Via Research Recognition Day 2024 VCOM-Carolinas

Clinical Case-Based Reports

Gastrosplenic Fistula in the Setting of Undiagnosed Lymphoma Mackenzie Lecher OMSIII 2 , Lindsay Tjiattas-Saleski DO 2 , Brian Lecher DO 1 . FastER Care 1 , Sumter, SC, Edward Via College of Osteopathic Medicine – Carolinas Campus 2

Abstract

Management

Discussion

A gastrosplenic fistula (GSF) is a pathologic connection between the spleen and stomach that can lead to life threatening complications. GSFs can arise spontaneously but are often secondary to a variety of etiologies. GSFs most commonly arise from a gastric or splenic non- Hodgkin’s diffuse large B -cell lymphoma (DLBCL). Only 46 cases of GSFs have been published to date and due to its rarity, extensive literature review is insufficient for characterization. This case discusses a patient with intermittent abdominal pain and weight loss which led to the diagnosis and treatment of a GSF and DLBCL. The patient later went into remission for his DLBCL but succumbed to respiratory failure from a secondary abdominal-pleural fistula formation. GSFs have the potential to cause fatal massive upper gastrointestinal (GI) hemorrhages, infections, fistulas, or obstructions. Delayed diagnosis corresponds with a higher morbidity and mortality; thus, prompt detection and treatment are imperative. The management of GSFs is complex due to their rare nature and requires a multidisciplinary approach to care. The intention of this report is to provide information and increase awareness of GSFs in the medical community to facilitate their diagnosis. A 59-year-old Caucasian male with a past medical history of hypertension, coronary artery disease, obesity, and psoriatic arthritis presented to an urgent care with intermittent left upper and lower quadrant abdominal pain, fatigue, and nausea which had gradually increased in severity over 5 weeks. Over the course of two months, the patient reported a weight loss of 25lbs but denied any melena, hematochezia, or tenesmus. Vitals: BP: 145/88, HR: 90, RR: 18, 37 ° C, 99% oxygen saturation on room air. Physical exam findings: normal bowel sounds, moderate upper and lower abdominal tenderness, no palpable masses or organomegaly. Laboratory testing revealed a WBC: 15.0 x10^3/uL, Hgb: 12.8 g/dL, amylase: 35 U/L, lipase: 22 U/L, creatinine: 0.79 mg/dL. All remaining laboratory values including CBC, BMP, and coagulation studies were normal. Radiologic Imaging: CT of the abdomen and pelvis with and without IV contrast shows a gastrosplenic fistula and left pleural effusion (Figures 1-3). Case Report

This patient had a GSF secondary to DLBCL. While GSF remains a rare occurrence, DLBCL is associated with numerous risk factors such as age, gender, race, family history, auto-immune disease, and medication usage 1,2 . Current data indicates DLBCL occurs most frequently in men with a median age of 55, Caucasians, and individuals with a BMI >30 which all put the patient in this case at increased risk 1 . GSF patients treated with surgery generally have a good prognosis but often have a propensity for developing complications from surgery due to a weakened immune system 3,4 . Potential complications of the surgery include infection, bleeding, venous thromboembolism, cancer, anastomotic leakage, stenosis, dumping syndrome, abscess, perforation, renal dysfunction, respiratory complications, and anemia 4,5 . It is imperative that post-splenectomy patients remain up to date on their immunizations, particularly against encapsulated bacteria such as Pneumococcus, Meningococcus, and Haemophilus influenzae 6 . In a systematic review conducted in 2017, the most common cause of death from GSFs was gastric perforation followed by pulmonary infection with multi-organ failure 3 . Overall, when initial presentation of the GSF is not massive GI hemorrhaging, patients have an 82% survival rate 7 . The patient in this case achieved remission for his lymphoma after surgery and chemotherapy but went on to develop complications which lead to his death.As a result of inflammation and leakage from his anastomosis, a jejunal-pleural fistula and colo-pleural fistula were formed and treatment with stenting and diverting transverse colostomy failed. After treatment in the ICU for hypoxemia, the patient was transferred to the floor of the hospital in stable condition but subsequently developed acute respiratory failure which ultimately proved to be fatal. The patient passed away approximately 3 years after his initial diagnosis of a GSF.

