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

Jutant EM, Puyo P, El Hajjam M, Blivet S, Houdart E, Aubier M, Lacombe P, Chinet T - BMC Pulm Med (2015)

Bottom Line: Despite extensive investigations, no cause was found other than reperfusion of the fistula in the left upper lobe.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.

View Article: PubMed Central - PubMed

Affiliation: APHP, Department of Pulmonology and Thoracic Oncology, Ambroise Paré Hospital, Boulogne, France. emjutant@gmail.com.

ABSTRACT

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.

No MeSH data available.


Related in: MedlinePlus

CT of the chest in 2012 showing a sub-pleural fistula in the left upper lobe (arrow).
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Fig3: CT of the chest in 2012 showing a sub-pleural fistula in the left upper lobe (arrow).

Mentions: In 2010, the patient was referred to our research team because of the reappearance of a dry cough that was more pronounced during exercise and exposure to volatile irritants, and absent during the night. The cough was so severe that it affected her professional activities, preventing her from contact with the public. Laboratory tests showed a normal blood count and the absence of inflammation. CT of the chest revealed a sub-pleural fistula in the anterior segment of the left upper lobe and a sub-pleural fistula in the right lower lobe (Figure 3).Figure 3


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

Jutant EM, Puyo P, El Hajjam M, Blivet S, Houdart E, Aubier M, Lacombe P, Chinet T - BMC Pulm Med (2015)

CT of the chest in 2012 showing a sub-pleural fistula in the left upper lobe (arrow).
© Copyright Policy - open-access
Related In: Results  -  Collection

License 1 - License 2
Show All Figures
getmorefigures.php?uid=PMC4380104&req=5

Fig3: CT of the chest in 2012 showing a sub-pleural fistula in the left upper lobe (arrow).
Mentions: In 2010, the patient was referred to our research team because of the reappearance of a dry cough that was more pronounced during exercise and exposure to volatile irritants, and absent during the night. The cough was so severe that it affected her professional activities, preventing her from contact with the public. Laboratory tests showed a normal blood count and the absence of inflammation. CT of the chest revealed a sub-pleural fistula in the anterior segment of the left upper lobe and a sub-pleural fistula in the right lower lobe (Figure 3).Figure 3

Bottom Line: Despite extensive investigations, no cause was found other than reperfusion of the fistula in the left upper lobe.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.

View Article: PubMed Central - PubMed

Affiliation: APHP, Department of Pulmonology and Thoracic Oncology, Ambroise Paré Hospital, Boulogne, France. emjutant@gmail.com.

ABSTRACT

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.

No MeSH data available.


Related in: MedlinePlus