Rodriguez Erika F, Blakeley Jaishri, Langmead Shannon, Olivi Alessandro, Tufaro Anthony, Tabbarah Abeer, Berkenblit Gail, Sacks Justin M, Newsome Scott D, Montgomery Elizabeth, Rodriguez Fausto J
Departments of Pathology, Johns Hopkins University School of Medicine, Baltimore, MD 21231.
Department of Neurology, Johns Hopkins University School of Medicine, Baltimore, MD 21231; Department of Oncology, Johns Hopkins University School of Medicine, Baltimore, MD 21231.
Hum Pathol. 2017 Feb;60:121-128. doi: 10.1016/j.humpath.2016.08.010. Epub 2016 Sep 22.
Leptomeningeal dissemination of low-grade Schwann cell neoplasms is an exceptionally rare occurrence and has not been well documented in the literature. We encountered 2 cases of leptomeningeal dissemination of low-grade Schwann cell neoplasms. Patient 1 was a 63-year-old woman with neurofibromatosis type 1 and a progressive low-grade malignant peripheral nerve sheath tumor developing from a diffuse/plexiform orbital neurofibroma that arose in childhood. The neoplasm demonstrated local and leptomeningeal dissemination intracranially leading to the patient's death. There was partial loss of H3K27 tri-methylation, p16 and collagen IV. Patient 2 was a 60-year-old man without neurofibromatosis type 1 who presented with cranial nerve symptoms and a disseminated neoplasm with a Schwann cell phenotype. The neoplasm stabilized after irradiation and chemotherapy, but the patient died of medical complications. Autopsy findings documented disseminated leptomeningeal disease in the intracranial and spinal compartment. H3K27M tri-methylation was preserved. The clinicopathologic and autopsy findings are studied and presented, and the literature is reviewed.
低级别施万细胞瘤的软脑膜播散极为罕见,文献中对此记载不多。我们遇到了2例低级别施万细胞瘤的软脑膜播散病例。病例1是一名63岁女性,患有1型神经纤维瘤病,患有由儿童期出现的弥漫性/丛状眼眶神经纤维瘤发展而来的进展性低级别恶性外周神经鞘瘤。该肿瘤在颅内表现出局部和软脑膜播散,导致患者死亡。H3K27三甲基化、p16和IV型胶原存在部分缺失。病例2是一名60岁男性,无1型神经纤维瘤病,表现为颅神经症状和具有施万细胞表型的播散性肿瘤。肿瘤在放疗和化疗后病情稳定,但患者死于医疗并发症。尸检结果显示颅内和脊髓腔存在播散性软脑膜疾病。H3K27M三甲基化得以保留。对临床病理和尸检结果进行了研究和展示,并对文献进行了综述。