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Propionyl-CoA carboxylase deficiency

MedGen UID:
1638582
Concept ID:
C4551877
Finding
Synonyms: Deficiency of propionyl-CoA carboxylase; PCC deficiency
SNOMED CT: Deficiency of propionyl-CoA carboxylase (124718009); Deficiency of propionyl-coenzyme A carboxylase (124718009)
 
HPO: HP:0003353
OMIM®: 606054

Definition

Propionic acidemia is an autosomal recessive metabolic disorder caused by defective functioning in the mitochondrial enzyme propionyl CoA carboxylase (PCC), resulting in the accumulation of propionic acid metabolites, and dysfunction in the respiratory chain and urea cycle pathways. The disorder is clinically heterogeneous. A neonatal-onset form is characterized by poor feeding, vomiting, and fatigue in the first days of life in a previously healthy infant, and if untreated, may be followed by lethargy, seizures, coma, and death. The neonatal form is frequently accompanied by metabolic acidosis with anion gap, ketonuria, hypoglycemia, hyperammonemia, and cytopenia. A late-onset form in older children and adults has a milder phenotype, is less common, and may present with developmental regression, chronic vomiting, protein intolerance, failure to thrive, hypotonia, and occasionally basal ganglia infarction, which may result in dystonia and choreoathetosis, and cardiomyopathy. Metabolically unstable individuals can have an acute decompensation that resembles the neonatal presentation, often precipitated by a catabolic stress such as infection, injury, or surgery, or an excessive intake of intact (i.e., complete, dietary, or natural) protein. Long-term manifestations of neonatal and late onset of propionic acidemia can include growth impairment, intellectual disability, seizures, basal ganglia lesions, pancreatitis, and cardiomyopathy. Other less common manifestations include optic atrophy, hearing loss, premature ovarian insufficiency, and chronic renal failure (summary by Jurecki et al., 2019). [from OMIM]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVPropionyl-CoA carboxylase deficiency

Conditions with this feature

Propionic acidemia
MedGen UID:
75694
Concept ID:
C0268579
Disease or Syndrome
The spectrum of propionic acidemia (PA) ranges from neonatal-onset to late-onset disease. Neonatal-onset PA, the most common form, is characterized by a healthy newborn with poor feeding and decreased arousal in the first few days of life, followed by progressive encephalopathy of unexplained origin. Without prompt diagnosis and management, this is followed by progressive encephalopathy manifesting as lethargy, seizures, or coma that can result in death. It is frequently accompanied by metabolic acidosis with anion gap, lactic acidosis, ketonuria, hypoglycemia, hyperammonemia, and cytopenias. Individuals with late-onset PA may remain asymptomatic and suffer a metabolic crisis under catabolic stress (e.g., illness, surgery, fasting) or may experience a more insidious onset with the development of multiorgan complications including vomiting, protein intolerance, failure to thrive, hypotonia, developmental delays or regression, movement disorders, or cardiomyopathy. Isolated cardiomyopathy can be observed on rare occasion in the absence of clinical metabolic decompensation or neurocognitive deficits. Manifestations of neonatal and late-onset PA over time can include growth impairment, intellectual disability, seizures, basal ganglia lesions, pancreatitis, and cardiomyopathy. Other rarely reported complications include optic atrophy, hearing loss, premature ovarian insufficiency, and chronic renal failure.
3-methylcrotonyl-CoA carboxylase 2 deficiency
MedGen UID:
347898
Concept ID:
C1859499
Disease or Syndrome
3-Methylcrotonylglycinuria is an autosomal recessive disorder of leucine catabolism. The clinical phenotype is highly variable, ranging from neonatal onset with severe neurologic involvement to asymptomatic adults. There is a characteristic organic aciduria with massive excretion of 3-hydroxyisovaleric acid and 3-methylcrotonylglycine, usually in combination with a severe secondary carnitine deficiency. MCC activity in extracts of cultured fibroblasts of patients is usually less than 2% of control (summary by Baumgartner et al., 2001). Also see 3-methylcrotonylglycinuria I (MCC1D; 210200), caused by mutation in the alpha subunit of 3-methylcrotonyl-CoA carboxylase (MCCC1; 609010).

