Cholesteryl esters are water-insoluble lipids formed by the esterification of cholesterol with long-chain fatty acids via an esterbond between the cholesterol hydroxyl group and the fatty acid carboxylate, enabling efficient storage and transport of cholesterol in biological systems.[1] Their structure renders them more hydrophobic than free cholesterol, allowing them to form the non-polar core of lipoproteins and cytoplasmic lipid droplets surrounded by a phospholipidmonolayer.[2]The formation of cholesteryl esters occurs through enzymatic processes, primarily involving lecithin-cholesterol acyltransferase (LCAT) in plasma, which esterifies about 70% of circulating cholesterol, and acyl-CoA:cholesterol acyltransferase (ACAT) intracellularly for storage.[3]Cholesteryl ester transfer protein (CETP) facilitates the exchange of cholesteryl esters between lipoproteins, such as from HDL to LDL and VLDL, playing a key role in cholesterol distribution and homeostasis.[2] These processes prevent the toxicity of excess free cholesterol by converting it into a neutral, storable form that undergoes a continual cycle of hydrolysis and re-esterification with a typical half-life of around 24 hours in cells.[3]In biological function, cholesteryl esters serve as the primary transport form within lipoproteins like HDL and LDL for reverse cholesterol transport and delivery to tissues, while intracellularly they accumulate in lipid droplets for long-term storage, particularly in cells like macrophages.[1] They exhibit low solubility in phospholipid monolayers (approximately 7 wt% or 0.8–8 mol%), adopting conformations such as horseshoe or extended shapes that influence their hydrolysis and mobilization.[1]Hydrolysis by enzymes like cholesterol esterase reverses this process, releasing free cholesterol for cellular use or membrane incorporation.[2]Dysregulation of cholesteryl ester metabolism contributes to diseases, notably atherosclerosis, where excessive accumulation in macrophage foam cells forms lipid-laden plaques in arterial walls.[4] Oxidized cholesteryl esters, present in atherosclerotic lesions (up to 23% of cholesteryl linoleate), are biomarkers for cardiovascular disease, with plasma levels ranging from 3–920 nmol/L and reduced by treatments like atorvastatin.[3] Inherited disorders such as cholesteryl ester storage disease further highlight their role, involving lysosomal accumulation due to deficient hydrolysis.[2]
Structure and properties
Chemical structure
Cholesteryl esters are esters formed by the esterification of the 3β-hydroxyl group at the C3 position of cholesterol with the carboxyl group of a fatty acid, yielding a molecule that is more hydrophobic and less polar than free cholesterol.[5][6]The general chemical formula of cholesteryl esters is \ce{C27H45O-OCOR}, where R denotes the alkyl chain of the fatty acid; common examples include palmitate (16:0), oleate (18:1), and linoleate (18:2), which predominate in human plasma cholesteryl esters.90002-X)00538-5/fulltext)The molecular structure features a rigid four-ring cyclopentanoperhydrophenanthrene sterol nucleus derived from cholesterol, with an eight-carbon isooctyl side chain attached at C17, an ester linkage at C3 connecting to the fatty acyl group, and an acyl chain of variable length typically spanning 14 to 24 carbons.90002-X)Cholesteryl esters exhibit structural variations based on the fatty acid moiety, such as saturated versus mono-unsaturated chains; saturated esters promote denser molecular packing through linear chain alignment, whereas mono-unsaturated esters, with their characteristic kinks from double bonds (e.g., at Δ⁹ in oleate), reduce packing density and influence phase behavior.90002-X)
Physical properties
