To the best of our knowledge, this is the first case of isolated SAD which was successfully treated by endovascular therapy. Although the etiology of SAD remains unclear, hypertension is considered to be a common risk factor [2]. This is consistent with the history of hypertension in the current case. Ruptured SAD usually presents with upper abdominal or left flank pain and causes intraperitoneal or retroperitoneal hemorrhage [2]. However, in the current case, it presented with intermittent hematochezia and resulted in digestive hemorrhage. To our knowledge, SAD resulting in digestive hemorrhage has not been reported in literature. Herein, we present the first case of SAD presenting as recurrent digestive hemorrhage. Ruptured SAD should be considered in the differential diagnosis as a rare cause of digestive hemorrhage.
Splenic artery diseases rarely result in digestive hemorrhage. Most of them are false splenic artery aneurysms (SAAs) caused by pancreatitis or pancreatic tumors [3,4,5,6,7]. SAAs can rupture into the stomach or the pancreatic duct and lead to gastric or duodenal hemorrhage [3,4,5,6,7]. The latter hemorrhage is also termed “hemosuccus pancreaticus” [4, 6, 7]. The diagnosis of hemosuccus pancreaticus remains challenging. It can be difficult to identify the bleeding site via endoscopy or digital subtraction angiography because of its rarity, anatomical location, and intermittent characteristics [4, 7]. Endoscopy was able to detect active bleeding via the papilla in only 30% of cases [7].
In this case, endoscopies failed to identify the bleeding site, and histopathological examination was not performed because of endovascular treatment. However, the authors believe that digestive hemorrhage was caused by rupture of SAD. The potential mechanism is considered to involve hemosuccus pancreaticus: SAD ruptured into the pancreatic tail, and the blood from the hematoma entered into the duodenum via the pancreatic duct. This is supported by several observations. First, the hematoma was completely within the pancreas. The site is similar to Yoshikazu’s cases [6]. The SAAs observed in their cases were treated by open surgery, and the communication between the aneurysm and the pancreatic duct was proven by histological examination. Second, hemosuccus pancreaticus is usually intermittent and repetitive [4, 7]. These characteristics were consistent with the present case. Third, primary gastrointestinal or pancreatic lesions were excluded via endoscopies (gastroduodenoscopy, colonoscopy, and capsule endoscopy) and radiological examination (digital subtraction angiography, repeated CT, and MR). SAD rupturing into the stomach was also excluded based on the symptoms (hematochezia rather than hematemesis), endoscopies, and the range of the lesion, which did not exceed the superior edge of the pancreas on CT and MR. Finally, digestive hemorrhage was cured after the use of endovascular coil embolization for SAD. The efficacy has been demonstrated by 1-year follow-up.
Several methods have been reported in the treatment of SAAs, including open resection, laparoscopic resection, and endovascular treatment. For hemosuccus pancreaticus associated with SAAs, if the source of the bleeding is identified on angiography, endovascular treatment should be considered as the first choice of treatment [4, 6, 7]. Surgical treatment remains an important supplement if angiography fails to identify the source of the bleeding or if endovascular treatment is not successful. For SAD, only one patient was successfully managed with open surgery [2]. In the current case, it was successfully treated by endovascular coil embolization. Endovascular treatment is a safe and effective option to ruptured SAD.
In conclusion, isolated SAD is extremely rare but life-threatening. Ruptured SAD should be considered in the differential diagnosis as a rare cause of digestive hemorrhage. Selective angiography is recommended for diagnosis, and endovascular embolization is a safe and effective treatment for ruptured SAD.