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Multicentric Retrospective Evaluation of Five Classic Infantile Pompe Disease Subjects Under Enzyme Replacement Therapy With Early Infratentorial Involvement.
Paoletti, Matteo; Pichiecchio, Anna; Colafati, Giovanna Stefania; Conte, Giorgio; Deodato, Federica; Gasperini, Serena; Menni, Francesca; Furlan, Francesca; Rubert, Laura; Triulzi, Fabio Maria; Cinnante, Claudia.
Afiliação
  • Paoletti M; Advanced Imaging and Radiomics Center, Neuroradiology Department, Istituto di Ricovero e Cura a Carattere Scientifico Mondino Foundation, Pavia, Italy.
  • Pichiecchio A; Advanced Imaging and Radiomics Center, Neuroradiology Department, Istituto di Ricovero e Cura a Carattere Scientifico Mondino Foundation, Pavia, Italy.
  • Colafati GS; Department of Brain and Behavioral Sciences, University of Pavia, Pavia, Italy.
  • Conte G; Oncological Neuroradiology Unit, Imaging Department, Istituto di Ricovero e Cura a Carattere Scientifico Ospedale Pediatrico Bambino Gesù, Rome, Italy.
  • Deodato F; Fondazione Istituto di Ricovero e Cura a Carattere Scientifico Ca' Granda Ospedale Maggiore Policlinico, Neuroradiology Unit, Milan, Italy.
  • Gasperini S; Unit of Metabolic Disease, Istituto di Ricovero e Cura a Carattere Scientifico Ospedale Pediatrico Bambino Gesù, Rome, Italy.
  • Menni F; Pediatric Rare Diseases Unit, Department of Pediatrics, Fondazione Monza e Brianza per il Bambino e la sua Mamma, San Gerardo Hospital, Monza, Italy.
  • Furlan F; Pediatric Highly Intensive Care Unit, Department of Pathophysiology and Transplantation, University of Milan, Istituto di Ricovero e Cura a Carattere Scientifico Ca' Granda Ospedale Maggiore Policlinico, Milan, Italy.
  • Rubert L; Pediatric Highly Intensive Care Unit, Department of Pathophysiology and Transplantation, University of Milan, Istituto di Ricovero e Cura a Carattere Scientifico Ca' Granda Ospedale Maggiore Policlinico, Milan, Italy.
  • Triulzi FM; Division of Inherited Metabolic Diseases, Department of Diagnostic Services, University Hospital of Padua, Padua, Italy.
  • Cinnante C; Fondazione Istituto di Ricovero e Cura a Carattere Scientifico Ca' Granda Ospedale Maggiore Policlinico, Neuroradiology Unit, Milan, Italy.
Front Neurol ; 11: 569153, 2020.
Article em En | MEDLINE | ID: mdl-33329311
White matter (WM) abnormalities and ventricular enlargement in brain MRI are well-known features in infantile-onset Pompe disease (IOPD) in the era of enzyme replacement therapy (ERT). In this multicentric observational retrospective study, we report a small cohort of IOPD subjects under ERT treatment (n = 5, median age at MRI = 7.4 years, median period of treatment = 85 months) that showed the classic features of extensive supratentorial WM abnormalities but also unusual findings such as early infratentorial WM abnormalities and late supratentorial U-fiber involvement. Given the recent implementation of ERT and the rarity of the disease, a complete spectrum of presentation and understanding of progressive pathology in the brain of IOPD subjects in treatment remains underacknowledged. The availability of long-term follow-up of IOPD subjects under ERT treatment allows a better insight into the evolution of brain abnormalities in such disease.
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Texto completo: 1 Base de dados: MEDLINE Idioma: En Ano de publicação: 2020 Tipo de documento: Article

Texto completo: 1 Base de dados: MEDLINE Idioma: En Ano de publicação: 2020 Tipo de documento: Article