A 40-year-old man presented with drooping of both eyelids and generalized weakness of two weeks duration. The symptoms were worse during the evening. He had been diagnosed with chronic myeloid leukaemia (CML) in chronic phase, four months previously, and was receiving regular imatinib, 400 mg/day. After three weeks of therapy, he had achieved a complete haematological response, and his constitutional symptoms had subsided. After three months of therapy, he showed a two-log reduction of BCR-ABL1 transcript. On presentation, he had bilateral ptosis (left). Physical examination was otherwise normal. A provisional diagnosis of ocular myasthenia gravis was made. He showed improvement in ptosis after administration of neostigmine. Repetitive nerve stimulation and anti-acetylcholine receptor antibody serology were positive, confirming the diagnosis of myasthenia gravis. He was started on pyridostigmine and prednisolone. Computed tomography of his chest showed a 2Á7 9 1Á7 cm mass in the anterior mediastinum, for which he underwent thymectomy. Histopathological examination showed thymic hyperplasia. He is currently asymptomatic with marked improvement in ptosis (right) and has achieved a partial molecular response (BCR-ABL1 transcript level, 1Á113%).Many cases of drug-induced myasthenia gravis have been reported. Drugs implicated include ribavirin, statins, fluoroquinolones, interferon, fludarabine, chloroquine, penicillamine, busulfan and carbamazepine. Development of myasthenia gravis during treatment with nilotinib has also been reported in a patient with CML. In the current case, myasthenia gravis was unmasked when the patient was treated with imatinib. Triggering of myasthenia by tyrosine kinase inhibitors and certain other drugs may be related to a disturbance of immune function. However, other possibilities, including a paraneoplastic manifestation of CML or co-incidental occurrence of myasthenia gravis and CML, cannot be ruled out.
Minimally invasive parathyroidectomy (MIP) is the standard of care for primary hyperparathyroidism (PHPT). Four dimensional computed tomography(4DCT) and F-18 Fluorocholine positron emission tomography/computed tomography (FCH PET/CT) localize adenomas accurately to perform MIP. We aimed to conduct a systematic review and metanalysis to evaluate the diagnostic performance of 4DCT and FCH PET/CT scan for quadrant wise localisation in PHPT patients and to do head-to-head comparison between these two modalities.Design, Patients and Measurement : After searching through PubMed and EMBASE databases, 46 studies (using histology as a gold standard) of 4DCT and FCH PET/CT were included.Results: Total number of patients included were 1651 and 952 for 4DCT scan (studies n = 26) and FCH PET/CT scan (studies n = 24) respectively. In per patient analysis, FCH PET/CT and 4DCT had pooled sensitivities of 92% (88−94) and 85% (73−92) respectively and in per lesion analysis, 90% (86−93) and 79% (71−84), respectively. In the subgroup with negative conventional imaging/persistent PHPT, FCH PET/CT had comparable sensitivity to 4DCT (84% [74−90] vs. 72% [46−88]). As per patient wise analysis, FCH PET/CT had better detection rates than 4DCT ([92.4 vs. 76.85], odds ratio −3.89 [1.6−9.36] p = .0024) in the subpopulation where both FCH PET/CT and 4DCT were reported. Conclusion:Both 4DCT and FCH PET/CT scan performed well in newly diagnosed patients, patients with persistent disease and in those with inconclusive conventional imaging results. FCH PET/CT scan had a higher pooled sensitivity than 4DCT in detecting patients with PHPT in head to head comparison.
Background Leucovorin (folinic acid) is a commonly used antidote for severe toxicity with low-dose methotrexate, but its optimum dose is unclear, varying from 15 to 25 mg every 6-h. Methods Open-label RCT included patients with severe low-dose (≤ 50 mg/week) methotrexate toxicity defined as WBC ≤ 2 × 10^9/L or platelet ≤ 50 × 10^9/L and randomized them to receive either usual (15 mg) or high-dose (25 mg) intravenous leucovorin given every 6-h. Primary outcome was mortality at 30-days and secondary outcomes were hematological recovery and mucositis recovery. Trial Registration number: CTRI/2019/09/021152. Results Thirty-eight patients were included, most with underlying RA who had inadvertently overdosed MTX (taken daily instead of weekly). At randomization, the median white blood and platelet count were 0.8 × 10^9/L and 23.5 × 10^9/L. 19 patients each were randomized to receive either usual or high-dose leucovorin. Number (%) of deaths over 30-days was 8 (42) and 9 (47) in usual and high-dose leucovorin groups (Odds ratio 1.2, 95% CI 0.3 to 4.5, p = 0.74). On Kaplan–Meier, there was no significant difference in survival between the groups (hazard ratio 1.1, 95% CI 0.4 to 2.9, p = 0.84). On multivariable cox-regression, serum albumin was the only predictor of survival (hazard ratio 0.3, 95% CI 0.1 to 0.9, p = 0.02). There was no significant difference in hematological or mucositis recovery between the two groups. Conclusion There was no significant difference in survival or time-to hematological recovery between the two doses of leucovorin. Severe low-dose methotrexate toxicity carried a significant mortality.
