- Email: [email protected]
Biliary fascioliasis: diagnosis, treatment and follow-up by ERCP
usefulness of endoscopy in d i a g n o s i n g a n d t r e a t i n g Dieulafoy's lesion in the rectum, m u c h like the effectiveness of e s o p h a g o g a s t r o d u o d e n o s c o p y for lesions in the u p p e r g a s t r o i n t e s t i n a l tract. Injection of 1:20,000 e p i n e p h r i n e a r o u n d the visible vessel was entirely successful in p r e v e n t i n g f u r t h e r h e m o r r h a g e w i t h o u t the use of t h e r m a l or electrocoagulation. I n o t h e r reports, h e m o s t a s i s w a s achieved with B I C A P cauterization or sclerotherapy, 14 combined s c l e r o t h e r a p y a n d electrocoagnlation, 16 h e a t e r - p r o b e coagulation, 17 or combined e p i n e p h r i n e injection a n d electrocoagnl a t i o n ) 9 We agree t h a t endoscopic t r e a t m e n t should be considered the t r e a t m e n t of choice for a bleeding Dieulafoy's lesion.
1.
2.
3. 4. 5.
6.
REFERENCES Dieulafoy G. Exulceratio simplex. L'intervention chirurgicale dans les h6mat~m~ses foudroyantes consgcutives l'exulceration simple de l'estomac. Bull Acad M6d 1898;49: 49-84 (quoted from reference 2). Veldhuyzen van Zanten SJO, Bartelsman JFWM, Schipper MEI, Tytgat GNJ. Recurrent massive hematemesis from Dieulafoy vascular malformations--a review of 101 cases. Gut 1986;27:213-22. Reilly HF, Al-Kawas FH. Dieulafoy's lesion: diagnosis and management. Dig Dis Sci 1991;36:1702-7. Bozkurt TP, Lederer CP, Lux G. Esophageal visible vessel as a cause of massive upper gastrointestinal hemorrhage. Endoscopy 1991;23:16-8. Rabago L, Mora P, Gea F, Martinez P, Ruiperez J, Campos R. An unusual source of gastrointestinal bleeding from the esophagns: two new cases of esophagus visible vessel. Endoscopy 1993;25:197-8. McClave SA, Goldschmid S, Cunningham JT, Boyd WP. Dieu-
Biliary fascioliasis: diagnosis, treatment and follow-up by ERCP Luis Moreira Dias, Rui Silva, Helena Lomba Viana, Manuel Palhinhas, Rafael Lomba Viana,
MD MD MD MD MD
Fascioliasis is a widely d i s s e m i n a t e d zoonosis c a u s e d by F a s c i o l a h e p a t i c a , a t r e m a t o d e t h a t comm o n l y infests cattle, goats, a n d sheep, the a n i m a l res-
From the Departments of Gastroenterology and Radiology, Instituto Portugu~s de Oncologia do Porto, Portugal. Reprint requests: Luis Moreira Dias, MD, Rua da Torrinha, 80 6 ° Dto Frente, 4050 Porto, Portugal. 0016-5107/96/4306-061655.00 + 0 GASTROINTESTINAL ENDOSCOPY Copyright © 1996 by the American Society for Gastrointestinal Endoscopy
37/4/66183 616 G A S T R O I N T E S T I N A L E N D O S C O P Y
7. 8. 9. 10. 11. 12. 13.
14. 15. 16. 17. 18. 19.
lafoy's cirsoid aneurysm of the duodenum. Dig Dis Sci 1988;33: 801-5. Pollack R, Lipsky H, GoldbergRI. Duodenal Dieulafoy'slesion. Gastrointest Endosc 1993;39:820-2. Rathmel RK, Horwell RJ, Greeley JP. Congenital aneurysm of the jejeunum producing fatal intestinal hemorrhage. Arch Pathol 1951;51:461-5. Matuchansky C, Babin P, Abadie JC, et al. Jejunal bleeding from a solitary large submucosa] artery. Gastroenterology 1978;75:110-3. Barbier P, Luder P, Triller J, Ruchti Ch, Hassler H, Stafford A. Colonic hemorrhage from a solitary minute ulcer. Report of three cases. Gastroenterology 1985;88:1065-8. Richards WO, Grove-Mahoney D, Williams LF. Hemorrhage from a Dieulafoy-like lesion in the colon: a new cause of lower gastrointestinal bleeding. Am Surg 1988;54:121-4. Franko E, Chardavoyne R, Wise L. Massive rectal bleeding from a Dieulafoy'stype ulcer of the rectum: a review of this unusual disease. Am J Gastroenterol 1991;86:1545-7. Abdulian JD, Santoro MJ, Chen YK, Collen MJ. Dieulafoy-like lesion of the rectum presenting with exsangninating hemorrhage: successfulendoscopicsclerotherapy. Am J Gastroenterol 1993;88:1939-41. Boron B, Mobarhan S. Endoscopic treatment of Dieulafoy's hemorrhage. J Clin Gastroenterol 1987;9:518-20. Eidus LB, Rasuli P, Manion D, Heringer R. Caliber-persistent artery of the stomach (Dieulafoy's vascular malformation). Gastroenterology 1990;99:1507-10. Pointner R, Schwab G, Konigsrainer A, Dietze O. Endoscopic treatment of Dieulafoy's disease. Gastroenterology 1988;94: 563-6. Lin HJ, Lee FY, Tsai YT, Lee SD, Lee CH, Kang WM. Therapeutic endoscopy for Dieulafoy's disease. J Clin Gastroenterol 1989;11:507-10. Jaspersen D. Dieulafoy's disease controlled by Doppler ultrasound endoscopictreatment. Gut 1993;34:857-8. Goldenberg SP, DeLuca VA, Marignani P. Endoscopic treatment ofDieulafoys lesion ofthe duodenum. Am J Gastroenterol 1990;85:452-4.
ervoirs of the disease. A large v a r i e t y of domestic a n d wild a n i m a l s can be infected. M a n is a n accidental final host in the life cycle of the p a r a s i t e a n d h u m a n infection u s u a l l y occurs after e a t i n g r a w vegetables, p a r t i c u l a r l y wild watercress, infested with metacercariae. A l t h o u g h infection m a y be a s y m p t o m a t i c , it m a y cause considerable morbidity and, rarely, mortality. We r e p o r t a case of chronic fascioliasis in a p a t i e n t with a 6-year h i s t o r y of i n t e r m i t t e n t biliary colic after h a v i n g ingested uncooked wild watercress. Fascioliasis m u s t be included in the differential diagnosis of the u n u s u a l u l t r a s o n o g r a p h i c (US) a n d c o m p u t e d tomographic (CT) findings presented, w h i c h include biliary dilation a n d a n i r r e g u l a r t h i c k e n i n g of the c o m m o n bile duct (CBD) wall. T h e y correlated well with the endoscopic r e t r o g r a d e cholangiographic (ERCP) pictures. We e m p h a s i z e the value of E R C P in the diagnosis a n d t r e a t m e n t of this parasitosis. A n impressive i m p r o v e m e n t of the biliary abnormalities is s h o w n at a long-term follow-up ERCP. VOLUME 43, NO. 6, 1996
Figure 1. Abdominal US. Dilated biliary tree and diffuse and irregular thickening of the CBD wall, which is 5 mm wide.
