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Recurrent villous adenoma of the duodenum
GASTROENTEROLOGY 1986;90:44C-2
CASE
REPORTS
Recurrent Villous Adenoma Duodenum JOSEPH
B. WEISS
and GREGORY
of the
G. SEMERDJIAN
Division of Gastroenterologv and Department of Family Practice, Scripps Clinic and Research Foundation, La Jolla, California
A patient is presented who in 1977 had a villous adenoma of the duodenum presenting with a diarrheal syndrome. Surgical removal of the polypoid mass lesion, which was pathologically a benign villous adenoma, resulted in resolution of her diarrhea. She remained asymptomatic until 6.5 yr later when diarrhea recurred. Radiographic and endoscopic examinations revealed a large villous adenoma within the proximal duodenum. After endoscopic polypectomy, her diarrhea promptly ceased. This is the first reported case of diarrhea associated with a villous adenoma of the duodenum. Villous adenomas of the duodenum are distinctly uncommon. Also described as a papillary adenoma, papilloma, adenomatous papilloma, and papillary tumor, they were first described by Perry in 1893 (1). Fewer than 50 cases have been reported in the literature before 1981.Varied clinical manifestations have been ascribed to these adenomas, including gastrointestinal (GI) bleeding, intestinal obstruction, intussusception, biliary tract involvement with obstructive jaundice, pancreatitis, and nonspecific GI complain6 (2-9). Villous adenomas involving the colon have been described previously in association with diarrhea and electrolyte loss (10).To our knowledge, no such association has been reported with villous adenomas involving the upper intestinal tract. We report here the case of a patient who at the age of 82 yr presented with a diarrhea1 illness possibly related to a large villous adenoma of the duodenum. Received March 7, 1985. Accepted July 26, 1985. Address requests for reprints to: Joseph B. Weiss, M.D., Division of Gastroenterology, Scripps Clinic and Research Foundation, 10666 North Torrey Pines Road, La Jolla, California 92037. The authors acknowledge the helpful comments of Dr. William S. Haubrich and Dr. Parviz Foroozan, who reviewed the manuscript. They also thank Bonny Mower and Beverly Williams for their excellent secretarial assistance. 0 1986 by the American Gastroenterological Association 0016-5085/86/$3.50
After surgical excision, her diarrhea ceased. She remained asymptomatic until recurrence of diarrhea was noted at age 89 yr. Villous adenoma involving the duodenum was subsequently identified. After endoscopic removal of a large benign villous adenoma she again had prompt cessation of her diarrhea.
Case Report An 82-yr-old white woman presented to a community hospital in 1977 complaining of frequent, loose, and watery stools. An upper GI barium study revealed a polypoid lesion in the juxtapyloric area, which prolapsed into the duodenal bulb. Upon exploration, she was found to have a 3.5 x 3.5~cm irregular, finely villous polypoid lesion. The polyp was removed by simple excision. Pathologically it was a villous adenoma with atypia and without malignancy. A Heineke-Mikulicz pyloroplasty was performed. No other abnormalities were noted on exploration of the abdomen. The patient had prompt resolution of her diarrhea. Periodic surveillance until 1980, both by upper GI barium study and endoscopy revealed no local recurrence or metachronous lesions. She was asymptomatic until 1984 at which time she presented with a l-mo history of loose, frequent, watery, and mucoid stools. She passed, rectally, voluminous quantities of a mucoid material, which at times resulted in soiling her underclothing. She had 6-12 bowel movements daily and regularly awoke during the night to defecate. She related that her diarrhea symptoms were identical to those present before her initial surgery. Physical examination was entirely unremarkable except for the presence of her surgical scar. Her stool was nonreactive for occult blood although mucoid material was present within the rectum. The serum potassium level was 3.4 mEq/L. A serum assay for vasoactive intestinal peptide was within normal limits. A barium enema of the colon and a flexible sigmoldoscopy (Olympus OES PlOS Sigmoidoscope, Olympus Corp. of America, New Hyde Park, N.Y.) to 60 cm were unremarkable. An upper GI barium study showed a lobulated 2.5 x 3.0-cm mass in the
