Pathologic bronchial vasculature in a case of massive hemoptysis due to chronic bronchitis

The cause of bleeding in a patient with recurrent massive hemoptysis was not apparent after bronchoscopy and gross examination of the lobectomy specimen. Histologic submission of all major bronchi uncovered dilated, tortuous bronchial arteries just below the bronchial mucosa with sites of both current and healing arterial rupture. This bronchial arterial abnormality is common to several chronic pulmonary diseases, but is rarely diagnosed as a cause of massive hemoptysis. Careful pathologic examination of major bronchi in the setting of hemoptysis of unknown causation is recommended.     

Churg–Strauss syndrome associated with leukotriene receptor antagonists (LTRA)

Churg–Strauss syndrome (CSS) is a rare vasculitic disorder that generally occurs in patients with bronchial asthma. CSS is being increasingly recognized in asthmatic patients treated with leukotriene receptor antagonists. However, the nature of this relationship remains to be elucidated. The present report describes three asthmatic patients who developed clinical manifestations highly suggestive of CSS, although one patient lacked the presence of eosinophilia. The patient, however, exhibited biopsy-proven cutaneous necrotizing vasculitis, which improved after withdrawal of montelukast. The second patient presented with systemic constitutional signs including fever, malaise, arthralgias, clinical jaundice, peripheral blood eosinophilia, and biopsy-proven eosinophilic hepatitis. The third patient also had circulating eosinophilia, scleritis, and arthritis. All patients improved after discontinuation of the leukotriene receptor antagonist (montelukast).     

A case of massive hemoptysis occurring after lung injury due to a torn segment of pleural calcification

A 64-year-old man was referred to our hospital because of little improvement of hemoptysis lasting three days after drug therapy. A chest roentgenogram and fiberoptic bronchoscopic examination performed on the second hospital day when the patient experienced a massive hemoptysis of about 2,000 ml revealed arterial bleeding from the left upper lobe. Even after extensive embolizations of the left upper bronchial, the first, second, third and 4th intercostal arteries, the patient's hemoptysis did not improve. On the 73rd hospital day the patient underwent left upper lobectomy. Macroscopic and microscopic examinations in the resected specimen revealed lung injury due to a torn segment of pleural calcification after tuberculous pleuritis, resulting in the massive hemoptysis. Although physicians encounter many patients complaining of hemoptysis and/or hemosputum, this case is considered to be very rare.     

Preprocedural Planning with Prospectively Triggered Multidetector Row CT Angiography Prior to Bronchial Artery Embolization in Cystic Fibrosis Patients with Massive Hemoptysis

Study Objectives  

The aim of this study was to determine if electrocardiographically synchronized, prospectively triggered multidetector row computed tomography (ECG-MDR-CT) angiography of the aorta can accurately predict the location of ectopic bronchial arteries in patients with cystic fibrosis (CF) with massive hemoptysis prior to bronchial artery embolization (BAE).     

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.     

支气管Dieulafoy病活检大出血窒息复苏成功1例并文献复习

目的 探讨支气管Dieulafoy病临床表现、诊断及治疗方法.方法 结合我院收治的1例支气管Dieulafoy病的临床资料及国内外文献报道的病例进行综合分析.结果 患者男性,70岁,反复间断咯血30年,大咯血8 h住院,胸部平片及CT未发现异常,行支气管检查发现气管黏膜呈"蚯蚓"样突起,活检过程中突然出现大出血窒息,经抢救复苏成功,后经病理及肺血管造影证实气管黏膜血管异常,考虑支气管Dieulafoy病,行栓塞治疗后好转.结论 支气管Dieulafoy病文献报道较少,发病原因不详,可能与支气管肺动脉先天发育异常和(或)后天的慢性炎症刺激、损伤有关,文献报道少可能与认识不足有关.遇有不明原因大咯血需行气管镜检查时,气管镜检查发现气管黏膜呈"蚯蚓"样突起,应想到该病的可能,不要盲目活检,应想到有大咯血窒息可能,如确需活检,应备好抢救措施.     

