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Ando M, Enomoto N, Nohara K, Yagi S, Kato D, Koda H, Yamada K, Kokudo N. Primary malignant melanoma of the esophagus arising from melanocytosis: a case report and literature review. Clin J Gastroenterol 2025:10.1007/s12328-025-02129-8. [PMID: 40377881 DOI: 10.1007/s12328-025-02129-8] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/13/2024] [Accepted: 04/02/2025] [Indexed: 05/18/2025]
Abstract
Primary malignant melanoma of the esophagus (PMME), a rare histologic type of esophageal cancer, accounts for 0.1-0.8% of all primary esophageal tumors. Esophagectomy is the main treatment in the absence of distant metastasis. Black lesions in esophageal mucosa (melanosis) and presence of melanocytes in biopsy specimen (melanocytosis) may be precancerous but have rarely been demonstrated to be associated with PMME so far. Here, we present a case of PMME arising from melanocytosis during follow-up, and conduct a literature review of cases in which PMME developed during the follow-up of melanosis. A 79-year-old woman underwent esophagogastroduodenoscopy, which revealed a black pigmented lesion in the esophagus. Biopsy results revealed melanocytes without atypia, indicating melanocytosis. One-year follow-up revealed that the lesion had enlarged, and she was referred to our hospital. Further evaluation confirmed the diagnosis of malignant melanoma without metastasis. Thoracoscopic esophagectomy was performed, and retrosternal reconstruction was made with a gastric conduit. The histopathologic evaluation confirmed PMME. The patient experienced minor postoperative anastomotic leakage but recovered with conservative treatment. No recurrence was detected at last follow-up 2 years after surgery.
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Affiliation(s)
- Makoto Ando
- Department of Surgery, National Center for Global Health and Medicine, 1-21-1 Toyama, Shinjuku-ku, Tokyo, 162-8655, Japan
| | - Naoki Enomoto
- Department of Surgery, National Center for Global Health and Medicine, 1-21-1 Toyama, Shinjuku-ku, Tokyo, 162-8655, Japan
| | - Kyoko Nohara
- Department of Surgery, National Center for Global Health and Medicine, 1-21-1 Toyama, Shinjuku-ku, Tokyo, 162-8655, Japan
| | - Shusuke Yagi
- Department of Surgery, National Center for Global Health and Medicine, 1-21-1 Toyama, Shinjuku-ku, Tokyo, 162-8655, Japan
| | - Daiki Kato
- Department of Surgery, National Center for Global Health and Medicine, 1-21-1 Toyama, Shinjuku-ku, Tokyo, 162-8655, Japan
| | - Hanako Koda
- Department of Surgery, National Center for Global Health and Medicine, 1-21-1 Toyama, Shinjuku-ku, Tokyo, 162-8655, Japan
| | - Kazuhiko Yamada
- Department of Surgery, National Center for Global Health and Medicine, 1-21-1 Toyama, Shinjuku-ku, Tokyo, 162-8655, Japan.
| | - Norihiro Kokudo
- Department of Surgery, National Center for Global Health and Medicine, 1-21-1 Toyama, Shinjuku-ku, Tokyo, 162-8655, Japan
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Kazacheuskaya L, Arora K. Esophageal melanosis: Two case reports and review of literature. World J Gastroenterol 2024; 30:4557-4565. [PMID: 39563752 PMCID: PMC11572630 DOI: 10.3748/wjg.v30.i42.4557] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/26/2024] [Revised: 09/29/2024] [Accepted: 10/14/2024] [Indexed: 10/31/2024] Open
Abstract
BACKGROUND Esophageal melanosis (EM) is a rare condition characterized by melanin pigmentation in the esophageal mucosa. It is not well understood and has been documented in less than 100 cases worldwide. CASE SUMMARY We report two cases of African American patients who complained of significant weight loss (over 20 pounds in approximately six months) and abdominal pain during their first visit. The first case involves a 54-year female with a history of hepatic steatosis and polysubstance abuse, who also experiences nausea and vomiting. The second case is a 59-year-old male with hypertension and gastroesophageal reflux disease (GERD), who was diagnosed with esophageal squamous cell carcinoma. Both cases show benign melanocytes in the basal layer on the esophagus biopsy and are diagnosed as EM. CONCLUSION It is important to note that EM has been associated with malignancies such as carcinoma and melanoma. Therefore, accurate diagnosis and appropriate management are crucial. Patients with EM, especially those with concurrent risk factors (e.g., GERD, smoking), should be carefully monitored for any signs of malignancy.
