Literature DB >> 24458587

Hydrops fetalis in a preterm newborn heterozygous for the c.4A>G SHOC2 mutation.

Giancarlo Gargano1, Isotta Guidotti, Eleonora Balestri, Federica Vagnarelli, Simonetta Rosato, Giuseppina Comitini, Anita Wischmeijer, Giovanni Battista La Sala, Lorenzo Iughetti, Viviana Cordeddu, Cesare Rossi, Marco Tartaglia, Livia Garavelli.   

Abstract

Fetal hydrops is a condition resulting from interstitial fluid accumulation in fetal compartments secondary to increased capillary permeability and characterized by high rates of perinatal mortality and morbidity. Clinical features include skin edema, hydrothorax, pericardial effusion, ascites with or without polyhydramnios, and placental edema. While it may occur as associated feature in multiple disorders, it has been documented to recur in Noonan syndrome, the most common disorder among RASopathies, but also in cardiofaciocutaneous and Costello syndromes. Here, we report on the occurrence of severe hydrops in a newborn heterozygous for the invariant c.4A>G missense change in SHOC2 which underlies Noonan-like syndrome with loose anagen hair, documenting that it represents a clinically relevant complication in this condition, shared by RASopathies.
© 2014 Wiley Periodicals, Inc.

Entities:  

Keywords:  Noonan-like syndrome with loose anagen hair; RASopathies; SHOC2; fetal hydrops

Mesh:

Substances:

Year:  2014        PMID: 24458587     DOI: 10.1002/ajmg.a.36376

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  8 in total

Review 1.  The leucine-rich repeat signaling scaffolds Shoc2 and Erbin: cellular mechanism and role in disease.

Authors:  HyeIn Jang; Payton Stevens; Tianyan Gao; Emilia Galperin
Journal:  FEBS J       Date:  2020-07-06       Impact factor: 5.542

2.  Hematopoietic and neural crest defects in zebrafish shoc2 mutants: a novel vertebrate model for Noonan-like syndrome.

Authors:  HyeIn Jang; Erin Oakley; Marie Forbes-Osborne; Melissa V Kesler; Rebecca Norcross; Ann C Morris; Emilia Galperin
Journal:  Hum Mol Genet       Date:  2019-02-01       Impact factor: 6.150

Review 3.  A system-based approach to the genetic etiologies of non-immune hydrops fetalis.

Authors:  Anne H Mardy; Shilpa P Chetty; Mary E Norton; Teresa N Sparks
Journal:  Prenat Diagn       Date:  2019-06-26       Impact factor: 3.050

4.  ARAF recurrent mutation causes central conducting lymphatic anomaly treatable with a MEK inhibitor.

Authors:  Dong Li; Michael E March; Alvaro Gutierrez-Uzquiza; Charlly Kao; Christoph Seiler; Erin Pinto; Leticia S Matsuoka; Mark R Battig; Elizabeth J Bhoj; Tara L Wenger; Lifeng Tian; Nora Robinson; Tiancheng Wang; Yichuan Liu; Brant M Weinstein; Matthew Swift; Hyun Min Jung; Courtney N Kaminski; Rosetta Chiavacci; Jonathan A Perkins; Michael A Levine; Patrick M A Sleiman; Patricia J Hicks; Janet T Strausbaugh; Jean B Belasco; Yoav Dori; Hakon Hakonarson
Journal:  Nat Med       Date:  2019-07-01       Impact factor: 53.440

5.  The role of USP7 in the Shoc2-ERK1/2 signaling axis and Noonan-like syndrome with loose anagen hair.

Authors:  Patricia Wilson; Lina Abdelmoti; Rebecca Norcross; Eun Ryoung Jang; Malathy Palayam; Emilia Galperin
Journal:  J Cell Sci       Date:  2021-11-05       Impact factor: 5.285

Review 6.  Lymphatic Abnormalities in Noonan Syndrome Spectrum Disorders: A Systematic Review.

Authors:  Julia Sleutjes; Lotte Kleimeier; Erika Leenders; Willemijn Klein; Jos Draaisma
Journal:  Mol Syndromol       Date:  2021-09-10

7.  Pathological MAPK activation-mediated lymphatic basement membrane disruption causes lymphangiectasia that is treatable with ravoxertinib.

Authors:  Harish P Janardhan; Karen Dresser; Lloyd Hutchinson; Chinmay M Trivedi
Journal:  JCI Insight       Date:  2022-09-08

8.  The function of Shoc2: A scaffold and beyond.

Authors:  Eun Ryoung Jang; Emilia Galperin
Journal:  Commun Integr Biol       Date:  2016-05-18
  8 in total

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