Please find below a reference list of recently published, peer-reviewed papers relating to SIDS or SUDI. The references have kindly been supplied by members of ISPID and may include papers in press. Any publication will be listed here for no longer than 2 years.

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2022

A mouse zoonotic virus (LCMV): A possible candidate in the causation of SIDS. Goldwater PN. Med Hypotheses 2022; 158: 110735. doi:10.1016/j.mehy.2021.110735


2021

Infection, celestial influences, and sudden infant death syndrome: a new paradigm. Goldwater PN. Oberg EO. Cureus 2021; 13 (8): e17449. doi:10.7759/cureus.17449

Infants' sleep: Israeli parents' knowledge, attitudes and practices. Shatz A, Joseph L, Korn L. Children 2021; 8 (9): 803. doi:10.3390/children8090803


2020

Increased thymus weight in sudden infant death syndrome compared to controls: The role of sub-clinical infections. Goldwater PN, Kelmanson IA, Little BB. Am J Hum Biol 2020; e23528. doi:10.1002/ajhb.23528

SIDS, prone sleep position and infection: An overlooked epidemiological link in current SIDS research? Key evidence for the "Infection Hypothesis". Goldwater PN. Med Hypotheses 2020; 144: 110114. doi:10.1016/j.mehy.2020.110114
"Mainstream SIDS researchers appear to have overlooked the key relationship between prone sleep position and infection. This omission has major implications for current and future SIDS research."

Recurrent sudden unexpected death in infancy: a case series of sibling deaths. Garstang JJ, Campbell MJ, Cohen MC, Coombs RC, Daman Willems C, McKenzie A, Moore A, Waite A. Arch Dis Child 2020:archdischild-2019-318379. doi:10.1136/archdischild-2019-318379
"Objectives: To determine the rate of sudden unexpected death in infancy (SUDI) for infants born after a previous SUDI in the same family, and to establish the causes of death and the frequency of child protection concerns in families with recurrent SUDI. Conclusions: The SUDI rate for siblings is 10 times higher than the current UK SUDI rate. Homicide presenting as recurrent SUDI is very rare. Many parents continued to smoke and exposed infants to hazardous co-sleeping situations, with these directly leading to or contributing to the death of six siblings. SUDI parents need support to improve parenting skills and reduce risk to subsequent infants."

Sudden Infant Death Syndrome. Letter. Keten A, Okdemir E. Am J Forensic Med Pathol 2020; 41 (2): 141. doi:10.1097/PAF.0000000000000551
Letter to the editor highlighting research into organ weight as of possible interest in the pathology of SIDS.

How might non nutritional sucking protect from sudden infant death syndrome. Zavala Abed B, Oneto S, Abreu AR, Chediak AD. Med Hypotheses 2020; 143: 109868. doi:10.1016/j.mehy.2020.109868
Review proposing a mechanism to explain how pacifiers might help prevent SIDS.

Blood and urine biomarkers associated with long-term respiratory dysfunction following neonatal hyperoxia exposure: implications for prematurity and risk of SIDS. Collada A, Mayer CA, MacFarlane PM. Respir Physiol Neurobiol 2020: 103465. doi:10.1016/j.resp.2020.103465
"In the present study, we identified serotonin (5-HT), dopamine (DA) and hyaluronan (HA) as three potential biomarkers that may be increased by neonatal hyperoxia and tested whether they would be associated with an impaired HVR in a rat model of supplemental O2 exposure."

Cerebellar heterotopia of infancy in sudden infant death syndrome: an observational neuropathological study of four cases. Matschke J, Sperhake JP, Wilke N, Püschel K, Glatzel M. Int J Legal Med 2020. doi:10.1007/s00414-020-02316-x
"We report four cases of SIDS in which neuropathological investigation revealed cerebellar heterotopia of infancy, a distinct malformation of the cerebellum, and discuss the potential impact of this condition on the aetiology and pathogenesis of SIDS."