Churg-Strauss syndrome presenting as spontaneous subarachnoid haemorrhage

Churg–Strauss syndrome (CSS) is a systemic small-vessel vasculitis characterised by the presence of asthma and eosinophilia. Central nervous system involvement (cerebral infarctions or intracerebral haemorrhage) is rare in CSS. Spontaneous subarachnoid hemorrhage (SAH) has been described in other systemic vasculitides. SAH is exceptional in CSS. We present a 47-year-old woman with CSS presenting as a spontaneous SAH with cerebral angiography findings consistent with vasculitis of the basilar artery and without aneurysms or arteriovenous malformations. She received treatment with prednisone and cyclophosphamide, and 2 months later the basilar artery was normal on magnetic resonance angiography. Received: 27 May 2001 / Accepted: 17 November 2001     

Idiopathic hemoptysis in pregnant women: A distinct entity?

In pregnant women, the reported cases of hemoptysis were most often mild and had an identified cause. Between November 2003 and January 2006, three pregnant women at 16-20 weeks gestation were admitted to our respiratory intensive care unit for massive hemoptysis. One of the women had experienced mild hemoptysis, considered as idiopathic, during her first pregnancy, with no recurrence until her second pregnancy. In all three cases, hemoptysis was massive. CT scan after iodine injection did not reveal any cause. Opacification of the bronchial artery showed hyperemia from abnormally dilated and tortuous bronchial arteries. Bronchial artery embolization (BAE) was performed in all three patients, successfully in two. Intravenous vasopressin was used as second-line treatment for recurrent bleeding after BAE in one patient. The women carried the pregnancy to term with delivery of healthy infants. Further complete investigation after the births did not identify any possible local (pulmonary) or general cause of bleeding in these three patients. Although these cases could be considered idiopathic, the close association with duration of pregnancy suggests the hemoptysis may be related to hormonal changes.     

支气管动脉栓塞术治疗148例大咯血回顾性临床分析

目的 分析支气管动脉造影及支气管动脉栓塞术(BAE)的安全性及有效性.方法 回顾性分析我院2009年3月至2016年4月148例因大咯血行BAE患者的病因、临床特点、栓塞术细节、术后结果及并发症.结果 148例行支气管动脉造影及BAE,其中男118例,女30例,年龄14～91岁.活动性肺结核及肺结核相关后遗症如慢性纤维性病变、空洞是大咯血的主要病因,本资料共83例患者与肺结核相关.另外不明原因咯血30例(20.3％).对其所有相关动脉给予评价.造影中出现血管迂曲、肥厚、点状及片状渗出被认为是靶向血管特征.双侧支气管动脉(64例)是本资料最常见的责任血管动脉,其次是右侧支气管动脉(53例)、左侧支气管动脉(23例)、肋间后动脉(23例).栓塞材料中明胶海绵为所有BAE者均有使用.明胶海绵联合聚乙烯醇颗粒28例,明胶海绵联合弹簧圈68例.在148例行BAE治疗患者中,120例术后立即止血.25例术后再发出血,其中14例接受保守治疗(6例成功,8例死亡),7例再次行BAE,4例行外科手术治疗(3例成功,1例死亡).术后记录到3例严重的并发症,脊髓横贯性截瘫1例,急性青光眼1例,脑梗死1例.共出现63例轻度并发症,胸痛32例和低热31例.结论 BAE对于控制大咯血是有效的治疗方法.重复BAE的有效性及安全性同样值得肯定.但术后严重并发症仍需我们关注.     

Bronchial artery embolization in the management of hemoptysis in cystic fibrosis

Massive hemoptysis and/or recurrent expectoration of measurable amounts of blood are common complications of chronic bronchopulmonary infections in cystic fibrosis (CF). When conservative treatment fails to control bleeding, surgery or bronchial artery embolization (BAE) is frequently considered. We present our experience and long-term follow up of BAE in 14 CF patients (age range 15–39 years) with massive (6 subjects) and/or recurrent (8 subjects) hemoptysis not responsive to medical treatment. Seven had chronic hypercapnic respiratory failure. After angiographic evaluation, polyvinyl alcohol particles (halon) were injected to embolize obviously enlarged bronchial arteries. Seventeen procedures were performed in 14 patients and 36 bronchial arteries were embolized. All the patients stopped bleeding immediately upon BAE. Most of the patients had postembolization fever, dysphagia, and transient chest pain which were managed symptomatically. After median follow-up period of 10.5 months (range 0.5–38 months), no recurrence of hemoptysis was observed in 8 patients who are still alive. In 3 patients hemoptysis recurred and they underwent reembolization after 3,22, and 25 months, respectively. Three subjects died of respiratory failure within 5 months from BAE. Presently, 50% of patients studied had a ≥ 1 year interval free of major hemoptysis after the first BAE. Our experience indicates that massive and/or recurrent hemoptysis in C:F patients can be safety and effectively managed by BAE if the procedure is performed by skilled practitioner. The procedure was well tolerated and resulted in prolonged and satisfactory bleeding control in most patients. © 1995 Wiley-Liss, Inc.     

