Epidemiologic research into sudden infant death syndrome (SIDS) has resulted in the recognition of factors that place an infant at increased risk of SIDS. The avoidance of these risk factors promulgated through public education programs, such as the “Reduce the Risk” campaign in Australia, New Zealand, and Western Europe as well as the “Back to Sleep” campaign in the United States, led to astounding reductions in SIDS rates in these communities.1
Although these risk factors may provide clues to causation, the ultimate clarification of the mechanisms of death in SIDS will require basic research in pathology. For example, prone sleep position is a very important risk factor for SIDS, but its role in the cause of death remains an enigma. External airway obstruction,2,3 rebreathing into soft sleep surfaces,4–6 hyperthermia,7 and increased frequency of mild infectious symptoms combined with a stimulated mucosal immunoglobulin A system of the larynx, inducing release of cytokines in the brain,8–11 are among a number of proposed but not universally accepted mechanisms of death in SIDS infants who are found prone. The reduction of serotonergic, kainate, and muscarinic cholinergic neurotransmitter receptors in a subset of SIDS cases has also been proposed to link prone sleep position and sudden death.12–14 Similarly, the explanation of the relationship between SIDS and other risk factors, including premature birth, low birth weight, and smoke exposure, awaits new research into basic pathology. Answers to these questions await a new research using very sophisticated methods into the pathology of postmortem specimens from these infants.
Unfortunately, research in a number of countries, including the United Kingdom and Australia, has been dramatically reduced as a result of recent events involving retention of postmortem specimens. Some of the most important current research into SIDS depends on postmortem specimens. Delay or improper processing of these specimens will diminish or preclude their value for research purposes. The dramatic decline in rates has also resulted in a reduction of SIDS cases available for research purposes. Both of these factors conspire to prevent parents who are the most motivated to understand why their infants died from getting answers to their questions.
Given this impasse, where do we go from here? It seems to us that the parents of infants who die of SIDS and investigators who partner together hold the key to future research in pathology. A suggestion that we strongly support is that together, they should advocate for statutory authorization to use these specimens legally for research without having to obtain consent from each individual family. There is precedent for this. In California, parents of infants who die of SIDS were the driving force for legislation that authorized research using postmortem specimens of infants who had died suddenly. In collaboration with investigators, these bereaved yet visionary parents helped to write the senate bills, lobbied their legislators, testified before government committees, and assisted in the creation of standardized protocols for scene investigation and postmortem examination. Without their commitment and support, some of the most important SIDS research during the past decade could not have taken place. It seems that broadening this experience beyond California is a reasonable goal.
How can this be accomplished? Investigators need to communicate these research-related issues to the SIDS parent community. Just as collaboration of SIDS researchers with national parent education programs that address infant care produced stunning and significant reductions in SIDS rates, we believe that the collaboration of parents and investigators can result in legislation that authorizes research using postmortem specimens. The methods used in these studies often depend on postmortem specimens that are collected and processed as soon as possible after death to yield credible and reproducible results. It is because of this that seeking authorization on a case-by-case basis is often impractical and will lead to a reduction of useful specimens for research purposes. There are many SIDS organizations, composed of parents of infants who died of SIDS, that can serve as an interface between newly bereaved survivors and legislative bodies and research laboratories.
We should emphasize that we understand that retaining postmortem specimens may be in potential conflict with religious beliefs, cultural mores, and personal attitudes. Recruitment of religious scholars and ethicists to assist parents, researchers, and legislators may help to facilitate achieving acceptable compromises. Public lectures and more informal workshops with smaller groups are undertakings that have assisted parents in understanding issues that researchers confront. These activities serve to demystify the scientific method and to allow parents to be more informed and involved in studies that have extreme emotional significance to families. Dialogue between researchers and parents will only serve to strengthen SIDS research and circumvent some of the unnecessary and destructive episodes that have damaged considerably what has in the past been a very fruitful and harmonious collaboration.
- Received December 19, 2003.
- Accepted December 19, 2003.
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- ↵Byard RW, Krous HF, eds. Sudden Infant Death Syndrome. Problems, Progress & Possibilities. London, UK: Arnold; 2001
- ↵Tonkin SL, Davis SL, Gunn TR. Upper airway radiographs in infants with upper airway insufficiency. Arch Dis Child.1994;70 :523– 529
- Kemp JS, Unger B, Wilkins D, et al. Unsafe sleep practices and an analysis of bedsharing among infants dying suddenly and unexpectedly: results of a four-year, population-based, death-scene investigation study of sudden infant death syndrome and related deaths. Pediatrics.2000;106(3) . Available at: pediatrics.org/cgi/content/full/106/3/e41
- ↵Kemp JS, Thach BT. Rebreathing of exhaled air. In: Byard RW, Krous HF, eds. Sudden Infant Death Syndrome: Problems, Progress & Possibilities. London, UK: Arnold; 2001:138–155
- ↵Kadim H, Kahn A, Sébire G. Distinct cytokine profile in SIDS brain: a common denominator in a multifactorial syndrome? Neurology.2003;61 :1256– 1259
- ↵Kinney HC, Filiano JJ, Sleeper LA, Mandell F, Valdes-Dapena M, White WF. Decreased muscarinic receptor binding in the arcuate nucleus in sudden infant death syndrome. Science.1995;269 :1446– 1450
- Panigrahy A, Filiano JJ, Sleeper LA, et al. Decreased kainate receptor binding in the arcuate nucleus of the sudden infant death syndrome. J Neuropathol Exp Neurol.1997;56 :1253– 1261
- ↵Panigrahy A, Filiano J, Sleeper LA, et al. Decreased serotonergic receptor binding in rhombic lip-derived regions of the medulla oblongata in the sudden infant death syndrome. J Neuropathol Exp Neurol.2000;59 :377– 384
- Copyright © 2004 by the American Academy of Pediatrics