Recurrent hemorrhagic pericardial effusion in a child due to diffuse lymphangiohemangiomatosis: a case report

Abstract

IntroductionRecurrent hemorrhagic pericardial effusion in children with no identifiable cause is a rare presentation.Case presentationWe report the case of a 4-year-old Indian girl who presented with recurrent hemorrhagic pericardial effusion. Diffuse lymphangiomatosis was suspected when associated pulmonary involvement, soft tissue mediastinal mass, and lytic bone lesions were found. Pericardiectomy and lung biopsy confirmed the diagnosis of diffuse lymphangiohemangiomatosis. Partial clinical improvement occurred with thalidomide and low-dose radiotherapy, but our patient died from progressive respiratory failure.ConclusionDiffuse lymphangiohemangiomatosis should be considered in the differential diagnosis of hemorrhagic pericardial effusion of unclear cause.

A gene missense mutation in diffuse pulmonary lymphangiomatosis with thrombocytopenia

“…[ 2 , 6 ] Like the present patient, 1 case with lymphangiomatosis had only thrombocytopenia without DIC and splenomegaly; she was not treated for thrombocytopenia and died from progressive respiratory failure. [ 3 ] The current patient had thrombocytopenia during follow-up, but no DIC or spleen involvement; he had no bleeding symptoms or progressive respiratory failure, and received no therapy for thrombocytopenia. Fortunately, he showed no deterioration during follow-up.…”

“…Based on clinical findings, pathology, and immunohistochemical data, the overall picture favored the diagnosis of lymphangiomatosis. [ 3 ] After right side thoracentesis, anti-infective treatment with ceftriaxone and clindamycin, and atomization for cough, no pericardial and pleural effusions were found in ultrasound examinations. His condition was improved and he was discharged on March 13, 2013.…”

“…To date, 8 cases of DPL have been reported with thrombocytopenia and DIC. [ 1 – 9 ] In addition, only thrombocytopenia was observed in 1 patient [ 3 ] (Table 1 ). Here, we present the case of a young male with DPL involving the mediastinum and pulmonary interstitium, accompanied by thrombocytopenia.…”

Coxsackie B viral infection presenting with hemorrhagic pericardial effusion and pleural effusion

“…Common causes of hemorrhagic pericardial effusion in children include tuberculosis and other bacteria such as Staphylococcus aureus ,[ 1 ] malignancy, trauma, drugs, and collagen vascular diseases. [ 2 ] There has been a report of hemorrhagic pericardial effusion due to Chlamydophila pneumoniae in an 8-year-old child. [ 3 ] Viruses are less commonly implicated in the etiology of hemorrhagic pericardial effusions in children.…”

Novel molecular pathways in Gorham disease: Implications for treatment

“…It also acts as an antiangiogenic agent by inhibiting IL‐12 and bFGF and is used in the treatment of multiple myeloma . Thalidomide did not lead to sustained clinical improvement in two cases of Gorham disease, either administered together with celecoxib or as monotherapy . In contrast, another report described full disease stabilization by thalidomide in a case of disseminated lymphangiomatosis accompanied by chylothorax, refractory to interferon‐α .…”