Figure 2. Abdominal CT shows an enlarged spleen with gas contiguous with gas in the greater curvature of the proximal stomach.

Figure 3 . Abdominal CT shows pleural effusion of the left lung base with atelectasis.

After admission to the hospital, an esophagogastroduodenoscopy (EGD) was performed showing inflammation at the gastric fundus and a perforated ulcer with abscess formation and abnormal mucosa. A large gastric tumor 3 or 4-inches inferior to the gastroesophageal junction was also visualized. A biopsy was obtained and tested positive for tumor markers Bcl6, CD20, CD10, MUM1, Ki-67 >90% and pathology consistent with DLBCL

Conclusion

Insufficient data exists regarding GSFs, therefore it is important to document cases to expedite early detection and foster the development of improved therapeutic strategies. It is essential to keep a high level of suspicion for GSFs in patients with DLBCL to prevent fatalities. Further studies should be conducted to increase awareness of GSFs and improve patient care.

Figure 4 . Visualization of a large gastric tumor by EGD

The patient underwent a splenic embolization followed by laparoscopic converted to open distal esophagectomy, total gastrectomy, omental pedicle flap, Roux-en-Y esophagojejunostomy, splenectomy, distal pancreatectomy, feeding jejunostomy tube placement, esophagogastroduodenoscopy, small bowel resection, and partial left hepatectomy. Surgeons were able to visualize perforation of the gastric tumor into the splenic capsule causing splenomegaly, and tumor invasion into the diaphragm on the left, the pancreatic tail, and the left lobe of the liver. He was later treated by oncology for stage-III DLBCL with chemotherapy.

1. Morton LM, Wang SS, Devesa SS, Hartge P, Weisenburger DD, Linet MS. Lymphoma incidence patterns by WHO subtype in the United States, 1992-2001. Blood . 2006;107(1):265-276. doi:10.1182/blood-2005-06-2508 2. Tokuhira, M., Saito, S., Suzuki, K., Higashi, M., Momose, S., Shimizu, T., Mori, T., Kimura, Y., Amano, K., Okamoto, S., Takeuchi, T., Tamaru, J.- I., & Kizaki, M. Clinicopathological features of clinical methotrexate-related lymphoproliferative disorders. Leuk. Lymphoma. 2019 ; 60 (10), 2508 – 2515. https://doi.org/10.1080/10428194.2019.1585841 3. Kang, D. H., Huh, J., Lee, J. H., Jeong, Y. K., & Cha, H. J. Gastrosplenic fistula occurring in lymphoma patients: Systematic review with a new case of extranodal NK/T-cell lymphoma. World J Gastroenterol. 2017; 23 (35), 6491 – 6499. https://doi.org/10.3748/wjg.v23.i35.6491 4. Kristinsson SY, Gridley G, Hoover RN, Check D, Landgren O. Long-term risks after splenectomy among 8,149 cancer-free American veterans: a cohort study with up to 27 years follow-up. Haematologica . 2014;99(2):392-398. doi:10.3324/haematol.2013.092460 5. Li SS, Costantino CL, Mullen JT. Morbidity and mortality of total gastrectomy: a comprehensive analysis of 90-day outcomes. J. Gastrointestl Surg. 2019;23(7):1340-1348. doi:10.1007/s11605-019-04228-7 6. Davies JM, Barnes R, Milligan D; British Committee for Standards in Haematology. Working Party of the Haematology/Oncology Task Force. Update of guidelines for the prevention and treatment of infection in patients with an absent or dysfunctional spleen. Clin Med (Lond). 2002;2(5):440-443. doi:10.7861/clinmedicine.2-5-440 7. Frenkel, A., Bichovsky, Y., Perry, Z. H., Peiser, J., Roy-Shapira, A., Brotfain, E., Koyfman, L., Binyamin, Y., Nalbandyan, K., & Klein, M. Management of gastrosplenic fistula in the emergency setting - A case report and review of the literature. Ann. Med. Surg. 2018; 29 , 26 – 29. https://doi.org/10.1016/j.amsu.2018.03.025

Figure 1. Abdominal CT shows an enlarged spleen with gas contiguous with gas in the greater curvature of the proximal stomach.

23

2024 Research Recognition Day