99mTc-SC lymphoscintigraphy and SPECT/CT findings in a case report of Gorham-Stout disease presenting with chylothorax and bone pain.

RATIONALE: Gorham-Stout disease (GSD) is a rare disorder characterized by multiple osteolytic lesions, sometimes complicated by chylothorax. The aim of this case report is to introduce a very rare case of Gorham-Stout syndrome, which involved several bones along with chylous pericardial and pleural effusions detected by Tc-sulfur colloid (SC) lymphoscintigraphy and single photon emission computed tomography/computed tomography (SPECT/CT).

PATIENT CONCERNS: A 15-year-old girl presented to our hospital complaining of shortness of breath and bone pain.

DIAGNOSIS: The CT showed multiple osteolytic lesions, left-sided pleural effusion, and pericardial effusion. Tc-SC lymphoscintigraphy showed discontinuation of thoracic duct and tracer accumulation on the left side chest. SPECT/CT revealed increased radioactivity uptake in pleural, pericardial effusions, and some thoracolumbar spines. Diagnostic thoracentesis to identify the nature of pleural effusion and histopathology of biopsy in the right femoral to that of the bone lesion were performed. Based on the clinical information, histopathologic, and radiographic findings, the diagnosis of GSD was made.

INTERVENTIONS: The patient received thoracic duct ligation and bisphosphonates treatment.

OUTCOMES: After receiving thoracic duct ligation and bisphosphonates treatment, the patient's symptoms of bone pain and dyspnea were relieved, and the pericardial and pleural fluid was diminished dramatically. At the 3-month and 9-month follow-up visit, the patient had nearly complete remission without any complication.

LESSONS: The Tc-SC lymphoscintigraphy and SPECT/CT could provide significant value assessing the lymphatic abnormity and evaluating the extent of disease, therefore aiding to guide decision making in the clinic.