An Extensive-stage Small-cell Lung Cancer Case With Preexisting Lambert-Eaton Myasthenic Syndrome Successfully Treated With an Immune Checkpoint Inhibitor

Sakaguchi, Tadashi; Kokubo, Yasumasa; Furuhashi, Kazuki; Nakamura, Yuki; Suzuki, Yuta; Ito, Kentaro; Fujiwara, Kentaro; Nishii, Yoichi; Taguchi, Osamu; Hataji, Osamu

doi:10.1016/j.cllc.2021.09.001

Cited by 9 publications

(9 citation statements)

References 8 publications

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“…Among these three cases, LEMS improved by combination immunotherapy in two Japanese cases, but deteriorated by ICI monotherapy in the other German case. Our case was very similar in clinical course, treatment regimen and outcomes to the other Japanese case by Sakaguchi et al [ 8 ]. The difference between these two Japanese cases was ICI drug, i.e., durvalumab vs. atezolizumab.…”

Section: Discussionsupporting

confidence: 84%

“…Then, she could continue chemotherapy without pembrolizumab. On the other hand, ours and the other two cases introduced ICIs soon after the confirmed diagnosis of malignancy and worsening LEMS [ 8 , 10 ]. Among these three cases, LEMS improved by combination immunotherapy in two Japanese cases, but deteriorated by ICI monotherapy in the other German case.…”

Section: Discussionmentioning

confidence: 98%

“…To our knowledge, there were only four malignant cases who had preexisting LEMS but had Durvalumab for Lambert-Eaton Syndrome J Med Cases. 2023;14(2):71-75 been treated by ICIs [7][8][9][10]. These rare cases suggest that ICI is a possible treatment option for cancer patients with LEMS.…”

Section: Discussionmentioning

confidence: 99%

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Durvalumab for Extensive-Stage of Small-Cell Lung Cancer With Lambert-Eaton Myasthenic Syndrome

Machiyama

Minami

2023

J Med Cases

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Durvalumab is an immune checkpoint inhibitor (ICI) of anti-programmed cell death protein 1 ligand antibody. ICI-combined chemotherapy has recently become a standard regimen for extensive-stage of small-cell lung cancer (ES-SCLC). SCLC is well known to be the most likely tumor associated with Lambert-Eaton myasthenic syndrome (LEMS), a rare autoimmune disease of a neuromuscular junction disorder. Although LEMS has been reported to be induced by ICI as immune-mediated adverse events, it remains unknown whether ICI can deteriorate preexisting paraneoplastic syndrome (PNS) of LEMS. Our rare case was successfully treated by durvalumab plus chemotherapy without exacerbation of preexisting PNS of LEMS. We report a 62-yearold female with ES-SCLC and preexisting PNS of LEMS. She started carboplatin-etoposide in combination with durvalumab. This immunotherapy achieved nearly complete response. However, multiple brain metastases were found after two courses of maintenance durvalumab. Her symptoms and physical examinations of LEMS improved despite of no significant change in compound muscle action potential amplitude in the nerve conduction study. The titer of anti-P/Q-type voltage-gated calcium channel (VGCC) antibody decreased from 1,419.2 to 263.5 pmol/L during the immunotherapy. In conclusion, ICI in combination with platinum doublet chemotherapy is still challenging but may be a treatment option for ES-SCLC patients complicated with PNS of LEMS.

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Section: Discussionsupporting

confidence: 84%

Section: Discussionmentioning

confidence: 98%

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Durvalumab for Extensive-Stage of Small-Cell Lung Cancer With Lambert-Eaton Myasthenic Syndrome

Machiyama

Minami

2023

J Med Cases

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“…Our institution experience was that IVIg was applied in acute phase to neutralize antibody for 5–7 days. Sakaguchi et al reported an extensive stage SCLC patient complicated with LEMS successfully treated by immune checkpoint inhibitor plus chemotherapy without a flare-up of LEMS [ 21 ].…”

