Clinical and experimental studies on respiratory involvement and alterations in immune status were carried out. Respiratory distress occurring in these patients has improved gradually for 14 years but still remains. Copious expectoration at an early stage of the disease may be related to the fact that a number of discrete polychlorinated biphenyls (PCBs) are distributed throughout the lung parenchyma. For accumulation in the bronchial mucosa, structural requirements and specific dose dependence of PCBs have been clearly shown; however, pathological and physiological studies have indicated that respiratory involvement in yusho is mainly small airway disease that may be caused by involvement of cellular component (Clara cells) in bronchioles and/or associated infection. Respiratory distress is often exacerbated by viral or bacterial infection. Changes in the immune status in PCB and polychlorinated dibenzofuran (PCDF) poisoning are as follows: IgA and IgM in the serum are decreased at an early stage of the disease and then return to normal; suppression of cellular immunity was reported in Taiwanese patients and some may remain in the later stages of the disease, as shown in our patients. PCDFs now appear to be the main causal agents in yusho. Rats given PCDFs showed necrosis of the Clara cells in bronchioles and marked thymus atrophy, while few such changes were noted in rats given PCBs. Therefore, further examination is needed for the difference of the toxic effects between two compounds.
A 37‐year‐old female with multiple pulmonary aneurysms, thrombotic obstruction of the inferior vena cava and mural thrombosis in the right heart was reported. Arterial blood culture repeatedly examined had been negative. The patient died of massive hemoptysis. This case was equivalent to Hughes‐Stovin syndrome and was the first female case of typical Hughes‐Stovin syndrome.
Review of the literature revealed 9 typical cases of this syndrome and 5 atypical cases who had solitary intrapulmonary aneurysm. It was obscure whether the typical cases were essentially different from the atypical ones or not, but the cases with solitary pulmonary aneurysm might proceed to the cases with multiple ones, as shown in this case.
Pathogenesis of the syndrome has been controversial. In the present case, development of pulmonary aneurysms seemed to be closely related to thromboembolization derived from venous thrombosis due to artificial abortion.
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