CASE REPORT Two weeks before being referred to us in April 1991, this 74-year-old patient had an episode of severe epigastric pain and vomiting. He attended the local hospital and improved over the following hours after receiving intravenous analgesics. His urine became dark, but he had no jaundice or fever. Blood tests showed a normal hemoglobin and white-cell count, without eosinophilia, and the following values: erythrocyte sedimentation rate, 25 mm; serum glutamate oxalocetate transaminase, 84 IU/L (normal <21); serum glutamate pyruvate transaminase, 68 IU/L (normal <22); gammaglutamyl transferase, 317 IU/L (normal <29). Abdominal US revealed a normal liver, gallbladder, and pancreas, and intrahepatic and extrahepatic biliary dilation. The CBD was 8 mm in diameter and its distal wall was diffusely and irregularly thickened (Fig. 1). The patient was then referred to us with suspected cholangiocarcinoma. The patient was in good general condition and lived in northern Portugal. He remembered two similar previous episodes of epigastric pain, 6 and 3 years before, but had remained completely asymptomatic in between them. Physical examination was normal. Laboratory findings included a normal hemogram, total and differential white blood cell count, liver function tests, and urinalysis. Stool specimens were negative for ova and parasites. Abdominal CT scan confirmed the presence of a normal liver, gallbladder, and pancreas, intrahepatic and extrahepatic biliary dilation, as well as an irregular thickening of the distal CBD wall. At ERCP, performed with antibiotic prophylaxis, the papilla of Vater, the pancreatogram, and the gallbladder were normal. There was dilation of the intrahepatic biliary tree, particularly on the left side. The CBD was dilated (10 mm in diameter), with an irregular and spiculated wall and a linear filling defect in its distal part (Fig. 2). A short sphincterotomy was performed and, with a balloon, a living, motile parasite was removed from the CBD. It was captured with a biopsy forceps and, on examination,
VOLUME 43, NO. 6, 1996
Figure 2, ERCP. Dilation of the intrahepatic biliary tree, particularly on the left side. Dilated CBD, with a spiculated wall and a linear filling defect in its distal portion.
proved to be an adult F. hepatica. Praziquantel, 25 mg/kg, three times a day for 1 day, was prescribed. There were no complications. Bile, collected during ERCP, contained F. hepatica eggs and showed no malignant cells. Indirect immunofluorescence and passive hemagglutination serological tests for F. hepatica were reactive at titers of 1/80 and 1/1280, respectively. ELISA test was also positive, with a value of 1.685 (normal <0.400). When specifically asked, the patient remembered the ingestion of wild watercress immediately before the first painful episode, 6 years ago. At a repeat ERCP, 18 months later, a patent sphincterotomy was seen. There was an impressive improvement of the biliary tract abnormalities, with no dilation and a very discrete remaining irregularity of the CBD wall (Fig. 3). Multiple passages with a balloon confirmed the absence of parasites. Bile collected was shown to be negative both for F. hepatica eggs and malignant cells. A repeat passive hemagglutination test revealed that the titer had dropped to 1/80. The patient remains asymptomatic at a 3-year follow-up; his liver function tests are normal.
DISCUSSION The province of Mirtho, n o r t h e r n Portugal, w h e r e this p a t i e n t lives, is a n endemic a r e a for h u m a n F. hepatica infection l"a and, in r u r a l villages, prevalences
GASTROINTESTINAL ENDOSCOPY 617
Figure 3. ERCP (18 months after Fig. 2). Balloon cholangiogram. Nondilated biliary tract. Discrete remaining irregularity of the CBD wall and absence of parasites. Marked improvement as compared with Figure 2.
as high as 33% have been described. 1 A focus of the disease has been found at Caminha, a small town where the patient lives. 1 The disease has not infrequently been reported in France, Spain, the United Kingdom, and many other countries in Europe and around the world, 3 but seems to be rare in the United States, where only one case was reported in the last two decades. 4 H u m a n hepatobiliary infection by the parasite F. hepatica includes two phases: an acute, invasive, hepatic phase, starting 1 to 3 weeks after infestation; and a chronic, biliary phase, beginning 3 to 4 months after the contaminating meal. Although some overlap m a y occur in endemic areas, where repetitive infection occurs and acute lesions are superimposed on chronic disease, 5 clinical and pathologic features of these two phases are different, resulting in distinct diagnostic challenges. 3 During the acute phase, immature worms penetrate the gastrointestinal wall and migrate through the peritoneal cavity, liver capsule, and parenchyma, until they reach the bile ducts. A toxi-infectious hepatitis results from the destruction of liver tissue by the migrating larvae, with general malaise, high fever, severe upper abdominal pain, allergic reactions, 618 GASTROINTESTINAL ENDOSCOPY
hepatomegaly, and intense eosinophilia being prominent features. During the chronic phase, adult flukes remain in the gallbladder and/or the bile ducts, laying eggs, causing inflammation and, eventually, episodes of biliary obstruction. The migration of parasites to organs other than the liver and biliary tract, known as ectopic fascioliasis, may occur. 3 Chatherjee 6 estimated that the life span of the adult fluke in man is between 9 and 13 years. One case with a 16-year evolution has been described. 7 Our patient had probably been infected for at least 6 years. A striking eosinophilia is characteristic of the invasive phase of the disease. It peaks at about 2 to 3 months and then declines progressively, s In the chronic stage, eosinophilia is often absent, as was the case with the patient described. In chronic fascioliasis, the diagnosis is usually based on the identification of F. hepatica eggs, in stool or in duodenal or biliary drainage, and on immunological tests. Because man is an accidental host for this parasite, the number of flukes reaching the biliary tree is usually small, and some of them never reach sexual maturity. In consequence, the number of eggs in the feces is also small and it is not surprising that fewer than 35% of the cases of chronic fascioliasis are diagnosed by parasitologic stool tests, s Duodenal intubation may be necessary for egg detection. The absence of either eosinophilia or detectable eggs in the stool contributed to the delay in diagnosis in our patient. Unlike parasitologic examination for F. hepatica ova, serologic tests allow an early diagnosis of h u m a n fascioliasis during the acute phase. They are also positive in chronic fascioliasis. A number of these tests are useful in clinical practice, immunofluorescence and ELISA being among those with the highest sensitivity and specificity. 3, s No consensus as to the optimal test has been reached and, whenever possible, several tests should be used. s Positive titers return to normal after successful treatment and are useful for monitoring evolution. 3, s Oral cholecystogram has been used to demonstrate endocholecystic fascioliasis, showing a leaf-like structure with a central linear opacity, corresponding to the alimentary canal of the parasite. 9 Both US and CT abnormalities have been described in fascioliasis. Most of the reports concern the acute phase, in which liver parenchyma lesions predominate.5, 10-19 US and CT may also be used in follow-up to evaluate the efficacy of medical therapy.5, 10, 12, 14, 16-1s,20 In chronic fascioliasis, the number of reported cases with imaging findings is small. Abdominal US may either be normal 15, 21 or show mobile vermiform structures without acoustic shadowing within the gallbladder 5' 9, 15-17,20, 22-24 and in the bile ducts,15, 23 representing worms, that may be confused with stones. 25 Duct dilation may also be seen.15, 16, 20, 25, 2~ Adult flukes, either due to a direct irritating effect or perhaps to the induction of a high VOLUME 43, NO. 6, 1996
