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1.
Dermatomyositis in a patient undergoing nivolumab therapy for metastatic melanoma: a case report and review of the literature.
Kosche, C, Stout, M, Sosman, J, Lukas, RV, Choi, JN
Melanoma research. 2020;(3):313-316
Abstract
Checkpoint inhibitor immunotherapy is a transformative treatment for advanced malignancies, but can be associated with numerous immune-related adverse events (irAEs). The majority of irAEs include those that closely resemble known cutaneous and neurocutaneous autoimmune or autoinflammatory diseases, such as scleroderma, psoriasis, and dermatomyositis. We present the case of a 63-year-old man with metastatic melanoma undergoing treatment with nivolumab who developed significant motor weakness, paresthesias of both hands, swollen fingers, and a pruritic rash over the face, chest, and upper back after two cycles. Creatine kinase was elevated. Electromyography revealed a myopathic pattern, muscle biopsy of the deltoid revealed an inflammatory myopathy, and skin biopsy showed interface dermatitis. There were no detectable autoantibodies except positive antinuclear antibody. He was diagnosed with immunotherapy-induced dermatomyositis, nivolumab was held, and he was treated with oral prednisone and intravenous immunoglobulin with overall improvement in myopathic and cutaneous symptoms. Dermatomyositis is an inflammatory myopathy with a characteristic dermatologic presentation that can occur spontaneously, as a paraneoplastic phenomenon, or as a drug reaction. This is the second known case of nivolumab-induced dermatomyositis. A review of the literature revealed seven total cases of immunotherapy-induced dermatomyositis. Functionally disabling autoimmune adverse effects of this severity would frequently persuade providers to discontinue immunotherapy in patients with metastatic disease.
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2.
8-Year Follow-up for Woman with Spinal Meningeal Melanocytoma in S1 Nerve Root: Case Report and Literature Review.
Yin, M, Ma, J, Ye, J, Xu, H, Mo, W
World neurosurgery. 2019;:143-147
Abstract
BACKGROUND Primary melanocytic neoplasms in the central nervous system are rare lesions arising from leptomeningeal melanocytes. These lesions produce neural deficits that resemble those of a meningioma or a schwannoma radiologically. CASE DESCRIPTION A tumor around the left S1 root with an extension into the left paraspinal compartment was identified in a 32-year-old female with persistent left leg pain for 6 months. The tumor was hyperintense on T1-weighted image and hypointense on T2-weighted image with a homogeneous enhancement. The clinical features, radiologic presentations, treatment choice, and pathologic characteristic were illustrated. The treatment outcome was compared with those reported in the previous literature. The tumor was en-bloc resected with the S1 nerve root reserved. Grossly, the tumor was a soft, capsulated, well-circumscribed, black pigmented lesion. Immunohistochemistry revealed that the tumor cells were positive for HMB-45, S-100 protein, and vimentin. The patient's symptoms were greatly relieved postoperatively. No signs of local recurrence were observed. CONCLUSIONS Spinal meningeal melanocytoma inside the nerve root is rare and benign. It is difficult to diagnose and often misdiagnosed as schwannoma or meningioma. HMB-45 has been suggested as a significant marker for the diagnosis of meningeal melanocytoma. Complete surgical resection is recommended as the primary treatment. Radiotherapy, chemotherapy, and other treatments can be selected as adjuvant therapies, but their effects are controversial. The recurrence and metastasis rates also remain unclear.
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3.
Autoimmune diabetes induced by PD-1 inhibitor-retrospective analysis and pathogenesis: a case report and literature review.
Gauci, ML, Laly, P, Vidal-Trecan, T, Baroudjian, B, Gottlieb, J, Madjlessi-Ezra, N, Da Meda, L, Madelaine-Chambrin, I, Bagot, M, Basset-Seguin, N, et al
Cancer immunology, immunotherapy : CII. 2017;(11):1399-1410
Abstract
Anti-PD-1 antibody treatment is approved in advanced melanoma and provides median overall survival over 24 months. The main treatment-related side effects are immune-related adverse events, which include rash, pruritus, vitiligo, thyroiditis, diarrhoea, hepatitis and pneumonitis. We report a case of autoimmune diabetes related to nivolumab treatment. A 73-year-old man was treated in second line with nivolumab at 3 mg/kg every two weeks for metastatic melanoma. At 6 weeks of treatment, he displayed diabetic ketoacidosis. Nivolumab was withheld 3.5 weeks and insulin therapy was initiated, enabling a normalization of glycaemia and the disappearance of symptoms. Laboratory investigations demonstrated the presence of islet cell autoantibodies, while C-peptide was undetectable. Retrospective explorations on serum banked at week 0 and 3 months before the start of nivolumab, already showed the presence of autoantibodies, but normal insulin, C-peptide secretion and glycaemia. Partial response was obtained at month 3, and nivolumab was then resumed at the same dose. The clinical context and biological investigations before, at and after nivolumab initiation suggest the autoimmune origin of this diabetes, most likely induced by anti-PD-1 antibody in a predisposed patient. The role of PD-1/PD-L1 binding is well known in the pathogenesis of type 1 diabetes. Therefore, this rare side effect can be expected in a context of anti-PD-1 treatment. Glycaemia should be monitored during PD-1/PD-L1 blockade. The presence of autoantibodies before treatment could identify individuals at risk of developing diabetes, but systematic titration may not be relevant considering the rarity of this side effect.