An aggressive and lethal course of Churg–Strauss syndrome with alveolar hemorrhage,intestinal perforation,cardiac failure and peripheral neuropathy

Churg–Strauss syndrome (CSS) is a rare type of necrotizing vasculitis affecting small to medium-sized vessels typically characterized by asthma, lung infiltrates, necrotizing granulomas and hypereosinophilia. Herein, we describe a case of CSS presenting severe and aggressive course. A 35-year-old male patient with weight loss, dyspepsia, dyspnea and hemoptysis was admitted. The laboratory analyses indicated a remarkable eosinophilia, elevated levels of serum total IgE and positive cANCA. Thorax CT findings were suggestive of alveolar hemorrhage. Bronchoalveolar lavage revealed alveolar hemorrhage with eosinophilia and transbronchial lung biopsy showed eosinophilic vasculitis. Cardiac enzymes were increased and murmurs were audible revealing cardiomyopathy proven by echocardiography. Pulse cyclophosphamide and methyl prednisolone was immediately started. On the 21st day, intestinal perforation developed and urgent surgery was performed. During a follow-up, although a radiological improvement was observed in the chest X-ray, cardiac failure, peripheral neuropathy and skin lesions developed and high-dose intravenous immunoglobulin and anti-TNF therapy (adalimumab) were applied. Despite the therapy, he died from heart failure and septicemia at 68th day of therapy.     

Churg-Strauss syndrome successfully treated with rituximab

Churg-Strauss syndrome (CSS) is characterized by small-vessel vasculitis, extravascular granulomatous inflammation, and massive eosinophilia in tissues and blood, and is often accompanied by a history of preclinical asthma and/or allergic rhinitis lasting several years before the syndrome develops its full clinical picture. Corticosteroids, often in combination with cytotoxic agents, comprise standard therapy; however, a number of patients appear to be resistant to treatment and there is a need for more effective regimens. B cell depletion may be an effective treatment option for CSS. Here, we describe two patients resistant to conventional therapy who were treated with rituximab and experienced a rapid and substantial decrease in disease activity and asthmatic symptoms and an increase in physical capacity.     

Rare manifestations of Churg–Strauss syndrome: coronary artery vasospasm, temporal artery vasculitis, and reversible monocular blindness—a case report

Churg–Strauss syndrome (CSS) is a rare illness with clinical findings characterized by asthma, eosinophilia, and vasculitis affecting medium and small-sized arteries and veins in a variety of organs. Involvement of the temporal arteries by non-giant cell eosinophilic vasculitis in CSS is quite rare and has only been published as isolated case reports or small patient series. Myocardial infarction due to coronary artery vasospasm is an unusual manifestation of CSS. We describe a case of a 39-year-old woman who had two myocardial infarctions due to severe coronary artery vasospasm and was diagnosed with CSS based on a temporal artery biopsy. During the course of her treatment, she also had another rare manifestation of CSS, monocular blindness reversible with immunosuppressive therapy.     

Corynebacterium ulcerans infection of the lung mimicking the histology of Churg-Strauss syndrome

We report the first case of pulmonary Corynebacterium ulcerans infection mimicking Churg-Strauss syndrome (CSS). Productive cough, fever, general fatigue, and weight loss developed in a 50-year-old man. Laboratory data revealed prominent eosinophilia and elevated serum IgE. On chest images, multiple nodules and cavities were predominantly detected in the right lung. Histopathologic examination showed necrotizing granulomas and vasculitis with massive eosinophilic infiltration identical to the findings seen in CSS; however, clusters of Gram-positive, coryneform rods were observed in the alveolar spaces. A toxigenic strain of C ulcerans was isolated from lung tissue. The patient was treated with antibiotics, and a favorable clinical course ensued.     

