Spontaneous Pancreatic Hemorrhage: Successful Conservative Management in Two Cases

Background

Early diagnosis is difficult but necessary for acute abdominal diseases. Spontaneous retroperitoneal hemorrhage (SRH) is one of the acute abdominal diseases, which refers to spontaneous retroperitoneal blood vessel rupture and bleeding. It is a relatively rare disease with an incidence rate of about 0.1–6.6% and a mortality rate of about 20% [1]. The main causes of SRH include trauma, iatrogenic injury, cancer, dialysis and anticoagulant therapy. SRH usually occurs at parenchymal organs and blood vessels [2]. Previous reported cases of spontaneous parenchymal organ hemorrhage mostly occurred in the adrenal gland and kidney, while few cases of spontaneous pancreatic and peripancreatic hemorrhage (SPH) were reported. Moreover, most of the reported cases and studies with SPH were treated through digital subtraction angiography (DSA) and percutaneous embolization or surgery [3,4]. Here, we present 2 cases with SPH admitted in Union Shenzhen Hospital of Huazhong University of Science and Technology from March 2021 to March 2022, which were successfully reversed with conservative treatment.

Case Reports

CASE 1 (PATIENT 1):

A 39-year-old man was admitted with abdominal pain for 2 days without an obvious trigger such as trauma or alcoholism, who had no previous medical history or family or personal history of a connective tissue disorder. The patient underwent abdominal puncture in the external hospital before 1 day, which indicating bloody ascites combined with decreased hemoglobin. He was transferred to our hospital the next day after fasting and fluid resuscitation. Physical examination showed normal vital signs. Abdominal assessment showed only mild tenderness in the left upper quadrant. Blood test results showed a 123 000 per mL white blood cells, 9.0 mg/dL hemoglobin, and normal blood platelet and coagulation function. Amylase and lipase began to elevate to 256U/L and 1163U/L on hospital day 4. Abdominal magnetic resonance imaging (MRI) showed mixed hematoma and cystic lesions located between the pancreatic head and the descending duodenum (Figure 1A), 50×40 mm in size, which revealed hematoma from ruptured mucinous cystic neoplasms. Enhanced computed tomography (CT) showed extensive hematocele around the liver and abdominal pelvis (Figure 1B). Angiography showed no aneurysm or active bleeding.

On the second day of admission, he received peritoneal puncture therapy, and 450 ml of non-coagulant fluid was drained on the first day. The biochemical analysis of the ascites indicated that amylase, white cell count, and red blood cell count were 32 U/L, 3842 per mL, and 4 150 000 per mL, respectively, and Rivalta test was positive. During this period, the hemoglobin level decreased twice, with the lowest hemoglobin level of 8.2 mg/dl. The patient received the treatments of fasting, rehydration, intravenous injection of proton pump inhibitors to decrease gastric volume and somatostatin to reduce pancreas secretion. The patient’s condition improved and he was discharged 9 days after admission. After 1 year of follow-up, he was living and working normally.

CASE 2 (PATIENT 2):

A 44-year-old man presented with back pain for 5 h and right upper-quadrant pain for 4 h without any causes, accompanied by abdominal distension, profuse sweating, and vomiting.

There was no history of trauma, alcoholism, or anticoagulant use, nor family history or personal history of connective tissue disease. Physical examination revealed normal vital signs. An abdominal examination found tension of abdominal muscle, obvious tenderness and rebound pain in the right upper abdomen, but no obvious mass. Blood test results showed the white cell counts, blood platelets, and hemoglobin level were 178 000 per mL, 15.4g/L, and 256 000 per mL, respectively. The levels of amylase, lipase, coagulation function and tumor marker were normal at admission. CT suggested a small amount of peritoneal fluid and a hematoma located between in pancreatic head and the descending duodenum (Figure 2A). After admission, the patient received conservative treatment, including fasting, rehydration, transfusion, intravenous injection of proton pump inhibitors, and somatostatin. He developed abdominal pain again after 8 days, accompanied by abdominal distention, nausea, vomiting, and increasing abdominal pain after eating. Abdominal CT showed an enlarged hematoma (50×30 mm) between the pancreatic head and the descending duodenum, and accumulated gastric contents (Figure 2B).

Angiography showed no aneurysm or active bleeding. The patient could not receive enteral nutrition support through gastric tube due to gastric outlet obstruction. Therefore, he had to receive parenteral nutrition. The daily gastric residual volume was 500–2000 mL, and gradually decreased to 100 mL on the 27^th day of hospitalization. On days 31, he began to try an oral fluid diet, with alleviated abdominal pain and stable hemodynamics. He recovered and discharged on hospital day 37.

