Risk of hyperammonemic coma in the puerperium: two cases of women with diagnosed and undiagnosed deficiency of urea cycle enzymes

被引:16
作者
Tihtonen, Kati [1 ]
Uotila, Jukka [1 ]
Lahde, Jyrki [2 ]
Salo, Matti [2 ]
Keskinen, Paivi [2 ]
机构
[1] Tampere Univ Hosp, Dept Obstet & Gynecol, Tampere 33521, Finland
[2] Tampere Univ Hosp, Dept Pediat, Tampere 33521, Finland
关键词
Carbamoylphosphate synthetase deficiency; hyperammonemia; ornithine transcarbamylase deficiency; postpartum coma; ORNITHINE TRANSCARBAMYLASE DEFICIENCY; SYNTHETASE-I DEFICIENCY; PREGNANCY; GENE;
D O I
10.3109/00016340903540646
中图分类号
R71 [妇产科学];
学科分类号
100211 ;
摘要
Deficiencies of urea cycle enzymes are rare metabolic disorders. Inadequate function of these enzymes may in worst cases lead to hyperammonemic encephalopathy and death. The danger of urea cycle enzyme deficiencies is that previously healthy adults with no prior medical history suggesting these deficiencies may suddenly develop life-threatening complications during prolonged catabolic situations such as delivery or surgery. Since most of the metabolic disorders are diagnosed during childhood, it may sometimes be difficult to bear in mind these rare diseases as a cause of unconsciousness in adulthood. However, early diagnosis and prompt initiation of ammonia-lowering treatment are essential for survival of these patients. We present two pregnant women with urea cycle disorders: one with a known deficiency and an uncomplicated outcome, and another with a previously undiagnosed disorder and life-threatening course of the postpartum period.
引用
收藏
页码:404 / 406
页数:3
相关论文
共 11 条
[1]  
ARN P, 1990, NEW ENGL J MED, V7, P1652
[2]  
Bale AE, 1999, PRENATAL DIAG, V19, P1052, DOI 10.1002/(SICI)1097-0223(199911)19:11<1052::AID-PD693>3.0.CO
[3]  
2-2
[4]  
BRUSILOW S, 1995, METABOLIC MOL BASES, P1205
[5]   Ornithine transcarbamylase deficiency in pregnancy [J].
Cordero, DR ;
Baker, J ;
Dorinzi, D ;
Toffle, R .
JOURNAL OF INHERITED METABOLIC DISEASE, 2005, 28 (02) :237-240
[6]   Carbamyl phosphate synthase deficiency: Diagnosed during pregnancy in a 41-year-old [J].
Eather, G. ;
Coman, D. ;
Lander, C. ;
McGill, J. .
JOURNAL OF CLINICAL NEUROSCIENCE, 2006, 13 (06) :702-706
[7]   Survival after treatment with phenylacetate and benzoate for urea-cycle disorders [J].
Enns, Gregory M. ;
Berry, Susan A. ;
Berry, Gerard T. ;
Rhead, William J. ;
Brusilow, Saul W. ;
Hamosh, Ada .
NEW ENGLAND JOURNAL OF MEDICINE, 2007, 356 (22) :2282-2292
[8]  
HATA A, 1989, AM J HUM GENET, V45, P123
[9]  
Schimanski U, 1996, HEPATOLOGY, V24, P1413, DOI 10.1002/hep.510240618
[10]   CARBAMYL-PHOSPHATE SYNTHETASE-I DEFICIENCY WITH NO DETECTABLE MESSENGER-RNA ACTIVITY [J].
SUZUKI, Y ;
MATSUSHIMA, A ;
OHTAKE, A ;
MORI, M ;
TATIBANA, M ;
ORII, T .
EUROPEAN JOURNAL OF PEDIATRICS, 1986, 145 (05) :406-408