Ataxia, peripheral neuropathy, and anti-gliadin antibody. Guilt by association?

Lock, R J; Tengah, Dayangku Siti Nur Ashikin Pengiran; Unsworth, D J; Ward, Jennifer; Wills, Adrian

doi:10.1136/jnnp.2004.058487

Cited by 22 publications

(10 citation statements)

References 24 publications

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“…The controversy is caused by the finding of such antibodies in patients with ataxia due to a proven genetic cause [23,24],which has led to the hypothesis that these antibodies are epiphenomena, secondary to a primary degenerative process. We have reviewed 10 studies that addressed the question of prevalence and specificity of these antibodies in patients with ataxia [22][23][24][25][26][27][28][29][30][31]. Two studies were Class II [23,25] and the other eight Class III or IV [22,24,[26][27][28][29][30].…”

Section: Negative Family Historymentioning

confidence: 99%

EFNS/ENS Consensus on the diagnosis and management of chronic ataxias in adulthood

Warrenburg

Gaalen

Boesch

et al. 2014

Euro J of Neurology

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Background and objectives The ataxias are a challenging group of neurological diseases due the aetiological heterogeneity and the complexity of the genetic subtypes. This guideline focuses on the heredodegenerative ataxias. The aim is to provide a peer‐reviewed evidence‐based guideline for clinical neurologists and other specialist physicians responsible for the care of patients with ataxia. Methods This guideline is based on systematic evaluations of the relevant literature and on three consensus meetings of the task force. Diagnosis If acquired causes are ruled out, and if the disease course is rather slowly progressive, a (heredo)degenerative disease is likely. A positive family history gives much guidance. In the case of a dominant family history, first line genetic screening is recommended for spinocerebellar ataxia (SCA) 1, 2, 3, 6, 7 and 17 (level B), and in Asian patients also for dentatorubral‐pallidoluysian atrophy (DRPLA). In the case of recessive disease, a stepwise diagnostic work‐up is recommended, including both biochemical markers and targeted genetic testing, particularly aimed at Friedreich's ataxia, ataxia telangiectasia, ataxia due to vitamin E deficiency, polymerase gamma gene (POLG gene, various mutations), autosomal recessive spastic ataxia of Charlevoix−Saguenay (ARSACS) and ataxia with oculomotor apraxia (AOA) types 1 and 2. If family history is negative, we still advise to screen for the more common dominant and recessive ataxias. In addition, if onset is below 45 years we recommend the full work‐up for recessive ataxias; if onset is above 45 years we recommend to screen for fragile X mental retardation 1 FMR1 premutations (good practice points). In sporadic cases with an onset after 30 years, a diagnosis of multiple system atrophy should be considered (good practice point). In particular the genetic work‐up will change over the upcoming years due to the diagnostic utility of new techniques such as gene panel diagnostics based on next generation sequencing for routine work‐up, or even whole exome and genome sequencing for selected cases. Treatment Some of the rare recessive ataxias are treatable, but for most of the heredodegenerative ataxias treatment is purely symptomatic. Idebenone is not effective in Friedreich's ataxia (level A). Riluzole (level B) and amantadine (level C) might provide symptomatic relief, irrespective of exact etiology. Also, varenicline for SCA3 patients (level B) can be considered. There is level Class II evidence to recommend physiotherapy, and Class III data to support occupational therapy.

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Section: Negative Family Historymentioning

confidence: 99%

EFNS/ENS Consensus on the diagnosis and management of chronic ataxias in adulthood

Warrenburg

Gaalen

Boesch

et al. 2014

Euro J of Neurology

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“…14 Consistent with previous reports, in patients without CD, first-generation gliadin antibodies lacked specificity for neurologic disorders purported to be related to gluten exposure. 10,24,25 Using tissue immunofluorescence, we did not detect any novel IgA or IgG neural antibodies that might serve as biomarkers of gluten-triggered neurologic disorders. 20,26 For specificity, our contemporary tissue immunofluorescence protocol includes gastric and renal tissue controls (side by side with brain tissue) and preabsorption of serums to remove non-organ-specific antibodies.…”

Section: Statisticsmentioning

confidence: 78%

The neurologic significance of celiac disease biomarkers

et al. 2014

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“…After resolving initial disagreements, 11/26 (42.3%) studies9 11–14 17–22 were considered eligible (figure 1), representing 6.9% of the total search (11/159). The reasons for excluding 15/26 articles8 23–36 in the order of high to low percentage are as follows: studies only on IDCA cases without HA cases or controls (n=6, 40%),8 23 26 27 30 32 studies on cases of ataxia with or without coeliac disease instead of ataxia cases versus controls (n=3, 20%),31 33 34 studies that defined IDCA distinctively from the general consensus (ie, non-genetic ataxias caused by acquired conditions and sporadic neurodegenerative disorders)15 (n=2, 13.3%),15 34 a study that only tested for anti-deamidated gliadin peptide (DGP) antibody without testing for AGA (n=1, 6.7%),25 a study that only tested for anti TTG2 antibody without testing for AGA (n=1, 6.7%),29 a study that did not include IDCA cases (n=1, 6.7%)24 and a duplicated study with same patient groups but under different titles (n=1, 6.7%) 36. These 11 studies included in the meta-analysis were all rated as 7 stars or above on Newcastle-Ottawa Quality Assessment Scale Adapted for Cross-Sectional Studies and thus included for meta-analysis.…”

Section: Resultsmentioning

confidence: 99%

Serum antigliadin antibodies in cerebellar ataxias: a systematic review and meta-analysis

Lin

Wang

Tse

et al. 2018

J Neurol Neurosurg Psychiatry

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Background Gluten sensitivity refers to prominent immunological responses to gluten, usually in conjunction with elevated levels of serum antigliadin antibody (AGA). The association between AGA and cerebellar ataxias has been inconsistently reported. Methods We performed a systematic literature search and a meta-analysis to study the weighted pooled OR of idiopathic cerebellar ataxia (IDCA) cases to controls or to hereditary ataxia (HA) for AGA seropositivity using fixed effect model. Results Eleven studies were included, with a total of 847 IDCA cases, 1654 controls and 445 HA cases. IDCA cases had fourfold higher odds than controls (OR 4.28, 95% CI 3.10 to 5.90) and twofold higher odds than HA cases (OR 2.23, 95% CI 1.45 to 3.44) of having AGA seropositivity. Sensitivity analysis excluding the most weighted study, which accounted for 69% of the total weight, still showed similar associations (IDCA vs controls, OR 3.18, 95% CI 1.79 to 5.67 and IDCA vs HA, OR 1.72, 95% CI 1.03 to 2.86, respectively). The subgroup analysis showed that, when compared with controls, IDCA cases of both East Asian and Western countries had approximately threefold to fourfold higher odds to have AGA seropositivity (OR 3.41, 95% CI 1.67 to 6.97 and OR 4.53, 95% CI 3.16 to 6.49, respectively), suggesting the lack of ethnic heterogeneity. The odds of AGA seropositivity for HA cases was not significantly higher than controls (OR 1.41, 95% CI 0.82 to 2.44). Conclusion Our study indicates the association between AGA and IDCA, across different geographic regions.

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Ataxia, peripheral neuropathy, and anti-gliadin antibody. Guilt by association?

Cited by 22 publications

References 24 publications

EFNS/ENS Consensus on the diagnosis and management of chronic ataxias in adulthood

EFNS/ENS Consensus on the diagnosis and management of chronic ataxias in adulthood

The neurologic significance of celiac disease biomarkers

Serum antigliadin antibodies in cerebellar ataxias: a systematic review and meta-analysis

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