Neonatal subpial hemorrhage: Padua experience and systematic review

Subpial hemorrhage (SPH) is a rare subtype of intracranial hemorrhage, predominantly affecting term neonates and often associated with cortical-subcortical infarction. We described the epidemiology of SPH by analyzing cases referred to our hospital and concurrently conducting a systematic review of the cases reported in the literature. We also illustrated factors associated with adverse outcomes. A retrospective study was conducted on neonates with SPH referred to our hospital from 2013 to 2023 (cohort 1). Additionally, a systematic literature review on neonatal SPH was performed using PubMed, Scopus, Cochrane, and Web of Science up to April 2024 (cohort 2). Cohorts 1 and 2 were pooled for combined analysis. A total of 173 cases were analyzed, 10 original cases (cohort 1) and 163 literature cases (cohort 2). Ninety-two percent was term/late preterm neonates (59% male). Clinical presentations included seizures (36%), apnea (36%), and encephalopathy (18%). Ninety-four percent was diagnosed with brain magnetic resonance imaging and/or cranial ultrasound. Lesions were located in the temporal lobe in 60%, with infarctions adjacent to SPH in 90%. Sixteen percent died, 53% was diagnosed with neurological impairment, and 8% with epilepsy. In a subcohort of 67 patients (cohort 3) with available individual data (10/10 from cohort 1, 57/163 from cohort 2), low birth weight (LBW), seizures, neonatal infections, and parenchymal hemorrhage were significantly associated with adverse outcomes.

Neonatal SPH is rare, predominantly located in the temporal lobe, and frequently presents with seizures and apneas. Neurologic sequelae are common, and parenchymal hemorrhage was strongly associated with neurological impairment in our study.

• Subpial hemorrhage is a rare subtype of intracranial extra-axial bleeding, often associated with cortical-subcortical infarction in the adjacent parenchyma, predominantly affecting male term neonates. The temporal lobe is the most commonly involved area, frequently exhibiting the "yin-yang sign" on brain MRI.

• This is the first systematic review of neonatal subpial hemorrhage, emphasizing a distinctive clinical presentation marked by seizures and apneas (potentially of ictal origin), consistent with a high prevalence of temporal lobe involvement. Prognostically, a significant incidence of neurological impairment was observed, and the occurrence of parenchymal hemorrhage adjacent to subpial hemorrhage was strongly associated with adverse outcomes.