Literature DB >> 22945242

Huge aneurysm of the ascending aorta in a patient with adult-type Pompe's disease: histological findings mimicking fibrillinopathy.

Volkhard Goeber1, Yara Banz, Anja Kaeberich, Thierry Carrel.   

Abstract

Adult-type Pompe's disease (glycogen storage disease type II) has rarely been shown to present with dilatative arteriopathy, suggesting potential smooth muscle involvement in addition to lysosomal glycogen deposits usually restricted to skeletal muscle tissue. We report the case of a middle-aged man under enzyme replacement therapy presenting with an exceedingly large thoracic aortic aneurysm. Surprisingly, the histological work-up of resected aortic tissue revealed changes mimicking those observed in patients with classic connective tissue diseases. Enzyme replacement therapy, in addition to musculoskeletal and pulmonary treatment for patients with Pompe's disease, may prolong survival and lead to patients presenting with vascular alterations that may pose surgical and potential diagnostic challenges in the future.

Entities:  

Mesh:

Year:  2012        PMID: 22945242     DOI: 10.1093/ejcts/ezs489

Source DB:  PubMed          Journal:  Eur J Cardiothorac Surg        ISSN: 1010-7940            Impact factor:   4.191


  4 in total

1.  Huge ascending aortic aneurysm with an intraluminal thrombus in an embolic event-free patient.

Authors:  Vito Maurizio Parato; Edvin Prifti; Franco Pezzuoli; Benedetto Labanti; Arben Baboci
Journal:  Interv Med Appl Sci       Date:  2015-03-20

2.  Improved efficacy of a next-generation ERT in murine Pompe disease.

Authors:  Su Xu; Yi Lun; Michelle Frascella; Anadina Garcia; Rebecca Soska; Anju Nair; Abdul S Ponery; Adriane Schilling; Jessie Feng; Steven Tuske; Maria Cecilia Della Valle; José A Martina; Evelyn Ralston; Russell Gotschall; Kenneth J Valenzano; Rosa Puertollano; Hung V Do; Nina Raben; Richie Khanna
Journal:  JCI Insight       Date:  2019-03-07

3.  Aortopathies in mouse models of Pompe, Fabry and Mucopolysaccharidosis IIIB lysosomal storage diseases.

Authors:  Maria Paola Belfiore; Francesca Iacobellis; Emma Acampora; Martina Caiazza; Marta Rubino; Emanuele Monda; Maria Rosaria Magaldi; Antonietta Tarallo; Marcella Sasso; Valeria De Pasquale; Roberto Grassi; Salvatore Cappabianca; Paolo Calabrò; Simona Fecarotta; Salvatore Esposito; Giovanni Esposito; Antonio Pisani; Luigi Michele Pavone; Giancarlo Parenti; Giuseppe Limongelli
Journal:  PLoS One       Date:  2020-05-19       Impact factor: 3.240

4.  Glycogen accumulation in smooth muscle of a Pompe disease mouse model.

Authors:  Angela L McCall; Justin S Dhindsa; Aidan M Bailey; Logan A Pucci; Laura M Strickland; Mai K ElMallah
Journal:  J Smooth Muscle Res       Date:  2021
  4 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.