Thoracic Hemangiomatosis Masquerading as Interstitial Lung Disease

Vevaina, James R.; Mark, Eugene J.

doi:10.1378/chest.93.3.657

Cited by 32 publications

(17 citation statements)

References 10 publications

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“…The disease seems to be restricted to the lungs, al though pericardium and mediastinal involvement have been described [4,11], In our patient, characteristic histo pathologic findings were seen in several mediastinal lymph nodes.…”

Section: Discussionsupporting

confidence: 45%

“…Signs of airway ob struction, as reported by Vevaina and Mark [11] and Ma gee et al [12], were not encountered (residual volume was low and specific conductance was normal).…”

Section: Discussionmentioning

confidence: 77%

“…Hemoptysis, a common clinical manifestation that may be useful in the differentiation between pulmo nary capillary hemangiomatosis and pulmonary veno-occlusivc disease [6], was not present in our patient. The def inite diagnosis is established on the basis of pathological findings at autopsy or as a result of pneumonectomy dur ing life [2,3], Open lung biopsy is often misinterpreted [6,11], Radiologic features arc unreliable in separating he mangiomatosis from other diseases. Chest radiographs show interstitial infiltrates [11], and although Wagenaar ct al.…”

Section: Discussionmentioning

confidence: 99%

“…The def inite diagnosis is established on the basis of pathological findings at autopsy or as a result of pneumonectomy dur ing life [2,3], Open lung biopsy is often misinterpreted [6,11], Radiologic features arc unreliable in separating he mangiomatosis from other diseases. Chest radiographs show interstitial infiltrates [11], and although Wagenaar ct al. [3] suggested the possibility of unilateral involvement, we consider that the disease should be characterized as producing bilateral affection.…”

Section: Discussionmentioning

confidence: 99%

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Pulmonary Capillary Hemangiomatosis: Report of a Case and Review of the Literature

Domingo

Encabo²,

Roig³

et al. 1992

Respiration

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We describe a case of pulmonary capillary hemangiomatosis in a 60-year-old woman with a 1-year history of progressive exertional dyspnea. Four years before admission a diagnosis of breast cancer was made, and she underwent mastectomy plus radiation therapy and treatment with oral antiestrogens. The chest X-ray showed bilateral interstitial infiltrates. Pulmonary function studies revealed a severe restrictive pattern. Abundant red blood cells were found in the bronchoalveolar lavage fluid. On the basis of open lung biopsy, interstitial fibrosis was diagnosed. Cardiac catheterization revealed pulmonary hypertension. Steroids were prescribed, but the patient’s condition continued to deteriorate and she died approximately 3 years after presentation. The identification of proliferating and invasive capillaries, which are unique to pulmonary capillary hemangiomatosis, led to the correct diagnosis at autopsy.

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Section: Discussionsupporting

confidence: 45%

“…Signs of airway ob struction, as reported by Vevaina and Mark [11] and Ma gee et al [12], were not encountered (residual volume was low and specific conductance was normal).…”

Section: Discussionmentioning

confidence: 77%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

See 2 more Smart Citations

Pulmonary Capillary Hemangiomatosis: Report of a Case and Review of the Literature

Domingo

Encabo²,

Roig³

et al. 1992

Respiration

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“…It is a histological diagnosis based on the presence of numerous foci of proliferating thin walled capillaries which invade lung alveolar tissue, pleura, and bronchial and vascular walls. Since this lesion was first described by WAGENVOORT and co-workers in [1] 1978, 23 cases have been reported in the literature, 17 of which were diagnosed at autopsy, making the actual prevalence of PCH difficult to estimate [1][2][3][4][5][6][7][8][9][10][11][12][13][14][15] ABSTRACT: A 24 yr old white female presented with dyspnoea, orthopnoea, paroxysmal nocturnal dyspnoea, cough and fatigue. Transthoracic echocardiography revealed a sinus venosus atrial septal defect (ASD).…”

mentioning

confidence: 99%

Pulmonary capillary haemangiomatosis coexistence with sinus venosus ASD: morphometric analysis and literature review

Slovis

Чазова²,

Loyd³

et al. 1998

Eur Respir J

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aaPulmonary capillary haemangiomatosis (PCH) is a rare vascular abnormality confined to the lung which results in the development of chronic pulmonary hypertension. It is a fatal disease which is difficult to diagnose antemortem because the clinical features may be identical to those of primary pulmonary hypertension (PPH) or pulmonary venoocclusive disease (PVOD). It is a histological diagnosis based on the presence of numerous foci of proliferating thin walled capillaries which invade lung alveolar tissue, pleura, and bronchial and vascular walls. Since this lesion was first described by WAGENVOORT and co-workers in [1] 1978, 23 cases have been reported in the literature, 17 of which were diagnosed at autopsy, making the actual prevalence of PCH difficult to estimate [1][2][3][4][5][6][7][8][9][10][11][12][13][14][15] ABSTRACT: A 24 yr old white female presented with dyspnoea, orthopnoea, paroxysmal nocturnal dyspnoea, cough and fatigue. Transthoracic echocardiography revealed a sinus venosus atrial septal defect (ASD). Right heart catheterization confirmed severe pulmonary hypertension (80/37 mmHg). A chest radiograph showed enlarged pulmonary arteries with peripheral pruning. Surgical repair of the ASD and lung biopsy were performed. Two days later, she developed right heart failure and was treated with inhaled nitric oxide and then a calcium channel blocker. She failed to improve and was readmitted three months later with severe right heart failure and progressive dyspnoea. While waiting for lung transplantation, she developed haematochezia and died. Light microscopy of lung biopsy and autopsy tissue revealed the structural changes of pulmonary hypertension and focal increases in congested pulmonary capillaries consistent with the diagnosis of pulmonary capillary haemangiomatosis. Quantitative analysis demonstrated that the pathological changes were rapidly progressive. Eur Respir J 1998; 12: 240-244.

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Pathological Case of the Mo012

Weatherly

Stark

Rao

et al. 1992

Arch Pediatr Adolesc Med

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GRAVIDA 2, para 1 mother at 23 weeks' gestation delivered vaginally on termination of pregnancy a 520 g stillborn female fetus (Figure 1) with multiple skeletal and central nervous system anomalies. The pregnancy was uncomplicated without signs or symptoms of infection. The mother denied tobacco, alcohol, or other drug use. Postmortem radiographic studies (Figure 2) disclosed short lower extremities with anterior bowing of the femurs, tibias, and fibulas as well as 11 paired thin ribs. At autopsy, SPECIAL FEATURE

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Thoracic Hemangiomatosis Masquerading as Interstitial Lung Disease

Cited by 32 publications

References 10 publications

Pulmonary Capillary Hemangiomatosis: Report of a Case and Review of the Literature

Pulmonary Capillary Hemangiomatosis: Report of a Case and Review of the Literature

Pulmonary capillary haemangiomatosis coexistence with sinus venosus ASD: morphometric analysis and literature review

Pathological Case of the Mo012

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