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Late-stage esophageal neuroendocrine carcinoma in a patient treated with tislelizumab combined with anlotinib: a case report.

ABSTRACT: Esophageal neuroendocrine carcinoma (ENEC) is an extremely rare tumor with highly malignant potential, rapid growth, and a poor prognosis. Advanced extrapulmonary neuroendocrine carcinoma should be treated with chemotherapeutic regimens suitable for small cell lung cancer. However, ENEC has no clear second-line treatment options. The clinical application of immunotherapy and targeted therapy in small cell lung cancer has produced good therapeutic effects. We describe the case of an elderly woman with multiple metastatic advanced ENEC treated with tislelizumab combined with anlotinib as second-line therapy, achieving complete remission in a short period and long-term survival. In total, 21 cycles of tislelizumab combined with anlotinib were given to this patient. After two cycles, the patient's neuron-specific enolase level decreased from 181.8 to 22.9 µg/L and remained at normal levels throughout treatment. Progression-free survival and overall survival were 16 and 21 months, respectively, in this patient. No obvious side effects were observed. Thus, tislelizumab and anlotinib could represent a novel therapeutic option for advanced ENEC.

SUBMITTER: Zhang Y

PROVIDER: S-EPMC10387792 | biostudies-literature | 2023 Jul

REPOSITORIES: biostudies-literature

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Late-stage esophageal neuroendocrine carcinoma in a patient treated with tislelizumab combined with anlotinib: a case report.

Zhang Yanqi Y Liu Xiaoyu X Liang Honglu H Liu Weihua W Wang Haiyan H Li Tao T

The Journal of international medical research 20230701 7

Esophageal neuroendocrine carcinoma (ENEC) is an extremely rare tumor with highly malignant potential, rapid growth, and a poor prognosis. Advanced extrapulmonary neuroendocrine carcinoma should be treated with chemotherapeutic regimens suitable for small cell lung cancer. However, ENEC has no clear second-line treatment options. The clinical application of immunotherapy and targeted therapy in small cell lung cancer has produced good therapeutic effects. We describe the case of an elderly woman ...[more]

PMID: 37498227

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Project description:Pulmonary sarcomatoid carcinoma (PSC) is a rare subtype of lung malignant tumor. Conventional chemotherapy has a suboptimal effectiveness. PSC has the characteristics of rapid disease progression and poor prognosis. We herein report a 56-year-old male patient with substantial smoking history was pathologically diagnosed as PSC, cT4N0M0 IIIA stage. Peripheral blood NGS showed TP53 mutation. The patient had poor tolerance to the first-line chemotherapy regimen "albumin paclitaxel + cisplatin," but the severe anemia was significantly improved after 5 days of anti-angiogenic therapy with Anlotinib. At this time, the patient received anti-PD-1 immunotherapy with Tislelizumab. Half a month later, degree III liver injury occurred repeatedly. After excluding drug-induced liver injury, we found that HCV-RNA 3.10 × 105 IU/ml and suspended all anti-tumor therapy. After the start of anti-HCV treatment with Epclusa, the treatment of Tislelizumab combined with Anlotinib was restarted, and there was no liver injury after that. The patient received monthly maintenance therapy with Tislelizumab combined with Anlotinib to the present. The pulmonary lesions continued to decrease, and only one lung cavity is left. The patient has achieved clinical complete remission (CCR) with PSF over 20 months. Our findings suggest that Tislelizumab combined with Anlotinib may be a preferred strategy in PSC complicating TP53 mutation. Core tip: Immune-check point inhibitors (ICIs) have been reported for the treatment of PSC in a small number of case reports and retrospective analysis, but there are few reports of ICIs combined with anti-angiogenic drugs. This patient was diagnosed as locally advanced PSC complicated with TP53 mutation and hepatitis C. After 14 cycles of Tislelizumab combined with Anlotinib treatment (during the course of treatment, several courses were not treated on time for economic reasons, rather than adverse reactions), the patient has achieved CCR. III degree liver injury occurred during the treatment, and the liver function returned to normal range after anti-hepatitis C treatment, which did not affect the continued treatment of this regimen.

