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Spontaneous gastrosplenic fistula revealing high-grade centroblastic lymphoma: Endoscopic findings
trointestinal Endoscopy Clinics of North America 1993;4:73747. 4. Loizou LA, Grigg D, Atkinson M, Robertson C, Brown SG. A prospective comparison of laser therapy and intubation in endoscopic palliation for malignant dysphagia. Gastroenterology 1991;100:1303-10. 5. Buchi KH, Dixon JA. Argon laser treatment of hemorrhagic radiation proctitis. Gastrointest Endosc 1978;333:27-9. 6. Alexander TJ, Dwyer RM. Endoscopic Nd:YAG laser treatment of severe radiation injury of the lower gastrointestinal tract: long term follow-up. Gastrointest Endosc 1988;34:407-12.
Spontaneous gastrosplenic fistula revealing high-grade centroblastic lymphoma: endoscopic findings Alain Blanchi, MD Bruno Bour, MD Odile Alami, MD
Gastrosplenic fistula is a rare complication of malignant disease. It appears after chemotherapy or after spontaneous tumor necrosis. We report two patients whose gastrosplenic fistula was the first clinical presentation of splenic lymphoma.
7. Gostout CJ, Ahlquist DA, Radford CM, Viggiano TR, Bowyer BA, Balm RK. Endoscopic laser therapy for watermelon stomach. Gastroenterology 1989;96:1462-5. 8. Kashima HK, Kessis T, Mounts P, Shah K. Polymerase chain reaction identification of human papillomavirus DNA in C02 laser plume from recurrent respiratory papillomatosis. Otolaryngol Head Neck Surg 1991;104:191-5. 9. Hallmo P, Naess O. Laryngeal papillomatosis with human papillomavirus DNA contracted by a laser surgeon. Eur Arch Otorhinolaryngol 1991;248:425-7.
CASE REPORTS Case 1 A 62-year-old a s t h m a t i c m a n presented with acute left hypochondrial p a i n a n d fever. He denied a s t h e n i a or recent weight loss. Physical examination disclosed t e n d e r n e s s in the left hypochondrium. Abdominal u l t r a s o n o g r a p h y revealed an enlarged spleen with a central hypoechoic lesion. The CT scan of the abdomen d e m o n s t r a t e d contrast material in the splenic p a r e n c h y m a (Fig. 1). A gastrosplenic fis-
From the departments o f gastroenterology and radiology, CHG, Le Mans, France. Reprint requests: Alain Blanchi, MD, 194 Av. Rubillard, 72037 Le Mans, France. 0016-5107/95/4206-058755.00 + 0 GASTROINTESTINAL ENDOSCOPY Copyright © 1995 by the American Society for Gastrointestinal Endoscopy 37/4/62848
Figure 1. CT scan showing contrast material in the splenic parenchyma in case 1. V O L U M E 42, NO. 6, 1995
Figure 2. Endoscopic view of the gastric fistula case 1 (A), and case 2 (B). GASTROINTESTINAL ENDOSCOPY
587
Figure 3. UGI barium study showing a large ulcer cavity at the top of the fundus in case 2.
Figure 4. CT scan showing a large gastric cavity into the spleen in case 2.
Table 1. Endoscopic characteristics in lymphoma gastrosplenic fistula Case
Antecedents
Indication
Sex
Age (years)
Prior therapy
Our case 1
Asthma
Left abdominal pain, fever
Male
62
No
Our case 2
No
Epigastric pain
Male
45
No
Bubenik et al?
Histiocytic lymphoma
Left abdominal discomfort
Male
58
Radiotherapy, chemotherapy
Massive bleeding
Male
36
Chemotherapy
Hiltunen et al. 4 High-grade gastric lymphoma
Endoscopy
Histology
Direct communication 2 cm fundussplenic pulp Ulcerative cavity, upper greater curvature Direct communication 3 cm fundus-splenic pulp, necrotic material Gastric folds converging into greater curvature, bright red oozing
B cell centroblastic lymphoma* B cell centroblastic lymphomat No residual tumor*
No residual tumor*
*Resected specimen. tEndoscopic biopsies.
t u l a was suspected. Endoscopy revealed a direct communication between the fundus of the stomach a n d the ulcerated splenic cavity (Fig. 2 A). A t laparotomy, no ascites was found. The liver was n o r m a l and the spleen was densely adh e r e n t to the d i a p h r a g m a n d the g r e a t e r curvature of t h e stomach. A resection of spleen, tail of pancreas, a n d involved stomach was performed. Histologic examination of the spleen revealed a B cell high-grade centroblastic lymphoma. No infiltration by the t u m o r into the gastric mucosa was seen. The A n n Arbor stage was considered as IE. Chemot h e r a p y was s t a r t e d after surgery. Six months after the operation, the p a t i e n t was in complete remission.
