@article{Silsby2024,

title = {Upper and lower limb tremor in inflammatory neuropathies},

author = {Matthew Silsby and Con Yiannikas and Alessandro F. Fois and Matthew C. Kiernan and Victor S.C. Fung and Steve Vucic},

doi = {10.1016/j.clinph.2023.12.005},

issn = {1388-2457},

year  = {2024},

date = {2024-02-00},

journal = {Clinical Neurophysiology},

volume = {158},

pages = {69--78},

publisher = {Elsevier BV},

keywords = {},

pubstate = {published},

tppubtype = {article}

}

@article{Silsby2023,

title = {Imbalance and lower limb tremor in chronic inflammatory demyelinating polyradiculoneuropathy},

author = {Matthew Silsby and Con Yiannikas and Alessandro F. Fois and Karl Ng and Matthew C. Kiernan and Victor S. C. Fung and Steve Vucic},

doi = {10.1111/jns.12574},

issn = {1529-8027},

year  = {2023},

date = {2023-09-00},

journal = {J Peripheral Nervous Sys},

volume = {28},

number = {3},

pages = {415--424},

publisher = {Wiley},

abstract = {AbstractBackground and AimsImbalance is a prominent symptom of chronic inflammatory demyelinating polyradiculoneuropathy (CIDP). Although upper limb tremor in CIDP is described, lower limb tremor has not been assessed. The aim of this study was to determine whether lower limb tremor was present in CIDP and assess potential relationships with imbalance.MethodsThis was a cross‐sectional observational study of prospectively recruited consecutive patients with typical CIDP (N = 25). Clinical phenotyping, lower limb nerve conduction and tremor studies, and posturography analyses were performed. The Berg Balance Scale (BBS) divided CIDP patients into those with “good” and “poor” balance.ResultsLower limb tremor was evident in 32% of CIDP patients and associated with poor balance (BBSTremor 35 [23–46], BBSNo Tremor 52 [44–55], p = .035). Tremor frequency was 10.2–12.5 Hz with legs outstretched and on standing, apart from four patients with a lower frequency tremor (3.8–4.6 Hz) while standing. Posturography analysis revealed a high‐frequency spectral peak in the vertical axis in 44% of CIDP patients (16.0 ± 0.4 Hz). This was more likely in those with “good” balance (40% vs. 4%, p = .013).InterpretationLower limb tremor is present in one third of CIDP patients and is associated with poor balance. A high‐frequency peak on posturography is associated with better balance in CIDP. Lower limb tremor and posturography assessments could serve as important biomarkers of balance in a clinical setting.},

keywords = {},

pubstate = {published},

tppubtype = {article}

}

@article{Silsby2023b,

title = {Chronic inflammatory demyelinating polyradiculoneuropathy‐associated tremor: Phenotype and pathogenesis},

author = {Matthew Silsby and Alessandro F. Fois and Con Yiannikas and Karl Ng and Matthew C. Kiernan and Victor S. C. Fung and Steve Vucic},

doi = {10.1111/ene.15693},

issn = {1468-1331},

year  = {2023},

date = {2023-04-00},

journal = {Euro J of Neurology},

volume = {30},

number = {4},

pages = {1059--1068},

publisher = {Wiley},

abstract = {AbstractBackground and purposeTremor in chronic inflammatory demyelinating polyradiculoneuropathy (CIDP) is underrecognized, and the pathophysiology remains incompletely understood. This study evaluated tremor in CIDP and tested the hypothesis, established in other demyelinating neuropathies, that tremor occurs due to mistimed peripheral inputs affecting central motor processing. Additionally, the tremor stability index (TSI) was calculated with the hypothesis that CIDP‐related tremor is more variable than other tremor disorders.MethodsConsecutive patients with typical CIDP were prospectively recruited from neuromuscular clinics. Alternative causes of neuropathy and tremor were excluded. Cross‐sectional clinical assessment and extensive tremor study recordings were undertaken. Pearson correlation coefficient was used to compare nerve conduction studies and tremor characteristics, and t‐test was used for comparisons between groups.ResultsTwenty‐four patients with CIDP were included. Upper limb postural and action tremor was present in 66% and was mild according to the Essential Tremor Rating Assessment Scale. Tremor did not significantly impact disability. Surface electromyography (EMG) found high‐frequency spectral peaks in deltoid (13.73 ± 0.66 Hz), biceps brachii (11.82 ± 0.91 Hz), and extensor carpi radialis (11.87 ± 0.91 Hz) muscles, with lower peaks in abductor pollicis brevis EMG (6.07 ± 0.45 Hz) and index finger accelerometry (6.53 ± 0.42 Hz). Tremor was unchanged by weight loading but correlated with ulnar nerve F‐wave latency and median nerve sensory amplitude. TSI (2.3 ± 0.1) was significantly higher than essential tremor.ConclusionsPostural tremor is a common feature in CIDP. Tremor was unaffected by weight loading, typical of centrally generated tremors, although there was a correlation with peripheral nerve abnormalities. The high beat‐to‐beat variability on TSI and gradation of peak frequencies further suggest a complex pathophysiology. These findings may assist clinicians with the diagnosis of neuropathic tremor.},

