Пилефлебит, вторичный по отношению к острому аппендициту, случайно обнаруженный при томографии (клинический случай)

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Септический венозный портомезентериальный тромбоз, или пилефлебит, - это редкое патологическое состояние, вызванное внутрибрюшным инфекционным процессом и сопровождающееся значимыми осложнениями и смертностью. Клинические и лабораторные показатели не обладают достаточной специфичностью для выявления пилефлебита, в результате частота его развития недооценивается, диагноз ставится в основном с применением методов визуализации. Потенциально пилефлебит излечим, возможно уменьшить его осложнения, однако только в случае ранней диагностики заболевания.

В настоящей работе представлен клинический случай. Мужчина, 50 лет, проходил рентгенологическое обследование по поводу новообразования желудка, на котором был случайно обнаружен пилефлебит, вторичный по отношению к острому аппендициту. Применение внутривенной антибиотикотерапии, анти коагуляции и экстренной лапаротомии позволило достичь благополучного исхода.

Ключевые слова:пилефлебит, брыжеечная вена, антикоагулянт, острый аппендицит, экстренная лапаротомия

Финансирование. Исследование не имело спонсорской поддержки. 
Конфликт интересов. Авторы заявляют об отсутствии конфликта интересов.
Для цитирования: Бустаманте А.С., Флорес А.М., Кабальеро-Алварадо Х. Пилефлебит, вызванный острым аппендицитом, как случайная находка при томографии // Клиническая и экспериментальная хирургия. Журнал имени академика Б.В. Петровского. 2021. Т. 9, № 3. С. 131-135. DOI: https://doi.org/10.33029/2308-1198-2021-9-3-131-135

Pylephlebitis, also called portal venous septic thrombophlebitis, is a rare pathological entity, characterized by suppurative thrombophlebitis of the porto-mesenteric venous system [1]. The incidence is not adequately clarified; however, a study from 2009 reports it at 2.7 per 100,000 people, with a preference to the male sex [2, 3]. The clinical manifestations are not very specific and can manifest itself asymptomatically or as an acute abdomen [4]. Any organ whose venous drainage ends in the portal system is susceptible to generating pylephlebitis. Thus, we have diverticulitis, appendicitis, inflammatory bowel disease, pancreatitis, infectious enteritis, intestinal perforation and neoplasms as potential sources [5]. The treatment, currently not standardized, is based on antibiotic therapy, surgical control of the abdominal infectious focus and early anticoagulation. Over the years, each of these has been questioned, resulting in the judicious use of these therapies [6-9]. The objective of this report is to present the clinical case of an asymptomatic pylephlebitis as an incidental diagnosis by tomography.

Case report

A 50-year-old patient was admitted to the emergency room, referred from the radiology service due to the finding of suggestive signs of an abdominal inflammatory process compatible with acute appendicitis, and septic thrombophlebitis in the upper mesenteric and right ileocolic veins, seen on the contrast abdominal-pelvic multislice spiral tomography (fig. 1). The patient was undergoing an imaging study as a result of an anatomopathological report of a gastric biopsy, which led to a suggestive lesion of infiltrating tubular adenocarcinoma. There were no other comorbidities or previous surgeries.

Fig. 1. Contrast abdominal MSCT on admission. Coronal and axial images show upper mesenteric vein with non-contrast capturing hypodense image, suggestive of thromboembolism with alteration of mesenteric fat density

The review of the emergency clinical history found that 15 days before, he presented with colicky abdominal pain of sudden onset and moderate intensity, associated with nausea, vomiting and mucous diarrhea; being treated as an acute gastroenteroco-litis with antibiotic therapy (ciprofloxacin) and an-tispasmodics (scopolamine) orally for 5 days. The abdominal pain progressively decreased within 7 days, when he arrived at the oncologist's outpatient clinic, without any pain; however, he had recurrent fevers up to 38.5°C daily, which subsided with Paracetamol 1 gr orally.

The physical examination disclosed a soft, de-pressible abdomen, with mild pain on deep palpation in the right lower quadrant, no palpable masses, and negative Mc Burney and Blumberg signs. The laboratory tests found on admission are detailed in table. The treatment implemented was begun based on the diagnosis of pylephlebitis secondary to complicated acute appendicitis. Antibiotic therapy was started with Piperacillin-Tazobactam 4.5gr IV every 6 hours, analgesia with Metamizole 2gr IV C/8h. Enoxaparin 60UI subcutaneously every 12 hours was instituted and he was taken to the operating theater for an exploratory laparotomy via a midline infraumbilical incision. The intraoperative findings revealed a retroileal appendicular mass with a 8 x 1cm cecal appendix, perforated in its distal third and necrotic in its entire extension with an appendicular base in good condition. A 5cc abscess was contained within the mesoappendix. Histopathology of the appendix reported acute suppurative appendicitis.

