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Romanian Journal of Ophthalmology 2023Granulomatosis with polyangiitis (GPA) is an autoimmune disorder characterized by necrotizing granulomatous inflammation of small and medium-sized vessels. This... (Review)
Review
Granulomatosis with polyangiitis (GPA) is an autoimmune disorder characterized by necrotizing granulomatous inflammation of small and medium-sized vessels. This systematic review aimed to highlight the most common ophthalmic manifestations and to uncover their associations with antineutrophil cytoplasmic antibody (ANCA) positivity and the presence of granulomas. A literature search of PubMed, Web of Science, and Scopus electronic databases was performed from journal inception to March 21, 2021, for case reports and a series of ophthalmic GPAs. Cytoplasmic-ANCA (c-ANCA), perinuclear-ANCA (p-ANCA), and granulomas were analyzed against many ophthalmic signs and symptoms. 306 patients with GPA were retrospectively studied. Granulomas were present in 47.7% of our sample, c-ANCA in 59.2%, and p-ANCA in 10.8%. Scleritis was significantly associated with higher odds for c-ANCA positivity. Eye discharge, episcleritis, proptosis, and central nervous system (CNS) involvement were each significantly associated with lower odds for c-ANCA positivity. Orbital mass was significantly associated with lower odds for p-ANCA positivity. CNS involvement was significantly associated with higher odds for p-ANCA positivity (OR:3.08, 95% CI:1.02, 9.36, p=0.047) and orbital mass was significantly associated with lower odds for p-ANCA positivity. We recommend that clinicians should consider ocular or orbital GPA in patients presenting with non-specific eye complaints, such as vision impairment, orbital mass, or proptosis, and obtain further assessments to determine the possible presence of granuloma, c-ANCA, or p-ANCA. GPA = Granulomatosis with Polyangiitis, ANCA = antineutrophil cytoplasmic antibody, c-ANCA = cytoplasmic-ANCA, p-ANCA = perinuclear-ANCA, CNS = central nervous system, AAVs = ANCA-associated vasculitides, SD = standard deviation, GU = genitourinary, ENT = ear nose and throat, OR = odds ratio, CI = confidence interval.
Topics: Humans; Granulomatosis with Polyangiitis; Antibodies, Antineutrophil Cytoplasmic; Retrospective Studies; Orbital Diseases; Granuloma; Exophthalmos
PubMed: 37876507
DOI: 10.22336/rjo.2023.38 -
Frontiers in Neurology 2023The literature for immune-mediated neurological disorders is evolving like no other field of neurological illnesses. Many new antibodies or disorders have been described...
BACKGROUND
The literature for immune-mediated neurological disorders is evolving like no other field of neurological illnesses. Many new antibodies or disorders have been described in the last decade. The cerebellum is a brain structure susceptible to these immune-mediated pathologies, and anti-metabotropic glutamate receptor 1 (mGluR1) antibody has a predilection to the cerebellar tissue. Anti-mGluR1 encephalitis is a rare autoimmune disease affecting the central and peripheral nervous systems, triggering an acute or subacute cerebellar syndrome with varying degrees of severity. Anti-mGluR1 encephalitis is a rare autoimmune disease affecting the central nervous system. We aimed to systematically review reported cases of anti-mGluR1 encephalitis and summarize their clinical presentation, management, outcomes, and case reports.
METHODS
A search of the PubMed and Google Scholar databases was conducted and included all cases of anti-mGluR1 encephalitis published in English before October 1, 2022. A comprehensive systematic review was conducted using "metabotropic glutamate receptor type 1," "mGluR1," autoantibodies," "autoantibodies," "autoimmunity," and "antibody" as keywords. The risk of bias assessment of the evidence was performed using appropriate tools. The qualitative variables were presented as frequency and percentage.
RESULTS
Including our case, 36 cases of anti-mGluR1 encephalitis (19 males, median age 52.5 years, 11.1% pediatric cases) have been reported. The most common clinical manifestations are ataxia, dysarthria, and nystagmus. Initial imaging was normal in 44.4% of patients; however, 75% of patients showed abnormality later in the disease course. The first-line therapy options include glucocorticoids, intravenous immunoglobulin, and plasma exchange. Rituximab is the most commonly used second-line treatment. Complete remission was achieved in only 22.2% of patients, and 61.8% were disabled by the end of their course.