Cholesteryl esters are highly hydrophobic molecules, with octanol-water partition coefficients (logP) estimated at around 18 for common variants like cholesteryl palmitate, far exceeding the logP of approximately 8.7 for free cholesterol. This enhanced lipophilicity results in virtual insolubility in water, typically on the order of 10^{-12} mg/L or less, necessitating their incorporation into lipid carriers for aqueous environments. The structural basis for this hydrophobicity lies in the ester linkage to long-chain fatty acids, which amplifies the nonpolar surface area compared to unesterified cholesterol.[7][8][1]The physical state of cholesteryl esters is heavily influenced by the fatty acid chain length and saturation, leading to varied melting points. For instance, cholesteryl palmitate, esterified with a saturated C16 chain, exhibits a melting point of 75–77°C, forming a solid at room temperature. In contrast, cholesteryl oleate, with an unsaturated C18:1 chain, melts at 42–48°C, remaining liquid under physiological conditions near 37°C. These transitions reflect the packing efficiency of the alkyl chains, where unsaturation disrupts crystalline order and lowers the melting temperature.[9][10]Cholesteryl esters possess a density of approximately 0.93 g/cm³, consistent with their nonpolar, lipid-like composition. They also demonstrate optical activity stemming from the multiple chiral centers in the cholesterol backbone, yielding specific rotations of -24° (in chloroform) for cholesteryl palmitate and -20° (in THF) for cholesteryl oleate. Furthermore, these compounds readily form liquid crystalline phases, including cholesteric and smectic structures, alongside crystalline and isotropic liquid states, particularly in nonpolar solvents where they can self-assemble into ordered mesophases relevant to their phase behavior.[11][12][13]
Biosynthesis
Esterification reaction
The esterification of cholesterol represents a critical step in lipid metabolism, converting free cholesterol into cholesteryl esters for efficient storage and transport within cells and plasma. This reaction involves the covalent attachment of a fatty acyl chain to the hydroxyl group at the 3β-position of cholesterol, rendering the molecule more nonpolar and less toxic to cellular membranes. The process is essential in tissues where cholesterol influx exceeds immediate needs, preventing cytotoxicity from excess free cholesterol accumulation.[14]Cholesteryl ester biosynthesis occurs via two primary pathways: the intracellular acyl-CoA:cholesterol acyltransferase (ACAT)-mediated reaction and the plasma lecithin-cholesterol acyltransferase (LCAT)-mediated reaction. The ACAT pathway utilizes acyl-CoA as the acyl donor and predominates in cellular storage, while the LCAT pathway transfers the acyl group from phosphatidylcholine (lecithin) and is crucial for lipoprotein maturation in circulation.[15][14]The biochemical reaction for the ACAT pathway can be summarized by the following equation:\text{Cholesterol} + \text{R-CO-SCoA} \rightarrow \text{R-COO-cholesterol (cholesteryl ester)} + \text{CoA}Here, R denotes the fatty acyl chain, typically 16–18 carbons in length, derived from various fatty acids. This transacylation transfers the acyl group from the thioester bond of acyl-CoA to cholesterol, releasing free coenzyme A (CoASH). The reaction is reversible under in vitro conditions but is strongly favored in the forward direction in vivo, driven by cellular compartmentalization, CoA utilization in other pathways, and the physiological imperative to sequester surplus cholesterol.[14]In contrast, the LCAT pathway involves the transfer of a fatty acyl chain from the sn-2 position of phosphatidylcholine to cholesterol:\text{Cholesterol} + \text{1-acyl-sn-glycero-3-phosphocholine (from phosphatidylcholine)} \rightarrow \text{R-COO-cholesterol} + \text{2-lysophosphatidylcholine}This reaction occurs primarily on high-density lipoprotein (HDL) particles in plasma, where LCAT is activated by apolipoprotein A-I (apoA-I), and favors unsaturated fatty acids like linoleate for esterification.[16]At the molecular level, the ACAT mechanism proceeds through nucleophilic acyl substitution. The 3β-hydroxyl group of cholesterol is deprotonated by a histidine residue (e.g., His460 in ACAT1) acting as a general base within the enzyme's active site, generating an alkoxide ion. This nucleophile then attacks the electrophilic carbonyl carbon of acyl-CoA, forming a tetrahedral intermediate that collapses to yield the ester bond and expel the thiolate of CoA. The reaction occurs within the plane of the endoplasmic reticulum membrane, where both substrates and the catalyzing enzymes are localized.