proline concentration in bile, promote hyperplasia and hypertrophy of the duct epithelium and enveloping periductal fibrosis, 27, 28 resulting in thickening of the duct walls. Irregular thickening of the CBD wall on abdominal US and CT in biliary fascioliasis has previously been reported in only three other patients.16, 18,20 CT was normal in a fourth patient. 14 Similar abnormal US and CT findings have been reported in sclerosing cholangitis 29, 30 and AIDS-related cholangitisY These findings, although nonspecific, correlated well with the cholangiographic pictures obtained at ERCP in our case. ERCP was normal in two patients with acute fascioliasis. 12 In biliary fascioliasis, ERCP may show typical pictures of F. hepatica parasites in the gallbladder,23, 25 dilated bile ducts with small, radiolucent, linear or crescent-like shadows, suggesting worms,* and with jagged, irregular margins. 16, is, 20 Similar pictures have been described on percutaneous 26, 34 as well as perioperative 35,36 cholangiography. ERCP pictures of chronic fascioliasis have been misinterpreted as primary sclerosing cholangitis. 4 Chronic biliary fascioliasis may be asymptomatic in an unknown proportion of cases. When left without treatment, however, it m a y produce biliary complications, including iron deficiency anaemia, biliary obstruction and pain, cholangitis, s and portal f i b r o s i s . 37 Severe hemobilia and death have been described.3, 33, 38, 39 Associated lithiasis of the bile ducts or of the gallbladder is frequent, 3, 33 inasmuch as eggs or fragments of dead parasites m a y constitute the nucleus for calculi. Acute pancreatitis as a complication of biliary fascioliasis has been reported only three times in the literature. 4, 34, 40 A number of drugs have been proposed for the treatment of h u m a n fascioliasis, including emetine and dehydroemetine, chloroquine, bithionol, niclofolan, metronidazole, albendazole, triclabendazole, and praziquantel. 3 Reported efficacy for eradication of the parasite is variable, with some patients requiring repeated trials of the same or different drugs. 2° The most appropriate medical therapy remains to be established. Chronic infection is more difficult to treat t h a n acute disease. Biliary disease caused by unsuspected fascioliasis has frequently required surgery.4, 18, 33-35, 37, 39, 41 Endoscopic sphincterotomy was introduced for the treatment of CBD stones. Indications for this technique have progressively been widened to include other biliary disorders. It has been considered the optimal approach to treat biliary parasitosis, such as biliary ascariasis 42 and biliary hydatid disease. 43 Previously, ERCP and sphincterotomy have been used with success to extract F. hepatica parasites from the biliary tree in only one patient. 34 In fascioliasis, as in *References 16, 18, 20, 21, 23, 32-34.
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all other diseases with biliary obstruction, cholangitis may complicate ERCP if adequate drainage is not achieved. 2° In our experience, a short sphincterotomy is enough to achieve the removal of these soft parasites. Therefore the complication rate of this procedure is supposedly smaller than that of the larger sphincterotomies required for stone removal. A marked improvement of the CBD wall abnormalities, at a follow-up ERCP after sphincterotomy andF. hepatica removal, had not been reported so far. Our findings demonstrate that these abnormalities result from persistent infestation and obstruction and are potentially reversible after CBD cleaning. Residual CBD irregularity is possibly explained by irreversible periductal fibrosis. 27, 28 In conclusion, fascioliasis may be overlooked in chronic cases. It should be included in the differential diagnosis of irregular and thickened CBD wall images obtained by US or CT. ERCP findings are suggestive of biliary fascioliasis, particularly when typical iraages ofF. hepatica are seen. In cases of dubious diagnosis or of failed medical treatment, endoscopic sphincterotomy and removal of parasites should be considered the therapeutic option for biliary fascioliasis. ACKNOWLEDGMENTS
The authors are grateful to Dr. Isabel Gomes, Department of Microbiology, Instituto Portugu~s de Oncologia do Porto, and Dr. Maria Agueda, Instituto Nacional de Safide Dr. Ricardo Jorge-Porto, for their kind assistance. REFERENCES 1. Silva MLS, Capron A, Capron M. Human fascioliasis in Portugal. Arquivos do Instituto Nacional de Safide 1980;4:101-9. 2. Rombert PC, Gr~cio MA. Fascioliase hepgtiea humana: Sua distribuicao em Portugal. O M6dico 1984;110:77-83. 3. Chen MG, Mott KE. Progress in assessment of morbidity due to Fasciola hepatica infection: a review of recent literature. Tropical Diseases Bulletin 1990;87:R1-38. 4. Hauser SC, Bynum TE. Abnormalities on ERCP in a case of human fascioliasis. Gastrointest Endosc 1984;30:80-2. 5. Pulpeiro JR, Armesto V, Varela J, Corredoira J. Fascioliasis: findings in 15 patients. Br J Radiol 1991;64:798-801. 6. Chatterjee KD. Parasitology (protozoology and helminthology). 10th Ed. Calcutta: S. N. Guha Ray At Sree Saraswaty Press Ltd, 1975:146-8. 7. Reinhard GH, Graf V, Augustin HJ. Chronic fascioliasis with destructive cholangitis. Fortschr Med 1991;109:737-8. 8. Ambroise-Thomas P, Goullier A, Peyron F. Les Distomatoses. Encycl M6d Chir (Paris), Maladies Infectieuses 1986;8110:1-10. 9. Pandolfo I, Zimbaro G, Bartiromo G, et al. Ultrasonographic and cholecystographic findings in a case of fascioliasis. J Clin Ultrasound 1991;19:505-7. 10. de Miguel F, Carrasco J, Garcia N, Bustamante V, Beltr~n J. CT findings in human fascioliasis. Gastrointest Radio11984;9:157-9. 11. Karabinis A, Herson S, Brucker G, et al. Fasciola hepatica abscesses: value of hepatic ultrasonography. Ann Med Interne (Paris) 1985;136:575-8. 12. Takeyama N, Okumura N, Sakai Y, et al. Computed tomography findings of hepatic lesions in human fascioliasis: report of two cases. Am J Gastroenterol 1986;81:1078-81. 13. Cauquil P, Pariente D, Loyer E, Lallemand D. Unusual sonographic appearance of a case of hepatic distomatosis. J Radiol 1986;67:715-7.
