Reyes-Leiva David, Carbayo Álvaro, Vesperinas-Castro Ana, Rojas-García Ricard, Querol Luis, Turon-Sans Janina, Pla-Junca Francesc, Olivé Montse, Gallardo Eduard, Pujades-Rodriguez Mar, Cortés-Vicente Elena
Neuromuscular Diseases Unit, Department of Neurology, Hospital de la Santa Creu i Sant Pau; and Institut de Recerca Sant Pau, IR-SantPau, Barcelona, Spain.
Centro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER, Valencia, Spain.
Eur J Neurol. 2025 Jan;32(1):e16463. doi: 10.1111/ene.16463. Epub 2024 Dec 3.
Generalized myasthenia gravis (gMG) is characterized by fluctuating muscle weakness. Exacerbation frequency, adverse events (AEs) related to immunosuppressant therapy and healthcare resource utilization (HCRU) are not well understood. Our study aimed to describe long-term clinical outcomes, drug-related AEs and estimated HCRU in gMG patients.
This was a retrospective cohort analysis of clinical data from patients with gMG followed-up over eight consecutive years in a Spanish referral unit. Myasthenia Gravis Foundation of America (MGFA) clinical classification, MGFA post-interventional status (MGFA-PIS), Myasthenia Gravis Activities of Daily Living (MG-ADL) score, exacerbations, MG crises, therapies, AEs reported, specialist consultations and emergency room visits were studied biannually. An estimation of HRCU was made based on these data.
Some 220 patients newly diagnosed with gMG were included. Ninety percent were seropositive (84.5% anti-acetylcholine receptor [AChR], 5.9% anti-muscle-specific kinase [MuSK]). Baseline mean MG-ADL score was 5.04 points (SD 3.17), improving to 0.7 points (SD 1.40) after 8 years. Exacerbations were more frequent in years 1-2 (30.1%) but still occurred in years 7-8 (20.2%). Myasthenic crisis frequency remained 1% in years 7-8. Eighty-nine percent achieved MGFA-PIS minimal manifestations or better at 8 years. Fifty-one percent of patients reported at least one AE during the study period, leading to drug withdrawal in approximately 20% of cases. HCRU decreased between years 1-2 to years 7-8 with an estimated cost of MG from 8074.19 € per patient/year to 1679.46 €, respectively.
There is a group of MG patients that suffers from persistent symptoms and exacerbations (11%-20%) or MG crises, and drug AEs, which may increase disease burden and impact on the healthcare system.
全身性重症肌无力(gMG)的特征是肌肉无力波动。对于病情加重频率、与免疫抑制治疗相关的不良事件(AE)以及医疗资源利用(HCRU),目前了解尚少。我们的研究旨在描述gMG患者的长期临床结局、药物相关AE以及估计的HCRU。
这是一项对西班牙一家转诊单位连续八年随访的gMG患者临床数据进行的回顾性队列分析。每半年研究一次美国重症肌无力基金会(MGFA)临床分类、MGFA介入后状态(MGFA-PIS)、重症肌无力日常生活活动(MG-ADL)评分、病情加重情况、重症肌无力危象、治疗方法、报告的AE、专科会诊和急诊就诊情况。基于这些数据对HRCU进行了估计。
纳入了约220例新诊断的gMG患者。90%为血清学阳性(84.5%抗乙酰胆碱受体[AChR],5.9%抗肌肉特异性激酶[MuSK])。基线时MG-ADL平均评分为5.04分(标准差3.17),8年后改善至0.7分(标准差1.