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Affiliation(s)
- Liubou Kazacheuskaya
- Department of Pathology, Louisiana State University Health, Shreveport, LA 71103, United States
| | - Kshitij Arora
- Department of Pathology, Louisiana State University Health, Shreveport, LA 71103, United States
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3
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Cazzato G, Cascardi E, Colagrande A, Lettini T, Resta L, Bizzoca C, Arezzo F, Loizzi V, Dellino M, Cormio G, Casatta N, Lupo C, Scillimati A, Scacco S, Parente P, Lospalluti L, Ingravallo G. The Thousand Faces of Malignant Melanoma: A Systematic Review of the Primary Malignant Melanoma of the Esophagus. Cancers (Basel) 2022; 14:3725. [PMID: 35954389 PMCID: PMC9367585 DOI: 10.3390/cancers14153725] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/08/2022] [Revised: 07/27/2022] [Accepted: 07/28/2022] [Indexed: 02/01/2023] Open
Abstract
Primary Malignant Melanoma of the Esophagus (PMME) is an extremely rare cancer of the esophagus, accounting for 0.1−0.8% of all oro-esophageal cancers and <0.05% of all melanoma subtypes, with an estimated incidence of 0.0036 cases per million/year. We conduct a careful analysis of the literature starting from 1906 to the beginning of 2022, searching the PubMed, Science.gov, Scopus and Web of Science (WoS) databases. A total of 457 records were initially identified in the literature search, of which 17 were duplicates. After screening for eligibility and inclusion criteria, 303 publications were ultimately included, related to 347 patients with PMME. PMME represents a very rare entity whose very existence has been the subject of debate for a long time. Over time, an increasing number of cases have been reported in the literature, leading to an increase in knowledge and laying the foundations for a discussion on the treatment of this pathology, which still remains largely represented by surgery. In recent times, the possibility of discovering greater mutations in gene hotspots has made it possible to develop new therapeutic strategies of which nivolumab is an example. Future studies with large case series, with clinicopathological and molecular data, will be necessary to improve the outcome of patients with PMME.
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Affiliation(s)
- Gerardo Cazzato
- Section of Pathology, Department of Emergency and Organ Transplantation (DETO), University of Bari “Aldo Moro”, 70124 Bari, Italy; (A.C.); (T.L.); (L.R.); (G.I.)
| | - Eliano Cascardi
- Department of Medical Sciences, University of Turin, 10124 Turin, Italy;
- Pathology Unit, FPO-IRCCS Candiolo Cancer Institute, Str. Provinciale 142 lm 3.95, 10060 Candiolo, Italy
| | - Anna Colagrande
- Section of Pathology, Department of Emergency and Organ Transplantation (DETO), University of Bari “Aldo Moro”, 70124 Bari, Italy; (A.C.); (T.L.); (L.R.); (G.I.)
| | - Teresa Lettini
- Section of Pathology, Department of Emergency and Organ Transplantation (DETO), University of Bari “Aldo Moro”, 70124 Bari, Italy; (A.C.); (T.L.); (L.R.); (G.I.)
| | - Leonardo Resta
- Section of Pathology, Department of Emergency and Organ Transplantation (DETO), University of Bari “Aldo Moro”, 70124 Bari, Italy; (A.C.); (T.L.); (L.R.); (G.I.)
| | - Cinzia Bizzoca
- Section of General Surgery, Azienda Ospedaliero-Universitaria Policlinico di Bari, 70124 Bari, Italy;
| | - Francesca Arezzo
- Section of Gynecology and Obstetrics, Department of Biomedical Sciences and Human Oncology (DIMO), University of Bari “Aldo Moro”, 70124 Bari, Italy; (F.A.); (V.L.); (M.D.)
| | - Vera Loizzi
- Section of Gynecology and Obstetrics, Department of Biomedical Sciences and Human Oncology (DIMO), University of Bari “Aldo Moro”, 70124 Bari, Italy; (F.A.); (V.L.); (M.D.)
| | - Miriam Dellino
- Section of Gynecology and Obstetrics, Department of Biomedical Sciences and Human Oncology (DIMO), University of Bari “Aldo Moro”, 70124 Bari, Italy; (F.A.); (V.L.); (M.D.)
| | - Gennaro Cormio
- Oncology Unit IRCSS Istituto Tumori “Giovanni Paolo II”, Department of Interdisciplinary Medicine (DIM), University of Bari “Aldo Moro”, 70124 Bari, Italy;
| | - Nadia Casatta
- Innovation Department, Diapath S.p.A., Via Savoldini n.71, 24057 Martinengo, Italy; (N.C.); (C.L.)
| | - Carmelo Lupo
- Innovation Department, Diapath S.p.A., Via Savoldini n.71, 24057 Martinengo, Italy; (N.C.); (C.L.)
| | - Antonio Scillimati
- Department of Pharmacy-Pharmaceutical Sciences, University of Bari “Aldo Moro”, 70124 Bari, Italy;
| | - Salvatore Scacco
- Department of Basic Medical Sciences and Neurosciences, University of Bari “Aldo Moro”, 70124 Bari, Italy;
| | - Paola Parente
- Unit of Pathology, Fondazione IRCCS Ospedale Casa Sollievo della Sofferenza, 71013 San Giovanni Rotondo, Italy;
| | - Lucia Lospalluti
- Section of Dermatology and Venereology, Department of Biomedical Sciences and Human Oncology (DIMO), University of Bari “Aldo Moro”, 70124 Bari, Italy;
| | - Giuseppe Ingravallo
- Section of Pathology, Department of Emergency and Organ Transplantation (DETO), University of Bari “Aldo Moro”, 70124 Bari, Italy; (A.C.); (T.L.); (L.R.); (G.I.)
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Esophageal Melanocytosis: report of two cases and review of a rare and misunderstood entity. Acta Gastroenterol Belg 2022; 85:390-392. [DOI: 10.51821/85.2.10126] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/11/2022]
Abstract
Esophageal melanocytosis (EM) is a rare entity, which is characterized by a non-atypical melanocytic proliferation and melanin deposits in the esophageal mucosa. The confusion between the terms of melanosis and melanocytosis in the literature, the rarity of this lesion (less than 50 cases reported in the literature), its uncertain pathobiological course and the lack of experience of pathologists and gastroenterologists prompt us to draw the attention to this particular entity by reporting two cases and reviewing the literature. Magnifying endoscopy to observe intensive melanin accumulation followed by a biopsy are key for the diagnosis.
