Esophageal cancer complicated by cytomegalovirus esophagitis during chemoradiotherapy: case report

H Ohnuma, Y Sato, T Takayama, et al.

Brief Reports

gioscope used in t h e p r e s e n t case allows t a r g e t e d biopsy s p e c i m e n s to be o b t a i n e d u n d e r d i r e c t vision. W i t h i n c r e a s i n g a v a i l a b i l i t y of s u i t a b l e i n s t r u m e n t s , t h e optim a l a n d l e a s t invasive p r e o p e r a t i v e e v a l u a t i o n of these t u m o r s should shift from p e r c u t a n e o u s cholangioscopy to endoscopic r e t r o g r a d e cholangioscopy a t i n i t i a l ERCP.

Lehel Somogyi, MD Haytham Dimashkieh, MD Fredrick L. Weber, Jr., MD Joseph Buell, MD University of Cincinnati and Veterans Affairs Medical Center Cincinnati, Ohio REFERENCES

Figure 3. A, Photomicrograph of cross section of bile duct in resection specimen showing papillary mass arising from wall and projecting into lumen (H&E, orig. mag. x3.12). B, Photomicrograph at higher power showing well-differentiated adenocarcinoma invading subepitheliat connective tissue of common hepatic duct wall (H&E, orig. mag. x10). s t a g e d as p T l a NO. P o s t o p e r a t i v e recovery was uneventful a n d findings from biochemical tests of liver function r e t u r n e d to normal. Discussion. Biliary intraductal papillary mucinous t u m o r is u s u a l l y d i a g n o s e d a n d localized by p e r c u t a n e o u s or i n t r a o p e r a t i v e cholangioscopy. 4-6,s P e r c u t a n e o u s cholangioscopy, however, is invasive and t i m e consuming because t h e t r a c t m u s t be d i l a t e d g r a d u a l l y over a period of time t h a t m a y be as long as 2 weeks and, in addition, carries a r i s k of m a l i g n a n t seeding of the tract. 8 The case described h e r e d e m o n s t r a t e s the feasibility of endoscopic r e t r o g r a d e cholangioscopy for d i a g n o s i s a n d localization of a b i l i a r y i n t r a d u c t a l p a p i l l a r y m u c i n o u s tumor. The procedure can be performed d u r i n g t h e initial E R C P if a cholangioscope is available. It is less invasive t h a n p e r c u t a n e o u s c h o l a n g i o s c o p y a n d could f a c i l i t a t e o p e r a t i v e p l a n n i n g b a s e d on exact k n o w l e d g e of t u m o r extent, both p r o x i m a l l y a n d distally. This can be accomplished by o b t a i n i n g r a d i o g r a p h s w i t h the cholangioscope positioned at t h e p r o x i m a l a n d t h e n th~ distal m a r g i n of the tumor. A s i m i l a r a p p r o a c h facilitates optimal s u r g e r y for p a p i l l a r y mucinous t u m o r of t h e pancreas. The cholan622

