Literature DB >> 23753090

Cobalamin C defect presenting with isolated pulmonary hypertension.

Francesca G Iodice1, Luca Di Chiara, Sara Boenzi, Chiara Aiello, Lidia Monti, Paola Cogo, Carlo Dionisi-Vici.   

Abstract

Cobalamin C (cblC) defect is the most common inborn error of vitamin B12 metabolism. Clinical features vary as does the severity of the disease. In most cases, the clinical symptoms of cblC defect tend to appear during infancy or early childhood as a multisystem disease with severe neurologic, ocular, hematologic, renal, and gastrointestinal signs. The neurologic findings are common and include hypotonia, developmental delay, microcephaly, seizures hydrocephalus, and brain MRI abnormalities. We report a case of a young boy with cblC defect, who did not undergo newborn screening, presenting at the age of 2 years with isolated pulmonary hypertension as the leading symptom. This novel way of presentation of cblC defect enlarges the spectrum of inherited diseases that must be considered in the differential diagnosis of pulmonary hypertension.

Entities:  

Keywords:  cobalamin C defect; pulmonary hypertension

Mesh:

Substances:

Year:  2013        PMID: 23753090     DOI: 10.1542/peds.2012-1945

Source DB:  PubMed          Journal:  Pediatrics        ISSN: 0031-4005            Impact factor:   7.124


  13 in total

1.  Reversible pulmonary arterial hypertension in cobalamin-dependent cobalamin C disease due to a novel mutation in the MMACHC gene.

Authors:  Mehmet Gündüz; Filiz Ekici; Eda Özaydın; Serdar Ceylaner; Belen Perez
Journal:  Eur J Pediatr       Date:  2014-05-24       Impact factor: 3.183

2.  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

Review 3.  Versatile enzymology and heterogeneous phenotypes in cobalamin complementation type C disease.

Authors:  Anna J Esser; Srijan Mukherjee; Ilia A Dereven'kov; Sergei V Makarov; Donald W Jacobsen; Ute Spiekerkoetter; Luciana Hannibal
Journal:  iScience       Date:  2022-08-18

4.  Clinical presentation and outcome in a series of 88 patients with the cblC defect.

Authors:  Sabine Fischer; Martina Huemer; Matthias Baumgartner; Federica Deodato; Diana Ballhausen; Avihu Boneh; Alberto B Burlina; Roberto Cerone; Paula Garcia; Gülden Gökçay; Stephanie Grünewald; Johannes Häberle; Jaak Jaeken; David Ketteridge; Martin Lindner; Hanna Mandel; Diego Martinelli; Esmeralda G Martins; Karl O Schwab; Sarah C Gruenert; Bernd C Schwahn; László Sztriha; Maren Tomaske; Friedrich Trefz; Laura Vilarinho; David S Rosenblatt; Brian Fowler; Carlo Dionisi-Vici
Journal:  J Inherit Metab Dis       Date:  2014-03-06       Impact factor: 4.982

5.  A Highly Diverse Portrait: Heterogeneity of Neuropsychological Profiles in cblC Defect.

Authors:  Jenny Bellerose; Mathilde Neugnot-Cerioli; Karine Bédard; Catherine Brunel-Guitton; Grant A Mitchell; Luis H Ospina; Miriam H Beauchamp
Journal:  JIMD Rep       Date:  2015-11-26

Review 6.  Guidelines for diagnosis and management of the cobalamin-related remethylation disorders cblC, cblD, cblE, cblF, cblG, cblJ and MTHFR deficiency.

Authors:  Martina Huemer; Daria Diodato; Bernd Schwahn; Manuel Schiff; Anabela Bandeira; Jean-Francois Benoist; Alberto Burlina; Roberto Cerone; Maria L Couce; Angeles Garcia-Cazorla; Giancarlo la Marca; Elisabetta Pasquini; Laura Vilarinho; James D Weisfeld-Adams; Viktor Kožich; Henk Blom; Matthias R Baumgartner; Carlo Dionisi-Vici
Journal:  J Inherit Metab Dis       Date:  2016-11-30       Impact factor: 4.982

7.  Combined methylmalonic acidemia and homocysteinemia presenting predominantly with late-onset diffuse lung disease: a case series of four patients.

Authors:  Jinrong Liu; Yun Peng; Nan Zhou; Xiaorong Liu; Qun Meng; Hui Xu; Shunying Zhao
Journal:  Orphanet J Rare Dis       Date:  2017-03-21       Impact factor: 4.123

Review 8.  Biomarkers for pediatric pulmonary arterial hypertension - a call to collaborate.

Authors:  Kelley L Colvin; Melanie J Dufva; Ryan P Delaney; D Dunbar Ivy; Kurt R Stenmark; Michael E Yeager
Journal:  Front Pediatr       Date:  2014-02-03       Impact factor: 3.418

Review 9.  Renal thrombotic microangiopathy in patients with cblC defect: review of an under-recognized entity.

Authors:  Bodo B Beck; FrancJan van Spronsen; Arjan Diepstra; Rolf M F Berger; Martin Kömhoff
Journal:  Pediatr Nephrol       Date:  2016-06-11       Impact factor: 3.714

10.  Favorable course of previously undiagnosed Methylmalonic Aciduria with Homocystinuria (cblC type) presenting with pulmonary hypertension and aHUS in a young child: a case report.

Authors:  Luciano De Simone; Laura Capirchio; Rosa Maria Roperto; Paola Romagnani; Michele Sacchini; Maria Alice Donati; Maurizio de Martino
Journal:  Ital J Pediatr       Date:  2018-08-13       Impact factor: 2.638

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