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4.
CAVERNOUS HEMANGIOMALIKE CHANGES IN A CHOROIDAL MELANOMA AFTER BRACHYTHERAPY.
Lopez, M, Johnson, JG, Margo, CE, Pavan, PR
Retinal cases & brief reports. 2016;(1):15-7
Abstract
PURPOSE To describe a degenerative vascular change in a treated choroidal melanoma that clinically simulated late treatment failure. METHODS Observational case report. RESULTS A 79-year-old man with choroidal melanoma treated 15 years earlier with iodine-125 brachytherapy demonstrated substantial increase in size of this once stable tumor. The eye was removed because of concern of late treatment failure. Histologically, the tumor consisted of melanocytes with no mitotic activity and virtually no Ki-67 expression. Roughly half the lesion was composed of cavernous blood-filled spaces lined by bland CD34+ and CD31+ endothelial cells. CONCLUSION Late degenerative changes in the vasculature of treated uveal melanoma can result in tumor enlargement, which may raise clinical concerns over treatment failure and neoplastic potential.
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5.
Pigmented extramammary Paget disease of the thigh mimicking a melanocytic tumor: report of a case and review of the literature.
De la Garza Bravo, MM, Curry, JL, Torres-Cabala, CA, Ivan, DS, Drucker, C, Prieto, VG, Tetzlaff, MT
Journal of cutaneous pathology. 2014;(6):529-35
Abstract
IMPORTANCE Extramammary Paget disease (EMPD) is an uncommon tumor that presents in apocrine-rich skin as an irregular, pruritic plaque. Histopathologically, EMPD consists of an intraepidermal proliferation of atypical epithelioid cells. Rarely, the tumor cells contain intracytoplasmic melanin pigment, and the lesion clinically and histopathologically can mimic a melanocytic proliferation. OBSERVATIONS A 51-year-old female with a history of breast carcinoma presented with a pigmented patch on her right thigh of 6 months duration. The clinical impression was an atypical melanocytic nevus. Histopathologic examination revealed an intraepidermal proliferation of epithelioid cells along the dermal-epidermal junction with pagetoid migration. The tumor cells exhibited increased cytoplasm containing conspicuous melanin pigment and enlarged oval-irregular nuclei. Immunohistochemical studies showed the tumor cells to be strongly and diffusely positive for cytokeratin 8/18, cytokeratin 7 and p63; focally and weakly positive for epithelial membrane antigen (EMA), but negative for cytokeratin 5/6, Cam5.2, carcinoembryonic antigen (CEA), human melanoma black 45 (HMB-45), tyrosinase and Sox-10, supporting the diagnosis of pigmented EMPD. The lesion was subsequently excised, and the patient is free of disease after 24 months. CONCLUSION We present this unusual case of pigmented EMPD arising on the thigh to draw attention to the entity and to underscore the potentially misleading clinical, histopathologic and immunophenotypic features that mimic other cutaneous intraepidermal lesions.
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6.
[A case of anorectal melanoma localized in mucosa].
Ito, T, Kondo, A, Tanaka, M, Naganuma, T, Hashimoto, A, Kawamata, H, Isaji, S
Nihon Shokakibyo Gakkai zasshi = The Japanese journal of gastro-enterology. 2012;(3):435-41
Abstract
The patient was a 67-year-old woman. An elevated rectal lesion had been identified at a local clinic, and colonoscopy revealed a 7-mm black elevated lesion with blackening of the surrounding mucosa. Malignant melanoma was diagnosed by biopsy, and the patient was referred to our department. An invasion depth of M was diagnosed on the basis of preoperative imaging. Local excision was considered, but abdominoperineal resection was performed in light of its nature, to perform curative resection. Pathological findings showed abnormal cells staining positive for HMB-45 in the tumor and surrounding mucosa, but these were localized within the mucosa. As of 1 year and 6 months after surgery, the patient has survived without recurrence.
