Paroxysmal hemicrania in children and adolescents: a systematic review of case series and case reports

Abstract

Background It has been so long since a child was first diagnosed with paroxysmal hemicrania, however, it is unknown whether children's paroxysmal hemicrania follows the same patterns as adults. We aim to report the accessible demographic, clinical and radiological characteristics of reported pediatric paroxysmal hemicrania.Methods This study followed the latest version of PRISMA. PubMed, Web of Science, and Scopus were searched systematically without time limitation. We included all peer-reviewed articles including observational or interventional studies reporting paroxysmal hemicrania cases in children or adolescents based on ICHD criteria. Data extracted included paroxysmal hemicrania class, gender, age, age of onset, frequency, duration, site, severity, and quality of pains, triggers, autonomic and migrainous symptoms, as well as a sense of restlessness/agitation, response to treatment, laboratory investigations, imaging, comorbidity, and family history. For quality assessment, two independent reviewers assessed the methodological quality of the included studies through the Joanna Briggs Institute critical appraisal checklist. ResultsA total of 182 records were identified and reduced to 116 after removing duplicates. After screening, 22 articles met the inclusion criteria. Overall, the studies represent 35 children or adolescents with paroxysmal hemicrania. We found a male to female sex ratio of 1.125:1. Onset occurred at a broad range of 1–14 years. The mean age of onset among reported cases in children and adolescents was 6.30 years, while the mean age of diagnosis was 7.96 years. The attacks frequency and duration were greatly varied. The left-sided pain was approximately reported twice more than the right-sided headaches. The characteristic of the pain was usually severe in intensity making children restless or agitated during an attack. In nearly all of the cases, it was accompanied by ipsilateral cranial autonomic features. While the majority of attacks are spontaneous, there were some common triggers. The physical examination, EEG, and brain MRI were normal. Almost all patients benefited from Indomethacin and showed complete response to treatment, while some needed combination treatment of Indomethacin with other medications.ConclusionAlthough pediatric-onset paroxysmal hemicrania has similar features to adult-onset paroxysmal hemicrania, there are some misleading challenges with ICHD criteria for younger children, limiting the diagnosis. Moreover, due to concomitant migrainous features paroxysmal hemicrania may be confused with migraine in children and adolescents.Trial RegistrationThis study was registered as a systematic review in PROSPERO.