Discussion

The causes of SPH include trauma, iatrogenic injury, cancer, dialysis, and anticoagulant therapy, which can be divided into neoplastic and non-neoplastic factors. The main non-neoplastic factors are pancreatic pseudocysts and peripancreatic pseudoaneurysms. The causes of aneurysms in peripancreatic arteries are atherosclerosis, pancreatitis, celiac compression, congenital, mycotic, trauma, rheumatoid vasculitis, iatrogenic, and fibromuscular hyperplasia [5]. Pancreatic cystic lesions are often found in spontaneous rupture and hemorrhage of pancreatic tumors. The most common lesions are serous cyst-adenomas (SCAs), intraductal papillary mucinous neoplasms (IPMNs), and mucinous cystic neoplasms (MCNs). Cha et al reported a case of SPH with SCAs, IPMNs, and pNETs [4]. The patients in the present report were all males and had no specific history of trauma or anticoagulant therapy. In Patient 1, MCN was first found by abdominal MRI. About 80–90% of MCN lesions occur in the tail of the pancreas, manifested as single or multiple thick-walled cysts with thick cystic fluid, high CEA, and low amylase [6]. However, no obvious cause was found in Patient 2 and the hematoma might have been idiopathic.

Based on anatomical location, the foramen of Winslow allows communication between the lesser sac and the subhepatic space [7]. Usually, the retroperitoneum is not connected to the abdominal cavity. When the content of the retroperitoneal space increases, it can enter the abdominal cavity through the foramen of Winslow, which is why the free abdominal fluid accumulated after SPH in these 2 Patients. Asymptomatic patients with small lesions were regularly observed and received conservative treatment, but when the lesion rapidly increases in the short term, patients with obvious symptoms and complications (eg, portal hypertension, biliary obstruction, gastrointestinal obstruction, hemorrhagic shock, septic shock) need invasive intervention [7].

SRH has various clinical manifestations and is easily misdiagnosed, with subsequent delayed treatment [8,9]. It usually consists of a sudden onset of severe upper abdominal pain, which often radiates to the lateral lower back and causes diaphragm irritation, and is sometimes accompanied with gastrointestinal symptoms and descending hemodynamics. If the hematoma compresses the femoral nerve, it will cause pain, paralysis, and dyskinesia in the lower extremities and hip joints. The mortality rate of SRH was reported to be 5.6% within 7 days and 19.1% within 6 months [10]. Therefore, rapid and accurate diagnosis is critical for the prognosis of SRH. Emergency abdominal ultrasound is usually the first examination method, with low sensitivity in retroperitoneal diseases [11]. In the 2 patients in this report, free peritoneal effusion could be detected by ultrasound, but due to the interference of gas in the gastrointestinal tract, it is difficult to detect pancreatic lesions. CT scanning is considered to be the most reliable imaging method to diagnose SRH, and it plays a crucial role in detection of active bleeding and pathological conditions [2]. Moreover, in patients with unstable hemodynamics, DSA can be used to identify the bleeding site, which can then be treated via arterial embolization or stent placement [12]. In the patients in this report, we promptly performed CT and MRI examinations to determine the characteristics and location of the hematoma, and angiography was used to determine if there was active bleeding.

The treatment of SRH consists of conservative treatment, interventional radiology, and surgery, depending on the etiology and hemodynamic stability of the hemorrhage. Interventional radiology is a safe and effective treatment, involving selective intra-arterial embolization and deployment of stent-grafts over the punctured vessel [3]. Only patients who develop severe complications such as abdominal hypertension syndrome and obstruction after conservative or interventional treatment require surgical treatment [4,13]. A 74-year-old man underwent pancreaticoduodenectomy due to the risk of malignancy and the possibility of re-bleeding [4]. However, in SRH patients in stable condition, conservative treatment is more likely to achieve clinical improvement through correcting coagulation function, fluid resuscitation, and blood transfusion [14]. Similarly, our 2 patients were treated successfully through conservative treatment, although the etiologies were different. These cases are compared and displayed in the Table 2.

Conclusions

SPH is special type of SRH that can present with a variety of clinical manifestations, even with minimal bleeding. It is important to quickly diagnose and differentiate this disease from other pancreatic diseases. Moreover, under stable hemodynamics, conservative treatment can enable patients to achieve improvement.