[14][17]LCAT employs a similar nucleophilic mechanism but uses a serine-histidine-aspartate catalytic triad to activate the cholesterol hydroxyl for attack on the sn-2 acyl ester of phosphatidylcholine, producing lysophosphatidylcholine as a byproduct. LCAT is a soluble enzyme secreted by the liver and circulates bound to HDL.[18]The primary substrates for ACAT are free cholesterol and acyl-CoA thioesters. Free cholesterol is sourced from dietary absorption in intestinal enterocytes, endogenous synthesis via the mevalonate pathway in hepatocytes, or receptor-mediated uptake from lipoproteins such as LDL in macrophages and other peripheral cells. Acyl-CoA is generated through the ATP-dependent activation of free fatty acids by acyl-CoA synthetases, providing a diverse pool of acyl chains that influence the composition of resulting cholesteryl esters. For LCAT, substrates include free cholesterol on nascent HDL and plasma phosphatidylcholine, with activity regulated by HDL maturation state.[14][19][16]This esterification predominantly takes place in the endoplasmic reticulum of key cell types involved in cholesterol handling for ACAT: enterocytes for packaging into chylomicrons, hepatocytes for incorporation into very low-density lipoproteins, and macrophages for storage in lipid droplets during foam cell formation. LCAT acts extracellularly in plasma. The ER localization for ACAT ensures proximity to cholesterol synthesis machinery and lipid droplet biogenesis sites, facilitating rapid response to fluctuating sterol levels.[14][20]Regulation of the esterification reaction balances cholesterolhomeostasis, with high free cholesterol levels acutely stimulating enzyme activity to promote ester formation and avert membrane disruption. Transcriptionally, the ACAT process is modulated via the sterol regulatory element-binding protein (SREBP) pathway: elevated free cholesterol suppresses SREBP activation and nuclear translocation, thereby inhibiting expression of ACAT2 (the predominant isoform in liver and intestine) and providing negative feedback to limit further esterification capacity under chronic excess. LCAT activity is regulated post-translationally by apoA-I activation and inhibited by excess free cholesterol or proteolysis. This dual regulation—post-translational activation by substrate availability and transcriptional restraint via SREBP for ACAT, and allosteric activation for LCAT—ensures adaptive control without over-accumulation of esters.[14][21][18]
Enzymes involved
The key enzymes responsible for cholesteryl ester synthesis are lecithin-cholesterol acyltransferase (LCAT) in plasma and the acyl-CoA:cholesterol acyltransferase (ACAT) isoforms, ACAT1 and ACAT2, in the endoplasmic reticulum membrane.[14][16] LCAT and ACATs were first identified in the mid-20th century as mediators of cholesterol esterification, with molecular cloning and characterization occurring in the 1990s for ACATs and 1970s for LCAT. Crystal structures of ACAT isoforms, solved in the early 2020s using cryo-electron microscopy, reveal a conserved active site featuring a histidine-serine catalytic dyad essential for acyl transfer, embedded within a bundle of transmembrane helices that segregate the active sites to the cytosolic leaflet. LCAT structures, determined by X-ray crystallography in the 2010s, show a α/β hydrolase fold with a liddomain that opens upon substrate binding on HDL surfaces.[22][18]LCAT, encoded by the LCAT gene on human chromosome 16, is synthesized in the liver and circulates as a glycoprotein associated with HDL. It plays a central role in reverse cholesterol transport by esterifying free cholesterol on nascent HDL particles, promoting their maturation into spherical HDL2 and facilitating cholesterol efflux from peripheral tissues. LCAT exhibits a strong preference for polyunsaturated fatty acids such as linoleic acid (18:2), resulting in cholesteryl linoleate as the predominant ester in plasma HDL.