Management of ANCA-associated vasculitis (AAV) during the COVID-19 pandemic poses unique therapeutic challenges. An online survey was conducted to understand physician’s choices for treating AAV during the COVID-19 pandemic. Web-based survey featuring nineteen questions was circulated amongst physicians across various specialties. The responses regarding immunosuppressive therapy for remission induction and maintenance, COVID-19 testing, and preventive measures were recorded. A total of 304 responses were recorded. Most of the respondents were from India (83.9%) and comprised rheumatologists (66%) in practice for ≥ 5 years (71%). Though a majority preferred Rituximab or intravenous cyclophosphamide (CYC) as a remission induction agent, a significant proportion opted for oral CYC and mycophenolate mofetil (MMF) also. Only one-third wanted to test for COVID-19 before initiating immunosuppressive therapy in patients with organ/life-threatening manifestations. Rituximab was the most favored maintenance therapy (47%), followed by azathioprine, MMF, and methotrexate. The results of this focused survey of managing AAV patients depict the real-world dilemmas and physicians’ choices in this setting.
M ycobacterium tuberculosis infects approximately 1.7 billion people in the world and causes disease in 10 million annually. Musculoskeletal tuberculosis accounts for 2% to 5% of cases, generally presenting as Pott's spine or large joint arthritis 1 ; the involvement of muscle by tubercle bacilli, referred to as tuberculous myositis, is rare. Muscle involvement can occur either due to hematogenous spread (primary) or as a result of extension from a contiguous site (secondary); the former being less common. 2 We report a case of primary tuberculous myositis and review all cases published in the last 25 years. CASE REPORTA 49-year-old man was admitted with swelling and dullaching pain over the right gluteal region, fever, and loss of appetite for 6 months. Three years prior to the current presentation, he had been diagnosed with dermatomyositis and treated with corticosteroids and azathioprine; these drugs were tapered and stopped after 2 years. With a possibility of relapse, corticosteroids were restarted 1 month before his admission.On examination, a localized tender swelling was present over the right gluteal area and posterior thigh, extending to the knee. Magnetic resonance imaging of this region revealed active myositis of the gluteal muscle and muscles in the posterior compartment of the right thigh (Figs. 1A, B). Positron emission tomographycomputed tomography showed uptake in these muscles, and an image-guided biopsy was performed. This revealed granulomatous inflammation on histopathology (Fig. 2), and a cartridge-based nucleic acid amplification test GeneXpert MTB/RIF (Cepheid, Sunnyvale, CA) detected M. tuberculosis. Computed tomography of the thorax did not show any pulmonary tuberculosis; however, tuberculin skin test was positive. Antitubercular therapy was started with isoniazid, rifampicin, ethambutol, and pyrazinamide for 2 months, followed by isoniazid, rifampicin, and ethambutol for 8 months. The fever resolved within 2 weeks, and over the next few months, the swelling subsided, and the patient gained weight. At 1 year, he was found to be asymptomatic and disease-free. METHODS FOR SYSTEMATIC REVIEWWe carried out a search in PubMed (MEDLINE), Scopus, EMBASE, and Web of Science, using the words "tubercular myositis," "tubercular pyomyositis," "tuberculous myositis," or "tuberculous pyomyositis." This review was restricted to articles in English language that were published between January 1995 and December 2019. Abstracts and full texts of relevant articles were examined by 2 authors (H.S., V.D.), and those articles that described cases of primary tuberculous myositis confirmed by microbiological tests (or on histopathology) were included for review. The outcome of cases was noted, factors associated with death analyzed, and odds ratios (ORs) (95% confidence intervals [CIs]) calculated. Analysis was done by GraphPad Prism (version 8 for Windows, La Jolla, CA).
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