CONCLUSION
Anti-mGluR1 encephalitis manifests as symptoms of cerebellar pathology. Although the natural history has not been completely elucidated, early diagnosis with prompt initiation of immunotherapy could be imperative. Any patient suspected to have autoimmune cerebellitis should be tested for the presence of anti-mGluR1 antibody in the serum and cerebrospinal fluid. Escalation to an aggressive therapy approach should be applied in cases that do not respond to first-line therapies, and extended follow-up durations are required in all cases.
PubMed: 37139064
DOI: 10.3389/fneur.2023.1142160 -
Medicine Dec 2023Hashimoto's thyroiditis (HT) is the prevailing form of autoimmune thyroiditis and the leading cause of hypothyroidism in iodine-sufficient regions worldwide. This study... (Meta-Analysis)
Meta-Analysis
BACKGROUND
Hashimoto's thyroiditis (HT) is the prevailing form of autoimmune thyroiditis and the leading cause of hypothyroidism in iodine-sufficient regions worldwide. This study aims to evaluate the efficacy of vitamin D supplementation on HT through a meta-analysis of randomized controlled trials (RCTs).
METHODS
The databases searched included PubMed, and others. We included RCTs that the treatment group received vitamin D, while the control group received either a placebo or no treatment. The studies measured the baseline and endpoint levels of 25-hydroxyvitamin D [25(OH)D], thyroid-stimulating hormone (TSH), free thyroxine (FT4), free triiodothyronine (FT3), anti-thyroid peroxidase antibody (TPO-Ab), and thyroglobulin antibody (TG-Ab). We performed a meta-analysis to calculate the standardized mean difference (SMD) and 95% confidence interval (CI).
RESULTS
A total of 12 studies involving 862 individuals were included. Vitamin D supplementation has a significant impact on reducing the titers of TPO-Ab (SMD = -1.084, 95% CI = -1.624 to -0.545) and TG-Ab (SMD = -0.996, 95% CI = -1.579 to -0.413) in patients with HT, and it also improves thyroid function by decreasing TSH level (SMD = -0.167, 95% CI = -0.302 to 0.031) and increasing FT3 (SMD = 0.549, 95% CI = 0.077-1.020) and FT4 (SMD = 0.734, 95% CI = 0.184-1.285) levels. Active vitamin D (calcitriol) significantly reduces the titer of TPO-Ab compared to naive forms of vitamin D (vitamin D2 or D3); treatment durations > 12 weeks result in a more effective reduction of TPO-Ab levels and a more significant increase in FT4 and FT3 levels in patients with HT (meta-regression P < .05).
CONCLUSION
Vitamin D supplementation may have beneficial effects on HT patients by modulating immune responses and improving thyroid function.
Topics: Humans; Autoantibodies; Dietary Supplements; Hashimoto Disease; Thyrotropin; Vitamin D
PubMed: 38206745
DOI: 10.1097/MD.0000000000036759 -
Epilepsia Open Dec 2023Several reports have described the autoimmune encephalitis' (AE) possible onset during pregnancy. In this systematic review, we summarize the available data on the... (Review)
Review
Several reports have described the autoimmune encephalitis' (AE) possible onset during pregnancy. In this systematic review, we summarize the available data on the diagnostic and therapeutic approach to AE during pregnancy, highlighting the associated maternal and fetal clinical outcomes. A systematic search of the literature was performed. The following databases were used: PubMed, Google Scholar, EMBASE, and CrossRef. The revision was registered on the PROSPERO platform (CRD42022336357). Forty-nine patients were included. AE onset was mainly observed during the first and the second trimester of pregnancy with psychiatric manifestations and seizures as main onset symptoms. CSF analysis showed AE-specific autoantibody positivity in 33 patients (anti-NMDA receptor as the most frequent). EEG generally showed normal findings. MRI revealed pathological findings in less than half of patients. Tumor screening was positive in 14 cases. First-line immunotherapy (single or combined) was generally employed while second line was administered in a minority of patients. Levetiracetam was the most used antiseizure medication. Cesarean section was performed in 18 women. Most of the women had an excellent early outcome after delivery but 22 showed persistent neurological deficits in long-term follow-up. Fetal outcome was positive in 33 cases, whereas 12 cases of fetal death were reported. A logistic regression showed that no variable significantly influenced the odds of good/bad maternal and fetal clinical outcome. Diagnosis and treatment of AE during pregnancy is challenging. The rate of miscarriage in women with AE seems to be higher than the general population. In addition, mothers may show long-term neurological deficits.