Maternal intentions towards infant sleeping practices in Ireland. O'Brien N, McGarvey C, Hamilton K, Hayes B. Acta Paediatr 2020. doi:10.1111/apa.15352
"A cross-sectional survey of post-partum mothers was performed in the Rotunda Hospital over four months. Educational campaigns on safe sleep for infants in Ireland need to address modifiable SIDS risks factors, focusing on younger, non-Irish mothers, with lower educational attainment."

Maternal bereavement the year before or during pregnancy and total and cause-specific infant mortality: a cohort study from Denmark and Sweden. László KD, Johansson S, Miao M, Li J, Olsen J, Yuan W, Cnattingius S. Psychosom Med 2020. doi:10.1097/PSY.0000000000000822
Cohort study investigating associations between maternal bereavement the year before or during pregnancy and total and cause-specific infant mortality.

Pacifiers: a cause for confusion. Wild BM, Kornfeld B. Pediatr Ann 2020; 49 (5): e204-e206. doi:10.3928/19382359-20200419-01
"Pacifier avoidance is recommended in the newborn nursery to optimize exclusive breast-feeding rates according to the Baby-Friendly Hospital Initiative. There are many reasons why parents may choose to provide a pacifier to their infant, particularly due to the association between pacifier use with sleep and risk reduction for sudden infant death syndrome. Early use of a pacifier does not likely cause direct harm to infants or lead to difficulty breast-feeding. Pediatricians should partner with families to share a clear message in support of establishing exclusive breast-feeding, ideally before introduction of a pacifier."

Mother-infant co-sleeping and maternally reported infant breathing distress in the UK Millennium Cohort. Waynforth D. Int J Environ Res Public Health 2020; 17 (9): 2985. doi:10.3390/ijerph17092985
"The hypothesis that mother-infant co-sleeping will be associated with a lower probability of infant breathing distress is tested in the UK Millennium Cohort Study (n = 18,552 infants). Maternal, infant, family, and socio-economic covariates were included in logistic regression analysis, and in a machine learning algorithm (Random Forest) to make full use of the number of variables available in the birth cohort study data. Results from logistic regression analysis showed that co-sleeping was associated with a reduced risk of breathing difficulties (OR = 0.69, p = 0.027)."

Obstetricians' and gynecologists' communication practices around smoking cessation in pregnancy, secondhand smoke and sudden infant death syndrome (SIDS): a survey. Sontag JM, Singh B, Ostfeld BM, Hegyi T, Steinberg MB, Delnevo CD. Int J Environ Res Public Health 2020; 17 (8): 2908. doi:10.3390/ijerph17082908
USA: "An increased awareness of statewide cessation resources by ob/gyns is needed to assist patients with cessation. The development of standardized risk messaging may reduce the variation in communication practices among ob/gyns."

Pathology of unexpected sudden cardiac death: obstructive sleep apnea is part of the challenge. Ottaviani G, Buja LM. Cardiovasc Pathol 2020: 107221. doi:10.1016/j.carpath.2020.107221
Editorial reviewing "the pathology of SCD, including sudden infant death syndrome (SIDS) and sudden intrauterine unexplained death (SIUD); OSA with its cardiovascular consequences; the possible link between SCD and OSA, discussing the potential mechanisms underlying these two frequent, but yet overlooked pathologies."

Factors associated with age of death in sudden unexpected infant death. Allen K, Anderson TM, Chajewska U, Ramirez JM, Mitchell EA. Acta Paediatr 2020. doi:10.1111/apa.15308
"Factors that varied with age of death are well-documented risk factors for SUID. The majority of these risk factors were associated with younger age at death after allowing for gestational age at birth."

