Radiographic Features of Gastric Polyps in. Familial Adenomatosis Coli

Transcription

1 Radiographic Features of Gastric Polyps in Familial Adenomatosis Coli YUJI ITAI, TAKASHI KOGURE, YAMAJI OKUYAMA,2 AND TETSUICHIRO MUTO3 Recent reports have noted a high frequency of elevated mucosal lesions of the stomach in patients with familial adenomatosis coli. This paper describes an additional 10 cases from six different families. Nine patients had sessile s in various portions of the stomach. Six of these have been examined histologically; two were adenomatous and four hyperplastic. The need for meticulous examination of the stomach in patients with multiple colonic osis is emphasized. Familial adenomatosis coii is characterized by autosomal dominant inheritance and the eventual development of colon carcinoma. While gastric osis is frequently associated with the Peutz-Jeghers and Cronkhite-Canada syndromes, gastric involvement in familial adenomatosis coli has only been documented in sporadic cases [1-6]. This paper describes six affected families with a high frequency of gastric s and characterizes the radiographic appearance. Radiographic Observations Ten patients with proven adenomatosis coli from six families underwent radiologic and/or endoscopic examinations of the stomach at hospitals affiliated with Tokyo University. Nine of the patients had gastric s. The s were sessile, had smooth, well defined margins, and often showed central umbilication. Their size, number, and location varied widely. Radiologic features are summarized in table 1, and five patients are described and illustrated in detail. Case 1 Case Reports A 60-year-old woman had an upper gastrointestinal oxamination because of anemia. Gastric osis was readily demonstrated (fig. 1A) and partial gastrectomy was performed. Approximately 80 hemispheric s were noted in the surgical specimen; histologically they were adenomas (fig. 1B). Size of the s varied from 2 to 17 mm. They were limited to the distal two-thirds of the stomach. Despite gastrectomy, anemia did not improve. Colonic adenomatosis and sigmoid cancer were discovered 2 years later. The patient is regarded as having Gardner syndrome because of a lipoma on her back and osteoma of hen frontal bone. Case 2 A 25-year-old woman underwent barium enema which revealed diffusely scattered s in the entire colon and rectum. Endoscopic biopsy confirmed they were adenomas. Upper gastrointestinal series showed 10 s in the prepylonic region (fig. 2A) and numerous small s in the fundus and body of the stomach (fig. 28). The prepylonic s measured up to 7 mm and had obvious central depression. The others ranged from 1.5 to 3 mm and showed smooth surfaces. Case 3 A 23-year-old man with colonic adenomatosis underwent upper gastrointestinal examination. A solitary with deep central depression was noted in the antrum (fig. 3). It was round, measured 1 cm, and had a steeply elevated rim. Endoscopic examination confirmed the ; biopsy identified it as hyperplastic. In addition, a few tiny mucosal elevations were noticed in the prepylonic region. Case 4 A 36-year-old man was referred for radiotherapy of rectal cancer after exploratory laparotomy at another hospital. Barium enema showed an obstructing carcinoma at the rectosigmoid junction, as well as numerous tiny s in the neighboring mucosa. Upper gastrointestinal series revealed innumerable s in the fundus and the greater curvature of body and distal antrum (fig. 4). Gastric biopsy specimen was interpreted as hyperplastic. Case 5 A 42-year-old man with complaints of general malaise had been diagnosed about 20 years earlier as having osis coli. Upper gastrointestinal series revealed gastric cancer as well as many s in the antnum, and partial gastrectomy was performed. In the surgical specimen multiple s were noted in the prepyloric region along with advanced ulcerating cancer (fig. 5). Histologically the s were hyperplastic. He developed cecal cancer 2 years later and died of liver metastasis soon after right hemicolectomy. Discussion In familial adenomatosis coli, primary attention has been directed to the colon because of the very high potential for malignancy (approaching 100%). In 1950 Halsted et al. [1] endoscopically identified gastric s in five members of a family with multiple colonic Received May ; accepted after revision September 16, I Department of Radiology, Faculty of Medicine, University of Tokyo Hongo. Bunkyo-ku, Tokyo. Japan. 2 Department of Geriatrics, Faculty of Medicine. University of Tokyo. Tokyo, Japan. 3 Department of Surgery. Faculty of Medicine. University of Tokyo. Tokyo. Japan. Am J Roentg.nol 128:73-76, January

