Abstract Background Lymphangioma is a multi-systemic disease mostly affecting the mesentery, omentum, mesocolon and retroperitoneum and rarely involving bone, with fewer than 30 cases reported so far. Lymphangioma usually has no specific clinical manifestation and could be easily misdiagnosed. Lymphangioma with myxoid degeneration was not reported. We report a histopathologically proved case of lymphangioma of 6th and 7th cervical vertebra (C6–7) with myxoid degeneration. Case presentation A 45-year-old male who suffered from pain in the right shoulder for 1 month, aggravated with numbness in the right little finger and ring finger and radiating pain for half a month. CT showed multiple cystic bone destruction areas in C6–7 vertebral right part and appendage with marginal osteosclerosis and surrounded by soft tissue density. The size of the lesion was about 36 mm × 41 mm . MRI: the margin of the lesion appears lobulated. Lesion showed slightly hypointense on T1WI, slightly hyperintense on T2WI and STIR. Multiple patchy T1WI hypointense and T2WI hyperintense shadows were seen within the lesion. No enhancement was observed in the lesion after injection of contrast, and there was no abnormal signal in surrounding soft tissue and bone. The histopathological examination confirmed the diagnosis of lymphangioma with myxoid degeneration after surgical resection. After over 2 years of follow-up, there were no signs of disease recurrence and progression. Conclusions To the best of our knowledge, this is the first case reported in English language literature of lymphangioma with myxoid degeneration. 

/pdf/cervical-lymphangioma-with-myxoid-degeneration-a-rare-case-1rkgkgwc.pdf

Cervical lymphangioma with myxoid degeneration: a rare case report and literature review

https://bnrc.springeropen.com/track/pdf/10.1186/s42269-022-00736-w

Background Chylopericardium refers to the accumulation of chylous fluid in the pericardial cavity. Non-enhanced magnetic resonance lymphangiography (MRL) can show neck and thoracic lymphatic abnormalities in the primary chylopericardium. It is not clear whether there is a relationship between neck and thoracic lymphatic abnormalities in primary chylopericardium and thoracic duct terminal release surgery. This study aimed to explore the correlation between the severity of neck and thoracic lymphatic abnormalities observed in non-enhanced MRL and the surgical outcomes in primary chylopericardium. Methods This is a retrospective cohort study. A retrospective analysis was conducted on fifty-six patients diagnosed with primary chylopericardium between January 2016 and December 2021, all of whom underwent thoracic duct terminal release surgery. Ultrasonography, chest computed tomography (CT) and non-enhanced MRL were performed prior to the surgical intervention. Patients were categorized into four types based on the severity of neck and thoracic lymphatic abnormalities observed in the non-enhanced MRL. Clinical and laboratory examinations and surgical outcomes were compared across different types using χ2-test or Fisher’s exact test, t-test, and Kruskal-Wallis H-test. Additionally, independent factors influencing surgical outcomes were analyzed. Results Among primary chylopericardium cases (n=56), 22 (39.2%) were classified as type I or II, 17 (30.4%) as type III, and 17 (30.4%) as type IV. Surgical outcomes were more favorable for type I or II patients than those with type III or IV, accompanied by a reduction in postoperative primary chylopericardium volume (P=0.002). Postoperative chest CT scans indicated that type I or II patients had fewer instances of large grid shadows, small grid shadows, and bronchovascular bundle thickening compared to preoperative scans (P=0.001, P=0.02, P=0.03). Age and bronchomediastinal trunk dilation emerged as independent factors influencing surgical outcomes [odds ratio (OR) 0.03, 95% confidence interval (CI): 0.003–0.220, P=0.001; OR 11.10, 95% CI: 1.70–72.39, P=0.01, respectively]. Conclusions A more severe degree of neck and thoracic lymphatic abnormalities is associated with worse surgical outcomes. Moreover, age and bronchomediastinal trunk dilatation are independent predictors of surgical outcomes. Preoperative utilization of non-enhanced MRL for severity of lymphatic abnormalities classification in primary chylopericardium patients offers a noninvasive means of assessing surgical risk.

Association between lymphatic abnormalities in the neck and thorax in primary chylopericardium and surgical outcomes evaluated by non-enhanced magnetic resonance (MR) lymphangiography

Background Chylopericardium due to generalized lymphangiomatosis is a rare clinical entity. Its aetiology and treatment remain unclear. Case summary We report one case of a 51-year-old man who was diagnosed with generalized lymphangiomatosis with idiopathic chylopericardium by bilateral inguinal intranodal contrast-enhanced magnetic resonance lymphangiography. Magnetic resonance lymphangiography demonstrated abnormal communications between the pericardial sac and the thoracic duct. The patient with idiopathic chylopericardium was therefore successfully treated by exclusive surgical ligation of the abnormal communications and partial pericardiectomy by thoracotomy. The patient's postoperative recovery was uneventful, and no recurrence of pericardial effusion occurred during the 13-month follow-up. Discussion Magnetic resonance lymphangiography showed a good capability for evaluating the extent of generalized lymphangiomatosis and therefore is helpful for delineating the anatomy of the thoracic duct and identifying chyle leakage and abnormal communications between the thoracic duct and the pericardial sac. This makes a simple ligature of the abnormal communications, instead of thoracic duct mass ligation possible in the treatment of chylopericardium.

/pdf/a-case-report-of-generalized-lymphangiomatosis-with-1unzrx4679.pdf

A case report of generalized lymphangiomatosis with chylopericardium: the crucial role of magnetic resonance lymphangiography.

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A case report of generalized lymphangiomatosis with chylopericardium: the crucial role of magnetic resonance lymphangiography.