[16][18]ACAT1, encoded by the SOAT1 gene on human chromosome 1, is ubiquitously expressed but plays a prominent role in steroidogenic tissues such as the adrenal glands and gonads, where it facilitates cholesterol storage for hormonebiosynthesis, as well as in macrophages to prevent free cholesterol toxicity.[23] It exhibits a preference for oleic acid (18:1) as a fatty acyl substrate, contributing to the formation of cholesteryl oleate droplets in lipid-laden cells.[24] In contrast, ACAT2, encoded by the SOAT2 gene on chromosome 12, is primarily expressed in hepatocytes and enterocytes of the liver and small intestine, where it selectively esterifies cholesterol for incorporation into apolipoprotein B-containing lipoproteins like very low-density lipoprotein (VLDL) and chylomicrons, aiding dietary and hepatic cholesterol export.[25] ACAT2 shows broader substrate specificity toward unsaturated fatty acids, including polyunsaturated ones such as arachidonic (20:4) and eicosapentaenoic (20:5) acids, resulting in cholesteryl esters enriched in these moieties within circulating lipoproteins.[24]Efforts to therapeutically target ACATs for anti-atherosclerosis applications have focused on isoform-selective inhibitors, as ACAT1 inhibition in macrophages may reduce foam cell formation while ACAT2 inhibition in the intestine and liver could limit atherogenic lipoprotein production; however, early non-selective inhibitors like avasimibe failed in clinical trials due to adverse effects. Ongoing research explores refined inhibitors to exploit these tissue-specific roles without disrupting essential cholesterolhomeostasis. LCAT modulation is also under investigation, with genetic deficiencies linking low activity to increased cardiovascular risk.[26][27][18]
Biological functions
Role in lipid storage
Cholesteryl esters (CEs) serve as the primary intracellular storage form of cholesterol in various cell types, particularly where excess cholesterol must be sequestered to maintain cellular homeostasis. In non-hepatic cells such as adipocytes, foam cells (lipid-laden macrophages), and steroidogenic cells, CEs accumulate in the neutral lipid core of cytoplasmic lipid droplets (LDs), which are surrounded by a phospholipidmonolayer. This storage mechanism prevents the accumulation of free cholesterol, which can disrupt membrane integrity and induce cytotoxicity at high levels.[28][29][30]These LDs in adipocytes, foam cells, and steroidogenic cells enable efficient cholesterol buffering, allowing cells to handle fluctuations in cholesterol influx without altering membrane fluidity or function. For instance, during lipid loading in macrophages, the majority of incoming cholesterol can be esterified and stored as CEs within LDs, minimizing toxicity and supporting cellular demands. The evolutionary advantage of this system lies in its ability to tightly regulate intracellular cholesterol levels, a critical adaptation for preventing membrane perturbations in diverse physiological contexts.[30][31][32]Mobilization of stored CEs occurs through regulated hydrolysis, providing free cholesterol for membranesynthesis or export as needed. LDs are coated with perilipin family proteins, such as perilipin 1 and 2, which stabilize the droplets and control access by lipases during this process. This dynamic storage and retrieval system ensures cholesterol availability without compromising cellular viability.[33][34]
Role in lipoprotein transport
Cholesteryl esters (CEs) serve as a primary core component in major plasma lipoproteins, including high-density lipoprotein (HDL), low-density lipoprotein (LDL), and very low-density lipoprotein (VLDL), where they occupy the hydrophobic interior and constitute approximately 15-48% of the particle mass depending on the lipoprotein class.[35] In LDL, CEs comprise 37-48% of the total mass, forming the bulk of the neutral lipid core alongside triglycerides, while in HDL they account for 15-30%, and in VLDL for 12-15%.