588 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
Case 2 A 45-year-old m a n presented with epigastric p a i n and weight loss of 12 lbs within 1 month. Physical examination was normal. Liver and pancreatic laboratory tests were normal. U p p e r gastrointestinal endoscopy revealed a n ulcerative cavity at the top of the g r e a t e r curvature of the fundus (Fig. 2 B). No t u m o r infiltration into the gastric mucosa was seen. B a r i u m m e a l confirmed t h e endoscopic appearance (Fig. 3). Histologic e x a m i n a t i o n of the endoscopic biopsies from the ulcer showed a B cell high-grade centroblastic lymphoma. Abdominal CT scan showed e n l a r g e m e n t of the spleen with heterogeneous p a r e n c h y m a closely related to
VOLUME 42, NO. 6, 1995
the gastric ulcer (Fig. 4). The Ann Arbor classification stage was IIB. Chemotherapy was instituted.
DISCUSSION Spontaneous perforation of splenic lymphoma into the stomach is a rare event.l, 2 Clinical presentation of B cell high-grade splenic lymphoma usually consists of acute onset of fever with left upper quadrant pain due to extensive necrosis. In a previous report of 10 cases, ~ these presenting symptoms were noted in 6 cases, and at laparotomy an invasion of the stomach was found in 4 cases with only one perforation. The presence of orally administered contrast material within the splenic cavity on CT scan is suggestive of communication between the stomach and the spleen. 3 Gastroscopic findings in lymphomatous gastrosplenic fistula has been reported only twice before. 3, 4 Table 1 summarizes the endoscopic appearances. They were sire-
Successful use of an internal biliary stent in Caroli's disease David M. Gold, MD Bernard Stark, MD Michael J. Pettei, MD, PhD Jeremiah J. Levine, MD C o n g e n i t a l dilatation of t h e i n t r a h e p a t i c bile ducts is a r a r e disorder first described b y Caroli et al. in 1958.1 I n t h e m a j o r i t y of cases, it is a s s o c i a t e d w i t h o t h e r a b n o r m a l i t i e s such as congenital h e p a t i c fibrosis, 2 r e n a l t u b u l a r ectasia, 3 or e x t r a h e p a t i c choledochal cyst. 4 I n a b o u t 15% of cases, t h e a b n o r m a l i t y is limited to the i n t r a h e p a t i c bile ducts. 5 T h e l o n g - t e r m m a n a g e m e n t of t h e s e p a t i e n t s is controversial, b u t m o s t f r e q u e n t l y involves t r e a t m e n t of r e c u r r e n t episodes of cholangitis w i t h e v e n t u a l surgical i n t e r v e n tion, including p a r t i a l resection or h e p a t i c t r a n s p l a n tation. 6 T h e t i m e course before t h e n e e d for Surgery is h i g h l y v a r i a b l e a n d m a y be y e a r s a f t e r t h e diagnosis
From the Division of Pediatric Gastroenterology and Nutrition, Schneider Children's Hospital, and the Division of Gastroenterology, Long Island Jewish Medical Center, Long Island campus for the Albert Einstein College of Medicine, New Hyde Park, New York. Reprint requests: David Gold, MD, Pediatric Gastroenterology and Nutrition, Schneider Children's Hospital, Room 229, New Hyde Park, N Y 11042. 0016-5107/95/4206-058955.00 + 0 GASTROINTESTINAL ENDOSCOPY Copyright © 1995 by the American Society for Gastrointestinal Endoscopy 37/4/64357
VOLUME 42, NO. 6, 1995
ilar to the findings in our two patients: (1) direct communication of the fundus and a necrotic cavity inside the splenic pulp, (2) no tumor in the stomach, and (3) no stigmata of bleeding. In case 2, endoscopic biopsies provided a diagnosis of lymphoma and avoided laparotomy. REFERENCES 1. Scoville ADE, Bovy P, Demeester P. Radiologic aerosplenomegaly caused by necrotizing splenic lymphosarcoma with double fistulization into the digestive tract. Acta Gastroenterol Belg 1967;30:840-6. 2. Harris NL, Aisenberg AC, Meyer JE, Ellman L, Elman A. Diffuse large cell (histiocytic) lymphoma ofthe spleen. Clinical and pathologic characteristics often cases. Cancer 1984;54:2460-7. 3. Bubenik 0, Lopez MJ, Greco AO, Kraybill WG, Cherwitz DL. Gastrosplenic fistula following successful chemotherapy for disseminated histiocytic lymphoma. Cancer 1983;52:994-6. 4. Hiltunen KM, Mattila J, Helve O. Massively bleeding gastrosplenic fistula following cytostatic chemotherapy of malignant lymphoma. J Clin Gastroenterol 1991;13:478-81.
