Chrysanthi Bountziouka, Despina Fotiou, Foteini Theodorakakou, Asimina Papanikolaou, Maria Gavriatopoulou, Evangelos Terpos, Meletios A Dimopoulos, Efstathios Kastritis, Panagiotis Kokotis
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引用次数: 0
摘要
免疫球蛋白轻链淀粉样蛋白(AL)淀粉样变性常累及周围神经系统(PNS)。PNS受累,特别是小纤维神经病变(SFN)在临床上常常被低估,需要标准化的方法进行全面评估。我们通过临床检查、神经传导检查(NCSs)、定量感觉测试(QST)检查和远端腿皮肤活检,对81例新诊断AL淀粉样变性患者SFN的患病率和临床意义进行了前瞻性评估。89%的患者有神经病变,65%的患者有SFN。合并大、小纤维神经病占48.1%,纯大纤维神经病占20%,纯SNF占10%。年龄较大是SFN的重要危险因素(OR 1.06, 95% CI 1.01-1.12, p = 0.014);SFN患者也更有可能累及软组织(OR 7.1, 95% CI 1.5-33.4, p = 0.013)。中位随访37.5个月后,SFN与较差的总生存期(OS)相关,并成为早期死亡的独立预后因素(p = 0.023)。我们的研究证明了SFN作为生存不利因素的高患病率和临床意义,并提示在AL淀粉样变患者诊断时需要进行多参数神经学评估。
A prospective study of small fiber neuropathy in AL amyloidosis.
The peripheral nervous system (PNS) is commonly affected in immunoglobulin light chain amyloid protein (AL) amyloidosis. PNS involvement and particularly small fiber neuropathy (SFN) is often clinically underestimated, requiring a standardized approach for comprehensive assessment. We prospectively evaluated the prevalence and clinical significance of SFN in 81 patients with newly diagnosed AL amyloidosis using clinical examination, nerve conduction studies (NCSs), quantitative sensory testing (QST) examination and distal-leg skin biopsy. Neuropathy was detected in 89% of patients and SFN in 65%. Combined small and large fiber neuropathy was seen in 48.1%, pure large fiber neuropathy in 20% and pure SNF in 10%. Older age was a significant risk factor for SFN (OR 1.06, 95% CI 1.01-1.12, p = .014); patients with SFN were also more likely to have soft tissue involvement (OR 7.1, 95% CI 1.5-33.4, p = .013). After a median follow-up of 37.5 months, SFN was associated with poorer overall survival (OS) and it emerged as an independent prognostic factor for early mortality (<12 months) in multivariate analysis (HR 4.3 95% CI 1.23-15.04, p = .023). Our study demonstrates the high prevalence and clinical significance of SFN as an adverse factor for survival and indicates the need for multiparametric neurological evaluation in patients with AL amyloidosis at diagnosis.
期刊介绍:
Amyloid: the Journal of Protein Folding Disorders is dedicated to the study of all aspects of the protein groups and associated disorders that are classified as the amyloidoses as well as other disorders associated with abnormal protein folding. The journals major focus points are:
etiology,
pathogenesis,
histopathology,
chemical structure,
nature of fibrillogenesis;
whilst also publishing papers on the basic and chemical genetic aspects of many of these disorders.
Amyloid is recognised as one of the leading publications on amyloid protein classifications and the associated disorders, as well as clinical studies on all aspects of amyloid related neurodegenerative diseases and major clinical studies on inherited amyloidosis, especially those related to transthyretin. The Journal also publishes book reviews, meeting reports, editorials, thesis abstracts, review articles and symposia in the various areas listed above.