Infant mortality related to NO2 and PM exposure: systematic review and meta-analysis. Kihal-Talantikite W, Marchetta GP, Deguen S. Int J Environ Res Public Health 2020; 17 (8): 2623. doi:10.3390/ijerph17082623
"In spite of a few number of epidemiological studies selected in the present literature review, our finding is in favor of a significant increase of infant death with the increase of air pollution exposure during either the pregnancy period or the first year of a newborn's life. Our findings have to be interpreted with caution due to weaknesses that could affect the strength of the associations and then the formulation of accurate conclusions. Future studies are called to overcome these limitations; in particular, (i) the definition of infant adverse outcome, (ii) exposure assessment, and (iii) critical windows of exposure, which could affect the strength of association."

Baby-box schemes in England: parent and practitioner experiences, and recommendations. Ball HL, Taylor CE. BMC Pediatr 2020; 20 (1): 154. doi:10.1186/s12887-020-02064-2
"Many assumptions exist about the origins and purpose of baby-boxes; this misinformation needs correcting, especially as it relates to infant death reduction and safe infant sleep. Baby-box schemes take multiple forms from those motivated by social welfare to those motivated by commercial profit. The English experience of partnership schemes between healthcare facilities and commercial box-providers reveals some success stories, along with multiple points of ambiguity, unanticipated difficulty, and concerns for infant safety."

No association to sudden infant death syndrome detected by targeted amplicon sequencing of 24 genes. Ferrante L, Opdal SH, Nygaard V. Acta Paediatr 2020. doi:10.1111/apa.15295
"There was no association between rare variants in the included genes and SIDS."

Death-scene investigations contribute to legal protection in unexpected child deaths in Norway. Bøylestad L, Stray-Pedersen A, Vege Å, Osberg S, Rognum T. Acta Paediatr 2020. doi:10.1111/apa.15284
"Death-scene investigations illuminate uncertainty about the cause of death, especially in grey-area cases where accidental suffocation, neglect or abuse is suspected. Knowledge about the course of events and the cause of death enhances both the child's and the caregiver's legal protection. Death-scene investigations should therefore be mandatory."

Multidisciplinary study of sudden unexpected infant death in Liguria (Italy): an nine-year report. Ventura F, Barranco R, Smith A, Ceccherini I, Bandettini R, Coviello D, Morando A, Nozza P, Buffelli F, Fulcheri E, Palmieri A. Minerva Pediatr 2020. doi:10.23736/S0026-4946.20.05599-1
"We conducted a retrospective analysis of cases of SUID referred to the SIDS-ALTE Center of the Liguria Region (Italy) from 2010 to 2018."

Prevalence of risk factors for sudden infant death among Indigenous and non-Indigenous people in Australia. Shipstone RA, Young J, Kearney L, Thompson JMD. Acta Paediatr 2020. doi:10.1111/apa.15274
"Aim: To examine differences in the prevalence of risk factors for sudden unexpected death in infancy (SUDI) between Aboriginal and Torres Strait Islander and non-Indigenous infants."

Risk and protective factors for sudden infant death syndrome. Oliveira AMF, Andrade PR, Pinheiro EM, Avelar AFM, Costa P, Belela-Anacleto ASC. Rev Bras Enferm 2020; 73 (2) : e20190458. doi:10.1590/0034-7167-2019-0458
Brazil: "Objectives: To verify the occurrence of the risk and protective factors for sudden infant death syndrome during nursing consultation. Methods: Retrospective cohort study conducted based on medical records from a primary care unit in the municipality of São Paulo."

SIDS associated RYR2 p.Arg2267His variant may lack pathogenicity. Kohli U, Nayak HM. J Electrocardiol 2020; 60: 23-26. doi:10.1016/j.jelectrocard.2020.03.007
"We report a family with 5 members (mother and 4 children) who are carriers of a rare RYR2 variant (c.6800G > A, p.Arg2267His [Exon: 45], heterozygous) which has previously been identified in a SIDS victim and shown to confer a gain-of-function CPVT phenotype in vitro. All of these 5 family members including the mother (age range 7 to 41 years) have had negative exercise stress tests, echocardiograms and Holter monitors. These findings suggest that caution should be exercised in inferring pathogenicity of rare RYR2 variants based on in vitro functional data which does not always translate to human phenotype."

