BMJ case reports
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Case Reports
Ruptured intercostal artery pseudoaneurysm in a patient with blunt thoracic trauma: diagnosis and management.
Intercostal artery pseudoaneurysm is an extremely unusual condition, with less than 10 reported cases to our knowledge. Most of them have been associated with surgical interventions or blunt thoracic trauma. The bleeding risk in this kind of lesions is considerable, the majority of them presenting as haemothorax. ⋯ The identification of a small artery pseudoaneurysm as the cause of haemothorax requires knowledge of this possible aetiology as well as detailed attention to the CT technique. Embolisation is considered to be the first therapeutic method in the management of a ruptured pseudoaneurysm. To reduce the risk of failure, the anatomic features and adjacent vessels providing collateral branches must be studied and embolised if needed, with important attention to collateral blood supply arising from the musculophrenic and anterior intercostal arteries.
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A 3-month-old infant was admitted to the respiratory unit for dyspnoea and vomiting after her second DTaP-Polio vaccine shot. The chest X-rays showed a white right lung with a left mediastinal shift. A pleural aspiration assessed the diagnosis of chylothorax. ⋯ Ten months later, her physical and biological conditions were normal and her chest X-rays dramatically improved. This case highlights the difficult management of infant chylothorax. Although conservative treatment has to be tried first, surgical procedures as pleuroperitoneal shunting and rarely pleurodesis have to be discussed.
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Case Reports
Recovery of chylous pulmonary congestion in tuberous sclerosis complex-associated lymphangioleiomyomatosis.
Chyloptysis and chylous pulmonary congestion are extremely rare complications of lymphangioleiomyomatosis (LAM). We report a case of a 50-year-old woman with tuberous sclerosis complex-associated LAM, who presented with expectorating milky-white bronchial casts. ⋯ Her symptoms and lung infiltration were improved by oral sirolimus therapy; moreover, serum Krebs von den Lungen-6 (KL-6) levels paralleled the symptoms and lung infiltration of these complications. We suggest that serum KL-6 may be a useful monitoring biomarker of chyloptysis and chylous pulmonary congestion in LAM.
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Vaccine failure is a rare condition and the need to investigate a primary immunodeficiency is controversial. We present the case of a 4-year-old boy, with complete antipneumococcal vaccination, who had necrotising pneumonia with pleural effusion and severe pancytopaenia with need for transfusion. A vaccine-serotype Streptococcus pneumoniae was isolated in the blood culture. ⋯ Dyskeratosis congenita is an inherited disorder that causes shortening or dysfunction of telomeres, affecting mainly rapidly dividing cells (particularly in the skin and haematopoietic system). It leads to bone marrow failure, combined immunodeficiency and predisposition to cancer. The confirmation of this diagnosis allows genetic counselling and medical monitoring of these patients, in order to detect early complications such as bone marrow aplasia or malignancies.
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We report a case of two patients with foot drop due to peroneal nerve infarct as early sign of two different forms of primary systemic vasculitides: a predominantly small-vessel p-antineutrophil cytoplasmic antibody-positive vasculitis (microscopic polyangiitis) and a predominantly medium-vessel vasculitis (polyarteritis nodosa).