VILLOUS ADENOMA OF THE DUODENUM
February 1986
Figure
1. An upper GI series demonstrating defect in the duodenum.
polypoid
filling
duodenal region [Figure 1) with characteristic “soap bubble” or “mousse de savon” sign seen with villous adenomas (Figure 2). Upper GI endoscopy (Olympus OES-GIF-XQ-10 endoscope) was performed after the patient underwent a 12-h fast. Approximately 500 ml of a viscous mucoid liquid was aspirated from the stomach. A z-cm soft lobulated mass lesion (Figure 31 within the duodenal bulb was subsequently apparent. Using an electrocautery snare, the majority of the polypoid mass was removed in piecemeal fashion without difficulty and was retrieved by suction and repetitive removal and reinsertion of the endoscope. Good hemostasis was apparent after the polyp had been removed. Histologically, the lesion was a benign villous adenoma without evidence of atypia or malignancy (Figure 4). The majority of the cells were columnar in nature and contained mucus droplets. Within 24 h of the endoscopic polypectomy, the patient noted complete resolution of her mucoid discharge and diarrhea. Indeed, the second day after the polypectomy, she complained of constipation. Two months later mucoid diarrhea recurred. Endoscopic examination after a 12-h fast revealed 250 ml of mucoid material within the stomach and proximal duodenum. The gastric pH was 6. A sizable mass, almost 1.5cm at its base, was seen at the site of previous polypectomy. The polyp was snared and severed from its base by electrocoagulation and then removed. Good hemostasis was noted and no residual polypoid tissue was apparent on gross examination. The patient once again noted prompt cessation of mucoid diarrhea
and
onset
of
constipation
requiring
441
digital
Figure 2. Polypoid filling defect with the characteristic “soap bubble” or “mousse de savon” sign seen in villous adenomas. disimpaction. An upper GI barium study documented removal of the polypoid mass. She has remained asymptomatic since the endoscopic removal of the residual polyp more than 8 mo ago.
Figure 3. Endoscopic
appearance of the multilobulated adenoma arising in the duodenum.
villous
442
WEISS AND SEMERDJIAN
GASTROENTEROLOGY Vol. 90. No. 2
gery and subsequent endoscopic polypectomy on two separate occasions, is suggestive of cause and effect. In addition, the endoscopic findings of large quantities of mucoid material would suggest that the villous adenoma may have been actively secreting the mucus material. Under normal circumstances one would expect proteolytic degradation of the mucus glycoprotein and subsequent intestinal absorption or utilization by the enteric flora. The patient’s relative achlorhydria and advanced age may have contributed to a diminution in proteolytic activity, resulting in the large quantity of mucus being passed through the intestinal tract. In view of the patient’s advanced age, endoscopic polypectomy was performed as previously described by Haubrich et al. (12), and subsequently by others (13). With no histologic evidence of malignancy, and with complete resolution of her symptoms over an 8-mo period following endoscopic removal, no surgical intervention is contemplated at this time.
References 1. Perry
Figure 4. Histologic section of villous adenoma demonstrating typical villous stroma. No evidence of malignancy was apparent.
Discussion Villous adenomas of the duodenum are uncommon. Villous adenomas of the small intestine are reported to cause GI bleeding, intestinal obstruction, intussusception, biliary tract involvement with obstructive jaundice, pancreatitis, and nonspecific GI complaints. To our knowledge, although previously described in colonic lesions, there have been no reports of an association between a diarrhea1 syndrome and villous adenomas involving the upper intestine. Indeed, a recent review comments on the lack of this feature in villous adenomas of the duodenum (11). Although inconclusive, the patient’s clinical course of diarrhea, associated with the presence of a villous adenoma of the duodenal region with prompt resolution following debulking of the tumor at sur-
EC. Papilloma of the duodenum. Trans Path01 Sot [London] 1892-1893;44:84. 2. Makkar J, Song KT, Cogbill CL. Villous adenoma of the duodenum: report of a case with massive hemorrhage. Am Surg 1969;35:656-9. 3. Mir-Madjlessi S, Farmer RG, Hawk WA. Villous tumors of the duodenum and jejunum. Report of four cases and review of the literature. Am J Dig Dis 1973;18:467-76. 4. Macumber HH, Stoll RG, Helvig FC. Papillary adenoma of the duodenum producing obstruction. Gastroenterology 1949; 13:468-70. 5. Greenwald ES, Parker JG, Schultz
6.