[35] This core structure allows CEs to solubilize and transport cholesterol systemically, preventing its precipitation in aqueous plasma.Assembly of CEs into lipoproteins occurs primarily in the intestine and liver. In enterocytes, acyl-CoA:cholesterol acyltransferase 2 (ACAT2) esterifies absorbed free cholesterol, incorporating the resulting CEs into the core of chylomicrons for secretion into the lymph and subsequent entry into circulation.[27] Similarly, in hepatocytes, ACAT2 catalyzes CE formation that is packaged into VLDL particles, enabling hepatic export of cholesterol to peripheral tissues.[27] For reverse cholesteroltransport, nascent discoidal HDL particles acquire free cholesterol from peripheral cells and are matured by lecithin-cholesterol acyltransferase (LCAT), which esterifies the cholesterol to form CEs that drive the particles into spherical HDL with a stable core.[36]The presence of CEs in the lipoprotein core contributes to their buoyancy and density gradient separation during ultracentrifugation, as the low-density neutral lipids displace higher-density proteins and phospholipids toward the surface.[37] For instance, HDL exhibits a higher CE-to-protein ratio than LDL, resulting in relatively lower density despite its smaller size, which facilitates efficient reverse transport.[37] CEs enable the solubilization of otherwise insoluble cholesterol within amphipathic lipoprotein envelopes, allowing its safe delivery to peripheral tissues for membranesynthesis and steroidogenesis while minimizing cytotoxicity.
Metabolism
Hydrolysis
The hydrolysis of cholesteryl esters (CE) represents the catabolic reversal of esterification, breaking down stored or internalized CE into free cholesterol and a fatty acid via enzymatic cleavage of the ester bond. This process is essential for liberating cholesterol for cellular use, such as membrane synthesis or efflux from cells like macrophages. The general reaction catalyzed by hydrolases is:\text{Cholesteryl ester} + \text{H}_2\text{O} \rightarrow \text{Cholesterol} + \text{R-COOH}where R-COOH denotes the released fatty acid chain.[38]In eukaryotic cells, CE hydrolysis occurs primarily through two distinct enzymes: lysosomal acid lipase (LAL) and neutral cholesterol ester hydrolase 1 (NCEH1), each adapted to specific cellular compartments and physiological contexts. LAL, encoded by the LIPA gene, is the principal lysosomal enzyme responsible for hydrolyzing CE and triglycerides derived from endocytosed lipoproteins, such as low-density lipoprotein (LDL) particles, within the acidic milieu of lysosomes. It exhibits an optimal activity at pH 4.5–5.0, aligning with the lysosomal environment, and plays a critical role in intracellular lipid degradation to prevent toxic accumulation.[39][40] Mutations in LIPA that severely impair LAL function lead to Wolman disease, a rare autosomal recessive lysosomal storage disorder characterized by massive CE buildup in organs like the liver, spleen, and adrenal glands, often resulting in infantile lethality.[41][42]In contrast, NCEH1, encoded by the NCEH1 gene (also known as AADACL1 or KIAA1363), functions as a neutral pH-active hydrolase with an optimum around pH 7.2, localized primarily to the endoplasmic reticulum membrane (with its catalytic domain facing the lumen) and exhibiting some cytosolic association. This enzyme mobilizes CE stored in cytoplasmic lipid droplets, particularly in macrophages and steroidogenic tissues, facilitating the release of free cholesterol for reverse cholesterol transport or hormonal synthesis.[43][44][45] Unlike LAL, NCEH1 operates independently of endocytosis and is vital for post-lysosomal CE processing, contributing to the dynamic turnover of intracellular lipid stores during lipidhomeostasis.[46]Regulation of CE hydrolysis integrates with broader lipid metabolic pathways, particularly in macrophages where foam cell formation is regulated. For instance, peroxisome proliferator-activated receptor γ (PPARγ) and PPARα agonists have been shown to modulate hydrolase activities, with PPARα directly upregulating neutral CE hydrolase expression to enhance hydrolysis and cholesterol efflux.[47] Statins, by inhibiting HMG-CoA reductase and reducing overall cholesterol synthesis, indirectly limit substrate availability for CE formation and subsequent hydrolysis, though direct effects on hydrolase enzymes remain context-dependent. This enzymatic breakdown is integral to mobilizing stored CE for efflux, preventing excessive lipid retention in cells.[48]
