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Sulfide quinone oxidoreductase deficiency(SQORD)

MedGen UID:
1780603
Concept ID:
C5543168
Disease or Syndrome
Synonym: SULFIDE:QUINONE OXIDOREDUCTASE DEFICIENCY
 
Gene (location): SQOR (15q21.1)
 
Monarch Initiative: MONDO:0030982
OMIM®: 619221

Definition

Sulfide:quinone oxidoreductase-deficiency (SQORD) is characterized by a variable phenotype ranging from no clinical symptoms to episodes of encephalopathy and Leigh syndrome-like (see 256000) brain lesions, with acute symptoms triggered by infections and fasting. Other features may include lactic acidosis and decreased mitochondrial respiratory chain complex IV activity in tissues. Most affected individuals are asymptomatic. Patients with encephalopathy may recover or die in childhood (Friederich et al., 2020). [from OMIM]

Clinical features

From HPO
Coma
MedGen UID:
1054
Concept ID:
C0009421
Disease or Syndrome
The complete absence of wakefulness and consciousness, which is evident through a lack of response to any form of external stimuli.
Migraine
MedGen UID:
57451
Concept ID:
C0149931
Disease or Syndrome
Migraine is a chronic neurological disorder characterized by episodic attacks of headache and associated symptoms.
Tonic seizure
MedGen UID:
82855
Concept ID:
C0270844
Disease or Syndrome
A tonic seizure is a type of motor seizure characterized by unilateral or bilateral limb stiffening or elevation, often with neck stiffening.
Acute encephalopathy
MedGen UID:
224930
Concept ID:
C1306587
Disease or Syndrome
A life-threatening disorder characterized by delirium, seizures, and neuromuscular changes.
Elevated brain lactate level by MRS
MedGen UID:
868368
Concept ID:
C4022762
Finding
An increase in the level of lactate in the brain identified by magnetic resonance spectroscopy (MRS).
Lactic acidosis
MedGen UID:
1717
Concept ID:
C0001125
Disease or Syndrome
An abnormal buildup of lactic acid in the body, leading to acidification of the blood and other bodily fluids.
Elevated circulating creatine kinase concentration
MedGen UID:
69128
Concept ID:
C0241005
Finding
An elevation of the level of the enzyme creatine kinase (also known as creatine phosphokinase (CK; EC 2.7.3.2) in the blood. CK levels can be elevated in a number of clinical disorders such as myocardial infarction, rhabdomyolysis, and muscular dystrophy.

Recent clinical studies

Etiology

González-García P, Hidalgo-Gutiérrez A, Mascaraque C, Barriocanal-Casado E, Bakkali M, Ziosi M, Abdihankyzy UB, Sánchez-Hernández S, Escames G, Prokisch H, Martín F, Quinzii CM, López LC
Hum Mol Genet 2020 Nov 25;29(19):3296-3311. doi: 10.1093/hmg/ddaa214. PMID: 32975579Free PMC Article
Friederich MW, Elias AF, Kuster A, Laugwitz L, Larson AA, Landry AP, Ellwood-Digel L, Mirsky DM, Dimmock D, Haven J, Jiang H, MacLean KN, Styren K, Schoof J, Goujon L, Lefrancois T, Friederich M, Coughlin CR 2nd, Banerjee R, Haack TB, Van Hove JLK
J Inherit Metab Dis 2020 Sep;43(5):1024-1036. Epub 2020 Apr 15 doi: 10.1002/jimd.12232. PMID: 32160317Free PMC Article

Diagnosis

Libiad M, Vitvitsky V, Bostelaar T, Bak DW, Lee HJ, Sakamoto N, Fearon E, Lyssiotis CA, Weerapana E, Banerjee R
J Biol Chem 2019 Aug 9;294(32):12077-12090. Epub 2019 Jun 18 doi: 10.1074/jbc.RA119.009442. PMID: 31213529Free PMC Article

Clinical prediction guides

Combi Z, Potor L, Nagy P, Sikura KÉ, Ditrói T, Jurányi EP, Galambos K, Szerafin T, Gergely P, Whiteman M, Torregrossa R, Ding Y, Beke L, Hendrik Z, Méhes G, Balla G, Balla J
Redox Biol 2023 Apr;60:102629. Epub 2023 Feb 8 doi: 10.1016/j.redox.2023.102629. PMID: 36780769Free PMC Article
Kožich V, Schwahn BC, Sokolová J, Křížková M, Ditroi T, Krijt J, Khalil Y, Křížek T, Vaculíková-Fantlová T, Stibůrková B, Mills P, Clayton P, Barvíková K, Blessing H, Sykut-Cegielska J, Dionisi-Vici C, Gasperini S, García-Cazorla Á, Haack TB, Honzík T, Ješina P, Kuster A, Laugwitz L, Martinelli D, Porta F, Santer R, Schwarz G, Nagy P
Redox Biol 2022 Dec;58:102517. Epub 2022 Oct 18 doi: 10.1016/j.redox.2022.102517. PMID: 36306676Free PMC Article
González-García P, Hidalgo-Gutiérrez A, Mascaraque C, Barriocanal-Casado E, Bakkali M, Ziosi M, Abdihankyzy UB, Sánchez-Hernández S, Escames G, Prokisch H, Martín F, Quinzii CM, López LC
Hum Mol Genet 2020 Nov 25;29(19):3296-3311. doi: 10.1093/hmg/ddaa214. PMID: 32975579Free PMC Article
Ziosi M, Di Meo I, Kleiner G, Gao XH, Barca E, Sanchez-Quintero MJ, Tadesse S, Jiang H, Qiao C, Rodenburg RJ, Scalais E, Schuelke M, Willard B, Hatzoglou M, Tiranti V, Quinzii CM
EMBO Mol Med 2017 Jan;9(1):96-111. doi: 10.15252/emmm.201606356. PMID: 27856618Free PMC Article
Palmfeldt J, Vang S, Stenbroen V, Pavlou E, Baycheva M, Buchal G, Monavari AA, Augoustides-Savvopoulou P, Mandel H, Gregersen N
J Proteome Res 2011 May 6;10(5):2389-96. Epub 2011 Mar 28 doi: 10.1021/pr101218d. PMID: 21410200

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