Clinical Case Summary

Introduction: Primary splenic angiosarcoma is a rare neoplasm of vascular origin with aggressive behavior, atypical clinical presentation and poor prognosis because of its high metastatic potential. This disease often presents with spontaneous rupture and lethal hemorrhage.^1-5
Case report: We report an 81-year-old woman who presented with a 1-month history of melena. The patient was hemodynamically stable and laboratory investigations showed a significant drop in hematocrit (Hct: 19.4%, Hb: 6,5 g/dL). The patient underwent upper GI endoscopy which revealed two hemorrhagic polypoid lesions, one in the greater curvature of the stomach and one in the second part of the duodenum (Figure A, B). Then, a polypectomy was performed for each lesion and specimens were sent for histological examination. Two weeks later, the patient experienced a recurrence of bleeding and a re-endoscopy was performed which revealed “regeneration” of the previously resected polypoid lesions. The bleeding gastric polyp had a visible vessel where an over-the-scope clip (OTSC) placement was successfully performed. After the gastrointestinal bleeding subsided in the next few days, the patient presented with acute abdominal pain and abdominal distension, with a dramatic drop in hematocrit and hemοdynamic instability. An urgent CT angiography was performed, showing (spontaneous) rupture of the spleen and hemoperitoneum (Figure C). Finally, the patient underwent splenectomy and from the histopathological evaluation of the surgical specimen and from the gastric and duodenal specimens she was diagnosed with primary splenic angiosarcoma with synchronous gastric and duodenal metastases.
Conclusions: Splenic angiosarcoma is a very rare malignancy and the majority of patients have metastases at the time of diagnosis. We recommend a high degree of vigilance in patients with multiple sites of gastrointestinal bleeding and early investigation of possible underlying disease.

References

1. Duan YF, Jiang Y, Wu CX, Zhu F. Spontaneous rupture of primary splenic angiosarcoma: a case report and literature review. World J Surg Oncol. 2013 Mar 4;11:53.
2. Bergas A, Escrihuela-Vidal F, Fernández Calvo D, Capdevila Pons O, Corbella X. Relapsing Haemothorax as an Unusual Presentation of Primary Angiosarcoma of the Spleen. Eur J Case Rep Intern Med. 2020 Oct 29;7(12):001944.
3. Maeyashiki C, Nagata N, Uemura N. Angiosarcoma involving solid organs and the gastrointestinal tract with life-threatening bleeding. Case Rep Gastroenterol. 2012 Sep;6(3):772-7.
4. Juin Hsien BL, Shelat VG. Spleen angiosarcoma: a world review. Expert Rev Gastroenterol Hepatol. 2021 Oct;15(10):1115-1141.
5. Fiorentino MD, Monteiro JMC, de Siqueira REB, Kim EIM, Curi AP, Ferrreira CR, Nardo M, de Campos FPF. Primary splenic angiosarcoma: a rare entity often associated with rupture and hemoperitoneum. Autops Case Rep. 2019 Jul 12;9(3):e2019100.

Clinical Case Summary

Introduction: Hepatic artery pseudoaneurysms are a low incidence (0.3–2.5%) but life-threatening complication. Fistulization of such pseudoaneusrysms into the gallbladder and duodenum is extremely rare and is an unusual cause of hemobilia.^1-7
Case report: A 54-year-old man, without history of traumatism, hepatobiliary surgery or intervention on the hepatobiliary tract, presented with Quincke’s triad of symptoms, upper gastro-intestinal hemorrhage (melena), jaundice and upper abdominal pain. The patient was haemodynamically unstable (BP: 86/49 mmHg, HR: 113/min), while the laboratory tests showed significant reduction in hematocrit (Hct: 25,4%, Hb: 8,3 g/dL), elevated markers of inflammation (WBCs: 20.500/μL, CRP: 25 mg/dL) and abnormal liver function tests (TBL: 3,9 mg/dL, DBL: 3 mg/dL, SGOT: 220 U/l, SGPT: 413 U/l, γGT: 497 U/l, ALP: 211 U/l). The patient was resuscitated and after stabilization he underwent upper GI endoscopy which showed an attached thrombus in the second part of the duodenum, with a pulsating duodenal wall (Figure A). Thrombus detachment revealed an external orifice of a fistula. A vascular lesion was then suspected and an urgent CT scan was performed. The CT scan confirmed the presence of a common hepatic artery pseudoaneurysm, 2.5cm in diameter, localized in the region of the gallbladder fossa, complicated with cholecystoduodenal fistula due to eroding cholecystitis (Figure B). Arterial catheterization and angiographic evaluation of the common hepatic artery pseudoaneurysm was performed, followed by a fully covered stent placement (Figure C). The postoperative course was uneventful and the patient was discharged on the 10^th postoperative day.
Conclusions: Pseudoaneurysm of the common hepatic artery is rare but potentially fatal. In the literature, there is no other case o hemobilia originating from a common hepatic artery pseudoaneurysm with cholecystoduodenal fistula due to eroding cholecystitis.

