A 9-month and 3-day-old male infant with a chief complaint of "intermittent vomiting blood for 6 h" was admitted to Wuhan Children's Hospital on February 8, 2019. Without obvious incentives, the child presented hemoptysis after coughing with bright red blood a total of four to five times. Three of them were quite serious, and blood clots could be observed. No fever, rash, hematuria, or syncope was found. The boy was treated with anti-infectious drugs, hemostasis, and fluid supplementation at clinics. Then, he was transferred to our hospital for further treatment. He was hospitalized after hypervolemic treatment in the emergency room. The boy had been in good health, with regular vaccinations and no history of exposure to infectious diseases.
On physical examination, the boy's heart rate was 160 beats per minute, blood pressure was 97/34 mmHg, and respiratory rate was 35 breaths per minute. He was conscious but pale in color, with reduced skin elasticity. There were no bleeding points throughout the body, and the capillary filling time was 2 s. The superficial lymph nodes throughout the body were not swollen and enlarged, and the sclera had no yellow staining. The pupils had normal reflection of light. There was no congestion in the pharynx. The bilateral breathing movements were symmetrical. The lungs were clear. His heart rate was regular and strong, without a murmur. His abdomen was tender. A hemangioma was detected in the abdominal wall, ~1.5 x 1.5 cm in size, and there was no organomegaly. There were no abnormalities in the extremities and no restrictions on movement.
The hemoglobin level was 104 g/L, the hematocrit was 31.6%, the white-cell count was 7.70 x 109/L, and the platelet count was 177 x 109/L. The results of the coagulation test were as follows: the prothrombin time (PT) was 13.5 s, the activated partial thromboplastin time (APTT) was 28.8 s, the thrombin time (TT) was 22.1 s, and the fibrinogen (FIB) level was 1.02 g/L. Liver function tests showed that the total protein (TP) level was 43.5 g/L, the albumin (ALB) level was 26.4 g/L, the total bilirubin (TBIL) level was 3.8 mumol/L, alanine transaminase (ALT) was 31 U/L, and aspartate aminotransferase (AST) was 22 U/L. The X-ray slide of the chest noted bronchitis. Computerized tomography (CT) of the abdomen detected an abnormal density shadow on the left abdominal wall, indicating the possibility of hemangioma. Chest CT showed poor texture, uneven transmission, and ground-glass opacification (GGO) of both lungs. The GGO was more significant in the right lower lobe. Using the CT coronal scan to simulate bronchoscopy, no obstruction signs in the larynx, trachea, or main bronchi were found (Figure 2).
After admission, the infant did not exhibit hematemesis again, although the coughing was still continuous. Black stool was observed once. Methods, such as hemostasis, acid suppression, and other symptomatic treatments, were received. An emergency gastrointestinal examination (Figure 3A) showed that there was patchy erythema near the corner of the stomach with a smooth surface and suspected vascular malformation. A biopsy was taken from the surrounding area. The pathology identified mild chronic inflammation of the mucosal tissue (Figures 3B,C). By conducting a thorough bronchoscopy (Figures 4A-F), we found a smooth mucosal protrusion in the opening of the right lower lobe. It was suspected of being a new formation, and bleeding after contact was obvious (Figure 4F). After spraying epinephrine and thrombin, the bronchoscope was removed until no more bleeding was observed.
After bronchoscopy, the infant was transferred to the intensive care unit. The blood test was rechecked. The hemoglobin level was 70 g/L, the hematocrit was 22.6%, the white cell count was 5.79 x 109/L, and the platelet count was 189 x 109/L. Hemoptysis diminished, and the stool turned yellow. After 5 days, he developed recurrent hemoptysis three times after coughing, with ~20-30 ml of blood each time, accompanied by cyanotic lips and shortness of breath, ~60 times per minute. Emergency tracheal intubation, mechanical ventilation, volume expansion, and hemostasis were given. Urgent blood analysis showed that the hemoglobin level was 81 g/L, the hematocrit was 25.6%, the white cell count was 8.06 x 109/L, and the platelet count was 315 x 109/L. Therefore, under general anesthesia, the right femoral artery was punctured using the Seldinger technique, and a 5F Cobra catheter and a microcatheter were superselectively inserted into the bronchial artery. The angiogram showed that the bilateral bronchial arteries showed distortion; the distal end was disordered, blurred, and irregularly stained; and more contrast agents showed signs of spillage (Figure 5A). The 5F Cobra catheter was placed in the opening of the bilateral bronchial arteries; the microcatheter was inserted superselectively to avoid the intercostal artery; PVA (300 mum) and gelatin sponge particles (560-710 mum) + dexamethasone (5 mg) + contrast mixed embolization agent were used for arterial embolization via microcatheter; and the embolization was stopped when the contrast agent slowed down significantly. Re-examination angiography showed satisfactory embolization of the bilateral bronchial arteries (Figure 5B). Bronchoscopy was performed again after the operation. The new formation at the opening of the right lower lobe showed no significant bleeding. After 3 days, the trachea intubation was removed. The infant was discharged after the chest radiograph was found to be normal.
CT coronal scan of the lung (A,B).