Unusual Complication of Pulmonary Arteriovenous Aneurysm: Intrapleural Rupture

Abstract

Arteriovenous aneurysm of the lung may give rise to sequelae such as cyanosis, clubbed fingers and toes, polycythemia, dyspnea, hypertrophy of the heart and neurological changes resulting from paradoxical embolic processes. A true complication of arteriovenous aneurysm is a rupture, which can result in hemoptysis or hemothorax. Hemoptysis due to intrabronchial rupture is a relatively frequent complication. According to Leutscher (1954) - in a monograph presenting a detailed analysis of 130 of the cases of arteriovenous aneurysm published up to 1954 hemoptysis is mentioned as a complication in 25 per cent of cases. Hemoptysis occurred in three of the five cases hitherto reported from this clinic. Intrapleural rupture of the arteriovenous aneurysm, resulting in hemothorax, however, proves to be an exceedingly rare complication which Leutscher found mentioned in only three of the 130 cases; it has not previously occurred in our material. This paper describes a woman showing this unusual complication. The description is given both in view of the rarity and possible difficulties in diagnosing this condition, and in view of the fact that an emergency operation in recognized cases may be life saving. CASE REPORT A woman, aged 37, had Rendu-Osler disease and frequent epistaxis. In 1946 (at age 25) she began to cough, with occasional expectoration of blood-tinged sputum. In 1947, while walking in the open, she suffered a violent hemoptysis resulting in a collapse. X-ray film examination of the thorax at the time revealed a homogeneous, virtually round opacity with a diameter of about one inch beside the hilum of the left lung. There was a markedly positive Mantoux tuberculin reaction. There was no fever, nocturnal hyperhidrosis or emaciation. Repeated testing of sputum and fasting gastric contents for acid-fast rods remained negative. The process was nevertheless regarded as tuberculosis, and she was given treatment accordingly, by rest for 18 months. Subsequently she remained asymptomatlc for several years. About 1950, she began to notice that dyspnea ensued more rapidly than previously. During the last five months preceding our examination, the left hemithorax was painful both in repose and during movements “as 11 Inflamed.” The pain radiated to the back, between the shoulder blades. We saw a slightly cyanotic, somewhat dyspneic woman in good nutritional condition, with small telanglectases on the tip of the tongue, the upper and the lower lip and in the nail bed of several fingers. Fingers and toes showed no sign of clubbing. Physical examination of the thorax (Cardiological Department, Prof. Dr. D. Durrer) yielded the following findings. Lungs: vesicular respiration; resonant percussion sounds; lung-liver borderline: sixth rib in front and 12th thoracic posteriorly. There was moderate displacement of the pulmonary outline. Heart: one flngerbreadth enlargement to the left, with a diastolic souffle (grade 1) at the apex, A2 of greater intensity than P2. In the left anterior second intercostal space and on the back, medial to the apex of the right scapula, a vascular murmur was audible which occurred late during systole and did not resemble a typical diamond-shaped “stenotic” souffle. Pulmonary function was normal (vital capacity 116 per cent; one-second value 78 per cent). ECG: intermediary position of the heart; no anomaly. The oxygen saturation in the brachial artery was 92.5 per cent In repose, 88.5 per cent after two minutes’ effort, and 97.5 per cent after 10 minutes’ oxygen respiration. X-ray film examination revealed an opacity in the left upper and the right lower lobe; It appeared to be homogeneous, with smooth contours and showing pulsations at fluoroscopy. The condition was diagnosed as bilateral pulmonary arterlovenous aneurysm. This diagnosis was confirmed by angiocardiography. In the left upper and the right lower lobe, sFi.om the Surgical Clinic A of the Wilhelmina Gasthuis, University of Amsterdam.