GASTROINTESTINAL ENDOSCOPY 619
14. Serrano MAP, Vega A, Ortega E, Gonzalez A. Computed tomography of hepatic fascioliasis. J Comput Assist Tomogr 1987;11:269-72. 15. Bassily S, Iskander M, Youssef FG, E1-Masry N, Bawden M. Sonography in diagnosis of fascioliasis. Lancet 1989;1:1270-1. 16. Van Beers B, Pringot J, Geubel A, Trigaux J-P, Bigaignon G, Dooms G. Hepatobiliary fascioliasis: noninvasiveimaging findings. Radiology 1990;174:809-10. 17. Rebollo MLR, Uriarte MLB, Schneider FB, Landeta LM, Urla JDC. Fontaneda ED. Ecografia en el diagn6stico de la fascioliasis. Rev Esp Enferm Dig 1991;79:297-8. 18. Han JK, Choi BI, Cho JM, Chung KB, Han MC, Kim CW. Radiological findings in human fascioliasis. Abdom Imaging 1993;18:261-4. 19. Stark ME, Herrington DA, Hillyer GV, McGill DB. An international traveler with fever, abdominal pain, eosinophilia and a liver lesion. Gastroenterology 1993;105:1900-8. 20. De Ronde T, M6lange M, Beers BV, et al. Distomatose des voles biliaires. Interet de la cholangiographie retrograde. Efficacite du triclabendazole. Acta C]in Belg 1992;47:209-14. 21. Ros6s LL, Alonso D, Ifiiguez F, Mateos A, Bal M, Agtiero J. Hepatic fascioliasis of long term evolution: diagnosis by ERCP. Am J Gastroenterol 1993;88:2118-9. 22. Eisenscher A, Sauget Y. Aspect ultrasonore des ascaridoses et distomatoses des voles biliaires. J Radiol 1980;61:319-22. 23. Bonniaud P, Barth616my C, Veyret C, Audigier JC, Fraisse H. Aspect ultrasonore de la distomatose des voles biliaires. J Radiol 1984;65:589-91. 24. Fawzy RK, Salem AE, Osman MM. Ultrasonographic findings in the gallbladder in human fascioliasis. J Egypt Soc Parasitol 1992;22:827-31. 25. Heredia D, Bordas JM, Mondelo F, Rod6s J. Distomatosis vesicular en una paciente portadora de cirrosis hepatica. Med Clin (Barc) 1984;82:768-70. 26. Condomines J, Refie-Espinet JM, Espinos-Perez JC, Vilardell F. Percutaneous cholangiography in the diagnosis of hepatic fascioliasis. Am J Gastroenterol 1985;80:384-6. 27. IsseroffH, Sawma JT, Reino D. Fascioliasis--role ofproline in bile duct hyperplasia. Science 1977;198:1157-9. 28. Foster JR. A study ofthe initiation ofbiliary hyperplasia in rats infected with Fasciola hepatica. Parasitology 1981;83:253-8.
29. Carrol BA, Oppenheimer DA. Sclerosing cholangitis: sonographic demonstration of bile duct wall thickening. AJR Am J Roentgenol 1982;139:1016-8. 30. Teefey SA, Baron RL, Rohrmann CA, Schuman WP, Freeny PC. Sclerosing cholangitis: CT findings. Radiology 1988;169:635-9. 31. Dolmatch BL, Laing FC, Federle MP, Jeffrey RB, Cello J. AIDS-related cholangitis: radiographic findings in nine patients. Radiology 1987;163:313-6. 32. Orive V, Goyeneche N, Cosme A, Alzate LF, Enciso C, Arenas JI. Aportacion de la CPRE al diagn6stico de la fasciolasis cronica [Abstract]. Rev Esp Enferm Dig 1984;65(suppl II):58. 33. Wong RKH, Peura DA, Mutter ML, Heit HA, Birns MT, Johnson LF. Hemobilia and liver flukes in a patient from Thailand. Gastroenterology 1985;88:1958-63. 34. Veerappan A, Siegel JH, Podany J, Prudente R, Gelb A. Fasciola hepatica pancreatitis: endoscopic extraction of live parasites. Gastrointest Endosc 1991;37:473-5. 35. Belgraier AH. Common bile duct obstruction due to Fasciola hepatica. N Y State J Med 1976;76:936-7. 36. Donnelly B, Hederman WP. Liver fluke in the common bile duct. Ir Med J 1977;70:507-9. 37. Jones EA, Kay JM, Milligan HP, Owens D. Massive infection with Fasciola hepatica in man. Gastroenterology 1977;63:83642. 38. Goodman MA, Henderson JI, Cu]lity GJ. Fascioliasis causing jaundice and intestinal bleeding. Med J Aust 1973;2:547-50. 39. Acuna-Soto R, Braun-Roth G. Bleeding ulcer in the common bile duct due to Fasciola hepatica. Am J Gastroenterol 1987; 82:560-2. 40. Maroy R, Moullot P, Daloubeix H, Mathey JC. Pancreatite aigue compliquant une distomatose biliare a Fasciola hepatica chez un patient porteur d'un diverticule choledocien [Abstract]. Ann Gastroenterol Hepatol (Paris) 1987;23:67-70. 41. Kayabali I, Gokcora IH, Yerdel MA, Ormeci N. Hepatic fascioliasis and biliary surgery. Int Surg 1992;77:154-7. 42. Khuroo MS, Zargar SA, Mahajan R. Hepatobiliary and pancreatic ascariasis in India. Lancet 1990;335:1503-6. 43. A1Karawi MA, Yasawy MI, Mohamed ARES. Endoscopic management of biliary hydatid disease: report of six cases. Endoscopy 1991;23:278-81.
Gastrointestinal lymphangioma: endosonographic demonstration and endoscopic removal
Lymphangiomas commonly develop in the head or neck where the primitive lymph sac is formed, whereas gastrointestinal lymphangiomas are extremely rare. Fleming and Carlson 1 reported that radiographic recognition was less than one case per 100,000 individuals examined. Recently, the advance and widespread use of endoscopy and endoscopic polypectomy has enabled physicians to diagnose the tumor precisely. 25 It was anticipated that endoscopic ultrasonography (EUS) would provide valuable findings in this peculiar tumor. However, because of its rarity, detailed descriptions of the tumor are as yet extremely limited. 67 , We describe five patients with lymphangioma in whom the tumors were located in the stomach in two, the duodenum in one, and the colon in two. All the lesions were evaluated with EUS before endoscopic polypectomy.
