LAUSR

Introduction Gorham-Stout disease is a rare disease that was first described by Jackson in 1838. The first case in jaws was reported in 1924. There is no clear consensus on the etiopathogenesis of the disease. A few proposed theories include bone destruction secondary to hyperemia from hemangiomatosis, increased osteoclastic activity mediated by IL-6, and lymphangiogenesis mediated by vascular endothelial growth factor, platelet-derived growth factor, and IL-6. The disease presents as an osteolytic lesion without cortical expansion. Here, we present a case of Gorham-Stout disease exhibiting hemorrhagic gingival overgrowth in the mandible. Case Report A 46-year-old female patient presented with a chief complaint of blisters and bubbles in her lower gums. Clinical examination revealed multiple loose teeth in the anterior and left mandible with hemorrhagic and hyperplastic gingival tissue. Upon radiographic evaluation, significant bone loss was recognized in the area, giving the appearance of “teeth floating in air.” The differential diagnosis included Gorham-Stout disease, osteomyelitis, Langerhans cell histiocytosis, and other conditions with osteolytic changes. Histologic examination of the biopsy specimen revealed atypical vascular proliferation with mixed acute and chronic inflammation favoring exuberant granulation tissue. Her lab workup confirmed elevated levels of serum IL-6 and vascular endothelial growth factor. A diagnosis of Gorham-Stout disease was made based on these findings. The patient presented with a pathologic fracture of her left mandible 3 months after her initial consultation. She was referred to rheumatology and treatment with bisphosphonates was initiated. However, the patient failed to return for further treatment with the physician and was lost to follow-up.