Chylothorax From Gorham-Stout Disease

Kotaru, Arun C; Rajput, Ashok Kumar

doi:10.1097/lbr.0000000000000506

Cited by 5 publications

(4 citation statements)

References 10 publications

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“…The most common traumatic causes of chylothorax are cardiothoracic surgical procedures. Non-traumatic causes include malignancy, tuberculosis, liver cirrhosis, and malformations[ 3 , 11 , 12 ]. Romero et al [ 13 ] reported that chylous ascites could present as chylothorax in patients with liver cirrhosis, due to communications between the thoracic and peritoneal cavity and lower pressure in the pleural cavity.…”

Section: Discussionmentioning

confidence: 99%

Chylothorax following posterior low lumbar fusion surgery: A case report

Huang¹,

Luo²,

Ran³

et al. 2021

WJCC

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BACKGROUND Postoperative chylothorax is usually regarded as a complication associated with cardiothoracic surgery; however, it is one of the rare complications in orthopedic surgery. This case report describes a female patient who developed chylothorax after a successful L4-S1 transforaminal lumbar interbody fusion surgery. The etiology, diagnosis, and treatment were analyzed and discussed. CASE SUMMARY A 50-year-old woman was admitted with repeated back and leg pain. She was diagnosed with L4 degenerative spondylolisthesis, L4/L5 and L5/S1 intervertebral disc herniation and L5 instability, and underwent successful posterior L4-S1 instrumentation and fusion surgery. Unfortunately, thoracic effusion was identified 2 d after operation. The thoracic effusion was finally confirmed to be chylous based on twice positive chyle qualitative tests. The patient was discharged after 12-d persisting drainage, 3-d total parenteral nutrition and fasting, and other supportive treatments. No recurring symptoms were observed within 12 mo follow-up. CONCLUSION Differential diagnosis is crucial for unusual thoracic effusion. Comprehensive diagnosis and treatment of chylothorax are necessary. Thorough intraoperative protection to relieve high thoracic pressure caused by the prone position is important.

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Section: Discussionmentioning

confidence: 99%

Chylothorax following posterior low lumbar fusion surgery: A case report

Huang¹,

Luo²,

Ran³

et al. 2021

WJCC

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“…[24] The mortality rate of GSD is high and the prognosis is poor in patients complicated with chylothorax. [12] The appearance of chylothorax was always fatal with death resulting from respiratory insufficiency, malnutrition, lymphopenia, and superimposed infection. [25]…”

Section: Discussionmentioning

confidence: 99%

“…[8,9] The chylous pericardial and pleural effusions can complicate the disease and lead to death in severe cases. [10–12] The medical treatment for GSD includes anti-osteoclastic medications (bisphosphonates), radiotherapy, thoracic duct ligation, interferon, and cement augmentation. [13] In this report, we show a case of Gorham disease involving thoracic spine with chylous pericardial and pleural effusions detected by 99m Tc-sulfur colloid (SC) single photon emission computed tomography/computed tomography (SPECT/CT).…”

Section: Introductionmentioning

confidence: 99%

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

2019

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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 99m 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. 99m 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 99m 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.

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“…Surgery also plays a key role in thoracic complications, such as chylothorax or even minor pleural fluid leakage, which have been treated with pleural drainage (119,149,151,152), pleurectomy, pleurodesis (64,153,154) and/ or thoracic duct ligation or embolization (14,27,65,155). In most of the cases, the best outcome has been obtained by combining these surgical techniques rather than using them in isolation (154,156).…”

Section: Surgical Treatmentmentioning

confidence: 99%

Current concepts from diagnosis to management in Gorham–Stout disease: a systematic narrative review of about 350 cases

et al. 2022

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Patients with Gorham–Stout disease (GSD) present progressive destruction and resorption of bone. Typical bone-related symptoms include swelling, pain and functional impairment in the region involved. The three aspects of GSD etiopathology are osteoclasts, angiogenesis/lymphangiogenesis and osteoblast function. Multi-targeted pharmacological approach includes innovative options and represent milestones of treatment, sometimes associated with radiotherapy. Surgery is mainly used to treat complications: pathologic/impending fractures, spinal instability or deformities and chylothorax. In this narrative review, we highlight current standards in diagnosis, clinical management and therapeutic strategies.

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Chylothorax From Gorham-Stout Disease

Cited by 5 publications

References 10 publications

Chylothorax following posterior low lumbar fusion surgery: A case report

Chylothorax following posterior low lumbar fusion surgery: A case report

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

Current concepts from diagnosis to management in Gorham–Stout disease: a systematic narrative review of about 350 cases

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