Severe, chronic cough caused by pulmonary arteriovenous malformations in a patient with hereditary haemorrhagic telangiectasia: case report

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• 作者：Etienne-Marie Jutant (1)
  Philippe Puyo (2)
  Mostafa El Hajjam (3)
  Sandra Blivet (1)
  Emmanuel Houdart (4)
  Michel Aubier (5)
  Pascal Lacombe (3)
  Thierry Chinet (1)
  
  1. APHP ; Department of Pulmonology and Thoracic Oncology ; Ambroise Par茅 Hospital ; Boulogne ; France
  2. Foch Hospital ; Department of Thoracic Surgery ; Suresnes ; France
  3. APHP ; Department of Radiology ; Ambroise Par茅 Hospital ; Boulogne ; France
  4. APHP ; Department of Neuroradiology ; Lariboisi猫re Hospital ; Paris ; France
  5. APHP ; Department of Pulmonology ; Bichat Hospital ; Paris ; France
  
• 关键词：Cough ; Hereditary haemorrhagic telangiectasia ; Pulmonary arteriovenous malformation
• 刊名：BMC Pulmonary Medicine
• 出版年：2015
• 出版时间：December 2015
• 年：2015
• 卷：15
• 期：1
• 全文大小：1,365 KB
• 参考文献：1. Gossage, JR, Kanj, G (1998) Pulmonary arteriovenous malformations. Am J Respir Crit Care Med 158: pp. 643-61 CrossRef
  2. Faughnan, ME, Palda, VA, Garcia-Tsao, G, Geisthoff, UW, McDonald, J, Proctor, DD (2011) International guidelines for the diagnosis and management of hereditary hemorragic telangiectasia. J Med Genet 48: pp. 73-87 CrossRef
  3. Wong, HH, Chan, RP, Klatt, R, Faughnan, ME (2011) Idiopathic pulmonary arteriovenous malformations: clinical and imaging characteristics. Eur Respir J 38: pp. 368-75 CrossRef
  4. Faughnan, ME, Granton, JT, Young, LH (2009) The pulmonary vascular complications of hereditary haemorrhagic telangiectasia. Eur Respir J 33: pp. 1186-94 CrossRef
  5. Blivet, S, Cobarzan, D, Beauchet, A, Hajjam, M, Lacombe, P, Chinet, T (2014) Impact of arteriovenous malformations on respiratory-related quality of life in patients with hereditary haemorrhagic telangiectasia. PLoS One 9: pp. e90937 CrossRef
  6. Moussoutas, M, Fayad, P, Rosenblatt, M, Hashimoto, M, Pollak, J, Henderson, K (2000) Pulmonary arteriovenous malformations. Cerebral ischemia and neurologic manifestations. Neurology 55: pp. 959-64 CrossRef
  7. Lacombe, P, Lacout, A, Marcy, PY, Binsse, S, Sellier, J, Bensalah, M (2013) Diagnosis and treatment of pulmonary arteriovenous malformations in hereditary hemorrhagic telangiectasia: an overview. Diagn Interv Imaging 94: pp. 835-48 CrossRef
  8. Lacombe, P, Lagrange, C, Hajjam, M, Chinet, T, Pelage, JP (2005) Reperfusion of complex pulmonary arteriovenous malformations after embolization: report of three cases. Cardiovasc Intervent Radiol 28: pp. 30-5 CrossRef
  9. Niimi, A, Kihara, Y, Sumita, Y, Okano, Y, Tambara, K, Fujita, M (2005) Cough reflex by ventricular premature contractions. Int Heart J 46: pp. 923-6 CrossRef
  
• 刊物主题：Pneumology/Respiratory System; Internal Medicine;
• 出版者：BioMed Central
• ISSN：1471-2466

文摘

Background Patients with pulmonary arteriovenous malformations usually complain of dyspnoea upon exertion, fatigue or migraine, or may be asymptomatic. We describe a patient with an unreported manifestation of a pulmonary arteriovenous malformation: a severe chronic cough. Case presentation A 51-year old Caucasian non-smoking female police officer presented with a chronic cough. She had been diagnosed with hereditary haemorrhagic telangiectasia in 1992. She complained of a severe, dry cough at the time of the diagnosis and a pulmonary arteriovenous malformation in the upper left lobe as demonstrated by CT of the chest. The fistula was occluded and the cough disappeared rapidly but resumed in 1994. Recanalisation of the fistula led to a new embolisation procedure, and the cough disappeared. Similar episodes occurred in 1998 and 2004, leading to embolisation of a fistula in the right lower lobe and reperfused fistula in the upper left lobe, respectively. The patient was referred to our research team in 2010 because of reappearance of her dry cough that was more pronounced during exercise and exposure to volatile irritants, and absent during the night. Despite extensive investigations, no cause was found other than reperfusion of the fistula in the left upper lobe. The malformation was not accessible to embolisation, leading us to recommend surgical excision of the malformation. A surgeon undertook atypical resection of the left upper lobe in 2012. The cough disappeared immediately after surgery and has not recurred. Conclusion Physicians caring for patients with pulmonary arteriovenous malformations should know that a severe, chronic cough can be caused by the malformation. A cough associated with a pulmonary arteriovenous malformation can be treated effectively by embolisation but may resume in cases of reperfusion of the malformation. In our case, the severity of the cough led to surgical excision because embolisation was not possible. The mechanism of action of this cough remains to be determined.