is made. We describe a child with Caroli's disease involving only the intrahepatic bile ducts who is being managed medically and whose treatment includes the placement of internal biliary stents by endoscopic retrograde cholangiopancreatography (ERCP). CASE REPORT A 10-year-old boy was admitted to Schneider Children's Hospital for evaluation of fever of unknown origin. He presented with a several week history of intermittent fever to 103°F, nausea and vomiting, transient jaundice, and intermittent periumbilical abdominal pain. On physical examination, his liver was palpable 7 cm below the right costal margin. The spleen tip was also palpable. An ultrasound examination revealed a nonhomogeneous liver with multiple echogenic areas throughout both lobes, as well as a prominent spleen. An abdominal CT scan showed multiple intrahepatic areas with attenuation most consistent with cysts or malignancy (Fig. 1); a laparotomy was performed. Surface inspection of the liver revealed multiple abscesses. The extrahepatic biliary tree appeared normal. The abscesses were aspirated and cultures grew F u s o b a c t e r i u m n u c l e a t u m and EikeneUa corrodens. Histologic examination of the liver revealed acute inflammation with abscess formation but no evidence of fibrosis. The patient was treated with intravenous antibiotics (initially metronidazole, ceftriaxone, and gentamicin for 7 days, then metronidazole, cefotaxime, and penicillin G) without improvement, until a percutaneous drain was placed into one of the large abscess cavities in the right liver lobe. Fever resolved within 24 hours of placement of this drain. A diagnosis of Caroli's disease was confirmed by a percutaneous trans-hepatic cholangiogram through the external drain, which demonstrated dilated intrahepatic bile ducts in
GASTROINTESTINAL ENDOSCOPY
589
Spontaneous gastrosplenic fistula revealing high-grade centroblastic lymphoma: endoscopic findings Alain Blanchi, MD Bruno Bour, MD Odile Alami, MD
Gastrosplenic fistula is a rare complication of malignant disease. It appears after chemotherapy or after spontaneous tumor necrosis. We report two patients whose gastrosplenic fistula was the first clinical presentation of splenic lymphoma.
7. Gostout CJ, Ahlquist DA, Radford CM, Viggiano TR, Bowyer BA, Balm RK. Endoscopic laser therapy for watermelon stomach. Gastroenterology 1989;96:1462-5. 8. Kashima HK, Kessis T, Mounts P, Shah K. Polymerase chain reaction identification of human papillomavirus DNA in C02 laser plume from recurrent respiratory papillomatosis. Otolaryngol Head Neck Surg 1991;104:191-5. 9. Hallmo P, Naess O. Laryngeal papillomatosis with human papillomavirus DNA contracted by a laser surgeon. Eur Arch Otorhinolaryngol 1991;248:425-7.
CASE REPORTS Case 1 A 62-year-old a s t h m a t i c m a n presented with acute left hypochondrial p a i n a n d fever. He denied a s t h e n i a or recent weight loss. Physical examination disclosed t e n d e r n e s s in the left hypochondrium. Abdominal u l t r a s o n o g r a p h y revealed an enlarged spleen with a central hypoechoic lesion. The CT scan of the abdomen d e m o n s t r a t e d contrast material in the splenic p a r e n c h y m a (Fig. 1). A gastrosplenic fis-
From the departments o f gastroenterology and radiology, CHG, Le Mans, France. Reprint requests: Alain Blanchi, MD, 194 Av. Rubillard, 72037 Le Mans, France. 0016-5107/95/4206-058755.00 + 0 GASTROINTESTINAL ENDOSCOPY Copyright © 1995 by the American Society for Gastrointestinal Endoscopy 37/4/62848
Figure 1. CT scan showing contrast material in the splenic parenchyma in case 1. V O L U M E 42, NO. 6, 1995
Figure 2. Endoscopic view of the gastric fistula case 1 (A), and case 2 (B). GASTROINTESTINAL ENDOSCOPY
587
Figure 3. UGI barium study showing a large ulcer cavity at the top of the fundus in case 2.
