scholarly journals Paraneoplastic Necrotizing Autoimmune Myopathy in a Patient Undergoing Laparoscopic Pancreatoduodenectomy for Distal Cholangiocarcinoma

2016 ◽  
Vol 10 (3) ◽  
pp. 525-530 ◽  
Author(s):  
Stefan van Dijk ◽  
Anneke J. van der Kooi ◽  
Eleonora Aronica ◽  
Thomas M. van Gulik ◽  
Olivier R. Busch ◽  
...  
Keyword(s):  
Single Dose ◽  
Muscle Strength ◽  
Intravenous Immunoglobulin ◽  
General Condition ◽  
Pulse Therapy ◽  
Distal Cholangiocarcinoma ◽  
Necrotizing Autoimmune Myopathy ◽  
Laparoscopic Pancreatoduodenectomy ◽  
Autoimmune Myopathy ◽  
Dexamethasone Pulse Therapy

A 73-year-old male presented with jaundice and severe muscle weakness. He was diagnosed with distal cholangiocarcinoma and paraneoplastic necrotizing autoimmune myopathy (NAM). Treatment of NAM consisted of dexamethasone pulse therapy, prednisone, and single-dose intravenous immunoglobulin. The distal cholangiocarcinoma was resected through a total laparoscopic pancreatoduodenectomy. After hospital discharge, muscle strength initially increased postoperatively; however, pneumonia resulted in the deterioration of his general condition and death 5 months after the diagnosis of paraneoplastic NAM.

Combined Central Retinal Vein and Cilioretinal Artery Occlusion in the Setting of Intravenous Immunoglobulin Administration

2021 ◽  
pp. 247412642199860
Author(s):  
Jonathan Cin-Bon Tsui ◽  
Gordon Scott Crabtree
Keyword(s):  
Intravenous Immunoglobulin ◽  
Vascular Occlusion ◽  
Artery Occlusion ◽  
Necrotizing Autoimmune Myopathy ◽  
Cilioretinal Artery Occlusion ◽  
Cilioretinal Artery ◽  
Optical Coherence Tomography Imaging ◽  
Autoimmune Myopathy ◽  
Central Retinal Vein ◽  
Ivig Administration

Purpose: This work reports a case of combined vascular occlusion in the setting of intravenous immunoglobulin (IVIg) administration. Methods: The authors describe a case of combined central retinal vein and cilioretinal artery that occurred in the setting of IVIg administration. Results: A 52-year-old White man presented with a unilateral subjective scotoma that began during IVIg administered for the treatment of statin-induced necrotizing autoimmune myopathy. Examination and optical coherence tomography imaging revealed a combined nonischemic central retinal vein and cilioretinal artery occlusion. Conclusions: To the authors’ review and knowledge, this is the first reported case of combined central retinal vein and cilioretinal artery occlusion occurring in the setting of IVIg administration. This rare adverse effect is an entity to be considered in patients who are treated with IVIg.

Rapidly Progressive Proximal Weakness

2021 ◽  
pp. 152-153
Author(s):  
Teerin Liewluck ◽  
Margherita Milone
Keyword(s):  
Creatine Kinase ◽  
Mycophenolate Mofetil ◽  
Intravenous Immunoglobulin ◽  
Muscle Weakness ◽  
Creatine Kinase Level ◽  
Coenzyme A ◽  
Immune Mediated ◽  
Necrotizing Autoimmune Myopathy ◽  
History Of ◽  
Autoimmune Myopathy

A 53-year-old woman had development of subacute-onset muscle weakness resulting in difficulty climbing stairs, rising from a chair, and reaching over her shoulders. She reported no dysphagia, dysarthria, dyspnea, or diplopia. She also disclosed no rash, joint pain, or urine discoloration. She had no history of statin exposure. There was no family history of neuromuscular disorders, early cataracts, cardiac arrhythmia, or cardiomyopathy. Two months of treatment with prednisone had resulted in no clinical improvement. Neurologic examination indicated moderate neck flexor, shoulder, and hip girdle muscle weakness, with sparing of cranial muscles. There was no action- or percussion-induced myotonia. Needle electromyography showed short-duration, low-amplitude, and complex motor unit potentials, predominantly affecting proximal muscles, associated with fibrillation potentials and myotonic discharges in proximal and axial muscles. Her creatine kinase level was increased. Biopsy of the left quadriceps showed variation in muscle fiber size, a moderate increase in internalized nuclei, fiber splitting, and scattered necrotic and regenerating fibers. There was a mild increase in perimysial fibrous and fatty connective tissue. 3-Hydroxy-3-methylglutaryl–coenzyme A reductase antibodies were strongly positive. The patient was diagnosed with hydroxy-3-methylglutaryl–coenzyme A reductase antibody–positive necrotizing autoimmune myopathy. The patient received intravenous immunoglobulin and mycophenolate mofetil while continuing prednisone. At 1-year follow-up, she had no weakness, and her creatine kinase value was normal while she continued taking prednisone, mycophenolate mofetil, and intravenous immunoglobulin. Necrotizing autoimmune myopathy, or immune-mediated necrotizing myopathy, is a subtype of immune-mediated myopathy, clinically characterized by subacute, progressive, proximal limb weakness and persistently increased creatine kinase level. Pathologically, it is characterized by myonecrosis with minimal or no inflammation. One-third of patients with necrotizing autoimmune myopathy have myalgia.