大咯血的急诊介入治疗

目的探讨支气管动脉栓塞治疗大咯血的适应症、并发症和疗效等。方法分析自2001年7月至2004年7月我院收治的急性大咯血患者63例。24小时咯血量为500-1550ml,平均610ml,以Seldinger技术穿刺右股动脉插管,再行患侧支气管动脉选择性插管,造影并栓塞出血血管。结果栓塞后,所有病例咯血量明显减少,24小时咯血量<50ml,并在3-5天内咯血停止,5例患者栓塞后1-2个月症状复发,行再次栓塞后未再咯血。全组无严重并发症。结论支气管动脉栓塞治疗大咯血安全有效。     

Microscopic polyangiitis complicated with bilateral brachial plexopathy: a case report and review of the literature

Microscopic polyangiitis is a small-vessel necrotizing vasculitis associated with anti-neutrophil cytoplasmic antibodies and presents itself with glomerulonephritis and hemorrhagic pulmonary capillaritis. Peripheral nervous system involvement is common in anti-neutrophil cytoplasmic antibodies-associated vasculitis, but brachial plexopathy is unusual. We present the case of a 22-year-old man with known microscopic polyangiitis who was under maintenance therapy with prednisolone and cyclophosphamide and developed cough, dyspnea, and hemoptysis which increased in 6 days accompanying pain and paresthesia in the upper limbs. His physical examination revealed hypoesthesia, absence of deep tendon reflexes, and decreased muscle strength in the upper limbs. His chest computed tomography scan showed ground glass pattern in the lower and middle lobes. Electromyography and nerve conduction study showed bilateral brachial plexopathy with involvement of all of the cervical roots that were more severe in the lower roots and left side.     

重视肺动脉瘤性大咯血的诊断与介入治疗

肺部高压的支气管动脉与肺静脉构成体循环,低压的肺动脉与肺静脉构成肺循环,支气管动脉、肺动脉和肺静脉三种血管构成肺部两套循环系统。体循环的高压动脉形成动静脉畸形包括支气管动脉-肺静脉畸形和支气管动脉-肺动脉畸形,动静脉畸形的供养动脉、畸形血管团和引流静脉极易形成动脉瘤,动脉瘤破裂引发大咯血。而肺动脉瘤少见,易被漏诊,肺动脉瘤的血供可单独来源于肺循环或体循环,也可同时累及肺循环和体循环,肺动脉瘤破裂大咯血单纯体循环动脉栓塞难以控制。因此,要重视大咯血患者肺动脉瘤的诊断与介入治疗。     

Massive haemoptysis in Beh?et syndrome: case report

Beh?et syndrome in association with pulmonary manifestations is rare. We describe a patient suffering from recurrent oral and genital ulcerations, conjunctivitis, thrombophlebitis and fluctuating radiological opacities in the lungs who died after massive haemoptysis. The autopsy showed a necrotizing vasculitis involving pulmonary arteries, muscular arteries and veins. It was complicated by arterial thromboses, bronchial erosions by pulmonary artery aneurysms and formation of a large arteriobronchial fistula. We conclude that the spread of the inflammatory infiltrate from the wall of the vessels to the adjacent bronchi is responsible for these complications and that haemoptysis must be considered as the expression of dramatic progression of the disease.     

A case of multidrug-resistant (MDR) tuberculosis with collapse of the left lung after hemoptysis

An 82-year-old female was admitted to our hospital with multidrug-resistant (MDR) tuberculosis, defined as resistance to both isoniazid and rifampicin. Chest X-ray showed massive infiltrates with a large cavitary lesions in the left lung field. No antituberculous agents were useful in improving her clinical condition and at 6th months after admission, she exhibited sudden onset of massive hemoptysis, which was successfully treated by bronchial artery embolization. After hemoptysis, her chest X-ray showed collapse of the left lung and computed tomography showed a coagula-like shadow in the left main bronchus, and sputum examination revealed no Mycobacterium tuberculosis colonies. The patient was discharged 5 months after the onset of hemoptysis.