Toshio HAMADA, M. D.
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1 Toshio HAMADA, M. D.
2
3 皮 膚 第10巻 第3号 第1図 昭和43年8月 患児 お よび そ の母 親 を 示 す 第2図 鞍 鼻 が あ り,ロ 造 で,と 第3図 腹 部,陰 股 部 及 び両 下肢 前面 特 に両 下 肢 で は 潮紅,鱗 屑 状 を 強 く呈 し, 破 痕 も著 明 に見 られ る 顔 面 唇 は厚 い 皮 膚 は粗 ころ に よ って 鱗 屑 状 を示 す 第4図 口 部 歯 芽 は 認 め られ な い
4 -356- 皮 膚 第10巻 第5図 頭 部,顔 面 の 骨 レ線 像 上 下顎 に各 4本 の歯 の 胚 芽 が僅 か に認 め られ る, 第3号 昭 和43年8月 第6図 発 汗 試 験 と して1%塩 酸 ピロ カ ル ピ ン0.1cc皮 下 注 射20分 後 の 左 腋 窩 部 ご く僅 か に し め り を 感 じた 程 度 で あ る 第7図 右 腋 窩 部 皮 膚 の 組 織 像,H.E.染 色( 40) 真 皮 に は 脂 線 お よ び エ ッ ク リ ン,ア 両 汗 腺 は い ず れ も全 く認 め ら れ ず,毛 成 不全 を示 して い る 第8図 左 大 腿 部 前 面 の 発 赤,鱗 織 像,H.E.染 屑 状 を示 す 皮 膚 の組 色( 150) 不 全 角 化,有 棘 層 肥 厚,有 棘 層 細 胞 内 の 浮 腫, 表 皮 突 起 の 軽 度 延 長 等 が み ら れ,真 皮上層に は 主 と して リ ン パ 球 か らな る 細 胞 浸 潤 も強 い ポ ク リン 嚢 も形
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6 1) Weech, A. A.: Hereditary Ectodermal Dysplasia (Congenital Ectodermal Defect). A Report of Two Cases, Am. J. Dis. Child., 37: , ) Cole, H. N., Driver, J. R., Griffen, H. K., Norris, C. B. and Stroud, G. : Ectoderml and Mesodermal Dysplasia with Osseous Involvement, Arch. Dermat. & Syph., 44 : , ) Osbourn, R. A. : Congenital Ectodermal Defect with Amastia, J. A. M. A., 148 : , ) Malagon, V. and Taveras, J. E. : Congenital Anhidrotic Ectodermal and Mesodermal Dysplasia. Report of Two Cases with Atrichia and Amastia, A. M. A. Arch. Dermat., 74 : , ) Upshaw, B. Y. and Montgomery, H. : Hereditary Anhidrotic Ectodermal Dysplasia. A Clinical and Pathologic Study, Arch. Dermat. & Syph., 60 : , ) Montgomery, H. : Congenital Ectodermal and Mesodermal Dysplasias, Dermatopathology, I, New York, Harper & Row, 1967, ) Butterworth, T. and Strean, L. P. : Hereditary Anhidrotic Ectodermal Dysplasia, Clinical Genodermatology, Baltimore, Williams & Wilkins, 1962, ) Perabo, F., Velasco, J. A. and Prader, A. : Ektodermale Dysplasie vom anhidrotischen Typus 5 neue Beobachtungen. Helvet. paediatr. acta, 11 : 604, ) Allen, A. C. : Hereditary Anhidrotic Ectodermal Dysplasia, The Skin. A Clinicopathological Treatise, New York, Crune & Stratton, 1965, ) Sunderman, F. W. : Persons Lacking Sweat Glands. Hereditary Ectodermal Dysplasia of the Anhidrotic Type, Arch. Int. Med., 67 : , ) Clouston, H. R. : The Major Forms of Hereditary Ectodermal Dysplasia (With an Autopsy and Biopsies on the Anhydrotic Type), Canadian Med. Assoc. J., 40 : 1-7, ) Franken, E. : Anhidrotische Form der ektodermalen Dysplasie mit Hypogammaglobulinamie and Properdinmangel, Dermat. Wschr., 152 : , ) Wells, R. S. and Kerr, C. B. : Genetic Classification of Ichthyosis, Arch, Dermat., 92 : 1-6, ) Goeckermann, W. H. : Congenital Ectodermal Defect, with Report of a Case, Arch. Dermat. & Syph., 1 : , ) Kaalund-Jorgensen, O. and Christensen, J. F. : Congenital Ectodermal Dysplasia of the Anhidrotic Type, Acta dermat-venereol., 22 : 1-23, 1941.
7 A Case of Hereditary Anhidrotic Ectodermal Dysplasia Toshio HAMADA, M. D. Department of Dermatology, Osaka City University Medical School (Director : Prof. Tadao SAITO, M. D.) The patient is a 15-month-old boy. Since the age of two months, he has developed erythematosquamous lesions with excoriation on the face, nape and extremities. And since three months of age, he has had intermittent fever, especially on the daytime of summer, and no sweating has been recognized. The facies of the patient was just like that of the older with a distinctive saddle-shaped nose. The hair of the scalp and the eyebrows were sparse and fine. No teeth were observed, but four dental germs in both of the upper and lower jaw were found with X-ray examination. In sweating test with pilocarpine injection, there was no sweating over the entire body surface, except on the axillary fossae, where the skin surface became a little wet. Histologic studies showed neither of sebaceous gland, eccrine nor apocrine gland in the cutis of the axillar region. Biopsy from the left thigh revealed a histological picture of chronic dermatitis in addition to anhidrotic ectodermal dysplasia. Repeated asthmalike fits and extensive erythematosquamous lesions suggest the complication with asthma and atopic dermatitis. No consanguity was found in the family, but his mother has a delayed dentition, dry skin and a conspicuous saddle-shaped nose. This abnormality was reported to show transmission by healthy females to affected males, but these females may be affected less severely. It seems that inheritance of the disease may not be recessive.
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