Key result
Dietary fat restriction resolves recurrent perianal abscesses alongside dramatic triglyceride reduction.
Why the study?
Does reduction of plasma triglycerides resolve recurrent perianal infections in a neonate with LPL deficiency and severe hypertriglyceridaemia?
Case Report (n=1)
Does reduction of plasma triglycerides resolve recurrent perianal infections in a neonate with LPL deficiency and severe hypertriglyceridaemia?
This case report suggests a potential pathophysiological link between severe hypertriglyceridaemia and immune dysfunction in LPL deficiency, which may have implications for its management.
Answers are grounded in this paper and cite their sources.
Hypothesis-generating for hypertriglyceridaemia impairing immunity in neonatal LPL deficiency; prospective studies needed before changing management.
Lipoprotein lipase (LPL), a member of the triglyceride lipase gene family, is synthesised by parenchymal cells of the heart, skeletal muscle and adipose tissues before being transported to luminal surfaces of vascular endothelial cells to exert its main physiological function to hydrolyse plasma lipoproteins. LPL deficiency is a rare autosomal recessive disorder, resulting in severe hypertriglyceridaemia from birth. The effect of marked hypertriglyceridaemia on the immune function in children has not been described. We present a case of a neonate with LPL deficiency and grossly elevated plasma triglyceride levels, presenting with recurrent and recalcitrant perianal abscesses suggestive of underlying immunodeficiency. With reduced levels of plasma triglycerides, the recurrent perianal infections resolved. This case report reviews evidence for potential deleterious effects of hypertriglyceridaemia on immune function, however, underlying mechanisms are poorly understood. Whether hypertriglyceridaemia contributes to immune dysfunction in this context is unknown. If there is a pathophysiological link, this may have implications for hypertriglyceridaemia management.
No takes yet. Share an insight, caveat, or question.
Akesson et al. (2016) conducted a case report in Lipoprotein lipase deficiency (n=1). Dietary fat restriction was evaluated on Resolution of recurrent perianal abscesses and plasma triglyceride levels. Dietary fat restriction successfully reduced plasma triglycerides from 332 mmol/L to 10-20 mmol/L and coincided with the complete resolution of recurrent, recalcitrant perianal abscesses.
Synapse has enriched 10 related papers. Consider them for comparative context: