ArticleForensic science, medicine, and pathology2016
Hippocampal malformation associated with sudden death in early childhood: a neuropathologic study: Part 2 of the investigations of The San Diego SUDC Research Project.
Article in Forensic science, medicine, and pathology, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 35 papers.
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35 citing papers in PubMed, 50 citations in OpenAlex.
- Forensic neuropathology: 2026 update.Free neuropathology · 2026Review
- Sudden unexpected infant death, sudden unexplained death in childhood, and sudden unexpected death in epilepsy.Developmental medicine and child neurology · 2025Review
- Developmental mechanisms underlying pediatric epilepsy.Frontiers in neurology · 2025Review
- Raphe and ventrolateral medulla proteomics in sudden unexplained death in childhood with febrile seizure history.Acta neuropathologica · 2024Article
- Video Analyses of Sudden Unexplained Deaths in Toddlers.Neurology · 2024Article
- Sudden Unexplained Death in Childhood: Current Understanding.Pediatric emergency care · 2023Article
- Epilepsy-associated death in the Southwestern Ontario: A clinicopathological correlation study.Brain pathology (Zurich, Switzerland) · 2023Article
- The fundamental need for unifying phenotypes in sudden unexpected pediatric deaths.Frontiers in medicine · 2023Article
- Proteomic differences in hippocampus and cortex of sudden unexplained death in childhood.Acta neuropathologica · 2022Article
- Genetic Determinants of Sudden Unexpected Death in Pediatrics.Genetics in medicine : official journal of the American College of Medical Genetics · 2022Article
- Blinded review of hippocampal neuropathology in sudden unexplained death in childhood reveals inconsistent observations and similarities to explained paediatric deaths.Neuropathology and applied neurobiology · 2022Article
- Considering the Role of Extracellular Matrix Molecules, in Particular Reelin, in Granule Cell Dispersion Related to Temporal Lobe Epilepsy.Frontiers in cell and developmental biology · 2022Review
- Seizure-related deaths in children: The expanding spectrum.Epilepsia · 2021Article
- Neuropathology in the North American sudden unexpected death in epilepsy registry.Brain communications · 2021Article
- Medullary Serotonergic Binding Deficits and Hippocampal Abnormalities in Sudden Infant Death Syndrome: One or Two Entities?Frontiers in pediatrics · 2021Article
- Genetic Factors Underlying Sudden Infant Death Syndrome.The application of clinical genetics · 2021Review
- A de novo BRPF1 variant in a case of Sudden Unexplained Death in Childhood.European journal of medical genetics · 2020Article
- Hippocampal abnormalities and seizures: a 16-year single center review of sudden unexpected death in childhood, sudden unexpected death in epilepsy and SIDS.Forensic science, medicine, and pathology · 2020Article
- Hippocampal granule cell dispersion: a non-specific finding in pediatric patients with no history of seizures.Acta neuropathologica communications · 2020Article
- A human amygdala site that inhibits respiration and elicits apnea in pediatric epilepsy.JCI insight · 2020Article
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10 authors at 6 institutions in 1 country.
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Abstract
purposeSudden unexplained death in childhood (SUDC), while rare, accounts for an important fraction of unexpected deaths in children >1 year of age. Previously we reported an association between febrile seizures, hippocampal maldevelopment, and sudden, unexpected deaths in young children (1-6 years), termed "hippocampal maldevelopment associated with sudden death (HMASD)." Here, we characterize in greater detail the hippocampal pathology in a large cohort of cases (n = 42) of this entity, and attempt to define possible new entities responsible for sudden, unexplained death in young children without HMASD/febrile seizure phenotypes.
methodsWe performed comparative analysis on cases, which we classified in a cohort of 89 sudden and unexpected deaths as HMASD, explained deaths, SUDC with febrile seizure phenotype (SUDC-FS) but without hippocampal pathology, and SUDC (without hippocampal pathology or febrile seizure phenotype).
resultsThe frequency of each subgroup was: HMASD 48% (40/83); SUDC 27% (22/83); SUDC-FS 18% (15/83); explained 7% (6/83). HMASD was characterized clinically by sudden, sleep-related death, term birth, and discovery in the prone position. Key morphologic features of HMASD were focal granule cell bilamination of the dentate gyrus with or without asymmetry and/or malrotation of the hippocampus, associated with significantly increased frequencies of 11 other developmental abnormalities. We identified no other distinct phenotype in the unexplained categories, except for an association of febrile seizures without hippocampal maldevelopment.
conclusionsHMASD is a distinct clinicopathologic entity characterized by a likely developmental failure of neuronal migration in the dentate gyrus. Future research is needed to determine the causal role of HMASD in sudden death in early childhood.
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