[Case of protein-losing gastroenteropathy associated with scleroderma in which central serous chorioretinopathy developed].

A 44-year-old woman noticed edema of the lower limbs in May 1999 and visited our hospital in September 1999 to undergo further examination. On admission, severe hypoalbuminemia (1.9 g/dl) was detected with a negative urinary protein level. Protein leakage into the gastrointestinal tract and deposition of immune complex in the colonic mucosa were shown by the fluorescent antibody method. In addition, anti-centromere antibody, sclerodactyly, and findings indicative of histological sclerotic changes on a skin biopsy were observed. These findings supported a diagnosis of protein-losing gastroenteropathy complicated by scleroderma. Administration of oral corticosteroids was begun one month after admission and the patient experienced diminished visual acuity immediately after steroid pulse therapy in November. Central serous chorioretinopathy (CSC) was diagnosed at the Department of Ophthalmology of our hospital, and the administration of corticosteroids was suspected as a possible cause of CSC. Considering the severity of hypoproteinemia, the corticosteroid treatment was continued despite corticosteroids being strogly suspected as the primary cause of CSC. A complete disappearance of CSC was achieved in 30 days after the onset of symptoms despite continuation of the steroid therapy, and her serum albumin and complement levels both normalized. We concluded that damage to the retinal pigment epithelium secondary to the vascular lesion at the choroidal level plays a causative role in CSC. In the present case, the findings suggested that the deposition of immune complex in choroidal tissues as well as the gastrointestinal tract caused hyperpermeability of choroidal vessels and led to the development of CSC.

[Polyarthralgia as the initial manifestation of isolated ACTH deficiency].

A 57-year-old man, employed as a taxi driver, noticed arthralgia of his fingers beginning in May 1999. He was unable to work due to the arthralgia and the accompanying general malaise and anorexia, and was thus admitted to a local hospital in July 1999. Since a diagnosis of rheumatic disease was suspected due to elevated inflammatory reactions and joint symptoms, he was referred to our hospital in September 1999. Although no joint swelling was observed, severe tenderness was present in both the fingers and wrists. His grasping power had decreased markedly and fever was intermittently observed. All autoantibodies aside from antinuclear antibody were negative. Given that hyponatremia (126 mEq/l) and fasting hypoglycemia were demonstrated, an endocrinological examination, in particular for hypopituitary-adrenal function, was performed. Both plasma and urinary cortisol concentrations were very low, and an associated low concentration of plasma ACTH (6.0 pg/ml) was noted. The ACTH circadian rhythm was absent and there was no response to the administration of corticotropin releasing hormone. All other pituitary hormones were secreted at normal levels and brain MRI revealed a normal appearance of a pituitary gland. Based on these findings, the patient was diagnosed as having isolated ACTH deficiency. Arthralgia and general malaise both improved soon after replacement of glucocorticoid, and CRP levels were normalized. Isolated ACTH deficiency should be considered in the differential diagnosis of patients suffering from polyarthralgia, given that fever and increased inflammatory reactions occasionally develop and that rheumatic symptoms are also present, as in the present case.