Transfer mechanisms
Cholesteryl esters (CEs) are transferred between lipoprotein particles in plasma through non-hydrolytic mechanisms mediated primarily by specialized proteins, enabling the redistribution of neutral lipids without degradation. The cholesteryl ester transfer protein (CETP), a plasmaglycoprotein synthesized in the liver, facilitates the bidirectional exchange of CEs from high-density lipoprotein (HDL) to very low-density lipoprotein (VLDL) and low-density lipoprotein (LDL) in return for triglycerides (TGs) from those apoB-containing lipoproteins.[49] This neutral lipid exchange process contributes to the remodeling of lipoprotein cores, where CEs enrich the core of VLDL and LDL while TGs accumulate in HDL.[50] The CETP gene, located on chromosome 16q13.1, encodes this 493-amino-acid protein, which circulates at concentrations of approximately 1-2 μg/mL in human plasma.[51]CETP operates through a shuttle mechanism, where its tunnel-shaped structure—featuring a hydrophobic binding pocket approximately 60 Å long—binds CEs and TGs, allowing transfer down concentration gradients between lipoprotein particles.[52] The protein's boomerang-like conformation enables it to interact sequentially with donor and acceptor lipoproteins, promoting neutral lipid unloading into the acceptor core via transient pores or direct diffusion.[53] This process is inhibited by pharmaceutical agents such as anacetrapib, a potent CETP inhibitor that binds to the protein's lipid-binding site, blocking CE transfer and thereby elevating HDL cholesterol levels by up to 140% in clinical studies.[54] Polymorphisms in the CETP gene, such as the TaqIB variant (rs708272), are associated with reduced CETP activity and higher HDL cholesterol concentrations; for instance, the B2 allele correlates with a 3.1 mg/dL per-allele increase in HDL-C and a 24% lower risk of coronary events in population studies.[55]CETP was first identified in the early 1980s through in vitro assays demonstrating CE transfer activity in lipoprotein-deficient plasma, with key reports from 1980 attributing the phenomenon to a specific factor in human and rabbitplasma.[56] Its three-dimensional structure was first elucidated in 2010 using X-ray crystallography, revealing a neutral lipid-binding pocket that accommodates two CE molecules and supports the tunnel-mediated transfer model.[57]In addition to CETP, other proteins contribute to CE movement, albeit through distinct pathways. Phospholipid transfer protein (PLTP), another liver-derived plasma protein with partial sequence homology to CETP, primarily transfers phospholipids but also exhibits minor CE transfer activity, aiding in HDL remodeling and size regulation during postprandial lipolysis.[58] Scavenger receptor class B type I (SR-BI), a cell-surface receptor expressed on hepatocytes and steroidogenic tissues, mediates selective uptake of CEs from HDL into cells without internalization of the entire lipoprotein particle, facilitating reverse cholesterol transport to the liver.[59]
Clinical significance
In cardiovascular disease
Cholesteryl esters (CE) play a central role in the pathogenesis of atherosclerosis, a key process in cardiovascular disease (CVD). In the arterial wall, macrophages take up oxidized low-density lipoprotein (oxLDL), leading to excess free cholesterol accumulation. This cholesterol is then esterified by acyl-CoA:cholesterol acyltransferase 1 (ACAT1) into CE, which forms lipid droplets within the cells, transforming macrophages into foam cells—a hallmark of early atherosclerotic lesions.[60] This foam cell formation promotes plaque buildup by contributing to the inflammatory response and extracellular matrix deposition in the intima.