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Chu YM, Hung CS, Huang CS. Primary malignant melanoma of the esophagogastric junction: A case report. Medicine (Baltimore) 2021; 100:e26467. [PMID: 34160452 PMCID: PMC8238344 DOI: 10.1097/md.0000000000026467] [Citation(s) in RCA: 4] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/20/2021] [Accepted: 06/07/2021] [Indexed: 01/04/2023] Open
Abstract
RATIONALE Most gastrointestinal melanomas are metastatic from an oculocutaneous primary lesion; however, primary gastrointestinal melanomas have been found in all levels of the gastrointestinal tract. We present the case of Primary malignant melanoma of the esophagus and discuss the diagnostic methods, differentiation from metastatic lesions and treatment options. PATIENT CONCERNS A 78-year-old male patient presented with fresh blood vomiting and tarry stools for 1 day. DIAGNOSES Esophagogastroduodenoscopy of this patient revealed a tumor ∼4 cm in size at the cardia side of the esophagogastric junction with dark-red and gray pigmentation. Immunohistochemical stains of the biopsy specimens were positive for S-100 and HMB-45, which are specific markers of melanoma. INTERVENTIONS Laparotomy with proximal gastrectomy was performed by the surgeon. Histological examination of the surgical specimen revealed the tumor arose from the distal esophagus with invasion of the proximal stomach. Primary malignant melanoma of the esophagus was diagnosed after a full skin and ophthalmic examination and positron emission tomography, which revealed no lesions elsewhere in the body. OUTCOMES No tumor recurrence was noted at the 1-year follow-up. LESSONS Primary malignant melanoma of the esophagus is an extremely rare but highly aggressive tumor. The special pattern of pigmentation should be recognized while performing endoscopy. Early detection and radical resection of the tumor are critical to ensure favorable outcomes.
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Affiliation(s)
- Yu-Ming Chu
- Division of Digestive Medicine, Department of Internal Medicine, Cathay General Hospital
| | - Chih-Sheng Hung
- Division of Digestive Medicine, Department of Internal Medicine, Cathay General Hospital
- School of Medicine, Fu-Jen Catholic University, New Taipei City
| | - Ching-Shui Huang
- Division of General Surgery, Department of Surgery, Cathay General Hospital, Taipei City, Taiwan
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Abstract
Primary malignant melanoma of esophagus (PMME) is a rare malignant tumor of esophagus. This study aimed to investigate the clinic pathologic characteristics and analyze the factors that might affect the prognosis of PMME patients.A total of 20 PMME patients who underwent surgical treatment in our hospital from 1975 to 2017 were analyzed. The clinical data, surgical and pathologic features of all patients were collected.For 20 PMME patients, the average age was 57.3 ± 10.7 years, and the male patients account for 75%. Most of the tumors (95%) were located in the middle and lower of the esophagus. There were 7 patients with primary tumor invasion beyond the muscular layer (T3 + T4) and 10 patients with lymph node metastasis (LNM). The median survival time of 20 patients was 12 months, and the 1-year and 5-year survival rates were 50% and 16.9%, respectively. The probability of LNM in tumors confined to submucosa (T1) and myometrium (T2) was lower than that in tumors with deeper invasion (T3, T4) (P = .035). Multivariate analysis showed that tumor node metastasis (TNM) staging was the independent prognostic factor for survival of PMME patients (hazard ratio [95% confidence interval], 4.15 [1.36-12.67]; P = .012).For PMME patients, tumors with deeper invasion were more likely to have LNM, and TNM staging was an independent predictor of prognosis for survival. Early detection of the disease and radical resection of the tumor are critical for better survival of the PMME patients.
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7
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Rakesh CR, Sharma AK, Pathak N, Gahlot GPS. Primary malignant melanoma of the gastroesophageal junction: A rare and unusual disease. Med J Armed Forces India 2020; 77:367-370. [PMID: 34305293 DOI: 10.1016/j.mjafi.2020.03.011] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/19/2017] [Accepted: 03/24/2020] [Indexed: 10/24/2022] Open
Abstract
Malignant melanoma, an aggressive tumor of skin, is also seen rarely in extra cutaneous sites like the gastrointestinal tract (GIT). Primary melanoma of the GIT by itself is a rare tumor; often metastatic at presentation and if found non-metastatic, it is rarely resectable. We are reporting a histopathologically confirmed case of primary malignant melanoma affecting the 'gastroesophageal junction', which we operated on.
