Cutaneous necrotising vasculitis induced by levamisoleThe anthelmintic agent levamisole has immunostimulant properties in patients with defective cell-mediated immune responses. We are assessing the drug in patients with breast cancer to see whether it can maintain surgically induced remission. One such patient developed a severe cutaneous necrotising vasculitis, which disappeared once the drug was withdrawn. Case reportA 59-year-old woman had been receiving thrice-weekly levamisole 150 mg/day for three months, when in May 1975 she developed fever and a severe rash. Cutaneous necrotising vasculitis was diagnosed. Biopsy of one of the lesions showed intense neutrophil and eosinophil infiltration of the vessel wall with obliteration of the lumen. There were no other physical abnormalities, and no sign of the original disease was noted. She had not been taking any other drugs.Haemoglobin was 13 g/dl and white cell count 3 0 x 109/1 (3000/mm3; 30 % segmented neutrophils, 8 0o eosinophils, 10 Po monocytes, 52°lymphocytes). Results of complement studies were within normal limits, and other immunological and biochemical values were normal. A bone-marrow aspirate showed a normal distribution of white and red cells but a moderate increase in eosinophils. A chest radiograph was normal.Levamisole was discontinued and the patient given a short course of prednisone 40 mg daily. After two weeks the clinical picture returned to normal, and three months later the skin lesions showed no signs of recurrence and the white cell count was normal. CommentLevamisole-induced vasculitis has not been reported, despite wide use of the drug in various conditions, including malignant and rheumatic diseases.1-3 The pathogenesis of our patient's skin reaction is unknown, though histologically it was similar to an Arthus-type reaction, in which immune-complex formation or complement activation is usually implicated. We found no complement abnormalities in our patient, but the tests did not exclude a local type III reaction.The peripheral blood neutropenia in our patient also implicates levamisole as the causal agent, since neutropenia and agranulocytosis are associated with levamisole treatment.3 The exact mechanism of this reaction is unknown, but the peripheral and central eosinophilia suggest a hypersensitivity reaction. Although we did not challenge the patient (for ethical reasons) we think that levamisole was the likely cause of the vasculitis.
Flow diverters are new generation stents that have recently garnered a large amount of interest for use in treatment of intracranial aneurysms. Flow diverters reduce blood flow into the aneurysm, with redirection along the path of the parent vessel. Flow stagnation into the aneurysm and neck coverage with subsequent endothelialization are the important synergistic mechanisms by which the therapy acts. Several studies have examined the mechanisms by which flow diverters subsequently lead to aneurysm occlusion. This review aims to provide a general overview of the flow diverters and their mechanism of action and potential implications. ANN NEUROL 2019;85:793-800 View this article online at wileyonlinelibrary.com.
Bilateral thalamic primary gliomas are an exceedingly rare entity. Symptomology heralding a workup and diagnosis of bithalamic gliomas is diverse and varies between the pediatric and adult populations. Herein, we present a case of a 63-year-old female patient who presented with progressive gait imbalance and fatigue, prompting an outpatient brain MRI, remarkable for marked expansion of the bilateral thalami secondary to non-enhancing, T2-weighted-fluid-attenuated inversion recovery (T2-FLAIR) bright bithalamic lesions. The patient underwent a right frontal frameless stereotactic biopsy of the right thalamic lesion, with immuno-histology indicating a high-grade anaplastic astrocytoma with molecular features of glioblastoma (GBM). The patient’s functional status declined precipitously in the month following her diagnostic biopsy, precluding any therapy, and the patient ultimately pursued home hospice care without further treatment. This case details the clinical management of a very rare tumor, supplementing the available literature on the progression and treatment of this rare disease.
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BACKGROUND Klippel-Trénaunay syndrome (KTS) is a combined capillary-lymphatic-venous malformation disorder traditionally associated with high surgical morbidity. Although rare, pathologic involvement of the spinal cord has been reported in the literature. However, the safety of surgical intervention remains unclear. We report a case of successful decompression of a thoracic epidural lesion in an individual with KTS who presented with spastic paraparesis. OBSERVATIONS The patient is a 38-year-old male, diagnosed with KTS as an infant, who presented with spastic paraparesis secondary to a thoracic epidural lesion. He underwent laminectomies and resection of the lesion with subsequent improvement of his symptoms and without significant postoperative morbidity. Histopathology confirmed the lesion to be a benign vascular malformation. LESSONS Currently, the literature regarding management of symptomatic vascular lesions in individuals with KTS supports nonoperative management, due to the increased risk of operative morbidity associated with this syndrome. This case presents evidence for safe and appropriate surgical management of a thoracic epidural vascular malformation in a patient with KTS in the setting of progressive neurological decline, establishing a role for neurosurgical intervention in this high-risk population when no conservative management portends further neurological deterioration.
Epidermoid cysts of the pineal region are a rare entity. Herein, the authors describe the endoscopic resection of a recurrent pineal region epidermoid by way of a supracerebellar infratentorial approach. The patient was positioned in the semiseated upright position with head tilted to the right and slightly flexed, maximizing gravity-based cerebellar retraction, and a paramedian craniotomy was performed owing to the gradual flattening of the tentorium from medial to lateral. This setup, in tandem with the enlarged viewing window achieved by use of 0°, 30°, and 70° endoscopes, afforded the necessary access to achieve a satisfactory resection through this anatomical corridor. The video can be found here: https://stream.cadmore.media/r10.3171/2021.4.FOCVID2131.
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