7. 8.
9.
10. 11. 12. 13.
S, Reed GE. Benign papillary adenoma of the ampullary region of the duodenum with intussusception. Gastroenterology 1962;43:344-50. Sobol S, Cooperman AM. Villous adenoma of the ampulla of Vater, an unusual case of biliary colic and obstructive jaundice. Gastroenterology 1978;75:107-9. Case Records of the Massachusetts General Hospital (Case 30-1984). N Engl J Med 1984;311:244-51. Cooperman M, Clausen KP, Hecht C, Lucas JG, Keith LM. Villous adenomas of the duodenum. Gastroenterology 1978; 74:1295-7. White SH, Nazarian NA, McEwen-Smith A, Balfour TW. Periampullary adenoma causing pancreatitis. Br Med J 1981;283:527. Fitzgerald MG. Extreme fluid and electrolyte loss due to villous adenoma of the rectum. Br Med J 1955;1:831. Reddy RR, Schuman BM, Priest RJ. Duodenal polyps: diagnosis and management. J Clin Gastroenterol 1981;3:139-45. Haubrich WS, Johnson RB, Foroozan P. Endoscopic removal of a duodenal adenoma. Gastrointest Endosc 1973;19:201. Duras JL, Marti R, Capron JP, Delamarre J. Villous adenoma of the duodenum, endoscopic diagnosis and resection. Endoscopy 1977;9:245-7.
CASE
REPORTS
Recurrent Villous Adenoma Duodenum JOSEPH
B. WEISS
and GREGORY
of the
G. SEMERDJIAN
Division of Gastroenterologv and Department of Family Practice, Scripps Clinic and Research Foundation, La Jolla, California
A patient is presented who in 1977 had a villous adenoma of the duodenum presenting with a diarrheal syndrome. Surgical removal of the polypoid mass lesion, which was pathologically a benign villous adenoma, resulted in resolution of her diarrhea. She remained asymptomatic until 6.5 yr later when diarrhea recurred. Radiographic and endoscopic examinations revealed a large villous adenoma within the proximal duodenum. After endoscopic polypectomy, her diarrhea promptly ceased. This is the first reported case of diarrhea associated with a villous adenoma of the duodenum. Villous adenomas of the duodenum are distinctly uncommon. Also described as a papillary adenoma, papilloma, adenomatous papilloma, and papillary tumor, they were first described by Perry in 1893 (1). Fewer than 50 cases have been reported in the literature before 1981.Varied clinical manifestations have been ascribed to these adenomas, including gastrointestinal (GI) bleeding, intestinal obstruction, intussusception, biliary tract involvement with obstructive jaundice, pancreatitis, and nonspecific GI complain6 (2-9). Villous adenomas involving the colon have been described previously in association with diarrhea and electrolyte loss (10).To our knowledge, no such association has been reported with villous adenomas involving the upper intestinal tract. We report here the case of a patient who at the age of 82 yr presented with a diarrhea1 illness possibly related to a large villous adenoma of the duodenum. Received March 7, 1985. Accepted July 26, 1985. Address requests for reprints to: Joseph B. Weiss, M.D., Division of Gastroenterology, Scripps Clinic and Research Foundation, 10666 North Torrey Pines Road, La Jolla, California 92037. The authors acknowledge the helpful comments of Dr. William S. Haubrich and Dr. Parviz Foroozan, who reviewed the manuscript. They also thank Bonny Mower and Beverly Williams for their excellent secretarial assistance. 0 1986 by the American Gastroenterological Association 0016-5085/86/$3.50
After surgical excision, her diarrhea ceased. She remained asymptomatic until recurrence of diarrhea was noted at age 89 yr. Villous adenoma involving the duodenum was subsequently identified. After endoscopic removal of a large benign villous adenoma she again had prompt cessation of her diarrhea.