Project description:The utilization of immune checkpoint inhibitors in oncological treatment has increased in recent years. The therapeutic strategy of targeting the programmed death-1 (PD-1)/programmed death-ligand 1 (PD-L1) pathway has altered the management of advanced non-small cell lung carcinoma (NSCLC). Tislelizumab, a novel anti-PD-1 monoclonal antibody developed in China, has demonstrated efficacy in treating advanced NSCLC. However, its potential role as a neoadjuvant therapy for locally advanced NSCLC has not been definitively established. Current guidelines do not specify which patient populations may gain the most benefit from neoadjuvant immunotherapy coupled with chemotherapy, nor do they indicate the optimal timing, dose or duration of adjuvant maintenance therapy post-NSCLC surgery. Similarly, data concerning the safety and practicability of surgical resection following neoadjuvant tislelizumab treatment for NSCLC remain limited. The present study describes the case of a patient diagnosed with stage IIIB NSCLC, which was initially deemed unresectable. A preoperative biopsy of the tumor mass revealed squamous cell carcinoma and a negative PD-L1 gene test. Notably, after two cycles of neoadjuvant tislelizumab treatment coupled with chemotherapy, the tumor exhibited marked shrinkage. This permitted the patient to undergo thoracoscopic radical lung cancer resection, which resulted in a pathological complete response. Postoperative pathology identified a large infiltration of lymphoplasmacytic cells and foamy histiocytes. The patient experienced grade 2 myelosuppression, a condition that was successfully addressed with the administration of recombinant human granulocyte colony-stimulating factor. The present case indicates the safety and feasibility of neoadjuvant immunotherapy integrated with chemotherapy for patients with locally advanced, PD-L1-negative NSCLC prior to surgical intervention. Moreover, the case suggests the potential of this therapeutic combination to alter the tumor microenvironment. However, the generalization of these findings necessitates further validation through randomized multicenter trials.

Project description:IntroductionNeuroendocrine neoplasms encompass well-differentiated tumors (NETs) and poorly differentiated carcinomas (neuroendocrine carcinomas [NECs]), which are distinguished by their clinical behavior and molecular characteristics. They can cause paraneoplastic syndromes, such as ectopic adrenocorticotropic hormone (ACTH)-dependent Cushing's syndrome (CS), necessitating prompt recognition and management due to severe hypercortisolism.Case presentationA 66-year-old patient with a 3-year history of metastatic mixed neuroendocrine-non-neuroendocrine neoplasm with a NEC and adenocarcinoma component originating from the vulva presented to the emergency department with dyspnea and fatigue. Upon clinical examination, we found widespread hyperpigmentation, a moon-face appearance, hirsutism, buffalo hump, and muscle atrophy. Laboratory investigations revealed severe hypokalemia (2.3 mmol/L), elevated serum cortisol (1,726 nmol/L) and ACTH (194 ng/L) levels. Urinary free cortisol measurement was 21-fold the upper limit of the reference range (3,614.0 nmol/24 h), and cortisol concentration did not decrease after 1mg-dexamethasone suppression test (1,812 nmol/L for an expected value <50 nmol/L), confirming the ACTH-dependent CS. Thoracoabdominal computed tomography (CT) scan demonstrated progressive neoplastic disease in the liver, kidney, lymph nodes, peritoneum, and lungs. Brain magnetic resonance imaging indicated multifocal metastatic infiltration but no evidence of pituitary adenoma. Interestingly, despite a previously negative 68Ga-DOTATATE positron emission tomography (PET)/CT performed 1 year prior, there was moderate somatostatin receptor (SSTR) expression in lymphatic, pulmonary, peritoneal, and bone tissues, suggesting the presence of a component with redifferentiation and re-expression of the SSTR. After the workup, the patient was admitted to a supportive care facility. Hypercortisolism symptoms were effectively managed with an adrenal enzyme inhibitor (ketoconazole) in combination with somatostatin analogs. Unfortunately, the patient was too frail to benefit from peptide receptor radionuclide therapy (PRRT).ConclusionThis redifferentiation phenomenon in neuroendocrine tumors should be further investigated as patients might be, under certain conditions, eligible for PRRT. Therefore, we suggest that newly occurring paraneoplastic syndromes in patients with NEC should always be evaluated using 68Ga-DOTATATE PET/CT.

Dataset Information

Late-stage esophageal neuroendocrine carcinoma in a patient treated with tislelizumab combined with anlotinib: a case report.

Publications

Late-stage esophageal neuroendocrine carcinoma in a patient treated with tislelizumab combined with anlotinib: a case report.

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