Approach to diagnosis and pathological examination in bronchial Dieulafoy disease: a case series

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Abstract

Background

There are limited series concerning Dieulafoy disease of the bronchus. We describe the clinical presentation of a series of 7 patients diagnosed with Dieulafoy disease of the bronchus and provide information about the pathological diagnosis approach.

Patients and methods

A retrospective review of patients who underwent surgery for massive and unexplained recurrent hemoptysis in a referral center during a 11-year period.

Results

Seven heavy smoker (49 pack years) patients (5 males) mean aged 54 years experienced a massive hemoptysis (350–1000 ml) unrelated to a known lung disease and frequently recurrent. Bronchial contrast extravasation was observed in 3 patients, combining both CT scan and bronchial arteriography. Efficacy of bronchial artery embolization was achieved in 40% of cases before surgery. Pathological examination demonstrated a minute defect in 3 cases and a large and dysplasic superficial bronchial artery in the submucosa in all cases.

Conclusion

Dieulafoy disease should be suspected in patients with massive and unexplained episodes of recurrent hemoptysis, in order to avoid hazardous endoscopic biopsies and to alert the pathologist if surgery is performed.

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Most cited references 28

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Hemoptysis: etiology, evaluation, and outcome in a tertiary referral hospital.

Boaz Hirshberg, Iftah Biran, Mendel Glazer … (1997)

Hemoptysis, an important and alarming symptom, often indicates serious disease. This study was designed to assess the different causes of hemoptysis, the relative importance of the different diagnostic modalities employed, and the outcome in an Israeli population cohort. A retrospective analysis of 208 patients with hemoptysis at the Hadassah University Hospital, Jerusalem, Israel between January 1980 and August 1995. Bronchiectasis (20%), lung cancer (19%), bronchitis (18%), and pneumonia (16%) accounted for most causes of hemoptysis. In contrast to older studies, active tuberculosis was a rare finding (1.4%). Bronchiectasis and bleeding diathesis were major causes of moderate to severe hemoptysis while bronchitis and lung cancer were commonly associated with milder degrees of bleeding. CT scan was the most sensitive diagnostic test when employed alone, with a positive yield of 67%. However, it failed to locate at least three cases of lung cancer. When combining a CT study together with a bronchoscopy, the positive yield increased to 93%. The mortality rate for patients with mild to moderate hemoptysis was low (2.5% and 6%, respectively), while patients with massive hemoptysis had high mortality rates (38%). Patients with lung cancer or bleeding diathesis had higher mortality rates compared with the rest of the cohort. Hemoptysis is a common symptom with a good prognosis in most cases. However, patients exhibiting massive bleeding or those with lung malignancy and patients with bleeding diathesis had a poorer prognosis. Patients older than 50 years with a positive smoking history need an extensive evaluation and follow-up to exclude lung carcinoma. The combined use of bronchoscopy and chest CT has the best yield in evaluating hemoptysis.

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Clinical assessment and management of massive hemoptysis.

Keith Baptiste (2000)

Massive hemoptysis is a potentially lethal condition that deserves to be investigated thoroughly and brought under control promptly. The mortality rate depends mainly on the underlying etiology and the magnitude of bleeding. Although the diagnosis of hemoptysis may be established by chest radiograph, many pathologies may be missed. Because bronchoscopy and computed tomography are complementary, they may indicate pathologies not detectable by chest radiograph. Finding the etiology and site of the hemoptysis is imperative. Urgent bronchoscopy should be performed in unstable patients because it exacts a paramount role in the diagnostic search and therapy. It can be used to facilitate the introduction of balloon-tip catheters into the bleeding bronchus for tamponade of the hemorrhagic artery, protecting de facto the contralateral lung or nonbleeding bronchi from blood aspiration. Endobronchial tamponade should only be used as a temporary measure until a more specific treatment is instituted. In stable patients, computed tomography should be ordered before any bronchoscopic exploration. Surgery was once regarded as the treatment of choice in operable patients with massive hemoptysis. Bronchial artery embolization (BAE) is an excellent nonsurgical alternative; it is proven to be very effective and lacks the mortality and morbidity encountered in surgical interventions. Nevertheless, surgery is recommended in patients with massive hemoptysis caused by thoracic vascular injury, arteriovenous malformation, leaking thoracic aneurysm with bronchial communication, hydatid cyst, and other conditions in which BAE would be inadequate. MEDICAL MANAGEMENT: Conservative medical therapy may suffice in certain conditions, like bronchiectasis, coagulopathies, Goodpasture's syndrome, and acute bronchopulmonary infections. Preparation for other interventions (endobronchial tamponade, BAE, or surgery in eligible candidates) should be undertaken if the bleeding fails to respond to conservative measures. Supportive therapy should be applied vigorously to all patients with massive hemoptysis.

