Primary racemose hemangioma of the bronchial artery

A 44-year-old woman was admitted to our hospital for repeated hemoptysis. Selective angiography of the bilateral bronchial arteries showed them to be enlarged and convoluted, and revealed shunts between the bronchial and pulmonary arteries. The patient was diagnosed as having racemose hemangioma of the bronchial artery. Although embolization of the bronchial artery with gelatin sponges was performed, hemoptysis emerged again about one year later. Finally, segmentectomy of the right lower lung was performed and primary racemose hemangioma of the bronchial artery was histopathologically confirmed.     

Primary racemose hemangioma of bronchial artery]

A 38-year-old woman was admitted to the hospital because of massive hemoptysis. A chest X-ray film disclosed an infiltrative shadow. A bronchoscopic examination revealed a small, pulsatile, papillary protrusion in the orifice of the middle lobe bronchus, and massive bleeding from the protrusion was observed during the examination. Bronchial arteriography showed convolution and mild hypervascularization of the right bronchial artery. A middle lobectomy was performed, and the protrusion was histologically shown to be a primary racemose hemangioma of the bronchial artery.     

A case of primary racemose hemangioma of bronchial artery with recurrent hemoptysis]

A 42-year old man was admitted to our hospital because of hemoptysis. Bronchial arteriography revealed a tortuous and dilated left bronchial artery with a shunt formation between the bronchial and pulmonary arteries. Bronchial artery embolization using a sponge was performed three times to treat the hemoptysis, but all attempts failed. The patient therefore underwent left lower lobectomy, after which no hemoptysis was observed. Histopathologically, the resected tissue showed no inflammatory change. Interestingly, abnormal vessels resembling arteriovenous malformations were also found. Although the embolization therapy was effective in several reported cases, we concluded that surgery was required for this patient with persistent hemoptysis because of the development of collaterals and a bronchial-pulmonary artery shunt.     

A case of successful bronchial artery embolization for primary racemose hemangioma with massive hemoptysis]

We encountered a case of primary racemose hemangioma treatment with successful bronchial artery embolism for massive hemoptysis. A 56-year-old woman with massive hemoptysis was transported to our hospital. The source of the massive hemoptysis was observed to be from around a non-pulsatile polyp covered by normal mucosa occluding the truncus intermedius by fiberoptic bronchoscopy. We stopped the bleeding temporarily using differential lung ventilation, and then bronchial artery angiography was performed. The main right bronchial artery was enlarged, and enlarged and convoluted right peripheral bronchial vessels were also observed. We diagnosed the massive bleeding to be due to racemose hemangioma. A successful bronchial artery embolization (BAE) was performed with gelforms and metallic coils for the treatment of racemose hemangioma. There has been no recurrence of hemoptysis for one year after BAE. There have been many reports on massive hemoptysis as in this patient who were treated by lobectomy, nevertheless we would like to state BAE should be considered as a suitable treatments for primary racemose hemangioma with hemoptysis if there is no recognizable shunt artery.     

A case of asymptomatic racemose hemangioma treated by bronchial artery embolization]

A 69-year-old man had an abnormal shadow on chest X-ray and bronchoscopic examination showed that left B4 was completely occluded by a tumor. A non-pulsatile polypoid nodule was also found in right B'. The tumor in the left B4 was diagnosed as carcinoid, but the nodule in right B' was suspected to be hemangioma and biopsy was not performed. Bronchial arteriography revealed hypervascularization with dilated vessels distributing to the lingular lobe and convoluted and a dilated bronchial artery extending to the right upper lobe. From these findings, we diagnosed racemose hemangioma of the bronchial artery of the right upper lobe. After bronchial artery embolization of the right and left bronchial arteries, he underwent segmentectomy of the lingula and was discharged without complications. Two months after the operation, bronchoscopic examination showed that the racemose hemangioma had shrunk and the swelling in the surrounding mucosa had decreased. If a submucosal small nodule is observed during bronchoscopy, biopsy should be performed after bronchial arteriography, and if the nodule is diagnosed as racemose hemangioma, bronchial artery embolization should be performed.     