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Affiliation(s)
- C R Rakesh
- Classified Specialist (Surgery & GI Surgeon), Army Hospital (R&R), New Delhi, India
| | - A K Sharma
- Senior Advisor (Surgery & GI Surgeon), Army Hospital (R&R), New Delhi, India
| | - Nishant Pathak
- Classified Specialist (Surgery & GI Surgeon), Military Hospital Jaipur, India
| | - G P S Gahlot
- Classified Specialist (Pathology & Oncopath), Army Hospital (R&R), New Delhi, India
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8
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Hashimoto T, Makino T, Yamasaki M, Tanaka K, Miyazaki Y, Takahashi T, Kurokawa Y, Motoori M, Kimura Y, Nakajima K, Morii E, Mori M, Doki Y. Clinicopathological characteristics and survival of primary malignant melanoma of the esophagus. Oncol Lett 2019; 18:1872-1880. [PMID: 31423256 PMCID: PMC6614672 DOI: 10.3892/ol.2019.10519] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/22/2019] [Accepted: 05/13/2019] [Indexed: 11/06/2022] Open
Abstract
Primary malignant melanoma of the esophagus (PMME) has been reported to be a rare and highly malignant disease, and to date a standard treatment strategy has not been established due to limited evidence. The aim of the present study was to investigate the clinicopathological characteristics of this extremely rare disease. A total of 6 out of 2,093 patients with PMME treated in our institution between 1995 and 2016 were retrospectively analyzed and their clinicopathological parameters including treatment course and long-term survival were investigated. The major clinicopathological characteristics of patients were that they were >70 years of age, male sex, dysphagia at first diagnosis, and macroscopic black protruding tumors located in the lower third of the thoracic esophagus. Four of the five patients receiving pretherapeutic endoscopic biopsy were correctly diagnosed with PMME, and two patients received preoperative treatment with ineffective histopathological responses. There were two unresectable cases, one was treated with an immune-checkpoint inhibitor and the other received palliative care. Three of the four patients receiving curative surgery developed hematogenous recurrence within two years of surgery and only one patient with pT1aN0M0 achieved long-term survival. The median overall survival of all six patients was 19.6 (6.4–40.5) months. Patients with stage I disease exhibited significantly more favorable prognoses than those with stage II–IV (P=0.025) and surgically-treated patients had significantly better prognoses than those who did not receive surgery (P=0.018). In conclusion, PMME was associated with highly malignant features and tended to develop hematogenous metastases even after radical resection. Early diagnosis appears to be important to cure this refractory disease.
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Affiliation(s)
- Tadayoshi Hashimoto
- Department of Gastroenterological Surgery, Osaka University Graduate School of Medicine, Osaka 565-0871, Japan
| | - Tomoki Makino
- Department of Gastroenterological Surgery, Osaka University Graduate School of Medicine, Osaka 565-0871, Japan
| | - Makoto Yamasaki
- Department of Gastroenterological Surgery, Osaka University Graduate School of Medicine, Osaka 565-0871, Japan
| | - Koji Tanaka
- Department of Gastroenterological Surgery, Osaka University Graduate School of Medicine, Osaka 565-0871, Japan
| | - Yasuhiro Miyazaki
- Department of Gastroenterological Surgery, Osaka University Graduate School of Medicine, Osaka 565-0871, Japan
| | - Tsuyoshi Takahashi
- Department of Gastroenterological Surgery, Osaka University Graduate School of Medicine, Osaka 565-0871, Japan
| | - Yukinori Kurokawa
- Department of Gastroenterological Surgery, Osaka University Graduate School of Medicine, Osaka 565-0871, Japan
| | - Masaaki Motoori
- Department of Surgery, Osaka General Medical Center, Osaka 558-8558, Japan
| | - Yutaka Kimura
- Department of Surgery, Kindai University Faculty of Medicine, Osaka 589-8511, Japan
| | - Kiyokazu Nakajima
- Department of Gastroenterological Surgery, Osaka University Graduate School of Medicine, Osaka 565-0871, Japan
| | - Eiichi Morii
- Department of Pathology, Graduate School of Medicine, Osaka University, Osaka 565-0871, Japan
| | - Masaki Mori
- Department of Surgery and Science, Graduate School of Medical Sciences, Kyushu University, Fukuoka 812-8582, Japan
| | - Yuichiro Doki
- Department of Gastroenterological Surgery, Osaka University Graduate School of Medicine, Osaka 565-0871, Japan
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9
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Kozan R, Tatar Z. An unusual localization; esophageal melanocytosis. Turk J Surg 2018; 37:76-79. [PMID: 34585099 DOI: 10.5152/turkjsurg.2017.3807] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/03/2017] [Accepted: 05/11/2017] [Indexed: 11/22/2022]
Abstract
Esophageal melanocytosis is a rare clinical and pathological condition characterized by non-atypical melanocytic proliferation and increased melanin in the esophageal mucosa, which is normally histologically non-melanocytic. Intensive melanin accumulation and hyperpigmentation are necessary for endoscopic recognition. Due to the fact that it is a rare gastrointestinal system pathology, experience and knowledge about its diagnosis, treatment and course are also limited. Although it is argued that chronic stimulating factors have an influence, there is no clear information about its etiology and pathogenesis. Malignant melanomas and melanocytic nevus in particular come to the fore in the differential diagnosis. Opinions and findings indicating that melanocytosis may be a precursor for malignant melanoma make the recognition and follow-up of this clinical and pathological entity more important. In this article, a patient with esophageal melanocytosis diagnosed by endoscopic evaluation is presented, with the aim of increasing the awareness of clinicians, especially endoscopists and pathologists, on this subject.
