/s-0043-117951
                PMC ID: 5659865
                SO-VID: 4ddf32b7-e875-414b-ba6e-cd081217e8c2
                License:

                This is an open-access article distributed under the terms of the Creative Commons Attribution-NonCommercial-NoDerivatives License, which permits unrestricted reproduction and distribution, for non-commercial purposes only; and use and reproduction, but not distribution, of adapted material for non-commercial purposes only, provided the original work is properly cited.

                History
                Date received : 08 March 2017
                Date accepted : 26 June 2017
                Categories
                Subject: Case report

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