Kazuoki Hizawa, Kunihiko Aoyagi, Kouichi Kurahara, Hiroshi Suekane, Yasuyuki Kuwano, Shoutaro Nakamura, Masatoshi Fujishima,
MD MD MD MD MD MD MD
From the Second Department of Internal Medicine and the Second Department of Pathology, Faculty of Medicine, Kyushu University, Fukuoka, Japan. Reprint requests: Kazuoki Hizawa, MD, Second Department of Internal Medicine, Kyushu University, Maidashi 3-1-1, Fukuoka 812, Japan. 0016-5107/96/4306-062055.00 +0 GASTROINTESTINAL ENDOSCOPY Copyright © 1996 by the American Society for Gastrointestinal Endoscopy 37/4/66182
620 GASTROINTESTINAL ENDOSCOPY
PATIENTS AND METHODS Between November 1991 and August 1994, five patients with the histologic diagnosis of gastrointestinal lymphangiVOLUME 43, NO. 6, 1996
1.
2.
3. 4. 5.
6.
REFERENCES Dieulafoy G. Exulceratio simplex. L'intervention chirurgicale dans les h6mat~m~ses foudroyantes consgcutives l'exulceration simple de l'estomac. Bull Acad M6d 1898;49: 49-84 (quoted from reference 2). Veldhuyzen van Zanten SJO, Bartelsman JFWM, Schipper MEI, Tytgat GNJ. Recurrent massive hematemesis from Dieulafoy vascular malformations--a review of 101 cases. Gut 1986;27:213-22. Reilly HF, Al-Kawas FH. Dieulafoy's lesion: diagnosis and management. Dig Dis Sci 1991;36:1702-7. Bozkurt TP, Lederer CP, Lux G. Esophageal visible vessel as a cause of massive upper gastrointestinal hemorrhage. Endoscopy 1991;23:16-8. Rabago L, Mora P, Gea F, Martinez P, Ruiperez J, Campos R. An unusual source of gastrointestinal bleeding from the esophagns: two new cases of esophagus visible vessel. Endoscopy 1993;25:197-8. McClave SA, Goldschmid S, Cunningham JT, Boyd WP. Dieu-
Biliary fascioliasis: diagnosis, treatment and follow-up by ERCP Luis Moreira Dias, Rui Silva, Helena Lomba Viana, Manuel Palhinhas, Rafael Lomba Viana,
MD MD MD MD MD
Fascioliasis is a widely d i s s e m i n a t e d zoonosis c a u s e d by F a s c i o l a h e p a t i c a , a t r e m a t o d e t h a t comm o n l y infests cattle, goats, a n d sheep, the a n i m a l res-
From the Departments of Gastroenterology and Radiology, Instituto Portugu~s de Oncologia do Porto, Portugal. Reprint requests: Luis Moreira Dias, MD, Rua da Torrinha, 80 6 ° Dto Frente, 4050 Porto, Portugal. 0016-5107/96/4306-061655.00 + 0 GASTROINTESTINAL ENDOSCOPY Copyright © 1996 by the American Society for Gastrointestinal Endoscopy
37/4/66183 616 G A S T R O I N T E S T I N A L E N D O S C O P Y
7. 8. 9. 10. 11. 12. 13.
14. 15. 16. 17. 18. 19.
lafoy's cirsoid aneurysm of the duodenum. Dig Dis Sci 1988;33: 801-5. Pollack R, Lipsky H, GoldbergRI. Duodenal Dieulafoy'slesion. Gastrointest Endosc 1993;39:820-2. Rathmel RK, Horwell RJ, Greeley JP. Congenital aneurysm of the jejeunum producing fatal intestinal hemorrhage. Arch Pathol 1951;51:461-5. Matuchansky C, Babin P, Abadie JC, et al. Jejunal bleeding from a solitary large submucosa] artery. Gastroenterology 1978;75:110-3. Barbier P, Luder P, Triller J, Ruchti Ch, Hassler H, Stafford A. Colonic hemorrhage from a solitary minute ulcer. Report of three cases. Gastroenterology 1985;88:1065-8. Richards WO, Grove-Mahoney D, Williams LF. Hemorrhage from a Dieulafoy-like lesion in the colon: a new cause of lower gastrointestinal bleeding. Am Surg 1988;54:121-4. Franko E, Chardavoyne R, Wise L. Massive rectal bleeding from a Dieulafoy'stype ulcer of the rectum: a review of this unusual disease. Am J Gastroenterol 1991;86:1545-7. Abdulian JD, Santoro MJ, Chen YK, Collen MJ. Dieulafoy-like lesion of the rectum presenting with exsangninating hemorrhage: successfulendoscopicsclerotherapy. Am J Gastroenterol 1993;88:1939-41. Boron B, Mobarhan S. Endoscopic treatment of Dieulafoy's hemorrhage. J Clin Gastroenterol 1987;9:518-20. Eidus LB, Rasuli P, Manion D, Heringer R. Caliber-persistent artery of the stomach (Dieulafoy's vascular malformation). Gastroenterology 1990;99:1507-10. Pointner R, Schwab G, Konigsrainer A, Dietze O. Endoscopic treatment of Dieulafoy's disease. Gastroenterology 1988;94: 563-6. Lin HJ, Lee FY, Tsai YT, Lee SD, Lee CH, Kang WM. Therapeutic endoscopy for Dieulafoy's disease. J Clin Gastroenterol 1989;11:507-10. Jaspersen D. Dieulafoy's disease controlled by Doppler ultrasound endoscopictreatment. Gut 1993;34:857-8. Goldenberg SP, DeLuca VA, Marignani P. Endoscopic treatment ofDieulafoys lesion ofthe duodenum. Am J Gastroenterol 1990;85:452-4.
ervoirs of the disease. A large v a r i e t y of domestic a n d wild a n i m a l s can be infected. M a n is a n accidental final host in the life cycle of the p a r a s i t e a n d h u m a n infection u s u a l l y occurs after e a t i n g r a w vegetables, p a r t i c u l a r l y wild watercress, infested with metacercariae. A l t h o u g h infection m a y be a s y m p t o m a t i c , it m a y cause considerable morbidity and, rarely, mortality. We r e p o r t a case of chronic fascioliasis in a p a t i e n t with a 6-year h i s t o r y of i n t e r m i t t e n t biliary colic after h a v i n g ingested uncooked wild watercress. Fascioliasis m u s t be included in the differential diagnosis of the u n u s u a l u l t r a s o n o g r a p h i c (US) a n d c o m p u t e d tomographic (CT) findings presented, w h i c h include biliary dilation a n d a n i r r e g u l a r t h i c k e n i n g of the c o m m o n bile duct (CBD) wall. T h e y correlated well with the endoscopic r e t r o g r a d e cholangiographic (ERCP) pictures. We e m p h a s i z e the value of E R C P in the diagnosis a n d t r e a t m e n t of this parasitosis. A n impressive i m p r o v e m e n t of the biliary abnormalities is s h o w n at a long-term follow-up ERCP. VOLUME 43, NO. 6, 1996
Figure 1. Abdominal US. Dilated biliary tree and diffuse and irregular thickening of the CBD wall, which is 5 mm wide.