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Affiliation(s)
- Ramazan Kozan
- Clinic of General Surgery, Eren Hospital, İstanbul, Turkey
| | - Zeynep Tatar
- Division of Pathology, Patomer Pathology Center, İstanbul, Turkey
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10
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Sun H, Gong L, Zhao G, Zhan H, Meng B, Yu Z, Pan Z. Clinicopathological characteristics, staging classification, and survival outcomes of primary malignant melanoma of the esophagus. J Surg Oncol 2017; 117:588-596. [PMID: 29266237 DOI: 10.1002/jso.24905] [Citation(s) in RCA: 11] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/27/2017] [Revised: 09/19/2017] [Accepted: 10/11/2017] [Indexed: 12/14/2022]
Abstract
BACKGROUND Primary malignant melanoma of esophagus (PMME) is a remarkably rare and highly aggressive tumor. Studies related with clinicopathological findings, staging classification, and clinical outcomes are lacking. METHODS We reviewed 21 cases of PMME at the Tianjin Medical University Cancer Institute and Hospital from January 2002 to February 2017. RESULTS Nineteen patients (90.48%) presented a history of dysphagia for months, and two (9.52%) experienced retrosternal pain. Histologically, tumors were composed of atypical melanocytes with melanocytosis surrounding the tumor. The overall survival was 1-40 months, with the median time of 10 months. The mucosal staging classification for upper aerodigestive tract showed better distribution of overall survival with different stages than that of the American Joint Commission on Cancer staging classification for esophagus, but without statistical difference. Both the clinical and pathological characteristics were not highly consistent with overall survival. CONCLUSIONS PMME is a considerably aggressive tumor with poor prognosis. The staging classification of mucosal melanoma of the upper aerodigestive tract may be a good option for PMME patients.
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Affiliation(s)
- Haiyan Sun
- Department of Integrative Oncology, Tianjin Medical University Cancer Institute and Hospital, Key Laboratory of Cancer Prevention and Therapy of Tianjin City, Tianjin, China
| | - Lei Gong
- Department of Esophageal Cancer, Tianjin Medical University Cancer Institute and Hospital, Key Laboratory of Cancer Prevention and Therapy of Tianjin City, Tianjin, China
| | - Gang Zhao
- Department of Pathology, Tianjin Medical University Cancer Institute and Hospital, Key Laboratory of Cancer Prevention and Therapy of Tianjin City, Tianjin, China
| | - Hongdian Zhan
- Department of Esophageal Cancer, Tianjin Medical University Cancer Institute and Hospital, Key Laboratory of Cancer Prevention and Therapy of Tianjin City, Tianjin, China
| | - Bin Meng
- Department of Pathology, Tianjin Medical University Cancer Institute and Hospital, Key Laboratory of Cancer Prevention and Therapy of Tianjin City, Tianjin, China
| | - Zhentao Yu
- Department of Esophageal Cancer, Tianjin Medical University Cancer Institute and Hospital, Key Laboratory of Cancer Prevention and Therapy of Tianjin City, Tianjin, China
| | - Zhanyu Pan
- Department of Integrative Oncology, Tianjin Medical University Cancer Institute and Hospital, Key Laboratory of Cancer Prevention and Therapy of Tianjin City, Tianjin, China
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11
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Fukuda S, Ito H, Ohba R, Sato Y, Ohyauchi M, Igarashi T, Obana N, Iijima K. A Retrospective Study, an Initial Lesion of Primary Malignant Melanoma of the Esophagus Revealed by Endoscopy. Intern Med 2017; 56:2133-2137. [PMID: 28781311 PMCID: PMC5596272 DOI: 10.2169/internalmedicine.8378-16] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/07/2016] [Accepted: 12/18/2016] [Indexed: 11/06/2022] Open
Abstract
A 66-year-old man presented to his previous physician with epigastric discomfort in 2014. He was then referred to our hospital due to suspected primary malignant melanoma of the esophagus (PMME). A biopsy showed atypical cells containing melanin granules. A diagnosis of PMME was thus made. We investigated the endoscopic findings of the previous physician, which revealed a black point-like pigmentation at the same site since 2009. In 2010, black pigmentation was also observed at the same site. Although esophageal melanosis was suspected, no biopsy was performed. This case demonstrates the process by which esophageal melanomas develop into malignant melanomas.
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Affiliation(s)
- Sho Fukuda
- Department of Gastroenterology, Akita University Graduate School of Medicine, Japan
| | - Hirotaka Ito
- Department of Gastroenterology, Osaki Citizen Hospital, Japan
| | - Reina Ohba
- Department of Gastroenterology, Akita University Graduate School of Medicine, Japan
| | - Yuichirou Sato
- Department of Gastroenterology, Osaki Citizen Hospital, Japan
| | - Motoki Ohyauchi
- Department of Gastroenterology, Osaki Citizen Hospital, Japan
| | | | - Nobuya Obana
- Department of Gastroenterology, Osaki Citizen Hospital, Japan
| | - Katsunori Iijima
- Department of Gastroenterology, Akita University Graduate School of Medicine, Japan
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12
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Primary malignant melanoma of esophagus following chemoradiotherapy for esophageal squamous cell carcinoma: report of a case. Clin J Gastroenterol 2017; 10:336-341. [PMID: 28550655 DOI: 10.1007/s12328-017-0751-2] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/19/2017] [Accepted: 05/12/2017] [Indexed: 10/19/2022]
Abstract
Primary malignant melanoma of the esophagus (PMME) is a rare neoplasm. We observed a case of PMME that had occurred at an irradiated site in the thoracic esophagus. The patient was a 74-year-old man who had received definitive chemoradiotherapy for esophageal squamous cell carcinoma at the age of 68, and had completely recovered. Six years later, via periodic upper gastrointestinal endoscopy, he had been diagnosed with esophageal tumors. After the patient underwent esophagectomy, the removed esophageal tumors were immunohistologically diagnosed as PMME, and most of the tumor was observed to be located within the irradiated area corresponding to the previous radiotherapy. The patient has been free of recurrence and metastasis and alive for 20 months after the second surgery without any additional treatment. The clinical course of this patient suggests that therapeutic radiation may be a factor in the occurrence of PMME.
