Purely Meningeal Intracranial Relapse of Melanoma Brain Metastases After Surgical Resection and Immunotherapy as a Unique Disease Progression Pattern: Our Experience and Review of the Literature.
- 作者列表："Armocida D","Marzetti F","Pesce A","Caporlingua A","D'Angelo L","Santoro A
BACKGROUND:We present a case of 72-year-old man with a history of metastatic melanoma diagnosed in 2015 presenting a stable disease in treatment with dabrafenib. CASE DESCRIPTION:The patient had been surgically treated for a presumed intracranial parietooccipital metastasis. He presented 1 month later with a meningeal lesion associated with a subdural hematoma. A second surgical treatment confirmed the diagnosis of meningeal recurrence of metastatic melanoma. CONCLUSIONS:The most recent literature lacks studies defining the clinical phenomena of an early recurrence of intracranial melanoma with de novo involvement of dural compartment in patients in treatment with a target immunotherapy. The aim of this present study is to report a case of early recurrence of intracranial melanoma metastases with evidence of fast immunohistochemical and macroscopical mutation of pathologic elements, with an analysis of literature that shows the lack of well-described occurrences.
背景: 我们介绍了一例 2015 年诊断为转移性黑色素瘤病史的 72 岁男性患者，在dabrafenib治疗中疾病稳定。 病例描述: 患者曾因推定的颅内枕叶旁转移接受手术治疗。1 个月后，他出现脑膜病变伴硬膜下血肿。第二次手术治疗证实了转移性黑色素瘤脑膜复发的诊断。 结论: 最近的文献缺乏定义颅内黑色素瘤早期复发的临床现象的研究，在靶向免疫治疗的患者中，硬脑膜室重新受累。本研究的目的是报道一例颅内黑色素瘤转移早期复发的病例，该病例有快速免疫组化和病理成分宏观突变的证据，对文献的分析表明缺乏描述良好的事件。
METHODS::Blue rubber bleb naevus syndrome (BRBNS) is an extremely rare venous malformation that often manifests as multiple haemangioma-like lesions in the skin and gastrointestinal tract. The drug sirolimus plays a key role in the signalling pathway of angiogenesis and subsequent development of BRBNS and its use has been described in several case reports. We present a case series of four patients with BRBNS who exhibited good treatment response to sirolimus. All four patients were administered oral sirolimus at doses of 1.0-1.5 mg/m2 /day with a target drug level of 5-10 ng/mL and median treatment duration of 20 months. All patients had a reduction in the size of the lesions and a normalization of coagulopathy with tolerable drug adverse reactions at follow-up. Sirolimus may be effective and safe in paediatric patients with BRBNS. Further prospective studies are suggested to evaluate the long-term effectiveness of this drug.
METHODS:BACKGROUND:Human papillomavirus (HPV) infections are associated with common dermatologic and nondermatologic diseases. Although HPV vaccines are well established as preventive measures for genital warts and cervical neoplasia, their use as therapeutic agents deserves greater attention. OBJECTIVE:To evaluate the use of HPV vaccine(s) as a treatment modality for cutaneous and/or mucosal disease. METHODS:A primary literature search using the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines was conducted in January 2019 by using the PubMed and Cochrane databases. RESULTS:A total of 63 articles with 4439 patients were included. The majority of patients with cutaneous warts, recurrent respiratory papillomatosis, and squamous and basal cell carcinomas were successfully treated with HPV vaccination. Preliminary data on patients with pre-existing anogenital warts, cervical intraepithelial neoplasia, anal intraepithelial neoplasia, and vulvar intraepithelial neoplasia is promising. LIMITATIONS:This review was limited by the lack of controls, patients' previous HPV vaccination status, and publication bias. CONCLUSION:The commercially available three-dose, quadrivalent HPV vaccine is a potential therapeutic option for the treatment of cutaneous warts, recurrent respiratory papillomatosis, and squamous and basal cell carcinomas. Noncommercially available HPV vaccines demonstrate therapeutic response for treating anogenital warts, cervical intraepithelial neoplasia, anal intraepithelial neoplasia, and vulvar intraepithelial neoplasia. The vaccine's efficacy as an adjunct therapy for HPV-associated cutaneous and/or mucosal disease warrants further exploration.
METHODS::Our understanding of melanoma precursors and progression to melanoma has developed as a result of advances in the field of molecular diagnostics. We now better understand the potential for genetic heterogeneity within a single lesion. Combined tumors can pose a diagnostic challenge when deciding the line between benign and malignant, which in turn has direct implications for patient management. Primary cilia (PC) are ubiquitous sensory organelles that have essential functions in cellular proliferation, differentiation, and development. The ciliation index (percentage of ciliated melanocytes) has been shown to reliably differentiate melanoma, which fail to ciliate, from melanocytic nevi, which retain PC. We therefore analyzed the potential for using the ciliation index to differentiate benign and malignant components in combined melanocytic lesions. We collected patient samples (n = 10) of unequivocal combined lesions with both melanoma and associated nevus components. Melanocytes were highlighted with SOX10 and costained with gamma-Tubulin and acetylated alpha-Tubulin to highlight the basal body and cilium, respectively. The number of melanocytes retaining cilia under high-power microscopy was examined. The melanoma component had average of 4% ciliation (SD: 7%), whereas the associated nevus component was significantly higher with 59% ciliation (SD: 17%). These data show that PC may be a reliable means of distinguishing benign from malignant components within a single tumor. The ciliation index may be a helpful tool in distinguishing challenging cases of combined lesions of melanoma in situ with a dermal nevus component from invasive melanoma, thus promoting improved staging and clinical management.