Role of Intravenous Immunoglobulin in Necrotizing Autoimmune Myopathy

2021 ◽  
Vol Publish Ahead of Print ◽  
Author(s):  
Amanda Kocoloski ◽  
Silvia Martinez ◽  
Siamak Moghadam-Kia ◽  
David Lacomis ◽  
Chester V. Oddis ◽  
...  
Keyword(s):  
Intravenous Immunoglobulin ◽  
Necrotizing Autoimmune Myopathy ◽  
Autoimmune Myopathy

scholarly journals Extended single-dose toxicity study of [211At]NaAt in mice for the first-in-human clinical trial of targeted alpha therapy for differentiated thyroid cancer

2021 ◽  
Author(s):  
Tadashi Watabe ◽  
Kazuko Kaneda-Nakashima ◽  
Kazuhiro Ooe ◽  
Yuwei Liu ◽  
Kenta Kurimoto ◽  
...  
Keyword(s):  
Clinical Trial ◽  
Body Weight ◽  
Thyroid Cancer ◽  
Single Dose ◽  
General Condition ◽  
Blood Test ◽  
Differentiated Thyroid Cancer ◽  
Targeted Alpha Therapy ◽  
Evaluation Point ◽  
Alpha Therapy

Abstract Objective Astatine (211At) is a promising alpha emitter as an alternative to iodine (131I). We are preparing the first-in-human (FIH) clinical trial of targeted alpha therapy for differentiated thyroid cancer in consultation with Pharmaceuticals and Medical Devices Agency. Here, we performed an extended single-dose toxicity examination under a reliability standard, as a preclinical safety assessment of [211At]NaAt to determine the FIH dose. Methods [211At]NaAt solution was injected into normal 6-week-old mice (male (n = 50) and female (n = 50), body weight: male 33.2 ± 1.7 g, female 27.3 ± 1.5 g), which were then divided into four groups: 5 MBq/kg (n = 20), 20 MBq/kg (n = 20), 50 MBq/kg (n = 30), saline control (n = 30). The mice were followed up for 5 days (primary evaluation point for acute toxicity: n = 80) or 14 days (n = 20: evaluation point for recovery) to monitor general condition and body weight change. At the end of the observation period, necropsy, blood test, organ weight measurement, and histopathological examination were performed. For body weight, blood test, and organ weight, statistical analyses were performed to compare data between the control and injected groups. Results No abnormal findings were observed in the general condition of mice. In the 50 MBq/kg group, males (days 3 and 5) showed a significant decrease in body weight compared with the control. However, necropsy did not differ significantly beyond the range of spontaneous lesions. In the blood test, males (50 MBq/kg) and females (50 MBq/kg) showed a decrease in white blood cell and platelet counts on day 5, and recovery on day 14. In the testis, a considerable weight decrease was observed on day 14 (50 MBq/kg), and multinucleated giant cells were observed in all mice, indicating a significant change related to the administration of [211At]NaAt. Conclusions In the extended single-dose toxicity study of [211At]NaAt, administration of high doses resulted in weight loss, transient bone marrow suppression, and pathological changes in the testis, which require consideration in the FIH clinical trial.

Necrotizing autoimmune myopathy

2011 ◽  
Vol 23 (6) ◽  
pp. 612-619 ◽  
Author(s):  
Christina Liang ◽  
Merrilee Needham
Keyword(s):  
Necrotizing Autoimmune Myopathy ◽  
Autoimmune Myopathy

scholarly journals Necrotizing myopathy presenting as congestive heart failure and life-threatening ventricular arrhythmias: a case report

2021 ◽  
Vol 5 (6) ◽  
Author(s):  
Kyunghee Lim ◽  
Jong Sung Park ◽  
Byeol-A Yoon ◽  
Song-Hee Han
Keyword(s):  
Heart Failure ◽  
Congestive Heart Failure ◽  
Ventricular Arrhythmias ◽  
High Dose ◽  
Respiratory Impairment ◽  
Motor Weakness ◽  
High Dose Steroid ◽  
Necrotizing Autoimmune Myopathy ◽  
Life Threatening ◽  
Autoimmune Myopathy

Abstract Background Necrotizing autoimmune myopathy is a rare subtype of idiopathic inflammatory myopathy; however, it can be associated with fatal cardiac manifestations. Case summary A 58-year-old female patient was referred for congestive heart failure with dysrhythmia. Electrocardiograms showed ventricular arrhythmias of various QRS complex morphologies and coupling intervals with beat-to-beat differences. Despite optimal medical therapy for heart failure, the patient was admitted for the progression of dyspnoea and generalized motor weakness. The burden of non-sustained ventricular tachycardia gradually increased, and ventricular fibrillation eventually occurred. In view of a differential diagnosis of an inflammatory myocardial diseases such as sarcoidosis, a cardiac biopsy was performed. However, pathologic examinations revealed only necrotic muscle fibres without granuloma. Further examinations revealed proximal dominant motor weakness, an elevated serum creatinine-phosphokinase level, myogenic potentials on needle electromyography, and biceps muscle biopsy findings that were compatible with necrotizing autoimmune myopathy. High-dose steroid therapy improved the patient’s motor weakness, including her respiratory impairment, and successfully suppressed ventricular arrhythmias. Discussion This case suggests that intensive immunosuppressive therapy with high-dose steroid could be useful in the necrotizing autoimmune myopathy manifested as congestive heart failure and life-threatening ventricular arrhythmias.

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