Section: Discussionmentioning

confidence: 99%

Lambert‐Eaton Myasthenic Syndrome in Lung Cancer

Wang

et al. 2022

Contrast Media & Molecular Imaging

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The aim is to study lung cancer with Lambert-Eaton myasthenic syndrome (LEMS) with clinical and electrical characteristics of physiology and prognosis. Fourteen LEMS patients with lung cancer were studied retrospectively. The data including demographics, clinical presentation, treatments, and prognosis from the medical records were analyzed. Lung cancer coexisting with LEMS is more common in men (10/14). The median age was 67.1 years. Eleven (78.6%) patients experienced gradual onset of disease. Most patients presented nervous system lesions prior to occult tumors. The most common symptoms reported were proximal muscle weakness (92.9%), decreased or absent tendon reflexes (50%), and autonomic dysfunction (71.4%). All the patients showed reduction in action potential amplitude after repetitive peripheral never stimulation at low frequency and increased amplitude at high frequency. LEMS usually occurs prior to lung cancer with complicated and various clinical manifestations in our centers. We should improve awareness and knowledge of such disease to shorten the diagnostic delay and lead to few misdiagnoses.

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“…To summarize, in most cases treatment of the tumor reduced the LEMS-associated symptoms but none of these cases received treatment with avelumab. Some other case reports raises the concern regarding whether immunotherapy may cause LEMS (28)(29)(30)(31)(32), none of these are reported having MCC. The majority of the cases described had lung cancer and out of the case reports one cannot say whether LEMS was caused by the immunotherapy, only that it presented after initiation of treatment.…”

Section: Discussionmentioning

confidence: 99%

Combined treatment with radiotherapy, chemotherapy and avelumab results in regression of metastatic Merkel cell carcinoma and improvement of associated Lambert‑Eaton myasthenic syndrome: A case report

et al. 2022

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Merkel cell carcinoma (MCC) is a rare and highly aggressive neuroendocrine malignancy arising from mechanoreceptors in the basal epidermis. Due to a pronounced risk of spread and a high propensity for recurrence after treatment, immediate treatment is of utmost importance. Lambert-Eaton myasthenic syndrome (LEMS) is a paraneoplastic phenomenon affecting the muscles with autoimmune pathophysiology, and >50% of known cases are associated with an underlying malignancy. In the present report, the case of a 67-year-old man presenting with progressive proximal muscle weakness, autonomic dysfunction and involuntary weight loss is described. Symptoms and detection of voltage-gated calcium channel antibodies were consistent with LEMS. Distant metastases were found in the inguinal and iliac lymph nodes, and these were immunohistochemically confirmed to be of epithelial and neuroendocrine origin, consistent with MCC. Local radiotherapy and chemotherapy improved the symptoms; however, a change of treatment was required due to the side effects of the chemotherapy. Avelumab, an immune checkpoint inhibitor, was therefore introduced, and within a year the patient did not only experience tumor remission but also exhibited marked improvements in muscle strength and mobility. At present, 2 years later, the MCC is still in remission. To the best of our knowledge, the present report is the first to describe MCC with associated LEMS, which was successfully treated with avelumab after previous radiotherapy and chemotherapy, with both improved functional motor recovery and tumor reduction. In conclusion, the present case report demonstrated that the present treatment strategy is a potential treatment option and could thus be considered in similar cases.

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An Extensive-stage Small-cell Lung Cancer Case With Preexisting Lambert-Eaton Myasthenic Syndrome Successfully Treated With an Immune Checkpoint Inhibitor

Cited by 9 publications

References 8 publications

Durvalumab for Extensive-Stage of Small-Cell Lung Cancer With Lambert-Eaton Myasthenic Syndrome

Durvalumab for Extensive-Stage of Small-Cell Lung Cancer With Lambert-Eaton Myasthenic Syndrome

Lambert‐Eaton Myasthenic Syndrome in Lung Cancer

Combined treatment with radiotherapy, chemotherapy and avelumab results in regression of metastatic Merkel cell carcinoma and improvement of associated Lambert‑Eaton myasthenic syndrome: A case report

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