Triadin knockout syndrome is absent in a multi-center molecular autopsy cohort of sudden infant death syndrome and sudden unexplained death in the young and is extremely rare in the general population. Clemens DJ, Gray B, Bagnall RD, Tester DJ, Dotzler SM, Giudicessi JR, Matthews E, Semsarian C, Behr ER, Ackerman MJ. Circ Genom Precis Med 2020; 13 (2): e002731. doi:10.1161/CIRCGEN.119.002731
"Triadin knockout syndrome (TKOS) is a potentially lethal arrhythmia disorder caused by recessively inherited null variants in TRDN-encoded cardiac triadin. TKOS is an exceedingly rare clinical entity that does not contribute meaningfully to either sudden infant death syndrome or sudden unexplained death in the young. However, despite its rarity and absence in large sudden death cohorts, TKOS remains a malignant and potentially lethal disorder which requires further research to better care for these patients."

A review of long QT syndrome: everything a hospitalist should know. Sharma N, Cortez D, Disori K, Imundo JR, Beck M. Hosp Pediatr 2020; 10 (4): 369-375. doi:10.1542/hpeds.2019-0139
"In this article, we will review various aspects of long QT syndrome (LQTS) necessary for hospitalists who care for children, adolescents, and young adults who have known LQTS and also review presenting features that should make one consider LQTS as a cause of hospitalization."

Back to the breast: a historical overview of the perceived connections between sudden infant death syndrome and breastfeeding. Cowgill B. J Hum Lact 2020; 36 (2): 310-317. doi:10.1177/0890334420906837
Historical review article.

Explaining sudden infant death with cardiac arrhythmias: complete exon sequencing of nine cardiac arrhythmia genes in Dutch SIDS cases highlights new and known DNA variants. Liebrechts-Akkerman G, Liu F, van Marion R, Dinjens WNM, Kayser M. Forensic Sci Int Genet 2020; 46: 102266. doi:10.1016/j.fsigen.2020.102266
"By using targeted massively parallel sequencing (MPS) in 142 Dutch SIDS cases, we performed a complete exon screening of all 173 exons from 9 cardiac arrhythmias genes SCN5A, KCNQ1, KCNH2, KCNE1, KCNE2, CACNA1C, CAV3, ANK2 and KCNJ2 (∼34,000 base pairs), that were selected to harbour previously established SIDS-associated DNA variants."

Geographic variation in sudden unexpected infant death in the United States. Mitchell EA, Yan X, Ren SY, Anderson TM, Ramirez JM, Lavista Ferres JM, Johnston R. J Pediatr 2020; 220: 49-55. doi:10.1016/j.jpeds.2020.01.006
"Objectives: To assess the geographic variation of sudden unexpected infant death (SUID) and test if variation in geographic factors, such as state, latitude, and longitude, play a role in SUID risk across the US."

Central serotonin and autoresuscitation capability in mammalian neonates. Erickson JT. Exp Neurol 2020; 326: 113162. doi:10.1016/j.expneurol.2019.113162
"The purpose of this review is to discuss some of the methodological considerations associated with 5-HT-deficient animal models, to summarize major findings arising from their use, and to highlight several key issues related to 5-HT involvement in gasping and the autoresuscitation response."

Sudden infant death syndrome: do the parents follow the recommendations? [Spanish]. Ruiz Botia I, Cassanello Peñarroya P, Díez Izquierdo A, Martínez Sánchez JM, Balaguer Santamaria A. An Pediatr (Barc) 2020; 92 (4): 222-228. doi:10.1016/j.anpedi.2019.06.011
"The objective of this study is to describe the prevalence of prone position during sleep as well as other risk factors associated with SIDS in a sample of Spanish babies and infants. There is a high prevalence of modifiable risk factors for SIDS among the studied population. Personalized education should be promoted, along with other campaigns to raise awareness and prevent SIDS."

