Primary esophageal malignant melanoma successfully treated with anti-PD-1 antibody for retroperitoneal recurrence after esophagectomy: A case report

Endo, Fumitaka; Akiyama, Yuji; Onishi, Masazumi; Fujisawa, Ryosuke; Sasaki, Noriyuki; Nikai, Haruka; Baba, Shigeaki; Sugimoto, Ryo; Kimura, Toshimoto; Takahara, Taro; Iwaya, Takeshi; Otsuka, Koki; Nitta, Hiroyuki; Koeda, Keisuke; Sugai, Tamotsu; Sasaki, Akira

doi:10.1016/j.ijscr.2020.09.034

Cited by 6 publications

(9 citation statements)

References 19 publications

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“…Notably, one patient with distant LN and adrenal gland metastases received 24 months of Nivolumab as first-line therapy and succeeded in long-term survival of 34 months (Table 3 ). As other recent case studies consistently report the effectiveness of immunotherapy for metastatic PMME [ 9 , 11 ], further large scale studies are warranted to confirm the validity of immunotherapy for PMME. To date, it is unclear whether PD-L1 expression can be a predictive marker for immunotherapy response for mucosal melanoma [ 23 , 24 ].…”

Section: Discussionmentioning

confidence: 83%

“…In CheckMate 067 study including 79 advanced mucosal melanoma patients treated with immune checkpoint inhibitor, the five-year overall survival rate was 36% and the median overall survival was 22.7 months in the combination immunotherapy group (Nivolumab plus Ipilimumab), which was better than those of either monotherapy group [ 8 ]. For PMME, there have been only a few small-sized studies on the outcomes of immunotherapy [ 9 – 11 ]. In the largest study by Wang et al [ 11 ] (n = 12) patients who received programmed death (PD)-1 inhibitors for PMME showed mean progression-free survival of 15.6 months.…”

Section: Introductionmentioning

confidence: 99%

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Clinical characteristics and treatment outcomes of primary malignant melanoma of esophagus: a single center experience

et al. 2022

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Background Primary malignant melanoma of esophagus (PMME) is an extremely rare disease with poor prognosis. We aimed to determine the clinical characteristics and treatment outcomes of patients with PMME. Methods We retrospectively reviewed 17 patients diagnosed with PMME in Samsung Medical Center between 2000 and 2020 with median 34 months of follow-up. Survival outcomes were analyzed with Kaplan–Meier method. Results 15 patients (88.2%) were male and the most common presenting symptom was dysphagia (9/17, 52.9%). On endoscopy, tumors were mass-forming in 15 patients (88.2%) and diffusely infiltrative in two patients (11.8%). Lesions were melanotic in 13 patients (76.5%) and amelanotic in four patients (23.5%). The most common tumor location was lower esophagus (11/17, 64.7%). The disease was metastatic at the time of diagnosis in four patients (23.5%). As for treatment, 10 patients (58.8%) underwent surgery. In all 17 patients, the median overall survival was 10 months. In surgically treated patients, all patients experienced recurrence and the median disease-free survival was 4 months. There was no statistical difference in overall survival between patients with or without surgery. Patients with diffusely infiltrative tumor morphology had better overall survival compared to those with mass-forming tumor morphology (P = 0.048). Two patients who received immunotherapy as the first-line treatment without surgery showed overall survival of 34 and 18 months, respectively. Conclusions As radical resection for patients with PMME does not guarantee favorable treatment outcomes, novel treatment strategy is required. Further large-scale studies are warranted to determine the efficacy of immunotherapy for patients with PMME.

