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Thursday, 28 November 2013

Pulmonary vascular disease – Pulmonary veno-occlusive disease – The Causes

Pulmonary veno-occlusive disease
Pulmonary veno-occlusive disease (PVOD) is an extremely rare form of pulmonary hypertension, affecting mostly in children and young adults as a result of a progressive obstruction of small pulmonary veins that leads to elevation in pulmonary vascular resistance and right ventricular failure.
A. Causes
Although the cause of Pulmonary veno-occlusive disease (PVOD) is considered unknown, some studies suggested the following
1. Oral contraceptives
According to the study by the University of California San Francisco, there ia 2 cases of PVOD that developed in 2 young women soon after the initiation of oral contraceptives (OCs). The first patient is a 14-year-old girl, with no medical history, who started taking an OC 3 weeks before the onset of symptoms. The second patient is an 18-year-old girl, diagnosed 2 years previously with systemic lupus erythematosus and lupus anticoagulant, who started taking an OC 4 months before the onset of symptoms(7).
2. Idiopathic or complicating other conditions
According to the study by the Hôpital Antoine Béclère, Assistance Publique – Hôpitaux de Paris, Université Paris-Sud, Clamart, France, PVOD has been described as idiopathic or complicating other conditions, including connective tissue diseases, HIV infection, bone marrow transplantation, sarcoidosis and pulmonary Langerhans cell granulomatosis(8).
3. Infections, Toxic Exposures, Thrombotic Diathesis, Autoimmune Disorders may also be the possible causes of PVOD
a. Infection and Toxic Exposures
No convincing data have linked PVOD to a specific infectious insult, although an “influenzalike illness” has preceded the development of PVOD in many cases, and serologic evidence suggestive of recent infection with one of several agents (including Toxoplasma gondii and measles) has been documented around the time when PVOD was diagnosed(8a). According to the study by the Service de Pneumo-Allergologie, there is a report of case of a 27 year old male intravenous drug abuser with HIV infection and pulmonary hypertension. Open lung biopsy led to the diagnosis of pulmonary veno-occlusive disease. This second case of pulmonary veno-occlusive disease raises the question of a relationship between HIV infection and lesions involving the pulmonary veins. However, the pathogenesis of vascular changes remains to be elucidated(9). Other study report a case of a Caucasian female with a long history of progressive dyspnoea ultimately diagnosed as focal granulomatous venulitis leading to a pulmonary veno-occlusive disease-like pathology(10).
b. Thrombotic Diathesis
According to the study by research team lead by Tsou E, there is a report of a 23-year-old woman in her 27th week of gestation presented with clinical findings of progressive pulmonary hypertension. After cardiac catheterization she went into labor and was delivered by cesarean section. She died shortly thereafter from right heart failure. Pulmonary venoocclusive disease was found at autopsy. Hemodynamic changes during pregnancy, labor, delivery, and the postpartum period may have contributed to her deterioration and death(11).
c. Autoimmune Disorders
There is a report of a 26-year-old woman with systemic lupus erythematosus (SLE) developed dyspnea and hypoxemia on exertion. She died from rapidly progressive respiratory failure. Autopsy revealed right ventricular hypertrophy and occlusion of the pulmonary veins compatible with pulmonary venoocclusive disease (PVOD), according to the study by Keio University School of Medicine(12).
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Sources
(7) http://www.ncbi.nlm.nih.gov/pubmed/22884388
(8) http://www.ncbi.nlm.nih.gov/pubmed/19118230
(8a) http://www.atsjournals.org/doi/full/10.1164/ajrccm.162.5.9912045#h15
(9) http://www.ncbi.nlm.nih.gov/pubmed/8620974
(10) http://www.ncbi.nlm.nih.gov/pubmed/19251802
(11) http://www.ncbi.nlm.nih.gov/pubmed/6738961
(12) http://www.ncbi.nlm.nih.gov/pubmed/8014950