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Immediate and long-term results of bronchial artery embolization for life-threatening hemoptysis.

Hervé Mal, Isabelle Rullon, Francois Mellot … (1999)

Bronchial artery embolization (BAE) has been established as an effective technique in the emergency treatment of life-threatening hemoptysis, but few data concerning long-term results and complications of the procedure are available. The aim of this study was to analyze retrospectively the experience of BAE in our center with particular emphasis on medium-term and long-term results and on morbidity. University hospital. Fifty-six patients underwent bronchial arteriography from 1986 to 1996 in our center for the management of life-threatening hemoptysis. Of them, BAE was performed in 46 patients. Their mean age was 51 years (range, 19 to 89 years). The most frequent etiologies of hemoptysis were active or inactive tuberculosis, bronchiectasis, or idiopathic hemoptysis. BAE resulted in an immediate cessation of hemoptysis in 43 of the initial 56 patients (77%). During the first month after BAE, four patients who died from causes other than hemoptysis or who were referred to surgery were excluded from follow-up and in the 39 remaining patients, a complete cessation of hemoptysis was observed in 32 patients. A remission was noted in 28 of the 29 patients followed up between 30 and 90 days after BAE. Long-term control of bleeding was achieved in 25 of the initial 56 patients (45%) followed up beyond 3 months after BAE (median follow-up of 13 months; range, 3 to 76 months). Overall, complications of BAE consisted of two episodes of mediastinal hematoma and three episodes of neurologic damage, two of which improved without permanent sequelae. We conclude that BAE may result in long-term as well as immediate control of life-threatening hemoptysis but that complications are not unusual.

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Author and article information

Journal

Journal ID (nlm-ta): Respir Res

Title: Respiratory Research

Publisher: BioMed Central

ISSN (Print): 1465-9921

ISSN (Electronic): 1465-993X

Publication date (Print): 2008

Publication date (Electronic): 5 August 2008

Volume: 9

Issue: 1

Page: 58

Affiliations

[1 ]Service de Pneumologie et Unité de Réanimation Médicale, Hôpital Tenon, Assistance Publique – Hôpitaux de Paris and Université Pierre et Marie Curie, Paris, France

[2 ]Service d'Anatomie Pathologique, Hôpital Tenon, Assistance Publique – Hôpitaux de Paris and Université Pierre et Marie Curie, Paris, France

[3 ]Service de Radiologie, Hôpital Tenon, Assistance Publique – Hôpitaux de Paris and Université Pierre et Marie Curie, Paris, France

[4 ]Service de Chirurgie Thoracique, Hôpital Tenon, Assistance Publique – Hôpitaux de Paris and Université Pierre et Marie Curie, Paris, France

Article

Publisher ID: 1465-9921-9-58

DOI: 10.1186/1465-9921-9-58

PMC ID: 2529287

PubMed ID: 18681960

SO-VID: ba85903f-46cd-47df-a650-f95f980d4578

License:

This is an Open Access article distributed under the terms of the Creative Commons Attribution License ( http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Approach to diagnosis and pathological examination in bronchial Dieulafoy disease: a case series

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Abstract

Background

Patients and methods

Results

Conclusion

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Biomarkers for Inflammatory Lung Disease

Most cited references 28

Hemoptysis: etiology, evaluation, and outcome in a tertiary referral hospital.

Clinical assessment and management of massive hemoptysis.

Immediate and long-term results of bronchial artery embolization for life-threatening hemoptysis.

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