keywords = {},

pubstate = {published},

tppubtype = {article}

}

@article{Park2022,

title = {Prevalence of chronic inflammatory demyelinating polyneuropathy and multifocal motor neuropathy in two regions of Australia},

author = {Susanna B. Park and Tiffany Li and Matthew C. Kiernan and Nidhi Garg and Ian Wilson and Richard White and Michael Boggild and Andrew McNabb and Matthew Lee‐Archer and Bruce V. Taylor},

doi = {10.1002/mus.27698},

issn = {1097-4598},

year  = {2022},

date = {2022-11-00},

journal = {Muscle and Nerve},

volume = {66},

number = {5},

pages = {576--582},

publisher = {Wiley},

abstract = {AbstractIntroduction/AimsImmune‐mediated neuropathies such as chronic inflammatory demyelinating polyneuropathy (CIDP) and multifocal motor neuropathy (MMN) produce significant disability and often require maintenance treatment. There is a paucity of epidemiological data on these conditions in Australia.MethodsWe undertook a prevalence study of CIDP and MMN in North Queensland and Tasmania, coinciding with a national census. Diagnoses were classified against the diagnostic criteria of the European Federation of Neurological Societies/Peripheral Nerve Society. Case ascertainment was undertaken via multiple methods, including survey of local neurologists across public and private clinics, search of neurophysiology, neurology and hospital databases, search of admitted hospital database collections using ICD codes and through immunoglobulin therapy prescription lists.ResultsThe crude prevalence of CIDP was 5.00 per 100,000 (95% confidence interval [CI] 3.79–6.62) and the crude prevalence of MMN was 1.33 per 100,000 (95% CI 0.78–2.27). Prevalence was also investigated using National Blood Authority numbers of cases prescribed immunoglobulin therapy, indicating a CIDP prevalence of 5.72 per 100,000 (95% CI 4.41–7.43) and MMN prevalence of 1.94 per 100,000 (95% CI 1.24–3.03). There was no significant difference between these numbers and those calculated through access of patient records locally. There was no significant difference in prevalence between Tasmania and North Queensland for any category.DiscussionThis study updates the prevalence of CIDP and MMN in Australia. Understanding the distribution of CIDP and MMN patients and their need for treatment is essential for future resource planning and to enable monitoring and coordination of therapies such as immunoglobulin.},

keywords = {},

pubstate = {published},

tppubtype = {article}

}

@article{Kapoor2022b,

title = {Association of plasma neurofilament light chain with disease activity in chronic inflammatory demyelinating polyradiculoneuropathy},

author = {Mahima Kapoor and Aisling Carr and Martha Foiani and Amanda Heslegrave and Henrik Zetterberg and Andrea Malaspina and Laura Compton and Elspeth Hutton and Alexander Rossor and Mary M. Reilly and Michael P. Lunn},

doi = {10.1111/ene.15496},

issn = {1468-1331},

year  = {2022},

date = {2022-11-00},

journal = {Euro J of Neurology},

volume = {29},

number = {11},

pages = {3347--3357},

publisher = {Wiley},

abstract = {AbstractBackground and purposeThis study was undertaken to explore associations between plasma neurofilament light chain (pNfL) concentration (pg/ml) and disease activity in patients with chronic inflammatory demyelinating polyradiculoneuropathy (CIDP) and examine the usefulness of pNfL concentrations in determining disease remission.MethodsWe examined pNfL concentrations in treatment‐naïve CIDP patients (n = 10) before and after intravenous immunoglobulin (IVIg) induction treatment, in pNfL concentrations in patients on maintenance IVIg treatment who had stable (n = 15) versus unstable disease (n = 9), and in clinically stable IVIg‐treated patients (n = 10) in whom we suspended IVIg to determine disease activity and ongoing need for maintenance IVIg. pNfL concentrations in an age‐matched healthy control group were measured for comparison.ResultsAmong treatment‐naïve patients, pNfL concentration was higher in patients before IVIg treatment than healthy controls and subsequently reduced to be comparable to control group values after IVIg induction. Among CIDP patients on IVIg treatment, pNfL concentration was significantly higher in unstable patients than stable patients. A pNFL concentration > 16.6 pg/ml distinguished unstable treated CIDP from stable treated CIDP (sensitivity = 86.7%, specificity = 66.7%, area under receiver operating characteristic curve = 0.73). Among the treatment withdrawal group, there was a statistically significant correlation between pNfL concentration at time of IVIg withdrawal and the likelihood of relapse (r = 0.72, p < 0.05), suggesting an association of higher pNfL concentration with active disease.ConclusionspNfL concentrations may be a sensitive, clinically useful biomarker in assessing subclinical disease activity.},

