-
Annals of Clinical and Translational... Jun 2024Fabry disease is caused by enzymatic defects in alpha-galactosidase A that leads to the accumulation of glycosphingolipids throughout the body, resulting in a...
OBJECTIVE
Fabry disease is caused by enzymatic defects in alpha-galactosidase A that leads to the accumulation of glycosphingolipids throughout the body, resulting in a multisystemic disorder. The most common neurological manifestations are neuropathic pain, autonomic nervous system dysfunction and strokes, but some rarer neurological manifestations exist. Among these, aseptic meningitis is a possible complication. Our objectives were to measure the prevalence of this complication in a cohort of patients with Fabry disease, and to describe its clinical features.
METHODS
We conducted a retrospective review of Fabry disease patients followed at our tertiary referral center between 1995 and September 2023 with at least one episode of meningitis, and performed a systematic review to identify similar published cases.
RESULTS
Four patients out of 107 (3.7%) had at least one episode of aseptic meningitis. Our systematic review identified 25 other observations. The median age of these 29 patients was 29.0 years, the median cerebrospinal fluid leukocyte count was 24 cells/mm3 with a predominance of lymphocytes in 64.7% of cases. In 82.8% of the patients, the diagnosis of Fabry disease was unknown before the meningitis. Large artery stenosis was present in 17.2% of patients and 57.1% of patients had a recent stroke concomitant with the meningitis. Several differential diagnoses were evoked, such as multiple sclerosis or central nervous system vasculitis.
INTERPRETATION
Our study suggests that Fabry disease should be considered as a cause of aseptic meningitis. The pathophysiological mechanisms underlying meningeal inflammation remain largely unknown but may reflect the dysregulation of pro-inflammatory signaling pathways.
Topics: Humans; Fabry Disease; Meningitis, Aseptic; Adult; Male; Female; Retrospective Studies; Middle Aged; Young Adult; Adolescent; Aged; Child
PubMed: 38717582
DOI: 10.1002/acn3.52043 -
Clinical Reviews in Allergy & Immunology Apr 2024An acute aseptic meningitis has been occasionally observed on intravenous polyclonal human immunoglobulin therapy. Since case reports cannot be employed to draw... (Meta-Analysis)
Meta-Analysis Review
An acute aseptic meningitis has been occasionally observed on intravenous polyclonal human immunoglobulin therapy. Since case reports cannot be employed to draw inferences about the relationships between immunoglobulin therapy and meningitis, we conducted a systematic review and meta-analysis of the literature. Eligible were cases, case series, and pharmacovigilance studies. We found 71 individually documented cases (36 individuals ≤ 18 years of age) of meningitis. Ninety percent of cases presented ≤ 3 days after initiating immunoglobulin therapy and recovered within ≤ 7 days (with a shorter disease duration in children: ≤ 3 days in 29 (94%) cases). In 22 (31%) instances, the authors noted a link between the onset of meningitis and a rapid intravenous infusion of immunoglobulins. Cerebrospinal fluid analysis revealed a predominantly neutrophilic (N = 46, 66%) pleocytosis. Recurrences after re-exposure were observed in eight (N = 11%) patients. Eight case series addressed the prevalence of meningitis in 4089 patients treated with immunoglobulins. A pooled prevalence of 0.6% was noted. Finally, pharmacovigilance data revealed that meningitis temporally associated with intravenous immunoglobulin therapy occurred with at least five different products. In conclusion, intravenous immunoglobulin may cause an acute aseptic meningitis. The clinical features remit rapidly after discontinuing the medication.
Topics: Humans; Meningitis, Aseptic; Immunoglobulins, Intravenous; Acute Disease; Child; Adolescent; Pharmacovigilance; Child, Preschool; Immunization, Passive
PubMed: 38739354
DOI: 10.1007/s12016-024-08989-1 -
Turkish Archives of Pediatrics Nov 2023Given the relatively high frequency of central nervous system infections and considerable mor- tality and morbidity reported to be caused by herpes simplex viruses among...
Given the relatively high frequency of central nervous system infections and considerable mor- tality and morbidity reported to be caused by herpes simplex viruses among the other viral agents, having a clear knowledge about their epidemiological profile seems necessary. This systematic review and meta-analysis aimed to determine the relative frequency and preva- lence of herpes simplex encephalitis and meningitis in patients tested for viral etiologies. A comprehensive systematic review was performed in PubMed, Scopus, and Web of Science databases, searching for studies on the prevalence and relative frequency of herpes sim- plex virus 1 and herpes simplex virus 2 encephalitis and meningitis. Seventy-one studies were included. Overall, the prevalence of herpes simplex virus encephalitis among patients tested was 8% (95% confidence interval, 6%-11%; I2 = 98%) and the prevalence of herpes simplex virus meningitis among aseptic patients tested was 4% (95% confidence interval, 3%-7%; I2 = 95%), and a significant difference was observed by region. The results of our subgroup analysis for herpes simplex virus encephalitis revealed a prevalence of 8% for pediatric patients and ado- lescents and 12% for adults. The results for herpes simplex virus meningitis showed a prevalence of 4% for pediatric patients and adolescents and 9% for adults. We observed significant differ- ences in the frequency of herpes simplex virus 1 and herpes simplex virus 2 detection rates by region. Having high rates of missed cases due to inadequate, highly sensitive paraclinical tests performed on patients with suspected viral central nervous system infection is one of the pos- sible factors. More studies are needed to detect the possible flaws in the process of diagnosis in different regions.
PubMed: 37553966
DOI: 10.5152/TurkArchPediatr.2023.23007 -
Reviews in Medical Virology May 2024Serious adverse events following vaccination include medical complications that require hospitalisation. The live varicella vaccine that was approved by the Food and... (Review)
Review
Serious adverse events following vaccination include medical complications that require hospitalisation. The live varicella vaccine that was approved by the Food and Drug Administration in the United States in 1995 has an excellent safety record. Since the vaccine is a live virus, adverse events are more common in immunocompromised children who are vaccinated inadvertently. This review includes only serious adverse events in children considered to be immunocompetent. The serious adverse event called varicella vaccine meningitis was first reported in a hospitalised immunocompetent child in 2008. When we carried out a literature search, we found 15 cases of immunocompetent children and adolescents with varicella vaccine meningitis; the median age was 11 years. Eight of the children had received two varicella vaccinations. Most of the children also had a concomitant herpes zoster rash, although three did not. The children lived in the United States, Greece, Germany, Switzerland, and Japan. During our literature search, we found five additional cases of serious neurological events in immunocompetent children; these included 4 cases of progressive herpes zoster and one case of acute retinitis. Pulses of enteral corticosteroids as well as a lack of herpes simplex virus antibody may be risk factors for reactivation in immunocompetent children. All 20 children with adverse events were treated with acyclovir and recovered; 19 were hospitalised and one child was managed as an outpatient. Even though the number of neurological adverse events remains exceedingly low following varicella vaccination, we recommend documentation of those caused by the vaccine virus.
Topics: Adolescent; Child; Child, Preschool; Female; Humans; Male; Acyclovir; Antiviral Agents; Chickenpox; Chickenpox Vaccine; Herpesvirus 3, Human; Meningitis, Viral; Nervous System Diseases; Vaccination; Virus Activation
PubMed: 38658176
DOI: 10.1002/rmv.2538