Pathologically confirmed diffuse alveolar haemorrhage in lymphangioleiomyomatosis

被引:0
|
作者
Kobylianskii, Jane [1 ]
Hutchinson-Jaffe, Adam [1 ,2 ]
Cabanero, Michael [3 ,4 ]
Thenganatt, John [1 ,5 ]
机构
[1] Univ Toronto, Dept Med, Toronto, ON, Canada
[2] North York Gen Hosp, N York, ON, Canada
[3] Univ Toronto, Dept Lab Med & Pathobiol, Toronto, ON, Canada
[4] Univ Hlth Network, Div Anat Pathol, Toronto, ON, Canada
[5] Univ Hlth Network, Div Respirol, Toronto, ON, Canada
关键词
interstitial lung disease; pathology; ACUTE RESPIRATORY-FAILURE; HEMOPTYSIS; PART;
D O I
10.1136/bcr-2020-238713
中图分类号
R5 [内科学];
学科分类号
1002 ; 100201 ;
摘要
A 40-year-old woman was referred to pulmonology after presenting with dyspnoea and self-limiting haemoptysis. Chest CT revealed diffuse ground glass opacities and small thin-walled cysts. Bronchoalveolar lavage cultures were negative and cytology revealed haemosiderin-laden macrophages. Transthoracic echocardiogram was normal. Connective tissue disease and vasculitis work-up were negative. Vascular endothelial growth factor-D level was indeterminate. Lung function was normal. She underwent video-assisted thoracoscopic lung biopsy. In addition to findings consistent with lymphangioleiomyomatosis, histopathological examination identified haemosiderosis without capillaritis, confirming a diagnosis of diffuse alveolar haemorrhage in the context of the associated clinical and radiographic features. Follow-up imaging after 5 months showed resolution of the diffuse ground glass opacities. Pharmacotherapy with sirolimus was not initiated due to absence of deterioration in pulmonary function. Diffuse alveolar haemorrhage in patients with lymphangioleiomyomatosis is a rare but important presentation. The few previously reported cases progressed to respiratory failure requiring mechanical ventilation.
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页数:4
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