CASE REPORT Two weeks before being referred to us in April 1991, this 74-year-old patient had an episode of severe epigastric pain and vomiting. He attended the local hospital and improved over the following hours after receiving intravenous analgesics. His urine became dark, but he had no jaundice or fever. Blood tests showed a normal hemoglobin and white-cell count, without eosinophilia, and the following values: erythrocyte sedimentation rate, 25 mm; serum glutamate oxalocetate transaminase, 84 IU/L (normal <21); serum glutamate pyruvate transaminase, 68 IU/L (normal <22); gammaglutamyl transferase, 317 IU/L (normal <29). Abdominal US revealed a normal liver, gallbladder, and pancreas, and intrahepatic and extrahepatic biliary dilation. The CBD was 8 mm in diameter and its distal wall was diffusely and irregularly thickened (Fig. 1). The patient was then referred to us with suspected cholangiocarcinoma. The patient was in good general condition and lived in northern Portugal. He remembered two similar previous episodes of epigastric pain, 6 and 3 years before, but had remained completely asymptomatic in between them. Physical examination was normal. Laboratory findings included a normal hemogram, total and differential white blood cell count, liver function tests, and urinalysis. Stool specimens were negative for ova and parasites. Abdominal CT scan confirmed the presence of a normal liver, gallbladder, and pancreas, intrahepatic and extrahepatic biliary dilation, as well as an irregular thickening of the distal CBD wall. At ERCP, performed with antibiotic prophylaxis, the papilla of Vater, the pancreatogram, and the gallbladder were normal. There was dilation of the intrahepatic biliary tree, particularly on the left side. The CBD was dilated (10 mm in diameter), with an irregular and spiculated wall and a linear filling defect in its distal part (Fig. 2). A short sphincterotomy was performed and, with a balloon, a living, motile parasite was removed from the CBD. It was captured with a biopsy forceps and, on examination,
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Figure 2, ERCP. Dilation of the intrahepatic biliary tree, particularly on the left side. Dilated CBD, with a spiculated wall and a linear filling defect in its distal portion.
proved to be an adult F. hepatica. Praziquantel, 25 mg/kg, three times a day for 1 day, was prescribed. There were no complications. Bile, collected during ERCP, contained F. hepatica eggs and showed no malignant cells. Indirect immunofluorescence and passive hemagglutination serological tests for F. hepatica were reactive at titers of 1/80 and 1/1280, respectively. ELISA test was also positive, with a value of 1.685 (normal <0.400). When specifically asked, the patient remembered the ingestion of wild watercress immediately before the first painful episode, 6 years ago. At a repeat ERCP, 18 months later, a patent sphincterotomy was seen. There was an impressive improvement of the biliary tract abnormalities, with no dilation and a very discrete remaining irregularity of the CBD wall (Fig. 3). Multiple passages with a balloon confirmed the absence of parasites. Bile collected was shown to be negative both for F. hepatica eggs and malignant cells. A repeat passive hemagglutination test revealed that the titer had dropped to 1/80. The patient remains asymptomatic at a 3-year follow-up; his liver function tests are normal.
DISCUSSION The province of Mirtho, n o r t h e r n Portugal, w h e r e this p a t i e n t lives, is a n endemic a r e a for h u m a n F. hepatica infection l"a and, in r u r a l villages, prevalences
GASTROINTESTINAL ENDOSCOPY 617
Figure 3. ERCP (18 months after Fig. 2). Balloon cholangiogram. Nondilated biliary tract. Discrete remaining irregularity of the CBD wall and absence of parasites. Marked improvement as compared with Figure 2.
as high as 33% have been described. 1 A focus of the disease has been found at Caminha, a small town where the patient lives. 1 The disease has not infrequently been reported in France, Spain, the United Kingdom, and many other countries in Europe and around the world, 3 but seems to be rare in the United States, where only one case was reported in the last two decades. 4 H u m a n hepatobiliary infection by the parasite F. hepatica includes two phases: an acute, invasive, hepatic phase, starting 1 to 3 weeks after infestation; and a chronic, biliary phase, beginning 3 to 4 months after the contaminating meal. Although some overlap m a y occur in endemic areas, where repetitive infection occurs and acute lesions are superimposed on chronic disease, 5 clinical and pathologic features of these two phases are different, resulting in distinct diagnostic challenges. 3 During the acute phase, immature worms penetrate the gastrointestinal wall and migrate through the peritoneal cavity, liver capsule, and parenchyma, until they reach the bile ducts. A toxi-infectious hepatitis results from the destruction of liver tissue by the migrating larvae, with general malaise, high fever, severe upper abdominal pain, allergic reactions, 618 GASTROINTESTINAL ENDOSCOPY
hepatomegaly, and intense eosinophilia being prominent features. During the chronic phase, adult flukes remain in the gallbladder and/or the bile ducts, laying eggs, causing inflammation and, eventually, episodes of biliary obstruction. The migration of parasites to organs other than the liver and biliary tract, known as ectopic fascioliasis, may occur. 3 Chatherjee 6 estimated that the life span of the adult fluke in man is between 9 and 13 years. One case with a 16-year evolution has been described. 7 Our patient had probably been infected for at least 6 years. A striking eosinophilia is characteristic of the invasive phase of the disease. It peaks at about 2 to 3 months and then declines progressively, s In the chronic stage, eosinophilia is often absent, as was the case with the patient described. In chronic fascioliasis, the diagnosis is usually based on the identification of F. hepatica eggs, in stool or in duodenal or biliary drainage, and on immunological tests. Because man is an accidental host for this parasite, the number of flukes reaching the biliary tree is usually small, and some of them never reach sexual maturity. In consequence, the number of eggs in the feces is also small and it is not surprising that fewer than 35% of the cases of chronic fascioliasis are diagnosed by parasitologic stool tests, s Duodenal intubation may be necessary for egg detection. The absence of either eosinophilia or detectable eggs in the stool contributed to the delay in diagnosis in our patient. Unlike parasitologic examination for F. hepatica ova, serologic tests allow an early diagnosis of h u m a n fascioliasis during the acute phase. They are also positive in chronic fascioliasis. A number of these tests are useful in clinical practice, immunofluorescence and ELISA being among those with the highest sensitivity and specificity. 3, s No consensus as to the optimal test has been reached and, whenever possible, several tests should be used. s Positive titers return to normal after successful treatment and are useful for monitoring evolution. 3, s Oral cholecystogram has been used to demonstrate endocholecystic fascioliasis, showing a leaf-like structure with a central linear opacity, corresponding to the alimentary canal of the parasite. 9 Both US and CT abnormalities have been described in fascioliasis. Most of the reports concern the acute phase, in which liver parenchyma lesions predominate.5, 10-19 US and CT may also be used in follow-up to evaluate the efficacy of medical therapy.5, 10, 12, 14, 16-1s,20 In chronic fascioliasis, the number of reported cases with imaging findings is small. Abdominal US may either be normal 15, 21 or show mobile vermiform structures without acoustic shadowing within the gallbladder 5' 9, 15-17,20, 22-24 and in the bile ducts,15, 23 representing worms, that may be confused with stones. 25 Duct dilation may also be seen.15, 16, 20, 25, 2~ Adult flukes, either due to a direct irritating effect or perhaps to the induction of a high VOLUME 43, NO. 6, 1996
