Ochi H, Aizawa H, Matsumoto K, Hashimoto S, Hara N
Research Institute for Diseases of the Chest, Faculty of Medicine, Kyushu University, Fukuoka, Japan.
Nihon Kyobu Shikkan Gakkai Zasshi. 1995 May;33(5):576-82.
A 31-year-old man was admitted to our hospital because of a sudden onset of thirst, polyposia, and polyuria. Five years previously he had been admitted to our hospital because of a dry cough. On the first admission, the chest X-ray film had shown reticular shadows and bullous changes in both upper lung fields. Histological examination of a transbronchial lung biopsy specimen had revealed that the nodular lesion in the interstitium of the alveolar lesion consisted of an aggregate of many Langerhans cells with pale cytoplasm and partly convoluted nuclei. In addition, immunoperoxidase stain for S-100 protein had been strongly positive in numerous Langerhans cells in a bone biopsy specimen from a left mandibullar lesion, which is the same histological appearance as the lung lesion. A diagnosis of pulmonary eosinophilic granuloma had been made. The course after discharge was not progressive without treatment for 5 years, but the patient suddenly began to have thirst, polyposia, and polyuria. Dehydration, vasopressin tests, and the findings of MRI indicated diabetes insipidus due to a pathological change in the pituitary gland. Although diabetes insipidus is known to be a common complication of pulmonary eosinophilic granuloma, only 9 cases have been reported in Japan.
一名31岁男性因突然出现口渴、多饮和多尿而入院。5年前，他因干咳入院。首次入院时，胸部X线片显示双上肺野有网状阴影和大疱样改变。经支气管肺活检标本的组织学检查显示，肺泡病变间质中的结节性病变由许多细胞质淡、核部分卷曲的朗格汉斯细胞聚集而成。此外，左下颌骨病变骨活检标本中许多朗格汉斯细胞的S-100蛋白免疫过氧化物酶染色呈强阳性，其组织学表现与肺部病变相同。诊断为肺嗜酸性肉芽肿。出院后5年未经治疗病情未进展，但患者突然出现口渴、多饮和多尿。脱水、血管加压素试验及MRI检查结果提示垂体病变导致尿崩症。虽然尿崩症是肺嗜酸性肉芽肿的常见并发症，但在日本仅报道过9例。