keywords = {},

pubstate = {published},

tppubtype = {article}

}

@article{Kapoor2022,

title = {IVIg-exposure and thromboembolic event risk: findings from the UK Biobank},

author = {Mahima Kapoor and Ian Hunt and Jennifer Spillane and Laura Jayne Bonnett and Elspeth Jane Hutton and James McFadyen and John-Paul Westwood and Michael P Lunn and Aisling S Carr and Mary M Reilly},

doi = {10.1136/jnnp-2022-328881},

issn = {1468-330X},

year  = {2022},

date = {2022-08-00},

journal = {J Neurol Neurosurg Psychiatry},

volume = {93},

number = {8},

pages = {876--885},

publisher = {BMJ},

abstract = {BackgroundArterial and venous thromboembolic events (TEEs) have been associated with intravenous Ig use, but the risk has been poorly quantified. We aimed to calculate the risk of TEEs associated with exposure to intravenous Ig.MethodsWe included participants from UK Biobank recruited over 3 years, data extracted September 2020.The study endpoints were incidence of myocardial infarction, other acute ischaemic heart disease, stroke, pulmonary embolism and other venous embolism and thrombosis.Predictors included known TEE risk factors: age, sex, hypertension, smoking status, type 2 diabetes mellitus, hypercholesterolaemia, cancer and past history of TEE. Intravenous Ig and six other predictors were added in the sensitivity analysis.Information from participants was collected prospectively, while data from linked resources, including death, cancer, hospital admissions and primary care records were collected retrospectively and prospectively. Findings14 794 of 502 492 individuals had an incident TEE during the study period. The rate of incident events was threefold higher in those with prior history of TEE (8 .7%) than those without previous history of TEE (3.0%).In the prior TEE category, intravenous Ig exposure was independently associated with increased risk of incident TEE (OR=3.69 (95% CI 1.15 to 11.92), p=0.03) on multivariate analysis. The number needed to harm by exposure to intravenous Ig in those with a history of TEE was 5.8 (95% CI 2.3 to 88.3).Intravenous Ig exposure did not increase risk of TEE in those with no previous history of TEE.InterpretationIntravenous Ig is associated with increased risk of further TEE in individuals with prior history of an event with one further TEE for every six people exposed. In practice, this will influence how clinicians consent for and manage overall TEE risk on intravenous Ig exposure.},

keywords = {},

pubstate = {published},

tppubtype = {article}

}

@article{Kapoor2020,

title = {Dramatic clinical response to ultra-high dose IVIg in otherwise treatment resistant inflammatory neuropathies},

author = {Mahima Kapoor and Mary M. Reilly and Hadi Manji and Michael P. Lunn and Aisling S. and Carr},

doi = {10.1080/00207454.2020.1815733},

issn = {1543-5245},

year  = {2022},

date = {2022-04-03},

journal = {International Journal of Neuroscience},

volume = {132},

number = {4},

pages = {352--361},

publisher = {Informa UK Limited},

keywords = {},

pubstate = {published},

tppubtype = {article}

}

@article{Kapoor2021,

title = {Subcutaneous immunoglobulin dose titration to clinical response in inflammatory neuropathy},

author = {Mahima Kapoor and Ryan Keh and Laura Compton and Sarah Morrow and David Gosal and Hadi Manji and Mary M. Reilly and Michael P. Lunn and Tim M. Lavin and Aisling S. Carr},

doi = {10.1007/s00415-020-10318-3},

issn = {1432-1459},

year  = {2021},

date = {2021-04-00},

journal = {J Neurol},

volume = {268},

number = {4},

pages = {1485--1490},

publisher = {Springer Science and Business Media LLC},

keywords = {},

pubstate = {published},

tppubtype = {article}

}