Figure 4. CT scan showing a large gastric cavity into the spleen in case 2.
Table 1. Endoscopic characteristics in lymphoma gastrosplenic fistula Case
Antecedents
Indication
Sex
Age (years)
Prior therapy
Our case 1
Asthma
Left abdominal pain, fever
Male
62
No
Our case 2
No
Epigastric pain
Male
45
No
Bubenik et al?
Histiocytic lymphoma
Left abdominal discomfort
Male
58
Radiotherapy, chemotherapy
Massive bleeding
Male
36
Chemotherapy
Hiltunen et al. 4 High-grade gastric lymphoma
Endoscopy
Histology
Direct communication 2 cm fundussplenic pulp Ulcerative cavity, upper greater curvature Direct communication 3 cm fundus-splenic pulp, necrotic material Gastric folds converging into greater curvature, bright red oozing
B cell centroblastic lymphoma* B cell centroblastic lymphomat No residual tumor*
No residual tumor*
*Resected specimen. tEndoscopic biopsies.
t u l a was suspected. Endoscopy revealed a direct communication between the fundus of the stomach a n d the ulcerated splenic cavity (Fig. 2 A). A t laparotomy, no ascites was found. The liver was n o r m a l and the spleen was densely adh e r e n t to the d i a p h r a g m a n d the g r e a t e r curvature of t h e stomach. A resection of spleen, tail of pancreas, a n d involved stomach was performed. Histologic examination of the spleen revealed a B cell high-grade centroblastic lymphoma. No infiltration by the t u m o r into the gastric mucosa was seen. The A n n Arbor stage was considered as IE. Chemot h e r a p y was s t a r t e d after surgery. Six months after the operation, the p a t i e n t was in complete remission.
588 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
Case 2 A 45-year-old m a n presented with epigastric p a i n and weight loss of 12 lbs within 1 month. Physical examination was normal. Liver and pancreatic laboratory tests were normal. U p p e r gastrointestinal endoscopy revealed a n ulcerative cavity at the top of the g r e a t e r curvature of the fundus (Fig. 2 B). No t u m o r infiltration into the gastric mucosa was seen. B a r i u m m e a l confirmed t h e endoscopic appearance (Fig. 3). Histologic e x a m i n a t i o n of the endoscopic biopsies from the ulcer showed a B cell high-grade centroblastic lymphoma. Abdominal CT scan showed e n l a r g e m e n t of the spleen with heterogeneous p a r e n c h y m a closely related to
VOLUME 42, NO. 6, 1995
the gastric ulcer (Fig. 4). The Ann Arbor classification stage was IIB. Chemotherapy was instituted.
