Gorham-Stout syndrome of the shoulder

Brunner, U.; Rückl, Kilian; Konrads, Christian; Rudert, Maximilian; Plumhoff, Piet

doi:10.1051/sicotj/2016015

Cited by 9 publications

(5 citation statements)

References 26 publications

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“…The severity of the disease usually depends on the complicated structures and the progression of osteolysis, and there are generally no systemic symptoms. [10,11] Past trauma has been mentioned in specific individuals. [12] There was no history of trauma in our case, and the disease began after age 65.…”

Section: Discussionmentioning

confidence: 99%

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

et al. 2022

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Rationale: Gorham-Stout syndrome is a sporadic condition characterized by a tumor-like lesion with extensive osteolysis, severe symptoms, and a poor prognosis. Poor prognostic indicators include osteolytic lesions of the spine and pleura effusion.Patient concerns: A 67-year-old Chinese man with five months history of chest tightness presented to our institution with aggravated shortness of breath. Ultrasonography demonstrated hydrothorax on the right side. The patient's imaging studies (computerized tomography [CT] scan, magnetic resonance imaging, and positron emission tomography [PET]/CT) revealed osteolytic lesions (the skull, several spines, several ribs, both shoulder blades, and the pelvis).Diagnoses: Gorham-Stout syndrome. (4) Interventions: We advised the patient to follow a low-fat diet. On the patient, we performed a superior vena cava angiography. The injection of zoledronic acid was used to prevent bone loss.Outcomes: We found resolution of chylothorax after a low-fat diet, superior vena cava angiography and injection of zoledronic acid.Lessons: The possibility of Gorham -Stout syndrome should be ruled out in patients with clinical chylothorax. The relief of chylothorax requires comprehensive treatment.

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Section: Discussionmentioning

confidence: 99%

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

et al. 2022

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“…Las radiografías demostraron una osteólisis progresiva rápida de la cabeza humeral y fractura patológica en la espina de la escápula derecha. Tras el análisis histopatológico de distintas muestras de hueso, no se evidenció rastros de infección o malignidad, por lo que se asumió la relación con el síndrome de Gorham-Stout (11).…”

Section: Discussionunclassified

Correlación clínico-radiológica como primera ruta en el diagnóstico de Síndrome de Gorham-Stout

Lozada-Martínez

Nieves

Bolaño-Romero

et al. 2020

CES Med

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El síndrome de Gorham-Stout constituye una rara enfermedad ósea cuya etiología sigue siendo desconocida, que se caracteriza por presentar osteólisis y proliferación anormal de canales vasculares. Se describe el caso de un paciente masculino de 47 años quien consultó por un cuadro de aproximadamente 15 días de evolución consistente en edema, dolor, eritema y secreción sero-hemática por herida quirúrgica en hombro derecho, con diagnóstico reciente de artritis séptica. Refería antecedente de síndrome de Gorham-Stout, evidenciándose al examen físico y radiografía comparativa de hombros. La presentación clínica de estos pacientes depende del área afectada y la extensión de la enfermedad, convirtiéndose su diagnóstico en un desafío en caso de que el deterioro se presente en zonas no expuestas. El principal síntoma consiste en dolor localizado, acompañado de hinchazón, debilidad y deterioro funcional de las extremidades afectadas. Estos pacientes pueden permanecer asintomáticos hasta sufrir una fractura ósea espontáneamente o después de un trauma menor.

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“…The factors underlying the pathogenesis of GSD remain unknown, although various changes in molecular mechanisms have been reported. A high level of growth factors, such as VEGF-A (14), basic fibroblast growth factor (15), and platelet-derived growth factor-BB (16), have been reported in GSD patients, each of which may drive lymphatic endothelial cell proliferation via the mammalian target of rapamycin pathway (15). An increased interleukin (IL)-8 concentration (17) and an overexpression of CD105 (18) were also reported in GSD patients.…”

Section: Discussionmentioning

confidence: 99%

Gorham‑Stout disease in the rib and thoracic spine with spinal injury treated with radiotherapy, zoledronic acid, vitamin D, and propranolol: A case report and literature review

et al. 2019

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Gorham-Stout disease (GSD) is a rare bone condition that is characterized by the spontaneous, idiopathic and progressive proliferation of blood or lymphatic vessels, which replace the bone and marrow space. The precise etiology and pathophysiology of GSD remain poorly understood. Therefore, there is no consensus on the treatment of this disease. In the current study, a rare case of GSD in the rib and thoracic spine with spinal injury that was treated with radiotherapy, zoledronic acid, vitamin D and propranolol, is reported. A 77-year-old man visited the hospital complaining of constipation for 10 days. Within a few days, the patient had recognized complete paralysis of the bilateral lower legs. Radiologically, large osteolytic lesions were confirmed in the 6, 7 and 8th right ribs, the 6 and 7th thoracic vertebrae and in the liver and spleen. The lesions were diagnosed as GSD based on clinical findings, imaging characteristics and needle biopsy results. The patient was treated with zoledronic acid, activated vitamin D, propranolol and radiotherapy to the thoracic vertebrae. However, approximately 5 months after the first treatment, the rapidly increasing hemorrhagic pleural effusion compressed the left lung and caused a mediastinal shift to the right thorax. Unfortunately, the patient succumbed to the disease 7 months after first admission. To the best of our knowledge, this is the first reported case of GSD in the rib and thoracic spine with spinal injury to be treated with radiotherapy, propranolol, vitamin D and zoledronic acid. Furthermore, there have been no previous reports of a mediastinal shift caused by intralesional hemorrhage in GSD. For future reference, it should be noted that such processes may occur in GSD lesions of the thoracic spine and/or thorax. The present case will therefore contribute to a deeper understanding of GSD, a rare clinical entity.

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Gorham-Stout syndrome of the shoulder

Cited by 9 publications

References 26 publications

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

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

Correlación clínico-radiológica como primera ruta en el diagnóstico de Síndrome de Gorham-Stout

Gorham‑Stout disease in the rib and thoracic spine with spinal injury treated with radiotherapy, zoledronic acid, vitamin D, and propranolol: A case report and literature review

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