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1. Styne P, Warren GH, Kumpe DA, Halgrimson C, Kern F Jr. Obstructive cholangitis secondary to mucus secreted by a solitary papillary bile duct tumor. Gastroenterology 1986;90: 748-53. 2. Hadjis NS, Slater RN, Blumgart LH. Mucobilia: an unusual cause of jaundice. Br J Surg 1987;74:48-9. 3. Kokubo T, Itai Y, Ohtomo K, Itoh K, Kawauchi N, Minami M. Mucin-hypersecreting intrahepatic biliary neoplasms. Radiology 1988;168:609-14. 4. Park HJ, Seo DW, Lee SK, Kim MH. Mucin-hypersecreting cholangiocarcinoma. Gastrointest Endosc 2000;52:526. 5. Kim HJ, Kim MH, Lee SK, Yoo KS, Park ET, Lim BC, et al. Mucin-hypersecreting bile duct tumor characterized by a striking homology with an intraductal papillary mucinous tumor (IPMT) of the pancreas. Endoscopy 2000;32:389-93. 6. Chen MF, Jan YY, Chen TC. Clinical studies of mucin-producing cholangiocellular carcinoma: a study of 22 histopathologyproven cases. Ann Surg 1998;227:63-9. 7. Ko SF, Chen YS, Ng SH, Lee TY, Chen WJ, Cheng YF. Mucinhypersecreting papillary cholangiocarcinoma presenting as abdominal wall abscess: CT and spiral CT cholangiography. Abdom Imaging 1996;21:222-5. 8. Sakamoto E, Hayakawa N, Kamiya J, Kondo S, Nagino M, Kanai M, et al. Treatment strategy for mucin-producing intrahepatic cholangiocarcinoma: value of percutaneous transhepatic biliary drainage and cholangioscopy. World J Surg 1999;23:1038-43. 9. Sano T, Nimura Y, Hayakawa N, Kamiya J, Nagino M, Kanai M, et al. Clinical utility of percutaneous transhepatic cholangioscopy in defining tumor extent: a case of mucin-producing bile duct carcinoma originating in the left caudate lobe, Gastrointest Endosc 1997;46:455-8. 10. Heit P, McKinley M, Javors B. Mucoid biliary obstruction: direct endoscopic visualization of a mucin-hypersecreting bile duct tumor. Gastrointest Endosc 1990;36:164-6.

Esophageal cancer complicated by cytomegalovirus esophagitis duringchemoradiotherapy: case report C h e m o r a d i o t h e r a p y can be highly effective for advanced esophageal cancer b u t is often associated with a variety of Reprint requests: Internal Medicine, Chuo-ku, Sapporo, Copyright 9 2003 Endoscopy

Yoshiro Niitsu, MD, Fourth Department of Sapporo Medical University, South 1 West 16, 060-8543, Japan. by the American Society for Gastrointestinal 0016-5107/2003/$30.00 + 0 doi:10.1067 / mge.2003.150 VOLUME 57, N0. 4, 2003

Brief Reports

Figure 1. Endoscopic view of circumferential tumor in distal esophagus. treatment-related complications. Cytomegalovirus (CMV) esophagitis is believed to occur when systemic immune functions are severely compromised as may occur, for example, in patients infected with HIV and those undergoing bone marrow transplantation. A case is presented of fever of unknown origin and refractory esophagitis that occurred during chemoradiotherapy for esophageal cancer. CMV esophagitis was suspected based on endoscopic findings, which proved to be the correct diagnosis. CMV esophagitis should be considered a potential treatment-related complication during chemoradiotherapy for esophageal cancer. C a s e r e p o r t . A 60-year-old woman saw her primary care physician because of vomiting and dysphagia on ingestion of solid food, Esophageal carcinoma was diagnosed at upper endoscopy and the patient was referred to us for treatment. Examination did not reveal any significant abnormality. Laboratory tests were negative except for a serologic test for anti-CMV IgG antibodies, which was positive, whereas a test for anti-CMV IgM antibodies was negative, findings interpreted as suggestive of prior CMV infection. Upper endoscopy confirmed the presence of a circumferential, ulcerative, and infiltrating tumor at 35 cm from the incisors, extending to the gastroesophageal junction (Fig. 1). Histopathologic evaluation of endoscopic biopsy specimens disclosed moderately differentiated adenocarcinoma. Chest and abdominal CT revealed right cardiac and anterior bronchial lymphadenopathy. Chemoradiotherapy was initiated with the protocol of Schnirer et al.l: 5-Fluorouracil (300 mg/m2/day 5 times per week) and docetaxel (20 mg/m2/day once per week). A total radiation dose of 50.4 Gy/28F was scheduled and externalbeam radiation therapy was started on the 36th day after admission. Soon after treatment began, peripheral blood lymphocyte counts began to decrease markedly, to as low as 31/mm 3 (normal: 1500-4000 m m 3) on the 68th day, but neutrophil levels were maintained throughout therapy. About 3 weeks after initiation of chemoradiotherapy, nausea and vomiting developed. For these symptoms, the patient was treated frequently with dexamethasone (4 mg/day, once daily for 2 weeks). On the 68th day, fever VOLUME 57, NO. 4, 2003

H Ohnuma, Y Sato, T Takayama, et al.

