An 11-year-old boy was admitted to the pediatric respiratory department of the Shandong Provincial Hospital due to 2-day history of intermittent mild hemoptysis. Six days earlier, the child had a fever with the highest temperature of 38.0 C. He had no other symptoms, and no examination was performed or treatment was given. Three days before the admission, the child was accidentally injured in the abdomen by the elbow of a classmate while playing. The resulting pain subsided soon, and no special treatment was given. The next day, the boy began coughing up bright red or dark red blood, 2-4 times a day, with a 30-40 mL of blood loss over 24 hours. He did not experience heart palpitations, nosebleed, skin bleeding, coma, hematuria, bloody stool, weight loss, or other symptoms. The boy had been full-term at birth, healthy, and without a history of tuberculosis or measles. At the time of admission, the patient was not febrile and had a respiratory rate of 30 breaths/min, heart rate of 92 beats/min, blood pressure of 107/80 mmHg, and oxygen saturation of 99% in ambient air. The weight was 35 kg. The breath sound of the right lung was slightly lower, and the results of other physical examinations were unremarkable. The complete blood count showed white blood cells 10.18 x 109/L, hemoglobin 141 g/L, and platelets 324 x 109/L, and the C-reactive protein (CRP) level was less than 0.2 mg/L. The coagulation function was normal. The chest radiograph documented right lower pneumonia (Figure 1).
After the admission, the boy was treated by ceftriaxone, vitamin K1, and etamsylate and was told to remain in bed and avoid strenuous activity. On the night of admission, hemoptysis occurred more than 10 times, with 20-30 ml of blood each time. The events were accompanied by obvious chest tightness and pain. Physical examination revealed body temperature 36.8 C, heart rate 120 beats/min, respiratory rate 35 breaths/min, blood pressure 118/92 mmHg, oxygen saturation of 98% in ambient air, and low breathing sounds in the right lung. According to the result of chest radiography and the reduction in the right lung breath sound, the possibility of right lung hemorrhage was considered. The vital signs of the boy were stable, and no signs of respiratory failure were present. Therefore, the patient was subjected to the administration of oxygen via a nasal catheter, ECG monitoring, infusion of hemocoagulase, and continuous administration of pituitrin. He was placed in a right lateral position to protect the airway. Auxiliary examination indicated normal blood count (white blood cells 10.22 x 109/L, hemoglobin 134 g/L, platelets 337 x 109/L). Normal values were obtained in liver function tests, biochemical tests, myocardial enzyme test, urine test, antistreptolysin O test, autoantibodies, immunoglobulin, complement, prothrombin time, activated partial thromboplastin time, and erythrocyte sedimentation rate. The tests for pathogens including bacteria, mycoplasma, tuberculosis, and fungi were all negative. Cardiac ultrasound was normal. Acute CT angiography (CTA) showed that bronchial artery 1 originated from the thoracic aorta to the left at the level of the upper margin of T5 and entered the right hilum above the bifurcation of the trachea. Bronchial artery 2 arose from the right side of the thoracic aorta at T6 and passed up to the right and then down to the right hilum with a 2 mm widening of the primary and middle tube diameters (Figure 2). Bronchial artery 3 originated from the left side of the thoracic aorta under T6 and was divided into two branches going, respectively, to the right and left, without an evident expansion of the lumen. The pulmonary CTA did not identify significant abnormalities. The chest CTA also showed intra-alveolar hemorrhage, partial bronchial stenosis on the right side, atelectasis in the middle lobe, and partial consolidation in the lower lobe. The interventional therapy department physicians concluded that the massive hemoptysis might be caused by bronchial artery dilatation, and bronchial arteriography was recommended to obtain a definite diagnosis, with bronchial artery embolization (BAE) if necessary.
Bronchial arteriography was performed 4 hours after the discontinuation of pituitrin. The imaging demonstrated that the right lower bronchial artery had the same origin as the left bronchial artery, and the vascular lumen was dilated. The right lower lung branches were disorganized, with a small amount of light staining in the parenchymal phase (Figure 3). The distal end of the right inferior bronchial artery was embolized by microspheres combined with a metal coil, this occlusion was confirmed by a repeated bronchial arteriography. Aortic angiography visualized an ectopic bronchial artery originating from the right renal artery and entering the right lower lung, with tortuous and disordered peripheral vessels, and the pulmonary artery branch was apparent in the arterial phase (Figure 4). In consideration of the presence of a bronchial-pulmonary artery fistula, embolization was performed at the distal end of the vessel (Figure 5), and the subsequent revealed that the artery was occluded at the distal end. On this basis, it was concluded that bronchial artery 3 and the ectopic bronchial arteries originating from the kidney were the vessels responsible for the bleeding, and not the slightly dilated bronchial artery 2.
No incidence of hemoptysis occurred after BAE, but the child continued to have paroxysmal chest tightness and chest pain. Subsequent chest X-ray documented atelectasis in the right lung (Figure 6). To avoid rebleeding, bronchoscopy was performed 2 days after the BAE. A large number of blood clots were found in the right main bronchial lumen, the right upper lobe bronchial lumen, and the right middle bronchial lumen. The thrombus was removed using forceps, bronchoscopic brush, and freezing. After the bronchoscopy, the child did not experience chest tightness or other discomforts and occasionally coughed up a small amount of brown blood clot. The chest radiograph did not reveal any evidence of atelectasis on the second day of bronchoscopy (Figure 7). Bronchoscopy performed again 5 days later did not detect hemorrhagic points or bloody sputum suppository. Given the circumstances preceding the admission, the experienced trauma is considered to be the cause of bleeding from the malformed bronchial artery. The child was hospitalized for 10 days. No incidence of hemoptysis occurred during one and a half years of follow-up, and no abnormality was found in the chest radiograph.