DISCUSSION Spontaneous perforation of splenic lymphoma into the stomach is a rare event.l, 2 Clinical presentation of B cell high-grade splenic lymphoma usually consists of acute onset of fever with left upper quadrant pain due to extensive necrosis. In a previous report of 10 cases, ~ these presenting symptoms were noted in 6 cases, and at laparotomy an invasion of the stomach was found in 4 cases with only one perforation. The presence of orally administered contrast material within the splenic cavity on CT scan is suggestive of communication between the stomach and the spleen. 3 Gastroscopic findings in lymphomatous gastrosplenic fistula has been reported only twice before. 3, 4 Table 1 summarizes the endoscopic appearances. They were sire-
Successful use of an internal biliary stent in Caroli's disease David M. Gold, MD Bernard Stark, MD Michael J. Pettei, MD, PhD Jeremiah J. Levine, MD C o n g e n i t a l dilatation of t h e i n t r a h e p a t i c bile ducts is a r a r e disorder first described b y Caroli et al. in 1958.1 I n t h e m a j o r i t y of cases, it is a s s o c i a t e d w i t h o t h e r a b n o r m a l i t i e s such as congenital h e p a t i c fibrosis, 2 r e n a l t u b u l a r ectasia, 3 or e x t r a h e p a t i c choledochal cyst. 4 I n a b o u t 15% of cases, t h e a b n o r m a l i t y is limited to the i n t r a h e p a t i c bile ducts. 5 T h e l o n g - t e r m m a n a g e m e n t of t h e s e p a t i e n t s is controversial, b u t m o s t f r e q u e n t l y involves t r e a t m e n t of r e c u r r e n t episodes of cholangitis w i t h e v e n t u a l surgical i n t e r v e n tion, including p a r t i a l resection or h e p a t i c t r a n s p l a n tation. 6 T h e t i m e course before t h e n e e d for Surgery is h i g h l y v a r i a b l e a n d m a y be y e a r s a f t e r t h e diagnosis
From the Division of Pediatric Gastroenterology and Nutrition, Schneider Children's Hospital, and the Division of Gastroenterology, Long Island Jewish Medical Center, Long Island campus for the Albert Einstein College of Medicine, New Hyde Park, New York. Reprint requests: David Gold, MD, Pediatric Gastroenterology and Nutrition, Schneider Children's Hospital, Room 229, New Hyde Park, N Y 11042. 0016-5107/95/4206-058955.00 + 0 GASTROINTESTINAL ENDOSCOPY Copyright © 1995 by the American Society for Gastrointestinal Endoscopy 37/4/64357
VOLUME 42, NO. 6, 1995
ilar to the findings in our two patients: (1) direct communication of the fundus and a necrotic cavity inside the splenic pulp, (2) no tumor in the stomach, and (3) no stigmata of bleeding. In case 2, endoscopic biopsies provided a diagnosis of lymphoma and avoided laparotomy. REFERENCES 1. Scoville ADE, Bovy P, Demeester P. Radiologic aerosplenomegaly caused by necrotizing splenic lymphosarcoma with double fistulization into the digestive tract. Acta Gastroenterol Belg 1967;30:840-6. 2. Harris NL, Aisenberg AC, Meyer JE, Ellman L, Elman A. Diffuse large cell (histiocytic) lymphoma ofthe spleen. Clinical and pathologic characteristics often cases. Cancer 1984;54:2460-7. 3. Bubenik 0, Lopez MJ, Greco AO, Kraybill WG, Cherwitz DL. Gastrosplenic fistula following successful chemotherapy for disseminated histiocytic lymphoma. Cancer 1983;52:994-6. 4. Hiltunen KM, Mattila J, Helve O. Massively bleeding gastrosplenic fistula following cytostatic chemotherapy of malignant lymphoma. J Clin Gastroenterol 1991;13:478-81.
is made. We describe a child with Caroli's disease involving only the intrahepatic bile ducts who is being managed medically and whose treatment includes the placement of internal biliary stents by endoscopic retrograde cholangiopancreatography (ERCP). CASE REPORT A 10-year-old boy was admitted to Schneider Children's Hospital for evaluation of fever of unknown origin. He presented with a several week history of intermittent fever to 103°F, nausea and vomiting, transient jaundice, and intermittent periumbilical abdominal pain. On physical examination, his liver was palpable 7 cm below the right costal margin. The spleen tip was also palpable. An ultrasound examination revealed a nonhomogeneous liver with multiple echogenic areas throughout both lobes, as well as a prominent spleen. An abdominal CT scan showed multiple intrahepatic areas with attenuation most consistent with cysts or malignancy (Fig. 1); a laparotomy was performed. Surface inspection of the liver revealed multiple abscesses. The extrahepatic biliary tree appeared normal. The abscesses were aspirated and cultures grew F u s o b a c t e r i u m n u c l e a t u m and EikeneUa corrodens. Histologic examination of the liver revealed acute inflammation with abscess formation but no evidence of fibrosis. The patient was treated with intravenous antibiotics (initially metronidazole, ceftriaxone, and gentamicin for 7 days, then metronidazole, cefotaxime, and penicillin G) without improvement, until a percutaneous drain was placed into one of the large abscess cavities in the right liver lobe. Fever resolved within 24 hours of placement of this drain. A diagnosis of Caroli's disease was confirmed by a percutaneous trans-hepatic cholangiogram through the external drain, which demonstrated dilated intrahepatic bile ducts in
GASTROINTESTINAL ENDOSCOPY
589