Figure 2. Endoscopic view of distal esophagus (82nd day) showing multiple irregular, map-like erosions and ulcerations corresponding to radiation field. (38~ and odynophagia developed, and on the 70th day (34th day after initiation of chemoradiotherapy, at which point a radiation dose of 41.4 Gy had been given), vomiting worsened to 6 times per day. Therapy was therefore terminated. A bacterial or fungal infection of the GI tract was suspected and treatment with imipenem and fluconazole was begun, but the symptoms persisted. At upper endoscopy on the 82nd day, the esophageal tumor was markedly reduced in size and covered with flat, scarred, and regenerated epithelium (Fig. 2). However, proximal to the tumor but within the radiation field, the mucosa was almost circumferentially reddened, edematous, and friable with multiple sharply demarcated, irregular map-like erosions and ulcers. A biopsy specimen from an erosion revealed stromal edema with numerous infiltrating polymorphonuclear leukocytes and enlarged vascular endothelial cells. There were basophilic inclusion bodies in the nuclei and cytoplasm of the vascular endothelial cells (Fig. 3A). Immunohistochemical staining with anti-CMV antibodies identified CMV antigens that corresponded with the inclusion bodies (Fig. 3B). In situ hybridization also proved the presence of CMV-DNA (Fig. 3C). CMV antigenemia in circulating leukocytes was also demonstrated on the day that the biopsy specimens were obtained. A diagnosis was made of CMV esophagitis and treatment with ganciclovir (360 mg/day) was begun. On the 101st day, the patient became afebrile. Creactive protein returned to negative and the odynophagia improved. The patient then tested negative for CMV antigenemia, immunohistochemical staining, and in situ hybridization. On the 21st day after ganciclovir administration, upper endoscopy disclosed a slight remnant of the reddened edematous mucosa in only a portion of the distal esophagus. The erosions and ulcers were markedly improved. Several biopsy specimens were taken from this area; all showed mild inflammation but no inclusion bodies or CMV-DNA were identified. At 6 months after chemoradiotherapy, the patient is doing well with no progression of the tumor and no evidence of recurrence of the infection. GASTROINTESTINAL ENDOSCOPY 623

H Ohnuma, Y Sato, T Takayama, et al.

Figure 3. A, Photomicrograph of endoscopic biopsy showing prominent basophilic inclusion bodies within nucleus and cytoplasm of vascular endothelial cells (arrows) (H&E, orig. mag. • B, Staining of infected cells (brownish-red, arrows) using a monoclonal antibody against a CMV antigen (immunoperoxidase stain, orig. mag. x200). C, In situ hybridization showing enlarged nucleus (purple, arrows, orig. mag. •