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14
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Lee J, Kim JY, Bae JY, Kim JY, Choi JE, Jang J, Tae CH, Shim KN. Primary Malignant Melanoma of the Esophagus Treated by Early Diagnosis and Surgical Resection. THE KOREAN JOURNAL OF HELICOBACTER AND UPPER GASTROINTESTINAL RESEARCH 2015. [DOI: 10.7704/kjhugr.2015.15.2.127] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 11/22/2022]
Affiliation(s)
- Jihyun Lee
- Department of Internal Medicine, Ewha Womans University School of Medicine, Seoul, Korea
| | - Ji-Yoon Kim
- Department of Internal Medicine, Ewha Womans University School of Medicine, Seoul, Korea
| | - Ji Yun Bae
- Department of Internal Medicine, Ewha Womans University School of Medicine, Seoul, Korea
| | - Joo Young Kim
- Department of Internal Medicine, Ewha Womans University School of Medicine, Seoul, Korea
| | - Jung Eun Choi
- Department of Internal Medicine, Ewha Womans University School of Medicine, Seoul, Korea
| | - Jiyoung Jang
- Department of Internal Medicine, Ewha Womans University School of Medicine, Seoul, Korea
| | - Chung Hyun Tae
- Department of Internal Medicine, Ewha Womans University School of Medicine, Seoul, Korea
| | - Ki-Nam Shim
- Department of Internal Medicine, Ewha Womans University School of Medicine, Seoul, Korea
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Yang L, Sun X, Meng X. A case of primary malignant melanoma of the esophagus. Dig Dis Sci 2013; 58:3634-6. [PMID: 23861110 DOI: 10.1007/s10620-013-2780-0] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/29/2013] [Accepted: 06/25/2013] [Indexed: 12/09/2022]
Affiliation(s)
- Luoluo Yang
- Department of Gastroenterology, First Hospital of Jilin University, Changchun, 130021, Jilin, China
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16
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Wang S, Tachimori Y, Hokamura N, Igaki H, Kishino T, Kushima R. Diagnosis and surgical outcomes for primary malignant melanoma of the esophagus: a single-center experience. Ann Thorac Surg 2013; 96:1002-6. [PMID: 23810175 DOI: 10.1016/j.athoracsur.2013.04.072] [Citation(s) in RCA: 33] [Impact Index Per Article: 2.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/18/2013] [Revised: 04/25/2013] [Accepted: 04/29/2013] [Indexed: 02/05/2023]
Abstract
BACKGROUND We summarize the experience of diagnosis and surgical therapy for primary malignant melanoma of the esophagus (PMME). METHODS Clinical data of 13 patients diagnosed as having PMME treated by surgery as their primary therapy from 2000 to 2012 were retrospectively analyzed, and survival information was collected through follow-up. RESULTS The average age (±standard deviation) of participants in this study was 66.4±7.6 years, and 84.6% were male. Overall, 61.5% of tumors were located in the lower thoracic esophagus. The accuracies of clinical T stage, N stage, and TNM stage were 53.9%, 46.2%, and 38.5%, respectively, compared with pathological staging (kappa=0.252, p=0.023). Surgical mortality and morbidity were 7.7% and 53.9%, respectively. The incidence of lymph node metastasis for patients with tumor invading within the mucosa was 0, but increased to 42.9% (3 of 7) with tumor invading to the submucosal layer. Primary malignant melanoma of the esophagus in the mid third of the thoracic esophagus had a greater chance to metastasize to perigastric lymph nodes (2 of 5) than to middle mediastinal lymph nodes (1 of 5). For PMME located at the lower third of the thoracic esophagus, upper mediastinal lymph node metastasis was more likely to occur (2 of 4) with tumor invasion penetrating the proper muscle layer. Recurrence occurred within 1 year in all patients with tumor later than Stage Ib. The most common recurrent organ was the liver. The overall 1-year and 5-year postoperative survival rates were 54.0% and 35.9%, respectively, and lymph node metastasis was the independent predictive factor for postoperative survival (p=0.013; odds ratio, 15.05). CONCLUSIONS Despite the similarity in lymph node metastatic patterns to squamous cell carcinoma, PMME is more inclined to distant metastasis. Clinical staging was inconsistent with pathological staging for PMME based on endoscopy and computed tomography. Surgical therapy was the optimal treatment for PMME at an earlier stage. Early diagnosis and aggressive lymph node dissection were beneficial for accurate staging, potentially reducing recurrence and thus improving survival.
