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What is the etiology of this acute abdominal pain?

A 63-year-old Caucasian male presented to the emergency department (ED) with five days of fever (maximum 40°C), three days of jaundice, malodorous urine and altered mental status.

他沒有明顯的病史或手術史。他有一年30包的吸煙史,但否認飲酒或吸毒。體檢結果顯示,一名昏昏欲睡、麵容不好、患有黃疸的男性患者沒有出現急性窘迫。生命體征為:溫度40.8℃,血壓153/73,心率124,心率24,室內空氣中的血氧飽和度92%。患者表現為粘膜幹燥、鞏膜黃疸和右上象限壓痛,無腹膜體征。


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Laboratory data was significant for a leukocytosis of 15.5 K/μL with an 80% neutrophil predominance, hyponatremia of 128 mmol/L, and creatinine of 1.23 mg/dL elevated above his baseline of 1.00 mg/dL. His International Normalized Ratio (INR) was 1.71. Aspartate transaminase (AST) was 141 IU/L, alanine aminotransferase (ALT) was 153 IU/L, alkaline phosphatase was 239 IU/L, total bilirubin was 16.1 mg/dL, and direct bilirubin was 10.0 mg/dL. Lipase was within normal limits at 46 IU/L. Lactate was minimally elevated at 2.8.

RUQ護理點超聲(POCUS)顯示膽囊擴張,膽囊壁厚度正常,膽總管正常。膽泥無結石或息肉。超聲小組注意到膽道積氣和門靜脈氣體的超聲表現。

血管內灌注缺陷與多普勒缺席flow in the vasculature were highly suggestive of portal vein (PV) thrombosis (figure 1-3).


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Pylephlebitis CR -figure 1 air in PV, white arrows

Figure 1 – U/S air in the PV (white arrows)

Pylephlebitis CR -figure 2 thrombus in PSC red arrow

圖2-PSC/SV中的U/S血栓(紅色箭頭)

Pylephlebitis CR -figure 3 thrombus in portal vein, red arrow

Figure 3 – U/S thrombus in the PV (red arrow)

Computed tomography (CT) scan demonstrated thrombus and gas in the inferior mesenteric vein extending into the main PV and left PV( Figure 4-6). A comprehensive liver duplex scan further confirmed complete occlusion of the left PV and nonocclusive thrombi in the peripheral branches of the right PV.

幽門炎CR-圖4冠狀CT血栓在PV中(紅色箭頭)空氣在PV中(白色箭頭)

Figure 4 – CT thrombus in the PV (red arrow); air in the PV (white arrow)

Pylephlebitis CR -figure 5 coronal CT thrombus in PV (red arrow) air in __(white arrow)

圖5–PV中的CT血栓(紅色箭頭);腸係膜下靜脈中的空氣(白色箭頭)

Pylephlebitis CR -figure 6 inferior mesenteric vein occlusion_ red arrow

Figure 6 – CT air in the mesenteric vein (red arrow)

患者開始服用經驗性廣譜抗生素和治療性低分子量肝素(LMWH),並在血管升壓藥支持下進入內科重症監護病房(MICU)。血液培養變得泛敏感大腸杆菌.


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盡管采取了積極的複蘇措施,但在最初的24小時內,患者的乳酸從2.8上升到10.4。介入放射學(IR)團隊得出結論,鑒於血栓的位置和血栓負荷的嚴重性,技術上具有挑戰性的血栓切除術的風險大於益處。

The patient was intubated on hospital day (HD) three for acute hypoxic respiratory failure. Heparin was discontinued on HD5 after the patient passed a large volume of bright red blood from his rectum. The patient was diagnosed with antiphospholipid syndrome (APLS) based on the detection of anti-nuclear antibodies and anti-cardiolipin antibodies and underwent plasma exchange on HD7.

不幸的是,盡管越來越積極的抗生素治療方案,患者仍然存在菌血症,需要在HD9上添加第二種血管升壓藥。間隔成像顯示血栓進展。隨後,他出現彌散性血管內凝血,並於HD11因繼發於災難性APL的幽門炎而死亡。

討論

幽門腦炎是一種由腹腔內感染引發的化膿性血栓形成,最終流入門靜脈係統(PVS)。根的病因pyle-corresponds to the portal veins.

Most instances are associated with diverticulitis or appendicitis, but the disease also occurs as a complication of pancreatitis, inflammatory bowel disease, cholangitis, and spontaneous bacterial peritonitis. No infection was ever identified in our patient, which is true in less than 1% of documented cases.

Thrombophlebitis begins in the microvasculature at the source of infection and extends proximally to the PVS. More than 40% of patients have an underlying hypercoagulable disorder. Our patient had previously undiagnosed APLS. Hepatic abscesses are seen in 50% of cases and may require percutaneous or surgical drainage.

Other complications result from embolism, such as splenic infarct, and septic pulmonary embolism. Bowel infarct constitutes a large percentage of fatal outcomes, especially in cases involving mesenteric vein thrombosis that exacerbates the low perfusion state of septic shock.

Presentation and Work-Up

Fever and poorly localized abdominal pain are the two most common presenting symptoms, followed by nausea, vomiting, and jaundice. A fifth of patients present in shock. Although not always present, abdominal tenderness, hepatomegaly, and splenomegaly are important physical exam findings.

Laboratory studies often reveal leukocytosis with a left shift and elevations in hepatic enzymes, alkaline phosphatase, and bilirubin. In bacteremic patients, infection tends to be polymicrobial. Blood cultures are positive in 50% to 88% of cases, with microbial isolates being normal bowel flora such asBacteroides fragilisand大腸杆菌.

Diagnosis of pylephlebitis is made primarily via radiographic studies. Abdominal ultrasonography and contrast-enhanced CT scan both can demonstrate the presence of the pathognomonic combination of gas and thrombus within the PVS.