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D i s c u s s i o n . It is believed that 80% to 90% of adults are serologically positive for CMV. The virus usually remains latent, but reactivation of CMV can occur when patients are immunocompromised by conditions such as AIDS, organ transplantation, immunosuppressive therapy, and treatment with corticosteroids.2, 3 In such circumstances, active infection can occur in m a n y organs including the lungs, GI tract, adrenal glands, liver, and spleen. 4 Frequently, several organs may be involved concurrently. Among GI organs, the colon and esophagus are involved most often, followed by stomach and small intestine.5, 6 Although there are no specific symptoms attributable to CMV esophagitis, those often associated with this form of esophagitis include dysphagia, odynophagia, substernal pain, nausea, and vomiting. 7-9 Thus, this disease should be suspected when a patient who is immunocompromised presents with any of these symptoms. Diagnostic procedures for CMV esophagitis include serologic testing, endoscopy, histopathologic examination of biopsy specimens (cytomegalic cells), viral identification (CMV antigen, DNA), and isolation (culture). Multiple punched-out, irregular, map-like erosions and deep ulcers are described endoscopic findings of CMV esophagitis.3,5, i0-12 Iwasaki 5 and Roberts et al. 13 found that CMV mainly infects vascular endothelial cells. Damaged and enlarged endothelial cells narrow the blood vessel lumen and cause tissue ischemia followed by ulcer formation. As a result, GI ulcers caused by CMV bleed easily. Except for the shallow depth of the ulcer, the endoscopic findings in the present case were typical of CMV esophagitis. Many studies have confirmed the specificity of cytomegalic cells for the diagnosis of CMV infection.5,i4-17 It known, however, that not all CMV infected cells display such a change in form. Immunohistochemical staining and in situ hybridization to identify the CMV antigen and DNA are used together to enhance diagnostic sensitivity. It is reported that the immunohistochemical staining can be positive whereas routine staining with hematoxylin and eosin fails to show any cytomegalic cells.IS, i9 A similar increase in sensitivity was reported for in situ hybridization. 20 These tests are rapid and highly sensitive compared with viral culture from endoscopic biopsy specimens, a method that reportedly is highly specific but less reliable with regard to sensitivity. 21 Endoscopic biopsy specimens in the present case revealed characteristic basophilic cytomegalic inclusion bodies, so called "owls' eyes," in the vascular endothelial cells, which is the generally accepted standard for diagnosis of CMV infection. However, because the ulcer was less deep than expected, in situ hybridization and immunohistostaining were also performed to confirm the diagnosis. Reactivation of latent CMV virus infection is precluded by both humoral (neutralizing antibodies) and cellmediated (cytotoxic T-lymphocytes) immunity. Cellmediated immunity is believed to control latent infection and reactivation. 22 Lymphopenia caused by bone marrow suppression occurred after chemoradiotherapy was begun in the present case. The lymphocyte count was markedly decreased to 31/mm 3 when symptoms of the CMV infecVOLUME 57, NO. 4, 2003

Brief Reports

H Ohnuma, Y Sato, T Takayama, et al.

tion developed. In addition, t h e frequent a d m i n i s t r a t i o n of corticosteroids for n a u s e a induced cell-mediated i m m u n o deficiency. F u r t h e r , CMV is k n o w n to infect i n j u r e d tissue. 23-26 Because of t h e tissue i n j u r y a t the site corres p o n d i n g to r a d i a t i o n exposure, it is a s s u m e d t h a t t h e v i r u s a c c u m u l a t e d at this site a n d caused t h e esophagitis. T r e a t m e n t w i t h a n t i v i r a l drugs is i n d i c a t e d w h e n a n i m m u n o c o m p r o m i s e d s t a t e a n d infection p e r s i s t . D r u g s such as ganciclovir, which competes w i t h d G T P to i n h i b i t D N A p o l y m e r a s e , a n d foscarnet, which d i r e c t l y i n h i b i t s D N A p o l y m e r a s e , h a v e e x c e l l e n t a c t i v i t y a g a i n s t CMV. Controlled t r i a l s have d e m o n s t r a t e d resolution of sympt o m s a n d l e s i o n s in p a t i e n t s w i t h h i s t o p a t h o l o g i c a l l y c o n f i r m e d CMV d i s e a s e i n v o l v i n g t h e GI t r a c t . 6,27-29 Ganciclovir was highly effective in t h e p r e s e n t case a n d r e s u l t e d in histopathologic i m p r o v e m e n t . C o m b i n a t i o n s of c h e m o t h e r a p y a n d r a d i o t h e r a p y a r e b e c o m i n g s t a n d a r d t r e a t m e n t for a d v a n c e d e s o p h a g e a l cancer. However, d u r i n g such t h e r a p y t h e r e is a r i s k of infection. CMV esophagitis should be considered w h e n a p a t i e n t u n d e r g o i n g c h e m o r a d i o t h e r a p y develops fever of u n k n o w n origin a n d o d y n o p h a g i a , is u n r e s p o n s i v e to a n t i b a c t e r i a l or a n t i f u n g a l a g e n t s , a n d / o r w h e n endoscopic findings d e m o n s t r a t e m u l t i p l e punched-out, irregul a r m a p - l i k e erosions a n d ulcers.

