Liver and the defects of cholesterol and bile acids biosynthesis: Rare disorders many diagnostic pitfalls

被引:9
作者
Corso, Gaetano [1 ]
Dello Russo, Antonio [2 ]
Gelzo, Monica [2 ]
机构
[1] Univ Foggia, Dept Clin & Expt Med, I-71122 Foggia, Italy
[2] Univ Naples Federico II, Dept Mol Med & Med Biotechnol, Via Pansini 5, I-80131 Naples, Italy
关键词
Cholesterol; Bile acids; liver metabolism; gas chromatography coupled to mass spectrometry; liquid chromatography coupled to tandem mass spectrometry; LEMLI-OPITZ SYNDROME; DELTA(4)-3-OXOSTEROID 5-BETA-REDUCTASE DEFICIENCY; OXYSTEROL 7-ALPHA-HYDROXYLASE GENE; ABNORMAL STEROL-METABOLISM; ANTLEY-BIXLER-SYNDROME; CEREBROTENDINOUS XANTHOMATOSIS; INBORN ERROR; CHENODEOXYCHOLIC ACID; DENSITY-LIPOPROTEIN; 3-BETA-HYDROXY-DELTA(5)-C-27-STEROID OXIDOREDUCTASE;
D O I
10.3748/wjg.v23.i29.5257
中图分类号
R57 [消化系及腹部疾病];
学科分类号
摘要
In recent decades, biotechnology produced a growth of knowledge on the causes and mechanisms of metabolic diseases that have formed the basis for their study, diagnosis and treatment. Unfortunately, it is well known that the clinical features of metabolic diseases can manifest themselves with very different characteristics and escape early detection. Also, it is well known that the prognosis of many metabolic diseases is excellent if diagnosed and treated early. In this editorial we briefly summarized two groups of inherited metabolic diseases, the defects of cholesterol biosynthesis and those of bile acids. Both groups show variable clinical manifestations but some clinical signs and symptoms are common in both the defects of cholesterol and bile acids. The differential diagnosis can be made analyzing sterol profiles in blood and/or bile acids in blood and urine by chromatographic techniques (GC-MS and LC-MS/MS). Several defects of both biosynthetic pathways are treatable so early diagnosis is crucial. Unfortunately their diagnosis is made too late, due either to the clinical heterogeneity of the syndromes (severe, mild and very mild) that to the scarcity of scientific dissemination of these rare diseases. Therefore, the delay in diagnosis leads the patient to the medical observation when the disease has produced irreversible damages to the body. Here, we highlighted simple clinical and laboratory descriptions that can potentially make you to suspect a defect in cholesterol biosynthesis and/or bile acids, as well, we suggest appropriate request of the laboratory tests that along with common clinical features can help to diagnose these defects.
引用
收藏
页码:5257 / 5265
页数:9
相关论文
共 91 条
[1]   Conradi-Hunermann-Happle syndrome in males vs. MEND syndrome (male EBP disorder with neurological defects) [J].
Arnold, A. W. ;
Bruckner-Tuderman, L. ;
Has, C. ;
Happle, R. .
BRITISH JOURNAL OF DERMATOLOGY, 2012, 166 (06) :1309-1313
[2]  
BATTA AK, 1985, J LIPID RES, V26, P690
[3]   White matter lesions in spastic paraplegia with mutations in SPG5/CYP7B1 [J].
Biancheri, Roberta ;
Ciccolella, Marianna ;
Rossi, Andrea ;
Tessa, Alessandra ;
Cassandrini, Denise ;
Minetti, Carlo ;
Santorelli, Filippo M. .
NEUROMUSCULAR DISORDERS, 2009, 19 (01) :62-65
[4]   Cerebrotendinous xanthomatosis [J].
Bjorkhem, Ingemar .
CURRENT OPINION IN LIPIDOLOGY, 2013, 24 (04) :283-287
[5]   Lathosterolosis, a novel multiple-malformation/mental retardation syndrome due to deficiency of 3β-hydroxysteroid-Δ5-desaturase [J].
Brunetti-Pierri, N ;
Corso, G ;
Rossi, M ;
Ferrari, P ;
Balli, F ;
Rivasi, F ;
Annunziata, I ;
Ballabio, A ;
Dello Russo, A ;
Andria, G ;
Parenti, G .
AMERICAN JOURNAL OF HUMAN GENETICS, 2002, 71 (04) :952-958
[6]  
CALI JJ, 1991, J BIOL CHEM, V266, P7779
[7]   Liver Transplantation in Defects of Cholesterol Biosynthesis: The Case of Lathosterolosis [J].
Calvo, P. L. ;
Brunati, A. ;
Spada, M. ;
Romagnoli, R. ;
Corso, G. ;
Parenti, G. ;
Rossi, M. ;
Baldi, M. ;
Carbonaro, G. ;
David, E. ;
Pucci, A. ;
Amoroso, A. ;
Salizzoni, M. .
AMERICAN JOURNAL OF TRANSPLANTATION, 2014, 14 (04) :960-965
[8]   Complex inheritance of familial hypercholanemia with associated mutations in TJP2 and BAAT [J].
Carlton, VEH ;
Harris, BZ ;
Puffenberger, EG ;
Batta, AK ;
Knisely, AS ;
Robinson, DL ;
Strauss, KA ;
Schneider, BL ;
Lim, WA ;
Salen, G ;
Morton, DH ;
Bull, LN .
NATURE GENETICS, 2003, 34 (01) :91-96
[9]   Molecular genetics of 3β-hydroxy-Δ5-C27-steroid oxidoreductase deficiency in 16 patients with loss of bile acid synthesis and liver disease [J].
Cheng, JB ;
Jacquemin, E ;
Gerhardt, M ;
Nazer, H ;
Cresteil, D ;
Heubi, JE ;
Setchell, KDR ;
Russell, DW .
JOURNAL OF CLINICAL ENDOCRINOLOGY & METABOLISM, 2003, 88 (04) :1833-1841
[10]   Bile acid-CoA ligase deficiency-a new inborn error of bile acid metabolism [J].
Chong, Catherine P. K. ;
Mills, Philippa B. ;
McClean, Patricia ;
Gissen, Paul ;
Bruce, Christopher ;
Stahlschmidt, Jens ;
Knisely, A. S. ;
Clayton, Peter T. .
JOURNAL OF INHERITED METABOLIC DISEASE, 2012, 35 (03) :521-530