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Affiliation(s)
- Shaohua Wang
- Division of Esophageal Surgery, Department of Surgery, National Cancer Center Hospital, Tokyo, Japan
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Ishizaki M, Aibara Y, Furuya K. Primary malignant melanoma of the esophagogastric junction: Report of a case. Int J Surg Case Rep 2013; 4:700-3. [PMID: 23810918 DOI: 10.1016/j.ijscr.2013.04.037] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/25/2013] [Revised: 04/15/2013] [Accepted: 04/17/2013] [Indexed: 11/18/2022] Open
Abstract
INTRODUCTION Primary malignant melanoma of the gastrointestinal tract is very rare, especially in the stomach. We report an extremely rare case of primary malignant melanoma of the esophagogastric junction mainly situated in the stomach. PRESENTATION OF CASE The patient was a 72-year-old woman who complained of shortness of breath due to severe anemia. Upper endoscopy revealed a soft easy-bleeding polypoid tumor just adjacent to the esophagogastric junction in the stomach. Biopsy of the tumor did not indicate a definite result, except malignant tumor. We performed total gastrectomy with splenectomy, and histological and immunohistological examination revealed malignant melanoma of the esophagogastric junction. She had no remote metastasis or lymphnodal metastasis at the point of surgery; however, she died of multiple metastases 11 months after the operation. DISCUSSION A definite preoperative diagnosis of primary malignant melanoma was very difficult to make from the preoperative biopsy specimen. This present case was first misinterpreted as undifferentiated carcinoma, or malignant lymphoma. Following the diagnosis of malignant melanoma, the question arose as to whether this was primary or metastatic (as malignant melanoma from other sites is known to metastasize to the stomach). Finally this tumor was diagnosed as a primary one due to the pathologic characteristics such as the existence of junctional activities. CONCLUSION We report an extremely rare case of primary malignant melanoma of the esophagogastric junction present in the stomach.
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Affiliation(s)
- Masahiro Ishizaki
- Department of Surgery, Okayama Rosai Hospital, 1-10-25 Chikkou-midorimachi, Minami Ward, Okayama 702-8055, Japan.
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18
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Maroy B, Baylac F. Primary malignant esophageal melanoma arising from localized benign melanocytosis. Clin Res Hepatol Gastroenterol 2013; 37:e65-7. [PMID: 22959400 DOI: 10.1016/j.clinre.2012.08.003] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/18/2012] [Revised: 07/26/2012] [Accepted: 08/03/2012] [Indexed: 02/04/2023]
Abstract
A heavy drinker and smoker had a normal UGI-endoscopy in 1996. In 1999, a repeat examination disclosed distal esophageal benign melanocytosis, typical, macro- and microscopically. Endoscopic and microscopic features were stable in 2002 and 2004. In 2007, dysphagia prompted an endoscopy, which disclosed a poorly differentiated epidermoid carcinoma at 19 cm and black nodules of the cardia with a microscopy typical of malignant melanoma, uT3N1 at EUS. Radiochemotherapy led to an 8-month improvement, which did not prevent death one year after diagnosis.
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Affiliation(s)
- Bernard Maroy
- Cabinet d'anatomopathologie, 182, rue de Périgueux, 16000 Angoulême, France.
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Machado J, Ministro P, Araújo R, Cancela E, Castanheira A, Silva A. Primary malignant melanoma of the esophagus: A case report. World J Gastroenterol 2011; 17:4734-8. [PMID: 22180718 PMCID: PMC3233681 DOI: 10.3748/wjg.v17.i42.4734] [Citation(s) in RCA: 12] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/23/2011] [Revised: 04/19/2011] [Accepted: 04/26/2011] [Indexed: 02/06/2023] Open
Abstract
The authors present the clinical case of an 87-year-old Caucasian male admitted to the emergency room with hematemesis. He had a history of intermittent dysphagia during the previous month. Endoscopic evaluation revealed an eccentric, soft esophageal lesion located 25-35 cm from the incisors, which appeared as a protrusion of the esophagus wall, with active bleeding. Biopsies were acquired. Tissue evaluation was compatible with a melanoma. After excluding other sites of primary neoplasm, the definitive diagnosis of Primary Malignant Melanoma of the Esophagus (PMME) was made. The patient developed a hospital-acquired respiratory infection and died before tumor-directed treatment could begin. Primary malignant melanoma represents only 0.1% to 0.2% of all esophageal malignant tumors. Risk factors for PMME are not defined. A higher incidence of PMME has been described in Japan. Dysphagia, predominantly for solids, is the most frequent symptom at presentation. Retrosternal or epigastric discomfort or pain, melena or hematemesis have also been described. The characteristic endoscopic finding of PMME is as a polypoid lesion, with variable size, usually pigmented. The neoplasm occurs in the lower two-thirds of the esophagus in 86% of cases. PMME metastasizes via hematogenic and lymphatic pathways. At diagnosis, 50% of the patients present with distant metastases to the liver, the mediastinum, the lungs and the brain. When possible, surgery (curative or palliative), is the preferential method of treatment. There are some reports in the literature where chemotherapy, chemohormonotherapy, radiotherapy and immunotherapy, with or without surgery, were used with variable efficacy. The prognosis is poor; the mean survival after surgery is less than 15 mo.