proline concentration in bile, promote hyperplasia and hypertrophy of the duct epithelium and enveloping periductal fibrosis, 27, 28 resulting in thickening of the duct walls. Irregular thickening of the CBD wall on abdominal US and CT in biliary fascioliasis has previously been reported in only three other patients.16, 18,20 CT was normal in a fourth patient. 14 Similar abnormal US and CT findings have been reported in sclerosing cholangitis 29, 30 and AIDS-related cholangitisY These findings, although nonspecific, correlated well with the cholangiographic pictures obtained at ERCP in our case. ERCP was normal in two patients with acute fascioliasis. 12 In biliary fascioliasis, ERCP may show typical pictures of F. hepatica parasites in the gallbladder,23, 25 dilated bile ducts with small, radiolucent, linear or crescent-like shadows, suggesting worms,* and with jagged, irregular margins. 16, is, 20 Similar pictures have been described on percutaneous 26, 34 as well as perioperative 35,36 cholangiography. ERCP pictures of chronic fascioliasis have been misinterpreted as primary sclerosing cholangitis. 4 Chronic biliary fascioliasis may be asymptomatic in an unknown proportion of cases. When left without treatment, however, it m a y produce biliary complications, including iron deficiency anaemia, biliary obstruction and pain, cholangitis, s and portal f i b r o s i s . 37 Severe hemobilia and death have been described.3, 33, 38, 39 Associated lithiasis of the bile ducts or of the gallbladder is frequent, 3, 33 inasmuch as eggs or fragments of dead parasites m a y constitute the nucleus for calculi. Acute pancreatitis as a complication of biliary fascioliasis has been reported only three times in the literature. 4, 34, 40 A number of drugs have been proposed for the treatment of h u m a n fascioliasis, including emetine and dehydroemetine, chloroquine, bithionol, niclofolan, metronidazole, albendazole, triclabendazole, and praziquantel. 3 Reported efficacy for eradication of the parasite is variable, with some patients requiring repeated trials of the same or different drugs. 2° The most appropriate medical therapy remains to be established. Chronic infection is more difficult to treat t h a n acute disease. Biliary disease caused by unsuspected fascioliasis has frequently required surgery.4, 18, 33-35, 37, 39, 41 Endoscopic sphincterotomy was introduced for the treatment of CBD stones. Indications for this technique have progressively been widened to include other biliary disorders. It has been considered the optimal approach to treat biliary parasitosis, such as biliary ascariasis 42 and biliary hydatid disease. 43 Previously, ERCP and sphincterotomy have been used with success to extract F. hepatica parasites from the biliary tree in only one patient. 34 In fascioliasis, as in *References 16, 18, 20, 21, 23, 32-34.
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all other diseases with biliary obstruction, cholangitis may complicate ERCP if adequate drainage is not achieved. 2° In our experience, a short sphincterotomy is enough to achieve the removal of these soft parasites. Therefore the complication rate of this procedure is supposedly smaller than that of the larger sphincterotomies required for stone removal. A marked improvement of the CBD wall abnormalities, at a follow-up ERCP after sphincterotomy andF. hepatica removal, had not been reported so far. Our findings demonstrate that these abnormalities result from persistent infestation and obstruction and are potentially reversible after CBD cleaning. Residual CBD irregularity is possibly explained by irreversible periductal fibrosis. 27, 28 In conclusion, fascioliasis may be overlooked in chronic cases. It should be included in the differential diagnosis of irregular and thickened CBD wall images obtained by US or CT. ERCP findings are suggestive of biliary fascioliasis, particularly when typical iraages ofF. hepatica are seen. In cases of dubious diagnosis or of failed medical treatment, endoscopic sphincterotomy and removal of parasites should be considered the therapeutic option for biliary fascioliasis. ACKNOWLEDGMENTS
The authors are grateful to Dr. Isabel Gomes, Department of Microbiology, Instituto Portugu~s de Oncologia do Porto, and Dr. Maria Agueda, Instituto Nacional de Safide Dr. Ricardo Jorge-Porto, for their kind assistance. REFERENCES 1. Silva MLS, Capron A, Capron M. Human fascioliasis in Portugal. Arquivos do Instituto Nacional de Safide 1980;4:101-9. 2. Rombert PC, Gr~cio MA. Fascioliase hepgtiea humana: Sua distribuicao em Portugal. O M6dico 1984;110:77-83. 3. Chen MG, Mott KE. Progress in assessment of morbidity due to Fasciola hepatica infection: a review of recent literature. Tropical Diseases Bulletin 1990;87:R1-38. 4. Hauser SC, Bynum TE. Abnormalities on ERCP in a case of human fascioliasis. Gastrointest Endosc 1984;30:80-2. 5. Pulpeiro JR, Armesto V, Varela J, Corredoira J. Fascioliasis: findings in 15 patients. Br J Radiol 1991;64:798-801. 6. Chatterjee KD. Parasitology (protozoology and helminthology). 10th Ed. Calcutta: S. N. Guha Ray At Sree Saraswaty Press Ltd, 1975:146-8. 7. Reinhard GH, Graf V, Augustin HJ. Chronic fascioliasis with destructive cholangitis. Fortschr Med 1991;109:737-8. 8. Ambroise-Thomas P, Goullier A, Peyron F. Les Distomatoses. Encycl M6d Chir (Paris), Maladies Infectieuses 1986;8110:1-10. 9. Pandolfo I, Zimbaro G, Bartiromo G, et al. Ultrasonographic and cholecystographic findings in a case of fascioliasis. J Clin Ultrasound 1991;19:505-7. 10. de Miguel F, Carrasco J, Garcia N, Bustamante V, Beltr~n J. CT findings in human fascioliasis. Gastrointest Radio11984;9:157-9. 11. Karabinis A, Herson S, Brucker G, et al. Fasciola hepatica abscesses: value of hepatic ultrasonography. Ann Med Interne (Paris) 1985;136:575-8. 12. Takeyama N, Okumura N, Sakai Y, et al. Computed tomography findings of hepatic lesions in human fascioliasis: report of two cases. Am J Gastroenterol 1986;81:1078-81. 13. Cauquil P, Pariente D, Loyer E, Lallemand D. Unusual sonographic appearance of a case of hepatic distomatosis. J Radiol 1986;67:715-7.
