| Literature DB >> 28304197 |
Rie Aoki1, Kittipong Srivatanakul1, Takahiro Osada1, Kazuko Hotta1, Takatoshi Sorimachi1, Mitsunori Matsumae1, Hiroko Morisaki2,3.
Abstract
Background Loeys-Dietz syndrome (LDS) is a recently recognized autosomal dominant connective tissue disease. The manifestations of LDS include vascular tortuosity, scoliosis, craniosynostosis, aneurysm and aortic dissections. Clinical presentation A 35-year-old woman treated with Stanford type B aortic dissection and breast cancer was referred to us for Borden type II dural arteriovenous fistula (dAVF) draining to the vein of Galen, involving the midline of tentorium cerebelli. The dAVF was treated successfully by combined transarterial and transvenous embolization. Because of tortuosity of vertebral arteries, a genetic test was conducted confirming LDS type 2. Conclusions To our knowledge, this is the first case report of dAVF associated with LDS. The relationship between LDS and dAVF is unknown but this report shows the possibility that mutation of transforming growth factor β receptors 2 ( TGFBR2) related to LDS may be related to shunt diseases. Because intervention in LDS seems to be feasible compared to Ehlers Danlos syndrome and Marfan syndrome, it is important to make the correct diagnosis.Entities:
Keywords: Loeys-Dietz syndrome; dural arteriovenous fistula; endovascular therapy
Mesh:
Year: 2017 PMID: 28304197 PMCID: PMC5433611 DOI: 10.1177/1591019916686054
Source DB: PubMed Journal: Interv Neuroradiol ISSN: 1591-0199 Impact factor: 1.610