Parental knowledge of safe infant sleep and sudden infant death syndrome is inadequate in Croatia. Barbir I, Ball H, Zakarija-Grković I. Acta Paediatr 2020. doi:10.1111/apa.15207
"Sudden infant death syndrome (SIDS) is the leading category of death for post-neonatal babies in wealthy countries and rates range from 0.06 to 0.9/1000 liveborn infants (1). Most research into infant sleep safety and SIDS is conducted in large countries and published in English; smaller nations may not have the resources to conduct SIDS case-control studies, nor the population size to make them viable. Consequently, in some countries SIDS risk-reduction guidance can be patchy, and parental safe sleep knowledge may be ad-hoc and based on guidance issued in other countries."

Back to sleep: teaching adults to arrange safe infant sleep environments. Carrow JN, Vladescu JC, Reeve SA, Kisamore AN. J Appl Behav Anal 2020. doi:10.1002/jaba.681
"Behavioral skills training (BST) is an evidence-based teaching strategy shown to successfully teach various safety skills to children and adults. The current study evaluated the effectiveness of behavioral skills training to teach safe infant sleep practices to typically developing adults."

T-wave alternans in nonpathological preterm infants. Marcantoni I, Sbrollini A, Agostinelli G, Surace FC, Colaneri M, Morettini M, Pozzi M, Burattini L. Ann Noninvasive Electrocardiol 2020: e12745. doi:10.1111/anec.12745
"Our preliminary retrospective study suggests that nonpathological PTI show TWA of few tens of µV, the interpretation of which is still an open issue but could indicate a condition of cardiac risk possibly related to the low development status of the infant. Further investigations are needed to solve this issue."

Transitional objects of grief. Goldstein RD, Petty CR, Morris SE, Human M, Odendaal H, Elliott AJ, Tobacco D, Angal J, Brink L, Prigerson HG. Compr Psychiatry 2020; 98: 152161. doi:10.1016/j.comppsych.2020.152161
"Bereaved parents often keep, cherish and visit saved objects of their deceased child. This research examined the hypothesis that these objects behave as transitional objects of grief in bereaved mothers during three years following their infants' deaths from Sudden Infant Death Syndrome."

A standardized postmortem protocol to assess the real burden of sudden infant death syndrome. Rizzo S, De Gaspari M, Carturan E, Paradiso B, Favretto D, Thiene G, Basso C. Virchows Arch 2020. doi:10.1007/s00428-020-02747-2
"In conclusion, since the application of a standardized protocol of post-mortem investigation, inflammatory, mostly infective, cardio-pulmonary diseases have been identified as the most common cause of SUID, with SIDS falling from 94 to 42% of SUID. Efforts must be made to implement a uniform autopsy protocol to provide reliable epidemiological data on SIDS."

Infant care practices and parent uptake of safe sleep messages: a cross-sectional survey in Queensland, Australia. Cole R, Young J, Kearney L, Thompson JMD. BMC Pediatr 2020; 20 (1): 27. doi:10.1186/s12887-020-1917-5
"This study aims to describe contemporary infant care practices employed by families related to the current public health SUDI prevention program."

The SIDS summit. Hageman JR. Pediatr Ann 2020; 49 (1): e1-e2. doi:10.3928/19382359-20191211-02
"Report on the 3rd Annual SIDS Summit organized by Dr. Jan-Marino (Nino) Ramirez, the Director of the Center for Integrative Brain Research at Seattle Children's Hospital, and John Kahan, the Chief Data Analytics Officer at Microsoft and the president of Aaron Matthew SIDS Research Guild of Seattle Children's Hospital."

