Gastrosplenic fistula due to diffuse large B-cell lymphoma: case report

Abstract

Background

Gastrosplenic fistula (GSF) is a rare complication of various benign and malignant etiologies. Splenic lymphoma is the most common cause of GSF. Most cases are due to diffuse large B-cell lymphoma (DLBCL).

Case presentation

A 39-year-old male patient presented at the emergency department with left upper quadrant abdominal pain. CT scan showed necrotic splenic mass with fistulous tract between the spleen and the gastric cavity. The patient underwent splenectomy and partial gastrectomy. The histopathological diagnosis was DLBCL.

Conclusions

Gastrosplenic fistula is a rare complication of diffuse large B-cell lymphoma. Radiologists should be aware of it and consider it in the diagnosis.

Background

Gastrosplenic fistula (GSF) is a rare complication of various benign and malignant etiologies [1]. The benign etiologies include splenic abscess, Crohn’s disease, and trauma, while the malignant etiologies include gastric lymphoma, gastric adenocarcinoma, and splenic lymphoma [2, 3]. Splenic lymphoma is the most common cause of GSF [4, 5]. Although primary gastric lymphoma is a rare disease constituting less than 15% of all primary gastric malignancies, it is the most common site of extranodal gastrointestinal lymphomas [4]. The majority of cases are due to diffuse large B-cell lymphoma (DLBCL) [6]. Here we present a case of DLBCL creating a fistulous tract between the spleen and the stomach which came to our tertiary center.

Case presentation

A 39-year-old male patient presented at the emergency department with left upper quadrant abdominal pain. Work-up revealed white blood cell count of 23.1 × 10³/μl and hemoglobin level of 10.8 g/dl. The CT scan showed splenic mass with air foci inside, and contrast-filled fistulous tract between the spleen and the stomach fundus (Fig. 1). The patient underwent splenectomy and partial gastrectomy. Pathological analysis revealed diffuse large B-cell lymphoma (DLBCL).

Fig. 1

Contrast-enhanced abdominal CT (A) axial image and (B) coronal image showing splenic mass with air foci inside, and a contrast-filled fistulous tract (arrow) between the spleen and the stomach fundus

Discussion

In 1962, Scoville et al. reported the first case of GSF. They described two cases of GSF due to lymphosarcoma and used the term “aerosplenomegalie” in one of the cases due to the presence of air in the enlarged spleen [7].

GSF can occur due to benign or malignant etiologies. Benign etiologies are much rarer and occur due to various diseases, e.g., peptic ulcer, splenic abscess, Crohn’s disease, trauma, and splenic tuberculosis [4, 8]. Malignancy of the spleen followed by the stomach are the commonest etiologies reported in the literature in 36 cases [5]. Lymphoma either splenic or gastric occurred in 31 out of 36 reported cases (86.11%) [5]. Splenic lymphoma occurred in 24 cases out of 36 malignant GSF (66.67%) [5]. Gastric lymphoma was reported in 6 of the 36 cases (16.67%) [5]. There is a case in which the author could not locate the site of the lymphoma, whether splenic or gastric [9]. Non-Hodgkin’s lymphoma (NHL) was the most reported histopathological subtype in malignant GSF in 63.89% (23/36), particularly diffuse large B-cell type 58.33% (21/36) [5]. A higher prevalence of GSF in male patients compared to female patients was noticed with no clear explanation for such different sex distribution [10].

GSF formation is considered to be an unfavorable prognostic event which occurs either spontaneously due to tumor progression or as a complication after administration of chemotherapy [1]. Spontaneous GSF cases are more common than post-chemotherapy GSF. Spontaneous GSF occurred in 75% of the reported cases [5].

GSF occurs spontaneously in splenic lymphoma due to extensive necrosis and invasion of the splenic capsule as well as all layers of the stomach [2, 11]. The gastric fundus and the greater curvature are the most common involved segments of the stomach. This is explained by their anatomic proximity to the spleen and the presence of the gasrtrosplenic ligament [12]. Although adenocarcinoma of the stomach is more common than primary gastric lymphoma, malignant GSF is much more common in lymphoma compared to adenocarcinoma [13]. This fact can be explained by the desmoplastic reaction that occurs in adenocarcinoma versus the rapid growth and tumor necrosis that occur in lymphoma [1]. On the other hand, it has been postulated that GSF after chemotherapy administration occurs due to rapid lysis of the tumor which exceeds the regenerative capacity of the gastric mucosal cells [14].

GSF has non-specific symptoms and is accidently discovered during imaging studies [14]. Common presenting clinical features include splenomegaly (up to 85% of cases), epigastric or left upper quadrant abdominal pain (up to 60% of cases), anorexia and weight loss (53% of cases) [3]. GSF can lead to hematemesis (8.3% of cases) and melena (5.55% of cases) [5].

Contrast-enhanced CT (CECT) is the best imaging modality for diagnosis of GSF [14]. The pathognomonic finding is the presence of contrast-filled tract between the stomach and the spleen [15]. Indirect signs include the presence of air or oral contrast in the spleen, and loss of fat planes between stomach and spleen [5, 16]. GSF on CECT may look like a splenic abscess, with a case previously reported which was initially diagnosed as an abscess in which a percutaneous catheter for drainage was inserted; then, the GSF was diagnosed by injecting contrast medium through the catheter and observing the fistulous tract on CT [17]. Endoscopy, upper gastrointestinal series, and barium studies can also demonstrate the GSF [18].

Conclusions

Gastrosplenic fistula is a rare complication of diffuse large B-cell lymphoma. Radiologists should be aware of this unusual presentation.