A 72-year-old male with right costalgia and fever presented to the department of gastroenterology and hepatology in our hospital. His previous medical history was unremarkable. After relevant examinations, extrahepatic cholangiocarcinoma was suspected.
Contrast-enhanced computed tomography (CT) showed a 2-cm-long mass with contrast enhancement in the central bile duct. There were no findings suggestive of enlarged lymph nodes or distant metastasis (Fig. 1a). Endoscopic retrograde cholangiopancreatography (ERCP) findings revealed narrowing of the central bile duct, without the cystic duct being visualized. The cytology of the bile duct brush and bile cytology were both class III (Fig. 1b).
The patient was diagnosed with distal bile duct cancer classified as cT2N0M0, Stage IB [2]. On the 21st day after the first visit, he was referred to the department of surgery. Blood biochemical tests at this time showed mild liver dysfunction. The serum level of carbohydrate antigen 19-9 was 55 IU/mL, which was slightly elevated. The patient underwent subtotal stomach-preserving PD on the 46th day after the first visit. The duration of surgery was 7 h and 52 min with a blood loss volume of 430 mL. 7.5Fr tube was inserted as the pancreatic duct stent for external drainage across the jejunum and removed 3 weeks after the surgery. We also inserted 3 mm internal stent as the biliary stent. Reconstruction following PD was performed by the modified Child method. Pancreaticojejunostomy was performed by the pancreaticojejunal mucosal anastomosis and the modified Kakita method. Choledochojejunostomy was performed with 24 stitches of 4-0 polydioxanone suture. 10 mm soft drains were inserted into the back side of the choledochojejunostomosis and the pancreaticojejunostomosis, respectively. Histopathological findings revealed well-differentiated tubular adenocarcinoma with a flat invasive lesion with indistinct borders in the distal bile duct, which was classified as fT2N0M0, Stage IB [2]. Neuroendocrine carcinoma was detected in some parts of the lesion, which indicated mixed adenoneuroendocrine carcinoma. The hepatic margin of the bile duct was negative.
Drainage amylase level of the pancreaticojejunal drain was 336 U/L and drainage total bilirubin level at the same site was 0.4 mg/dL, respectively, on the 3rd day after surgery. Drainage amylase level of the choledochojejunal drain was 42 U/L and drainage total bilirubin level at the same site was 39 mg/dL, respectively, on the same day. The serum bilirubin level at the same day was 1.0 mg/dL. Therefore, the patient was diagnosed with bile leakage [3].
A liquid diet was given on the 4th day after surgery. Drainage amylase level of the pancreaticojejunal drain was 4 U/L and drainage total bilirubin level at the same site was 21.8 mg/dL, respectively, on the 5th day after surgery. Drainage amylase level of the choledochojejunal drain was 163 U/L and drainage total bilirubin level at the same site was 1.2 mg/dL, respectively, on the same day. The serum bilirubin level at the same day was 0.8 mg/dL.
We removed the pancreaticojejunal drain and replaced the choledochojejunal drain on the 7th day after surgery. Drainage amylase level of the choledochojejunal drain was 14 U/L and drainage total bilirubin level at the same site was 7.1 mg/dL, respectively, on the same day. The serum bilirubin level at the same time was 0.6 mg/dL. As he was asymptomatic and the results of blood test on the 7th day after surgery were within normal limits as postoperative, we started to gradually increase the amount of solid diet. He ate almost half the amount.
Drainage amylase level of the choledochojejunal drain was 662 U/L and drainage total bilirubin level was 35.7 mg/dL on the 14th day after surgery. Therefore, the patient was diagnosed with pancreatic fistula [4]. The patient recovered conservatively with drain replacement and antimicrobial therapy, and he was discharged on the 36th day after surgery.
On the 44th day after surgery, abdominal pain developed in the right upper quadrant and the patient visited our outpatient clinic. Blood tests showed a white blood cell count of 16,400/μL, a hemoglobin level of 7.1 g/dL and a C-reactive protein level of 13.24 mg/dL. Contrast-enhanced CT revealed a pseudoaneurysm at the end of the gastroduodenal artery (GDA) with widespread hematoma in the surrounding area (Fig. 2a). Angiography also showed the pseudoaneurysm at the end of the GDA (Fig. 2b). Transcatheter arterial embolization (TAE) was performed through the GDA with a stretch-resistant coil for hemostasis. Although the patient did not rebleed after TAE, a large intra-abdominal abscess formed mainly in the right upper abdomen due to an infected hematoma. The patient developed necrotizing fasciitis of the right oblique abdominal muscle and costochondritis on the 13th day after TAE (Fig. 3a). The CT on the same day showed that the infected hematoma contacted to inner abdominal wall (Fig. 3b). Treatment with incisional drainage was started immediately with two subcutaneous 10 Fr Penrose drains. MRSA and Enterobacter species were detected in the wound on the 15th day after TAE and antimicrobial agents (vancomycin and clindamycin) were administered. The serum albumin level was 1.6 g/dL and extreme nutritional disturbance was observed. On the next day, we started to treat with debridement and negative pressure wound therapy (NPWT) (Fig. 4a, b). However, it was difficult to control the infection. The patient underwent debridement of the infected area on the 78th day after TAE, and simultaneous flap plasty with musculus latissimus dorsi and skin grafting from the right buttock were performed to close the wound from the right anterior chest to the upper abdomen in collaboration with the department of plastic surgery (Fig. 4c). The culture of the wound turned negative on the 98th day after TAE. Following postoperative wound care and rehabilitation, the patient was discharged on the 192nd day after TAE and followed up in the outpatient clinic. A small amount of discharge was observed from the wound on the 215th day after surgery and antimicrobial agent (levofloxacin) was administered. The discharge disappeared 3 weeks later. Multiple liver metastases were detected by CT on the 267th day after PD. Although chemotherapy was considered, CT also showed a subcutaneous abscess in the right 11th rib (Fig. 3c). Rib cartilage resection and debridement for right costochondral osteomyelitis were performed to control the infection. NPWT was postoperatively continued and resolved wound infection. Since rehabilitation and nutritional therapy were also performed during this period, the activities of daily living improved (Performance status 0). Therefore, chemotherapy with gemcitabine with cisplatin (GC) was initiated on the 460th day after PD. The patient received six courses of GC without serious adverse events. The therapeutic effect was assessed as PR (Fig. 5).