[61]The cholesteryl ester transfer protein (CETP) exacerbates atherogenesis by facilitating the exchange of CE from high-density lipoprotein (HDL) to low-density lipoprotein (LDL) particles, thereby enriching atherogenic LDL with CE while depleting protective HDL.[62] Genetic variants in the CETP gene, such as the TaqIB polymorphism (B2 allele), are associated with reduced CETP activity, higher HDL levels, and a lower risk of coronary heart disease (CHD), with carriers showing up to a 15% reduction in composite ischemic CVD events compared to non-carriers.[63] This variant highlights CETP's pro-atherogenic influence, as lower CETP function correlates with decreased CVD susceptibility.[64]Therapeutic strategies targeting CE metabolism have focused on CETP and ACAT inhibition, though with mixed outcomes. CETP inhibitors like torcetrapib, tested in the 2006 ILLUMINATE trial, raised HDL but increased CVD events due to off-target effects such as elevated blood pressure, leading to its discontinuation.[65] Similarly, evacetrapib in the 2015 ACCELERATE trial failed to reduce major adverse cardiovascular events despite HDL elevation and LDL reduction, underscoring challenges in translating lipid changes to clinical benefit.[66] ACAT inhibitors, such as avasimibe in the 2006 ACTIVATE trial, did not slow coronary atherosclerosis progression and potentially promoted it, highlighting risks of disrupting CE storage in macrophages.[67]Epidemiologically, elevated plasma CE levels, particularly in LDL particles, correlate with increased CHD risk; for instance, higher cholesteryl ester transfer activity is linked to a 22% greater incidence of coronary events in cohort studies, independent of traditional risk factors.[68] In the Framingham Heart Study, related lipid profiles influenced by CETP variants further support this association, with dysregulated CE distribution contributing to a 20-30% heightened CVD risk in susceptible populations.[69]
In storage disorders
Cholesteryl ester storage disease (CESD) represents the milder, later-onset form of lysosomal acid lipase (LAL) deficiency, an autosomal recessive disorder caused by pathogenic variants in the LIPA gene that impair the enzyme's ability to hydrolyze cholesteryl esters and triglycerides in lysosomes.[70] Affected individuals typically present in childhood or adulthood with hepatomegaly due to lipid-laden macrophages in the liver, elevated serum cholesterol levels (hypercholesterolemia), and dyslipidemia, including low high-density lipoproteincholesterol.[71] The estimated prevalence of CESD is approximately 1 in 40,000 among individuals of Caucasian descent, though it is often underdiagnosed due to its variable and nonspecific symptoms.[72]In contrast, Wolman disease is the severe, infantile form of LAL deficiency, manifesting within weeks of birth and leading to rapid progression with massive accumulation of cholesteryl esters in visceral organs such as the adrenals, liver, and spleen.[73] Infants exhibit hepatosplenomegaly, failure to thrive, vomiting, diarrhea, and adrenal calcification, with nearly all cases proving fatal by age 1 year due to multiorgan failure.[74]Diagnosis of both CESD and Wolman disease relies on demonstrating deficient LAL enzyme activity through biochemical assays on peripheral blood leukocytes, fibroblasts, or dried blood spots, often complemented by genetic sequencing to identify biallelic LIPA variants.[74] The treatment of LAL deficiency involves enzyme replacement therapy with sebelipase alfa, a recombinant human LAL approved by the U.S. Food and Drug Administration in December 2015 for long-term use in patients of all ages, which reduces hepatic fat content, improves lipid profiles, and enhances growth in affected individuals.[75]Pathophysiologically, the accumulation of cholesteryl esters and triglycerides within lysosomes due to LAL deficiency disrupts lysosomal integrity and cellular autophagy, leading to foam cell formation and progressive damage primarily in visceral organs like the liver and spleen, rather than vascular tissues.[70] This lysosomal storage impairs organelle function and contributes to inflammation and fibrosis, underscoring the multisystemic impact of unchecked lipid buildup.[71]