• Yasutomo Kumakura, Norio Hasuda, Kazuki Akita, Tetsuya Iijima, and Takashi Matsukawa.
• Department of Anesthesiology, Faculty of Medicine, University of Yamanashi, Chuo, Japan.
• Medicine (Baltimore). 2023 Jun 2; 102 (22): e33950e33950.

RationaleGorham-Stout disease (GSD) is a rare disease that causes massive osteolysis and proliferation of abnormal lymphangiomatous tissues. Patients with GSD often experience pain associated with bone fractures and chylothorax. However, bleeding caused by abnormal lymphangiomatous tissue or hematological dysfunction rarely occurs.Patient ConcernsA 22-year-old female patient with GSD presented with severe left hip and lower limb pain. The GSD had disappeared her right pelvic bone and femur, but no abnormalities were found in the bones at the site of the pain.DiagnosesThe patient presented with a chylothorax and cerebrospinal fluid leakage. She was treated with sirolimus and an epidural blood patch, and her symptoms resolved. Computed tomography and magnetic resonance imaging revealed an epidural hematoma extending from L3 to the caudal region, and blood results revealed a consumption coagulopathy.InterventionsWe presumed that the hematoma caused pain and prescribed pregabalin and morphine. The pain gradually subsided.OutcomesAn unexpected liver subcapsular hemorrhage occurred 4 months later, and the patient went into hemorrhagic shock. Transcatheter arterial embolization was promptly performed, and the patient recovered.LessonsGSD infrequently causes bleeding related to abnormal lymphangiomatous tissues and coagulopathy, yet it can lead to serious events if it occurs.Copyright © 2023 the Author(s). Published by Wolters Kluwer Health, Inc.

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