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Homocarnosinosis

MedGen UID:
75703
Concept ID:
C0268632
Disease or Syndrome
Synonym: Homocarnosinase deficiency
 
Monarch Initiative: MONDO:0009351
OMIM®: 236130
Orphanet: ORPHA2168

Definition

Homocarnosinosis, an elevation of homocarnosine, is a biochemical aberration of unknown significance. Only one such family has been reported (Sjaastad et al., 2018). Homocarnosinosis was previously thought to be a disorder characterized by marked elevation of homocarnosine in the cerebrospinal fluid along with spastic paraplegia, impaired intellectual development, and retinal pigmentation based on the report of one Norwegian family reported by Sjaastad et al. (1976). Sjaastad et al. (2018) performed genetic analysis postmortem in this family and identified a homozygous mutation in the SPG11 gene (610844). A reevaluation of the clinical symptoms and findings in the family correlated with spastic paraplegia-11 (SPG11; 604360). A study of other patients with SPG11 did not find elevated levels of homocarnosine. [from OMIM]

Clinical features

From HPO
Carnosinuria
MedGen UID:
758781
Concept ID:
C3495558
Finding
An increased concentration of carnosine in the urine.
Spastic paraplegia
MedGen UID:
20882
Concept ID:
C0037772
Disease or Syndrome
Spasticity and weakness of the leg and hip muscles.
Intellectual disability
MedGen UID:
811461
Concept ID:
C3714756
Mental or Behavioral Dysfunction
Intellectual disability, previously referred to as mental retardation, is characterized by subnormal intellectual functioning that occurs during the developmental period. It is defined by an IQ score below 70.
Abnormality of skin pigmentation
MedGen UID:
224697
Concept ID:
C1260926
Finding
An abnormality of the pigmentation of the skin.
Abnormality of retinal pigmentation
MedGen UID:
350681
Concept ID:
C1862475
Finding

Recent clinical studies

Etiology

Sjaastad O, Blau N, Rydning SL, Peters V, Rødningen O, Stray-Pedersen A, Krossnes B, Tallaksen C, Koht J
Acta Neurol Scand 2018 Sep;138(3):245-250. Epub 2018 May 6 doi: 10.1111/ane.12949. PMID: 29732542
Peters V, Zschocke J, Schmitt CP
J Inherit Metab Dis 2018 Jan;41(1):39-47. Epub 2017 Oct 13 doi: 10.1007/s10545-017-0099-2. PMID: 29027595

Diagnosis

Sjaastad O, Blau N, Rydning SL, Peters V, Rødningen O, Stray-Pedersen A, Krossnes B, Tallaksen C, Koht J
Acta Neurol Scand 2018 Sep;138(3):245-250. Epub 2018 May 6 doi: 10.1111/ane.12949. PMID: 29732542
Peters V, Zschocke J, Schmitt CP
J Inherit Metab Dis 2018 Jan;41(1):39-47. Epub 2017 Oct 13 doi: 10.1007/s10545-017-0099-2. PMID: 29027595
Jansen EE, Gibson KM, Shigematsu Y, Jakobs C, Verhoeven NM
J Chromatogr B Analyt Technol Biomed Life Sci 2006 Jan 18;830(2):196-200. Epub 2005 Nov 22 doi: 10.1016/j.jchromb.2005.10.053. PMID: 16309980
Pearl PL, Gibson KM
Curr Opin Neurol 2004 Apr;17(2):107-13. doi: 10.1097/00019052-200404000-00005. PMID: 15021235
Jakobs C, Jaeken J, Gibson KM
J Inherit Metab Dis 1993;16(4):704-15. doi: 10.1007/BF00711902. PMID: 8412016

Therapy

Lunde HA, Gjessing LR, Sjaastad O
Neurochem Res 1986 Jun;11(6):825-38. doi: 10.1007/BF00965207. PMID: 3736769

Prognosis

Sjaastad O, Blau N, Rydning SL, Peters V, Rødningen O, Stray-Pedersen A, Krossnes B, Tallaksen C, Koht J
Acta Neurol Scand 2018 Sep;138(3):245-250. Epub 2018 May 6 doi: 10.1111/ane.12949. PMID: 29732542
Peters V, Zschocke J, Schmitt CP
J Inherit Metab Dis 2018 Jan;41(1):39-47. Epub 2017 Oct 13 doi: 10.1007/s10545-017-0099-2. PMID: 29027595
Pearl PL, Gibson KM
Curr Opin Neurol 2004 Apr;17(2):107-13. doi: 10.1097/00019052-200404000-00005. PMID: 15021235

Clinical prediction guides

Caruso G, Caraci F, Jolivet RB
Prog Neurobiol 2019 Apr;175:35-53. Epub 2018 Dec 26 doi: 10.1016/j.pneurobio.2018.12.004. PMID: 30593839

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