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7.
Melanoma-associated retinopathy report of a case and review.
Bahig, H, Wein, F, Del Maestro, RF
The Canadian journal of neurological sciences. Le journal canadien des sciences neurologiques. 2011;(5):797-8
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8.
Colonic malignant melanoma, primary or metastatic? Case report.
Serin, G, Doğanavşargil, B, Calişkan, C, Akalin, T, Sezak, M, Tunçyürek, M
The Turkish journal of gastroenterology : the official journal of Turkish Society of Gastroenterology. 2010;(1):45-9
Abstract
Gastrointestinal malignant melanomas, either primary or metastatic, are rare and overlooked tumors. There is also controversy regarding the actual existence of primary melanoma in the gastrointestinal tract apart from the esophagus and anorectal regions, where melanocytes normally exist. A case of malignant melanoma in the cecum is presented. The patient was a 30- year-old male who presented to the hospital for abdominal pain and diarrhea. The tumor was located mainly in the submucosa and measured 14x11x4.5 cm. The cut surface was solid, gray-white and fleshy. Histologically, tumor cells were arranged in compact nests or wide cords surrounded by fibrous stroma. The tumor cells had pleomorphic nuclei and quite rich cytoplasm; multinucleated, giant tumor cells were intermingled. Although no tumor cells contained apparent brown pigment, most were found to be positive for S-100 protein, HMB-45, Melan-A, and vimentin. The possibility of a metastatic lesion was considered. While the patient had a history of a pathologically examined dorsal nevus excision two years before, there was no evidence of either cutaneous or ocular primary melanoma at the time of diagnosis. Moreover, a thorough postoperative investigation did not reveal any other lesion in any other site favoring a metastatic spread. There was also no evidence of recurrent disease or metastasis one year after the surgery. This case is presented in view of its rare occurrence in the cecum. The difficulties in the diagnostic course are discussed, together with a literature review on distinguishing a primary mucosal melanoma from a metastatic one from an unknown or regressed cutaneous primary tumor.
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9.
Diffuse primary leptomeningeal melanocytosis in a patient receiving a novel cancer cell vaccine for prostate cancer.
Michael, BD, Syndikus, I, Clark, A, Baborie, A
BMJ case reports. 2010
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Abstract
UNLABELLED A 75-year-old man, undergoing treatment for metastatic prostate cancer with a novel cancer cell vaccine, presented with a 4 week history of poor balance, gait disturbance and cognitive decline. Blood tests including HIV and onconeuronal and voltage gated potassium channel antibodies were normal. Computed tomography and two magnetic resonance images of the brain showed possible non-specific meningeal or vascular enhancement. Two cerebrospinal fluid analyses, including cytology, were negative, other than six lymphocytes in the former. Despite intravenous aciclovir and dexamethasone the patient deteriorated over 16 days, with worsening confusion and involuntary movements, and died. Postmortem examination showed that the leptomeninges overlying the brain and spinal cord were diffusely infiltrated by a melanocytosis with a focal area of melanomatosis. Moreover, there were two sites of metastases of a highly malignant clone present in the pulmonary parenchyma. TRIAL REGISTRATION NUMBER NCT00133224.
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10.
Significant response after treatment with the mTOR inhibitor sirolimus in combination with carboplatin and paclitaxel in metastatic melanoma patients.
Meier, F, Guenova, E, Clasen, S, Eigentler, T, Forschner, A, Leiter, U, Zielinski, C, Knaudt, B, Garbe, C, Berneburg, M
Journal of the American Academy of Dermatology. 2009;(5):863-8
Abstract
Melanoma is highly resistant to chemotherapy. In melanoma, the PI3K-AKT-mTOR signaling pathway is constitutively activated through multiple mechanisms. Several experimental studies suggest that targeting the PI3K-AKT-mTOR signaling pathway is a promising strategy to overcome chemoresistance. This is the first report describing a chemosensitizing effect of mTOR inhibition in patients with melanoma. We report two cases of patients with metastatic melanoma who showed significant remission after combination of carboplatin and paclitaxel with the mTOR inhibitor sirolimus. Our case report, together with the literature discussed, suggests that mTOR inhibition possibly enhances the sensitivity of melanoma cells to chemotherapy and should prompt in-depth and clinical investigation.