Hiroyuki Ohnuma, MD Yasushi Sato, MD, PhD Tetsuji Takayama, MD, PhD Rishu Takirnoto, MD, PhD Tomoyuki Abe, MD Seiya Hagiwara, MD, PhD Takehiro Kukitsu, MD Atsushi Nobuoka, MD, PhD Tsutornu Sato, MD, PhD Katsuhisa Kogawa, MD, PhD Junji Kato, MD, PhD Yoshiro Niitsu, MD, PhD Fourth Department of Internal Medicine Sapporo Medical University Chuo-ku, Sapporo, Japan REFERENCES 1. Schnirer II, Komaki R, Yao JC, Swisher S, Putnam J, Pisters PW, et al. Pilot study of concurrent 5-fluorouracil/paclitaxel plus radiotherapy in patients with carcinoma of the esophagus and gastroesophageal junction. Am J Clin Onco12001;24;91-5. 2. Ho M. Epidemiology of cytomegalovirus infections. Rev Infect Dis 1990;12:$701-10. 3. Grundy JE. Virologic and pathogenetic aspects of cytomegalovirus infection. Rev Infect Dis 1990;12:$711-9. 4. Wong TW, Warner NE. Cytomegalic inclusion disease in adults. Arch Pathol 1962;74:403-22. 5. Iwasaki T. Alimentary tract lesions in cytomegalovirus infection. Acta Pathol Jpn 1987;37:549-65. 6. Goodgame RW. Gastrointestinal cytomegalovirus disease. Ann Intern Med 1993;119:924-35. 7. Wilcox CM, Diehl DL, Cello JP, Margaretten W, Jacobson MA. Cytomegalovirus esophagitis in patients with AIDS. A clinical, endoscopic, and pathologic correlation. Ann Intern Med 1990;113:589-93. 8. Theise ND, Rotterdam H, Dieterich D. Cytomegalovirus VOLUME 57, NO. 4, 2003

