Pembrolizumab for primary malignant melanoma of the central nervous system

Habnouni, Chakib El; Bléchet, Claire; Bens, Guido

doi:10.1007/s11060-018-2848-y

Cited by 5 publications

(5 citation statements)

References 4 publications

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“…In the case of primary CNS melanoma, the use of immunotherapy has been limited to a few case reports, and responses have thus far been mixed. El Habnouni et al [32] treated a 69-year-old woman for 16 weeks with pembrolizumab, but the patient had continued tumor progression. In contrast, Krpan et al [33] report the case of a 72-year-old patient who experienced 2 years of stable disease following treatment with pembrolizumab.…”

Section: Discussionmentioning

confidence: 99%

Primary Diffuse Leptomeningeal Melanomatosis in a Child with Extracranial Metastasis: Case Report

Shahab,

Patil,

Fangusaro

et al. 2024

Current Oncology

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Primary meningeal melanomatosis is an extremely rare tumor with very few documented responses to treatment. A 3-year-old male with a complex past medical history, including prematurity and shunted hydrocephalus, was diagnosed with primary meningeal melanomatosis with peritoneal implants. Molecular testing revealed an NRAS Q61R mutation. The patient received proton craniospinal radiation followed by immunotherapy with nivolumab (1 mg/kg) and ipilimumab (3 mg/kg) IV every 3 weeks and, upon progression, he was switched to a higher dose of nivolumab (3 mg/kg IV every 2 weeks) and binimetinib (24 mg/m2/dose, twice a day). The patient had significant improvement of CNS disease with radiation therapy and initial immunotherapy but progression of extracranial metastatic peritoneal and abdominal disease. Radiation was not administered to the whole abdomen. After two cycles of nivolumab and treatment with the MEK inhibitor binimetinib, he had radiographic and clinical improvement in abdominal metastasis and ascitis. He ultimately died from RSV infection, Klebsiella sepsis, and subdural hemorrhage without evidence of tumor progression. This is the first report of a child with primary meningeal melanomatosis with extracranial metastatic disease with response to a combination of radiation, immunotherapy and MEK inhibitor therapy.

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Section: Discussionmentioning

confidence: 99%

Primary Diffuse Leptomeningeal Melanomatosis in a Child with Extracranial Metastasis: Case Report

Shahab,

Patil,

Fangusaro

et al. 2024

Current Oncology

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“…Of the 92 primary CNS melanoma cases in our compilation, 13 patients reported their genetic status (Burgos et al., 2021; de la Fouchardière et al., 2015; El Habnouni et al., 2018; Fortin Ensign et al., 2020; Goldman‐Lévy et al., 2016; Hanft et al., 2022; Kinsler et al., 2017; Schäfer et al., 2013) (Table 1). This included three cases with GNAQ mutations, five cases with NRAS mutations, one case with BAP1 deletion, one case with INPP4B Q204R mutation, and one case with BRAF V600E mutation.…”

Section: Molecular Pathologymentioning

confidence: 99%

“…To date, there have been no published reports of large‐sample studies on the tumor immune microenvironment (TIME) in primary CNS melanoma. Few relevant small‐sample studies and case reports exist, with only two patients having undergone PD‐L1 testing (El Habnouni et al., 2018; Fortin Ensign et al., 2020). However, findings from MBM may aid our understanding of the TIME in primary CNS melanoma.…”

Section: Immune Microenvironmentmentioning

confidence: 99%

“…However, only a few cases of primary CNS melanoma have been treated with immunotherapy. To address this gap, we collected data from Chakib, Remberto, Ashley, Heidi, and Kentaro on five cases of primary CNS melanoma treated with immunotherapy (Aaroe et al., 2021b; Burgos et al., 2021; El Habnouni et al., 2018; Fujimori et al., 2018; Küsters‐Vandevelde et al., 2017) (Table 3). These patients achieved a median OS of 58 months (5–63 months).…”

Section: Treatmentmentioning

confidence: 99%

“…The incidence of primary central nervous system (CNS) melanoma is approximately 0.01/100,000 per year, accounting for 1% of all melanomas (Balakrishnan et al., 2015; El Habnouni et al., 2018; Hsieh et al., 2015b; Rosenthal et al., 2003c). Diagnosis relies on histologic or cytopathologic biopsies due to the non‐characteristic clinical symptoms and imaging manifestations.…”

Section: Introductionmentioning

confidence: 99%

See 2 more Smart Citations

Clinicopathological features, current status, and progress of primary central nervous system melanoma diagnosis and treatment

Guo,

Wei,

Guo

et al. 2023

Pigment Cell Melanoma Res

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Primary central nervous system (CNS) melanoma is an extremely rare condition, with an incidence rate of 0.01 per 100,000 individuals per year. Despite its rarity, the etiology and pathogenesis of this disease are not yet fully understood. Primary CNS melanoma exhibits highly aggressive biological behavior and presents clinically in a distinct manner from other types of melanomas. It can develop at any age, predominantly affecting the meninges as the primary site, with clinical symptoms varying depending on the neoplasm’s location. Due to the lack of specificity in its presentation and the challenging nature of imaging diagnosis, distinguishing primary CNS melanoma from other CNS diseases. The combination of challenges in early detection, heightened tumor aggressiveness, and the obscured location of its origin contribute to an unfavorable prognostic outcome. Furthermore, there has been currently no consensus on a standardized treatment approach for primary CNS melanoma. Despite recent advancements in targeted therapy and immunotherapy for CNS melanoma, patients with primary CNS melanoma have limited treatment options due to their inadequate response to these therapies. Here, we provided a comprehensive summary of the epidemiology, clinical features, molecular pathological manifestations, and available diagnostic and therapeutic approaches of primary CNS melanoma. Additionally, we proposed potential therapeutic strategies for it.

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Primary leptomeningeal melanomatosis successfully treated with PD-1 inhibitor pembrolizumab

2020

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Rationale: Primary leptomeningeal melanoma is an extremely rare disease of the central nervous system. There are no standard treatment protocols with a poor prognosis in very few reported cases. Immunotherapy in primary brain melanoma has not been successfully applied so far. Patient concerns: We describe a female patient 72-year-old diagnosed in the Neurosurgery Department which presented with generalized seizures. Diagnoses: Histological examination confirmed atypical melanocytes immunohistochemically positive for melan A, HMB45 and S-100 protein in the meninges, BRAF V600E negative. Dermatological, ophthalmological examinations, and 18-FDG PET/CT were negative. Interventions: The patient was successfully treated with pembrolizumab 2 mg/kg every 3 weeks for 2 years. Outcomes: The disease was stable for 2 years and the patient had no significant toxicity. Lessons: Our report describes durable intracranial tumor response suggesting the efficacy of PD-1 inhibitor pembrolizumab for central nervous system primary leptomeningeal melanoma.

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Pembrolizumab for primary malignant melanoma of the central nervous system

Cited by 5 publications

References 4 publications

Primary Diffuse Leptomeningeal Melanomatosis in a Child with Extracranial Metastasis: Case Report

Primary Diffuse Leptomeningeal Melanomatosis in a Child with Extracranial Metastasis: Case Report

Clinicopathological features, current status, and progress of primary central nervous system melanoma diagnosis and treatment

Primary leptomeningeal melanomatosis successfully treated with PD-1 inhibitor pembrolizumab

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