Successful surgical ligation and transection of racemose hemangioma of bronchial artery]

We report two cases of bilateral racemose hemangioma in patients with hemoptysis. Case 1 was a 62-year-old woman who visited a local clinic complaining of hemoptysis. Bronchoscopy revealed multiple pulsating tumorous lesions and she was referred to our department. Chest Multidetector CT (MDCT) disclosed bilateral enlarged and convoluted, abnormal bronchial arteries and primary racemose hemangioma was diagnosed. Bronchial artery embolization was selected, but not conducted because of concern regarding the arteriovenous shunt and escape into the greater circulation. In combination with thoracoscopic mini-thoracotomy, ligation and separation of the bronchial artery were performed. Case 2 was a 68-year-old man who was transferred to our department with a chief complaint of hemoptysis. MDCT revealed bilateral bronchiectasis and a convoluted and enlarged, abnormal bronchial artery along the mediastinum. He was diagnosed as having secondary racemose hemangioma. First, bronchial artery embolization was conducted, but hemostasis was difficult, thus surgical ligation was conducted. In both cases, MDCT was effective for diagnosis and surgical ligation is very important as a therapeutic option for racemose hemangioma.     

Successful thoracoscopic ligation and transection of racemose hemangioma of bronchial artery]

We used thoracoscopy for the successful ligation and transection of a racemose hemangioma of bronchial artery. The patient was a 61-year-old woman who had been admitted to our hospital because of hemoptysis. Bronchoscopic examination revealed bulging lesions covered with normal bronchial mucosa in the right B5 and B8, and bronchial arteriography revealed a shunt between the right bronchial arteries and pulmonary arteries and veins. Ligation and transection of the right bronchial artery under thoracoscopy was performed. Hemoptysis has not recurred 9 months after the operation. Thoracoscopic ligation and transection of bronchial artery may be an effective and less invasive procedure for the treatment of racemose hemangioma.     

Selective gelfoam embolization of primary racemose haemangioma of the bronchial artery

A 54-year-old male presented with haemoptysis. Bronchial arteriography revealed a bent, meandering and dilated bronchial artery with vascular hyperplasia in multiple locations. The patient was diagnosed as having primary racemose haemangioma of the bronchial artery. Using a microcatheter, TorconNB (5 Fr) and Progreat (2.7 Fr), selective gelfoam embolization of the descending branch of the right bronchial artery was performed using the double catheter method. This approach would allow effective treatment of the haemorrhage and avoid spinal cord injury.     

Bronchial artery embolization can be effective for primary racemose hemangioma]

We report two cases of primary racemose hemangioma in patients with hemoptysis. In the first, a 59-year-old man had a pulsatile polypoid lesion in the bronchus of the lingula; and in the second, a 24-year-old woman had a nodule in the bronchus of the right lower lobe. In both patients, arteriography of the bronchial artery revealed enlargement and convolution of its branches. Primary racemose hemangioma was diagnosed in both patients. The arteriography also revealed a shunt between the pulmonary and bronchial arteries in the woman. Bronchial artery embolization (BAE) was an effective treatment for the hemoptysis. To date, eight months after the embolization, hemoptysis has not recurred. Bronchoscopy and arteriography of the bronchial artery are both useful for diagnosing primary racemose hemangioma. BAE seems to be effective in treating primary racemose hemangioma.     

Two cases of primary artery dissection

We experienced two cases of primary coronary artery dissection. (Case 1) 55-year-old man had frequent episodes of chest oppression at early morning and midnight. During chest oppression, electrocardiogram showed transient ST-segment elevation in leads II, III, and a VF. Then, he was diagnosed as having angina pectoris. This diagnosis was based on the fact that he presented coronary spastic syndrome. Right coronary angiogram demonstrated an intimal flap and false lumen at segment 3, and primary coronary dissection was confirmed. (Case 2) A 27-year-old woman complained of back pain while taking a bath. Electrocardiogram showed ST-segment elevation and abnormal Q in leads V2, V3 and V4. She was diagnosed as having acute anterior wall myocardial infarction. Presence of coronary artery dissection at segment 6 was identified by left coronary angiogram. Primary coronary artery dissection is clinically diagnosed by coronary angiogram very rarely. Only 27 such cases have been reported. It was speculated that, in case 1, vasospastic angina may be associated with primary coronary artery dissection. Case 2 had primary coronary artery dissection at segment 6 of the left anterior descending artery. Thus, her clinical picture was similar to those of previously reported cases.     