Topics: Humans; Pregnancy; Female; Cesarean Section; Encephalitis; Abortion, Spontaneous; Autoimmune Diseases of the Nervous System
PubMed: 37562964
DOI: 10.1002/epi4.12806 -
Frontiers in Immunology 2021The presence of anti-desmocollin (Dsc) antibodies is rarely described in autoimmune blistering diseases patients. Moreover, several clinical phenotypes of pemphigus may...
The presence of anti-desmocollin (Dsc) antibodies is rarely described in autoimmune blistering diseases patients. Moreover, several clinical phenotypes of pemphigus may be associated with these antibodies. In this review we analyze clinicopathological, immunologic and outcome features of anti-Dsc autoimmune blistering diseases patients, to improve their diagnosis and management. We conducted a systematic search of PubMed and Embase (1990-present) for studies reporting cases of autoimmune blistering diseases with anti-Dsc antibodies. We classified the selected patients as patients with exclusively anti-Dsc autoantibodies, and patients with anti-Dsc and other autoantibodies. Of 93 cases with anti-Dsc autoantibodies included, 38 (41%) had exclusively these antibodies. Only 18% of patients presented with the typical clinicopathological phenotype of pemphigus vulgaris or pemphigus foliaceous. Mucosal involvement was seen in approximately half of the patients. Up to 18% of cases were associated with neoplasms. Acantholysis was described in 54% of cases with histopathological information. Treatments and outcomes vary in the different clinical phenotypes. The presence of anti-Dsc antibodies must be suspected mainly in those patients with either atypical pemphigus, in special with clinical pustules, or in cases showing intraepithelial or dermal neutrophilic/eosinophilic infiltrate on histological examination and dual pattern by direct immunofluorescence examination.
Topics: Acantholysis; Animals; Autoantibodies; Autoimmunity; Desmocollins; Desmogleins; Eosinophils; Humans; Neutrophils; Pemphigus; Phenotype; Skin
PubMed: 34567003
DOI: 10.3389/fimmu.2021.740820 -
Journal of Clinical Immunology Oct 2023Anti-interferon gamma antibody (AIGA) is a rare cause of adult onset immunodeficiency, leading to severe disseminated opportunistic infections with varying outcomes. We...
PURPOSE
Anti-interferon gamma antibody (AIGA) is a rare cause of adult onset immunodeficiency, leading to severe disseminated opportunistic infections with varying outcomes. We aimed to summarize the disease characteristics and to explore factors associated with disease outcome.
METHODS
A systematic literature review of AIGA associated disease was conducted. Serum-positive cases with detailed clinical presentations, treatment protocols, and outcomes were included. The patients were categorized into controlled and uncontrolled groups based on their documented clinical outcome. Factors associated with disease outcome were analyzed with logistic regression models.
RESULTS
A total of 195 AIGA patients were retrospectively analyzed, with 119(61.0%) having controlled disease and 76 (39.0%) having uncontrolled disease. The median time to diagnosis and disease course were 12 months and 28 months, respectively. A total of 358 pathogens have been reported with nontubercular mycobacterium (NTM) and Talaromyces marneffei as the most common pathogens. The recurrence rate was as high as 56.0%. The effective rates of antibiotics alone, antibiotics with rituximab, and antibiotics with cyclophosphamide were 40.5%, 73.5%, and 75%, respectively. In the multivariate logistic analysis, skin involvement, NTM infection, and recurrent infections remained significantly associated with disease control, with ORs of 3.25 (95% CI 1.187 ~ 8.909, P value = 0.022), 4.74 (95% CI 1.300 ~ 17.30, P value = 0.018), and 0.22 (95% CI 0.086 ~ 0.551, P value = 0.001), respectively. The patients with disease control had significant AIGA titer reduction.
CONCLUSIONS
AIGA could cause severe opportunistic infections with unsatisfactory control, particularly in patients with recurrent infections. Efforts should be made to closely monitor the disease and regulate the immune system.