Estimating the burden of prematurity on infant mortality: a comparison of death certificates and child fatality review in Ohio, 2009-2013. Montgomery M, Conrey E, Okoroh E, Kroelinger C. Matern Child Health J 2020; 24 (2): 135-143. doi:10.1007/s10995-019-02851-z
"Among 456 deaths categorized as sudden infant death syndrome on death certificates, approximately 50% (230) were categorized as missing, unknown, or undetermined by Child Fatality Reviews."

U-shaped pillows and sleep-related infant deaths, United States, 2004-2015. Cottengim C, Parks SE, Erck Lambert AB, Dykstra HK, Shaw E, Johnston E, Olson CK, Shapiro-Mendoza CK. Matern Child Health J 2020; 24 (2): 222-228. doi:10.1007/s10995-019-02847-9
"An examination of the demographics and circumstances of 141 infant deaths during 2004-2015 in the US National Fatality Review Case Reporting System with u-shaped pillows in the sleep environment."

Jean W Keeling. Khong TY, Malcomson R. Pediatr Dev Pathol 2020; 23 (1): 4-7. doi:10.1177/1093526619892715
This is a short appreciation of Jean W Keeling as a pediatric pathologist.

Distinct populations of sudden unexpected infant death based on age. Lavista Ferres JM, Anderson TM, Johnston R, Ramirez JM, Mitchell EA. Pediatrics 2020; 145 (1). pii:e20191637. doi:10.1542/peds.2019-1637
"The aim of this study is to determine if there are statistically different subcategories of SUID that are based on the age of death of an infant. Proposes sudden unexpected early neonatal deaths (SUENDs; days 0-6) and postperinatal SUIDs (days 7-364) as separate categories for research."

Knowledge assessment of sudden infant death syndrome risk factors in expectant mothers: A prospective monocentric descriptive study. Gemble A, Hubert C, Borsa-Dorion A, Dessaint C, Albuisson E, Hascoet JM. Arch Pediatr 2020; 27 (1): 33-38. doi:10.1016/j.arcped.2019.10.012
France: "In this prospective, descriptive monocentric study, we aimed to evaluate the level of expectant women's knowledge about SIDS."

Considerations in Safe to Sleep® messaging: Learning from African-American mothers. Stiffler D, Matemachani SM, Crane L. J Spec Pediatr Nurs 2020; 25 (1): e12277. doi:10.1111/jspn.12277
"The purpose of this study was to identify why African-American mothers do not tend to follow the Safe to Sleep® recommendations and to begin to identify a way to frame the Safe to Sleep® message so that African-American mothers might be more likely to follow these recommendations."

Premature birth, homeostatic plasticity and respiratory consequences of inflammation. Gauda EB, McLemore GL. Respir Physiol Neurobiol 2020; 274:103337. doi:10.1016/j.resp.2019.103337
"We propose in this review that simply being born early in the last trimester can trigger homeostatic plasticity within the respiratory network tipping the balance toward inhibition that persists in infancy. We discuss the intersection of premature birth, homeostatic plasticity and biological mechanisms leading to respiratory depression during inflammation in former premature infants."

Risk of adverse birth outcomes after maternal varenicline use: A population-based observational study in Denmark and Sweden. Pedersen L, Petronis KR, Nørgaard M, Mo J, Frøslev T, Stephansson O, Granath F, Kieler H, Sørensen HT. Pharmacoepidemiol Drug Saf 2020; 29 (1): 94-102. doi:10.1002/pds.4894
"Use of varenicline during pregnancy does not appear to increase the risk of major congenital malformations or other adverse birth outcomes."

Modeling safe infant sleep in the hospital. Frey E, Hamp N, Orlov N. J Pediatr Nurs 2020; 50: 20-24. doi:10.1016/j.pedn.2019.10.002
"Quality improvement project to improve safe sleep practice (SSP) adherence by healthcare providers working with infants admitted to an inpatient pediatric unit in an urban academic center specifically increasing compliance on five core SSP (supine, alone in the crib, no objects in crib, appropriate bundling, and flat crib)."