Case Report An 82-yr-old white woman presented to a community hospital in 1977 complaining of frequent, loose, and watery stools. An upper GI barium study revealed a polypoid lesion in the juxtapyloric area, which prolapsed into the duodenal bulb. Upon exploration, she was found to have a 3.5 x 3.5~cm irregular, finely villous polypoid lesion. The polyp was removed by simple excision. Pathologically it was a villous adenoma with atypia and without malignancy. A Heineke-Mikulicz pyloroplasty was performed. No other abnormalities were noted on exploration of the abdomen. The patient had prompt resolution of her diarrhea. Periodic surveillance until 1980, both by upper GI barium study and endoscopy revealed no local recurrence or metachronous lesions. She was asymptomatic until 1984 at which time she presented with a l-mo history of loose, frequent, watery, and mucoid stools. She passed, rectally, voluminous quantities of a mucoid material, which at times resulted in soiling her underclothing. She had 6-12 bowel movements daily and regularly awoke during the night to defecate. She related that her diarrhea symptoms were identical to those present before her initial surgery. Physical examination was entirely unremarkable except for the presence of her surgical scar. Her stool was nonreactive for occult blood although mucoid material was present within the rectum. The serum potassium level was 3.4 mEq/L. A serum assay for vasoactive intestinal peptide was within normal limits. A barium enema of the colon and a flexible sigmoldoscopy (Olympus OES PlOS Sigmoidoscope, Olympus Corp. of America, New Hyde Park, N.Y.) to 60 cm were unremarkable. An upper GI barium study showed a lobulated 2.5 x 3.0-cm mass in the
VILLOUS ADENOMA OF THE DUODENUM
February 1986
Figure
1. An upper GI series demonstrating defect in the duodenum.
polypoid
filling
duodenal region [Figure 1) with characteristic “soap bubble” or “mousse de savon” sign seen with villous adenomas (Figure 2). Upper GI endoscopy (Olympus OES-GIF-XQ-10 endoscope) was performed after the patient underwent a 12-h fast. Approximately 500 ml of a viscous mucoid liquid was aspirated from the stomach. A z-cm soft lobulated mass lesion (Figure 31 within the duodenal bulb was subsequently apparent. Using an electrocautery snare, the majority of the polypoid mass was removed in piecemeal fashion without difficulty and was retrieved by suction and repetitive removal and reinsertion of the endoscope. Good hemostasis was apparent after the polyp had been removed. Histologically, the lesion was a benign villous adenoma without evidence of atypia or malignancy (Figure 4). The majority of the cells were columnar in nature and contained mucus droplets. Within 24 h of the endoscopic polypectomy, the patient noted complete resolution of her mucoid discharge and diarrhea. Indeed, the second day after the polypectomy, she complained of constipation. Two months later mucoid diarrhea recurred. Endoscopic examination after a 12-h fast revealed 250 ml of mucoid material within the stomach and proximal duodenum. The gastric pH was 6. A sizable mass, almost 1.5cm at its base, was seen at the site of previous polypectomy. The polyp was snared and severed from its base by electrocoagulation and then removed. Good hemostasis was noted and no residual polypoid tissue was apparent on gross examination. The patient once again noted prompt cessation of mucoid diarrhea
and
onset
of
constipation
requiring
441
digital
Figure 2. Polypoid filling defect with the characteristic “soap bubble” or “mousse de savon” sign seen in villous adenomas. disimpaction. An upper GI barium study documented removal of the polypoid mass. She has remained asymptomatic since the endoscopic removal of the residual polyp more than 8 mo ago.