References

1. Landolsi S, Landolsi M, Mannai S. Spontaneous right hepatic artery branch gallbladder fistula revealed by haemobilia and upper cataclysmic gastrointestinal bleeding. BMJ Case Rep. 2018 Jan 3;2018:bcr2017222134.
2. Totolici BD, NeamŢu C, Bodog FD, Bungău SG, Goldiş DS, Matei M, Andercou OA, Amza OL, Gyori Z, Coldea L. Hemobilia by idiopathic aneurysm of cystic artery, fistulized in the biliary ways - clinical case. Rom J Morphol Embryol. 2017;58(1):267-270.
3. Das M, Volmar FH, Walayat S, Nolte R. Hemobilia from a right hepatic artery pseudoaneurysm due to chronic cholecystitis. SAGE Open Med Case Rep. 2019 Aug 23;7:2050313X19872075.
4. Sawalha K, Kunnumpurath A, McCann R. An Unusual Cause of an Intraperitoneal Bleed: Bleeding Hepatic Artery Pseudoaneurysm Due to an Eroding Cholecystitis. J Investig Med High Impact Case Rep. 2020 Jan-Dec;8:2324709620982431.
5. Shelton J, Raviraj S. A Case Report: Hepatic artery pseudoaneurysm causing life-threatening haemobilia. Int J Surg Case Rep. 2021 Sep;86:106350.
6. Traa AC, Hoven-Gondrie ML, Diederik AL. A pseudoaneurysm of the right hepatic artery treated successfully with a stent graft - A case report. Int J Surg Case Rep. 2020;77:407-411.

Clinical Case Summary

Introduction: Swelling of the parotid glands has previously been reported as a rare complication of peroral endoscopy. Several mechanisms have been proposed such as retention of secretions causing a blockage of the salivary ducts, venous congestion of the salivary glands, dehydration causing thick secretions, parasympathetic stimulation resulting in parotid vasodilation.
Case report: We report the case of 81‑year‑old male patient who presented with a 3-day history of melena and dizziness. The patient was hemodynamically stable (BP: 140/85 mmHg, HR: 89/min) with positive digital rectal exam for blood. The laboratory tests showed significant reduction in hematocrit (Hct: 21.2%, Hb: 7.3 g/dL). The patient was supported with intravenous fluids and transfusions of RBCs and within 24 hours from admission he underwent esophagogastroscopy, with local xylocaine spray, which revealed a Dieulafoy’s lesion inside a diafragmatocele (Figure 1) treated with adrenaline injection and endoclip placement. The procedure was uneventful, but after 24 hours the patient developed pain followed by swelling around left angle of mandible, without fever. On examination, there was swelling of left parotid gland (Figure 2), with tender on touch suggestive of parotitis, but there was no crepitus in the neck. Ultrasonography revealed diffusely swollen parotid gland (Figure 3), without salivary duct calculi, abscess, fluid collection or enlarged lymph nodes. The patient was hydrated and swelling disappeared spontaneously after 72 hours. He received a total of 3 units of RBCs during hospitalization, melena resolved and the patient was discharged home. No other complication was
noted during follow-up.
Conclusion: Upper gastrointestinal endoscopy is a procedure mostly uneventful. Parotid gland swelling is a rare complication of endoscopy and awareness of this benign condition can help relieve any apprehension that the patient and the endoscopist may have.

References

1. Lamtha SC, Rai U, Hassan PT. Parotid gland swelling after upper gastrointestinal endoscopy. J Family Med Prim Care. 2019 Jan;8(1):274-275.
2. Pawar SV, Zanwar VG, Jain SS, Rathi PM. Transient Parotitis After Oesophagogastroduodenoscopy: A Rare Complication. J Clin Diagn Res. 2015 Nov;9(11):OJ01.
3. Can, G., Yilmaz, B., Posul, E., Taslamacioğlu, T., Korkmaz, U., & Kurt, M. (2014). Rare complication of upper gastrointestinal endoscopy: bilateral swelling of parotid glands. Acta gastro-enterologica Belgica, 77(3), 364.
4. Vadivel Kumaran, S., Sumathi, B., & Nirmala Natarajan, D. (2013). Transient parotitis after upper gastrointestinal endoscopy. Endoscopy, 45 Suppl 2 UCTN, E424–E425.