esophagitis in AIDS: diagnosis by endoscopic biopsy. Am J Gastroenterol 1991;86:1123-6. 9. Parente F, Cernuschi M, Rizzardini G, Lazzarin A, Valsecchi L, Bianchi Porro G, et al. Opportunistic infections of the esophagus not responding to oral systemic antifungals in patients with AIDS: their frequency and treatment. Am J Gastroenterol 1991;86:1729-34. 10. Goodgame RW, Ross PG, Kim HS, Hook AG, Sutton FM. Esophageal stricture after cytomegalovirus ulcer treated with ganciclovir. J Clin Gastroenterol 1991;13:678-81. 11. Bonacini M, Young T, Laine L. The cause of esophageal symptoms in human immunodeficiency virus infection. A prospective study of 110 patients. Arch Intern Med 1991;151:1567-72. 12. McDonald GB, Sharma P, Hackman RC, Meyers JD, Thomas ED. Esophageal infections in immunosuppressed patients after bone marrow transplantation. Gastroenterology 1985; 88:1111-7. 13. Roberts WH, Sneddon JM, Waldman J, Stephens RE. Cytomegalovirus infections of gastrointestinal endothelium demonstrated by simultaneous nucleic acid hybridization and immunohistochemistry. Arch Pathol Lab Med 1989;113:461-4. 14. Culpepper-Morgan JA, Kotler DP, Scholes JV, Tierney AR. Evaluation of diagnostic criteria for mucosal cytomegalovirus disease in the acquired immune deficiency syndrome. Am J Gastroenterol 1987;82:1264-70. 15. Jacobson MA, Mills J. Serious cytomegalovirus disease in the acquired immunodeficiency syndrome (AIDS). Clinical findings, diagnosis, and treatment. Ann Intern Med 1988;108:585-94. 16. Myerson D, Hackman RC, Nelson JA, Ward DC, McDougall JK. Widespread presence of histologically occult cytomegalovirus. Hum Pathol 1984;15:430-9. 17. Clayton F, Klein EB, Kotler DP. Correlation of in situ hybridization with histology and viral culture in patients with acquired immunodeficiency syndrome with cytomegalovirus colitis. Arch Pathol Lab Med 1989;113:1124-6. 18. Wu GD, Shintaku IP, Chien K, Geller SA. A comparison of routine light microscopy, immunohistochemistry, and in situ hybridization for the detection of cytomegalovirus in gastrointestinal biopsies. Am J Gastroenterol 1989;84:1517-20. 19. Francis ND, Boylston AW, Roberts AH, Parkin J, Pinching AJ. Cytomegalovirus infection in gastrointestinal tracts of patients infected with H1V-1 or AIDS. J Clin Pathol 1989;42: 1055-64. 20. Robert WH, Hammond S, Sneddon JM, Thesing J, Caldwell JH, Clausen KP. In situ DNA hybridization for cytomegalovirus in colonoscopic biopsies. Arch Pathol Lab Med 1988; 112: 1106-9. 21. Goodgame RW, Genta RM, Estrada R, Demmler G, Buffone G. Frequency of positive tests for cytomegalovirus in AIDS patients: Endoscopic lesions compared with normal mucosa. Am J Gastroenterol 1993;88:338-43. 22. Pasternack MS, Medearis DN Jr, Rubin RH. Cell-mediated immunity in experimental cytomegalovirus infections: a perspective. Rev Infect Dis 1990;12:$720-6. 23. Wakefield AJ, Fox JD, Sawyerr AM, Taylor JE, Sweenie CH, Smith M, et al. Detection of herpesvirus DNA in the large intestine of patients with ulcerative colitis and Crohn's disease using the nested polymerase chain reaction. J Med Virol 1992;38:183-90. 24. Diepersloot RJ, Kroes AC, Visser W, Jiwa NM, Rothbarth PH. Acute ulcerative proctocolitis associated with primary cytomegalovirus infection. Arch Intern Med 1990;150:1749-51. 25. Aynlo M, Aisner SC, Margolis K, Moravee C. Cytomegalovirus associated gastritis in a compromised host. JAMA 1980;243: 1364-7. 26. Hinnant KL, Rotterdam HZ, Bell ET, Tapper ML. CytoGASTROINTESTINAL ENDOSCOPY 625

F Willingham, A Opekun, D Graham

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megalovirus infection of the alimentary tract: a clinicopathological correlation. Am J Gastroenterol 1986;81:944-50. 27. Buhles WC Jr, Mastre BJ, Tinker AJ, Strand V, Koretz SH. Ganciclovir treatment of life- or sight-threatening cytomegalovirus infection: experience in 314 immunocompromised patients. Rev Infect Dis 1988;10:$495-504. 28. Nelson MR, Connolly GM, Hawkins DA, Gazzard BG. Foscarnet in the treatment of cytomegalovirus infection of the esophagus and colon in patients with the acquired immune deficiency syndrome. Am J Gastroenterol 1991;86:876-81. 29. Dieterich DT, Poles MA, Dicker M, Tepper R, Lew E. Foscarnet treatment of cytomegalovirus gastrointestinal infections in acquired immunodeficiency syndrome patients who have failed ganciclovir induction. Am J Gastroenterol 1993;88:542-48.