Professional guidelines

PubMed

Abacan M, Boneh A
Mol Genet Metab 2013 Aug;109(4):397-401. Epub 2013 Jun 6 doi: 10.1016/j.ymgme.2013.05.018. PMID: 23791308
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J Inherit Metab Dis 1982;5(2):121-4. doi: 10.1007/BF01800005. PMID: 6820422

Recent clinical studies

Etiology

Loyer M, Leclerc D, Gravel RA
Hum Mol Genet 1995 Jun;4(6):1035-9. doi: 10.1093/hmg/4.6.1035. PMID: 7655456
Ohura T, Kraus JP, Rosenberg LE
Am J Hum Genet 1989 Jul;45(1):33-40. PMID: 2741949Free PMC Article
Kalousek F, Orsulak MD, Rosenberg LR
Am J Hum Genet 1983 May;35(3):409-20. PMID: 6859037Free PMC Article
McKeon C, Eanes RZ, Wolf B
Biochem Genet 1982 Feb;20(1-2):77-94. doi: 10.1007/BF00484937. PMID: 7092804
Gravel RA, Lam KF, Scully KJ, Hsia Y
Am J Hum Genet 1977 Jul;29(4):378-88. PMID: 195466Free PMC Article

Diagnosis

Irum S, Rehman A, Aamir M, Haroon ZH, Chaudhry N, Bibi A
J Coll Physicians Surg Pak 2022 Aug;32(8):S180-S182. doi: 10.29271/jcpsp.2022.Supp2.S180. PMID: 36210688
Wolf B, Raetz H
Clin Chim Acta 1983 May 9;130(1):25-30. doi: 10.1016/0009-8981(83)90255-3. PMID: 6851181
Bergstrøm T, Greter J, Levin AH, Steen G, Tryding N, Wass U
Scand J Clin Lab Invest 1981 Apr;41(2):117-26. doi: 10.3109/00365518109092023. PMID: 7313494
Przyrembel H, Bremer HJ, Duran M, Bruinvis L, Ketting D, Wadman SK, Baumgartner R, Irle U, Bachmann C
Eur J Pediatr 1979 Jan 18;130(1):1-14. doi: 10.1007/BF00441893. PMID: 759179
Wadlington WB, Kilroy A, Ando T, Sweetman L, Nyhan WL
J Pediatr 1975 May;86(5):707-12. doi: 10.1016/s0022-3476(75)80354-4. PMID: 1133651

Therapy

Scholl-Bürgi S, Sass JO, Heinz-Erian P, Amann E, Haberlandt E, Albrecht U, Ertl C, Sigl SB, Lagler F, Rostasy K, Karall D
Amino Acids 2010 May;38(5):1473-81. Epub 2009 Oct 1 doi: 10.1007/s00726-009-0356-2. PMID: 19795187
Wolf B, Raetz H
Clin Chim Acta 1983 May 9;130(1):25-30. doi: 10.1016/0009-8981(83)90255-3. PMID: 6851181
Bergstrøm T, Greter J, Levin AH, Steen G, Tryding N, Wass U
Scand J Clin Lab Invest 1981 Apr;41(2):117-26. doi: 10.3109/00365518109092023. PMID: 7313494
Wolf B
J Pediatr 1980 Dec;97(6):964-6. doi: 10.1016/s0022-3476(80)80437-9. PMID: 7441430
Sweetman L, Bates SP, Hull D, Nyhan WL
Pediatr Res 1977 Nov;11(11):1144-7. doi: 10.1203/00006450-197711000-00006. PMID: 917614

Prognosis

Pérez-Cerdá C, Merinero B, Martí M, Cabrera JC, Peña L, García MJ, Gangoiti J, Sanz P, Rodríguez-Pombo P, Hoenicka J, Richard E, Muro S, Ugarte M
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Clinical prediction guides

Scholl-Bürgi S, Sass JO, Heinz-Erian P, Amann E, Haberlandt E, Albrecht U, Ertl C, Sigl SB, Lagler F, Rostasy K, Karall D
Amino Acids 2010 May;38(5):1473-81. Epub 2009 Oct 1 doi: 10.1007/s00726-009-0356-2. PMID: 19795187
Ohura T, Kraus JP, Rosenberg LE
Am J Hum Genet 1989 Jul;45(1):33-40. PMID: 2741949Free PMC Article
McKeon C, Eanes RZ, Wolf B
Biochem Genet 1982 Feb;20(1-2):77-94. doi: 10.1007/BF00484937. PMID: 7092804
Sweetman L, Weyler W, Nyhan WL, de Céspedes C, Loria AR, Estrada Y
Biomed Mass Spectrom 1978 Mar;5(3):198-207. doi: 10.1002/bms.1200050307. PMID: 630060
Duran M, Gompertz D, Bruinvis L, Ketting D, Wadman SK
Clin Chim Acta 1978 Jan 2;82(1-2):93-9. doi: 10.1016/0009-8981(78)90030-x. PMID: 618687

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