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20
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Yu H, Huang XY, Li Y, Xie X, Zhou JL, Zhang LJ, Fu JH, Wang X. Primary malignant melanoma of the esophagus: a study of clinical features, pathology, management and prognosis. Dis Esophagus 2011; 24:109-13. [PMID: 21040150 DOI: 10.1111/j.1442-2050.2010.01111.x] [Citation(s) in RCA: 23] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/11/2022]
Abstract
Primary malignant melanoma of the esophagus (PMME) is a rare disease that is characterized by aggressive invasion, early metastasis, and poor prognosis. Treatment protocols are not well-established. To understand this condition more precisely, we performed a retrospective review of eight cases of PMME diagnosed at the Thoracic Department of the Cancer Center at Sun Yat-Sen University between 1985 and 2009. Eight PMME patients (five men and three women) with a mean age of 58 years (range: 48 to 72 years) were included. Dysphagia was the most common presenting symptom. All patients underwent an Ivor-Lewis esophagogastrectomy and lymph node dissection with postoperative adjuvant chemotherapy. One patient with stage III/pT4N0M0 underwent postoperative chemotherapy plus radiotherapy. Four patients died of distant metastases. The median survival time was 28 months (range: 11 months to 6 years). Our data confirm that PMME is a highly aggressive disease with a poor prognosis. If the diagnosis is suspected or confirmed as PMME, and the patients have no distal metastases or extensive lymph node enlargement, we suggest that surgery should be the first choice of treatment. With regard to adjuvant therapy, we recommend the addition of chemotherapy. The role of radiotherapy remains questionable and requires further investigation.
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Affiliation(s)
- H Yu
- State Key Laboratory of Oncology in South China, Department of Thoracic Surgery, Cancer Center, Sun Yat-Sen University, Guangzhou, China
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Mamori S, Kahara F, Ohnishi K, Takeda A, Higashida A, Ashida C, Yamada H. Survivin expression in primary malignant melanoma of the esophagus. Scand J Gastroenterol 2010; 44:1497-8. [PMID: 19883277 DOI: 10.3109/00365520903330310] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/04/2023]
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Abstract
A 55-year-old man had dysphagia of 4-month duration. Endoscopy revealed a large polypoidal pedunculated blackish mass located in the lower thoracic esophagus. A barium esophagogram revealed a lower esophageal mass. Computed tomography revealed a large polypoidal esophageal mass without any evidence of local invasion or distant disease. Endoscopic biopsy established the diagnosis of melanoma. A radical resection of the esophagus with three-field lymph nodal dissection was undertaken. The patient made an uneventful recovery. Histopathology confirmed the diagnosis of melanoma and positive lymph nodes. Adjuvant chemotherapy was given. He succumbed to systemic recurrence after 69 months.
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Nevus-like appearance of primary malignant melanoma of the esophagus. Gastroenterol Res Pract 2009; 2009:285753. [PMID: 19644559 PMCID: PMC2716486 DOI: 10.1155/2009/285753] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/24/2009] [Accepted: 06/23/2009] [Indexed: 11/18/2022] Open
Abstract
The primary malignant melanoma of the esophagus (PMME) is a rare
malignant disease, accounting for only 0.1–0.2% of all
esophageal neoplasms, and the majority of the patients are
diagnosed at advanced stages with poor prognosis. We present here
a case of 56-year-old woman with epigastric pain and her
endoscopic finding revealed several flat and black pigmented
mucosal lesions within the distal portion of the esophagus which
looked like flat nevus. The histopathology and immunohistochemical
profile of the tissue specimens were diagnostic of malignant
melanoma.
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Tabatabaei SA, Moghadam NA, Ahmadinejad M, Mirmohammadsadeghi A, Masoudpour H, Adibi P. Giant esophageal squamous papilloma: a case report. J Dig Dis 2009; 10:228-30. [PMID: 19659792 DOI: 10.1111/j.1751-2980.2009.00390.x] [Citation(s) in RCA: 12] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/11/2022]
Abstract
Esophageal papilloma is a rare disorder that may cause hypopharyngeal symptoms. This patient was a 56-year-old man who presented with cough and choking symptoms. After the initial negative laryngoscopy, a fiberoptic endoscopy revealed a mass originating from the hypopharyngeal area, which was resected surgically and found to be non-malignant.
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Affiliation(s)
- Sayed Abbas Tabatabaei
- Department of Thoracic Surgery, Al-Zahra Hospital, Isfahan University of Medical Sciences, Isfahan, Iran
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Fredricks JRC, Bejarano PA. Primary malignant melanoma of the esophagus with separate foci of melanoma in situ and atypical melanocytic hyperplasia in a patient positive for human immunodeficiency virus: a case report and review of the literature. Arch Pathol Lab Med 2008; 132:1675-8. [PMID: 18834229 DOI: 10.5858/2008-132-1675-pmmote] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 02/28/2008] [Indexed: 11/06/2022]
Abstract
Primary malignant melanoma of the esophagus (PMME) is a rare neoplasm. The primary nature of esophageal melanoma has been questioned in the past because most reported cases have not been able to demonstrate local tumorigenesis or evolution of the malignancy from a preceding benign lesion or cell. In addition, the occurrence of metastatic melanoma to the esophagus is much more common than PMME. We report a case of PMME with separate foci of melanoma in situ and atypical melanocytic hyperplasia in a patient with human immunodeficiency virus infection. To our knowledge, this is the first reported case of PMME in a patient with a previous diagnosis of human immunodeficiency virus infection. In addition to adding data to the limited literature on this subject, we also provide histologic evidence that demonstrates the primary nature of some esophageal melanomas. This case also documents the occurrence of PMME in a patient positive for human immunodeficiency virus. We also provide a review of the relevant literature.
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