Search

Background: Alice in Wonderland syndrome (AIWS) is a disorienting neurological condition that affects human perception to the senses of vision, hearing, touch, and sensation and the phenomenon of time. Herein we report two pediatric cases of AIWS temporally related to severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) infection., Case Presentation: An eight-year-old-girl without history of migraine or epilepsy experienced some episodes of visual distortions (micropsia, macropsia, and teleopsia) and misperception of sound, sometimes associated with headache. The onset of symptoms began at the occurrence of fever (38°C), during SARS-CoV-2 infection. Another six-year-old girl, with no history of migraine or epilepsy, experienced short-term episodes of visual (metamorphopsia) and color disturbance (chromatopsia), during an otherwise asymptomatic SARS-CoV-2 infection. In both cases, clinical examination was unremarkable; surface electroencephalography showed normal findings, without any correlation between visual phenomena and cortical activity; and brain magnetic resonance was normal. The patients were given symptomatic treatment, consisting of anti-inflammatory drugs on demand. The frequency of episodes decreased progressively following a negative SARS-CoV-2 test, with full remission in a few weeks. At the moment of hospital admission, none of the patients had completed the two-dose vaccination schedule for SARS-CoV-2., Conclusion: Based on our clinical experience, we believe SARS-CoV-2 may be responsible for AIWS, in addition to other neurological symptoms more frequently documented in the literature. Pathogenesis is multifactorial and arises from the activation of inflammatory pathways. We therefore suggest also searching for SARS-CoV-2, among other viruses linked with AIWS, in children presenting with visual and/or auditory hallucinations, even as isolated symptoms., Competing Interests: Declaration of competing interest The authors declare that the research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors. All authors disclose any financial and personal relationships with other people or organizations that could inappropriately influence (bias) their work., (Copyright © 2024 Elsevier Inc. All rights reserved.)

Background: The aim of this study was to investigate the features of cyclic vomiting syndrome (CVS) in a pediatric cohort and assess the safety and efficacy of cyproheptadine regardless of age., Methods: This retrospective, monocentric study enrolled children diagnosed with CVS. After collecting data from the medical records, a telephone-based questionnaire interview with the caregivers was conducted to obtain missing or unclear data. Prodromal symptoms, triggers, and comorbidities were analyzed. The safety and efficacy of cyproheptadine were evaluated in the entire patient cohort and compared between children under 5 years old and those 5 years of age or older., Results: A cohort of 24 subjects with CVS were included. Prodromal symptoms were reported in 45.8%, triggers in 33.3%, and comorbidities in 58.3% of patients. Migraine was present in 54% of the patients. Cyproheptadine was prescribed to 15 of the 24 patients (62.5%), with 40% and 80% achieving a reduction in the frequency of vomiting clusters of over 75% after a median duration of 6.03 and 12.13 months respectively. Prophylactic therapy did not provide any benefit to 20% of the patients. No statistically significant differences were found in vomiting features or cyproheptadine response between children above and below 5 years, except for a negative correlation between age and the presence of diarrhea during CVS episodes. No serious adverse effects were reported., Conclusions: Assessment of prodromes, triggers, and comorbidities aids in CVS diagnosis in children. Cyproheptadine is effective and safe for the management of CVS regardless of age.

Background: Sturge-Weber syndrome (SWS) is a neurocutaneous disorder for which the neurological aspects, particularly headaches, remain poorly understood, despite significantly affecting morbidity. The present study aimed to elucidate the prevalence, characteristics and treatment strategies, as well as explore the pathogenesis of headaches, in SWS., Methods: Using Preferred Reporting Items for Systematic reviews and Meta-Analyses (PRISMA) guidelines, we systematically reviewed observational studies, case reports and series from eight databases (Cochrane Library, EBSCO, Embase, Medline, PubMed, Science Direct, Scopus and Web of Science), published from 1978 to 2023, to investigate the prevalence, characteristics, medication response and pathogenic theories of headaches in SWS., Results: The review analyzed 48 studies, uncovering headache prevalence between 37% and 71%. Migraine-like headache affected up to 52% of individuals. Prophylactic and acute treatments included non-steroidal anti-inflammatory drugs, triptans and antiepileptic drugs, despite the lack of established guidelines. Life-threatening headaches in SWS are uncommon, typically accompanied by other neurological symptoms. The pathogenesis of headaches in SWS is considered to involve venous congestion and neuronal hyperexcitability linked to leptomeningeal angiomas., Conclusions: Headaches occur more frequently in individuals with SWS than in the general population. Despite symptoms meeting migraine criteria, these headaches should be considered secondary to vascular conditions. Implementing acute and prophylactic treatment is advised to reduce the impact on patients' lives., Competing Interests: Declaration of conflicting interestsThe authors declared no potential conflicts of interest with respect to the research, authorship and/or publication of this article.