open access

Vol 77, No 3 (2009)
CASE REPORTS
Published online: 2009-04-23
Submitted: 2013-02-22
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Hemorrhagic parapneumonic effusion in a 64 year-old patient as the first symptom of hemophilia B

Elżbieta M. Grabczak, Rafał Krenke, Magdalena Jeleńska, Iwan Komarow, Urszula Ambroziak, Adam Fangrat, Ryszarda Chazan
Pneumonol Alergol Pol 2009;77(3):320-326.

open access

Vol 77, No 3 (2009)
CASE REPORTS
Published online: 2009-04-23
Submitted: 2013-02-22

Abstract

Hemophilia B is an inherited, X chromosome-linked disease. It is usually diagnosed in childhood, sometimes in adolescence. The commonest symptoms include spontaneous or post-traumatic bleeding into the joints and/or muscles, as well as mucosal bleeding. Respiratory symptoms are rarely reported.
We present the case of a 64 year-old man in whom bloody parapneumonic effusion (hemothorax) was the first symptom of hemophilia B. The reason for prolonged activated partial thromboplastin time (APTT) found on admission has not been elucidated. Since antibiotic therapy and pleural tube thoracostomy with intrapleural streptokinase were found to be ineffective, video-assisted thoracic surgery was performed with the right lung decortication. Post-operative treatment was complicated by massive pleural bleeding requiring two subsequent thoracotomies. Additional blood tests revealed factor IX deficiency and resulted in hemophilia B being diagnosed.
The presented case proves that hereditary bleeding disorders may be diagnosed even in late adulthood. Intrapleural bleeding related to pneumonia and pleural inflammation might be the first presenting symptom. Hemophilia should be considered as a potential cause of APTT prolongation, even in an elderly patient with atypical presentation. Explaining the reason for APTT prolongation before the surgical procedure could have allowed to avoid severe bleeding in the described patient.

Abstract

Hemophilia B is an inherited, X chromosome-linked disease. It is usually diagnosed in childhood, sometimes in adolescence. The commonest symptoms include spontaneous or post-traumatic bleeding into the joints and/or muscles, as well as mucosal bleeding. Respiratory symptoms are rarely reported.
We present the case of a 64 year-old man in whom bloody parapneumonic effusion (hemothorax) was the first symptom of hemophilia B. The reason for prolonged activated partial thromboplastin time (APTT) found on admission has not been elucidated. Since antibiotic therapy and pleural tube thoracostomy with intrapleural streptokinase were found to be ineffective, video-assisted thoracic surgery was performed with the right lung decortication. Post-operative treatment was complicated by massive pleural bleeding requiring two subsequent thoracotomies. Additional blood tests revealed factor IX deficiency and resulted in hemophilia B being diagnosed.
The presented case proves that hereditary bleeding disorders may be diagnosed even in late adulthood. Intrapleural bleeding related to pneumonia and pleural inflammation might be the first presenting symptom. Hemophilia should be considered as a potential cause of APTT prolongation, even in an elderly patient with atypical presentation. Explaining the reason for APTT prolongation before the surgical procedure could have allowed to avoid severe bleeding in the described patient.
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Keywords

spontaneous hemothorax; parapneumonic effusion; hemophilia B

About this article
Title

Hemorrhagic parapneumonic effusion in a 64 year-old patient as the first symptom of hemophilia B

Journal

Advances in Respiratory Medicine

Issue

Vol 77, No 3 (2009)

Pages

320-326

Published online

2009-04-23

Bibliographic record

Pneumonol Alergol Pol 2009;77(3):320-326.

Keywords

spontaneous hemothorax
parapneumonic effusion
hemophilia B

Authors

Elżbieta M. Grabczak
Rafał Krenke
Magdalena Jeleńska
Iwan Komarow
Urszula Ambroziak
Adam Fangrat
Ryszarda Chazan

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