Literature DB >> 35254599

A rare cause of hydrops fetalis in two Gaucher disease type 2 patients with a novel mutation.

Sebile Kılavuz1,2, Murat Basaranoglu3, Serdar Epcacan4, Derya Bako5, Arife Ozer6, Yasemin Nuran Donmez4, Emine Ipek Ceylan7, Ajlan Tukun8, Serdar Ceylaner9,10, Hadi Geylani11, Halise Neslihan Onenli Mungan12.   

Abstract

Gaucher disease type 2 is the most progressive and the rarest form of Gaucher disease, defined as the acute neuronopathic type. We presented two GD2 patients who died before three months of age due to severe septicemia, respiratory and liver failure. One was homozygous for a novel GBA variant c.590 T > A (p.197 K), and the second homozygous for the known GBA mutation c.1505G > A (p.R502H). Ichthyosis, hydrops fetalis, apnea, myoclonic seizures, and hepatosplenomegaly occurred in both patients, but hypertrophic cardiomyopathy was observed only in the second and unilateral cataract in the first patient. Due to the disease's early and rapid neurological progression, we did not administer ERT to our patients. It is strongly believed that early diagnosis is essential, and prenatal diagnosis makes genetic counselling possible for future pregnancies.
© 2022. The Author(s), under exclusive licence to Springer Science+Business Media, LLC, part of Springer Nature.

Entities:  

Keywords:  GBA; Gaucher disease; Ichthyosis; hydrops fetalis

Mesh:

Year:  2022        PMID: 35254599     DOI: 10.1007/s11011-022-00942-5

Source DB:  PubMed          Journal:  Metab Brain Dis        ISSN: 0885-7490            Impact factor:   3.584


  22 in total

Review 1.  Perinatal lethal Gaucher disease: a distinct phenotype along the neuronopathic continuum.

Authors:  Michael J Eblan; Ozlem Goker-Alpan; Ellen Sidransky
Journal:  Fetal Pediatr Pathol       Date:  2005 Jul-Oct       Impact factor: 0.958

2.  Four Gaucher disease type II patients with three novel mutations: a single centre experience from Turkey.

Authors:  Fatma Derya Bulut; Deniz Kör; Berna Şeker-Yılmaz; Özlem Hergüner; Serdar Ceylaner; Ferda Özkınay; Sebile Kılavuz; Neslihan Önenli-Mungan
Journal:  Metab Brain Dis       Date:  2018-04-14       Impact factor: 3.584

3.  A newborn case with perinatal-lethal Gaucher disease due to R463H homozygosity complicated by C677T homozygosity in the MTHFR gene.

Authors:  Arzu Akdag; Serife Suna Oğuz; Fatih Ezgü; Omer Erdeve; Nurdan Uraş; Uğur Dilmen
Journal:  J Pediatr Endocrinol Metab       Date:  2011       Impact factor: 1.634

4.  An update of Gaucher mutations distribution in the Ashkenazi Jewish population: prevalence and country of origin of the mutation R496H.

Authors:  Silvia Bronstein; Mazal Karpati; Leah Peleg
Journal:  Isr Med Assoc J       Date:  2014-11       Impact factor: 0.892

5.  Congenital ichthyosis in severe type II Gaucher disease with a homozygous null mutation.

Authors:  Sabine Haverkaemper; Thorsten Marquardt; Ingrid Hausser; Katharina Timme; Thomas Kuehn; Christoph Hertzberg; Rainer Rossi
Journal:  Neonatology       Date:  2011-04-01       Impact factor: 4.035

6.  Type 1 Gaucher disease: null and hypomorphic novel chitotriosidase mutations-implications for diagnosis and therapeutic monitoring.

Authors:  Marie E Grace; Manisha Balwani; Irina Nazarenko; Ainu Prakash-Cheng; Robert J Desnick
Journal:  Hum Mutat       Date:  2007-09       Impact factor: 4.878

7.  A model of neuronopathic Gaucher disease.

Authors:  P E Campbell; C M Harris; C M Harris; T Sirimanna; A Vellodi
Journal:  J Inherit Metab Dis       Date:  2003       Impact factor: 4.982

Review 8.  Mutation analysis and genotype/phenotype relationships of Gaucher disease patients in Spain.

Authors:  Pilar Alfonso; Sofía Aznarez; Manuel Giralt; Miguel Pocovi; Pilar Giraldo
Journal:  J Hum Genet       Date:  2007-04-11       Impact factor: 3.172

Review 9.  Diagnosing neuronopathic Gaucher disease: New considerations and challenges in assigning Gaucher phenotypes.

Authors:  Emily C Daykin; Emory Ryan; Ellen Sidransky
Journal:  Mol Genet Metab       Date:  2021-01-09       Impact factor: 4.797

10.  Perinatal-lethal Gaucher disease presenting as hydrops fetalis.

Authors:  Emira BenHamida; Imene Ayadi; Ines Ouertani; Maroua Chammem; Ahlem Bezzine; Riadh BenTmime; Leila Attia; Ridha Mrad; Zahra Marrakchi
Journal:  Pan Afr Med J       Date:  2015-06-10
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