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Polyneuropathy and Anasarca: Evidence for a New Connective-Tissue Syndrome and Vasculopathic Contribution

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Grant support: in part by grants AM-12049 and AM-05055 on the National Institute of Arthritis, Metabolism, and Digestive Diseases, Bethesda, Maryland.

▸Requests for reprints should be addressed to A. T. Masi, M.D., Division of Connective-Tissue Diseases, University of Tennessee Center for the Health Sciences, 858 Madison Avenue, Memphis, TN 38163.

Memphis, Tennessee

Ann Intern Med. 1976;84(3):271-274. doi:10.7326/0003-4819-84-3-271
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A 41-year-old woman manifested a polyneuropathy, anasarca, pseudotumor cerebri, hyperhidrosis and hyperpigmentation of the skin, generalized lymphadenopathy, distal esophageal dysphagia, pleuritis, platelike pulmonary atelectasis, fluctuating renal insufficiency, hepatosplenomegaly, amenorrhea, and slight fever suggesting a connective-tissue disorder. Extensive clinical and laboratory evaluation did not support the initial impression of progressive systemic sclerosis or systemic lupus erythematosus but did show nonnecrotizing vascular changes, mild polyclonal gammopathy, and low thyroxin levels similar to the syndrome of polyneuropathy and endocrine disturbances recently reported from Japan. The impressive response to moderate-dose corticosteroids and exacerbation on withdrawal require diagnostic awareness of this insidiously progressive multisystem disorder. A bland vasculopathic process resulting from metabolic or immunologic disturbances appears to be the best explanation for this new syndrome, which has previously been recognized only in Japan.





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