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A 45-year-old male patient with spontaneous chylothorax and osteolysis in the right 1st and 2nd ribs was diagnosed with Gorham-Stout disease based on clinical manifestations and bone biopsy. The chylothorax temporarily decreased after a successful selective lymphatic embolization. The patient presented with recurrent chylothorax, mild chest discomfort, and progressive osteolysis (despite administering sirolimus) during the follow-up period of 15 months.

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Generalized lymphatic anomaly (GLA) is an uncommon congenital disease secondary to the proliferation of lymphatic vessels in any organ except the central nervous system. GLA has a wide spectrum of clinical and radiological presentations, among which osteolytic lesions are the most widespread, being the ribs the most commonly affected bone. GLA is diagnosed mainly in children and young adults; nevertheless, on rare occasions it can remain asymptomatic and be detected incidentally in older patients.

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Gorham-Stout syndrome: A chylothorax disease with bony destruction: A case report.

Medicine (Baltimore)

December 2022

Department of Pulmonary and Critical Care Medicine, Zhongshan Hospital of Xiamen University, School of Medicine, Xiamen University; the Third Clinical Medical College of Fujian Medical University, Fujian, China.

Article Synopsis
  • - Gorham-Stout syndrome is a rare and serious condition that leads to painful tumor-like lesions and significant bone loss, resulting in unfavorable outcomes, especially when affecting the spine or causing fluid buildup in the chest.
  • - A 67-year-old man experienced severe chest tightness and shortness of breath, which was linked to hydrothorax and multiple osteolytic lesions revealed by imaging tests.
  • - Treatment involved dietary changes, angiography, and a medication called zoledronic acid, which successfully resolved the patient's symptoms, highlighting the need for thorough evaluation in similar cases.
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Gorham-Stout disease (GSD) is thought to be due to uncontrolled proliferation of vascular and lymphatic structures within bone tissue causing destruction and osteolysis of bone. We present a patient in her mid-40s who reported chronic shoulder pain, a pleural effusion and irregular periods. Investigations showed osteolysis of her ribs, pleural effusions, an ovarian mass and a raised carbohydrate antigen 125 (Ca-125).

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A 73-year-old man with multiple myeloma (initial diagnosis 21 months earlier) was referred to our center for a whole-body 18 F-FDG PET/CT. We detected a bilateral synchronous testicular manifestation, which was confirmed by histopathology after orchiectomy. Besides hypermetabolic lesions in the spine and ribs (most likely old fractures), known osteolysis showed no uptake.

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