Endoscopic demonstration of transient small bowel intussusception in a patient with adult celiac disease The manifestations of celiac disease (gluten enteropathy) relate to small-bowel damage that occurs as a result of a pathologic immune response to the ingestion of gluten. 1 Endoscopic features include scalloped folds, a mosaic pattern (grooves), and visualization of the underlying vasculature of the proximal small intestine.2,3 Transient entero-entero intussusception has been observed by contrast radiography of the small bowel in as many as 20% of adult patients with celiac disease, but this has not been described endoscopically.4 A wireless capsule endoscopy in a patient with cramping abdominal pain and a diagnosis of celiac disease is described. This revealed flattened mucosal folds that progressed to scalloped mucosa. Shortened villi became visible in the midjejunum. At the approximate point of transition from a flattened to short villous architecture, a transient entero-enteral intussusception was observed. Case r e p o r t . A 60-year-old man presented with dull, diffuse-cramping abdominal discomfort that was worse after meals. He reported a 30-pound weight loss over the preceding year. He weighed 125 pounds and had a body mass index of 17. A complete blood count, lipid panel, and routine laboratory studies were within normal ranges. Upper endoscopy demonstrated scalloped duodenal folds and a mosaic pattern in the second portion of the duodenum. Endoscopic biopsies revealed the villous atrophy and lymphocytic infiltration characteristic of gluten enteropathy. The antigliadin IgA level was 20 units (normal: <20 units), antigliadin IgG 53 units (<20 units), and the antiendomysial antibody titer was less than 1:10 (negative <1:10). The patient was referred for dietary counseling. Approximately 2 months after the initiation of a Supported in part by the Office of Research and Development Medical Research Service Department of Veterans Affairs and by Public Health Service grant DK56338 which funds the Texas Gulf Coast Digestive Diseases Center. Reprint requests: David Y. Graham, MD, Veterans Affairs Medical Center, RM 3A-320(111D), 2002 Holcombe Blvd., Houston, TX 77030. 0016-5107/2003/$30.00 + 0 doi:10.1067/mge.2003.152 626

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Figure 1. Capsule endoscopy view (orig. mag. • at elapsed time of 72 minutes showing flattening of mucosa and prominent vascularity in proximal small bowel. gluten-free diet, the abdominal pain had resolved and the patient had gained weight. Video capsule endoscopy (M2A, Given Imaging, Yoqneam, Israel) was performed to evaluate the extent of small bowel involvement and to investigate the cause of the abdominal pain. The capsule, ingested after an overnight fast, entered the duodenum at 37 minutes and passed through the ileocecal valve at approximately 6 hours and 31 minutes. After 4 hours a light meal consisting of a sandwich and a clear liquid was given. Scalloped duodenal folds were not definitely seen in the proximal small bowel. Rather, the mucosa was flattened, villi were absent, and the vascularity was prominent throughout the proximal small bowel (Fig. 1). At 96 minutes an entero-enteral intussusception was observed (Fig. 2). The intussusception spontaneously resolved after approximately 30 seconds. It occurred at approximately the same point at which the flattened mucosa transitioned to a pattern of mild scalloping or a mosaic pattern with short villi (i.e., at 97 minutes; Fig. 3). The normal villous architecture first became evident after approximately 3.5 hours elapsed time. No other abnormality, such as ulcerative jejunoileitis, was seen. D i s c u s s i o n . Clinically, jejunal intussusception is not frequently considered as being associated with celiac disease; however, the complication may be more common than recognized.4, 5 For example, in one radiographic series, intussusception was noted in 20% of adults with celiac disease, leading the investigators to suggest that the frequency of diagnosis may be related to the intensity of the effort to demonstrate the characteristic appearance. 4 In the present case, capsule endoscopy revealed flattened folds throughout the proximal small bowel, with a transition to a pattern of mild scalloping or a mosaic pattern with short villi in the jejunum. A spontaneous jejuno-jejunal intussusception was noted to occur in the region of transition from grossly effaced to a more raised mucosal architecture. Intussusception is thought to occur as a result of unbalanced contractions when a normal peristaltic wave reaches an abnormal segment of bowel.5,6 Thus, the transition VOLUME 57, NO. 4, 2003