A case of racemose hemangioma of a bronchial artery complicated with atrial septal defect and localized peripheral ground-glass opacity]

We report a case of racemose hemangioma of the right bronchial artery complicated with an atrial septal defect (ASD) and ground-glass opacity localized in the right upper lung lobe. A 42-year-old woman was admitted for the pre-operative assessment of ASD. Aortography and selective angiography revealed that the right bronchial artery was markedly dilated and convoluted, draining into the right intercostal arteries and the right upper pulmonary vein. The patient was suffering from exertional dyspnea, but produced no bloody sputum. Since videobronchoscopy showed normal mucosal findings without bronchiectasis, primary racemose hemangioma of the right bronchial artery was diagnosed, and was treated by ligation of the bronchial artery and patch-closure of the ASD. The ground-glass opacity observed preoperatively in the periphery of the right upper lung lobe vanished after surgery. This opacity was considered to have been localized pulmonary congestion due to augmented hydrostatic pressure in the right upper pulmonary vein, induced by increased pulmonary blood flow through the ASD and the drainage route of the racemose bronchial artery into right upper pulmonary vein.     

Two cases of primary pulmonary osteosarcoma

Two cases of primary pulmonary osteosarcoma are presented. In both cases, chest computed tomography revealed a calcified pulmonary mass and technetium-99m methylene diphosphonate bone scintigraphy showed intense uptake in the pulmonary mass. Primary pulmonary osteosarcoma was suspected on the basis of these radiographic findings. Microscopic examination of tumor specimens obtained by needle biopsy revealed histologic features of osteosarcoma, and this diagnosis was confirmed by postmortem examination of a second specimen in each case. Radiographic and histopathological findings enabled us to diagnose primary pulmonary osteosarcoma, which is one of the rarest types of cancer.     

Two cases of Kimura's disease associated with bronchial asthma]

We encountered two rare cases of Kimura's disease associated with bronchial asthma presenting eosinophilia and hyperimmunoglobulinemia E. Patient 1 was a 26-year-old man who had been admitted to our hospital with recurrent increase in left parotid mass in May 1997. He had previously undergone surgery for local excision at another hospital in September 1987; the excised specimens were re-evaluated and the diagnosis of Kimura's disease was confirmed. Because the patient was suffering from an acute asthma attack on admission, prednisolone (PSL) 30 mg/day was administered orally. PSL reduced the parotid mass and improved control of the asthma. Patient 2 was an 18 year-old man who had been given a diagnosis of Kimura's disease on the basis of histologic findings from a biopsy specimen of a subcutaneous tumor in the left cheek in 1988. Following the diagnosis, the patient was treated with methotrexate for the first several months, and then with loxioprofen for 9 years, but the size of the mass remained unchanged. Bronchial asthma developed in this patient in 1995 and had been treated with theophylline. However, because this therapy caused a deterioration of asthma control, the patient was admitted to our hospital in October 1997 for the treatment of bronchial asthma. Inhaled corticosteroids (beclometasone 0.8 mg/day) in addition to theophylline alleviated the patient's asthma symptoms and yielded improved lung function. Because few cases of Kimura's disease associated with bronchial asthma have been reported, patients with eosinophilia and hyperimmunoglobulinemia E were not necessarily considered at high risk for the onset of bronchial asthma.     

Two cases of bronchial asthma diagnosed as hyperventilation syndrome]

We report two cases of bronchial asthma masked by hyperventilation syndrome. Case 1 was a 43-year-old woman. She had repeatedly visited the emergency room complaining of dyspnea and had been given a diagnosis of hyperventilation syndrome for 8 years. Initially, her symptom was only coughing, however, dyspnea gradually appeared. We suspected her symptoms were caused by bronchial asthma on the basis of her wheezes, and we established a diagnosis based on the results of the airway reversible test. Case 2 was a 24-year-old woman. She also had repeatedly visited the emergency room, supposedly because of hyperventilation syndrome over the long term. The airway reversible test revealed that she had features of asthma. After treatment for bronchial asthma, the asthma attacks subsided. After that, neither patient made any emergency visits. We should auscultate the respiratory sounds of patients with hyperventilation syndrome carefully. Additionally, we may as well apply the spirometries and the airway reversible test during a period of stability to a patient who has repeated hyperventilation attacks.     

原发性支气管肺淀粉样变三例临床分析

目的 探讨原发性支气管肺淀粉样变的流行病学、诊断、治疗和预后。方法 对3例病理确诊为原发性支气管肺淀粉样变病人的临床表现、实验室检查及治疗进行分析总结。结果 3例病人均有发热、咳嗽咳痰、胸闷气急,均经纤支镜活检,见均质嗜伊红物沉积于支气管粘膜下,刚果红染色阳性,经MP方案、抗菌药、手术及支持治疗后病情均明显好转。结论 淀粉样变常累及呼吸系统,但临床少见,诊断依靠病理活检,目前尚无特异性治疗。