Autoimmune encephalitis associated with vitiligo?

Ren, Haitao; Liu, Huiqin; Qu, Tao; Xunzhe, Yang; Shao, Xiaoqiu; Li, Wei; Zhang, Jiewen; Guan, Hongzhi

doi:10.1016/j.jneuroim.2017.05.019

Cited by 18 publications

(11 citation statements)

References 12 publications

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“…Several cases with encephalitis preceding SV have been reported (62,63). It should, however, be noted that this is also the case for NSV (80) found. This offers however more a clue toward somatic mosaicism given their common origin from the neural crest than an argument for a neurogenic cause of SV (64).…”

Section: Neurogenic Theorymentioning

confidence: 61%

Autoimmunity in Segmental Vitiligo

et al. 2020

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The autoimmune basis of segmental vitiligo (SV) has only recently been recognized. Systemic autoimmune diseases are less frequently associated compared to non-segmental vitiligo (NSV), but localized skin disordersin particular linear morpheahave been repeatedly observed in patients with SV. The inflammatory response is documented on a clinical level with cases displaying erythematous borders or a hypochromic stage, on a histopathological level with predominantly CD8 lymphocytes migrating toward the basal layer and by flow cytometry demonstrating the antimelanocyte specificity of these cytotoxic T cells. The increased risk for halo naevi and NSV in these patients further underline the immunemediated mechanisms of SV. Nonetheless, the localized and unique distribution pattern points to somatic mosaicism. This places SV in a category of similar diseases such as lichen striatus, blaschkitis, linear lupus erythematosus, and linear scleroderma where an immune reaction against genetically mutated skin cells is believed to be the underlying cause. All these disorders are characterized by a young age of onset, a temporary disease activity with spontaneous resolution, limited response to treatment, and often long-term sequelae. Although challenging, genetic research proving this genetic mosaicism could offer crucial insights into the pathogenesis of both segmental and non-segmental vitiligo.

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Section: Neurogenic Theorymentioning

confidence: 61%

Autoimmunity in Segmental Vitiligo

et al. 2020

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“…For all antibody-defined AIE subgroups combined, we could identify 116 publications that matched our search criteria and contained relevant information for the intended analyses. Ten were identified for AIE with AMPAR antibodies (7–16), 15 for CASPR2 (16–30), eight for DPPX (31–38), four for GABA A R (15, 39–41) with patients reported by the Pettingil et al being excluded as these were not tested for GABA A R antibodies in CSF and represented a different phenotype (42), 15 for GABA B R antibodies (12, 15, 16, 43–54), 15 for GAD antibodies (19, 22, 55–67), five for GlyR antibodies (68–72), 14 for IgLON5 antibodies (73–86), 25 for LGI1 antibodies (5, 6, 15, 16, 19–22, 30, 52, 77, 87–100), and finally 27 for NMDAR antibodies (4, 15, 16, 19, 22, 52, 62, 88, 89, 101–118). The history of the data search and the results are depicted in Figure 1.…”

Section: Resultsmentioning

confidence: 99%

Cerebrospinal Fluid Findings in Patients With Autoimmune Encephalitis—A Systematic Analysis

2019

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Autoimmune encephalitides (AIE) comprise a group of inflammatory diseases of the central nervous system (CNS), which can be further characterized by the presence of different antineuronal antibodies. Recently, a clinical approach for diagnostic criteria for the suspected diagnosis of AIE as well as definitive AIE were proposed. These are intended to guide physicians when to order the antineuronal antibody testing and/or facilitate early diagnosis even prior to the availability of the specific disease-confirming test results to facilitate prompt treatment. These diagnostic criteria also include the results of basic cerebrospinal fluid (CSF) analysis. However, the different antibody-defined AIE subtypes might be highly distinct with regard to their immune pathophysiology, e.g., the pre-dominance of specific IgG subclasses, IgG1, or IgG4, or frequency of paraneoplastic compared to idiopathic origin. Thus, it is conceivable that the results of basic CSF analysis might also be very different. However, this has not been explored systematically. Here, we systematically reviewed the literature about the 10 most important AIE subtypes, AIE with antibodies against NMDA, AMPA, glycine, GABA A , and GABA B receptors as well as DPPX, CASPR2, LGI1, IgLON5, or glutamate decarboxylase (GAD), with respect to the reported basic CSF findings comprising CSF leukocyte count, total protein, and the presence of oligoclonal bands (OCB) restricted to the CSF as a sensitive measure for intrathecal IgG synthesis. Our results indicate that these basic CSF findings are profoundly different among the 10 different AIE subtypes. Whereas, AIEs with antibodies against NMDA, GABA B , and AMPA receptors as well as DPPX show rather frequent inflammatory CSF changes, in AIEs with either CASPR2, LGI1, GABA A , or glycine receptor antibodies CSF findings were mostly normal. Two subtypes, AIEs defined by either GAD, or IgLON5 antibodies, did not fit into this general pattern. In AIE with GAD antibodies, positive OCBs in the absence of other changes were typical, while the CSF in IgLON5 antibody-positive AIE was characterized by elevated protein.

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“…The literature search initially retrieved 113 studies published between 2014 and 2019, from which 35 were excluded as duplicates, 38 after reading the title and abstract and 22 after reading the whole article (Table S1). Therefore, 18 articles met the selection criteria (see Figure 1): three case series 1,7,8 and 15 case reports, [11][12][13][14][15][16][17][18][19][20][21][22][23][24][25][26][27][28] all with a SIGN level of evidence of 3, which described a total of 46 patients with IgLON5 treated with IT (Table 1).…”

Section: Searchmentioning

confidence: 99%

Response to immunotherapy in anti‐IgLON5 disease: A systematic review

et al. 2020

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The aim of this study was to evaluate the frequency of response to immunotherapy in patients with anti‐IgLON5 disease through a systematic review of the literature. MEDLINE and Embase databases were searched for studies that included patients with anti‐IgLON5 disease who received immunotherapy (IT). Review inclusion criteria were met by 18 studies. The main study variable was response to IT, defined as the frequency of patients with an improvement greater than mild in at least one of the main symptoms defined by the clinical phenotype. Data were also gathered on the rate of response to last follow‐up, the line(s) of IT received, the administration of monotherapy or combination therapy, and clinical and analytical characteristics. Selected studies included a total of 46 patients. A response to IT was observed in 20 (43.4%) and the presence of response to last follow‐up in 15 (32.6%). Response was achieved more frequently with combination therapy vs monotherapy (14/21 [66.6%] vs 7/22 [31.8%]) and second‐line therapy vs first‐line therapy (7/13 [53.8%] vs 15/46 [32.6%]). The response rate by drug was 34.2% (12/35) for steroids, 42.8% (9/21) for IVIg, 46% (7/15) for PLEX, 100% (5/5) for AZA and 75% (3/4) for MMF. Factors associated with a response to IT included the cognitive impairment and non‐classical phenotypes, presence of HLA‐DQB1*05:01 without HLA‐DRB1*10:01 and cerebral spinal fluid inflammation. Patients with anti‐IgLON5 disease respond to IT, and this response is associated with certain clinical and analytical characteristics of the patients. Also rate of response seems higher with second‐line and combination treatment. However, the quality of available studies is inadequate to allow definitive conclusions to be drawn.

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Autoimmune encephalitis associated with vitiligo?

Cited by 18 publications

References 12 publications

Autoimmunity in Segmental Vitiligo

Autoimmunity in Segmental Vitiligo

Cerebrospinal Fluid Findings in Patients With Autoimmune Encephalitis—A Systematic Analysis

Response to immunotherapy in anti‐IgLON5 disease: A systematic review

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