A case control study on autopsy findings in sudden unexplained nocturnal death syndrome

Abstract

Aim Sudden unexplained nocturnal death syndrome (SUNDS) has been linked to the Brugada syndrome. In some places, acute haemorrhagic pancreatitis is widely held to cause it. We conducted a systematic, controlled autopsy study on Filipino SUNDS victims to rule out structural heart findings as well as acute haemorrhagic pancreatitis as causes. Methods and results A case control autopsy study was conducted comparing SUNDS victims between 18 and 50 years of age who died within 1 h of symptom onset with age-and gender-matched controls. There were 24 SUNDS (mean age 34.5 years) and 24 controls (mean 32.7 years). The autopsy incidence of structural heart disease was 8.3% (95% CI (1% to 27%)) and focal pancreatic haemorrhage was 4.17% (95% CI (0.1% to 20%)) but zero for true acute haemorrhagic pancreatitis among SUNDS victims. Autopsy findings in SUNDS versus controls were not significantly different from each other, showing no diagnostic abnormality in any of the organs. There was no significant difference in the incidence of acute haemorrhagic pancreatitis in both the SUNDS and control groups. We did not find fetal dispersion of the atrioventricular (AV) node, sclerosis or fibrosis of the AV conduction system, in a substudy of SUNDS cases. Conclusions We have shown that there is no significant difference in the overall autopsy findings between SUNDS and controls. Autopsy findings were normal in 70% of SUNDS; no cardiac structural pathology was found in 87% of cases. Haemorrhagic pancreatitis is the cause of death in a minority of SUNDS. The cardiac conduction system is normal in a subgroup of SUNDS studied.

Sudden Unexplained Nocturnal Death Syndrome: Epidemiological and Morphological Characteristics in Thai Autopsy Cases

“…The finding is also in contrast to a study performed by Gervacio et al. who found that only one of 24 (4.2%) Filipino SUNDS cases had coronary atherosclerosis. Such previous findings suggest that coronary atherosclerosis found in Thai SUNDS cases might be coincident without pathologic signs of either recent or old myocardial infarction.…”

“…Most SUNDS cases in this study were located in the Northeastern region of Thailand, which is consistent with previous Thai studies . All SUNDS cases in this study only occurred in young or middle‐aged men, being consistent with previous Thai studies but not in other ethnic studies , which reported that men were 3.9–13.9 times more affected than women. Most SUNDS cases in this study were blue‐collar construction workers, who migrated from their original birth region to Bangkok for labor.…”

“…However, Gervacio et al. found 70% of SUNDS having structurally normal hearts with normal CCS in randomly selected five cases of SUNDS examined in his study.…”

A Case-Control Study of Sudden Unexplained Nocturnal Death Syndrome in the Southern Chinese Han Population

“…3 Previous study showed that mild respiratory tract infection or exposure to cigarette smoke may be the factors associated with these deaths 10 and that acute hemorrhagic pancreatitis, hemorrhagic congestion, edema of the lungs, infection with Pseudomonas pseudomallei, or cardiac conduction system abnormalities may explain the death of very rare SUNDS victims. 11,12 So far, there is no fatal pathological changes found in the vast majority of SUNDS decedents. 13 Brugada syndrome was reported to be phenotypically, genetically, and functionally the same disorder with SUNDS based on 3 pathogenic SCN5A mutations found in SUNDS cases.…”

Does Sudden Unexplained Nocturnal Death Syndrome Remain the Autopsy-Negative Disorder: A Gross, Microscopic, and Molecular Autopsy Investigation in Southern China

“…6 Kampuchea, Laos, and Hmong refugees in the United States were also reported to have a high incidence (59, 82, and 92 per 100,000 people years, respectively) of SUNDS. 7,8 The annual incidence of SUNDS is approximately 1–3 per 100,000 people in Southern China. 4,9 …”