Linear echogenic areas with posterior reverberations consistent with intraluminal gas can be seen on US. Color Doppler showed hyperechoic material within the lumen of the vein. US is fairly sensitive for PV thrombosis, however, diagnostic accuracy is operator-dependent and can be limited by the presence of bowel gas. CT scan provides the benefit of revealing underlying infectious processes as well as complications such as bowel ischemia or hepatic abscess. Interval CT scans, in this case, did demonstrate the progression of thrombus, but no source of infection.

Antimicrobials and Source Control

一旦懷疑或診斷出幽門腦炎,應開始使用廣譜腸外抗生素進行治療,並在獲得培養和敏感性結果後進行定製。標準抗生素方案尚未建立,可能會因醫院協議而有所不同,但覆蓋範圍應包括革蘭氏陰性菌和厭氧菌。甲硝唑、慶大黴素、呱拉西林、氨苄西林和亞胺培南均已成功應用。

對於沒有肝膿腫的患者,建議至少使用四周的抗生素,對於有肝膿腫的患者,建議至少使用六周的抗生素。我們的患者在急診室開始使用經驗性萬古黴素和呱拉西林-他唑巴坦。當培養物培養出泛敏感大腸杆菌時,抗生素的範圍縮小到阿莫西林-克拉維酸和甲硝唑。當無法實現源頭控製且患者失代償時,覆蓋範圍再次擴大到萬古黴素和美羅培南。

Management of these patients often requires a multidisciplinary approach, involving gastroenterology, radiology, hematology, pharmacy and infectious disease. Although surgical intervention may not be pursued, consultation and involvement of surgical colleagues is prudent.

Patients may require procedures to achieve source control or to treat a complication. Some studies have shown surgical thrombectomy to be associated with a higher risk of the rate of recurrent thrombosis and therefore advise against it. In our case, IR concluded that the risks of thrombectomy outweighed the benefits.

Anticoagulation

近一半的幽門腦炎患者被發現有潛在的高凝狀態,因此早期谘詢血液學是至關重要的。住院近一周後,我們的患者被診斷為APLS,並開始接受確定性治療,包括大劑量類固醇和緊急血漿置換。然而,鑒於他的病情危急,這些幹預措施幾乎沒有時間抵消他去世前的血栓前狀態。

考慮到出血的風險,抗凝治療仍有爭議。一些供應商主張在腸係膜靜脈血栓患者中使用AC,因為這些患者因腸缺血死亡的風險增加。

However, AC in these patients with poorly perfused bowel has a high risk of gastrointestinal bleed. Heparin therapy was discontinued in our patient after a large spontaneous bleed.

Outcomes data is underpowered due to the low incidence of pylephlebitis, but data suggests increased recanalization rates and improved mortality rates with the use of anticoagulation. Currently, there is no consensus on duration and no data on the use of thrombolytics.

Resources:

https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3716219/#:~:text=Pylephlebitis%20is%20defined%20as%20an,fatigue%2C%20nausea%2C%20and%20vomiting.

https://www.journalmc.org/index.php/JMC/article/view/3050/2378

https://medcraveonline.com/JLRDT/pylephlebitis-both-a-surgical-and-non-surgical-pathology-a-2-case-report-and-literature-review.html(死亡率)

https://www.journal-of-hepatology.eu/article/S0168-8278(00)80259-7/fulltext(pathophys)

https://www.researchgate.net/publication/45826696_Pylephlebitis_An_overview_of_non-cirrhotic_cases_and_factors_related_to_outcome(pathphys, causes)

https://www.amjmed.com/article/S0002-9343(14) 00090-4/pdf(臨床表現)

https://westjem.com/case-report/pylephlebitis-in-a-previously-healthy-emergency-department-patient-with-appendicitis.html(臨床表現)

https://westjem.com/case-report/pylephlebitis-in-a-previously-healthy-emergency-department-patient-with-appendicitis.html(臨床表現)

https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3789899/(臨床表現)

https://www.researchgate.net/publication/45826696_Pylephlebitis_An_overview_of_non-cirrhotic_cases_and_factors_related_to_outcome

https://academic.oup.com/cid/article-abstract/21/5/1114/357326?redirectedFrom=fulltext

https://bmcgastroenterol.biomedcentral.com/articles/10.1186/1471-230X-7-22(影像)

https://somepomed.org/articulos/contents/mobipreview.htm?39/45/40670?source=see_link(影像)

https://www.amjmed.com/article/S0002-9343(14) 00090-4/pdf(影像)

https://www.sciencedirect.com/science/article/pii/S2444050715001412(treatment)

https://www.hindawi.com/journals/criid/2019/5341281/(treatment)

https://www.hindawi.com/journals/crira/2013/627521/(手術適應症)

https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4882085/(surgery may increase re-thromb)

https://link.springer.com/article/10.1007/s11239-019-01949-z(反間諜)

ABOUT THE AUTHORS

Dr. Effron is the assistant professor of emergency medicine at Case Western Reserve University and attending physician in the department of emergency medicine at the MetroHealth Medical Center, in Cleveland, Ohio.

馬裏蘭州巴努·班達是俄亥俄州克利夫蘭市MetroHealth醫療中心的急診醫師。

Diane Gramer RDMS是俄亥俄州克利夫蘭市MetroHealth醫療中心的RVT和RT。

Clare Charbonnet, MD is an Emergency Medicine Resident at MetroHealth Medical Center in Cleveland, Ohio.

亞當·布魯姆醫學博士is a staff radiologist at MetroHealth Medical Center in Cleveland, Ohio.

喬納森·格拉布,醫學博士is a staff radiologist at MetroHealth Medical Center in Cleveland, Ohio.

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