Literature DB >> 33562640

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

Vasiliki Karava1, Antonia Kondou1, John Dotis1, Georgia Sotiriou1, Spyridon Gerou2, Helen Michelakakis3, Euthymia Vargiami1, Marina Economou1, Dimitrios Zafeiriou1, Nikoleta Printza1.   

Abstract

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.

Entities:  

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

Year:  2021        PMID: 33562640      PMCID: PMC7915400          DOI: 10.3390/children8020112

Source DB:  PubMed          Journal:  Children (Basel)        ISSN: 2227-9067


  30 in total

1.  An infant with methylmalonic aciduria and homocystinuria (cblC) presenting with retinal haemorrhages and subdural haematoma mimicking non-accidental injury.

Authors:  Peter J Francis; David M Calver; Peter Barnfield; Charles Turner; R Neil Dalton; Mike P Champion
Journal:  Eur J Pediatr       Date:  2004-04-09       Impact factor: 3.183

2.  Cobalamin C defect associated with hemolytic-uremic syndrome.

Authors:  M T Geraghty; E J Perlman; L S Martin; S J Hayflick; J F Casella; D S Rosenblatt; D Valle
Journal:  J Pediatr       Date:  1992-06       Impact factor: 4.406

3.  Combined methylmalonic aciduria and homocystinuria (cblC): phenotype-genotype correlations and ethnic-specific observations.

Authors:  Chantal F Morel; Jordan P Lerner-Ellis; David S Rosenblatt
Journal:  Mol Genet Metab       Date:  2006-05-22       Impact factor: 4.797

4.  Cobalamin disorder CblC presenting with hemolytic uremic syndrome and pulmonary hypertension.

Authors:  Zuriñe Martínez de Compañón; Miriam Poblet-Puig; Griselda Vallès; Mireia Del Toro; Ramón Vilalta; Antonio Moreno; Joan Balcells
Journal:  Nefrologia (Engl Ed)       Date:  2017-06-10

Review 5.  Combined methylmalonic acidemia and homocystinuria, cblC type. II. Complications, pathophysiology, and outcomes.

Authors:  Nuria Carrillo-Carrasco; Charles P Venditti
Journal:  J Inherit Metab Dis       Date:  2011-07-12       Impact factor: 4.982

Review 6.  Combined methylmalonic acidemia and homocystinuria, cblC type. I. Clinical presentations, diagnosis and management.

Authors:  Nuria Carrillo-Carrasco; Randy J Chandler; Charles P Venditti
Journal:  J Inherit Metab Dis       Date:  2011-07-12       Impact factor: 4.982

7.  Congenital methylmalonic aciduria--homocystinuria with megaloblastic anemia: observations on response to hydroxocobalamin and on the effect of homocysteine and methionine on the deoxyuridine suppression test.

Authors:  R Carmel; A A Bedros; J W Mace; S I Goodman
Journal:  Blood       Date:  1980-04       Impact factor: 22.113

8.  Identification of the gene responsible for methylmalonic aciduria and homocystinuria, cblC type.

Authors:  Jordan P Lerner-Ellis; Jamie C Tirone; Peter D Pawelek; Carole Doré; Janet L Atkinson; David Watkins; Chantal F Morel; T Mary Fujiwara; Emily Moras; Angela R Hosack; Gail V Dunbar; Hana Antonicka; Vince Forgetta; C Melissa Dobson; Daniel Leclerc; Roy A Gravel; Eric A Shoubridge; James W Coulton; Pierre Lepage; Johanna M Rommens; Kenneth Morgan; David S Rosenblatt
Journal:  Nat Genet       Date:  2005-11-27       Impact factor: 38.330

9.  Adult-onset eculizumab-resistant hemolytic uremic syndrome associated with cobalamin C deficiency.

Authors:  Emilie Cornec-Le Gall; Yahsou Delmas; Loïc De Parscau; Laurent Doucet; Hélène Ogier; Jean-François Benoist; Véronique Fremeaux-Bacchi; Yannick Le Meur
Journal:  Am J Kidney Dis       Date:  2013-11-06       Impact factor: 8.860

10.  Spectrum of mutations in MMACHC, allelic expression, and evidence for genotype-phenotype correlations.

Authors:  Jordan P Lerner-Ellis; Natascia Anastasio; Junhui Liu; David Coelho; Terttu Suormala; Martin Stucki; Amanda D Loewy; Scott Gurd; Elin Grundberg; Chantal F Morel; David Watkins; Matthias R Baumgartner; Tomi Pastinen; David S Rosenblatt; Brian Fowler
Journal:  Hum Mutat       Date:  2009-07       Impact factor: 4.878

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  2 in total

1.  Clinical features and outcomes of patients with cblC type methylmalonic acidemia carrying gene c.609G>A mutation.

Authors:  Yue Yu; Shiying Ling; Ruixue Shuai; Wenjuan Qiu; Huiwen Zhang; Lili Liang; Wenjun Ji; Yuchao Liu; Xuefan Gu; Lianshu Han
Journal:  Zhejiang Da Xue Xue Bao Yi Xue Ban       Date:  2021-08-25

2.  Case Report: Lipoprotein Glomerulopathy Complicated by Atypical Hemolytic Uremic Syndrome.

Authors:  Lara Kollbrunner; Patricia Hirt-Minkowski; Javier Sanz; Elena Bresin; Thomas J Neuhaus; Helmut Hopfer; Andreas W Jehle
Journal:  Front Med (Lausanne)       Date:  2021-06-02
  2 in total

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