Laboratory evolution

On the first postoperative day a diet was started, the surgical wound was without signs of phlogosis or bleeding, the abdomen was soft, without peritoneal signs and active ambulation was begun, with only mild pain in the surgical site. Piperacylin-Tazobactam 4.5gr IV was continued every 6 hours for 7 days and at discharge, laboratory results obtained were within normal limits (table) with tomographic findings reported as pericecal inflammatory sequelae and other organs evaluated without significant alterations (fig. 2). Rivaroxaban 20mg Q/24h x 30 days, Ciprofloxacin 500mg Q/12h x 3 more weeks, Paracetamol 1gr Q8h x 4 days are indicated; he returned to his outpatient visit 10 days later, without discomfort, and was referred back to his oncologist to continue with his gastric cancer treatment.

Fig. 2. Contrast abdominal MSCT control. Coronal and axial images show contrast capturing upper mesenteric vein, compatible with permeable vessel

Discussion

Pylephlebitis was described for the first time by Waller, in 1846, with the findings of liver abscesses in autopsies, considering it as an unusual complication of intra-abdominal infections [10]. In spite of the small number of cases, he reported a high mortality; from 30 to 50% of the cases [6, 11, 12]. Currently, sigmoid diverticulitis is the most frequent etiology (30%), followed by appendicitis (19%), inflammatory bowel disease (6%), pancreatitis (5%), infectious enteritis (4%), intestinal perforation and neoplasms (6%) [5].

A cohort of 95 patients identified the right portal vein as the most frequent site of thrombophlebitis, in 33% of the cases followed by the inferior mesenteric vein as the least common site with 8%. Septic thromboembolism in the superior mesenteric vein was documented in the literature with 30.5% [13].

The clinical manifestations are characterized by being ambiguous and not very specific; from the asymptomatic character, to the presence of abdominal pain (80.5%), fever (31.7%), muscular defense, nausea, diarrhea, anorexia or jaundice, with an average duration of 12.34±11.23 days [4, 13, 14]. Our patient described here presented with a 15-day clinical evolution, of regressive course and low intensity from the beginning, leading to a diagnostic error. The laboratory findings are also atypical, finding leukocytosis (80%), positive blood cultures (44-88%), alteration of hepatic enzymes (40-69%), elevation of total bilirubin (55%) and elevation of CRP [4, 13]. The organism isolated most frequently in blood cultures is Bacteroides fragilis over Escherichia coli and Streptococcus sp, resulting in bacteremia in 23 to 88% of patients [12]. Imaging studies have an important role and it is necessary to be attentive to their findings. The portal venous doppler ultrasound is able to identify portal venous thrombosis and, in association with the CT scan, are able to demonstrate the possible intra-abdominal septic focus and its affectation to the porto-mesenteric venous system. Both techniques facilitate and prioritize early diagnosis [15].

Currently there are no established protocols for the management of pylephlebitis, however, antibiotic therapy, anticoagulation and surgical control of the septic focus are recommended [8]. Antibiotic therapy should cover gram-negative and anaerobic flora utilizing metronidazole, gentamicin, piperacillin-tazobactam, imipenem-cylastatin, ceftizoxime and ampicillin for a period of 4 weeks in patients without liver abscess, and for 6 weeks if necessary [7]. The most controversial point of the therapeutic algorithm is undoubtedly anticoagulation. L. Naymagon et al., state that it should be used early whenever possible since it produces resolution rates of 58% vs 21% [16]. Likewise, T.C. Hall et al. describe the synergy with antibiotic therapy in the recanalization process [17].

On the other hand, N. Baril et al. suggest its use in selected patients diagnosed with pylephlebitis who meet criteria of hypercoagulability such as neoplasia or coagulation factor deficit. Our patient met the first described criterion and the diagnosis of pylephlebitis, thus justifying the use of this strategy [6, 18]. Upon discharge, Rivaroxaban VO was prescribed orally for 1 month, although some recommend Apixaban because of its greater range of safety in patients with atrial fibrillation and in the prevention of stroke over vitamin K antagonists K [19-21]. The surgical strategy for draining the septic focus is also not well clarified. Cases have been reported successfully treated by laparotomy or laparoscopy, both emergently and deferred; but all agree that intravascular septic emboli should not be delayed [22, 23].

Conclusion

This report illustrates a rare pathological finding, with potentially high morbidity and mortality complications, pylephlebitis. The poor specificity of the clinical picture points out that the imaging studies must be scrutinized. Acute appendicitis is the leading cause of surgical acute abdomen worldwide, so its presence should be suspected when there is thrombosis of the porto-mesenteric venous system. An incidental radiological diagnosis was made in our patient.

Литература/References

1.    Abdallah M., Gohar A., Naryana Gowda S., Abdullah H.M., Al-Hajjaj A. Pylephlebitis associated with inferior mesenteric vein thrombosis treated successfully with anticoagulation and antibiotics in a 37-year-old male. Case Rep Gastrointest Med. 2020; 2020: 1-3.

2.    Klinefelter H.J., Crawford W.E., Grose H. Pylephlebitis. Bull Johns Hopkins Hosp. 1960; 106: 65-73.

3.    Acosta S., Alhadad A., Svensson P., Ekberg O. Epidemiology, risk and prognostic factors in mesenteric venous thrombosis. Br J Surg. 2008; 95 (10): 1245-51.