Clinical Case Summary

Introduction: Pancreatitis is an inflammatory disorder of the pancreas resulting in various complications, such as parenchyma/peripancreatic fluid collections and necrosis pseudocyst formation, benign biliary stricture and vascular complications like venous thrombosis and pseudoaneurysmal bleeding.
Case report: A 48-year-old man with a history of chronic alcohol consumption presented with abdominal pain and melena. He had no history of traumatism, hepatobiliary surgery or intervention on the hepatobiliary tract, but suffered from recurrent pancreatitis. The patient manifested Quincke’s triad of symptoms, upper gastro-intestinal hemorrhage (melena), jaundice and upper abdominal pain, without a pulsatile mass. The patient was haemodynamically stable with positive digital rectal exam for blood, while the laboratory tests showed significant reduction in hematocrit (Hct: 23,6%, Hb: 7,7 g/dL), elevated serum amylase (AMY: 780 U/l) and hyperbilirubinemia (TBL: 3 mg/dL, DBL: 2,3 mg/dL, γ-GT: 531 U/l, ALP: 443 U/l). The patient underwent upper GI endoscopy which showed an attached thrombus in the papilla of Vater (Figure A), with a pulsating duodenal wall. A vascular lesion was then suspected and an urgent contrast-enhanced computed tomography (CT) scan was performed. The CT scan confirmed the presence of a pancreaticoduodenal arterial pseudoaneurysm (Figure B, red arrow) due to an eroding pancreatic pseudocyst (Figure B, green arrow) filled with blood clots, without active extravasation. Ultimately, we decided to treat the patient using endovascular therapy. Arterial catheterization and angiographic evaluation of the pancreaticoduodenal pseudoaneurysm was performed, followed by coil embolization (Figure C). The postoperative course was uneventful and the patient was discharged on the 16^th postoperative day.
Conclusions: Bleeding pseudoaneurysm is a rare but life-threatening complication of pancreatitis with high rates of mortality if left untreated.

References

1. Barka, M., Rguez, A., Ben Cheikh, Y., Chaouch, M. A., Jarrar, M. S., Ben Abdessalem, Z., Hamila, F., & Youssef, S. A case report of gastroduodenal artery pseudoaneurysm and giant pancreatic pseudocyst following acute pancreatitis revealed by obstructive jaundice. SAGE Open Med Case Rep. 2023 Jul 29:11:2050313X231188885.
2. Walia, D., Saraya, A., & Gunjan, D. Vascular complications of chronic pancreatitis and its management. World J Gastrointest Surg. 2023 Aug 27;15(8):1574-1590.
3. Dhali, A., Ray, S., Sarkar, A., Khamrui, S., Das, S., Mandal, T. S., Biswas, D. N., & Dhali, G. K. Peripancreatic arterial pseudoaneurysm in the background of chronic pancreatitis: clinical profile, management, and outcome. Updates Surg. 2022 Aug;74(4):1367-1373.
4. Shelar SS, Dhande R, Nagendra V, Suryadevara M, Shetty N. Giant Abdominal Pseudoaneurysm Secondary to Recurrent Pancreatitis: Imaging and Endovascular Intervention. Cureus. 2022 Dec 23;14(12):e32872.
5. Chiang, H. C., Lo, Y. N., Chen, C. Y., Chuang, C. H., Hong, T. C., & Kuo, H. Y. Acute pancreatitis complicated with peripancreatic pseudoaneurysm and subsequent intra-abdominal hematoma. JGH Open. 2023 May 9;7(5):393-394.