Topics: Humans; Adult; Mycobacterium Infections, Nontuberculous; Retrospective Studies; Reinfection; Autoantibodies; Interferon-gamma; Immunologic Deficiency Syndromes; Opportunistic Infections; Anti-Bacterial Agents
PubMed: 37365453
DOI: 10.1007/s10875-023-01537-0 -
Nutrients Oct 2023The incidence of type 1 diabetes mellitus in children is considerably increasing in western countries. Thus, identification of the environmental determinants involved... (Review)
Review
AIMS AND HYPOTHESIS
The incidence of type 1 diabetes mellitus in children is considerably increasing in western countries. Thus, identification of the environmental determinants involved could ultimately lead to disease prevention. Here, we aimed to systematically review (PROSPERO ID: CRD42022362522) the current evidence of the association between maternal dietary factors during gestation and the risk of developing type 1 diabetes and/or islet autoimmunity (IA) in murine and human offspring.
METHODS
In accordance with PRISMA guidelines, the present systematic review searched PubMed and Scopus ( = 343) for different combinations of MeSH terms, such as type 1 diabetes, diet, islet autoimmunity, prenatal, nutrient, gluten, gliadin, vitamin, milk, and fibers.
RESULTS
We found that the most investigated dietary factors in the present literature were gluten, dietary advanced glycosylated end products (dAGEs), vitamin D, fatty acids, and iron. The results concerning prenatal exposure to a gluten-free environment showed a consistently protective effect on the development of IA. Prenatal exposures to vitamin D and certain fatty acids appeared to protect against the development of IA, whereas in utero iron and fat exposures correlated with increased risks of IA.
CONCLUSION
We conclude that a definite association is not established for most factors investigated as the literature represents a heterogeneous pool of data, although fetal exposures to some maternal dietary components, such as gluten, show consistent associations with increased risks of IA. We suggest that human prospective dietary intervention studies in both cohort and clinical settings are crucial to better evaluate critical and protective prenatal exposures from the maternal diet during pregnancy.
Topics: Child; Pregnancy; Female; Humans; Animals; Mice; Diabetes Mellitus, Type 1; Autoimmunity; Islets of Langerhans; Vitamin D; Vitamins; Fatty Acids; Glutens; Iron; Autoantibodies; Risk Factors
PubMed: 37892409
DOI: 10.3390/nu15204333 -
Frontiers in Neuroscience 2021To review the available evidence on sensitivity and specificity of anti-NF155 antibody detection in diagnosing a specific subset of patients with chronic inflammatory...
To review the available evidence on sensitivity and specificity of anti-NF155 antibody detection in diagnosing a specific subset of patients with chronic inflammatory demyelinating polyradiculoneuropathy (CIDP) and to calculate the frequencies of different autoantibodies to paranodal proteins. Diagnosis of CIDP relies on clinical and neurophysiologic criteria and lacks useful diagnostic biomarkers. A subset of CIDP patients exhibit atypical clinical phenotypes and impaired response to conventional treatments. These patients were reported as having autoantibodies targeting paranodal protein neurofascin isoform 155 (NF155), contactin-1 (CNTN1), and contactin-associated protein-1 (CASPR1). Here, we conducted a meta-analysis to summarize evidence on the diagnostic and prognostic value of these autoantibodies, especially for anti-NF155 antibody. We searched the following electronic bibliographic databases: PubMed, EMBASE, Cochrane Central Register of Controlled Trials (CENTRAL), and Web of Science. Eligible studies provided information to calculate the frequencies of anti-NF155 antibody and anti-CNTN1 antibody, the sensitivity and specificity of anti-NF155 antibody, and the incidence of improvement and deterioration among anti-NF155 antibody seropositive CIDP patients. Heterogeneity was assessed using Q and statistics. The pooled frequency of anti-NF155 autoantibody across 14 studies was 7% [95% confidence interval (CI): 0.05-0.10] with high heterogeneity; the overall pooled sensitivity and specificity of anti-NF155 antibody for the diagnosis of a specific subgroup of CIDP patients were 0.45 (95% CI: 0.29-0.63) and 0.93 (95% CI: 0.86-0.97), respectively. For diagnosing of a specific subset of CIDP characterized by poor response to intravenous immunoglobulin (IVIg), we found a moderate sensitivity and a high specificity. The anti-NF155 antibody test should be used as a confirmatory test rather than a screening test. PROSPERO, identifier: CRD42020203385 and CRD42020190789.
