Abstract
Witnessed reports of sudden death are rare, but critical to deciphering its mechanism(s). We report such a death in a seemingly healthy 8-month-old boy in whom seizures and respiratory distress in the prone position were witnessed upon discovery during a sleep period. Following cardiopulmonary resuscitation, anoxic encephalopathy resulted in “brain death” and withdrawal of life support after 2 days. The autopsy did not reveal a primary anatomic cause of death. Metabolic evaluation failed to uncover an inborn error of ammonia, amino, organic, or fatty acid metabolism. Seizures in this case may have been secondary to cerebral hypoxia–ischemia complicating cardiorespiratory arrest of unknown etiology. Yet, they may represent the first manifestation of idiopathic epilepsy, triggering cardiopulmonary arrest, analogous to the terminal events postulated in sudden and unexplained death in epilepsy. This report alerts the forensic community to the possibility that sudden and unexplained death in infants may be due to seizures.
References
Krous HF, Beckwith JB, Byard RW, Rognum TO, Bajanowski T, Corey T, Cutz E, Hanzlick R, Keens TG, Mitchell EA. Sudden infant death syndrome and unclassified sudden infant deaths: a definitional and diagnostic approach. Pediatrics. 2004;114:234.
Kinney HC, Thach BT. The sudden infant death syndrome. N Engl J Med. 2009;361:795–805.
Kinney HC, Richerson GB, Dymecki SB, Darnall RA, Nattie EE. The brainstem, serotonin, and sudden infant death syndrome: a review. Annu Rev Pathol Mech Dis. 2009;4:517–49.
Randall B. Witnessed sudden infant death syndrome: a case report. J SIDS Infant Mortal. 1996;1:53–5.
Krous HF, Chadwick A, Hass EA. Sudden infant death while awake. Forensic Sci Med Pathol. 2008;4:40–6.
Stevens LH. Sudden unexplained death in infancy: observations on a natural mechanism of adoption of the face down position. Am J Dis Child. 1965;110:243–7.
Thach BT. Sleep apnea in infancy and childhood. Med Clin North Am. 1985;69:1289–315.
Kaur B, Taylor O. Retinal hemorrhages. Arch Dis Child. 1990;65:1369–72.
Krikham F. Cardiac arrest and post resuscitation of the brain. Eur J Neurol Pediatr. 2011;15:379–89.
Donner EF, Smith CR, Sneed O III. Sudden and unexplained death in children. Neurology. 2001;57:430–4.
Platt MW, Blair PS, Fleming PJ, et al. A clinical comparison of SIDS and explained sudden infant deaths: how healthy and how normal? Arch Dis Child. 2000;82:98–106.
Hewertson J, Poets CF, Samuels MP, Boyd SC, Neville BGR, Southall DP. Epileptic-induced hypoxemia in infants with apparent life-threatening events. Pediatrics. 1994;94:148–56.
Poets CF, Meny RG, Chobanian MR, Bonofigio RE. Gasping and other cardiorespiratory patterns during sudden infant deaths. Pediatr Res. 1999;45:350–4.
Rodriguez M, McMillan K, Crandall LA, Minter ME, Grafe MR, Poduri A, Kinney HC. Hippocampal asymmetry and sudden unexpected death in infancy: a case report. Forensic Med Pathol Sci. 2012;8:441–6.
Richerson GB, Buchanan GF. The serotonin-axis: shared mechanisms in seizures, depression, and SUDEP. Epilepsia. 2011;52(Suppl 1):28–38.
Acknowledgments
We deeply appreciate the parents’ courage in sharing their experience of the loss of their son in the hopes of advancing basic knowledge about sudden and unexpected death in infancy. We are grateful for the helpful comments of Drs. Joseph J. Volpe and Eugene E. Nattie in manuscript preparation. This study was supported by the Cooper Trewin Foundation for Sudden Unexplained Death in Childhood, program in Sudden and Unexpected Death in Pediatrics, Boston Children’s Hospital, MA, Eunice Kennedy Shriver Institute of Child Health and Development (P01-042774441) (HCK), and Intellectual and Developmental Disabilities Research Center, Boston Children’s Hospital, MA (P30-HD18655).
Author information
Authors and Affiliations
Corresponding author
Rights and permissions
About this article
Cite this article
Kinney, H.C., McDonald, A.G., Minter, M.E. et al. Witnessed sleep-related seizure and sudden unexpected death in infancy: a case report. Forensic Sci Med Pathol 9, 418–421 (2013). https://doi.org/10.1007/s12024-013-9448-0
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s12024-013-9448-0