Hemolytic Uremic Syndrome Due to Methylmalonic Acidemia and Homocystinuria in an Infant: A Case Report and Literature Review.

cobalamic C defect early-onset cblC deficit hemolytic uremic syndrome methylmalonic acidemia and homocystinuria thrombotic microangiopathy

Journal

Children (Basel, Switzerland)
ISSN: 2227-9067
Titre abrégé: Children (Basel)
Pays: Switzerland
ID NLM: 101648936

Informations de publication

Date de publication:
05 Feb 2021
Historique:
received: 28 12 2020
revised: 29 01 2021
accepted: 02 02 2021
entrez: 10 2 2021
pubmed: 11 2 2021
medline: 11 2 2021
Statut: epublish

Résumé

Methylmalonic acidemia and homocystinuria cobalamin C (cblC) type is the most common inborn error of the intracellular cobalamin metabolism, associated with multisystem involvement and high mortality rates, especially in the early-onset form of the disease. Hemolytic uremic syndrome (HUS) is a rare manifestation and needs to be distinguished from other causes of renal thrombotic microangiopathy. We describe a case of a 3-month-old infant, with failure to thrive, hypotonia and pallor, who developed HUS in the setting of cblC deficit, along with dilated cardiomyopathy, and presented delayed response to optic stimulation in visual evoked potentials, as well as enlarged bilateral subarachnoid spaces and delayed myelination in brain magnetic resonance imaging. Renal damage was reversed, while neurodevelopmental profile and eye contact improved after supplementation with parenteral hydroxycobalamin, oral folic acid, betaine and levocarnitine. Homozygous mutation of c.271dupA in the MMACHC gene was ultimately detected. In this report, we highlight the diagnostic challenges as well as the significance of early recognition and multidisciplinary management of this unusual condition. A brief review of published case reports of early-onset cblC deficit and related HUS is depicted, pointing out the initial clinical presentation, signs of renal damage and outcome, MMACHC gene type of mutations and accompanying extra-renal manifestations.

Identifiants

pubmed: 33562640
pii: children8020112
doi: 10.3390/children8020112
pmc: PMC7915400
pii:
doi:

Types de publication

Case Reports

Langues

eng

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Auteurs

Vasiliki Karava (V)

1st Department of Pediatrics, Aristotle University of Thessaloniki, Hippokratio General Hospital, 546 42 Thessaloniki, Greece.

Antonia Kondou (A)

1st Department of Pediatrics, Aristotle University of Thessaloniki, Hippokratio General Hospital, 546 42 Thessaloniki, Greece.

John Dotis (J)

1st Department of Pediatrics, Aristotle University of Thessaloniki, Hippokratio General Hospital, 546 42 Thessaloniki, Greece.

Georgia Sotiriou (G)

1st Department of Pediatrics, Aristotle University of Thessaloniki, Hippokratio General Hospital, 546 42 Thessaloniki, Greece.

Spyridon Gerou (S)

Gerou Analysis Medical S.A., Diagnostic-Research Clinics, 546 22 Thessaloniki, Greece.

Helen Michelakakis (H)

Department of Enzymology and Cellular Function, Institute of Child Health, 115 26 Athens, Greece.

Euthymia Vargiami (E)

1st Department of Pediatrics, Aristotle University of Thessaloniki, Hippokratio General Hospital, 546 42 Thessaloniki, Greece.

Marina Economou (M)

1st Department of Pediatrics, Aristotle University of Thessaloniki, Hippokratio General Hospital, 546 42 Thessaloniki, Greece.

Dimitrios Zafeiriou (D)

1st Department of Pediatrics, Aristotle University of Thessaloniki, Hippokratio General Hospital, 546 42 Thessaloniki, Greece.

Nikoleta Printza (N)

1st Department of Pediatrics, Aristotle University of Thessaloniki, Hippokratio General Hospital, 546 42 Thessaloniki, Greece.

Classifications MeSH