Gastroenterol. Endosc. 32 : 2632 `2639, 1990 Soichi HOTTA A Case of Cronkhite-Canada Syndrome Associated with an Early Cancer and Multiple Adenomas of
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1 Gastroenterol. Endosc. 32 : 2632 `2639, 1990 Soichi HOTTA A Case of Cronkhite-Canada Syndrome Associated with an Early Cancer and Multiple Adenomas of the Colon.
2 Vol.32(11),Nov.1990 Table 1 Laboratory 症例 findings 堀 田総 一 ほか 2633 on admission. Figure 5 Radiograph showing numerous, irregularshaped, sessile polyps which were under 5 mm and two semi -pedunculated polyps (arrows). 図). 注 腸 検 査:S状 結 腸 か ら盲 腸 に か け て,大 き さ5mmま で の 無 茎 性 の ポ リー プ が,無 1cm前 数 に 認 め ら れ,そ の他 に も 後 の 亜 有 茎 な い し有 茎 性 の ポ リー プ が,8個 認 め ら れ た(Figure5). 小 腸 二 重 造 影 検 査:異 常 な し. 大 腸 内 視 鏡 検 査:S状 結 腸 か ら 盲 腸 に か けて,浮 腫 状 で や や発 赤 した米 粒 大 の無 茎 性 ポ リープ が無 数 に認 め られ た(Figure6カ Figure 2 Radiograph showing nearly total ラ ー 附 図).こ 的 に 明 らか に 異 な る大 き さ1cm前 repla の 内6個 理 組 織 学 的 所 見 は,2個 と こ ろ,組 検 お よ び ポ リペ ク ト ミ ー を 施 行 し た 織 学 的 に は,嚢 形 成 性 腺 管 で,問 胞 状 拡 張 を 伴 う異 型 の 弱 い 過 質 の 浮 腫 と細 胞 浸 潤 が 認 め ら れ,若 性 ポ リー プ 様 の 所 見 を呈 して い た(Figure4カ 瘍 性 ポ リー プの 可 能 性 を 考 慮 し て す べ て ポ リ ペ ク ト ミー した(計8個 )(Table2).こ ラ ー 附 図).生 後 で 亜 有 茎 な い し有 茎 性 の や や 緊 満 感 を 呈 す る ポ リ ー プ は,腫 ment of gastric mucosa by numerous sessile polyps varying sizes which were under 1 cm in diameter. 3カ れ ら の ポ リー プ と は 肉 眼 年 ラ ー附 を 回 収 し た.切 除 ポ リー プの 病 は 異 型 性 の 弱 い 過 形 成 性 腺 管 で, 一 部 の 腺 管 の 拡 張 と 間 質 の 浮 腫 を 伴 う若 年 性 ポ リ ー プ 様 の 所 見 が 認 め ら れ た(Figure7カ ラ ー 附 図).こ れ らの ポ リー プ に 腺 腫 へ の 移 行 を 示 唆 す る 所 見 は 認 め ら れ な か っ た.3個 は 中 等 度 か ら 高 度 異 型 を伴 う管 状 腺 腫 で あ っ
