Treatment options for lactic acidosis and metabolic crisis in children with mitochondrial disease

被引:17
作者
Danhauser, Katharina [1 ]
Smeitink, Jan A. M. [2 ]
Freisinger, Peter [3 ]
Sperl, Wolfgang [4 ]
Sabir, Hemmen [1 ]
Hadzik, Berit [1 ]
Mayatepek, Ertan [1 ]
Morava, Eva [5 ]
Distelmaier, Felix [1 ]
机构
[1] Univ Dusseldorf, Univ Childrens Hosp, Dept Gen Pediat Neonatol & Pediat Cardiol, D-40225 Dusseldorf, Germany
[2] Radboud Univ Nijmegen, Med Ctr, Dept Pediat, Nijmegen Ctr Mitochondrial Disorders, NL-6525 ED Nijmegen, Netherlands
[3] Klinikum Reutlingen, Inherited Metab Dis Ctr, Dept Pediat, Reutlingen, Germany
[4] Paracelsus Med Univ, Dept Pediat, Salzburg, Austria
[5] Tulane Univ, Sch Med, Hayward Genet Ctr, New Orleans, LA 70118 USA
关键词
COMPLEX-I DEFICIENCY; PYRUVATE-CARBOXYLASE DEFICIENCY; RENAL REPLACEMENT THERAPY; SODIUM-BICARBONATE; OXIDATIVE-PHOSPHORYLATION; YOUNG-CHILDREN; INBORN-ERRORS; DICHLOROACETATE; ACIDEMIA; CITRATE;
D O I
10.1007/s10545-014-9796-2
中图分类号
R5 [内科学];
学科分类号
1002 ; 100201 ;
摘要
The mitochondrial pyruvate oxidation route is a tightly regulated process, which is essential for aerobic cellular energy production. Disruption of this pathway may lead to severe neurometabolic disorders with onset in early childhood. A frequent finding in these patients is acute and chronic lactic acidemia, which is caused by increased conversion of pyruvate via the enzyme lactate dehydrogenase. Under stable clinical conditions, this process may remain well compensated and does not require specific therapy. However, especially in situations with altered energy demands, such as febrile infections or longer periods of fasting, children with mitochondrial disorders have a high risk of metabolic decompensation with exacerbation of hyperlactatemia and severe metabolic acidosis. Unfortunately, no controlled studies regarding therapy of this critical condition are available and clinical outcome is often unfavorable. Therefore, the aim of this review was to formulate expert-based suggestions for treatment of these patients, including dietary recommendations, buffering strategies and specific drug therapy. However, it is important to keep in mind that a specific therapy for the underlying metabolic cause in children with mitochondrial diseases is usually not available and symptomatic therapy especially of severe lactic acidosis has its ethical limitations.
引用
收藏
页码:467 / 475
页数:9
相关论文
共 84 条
  • [1] Long-term safety of dichloroacetate in congenital lactic acidosis
    Abdelmalak, Monica
    Lew, Alicia
    Ramezani, Ryan
    Shroads, Albert L.
    Coats, Bonnie S.
    Langaee, Taimour
    Shankar, Meena N.
    Neiberger, Richard E.
    Subramony, S. H.
    Stacpoole, Peter W.
    [J]. MOLECULAR GENETICS AND METABOLISM, 2013, 109 (02) : 139 - 143
  • [2] Ahmad A, 1999, AM J MED GENET, V87, P331, DOI 10.1002/(SICI)1096-8628(19991203)87:4<331::AID-AJMG10>3.0.CO
  • [3] 2-K
  • [4] Andrade BVB, 2007, J PEDIAT-BRAZIL, V83, pS11, DOI [10.2223/JPED.1616, 10.1590/S0021-75572007000300003]
  • [5] DIALYSIS DISEQUILIBRIUM SYNDROME - CURRENT CONCEPTS ON PATHOGENESIS AND PREVENTION
    ARIEFF, AI
    [J]. KIDNEY INTERNATIONAL, 1994, 45 (03) : 629 - 635
  • [6] Dichloroacetate treatment for severe refractory metabolic acidosis during neonatal sepsis
    Arnon, S
    Litmanovits, I
    Regev, R
    Elpeleg, O
    Dolfin, T
    [J]. PEDIATRIC INFECTIOUS DISEASE JOURNAL, 2001, 20 (02) : 218 - 219
  • [7] Sodium bicarbonate: Basically useless therapy
    Aschner, Judy L.
    Poland, Ronald L.
    [J]. PEDIATRICS, 2008, 122 (04) : 831 - 835
  • [8] A guide to diagnosis and treatment of Leigh syndrome
    Baertling, Fabian
    Rodenburg, Richard J.
    Schaper, Joerg
    Smeitink, Jan A.
    Koopman, Werner J. H.
    Mayatepek, Ertan
    Morava, Eva
    Distelmaier, Felix
    [J]. JOURNAL OF NEUROLOGY NEUROSURGERY AND PSYCHIATRY, 2014, 85 (03) : 257 - 265
  • [9] Acute Renal Replacement Therapy in Pediatrics
    Basu, Rajit K.
    Wheeler, Derek S.
    Goldstein, Stuart
    Doughty, Lesley
    [J]. INTERNATIONAL JOURNAL OF NEPHROLOGY, 2011, 2011
  • [10] Therapeutic potential of dichloroacetate for pyruvate dehydrogenase complex deficiency
    Berendzen, Kristen
    Theriaque, Douglas W.
    Shuster, Jonathan
    Stacpoole, Peter W.
    [J]. MITOCHONDRION, 2006, 6 (03) : 126 - 135