2 74 ITAI ET AL. TABLE 1 Radiographic and Histologic Findings Patient No. Age and Sex Location of Polyps No. Size (mm) Central Depression Histology I (case 1 ) 60, F Antrum/body Adenoma 2 (case 2) 25, F Distal antrum Fundus/body Many Not examined 3 (case 3) 23. M Antrum f- Hyperplastic 4 27, M Antrum Adenoma 5 (case 4) 36, M Proximal antrum/fundus Many Hyperplastic 6 40, F Antrum Not examined 7 (case 5) 42, M Distal antrum Hyperplastic 8 28, M Distal antnum Not examined 9 20, M Proximal antrum/fundus Hyperplastic M... None N ote.-patients 1-4 from family A: patient s 5 and 6, family B; patien ts 7-10, families C-F, respectively. Fig. 1. -Case 1. A, Air contrast examination showing multiple mucosal nodules. B. Sucgical specimen of stomach. adenomatosis. Mayo et al. [2] found two cases of gastric s in nine patients with familial osis coli. Hoffmann and Goligher [6] reported three cases of familial osis coli with adenomatous s of the upper gastrointestinal tract and an additional 14 cases of gastric s from the literature. More frequent associations of gastric and colonic s have been noted recently in Japan. Utsunomiya et al. [rnl] reported gastric polys in 10 of 1 5 patients with colonic adenomatosis from five different families. Two other reports [8, 9] showed an association as high as 55% and 67%, respectively. This apparent difference in frequency may be due to genetic on environmental variables. A more systematic survey would be necessary to assess this clearly. Meticulous examination of thg stomachs of patients with familial colonic osis is recommended. Risk of malignant change of these s [10] and the actual incidence of gastric cancer in familial adenomatosis coli are now under investigation.

3 GASTRIC POLYPS IN ADENOMATOSIS COLI 75 Fig. 2.-Case 2. A. Multiple umbilicated s in distal antrum. B, Innumerable tiny s along greater curvature of body. Fig. 3.-Case 3. Solitary with irregular central depression. Fig. 4.-Case 4. Double contrast mucosal examination showing innumerable s in fundus.

4 76 ITAI ET AL. Fig. 5.-Case 5. Partial gastrectomy specimen showing many sessile s as well as ulcerating antral cancer. REFERENCES 1. Halsted JA, Harris EJ, Bartlett MK: Involvement of the stomach in familial osis of the gastrointestinal tract. Gastnoentenology 15: , Mayo CW, DeWeend JH, Jackman RJ: Diffuse familial osis of the colon. Sung Gyneco! Obstet 93:87-96, Boley SJ, McKinnon WMP, Manzulli VF: The management of familial gastrointestinal osis involving stomach and colon. Surgery 50: , Yonemoto RH, Slayback JB, Byron RL, Rosen RB: Familial osis of the entire gastrointestinal tract. Arch Sung 99: , Parks TG, Bussey HJR, Lockhart-Mummery HE: Familial osis coli associated with extracolonic abnonmalities. Gut 11: , Hoffmann DC, Golighen JC: Polyposis of the stomach and small intestine in association with familial osis coli. Br J Sung 58: , Utsunomiya J, Maki T, Iwama T, Matsunaga Y, lchikawa T, Shimomura T, Hamaguchi E, Aoki N: Gastric lesion of familial osis coli. Cancer 34: , Ushio K, Yamada T, Ichikawa H: Lesions associated with so-called familial osis of the colon-on lesions of the stomach, bones and teeth (in Japanese). Stomach and Intestine 9: , Ohsato K, Itoh H, Ikeda S. Nishimuna M: Follow-up study on upper gastrointestinal lesions in familial poiyposis of the colon (in Japanese). Jap J Gastroentenol 172: , Murphy ES, Mireles M, Beltran A: Familial osis of the colon and gastric carcinoma. Concurrent conditions in a 16-year-old boy. JAMA 179: , 1962

5 This article has been cited by: 1. Stephen E. RubesinCase [CrossRef] 2. Carina L. Butler, James L. BuckPolyposis Syndromes [CrossRef] 3. Alvin L. Watne The syndromes of intestinal osis. Current Problems in Surgery 24:5, [CrossRef] 4. Edwin W. Naylor, Emanuel Lebenthal Gardner's syndrome. Digestive Diseases and Sciences 25:12, [CrossRef]