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14. Serrano MAP, Vega A, Ortega E, Gonzalez A. Computed tomography of hepatic fascioliasis. J Comput Assist Tomogr 1987;11:269-72. 15. Bassily S, Iskander M, Youssef FG, E1-Masry N, Bawden M. Sonography in diagnosis of fascioliasis. Lancet 1989;1:1270-1. 16. Van Beers B, Pringot J, Geubel A, Trigaux J-P, Bigaignon G, Dooms G. Hepatobiliary fascioliasis: noninvasiveimaging findings. Radiology 1990;174:809-10. 17. Rebollo MLR, Uriarte MLB, Schneider FB, Landeta LM, Urla JDC. Fontaneda ED. Ecografia en el diagn6stico de la fascioliasis. Rev Esp Enferm Dig 1991;79:297-8. 18. Han JK, Choi BI, Cho JM, Chung KB, Han MC, Kim CW. Radiological findings in human fascioliasis. Abdom Imaging 1993;18:261-4. 19. Stark ME, Herrington DA, Hillyer GV, McGill DB. An international traveler with fever, abdominal pain, eosinophilia and a liver lesion. Gastroenterology 1993;105:1900-8. 20. De Ronde T, M6lange M, Beers BV, et al. Distomatose des voles biliaires. Interet de la cholangiographie retrograde. Efficacite du triclabendazole. Acta C]in Belg 1992;47:209-14. 21. Ros6s LL, Alonso D, Ifiiguez F, Mateos A, Bal M, Agtiero J. Hepatic fascioliasis of long term evolution: diagnosis by ERCP. Am J Gastroenterol 1993;88:2118-9. 22. Eisenscher A, Sauget Y. Aspect ultrasonore des ascaridoses et distomatoses des voles biliaires. J Radiol 1980;61:319-22. 23. Bonniaud P, Barth616my C, Veyret C, Audigier JC, Fraisse H. Aspect ultrasonore de la distomatose des voles biliaires. J Radiol 1984;65:589-91. 24. Fawzy RK, Salem AE, Osman MM. Ultrasonographic findings in the gallbladder in human fascioliasis. J Egypt Soc Parasitol 1992;22:827-31. 25. Heredia D, Bordas JM, Mondelo F, Rod6s J. Distomatosis vesicular en una paciente portadora de cirrosis hepatica. Med Clin (Barc) 1984;82:768-70. 26. Condomines J, Refie-Espinet JM, Espinos-Perez JC, Vilardell F. Percutaneous cholangiography in the diagnosis of hepatic fascioliasis. Am J Gastroenterol 1985;80:384-6. 27. IsseroffH, Sawma JT, Reino D. Fascioliasis--role ofproline in bile duct hyperplasia. Science 1977;198:1157-9. 28. Foster JR. A study ofthe initiation ofbiliary hyperplasia in rats infected with Fasciola hepatica. Parasitology 1981;83:253-8.
29. Carrol BA, Oppenheimer DA. Sclerosing cholangitis: sonographic demonstration of bile duct wall thickening. AJR Am J Roentgenol 1982;139:1016-8. 30. Teefey SA, Baron RL, Rohrmann CA, Schuman WP, Freeny PC. Sclerosing cholangitis: CT findings. Radiology 1988;169:635-9. 31. Dolmatch BL, Laing FC, Federle MP, Jeffrey RB, Cello J. AIDS-related cholangitis: radiographic findings in nine patients. Radiology 1987;163:313-6. 32. Orive V, Goyeneche N, Cosme A, Alzate LF, Enciso C, Arenas JI. Aportacion de la CPRE al diagn6stico de la fasciolasis cronica [Abstract]. Rev Esp Enferm Dig 1984;65(suppl II):58. 33. Wong RKH, Peura DA, Mutter ML, Heit HA, Birns MT, Johnson LF. Hemobilia and liver flukes in a patient from Thailand. Gastroenterology 1985;88:1958-63. 34. Veerappan A, Siegel JH, Podany J, Prudente R, Gelb A. Fasciola hepatica pancreatitis: endoscopic extraction of live parasites. Gastrointest Endosc 1991;37:473-5. 35. Belgraier AH. Common bile duct obstruction due to Fasciola hepatica. N Y State J Med 1976;76:936-7. 36. Donnelly B, Hederman WP. Liver fluke in the common bile duct. Ir Med J 1977;70:507-9. 37. Jones EA, Kay JM, Milligan HP, Owens D. Massive infection with Fasciola hepatica in man. Gastroenterology 1977;63:83642. 38. Goodman MA, Henderson JI, Cu]lity GJ. Fascioliasis causing jaundice and intestinal bleeding. Med J Aust 1973;2:547-50. 39. Acuna-Soto R, Braun-Roth G. Bleeding ulcer in the common bile duct due to Fasciola hepatica. Am J Gastroenterol 1987; 82:560-2. 40. Maroy R, Moullot P, Daloubeix H, Mathey JC. Pancreatite aigue compliquant une distomatose biliare a Fasciola hepatica chez un patient porteur d'un diverticule choledocien [Abstract]. Ann Gastroenterol Hepatol (Paris) 1987;23:67-70. 41. Kayabali I, Gokcora IH, Yerdel MA, Ormeci N. Hepatic fascioliasis and biliary surgery. Int Surg 1992;77:154-7. 42. Khuroo MS, Zargar SA, Mahajan R. Hepatobiliary and pancreatic ascariasis in India. Lancet 1990;335:1503-6. 43. A1Karawi MA, Yasawy MI, Mohamed ARES. Endoscopic management of biliary hydatid disease: report of six cases. Endoscopy 1991;23:278-81.
Gastrointestinal lymphangioma: endosonographic demonstration and endoscopic removal
Lymphangiomas commonly develop in the head or neck where the primitive lymph sac is formed, whereas gastrointestinal lymphangiomas are extremely rare. Fleming and Carlson 1 reported that radiographic recognition was less than one case per 100,000 individuals examined. Recently, the advance and widespread use of endoscopy and endoscopic polypectomy has enabled physicians to diagnose the tumor precisely. 25 It was anticipated that endoscopic ultrasonography (EUS) would provide valuable findings in this peculiar tumor. However, because of its rarity, detailed descriptions of the tumor are as yet extremely limited. 67 , We describe five patients with lymphangioma in whom the tumors were located in the stomach in two, the duodenum in one, and the colon in two. All the lesions were evaluated with EUS before endoscopic polypectomy.
Kazuoki Hizawa, Kunihiko Aoyagi, Kouichi Kurahara, Hiroshi Suekane, Yasuyuki Kuwano, Shoutaro Nakamura, Masatoshi Fujishima,
MD MD MD MD MD MD MD
From the Second Department of Internal Medicine and the Second Department of Pathology, Faculty of Medicine, Kyushu University, Fukuoka, Japan. Reprint requests: Kazuoki Hizawa, MD, Second Department of Internal Medicine, Kyushu University, Maidashi 3-1-1, Fukuoka 812, Japan. 0016-5107/96/4306-062055.00 +0 GASTROINTESTINAL ENDOSCOPY Copyright © 1996 by the American Society for Gastrointestinal Endoscopy 37/4/66182
620 GASTROINTESTINAL ENDOSCOPY
PATIENTS AND METHODS Between November 1991 and August 1994, five patients with the histologic diagnosis of gastrointestinal lymphangiVOLUME 43, NO. 6, 1996