4.    Ufuk F., Herek D., Karabulut N. Pylephlebitis complicating acute appendicitis: prompt diagnosis with contrast-enhanced computed tomography. J Emerg Med. 2016; 50 (3): e147-9.

5.    Falkowski A.L., Cathomas G., Zerz A., Rasch H., Tarr P.E. Pylephlebitis of a variant mesenteric vein complicating sigmoid diverticulitis. J Radiol Case Rep. 2014; 8 (2): 37-45.

6.    Baril N., Wren S., Radin R., Ralls P., Stain S. The role of anticoagulation in pylephlebitis. Am J Surg. 1996; 172 (5): 449-53.

7.    Zia A., Sohal S., Costas C. Pylephlebitis: a case of inferior mesenteric vein thrombophlebitis in a patient with acute sigmoid diverticulitis - a case report and clinical management review. Case Rep Infect Dis. 2019; 2019: 1-4.

8.    Hamera L., Abraham S., Jordan J. Pylephlebitis as a rare complication of ulcerative colitis: a case report. Cureus [Electronic resource]. 2019; 11 (5): e4792. URL: https://www.cureus.com/articles/19901-pylephlebitis-as-a-rare-complication-of-ulcerative-colitis-a-case-report (date of access April 17, 2020)

9.    Subercaseaux S., Zuniga S., Encalada R., Zuniga P., Berrrfos C. Pileflebitis asociada a apenaicitis aguda en una nina de 11 anos. Rev Chil Cir. 2010; 62 (2): 160-4.

10.    Bolt R.J. Diseases of the hepatic blood vessels. In: Bockus Gastroenterology. Philadelphia, PA, USA: W.B. Saunders, 1985: 3259-77.

11.    Imaoka K., Fukuda S., Tazawa H., Fukuhara S., Hirata Y., Fujisaki S. et al. A rare case of pylephlebitis as a complication of cholecystocolonic fistula. Case Rep Surg. 2018; 2018: 1-5.

12.    Plemmons R.M., Dooley D.P., Longfield R.N. Septic thrombophlebitis of the portal vein (pylephlebitis): diagnosis and management in the modern era. Clin Infect Dis. 1995; 21 (5): 1114-20.

13.    Choudhry A.J., Baghdadi Y.M.K., Amr M.A., Al-zghari M.J., Jenkins D.H., Zielinski M.D. Pylephlebitis: a review of 95 cases. J Gastrointest Surg. 2016; 20 (3): 656-61.

14.    Cho J.W., Choi J.J., Um E., Jung S.M., Shin Y.C., Jung S.-W., et al. Clinical manifestations of superior mesenteric venous thrombosis in the era of computed tomography. Vasc Spec Int. 2018; 34 (4): 83-7.

15.    Castro R., Fernandes T., Oliveira M.I., Castro M. Acute appendicitis complicated by pylephlebitis: a case report. Case Rep Radiol. 2013; 2013: 1-3.

16.    Naymagon L., Tremblay D., Schiano T., Mascarenhas J. The role of anticoagulation in pylephlebitis: a retrospective examination of characteristics and outcomes. J Thromb Thrombolysis. 2020; 49 (2): 325-31.

17.    Hall T.C., Garcea G., Metcalfe M., Bilk D., Rajesh A., Dennison A. Impact of anticoagulation on outcomes in acute non-cirrhotic and non- malignant portal vein thrombosis: a retrospective observational study. Hepatogastro-enterology. 2013; 60 (122): 311-7.

18.    Perez Aisa A., Rosales Zabal J.M. Pileflebitis. Gastroenterol Hepatol Contin. 2011; 10 (5): 246-9.

19.    Nery F., Valadares D., Morais S., Gomes M.T., De Gottardi A. Efficacy and safety of direct-acting oral anticoagulants use in acute portal vein thrombosis unrelated to cirrhosis. Gastroenterol Res. 2017; 10 (2): 141-3.

20.    Hale G.R., Sakkal L.A., Galanis T. Pylephlebitis treated with apixaban. Hosp Pract. 1995. 2019; 47 (4): 192-5.

21.    Granger C.B., Alexander J.H., McMurray J.J.V.. Lopes R.D., Hylek E.M., Hanna M., et al. Apixaban versus warfarin in patients with atrial fibrillation. N Engl J Med. 2011; 365 (11): 981-92.

22.    Simillis C., Symeonides P., Shorthouse A.J., Tek-kis P.P A meta-analysis comparing conservative treatment versus acute appendectomy for complicated appendicitis (abscess or phlegmon). Surgery. 2010; 147 (6): 818-29.

23.    Stitzenberg K.B., Piehl M.D., Monahan P.E., Phillips J.D. Interval laparoscopic appendectomy for appendicitis complicated by pylephlebitis. JSLS. 2006; 10 (1): 108-13.

ГЛАВНЫЙ РЕДАКТОР
ГЛАВНЫЙ РЕДАКТОР
Дземешкевич Сергей Леонидович
Доктор медицинских наук, профессор (Москва, Россия)
РОСМЕДОБР 2021
Вскрытие
Медицина сегодня
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