Clinical Case Summary

Aims: Intestinal malrotation is a congenital anomaly with only 0.2–0.5% of cases reported in adolescents and adults. Intestinal malrotation rarely has an asymptomatic course, in which the diagnosis is usually made incidentally. Duodenal varices account for only 17% of total ectopic variceal bleeding and have an estimated mortality rate of 40%. Management of bleeding ectopic varices usually requires an endoscopic treatment (band ligation or sclerotherapy) or radiologic intervention and rarely surgical intervention.
Methods: We report a female patient who presented with acute blood loss due to large bleeding duodenal varices fed by a portosystemic shunt from the superior mesenteric vein.
Case report: A 79-year-old female patient with a medical history of arterial hypertension and diabetes presented to the emergency department with melena. Physical examination revealed pallor and a hemodynamically stable patient (BP: 107/61 mmHg, HR: 88/min), with positive digital rectal exam for melena. The laboratory tests showed significant reduction in hematocrit (Hct: 22.6%, Hb: 7 g/dL) and elevated urea value (Urea: 137 mg/dL, Cre: 1,6 mg/dL). The patient was supported with intravenous fluids and transfusions of RBCs and within 12 hours from admission she underwent esophagogastroduodenoscopy which revealed spurting isolated enlarged duodenal varices (Figure) successfully treated with cyanoacrylate glue injection (Figure). Contrast-enhanced computerized tomography of the abdomen was suggestive of extensive varicose veins both around the head of the pancreas and the 2nd part of the duodenum (Figure) due to superior mesenteric vein occlusion, chronic mesenteric volvulus with “whirlpool sign” (Figure) and intestinal malrotation, with cecum and ascending colon in a high position in the right hypochondrium (Figure).
​Conclusions: Intestinal malrotation is a quite rare anomaly in adults, especially when is complicated with superior mesenteric vein occlusion and ectopic varices.

References

1. Dzwonkowski, M., Iqbal, U., Kaufer, S. W., Khan, Y. I., & Kumar, K. (2022). Endoscopic Band Ligation of Bleeding Duodenal Varices. Cureus, 14(2), e22009. https://doi.org/10.7759/cureus.22009
2. Nguyen, R. K., Crouse, R. M., Talbot, E. A., & Allard-Picou, A. K. (2022). Intestinal Malrotation in the Adult. The American surgeon, 88(6), 1367–1368. https://doi.org/10.1177/0003134820947406
3. Glosser, L. D., Lombardi, C. V., Knauss, H. M., Rivero, R., Liu, S., & Jones, T. J. (2022). Case report of congenital intestinal malrotation in an adult discovered three months status-post appendectomy. International journal of surgery case reports, 91, 106795. https://doi.org/10.1016/j.ijscr.2022.106795
4. Kafadar MT, Cengiz AY, Çaviş T, Bilgiç İ, Nadir I. Incidental intestinal malrotation in an adult: Midgut volvulus. Turk J Surg. 2018 Dec 1;34(4):337-339. doi: 10.5152/turkjsurg.2017.3468.

Disclosure

Conflict of Interest disclosure
The authors declare that there are not conflicts of interest.

Clinical Case Summary

Splenic artery (SA) pseudoaneurysms are rare but potentially fatal complications of pancreatitis, with a mortality rate up to 52%. A good prognosis relies on early diagnosis and treatment.
A 54-year-old man, recently splenectomized due to spleen rupture, and with a history of alcohol and tobacco abuse presented with hematemesis, melena and acute anemia. Upper endoscopy revealed a blood vessel without ulcer in the posterior gastric body (PGB), suggestive of Dieulafoy lesion, treated with adrenaline and through-the-scope clips (TTSC). During hospitalization there were 2 more hemorrhagic events: the first treated with TTSC; the second with an over-the-scope clip (OTSC).
Weeks after being safely discharged, he presented with hematemesis, epigastric pain and acute anemia. A presumed Dieulafoy lesion in the PGB was treated with an OTSC. A computed tomography (CT) scan was performed to exclude vascular malformations, revealing an hipodense heterogeneous area in the pancreas tail contacting the SA and the PGB, suggestive of intraductal papillary mucinous neoplasm with malignant transformation.
An endoscopic ultrasound with biopsy was scheduled but, before that happened, another episode of hematemesis with hemodynamic instability required a CT scan, revealing a pseudocyst adjacent to the PGB. Reviewing his medical records from another hospital: a CT scan performed at the time of the spleen rupture described this collection, consistent with a pseudocyst based on cytology.
Subsequent angiography confirmed a SA pseudoaneurysm communicating with PGB, treated with microcoil embolization. The patient was discharged after an uneventful recovery. No more hemorrhagic events were recorded for 10 months.
SA pseudoaneurysms require a high index of suspicion for early diagnosis. This case emphasizes the importance of reviewing medical records and considering previous evidence of pancreatitis in at risk patients. Prompt diagnosis and intervention can prevent life-threatening hemorrhage.

References

Endoscopic images (a total of 10-12 pictures); CT scan images (a total of 3-4).