TS-HDS autoantibody: clinical characterization and utility from real-world tertiary care center experience

Chompoopong, Pitcha; Rezk, Mohamed; Mirman, Igal; Berini, Sarah E.; Dyck, Peter J.; Mauermann, Michelle L.; Shouman, Kamal; Klein, Christopher J.; Mills, John R.; Dubey, Divyanshu

doi:10.1007/s00415-023-11798-9

Cited by 5 publications

(2 citation statements)

References 6 publications

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“… 3 , 45 However, their pathogenicity in SFN remains uncertain. 46 Moreover, we observed IgG cell membrane immunostaining. This contrasts with the reported anti-TS-HDS antibodies which are IgM.…”

Section: Discussionmentioning

confidence: 78%

Characterizing Acute-Onset Small Fiber Neuropathy

Gendre,

Lefaucheur,

Nordine

et al. 2024

Neurol Neuroimmunol Neuroinflamm

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Background and Objectives Immune-mediated small fiber neuropathy (SFN) is increasingly recognized. Acute-onset SFN (AOSFN) remains poorly described. Herein, we report a series of AOSFN cases in which immune origins are debatable. Methods We included consecutive patients with probable or definite AOSFN. Diagnosis of SFN was based on the NEURODIAB criteria. Acute onset was considered when the maximum intensity and extension of both symptoms and signs were reached within 28 days. We performed the following investigations: clinical examination, neurophysiologic assessment encompassing a nerve conduction study to rule out large fiber neuropathy, laser-evoked potentials (LEPs), warm detection thresholds (WDTs), electrochemical skin conductance (ESC), epidermal nerve fiber density (ENF), and patient serum reactivity against mouse sciatic nerve teased fibers, mouse dorsal root ganglion (DRG) sections, and cultured DRG. The serum reactivity of healthy subjects (n = 10) and diseased controls (n = 12) was also analyzed. Data on baseline characteristics, biological investigations, and disease course were collected. Results Twenty patients presenting AOSFN were identified (60% women; median age: 44.2 years [interquartile range: 35.7–56.2]). SFN was definite in 18 patients (90%) and probable in 2 patients. A precipitating event was present in 16 patients (80%). The median duration of the progression phase was 14 days [5–28]. Pain was present in 17 patients (85%). Twelve patients (60%) reported autonomic involvement. The clinical pattern was predominantly non–length-dependent (85%). Diagnosis was confirmed by abnormal LEPs (60%), ENF (55%), WDT (39%), or ESC (31%). CSF analysis was normal in 5 of 5 patients. Antifibroblast growth factor 3 antibodies were positive in 4 of 18 patients (22%) and anticontactin-associated protein-2 antibodies in one patient. In vitro studies showed IgG immunoreactivity against nerve tissue in 14 patients (70%), but not in healthy subjects or diseased controls. Patient serum antibodies bound to unmyelinated fibers, Schwann cells, juxtaparanodes, paranodes, or DRG. Patients' condition improved after a short course of oral corticosteroids (3/3). Thirteen patients (65%) showed partial or complete recovery. Others displayed relapses or a chronic course. Discussion AOSFN primarily presents as an acute, non–length-dependent, symmetric painful neuropathy with a variable disease course. An immune-mediated origin has been suggested based on in vitro immunohistochemical studies.

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Section: Discussionmentioning

confidence: 78%

Characterizing Acute-Onset Small Fiber Neuropathy

Gendre,

Lefaucheur,

Nordine

et al. 2024

Neurol Neuroimmunol Neuroinflamm

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“…Trisulfated heparin disaccharide IdoA2S-GlcNS6S IgM has been reported in association with painful, predominantly sensory, axonal polyneuropathy 44 . However, a 2023 study demonstrated that this antibody may have limited phenotypic or disease specificity 45 . Additional, prospective studies are necessary to further characterize the utility of this antibody in neurology practice.…”

Section: Peripheral Neuropathymentioning

confidence: 99%

Autoimmune Neuromuscular Disorders Associated With Neural Antibodies

Dubey

2024

CONTINUUM: Lifelong Learning in Neurology

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OBJECTIVE This article reviews autoimmune neuromuscular disorders and includes an overview of the diagnostic approach, especially the role of antibody testing in a variety of neuropathies and some other neuromuscular disorders. LATEST DEVELOPMENTS In the past few decades, multiple antibody biomarkers associated with immune-mediated neuromuscular disorders have been reported. These biomarkers are not only useful for better understanding of disease pathogenesis and allowing more timely diagnosis but may also aid in the selection of an optimal treatment strategy. ESSENTIAL POINTS Recognition of autoimmune neuromuscular conditions encountered in inpatient or outpatient neurologic practice is very important because many of these disorders are reversible with prompt diagnosis and early treatment. Antibodies are often helpful in making this diagnosis. However, the clinical phenotype and electrodiagnostic testing should be taken into account when ordering antibody tests or panels and interpreting the subsequent results. Similar to other laboratory investigations, understanding the potential utility and limitations of antibody testing in each clinical setting is critical for practicing neurologists.

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Etanercept/Pyridoxine

2024

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TS-HDS autoantibody: clinical characterization and utility from real-world tertiary care center experience

Cited by 5 publications

References 6 publications

Characterizing Acute-Onset Small Fiber Neuropathy

Characterizing Acute-Onset Small Fiber Neuropathy

Autoimmune Neuromuscular Disorders Associated With Neural Antibodies

Etanercept/Pyridoxine

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