Primary splenic lymphoma complicated by hematemesis and gastric erosion.

ABSTRACT

Gastrosplenic fistula resulting from erosion of a primary splenic lymphoma is a rare cause of massive upper gastrointestinal hemorrhage associated with benign peptic ulcer disease, gastric Crohn's disease, gastric adenocarcinoma, and primary gastric and splenic lymphomas. Upper intestinal hemorrhage can be successfully treated with splenic artery embolization, followed by splenectory and gastric resection.

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GASTROSPLENIC FISTULA is a rare cause of upper gastrointestinal hemorrhage. Herein, we describe the only known case of a primary splenic lymphoma presenting initially as massive upper intestinal hematemesis due to erosion into the stomach. The literature on this rare complication will be reviewed.

CASE REPORT

A 36-year-old morbidly obese, diabetic white man presented with a 2-week history of melena, fatigue, and a 20-pound weight loss. Esophagogastroduodenoscopy showed a 4 x 5 cm ulcerative, necrotic mass located superiorly along the greater curvature of the stomach. Computed tomography of the abdomen revealed massive splenomegaly, a large air-containing mass in the left upper quadrant, and marked periaortic lymphadenopathy (Fig 1). Massive hematemesis with hemodynamic instability necessitated a 6-unit red blood cell transfusion. After splenic artery embolization, the patient had a near-total gastrectomy, splenectomy, and esophagogastrostomy for a splenic mass with erosion into the stomach (Fig 2). Pathologic examination showed a diffuse, large cell splenic lymphoma (B-cell phenotype) with direct extension and erosion into the greater curvature of the stomach. The patient subsequently received chemotherapy and is disease free after 3 years.

DISCUSSION

Gastrointestinal hemorrhage from a gastrosplenic fistula is a rare occurrence. In 35 years, fewer than 15 cases have been described. Primary splenic lymphomas, (1-3) gastric lymphomas, (4,5) gastric adenocarcinoma, (6) gastric Crohn's disease, (7) and benign gastric ulcers (8,9) are known causes of gastrosplenic fistula formation. Gastric and splenic lymphomas are also known to erode into the colon (10) and the bronchus. (11)

Our patient is unique, however, because his is the only known case of massive hematemesis and a gastrosplenic fistula from a splenic lymphoma. Esophagogastroduodenoscopy was non-diagnostic of gastrosplenic fistula, but computed tomography did show an air-containing mass in the spleen, as described in previous reports. (1-4) Furthermore, this patient is intriguing because a gastric erosion formed before the institution of chemotherapy. Gastrosplenic fistula formation can occur after chemotherapy for either gastric or splenic lymphoma, (3-5) and spontaneous resolution of the fistula has been described after chemotherapy. (4)

CONCLUSION

This patient represents the only known case of primary splenic lymphoma presenting as significant upper gastrointestinal hemorrhage before institution of chemotherapy. The massive hemorrhage may be explained by the large size of the tumor mass and consequent tumor necrosis. Massive hematemesis due to malignant gastrosplenic fistula can be managed safely with splenic artery embolization followed by resection.

References

(1.) Blanchi A, Bour B, Alami O: Spontaneous gastrosplenic fistula revealing high-grade centroblastic lymphoma: endoscopic findings. Gastrointest Endosc 1995; 42:587-589

(2.) Scoville AD, Bovy P, Demeester P: [Radiologic "aerosplenomegaly" caused by necrotizing splenic lymphosarcoma with double fistulization into the digestive tract]. Acta Gastroenterol Belg 1967; 30:840-846

(3.) Bubenik O, Lopez MJ, Greco AO, et al: Gastrosplenic fistula following successful chemotherapy for disseminated histiocytic lymphoma. Cancer 1983; 52:994-996

(4.) Carolin KA, Prakash SH, Silva YJ: Gastrosplenic fistulas: a case report and review of the literature. Am Surg 1997; 63:1007-1010

(5.) Hiltunen KM, Airo I, Mattila J, et al: Massively bleeding gastrosplenic fistula following cytostatic chemotherapy of a malignant lymphoma (Letter). J Clin Gastroenterol 1991; 13:478-481

(6.) Krause R, Larsen CR, Scholz FJ: Gastrosplenic fistula: complication of adenocarcinoma of stomach. Comput Med Imaging Graph 1990; 14:273-276

(7.) Cary ER, Tremaine WJ, Banks PM, et al: Isolated Crohn's disease of the stomach. Mayo Clin Proc 1989; 64:776-779

(8.) Joffe N, Antonioli DA: Penetration into spleen by benign gastric ulcers. Clin Radiol 1981; 32:177-181

(9.) Glick SN, Levine MS, Teplick SK, et al: Splenic penetration by benign gastric ulcer: preoperative recognition with CT. Radiology 1987; 163:637-639

(10.) Karpeh MS Jr, Hicks DG, Torosian MH: Colon invasion by primary splenic lymphoma: a case report and review of the literature. Surgery 1992; 111:224-227

(11.) Cameron EW, Colby JM, Swanson RS: Gastrobronchial fistula in untreated lymphoma. J Thorac Imaging 1996; 11:150-152

RELATED ARTICLE: KEY POINTS

* Gastrosplenic fistula is a rare and life-threatening cause of massive hematemesis.

* Gastrosplenic fistula has been associated with benign peptic ulcer disease, gastric Crohn's disease and adenocarcinoma, and primary gastric and splenic lymphomas.

* Upper intestinal hemorrhage arising from a gastrosplenic fistula can be successfully managed with preoperative splenic artery embolization, followed by curative resection.

From the Departments of Surgery and Pathology, University of North Carolina at Chapel Hill.

Reprint requests to Kevin E. Behrns, MD, University of North Carolina at Chapel Hill, Department of Surgery, CB No. 7210, Chapel Hill, NC 27599-7210.