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Section: Discussionmentioning

confidence: 83%

Section: Introductionmentioning

confidence: 99%

Clinical characteristics and treatment outcomes of primary malignant melanoma of esophagus: a single center experience

et al. 2022

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“…Notably, one patient with distant LN and adrenal gland metastases received 24 months of Nivolumab as rst-line therapy and succeeded in long-term survival of 34 months (Table 3). As other recent case studies consistently report the effectiveness of immunotherapy for metastatic PMME [8,10], further large scale studies are warranted to con rm the validity of immunotherapy for PMME.…”

Section: Discussionmentioning

confidence: 88%

“…However, due to the extreme rarity of the disease, its clinical features are sparsely reported and treatment strategies are not standardized. Recently, treatment with immune-checkpoint inhibitor such as Pembrolizumab or Nivolumab for metastatic PMME have shown promising results [8][9][10]. However, as the number of cases were limited, more studies with consistent results are required to validate the e cacy of such treatments.In the present study, we reviewed the clinical and endoscopic features of 17 patients diagnosed with PMME in our institution and investigated their surgical and non-surgical outcomes.…”

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confidence: 99%

Clinical Characteristics and Treatment Outcomes of Primary Malignant Melanoma of Esophagus: A Single Center Experience Running head: Outcomes of primary esophageal melanoma

Kim

Min

et al. 2021

Preprint

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BackgroundPrimary malignant melanoma of esophagus (PMME) is an extremely rare disease with poor prognosis. The aim of this study was to determine the clinical characteristics and treatment outcomes of patients with PMME. MethodsWe retrospectively reviewed 17 patients diagnosed with PMME in Samsung Medical Center between 2000 and 2020. Clinical characteristics and survival outcomes were analyzed. Results15 patients (88.2%) were male and the most common presenting symptom was dysphagia (9/17, 52.9%). On endoscopy, tumors were mass-forming in 15 patients (88.2%) and diffusely infiltrative in two patients (11.8%). Lesions were melanotic in 13 patients (76.5%) and amelanotic in four patients (23.5%). The most common anatomic location of tumor was lower esophagus (11/17, 64.7%). The disease was metastatic at the time of diagnosis in four patients (23.5%). As for treatment, 10 patients (58.8%) underwent surgery. In all 17 patients, the median overall survival was 10 months. In surgically treated patients, all patients experienced recurrence and the median disease-free survival was 4 months. There was no statistical difference in overall survival between patients with or without surgery. Patients with diffusely infiltrative tumor morphology had better overall survival compared to those with mass-forming tumor morphology (P = 0.048). Two patients who received immunotherapy as the first-line treatment without surgery showed overall survival of 34 and 18 months, respectively. ConclusionsAs radical resection for patients with PMME does not guarantee favorable treatment outcomes, novel treatment strategy is required. Further large-scale studies are warranted to determine the efficacy of immunotherapy for patients with PMME.

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“…Therefore, these authors renewed the need to employ techniques such as massive gene sequencing to be able to search for biomarkers capable of predicting the response of PMME to anti-PD1 immunocheckpoint inhibitors. After another 38 cases of PMME [ 274 , 277 , 278 , 279 , 280 , 281 , 282 , 283 , 284 , 285 , 286 , 287 , 288 , 289 ], Endo F. et al [ 290 ] reported a case of a 70-year-old man who complained of dysphagia due to the presence of a polypoid lesion in the lower thoracic esophagus. Although the histopathological examination had provided the diagnosis of esophageal squamous carcinoma at first, with subsequent pT3N1M0 staging and clinical stage III, a histological re-evaluation after radical esophagectomy revealed the presence of atypical melanocyte cells positive for S-100 protein, Melan-A and HMB-45, and, therefore, PMME was diagnosed.…”

Section: Discussionmentioning

confidence: 99%

The Thousand Faces of Malignant Melanoma: A Systematic Review of the Primary Malignant Melanoma of the Esophagus

Cazzato

Cascardi

Colagrande

et al. 2022

Cancers

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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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Primary esophageal malignant melanoma successfully treated with anti-PD-1 antibody for retroperitoneal recurrence after esophagectomy: A case report

Cited by 6 publications

References 19 publications

Clinical characteristics and treatment outcomes of primary malignant melanoma of esophagus: a single center experience

Clinical characteristics and treatment outcomes of primary malignant melanoma of esophagus: a single center experience

Clinical Characteristics and Treatment Outcomes of Primary Malignant Melanoma of Esophagus: A Single Center Experience Running head: Outcomes of primary esophageal melanoma

The Thousand Faces of Malignant Melanoma: A Systematic Review of the Primary Malignant Melanoma of the Esophagus

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