Figure 3. Endoscopic
appearance of the multilobulated adenoma arising in the duodenum.
villous
442
WEISS AND SEMERDJIAN
GASTROENTEROLOGY Vol. 90. No. 2
gery and subsequent endoscopic polypectomy on two separate occasions, is suggestive of cause and effect. In addition, the endoscopic findings of large quantities of mucoid material would suggest that the villous adenoma may have been actively secreting the mucus material. Under normal circumstances one would expect proteolytic degradation of the mucus glycoprotein and subsequent intestinal absorption or utilization by the enteric flora. The patient’s relative achlorhydria and advanced age may have contributed to a diminution in proteolytic activity, resulting in the large quantity of mucus being passed through the intestinal tract. In view of the patient’s advanced age, endoscopic polypectomy was performed as previously described by Haubrich et al. (12), and subsequently by others (13). With no histologic evidence of malignancy, and with complete resolution of her symptoms over an 8-mo period following endoscopic removal, no surgical intervention is contemplated at this time.
References 1. Perry
Figure 4. Histologic section of villous adenoma demonstrating typical villous stroma. No evidence of malignancy was apparent.
Discussion Villous adenomas of the duodenum are uncommon. Villous adenomas of the small intestine are reported to cause GI bleeding, intestinal obstruction, intussusception, biliary tract involvement with obstructive jaundice, pancreatitis, and nonspecific GI complaints. To our knowledge, although previously described in colonic lesions, there have been no reports of an association between a diarrhea1 syndrome and villous adenomas involving the upper intestine. Indeed, a recent review comments on the lack of this feature in villous adenomas of the duodenum (11). Although inconclusive, the patient’s clinical course of diarrhea, associated with the presence of a villous adenoma of the duodenal region with prompt resolution following debulking of the tumor at sur-
EC. Papilloma of the duodenum. Trans Path01 Sot [London] 1892-1893;44:84. 2. Makkar J, Song KT, Cogbill CL. Villous adenoma of the duodenum: report of a case with massive hemorrhage. Am Surg 1969;35:656-9. 3. Mir-Madjlessi S, Farmer RG, Hawk WA. Villous tumors of the duodenum and jejunum. Report of four cases and review of the literature. Am J Dig Dis 1973;18:467-76. 4. Macumber HH, Stoll RG, Helvig FC. Papillary adenoma of the duodenum producing obstruction. Gastroenterology 1949; 13:468-70. 5. Greenwald ES, Parker JG, Schultz
6.
7. 8.
9.
10. 11. 12. 13.
S, Reed GE. Benign papillary adenoma of the ampullary region of the duodenum with intussusception. Gastroenterology 1962;43:344-50. Sobol S, Cooperman AM. Villous adenoma of the ampulla of Vater, an unusual case of biliary colic and obstructive jaundice. Gastroenterology 1978;75:107-9. Case Records of the Massachusetts General Hospital (Case 30-1984). N Engl J Med 1984;311:244-51. Cooperman M, Clausen KP, Hecht C, Lucas JG, Keith LM. Villous adenomas of the duodenum. Gastroenterology 1978; 74:1295-7. White SH, Nazarian NA, McEwen-Smith A, Balfour TW. Periampullary adenoma causing pancreatitis. Br Med J 1981;283:527. Fitzgerald MG. Extreme fluid and electrolyte loss due to villous adenoma of the rectum. Br Med J 1955;1:831. Reddy RR, Schuman BM, Priest RJ. Duodenal polyps: diagnosis and management. J Clin Gastroenterol 1981;3:139-45. Haubrich WS, Johnson RB, Foroozan P. Endoscopic removal of a duodenal adenoma. Gastrointest Endosc 1973;19:201. Duras JL, Marti R, Capron JP, Delamarre J. Villous adenoma of the duodenum, endoscopic diagnosis and resection. Endoscopy 1977;9:245-7.