3 Table 2 Location, size and histological findings of polypectomized colonic polyps.
4 Vol. 32(11), Nov.1990 Table 3 Reported cases of Cronkhite-Canada syndrome associated with gastrointestinal cancer. * early carcinoma **adenosquamous carcinoma
5 8) Katayama Y., Kimura M. and Konn M.: Cronkhite- Canada sydrome associated with a rectal cancer and adenomatous changes in colonic polyps. Am. J. Surgical pathology, 9 : 6571, ) Ikeda K., Sannohe Y. and Murayama H.: A case of Cronkhite-Canada sydrome developing after hemicolectomy., 13 : 251 `253, ) Koehler, P. R., Kyaw, M. M. and Fenlon, J. W.: Diffuse gastrointestinal polyposis with ectodermal changes. Cronkhite-Canada sydrome. Diagnostic Radiology, 103 : 589 `594, ) Cronkhite, L. W. and Canada, W. J.: Generalized gastrointestinal polyposis. An unusual syndrome of polyposis, pigmentation, alopecia and onychotrophia. New. Eng. J. Med, 252: 1011 `1015, ) Jarum, S. J. and Jensen, H.: Diffuse gastrointestinal polyposis with ectodermal changes. A case with severe malabsorption and enteric loss of plasma proteins and electrolytes. Gastroenterology, 50 : 107 `118, ) Malhotra, R, and Sheffield, A.: Cronkhite-Canada 13) Rappaport L. B., Sperling H. V. and Stavrides A.: Colon cancer in the Cronkhite-Canada sydrome.: J. Clin. Gastroenterol., 8 : 199 `202, ) Sagara K., Fujiyama S., Kamuro Y., Tashiro A, and Sato T.: Cronkhite-Canada sydrome associated with gastric cancer : report of a case. Gastroenterol. Jpn., 18:260 `266, sydrome associated with colon carcinoma and adenomatous changes in C-C polyps. Am. J. Gastroenterology, 83 : 772 `776, 1988.
6 Kazunobu Yamazakiほ か 論 文 附 図 本 文 掲 載 頁:p afig-lb SoichiHottaほ か 論 文 附 図 afig-2b 本 文 掲 載 頁:p Fig-1 Fig-3 Fig-4 Fig-6 Fig-7 Fig-8 Fig-9
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8 A CASE OF CRONKHITE-CANADA SYNDROME ASSOCIATED WITH AN EARLY CANCER AND MULTIPLE ADENOMAS OF THE COLON Soichi HOTTA, Hiromasa KASHIMURA, Yoko SAITO, Tsuguo EBIHARA, Akira NAKAHARA, Susumu YAMAGATA Syohei KOYAMA, Hiroshi MUTO, Hisayuki FUKUTOMI Toshiaki OSUGA, Shinichi BAN AND Kyoichi NAKAMURA,, Department of Gastroenterology, Institute of Clinical Medicine, University of Tsukuba, Ibaraki, Japan. Department Japan. of Pathology, Institute of Basic Medical Science, University of Tsukuba, Ibaraki, We experienced 49-year-old man suffered from Cronkhite-Canada syndrome associated with multiple adenomas and 1 early cancer of the colon. Upper gastrointestinal series and endoscopy revealed nearly total replacement of gastric mucosa by numerous polyps of various sizes which were under 1 cm in diameter. Biopsy specimens of these polyps showed hyperplastic glands without atypia and edematous stroma with inflammatory cell infiltration. These polyps resembled "juvenile polyps". Barium enema study revealed two pedunculated and six semi-pedunculated polyps about 1 cm in diameter in addition to numerous, irregular-shaped sessile polyps. Colonoscopic polypectomy was performed for these polyps. We could withdraw six of them. Histological findings showed that two of them were hyperplastic polyps with dilatated cystiform glands and edematous stroma, three were tubular adenomas with moderate to severe atypia and another one was tubular adenocarcinoma with mucosal invasion. There were no adenomatous or carcinomatous foci in non-neoplastic polyps in our case, indicating that all adenomas and carcinoma were supposed to be accidentaly associated with Cronkhite-Canada syndrome. Twenty-five cases of Cronkhite-Canada syndrome associated with gastrointestinal cancer have already been reported in the world according to our research. Only three of them were cases of early cancer. Accordingly, our case can be classified as a rare case. Figure 1 Nail of the patient showing mild atrophy. Figure 3 Endoscopic view in the stomach showing numerous edematous sessile polyps with redness. Figure 4 High power view of the gastric polyp showing hyperplastic glands without atypia and edematous stroma with inflammatory cell infiltration. Figure 6 Endoscopic view in the colon showing multiple small sessile polyps. Figure 7 Low power view of a pedunculated polyp in the descending colon showing hyperplastic glands without atypia, such as cystic dilatation of glands, stromal edema, and inflammatory cell infiltration. Figure 8 Endoscopic view of in the sigmoid colon showing a semi-pedunculated polyp which was diagnosed as adenocarcinoma histologically Figure 9 High power view of adenocarcinoma showing structual atypia such as crowded and irregular glands, and cellular atypia such as disarrangement of nucleus.
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