PubMed: 34108854
DOI: 10.3389/fnins.2021.637336 -
Frontiers in Endocrinology 2023Numerous studies have found an association between vitamin deficiency and thyroid disorders (TD). The presence of anti-parietal cell antibodies is indicative of reduced... (Meta-Analysis)
Meta-Analysis
BACKGROUND AND AIMS
Numerous studies have found an association between vitamin deficiency and thyroid disorders (TD). The presence of anti-parietal cell antibodies is indicative of reduced ability to absorb vitamin B12. Thus, this study reviewed the existing studies with the objective of assessing differences in the serum levels of vitamin B12 among patients with and without TD, the frequency of vitamin B12 deficiency in patients with TD, and the presence of anti-parietal cell antibodies in patients with TD.
METHODS
A meta-analysis of random-effects model was conducted to calculate pooled frequencies, mean differences (MD), and their respective 95% confidence intervals (CI). We identified 64 studies that met our inclusion criteria (n = 28597).
RESULTS
We found that patients with hypothyroidism had lower vitamin B12 levels than healthy participants (MD: -60.67 pg/mL; 95% CI: -107.31 to -14.03 pg/mL; p = 0.01). No significant differences in vitamin B12 levels were observed between healthy participants and patients with hyperthyroidism (p = 0.78), autoimmune thyroid disease (AITD) (p = 0.22), or subclinical hypothyroidism (SH) (p = 0.79). The frequencies of vitamin B12 deficiency among patients with hypothyroidism, hyperthyroidism, SH, and AITD were 27%, 6%, 27%, and 18%, respectively.
CONCLUSIONS
Patients with hypothyroidism had lower levels of vitamin B12 than healthy participants. No significant differences were observed between vitamin B12 levels and hyperthyroidism, AITD, or SH.
SYSTEMATIC REVIEW REGISTRATION
https://www.crd.york.ac.uk/prospero/display_record.php?RecordID=324422, identifier (CRD42022324422).
Topics: Humans; Hypothyroidism; Hyperthyroidism; Vitamin B 12; Vitamin B 12 Deficiency; Hashimoto Disease; Autoantibodies
PubMed: 36909313
DOI: 10.3389/fendo.2023.1070592 -
Clinical and Experimental Rheumatology Jun 2022Studies report that autoimmune thyroid disease and elevated levels of thyroid autoantibodies are associated with fibromyalgia and widespread chronic pain. The aim of... (Meta-Analysis)
Meta-Analysis Review
OBJECTIVES
Studies report that autoimmune thyroid disease and elevated levels of thyroid autoantibodies are associated with fibromyalgia and widespread chronic pain. The aim of this meta-analysis was to investigate the relationship between fibromyalgia and thyroid autoimmunity. Clinical symptoms and depression associated with fibromyalgia were also investigated in relation to the presence of thyroid autoantibodies.
METHODS
A literature search was conducted on PubMed and Embase for studies published between January, 1980 and February, 2020 on thyroid autoimmunity in fibromyalgia patients. Two reviewers independently screened and assessed the quality of the articles. Meta-analysis was performed to analyse the difference in frequency of thyroid autoantibody positivity between fibromyalgia patients and healthy controls. Clinical symptoms and depression were also analysed according to the presence of thyroid autoantibodies.
RESULTS
Data from 10 original studies were included in the systematic review, and 5 case-control studies that satisfied the selection criteria were subjected to meta-analysis. Thyroid autoantibody positivity was more common in fibromyalgia patients compared to healthy controls (thyroid peroxidase antibody: OR 3.41, 95% CI 1.97-5.90; thyroglobulin antibody: OR 2.23, 95% CI 1.23-4.01). The frequency of postmenopausal status was significantly higher in fibromyalgia patients with thyroid autoantibodies (OR 1.95, 95% CI 1.23-3.08). However, the severity of disease (pain and fatigue level, fibromyalgia impact questionnaire score, and disease duration) and prevalence of depression did not show a statistically significant difference according to thyroid autoantibody positivity.
CONCLUSIONS
Thyroid autoimmunity should be considered in fibromyalgia patients. The percentage of women in menopause was higher in thyroid autoantibody positive fibromyalgia patients.
Topics: Autoantibodies; Autoimmunity; Causality; Female; Fibromyalgia; Humans; Thyroid Gland
PubMed: 34369360
DOI: 10.55563/clinexprheumatol/y3gfva