Literature DB >> 9357476

Mucopolysaccharidosis presenting as pediatric multiple aortic aneurysm: first reported case.

J Engle1, H J Safi, O Abbassi, D C Iliopoulos, D Dorsay, J Cartwright, D Weilbaecher.   

Abstract

Within the pediatric population, the rare aortic aneurysm is most often brought on by congenital cardiovascular malformation or connective tissue disorder, trauma, inflammatory disease, or infection. Thus our 8-year-old patient who had multiple aortic aneurysms and evidence of mucopolysaccharidosis presented a doubly unique case. Three and one-half months after the patient underwent emergency aortic valve replacement, we performed resection and graft replacement of both her descending thoracic aorta and thoracoabdominal aorta. Histologic analysis of the aneurysm wall displayed severe medial degeneration with large deposits of acid mucopolysaccharides. Subsequent evaluation, although negative for connective tissue disorders, showed glycosaminoglycans, chondroitin sulfate, and heparan sulfate in the patient's urine. These findings are diagnostic for a heterogeneous group of storage diseases termed mucopolysaccharidoses, although testing of the patient's cultured fibroblasts failed to reveal any specific previously described enzymatic defect. After reviewing the literature, we believe that this is the first known successfully treated pediatric aortic aneurysm associated with mucopolysaccharidosis.

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Year:  1997        PMID: 9357476     DOI: 10.1016/s0741-5214(97)70074-0

Source DB:  PubMed          Journal:  J Vasc Surg        ISSN: 0741-5214            Impact factor:   4.268


  7 in total

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Review 4.  Possible mechanical roles of glycosaminoglycans in thoracic aortic dissection and associations with dysregulated transforming growth factor-β.

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5.  Clinical characterization of cardiovascular abnormalities associated with feline mucopolysaccharidosis I and VI.

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6.  Current and emerging management options for patients with Morquio A syndrome.

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Review 7.  Insights on the Pathogenesis of Aneurysm through the Study of Hereditary Aortopathies.

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  7 in total

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