原発性の腸管非定型抗酸菌症の1例―A New Clinical Entity

Primary Atypical Mycobacteriosis of the Intestine: A New Clinical Entity, Report of a Case

要旨　患者は23歳の女性．主訴は右下腹部痛と発熱．現症として四肢に結節性紅斑，会陰部に皮膚硬結を認めた．検査で貧血と炎症所見を認めた．ツ反陽性で，胸写と上部消化管X線検査は正常．注腸検査でBauhin弁は開大し，回腸末端は拡張していた．盲腸から上行結腸(近位半分)の管腔は著明に狭窄し，その粘膜面には半球状の隆起がみられた．横行結腸にも2病巣が飛躍して存在し，各々潰瘍瘢痕と炎症性ポリープの肉眼所見を呈していた．大腸生検でLanghans型巨細胞を伴う非乾酪性類上皮細胞肉芽腫が認められた．局所の腸液および生検材料の塗抹標本より，多数の非定型抗酸菌が検出された．病態は抗結核療法で改善した．原発性の腸管非定型抗酸菌症の報告は，国内外ともこれまで皆無で，new clinical entityと思われ，その最初の症例を報告した．

　A 23-year-old woman was admitted to our hospital complaining of dull pain in the right lower quadrant of the abdomen of several months' duration. She had erythema nodosum in the extremities and a hard granulomatous nodule in the perianal area. Laboratory findings were as follows: anemia, positive inflammatory reaction, and positive reaction against PPD. A chest x-ray film and an upper GI series showed no abnormality. A barium enema revealed a broad circular stenosis with a cobblestone appearance from the caecum to the ascending colon, destruction of Bauhin's valve, dilatation of the terminal ileum, ulcer scars in the righttransverse colon and inflammatory polyps in the lefttransverse colon. Biopsy specimens showed noncaseous epithelioid cell granulomas with Langhans' giant cells. A large number of atypical mycobacteria were demonstrated by Ziehl-Neelsen staining of the biopsy material and confirmed by culture. Atypical mycobacteria were considered pathogenic and the patient's general condition improved after the treatment with INH, RFP and SM. This is probably the first case of primary atypical mycobacteriosis of the intestine, representing new clinical entity, in the world.