Child Kidney Dis.
2020 Oct;24(2):120-125. 10.3339/jkspn.2020.24.2.120.
Gorham-Stout Syndrome with Focal Segmental Glomerulosclerosis: A Case Report
- Affiliations
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- 1Department of Pediatrics, Seoul National University Bundang Hospital, Seongnam, Korea
- 2Department of Pediatrics, Seoul National University Children's Hospital, Seoul, Korea
- 3Department of Pathology, Seoul National University Hospital, Seoul, Korea
- 4Kidney Research Institute, Medical Research Center, Seoul National University College of Medicine, Seoul, Korea
- 5Wide River Institute of Immunology, Seoul National University, Seoul, Korea
- KMID: 2508550
- DOI: http://doi.org/10.3339/jkspn.2020.24.2.120
Abstract
- Gorham-Stout syndrome is a rare bone disorder characterized by progressive massive osteolysis and proliferation of vascular and lymphatic vessels. A 15-year-old boy was initially diagnosed with Gorham-Stout at the age of 8 years based on clinical and radiological findings. Following diagnosis, he was treated with pamidronate, interferon alfa, propranolol, oral corticosteroids, and sirolimus. He developed proteinuria at the age of 15 and progressed into the nephrotic range 2 years later. A renal biopsy revealed focal segmental glomerulosclerosis, not otherwise specified variant. The sequential increase in proteinuria associated with medications suggested that the focal segmental glomerulosclerosis may be caused by pamidronate and sirolimus, but cannot completely rule out the possibility of kidney involvement of GSS itself.
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