Introduction
Acute bowel ischemia (ABI) is a relatively rare disease, but with a very serious prognosis. It may be caused by obstruction of the visceral artery (embolism, thrombosis, dissection, vasculitis), visceral artery spasm or decrease in visceral perfusion pressure, for example in the course of shock (NOMI, non-occlusive ischemia) or visceral venous thrombosis. In the majority of cases ABI leads to necrosis of intestine wall, peritonitis and patient’s death [1−3]. Much seldom it comes to focal ischemia of the small bowel. It is self-limiting condition, thanks to the partial restore of blood supply via collateral circulation [4]. In such cases, bowel ischemia concern mainly its mucosa and submucosa without of transmural necrosis and bowel perforation. However, even such submucosal ischemia may come to the acute and chronic focal ischemic ileitis, extensive mucosa ulcerations, secondary bowel stenosis and ileus, as well as to an increase of intestinal wall permeability for endotoxins or bacteria. Focal ischemic ileitis requires a differentiation with for example Leśniowski-Crohn’s disease [5, 6] and should be suspected in patients with even short but recurrent episodes of bowel ischemia. They may occur in cocaine, ergotamine, amphetamine or phenylephrine misusers (due to visceral artery spasm), in long-distance runners, in patients with Dunbar syndrome (entrapment of the celiac trunk by the median arcuate ligament of the diaphragm) [7], vasculitis [8] or in individuals with episodes of permeable ileus (intestine wall extension leads to increase of bowel wall intramural pressure and decrease in local perfusion) [1−3, 5, 9, 10]. They are also possible in patients with a history of all kind shocks, low cardiac output syndrome, aorta surgery, portal hypertension, etc., which are the causes of non-occlusive mesenteric ischemia (NOMI) [1−3, 11, 12]. Another cause of focal ischemic ileitis are atheroemboli (cholesterol embolization syndrome) or small thromboemboli (heart or ulcerated plaque originated), which may appear after large vessels catheterization in coronarography or in other endovascular interventions [1−3, 10−16].
The increasing prevalence of narcotic, some OTC and stupefying drugs misuse, which may cause spasm of splanchnic arteries, medicine progress, which allows to save patients in serious conditions, including various types of shock, and constantly increasing number of endovascular procedures with risk of arterial microembolisation, require the preparation of physicians for diagnosis and treatment their late complications, i.a. localized in digestive tract sub form of acute and chronic focal post-ischemic ileitis with bowel narrowing and hyperpermeability [5, 11−12]. In this paper we present a patient with recurrent febrile states, abdominal pain and distension, who finally was diagnosed as having ileus secondary to post-ischemic bowel narrowing and required long resection of the long intestine segment and parenteral nutrition application.
Case report
Seventy year-old patient was admitted to the ward on 12th of December 2012 in severe general condition, with breathless, septic fever, dehydration, abdominal colic pain, diarrhoea and substantial weight loss, accompanied by clinical, biochemical and immunological markers of protein-energy malnutrition. Patient had a past medical history of chronic obstructive pulmonary disease, chronic pancreatitis with cyst, and gastric ulcer disease complicated by pylorostenosis. Patient’s medical documentation analysis revealed also that between 28th of August and 11th of September 2012 he was hospitalized in the lung diseases ward due to severe pneumonia complicated by septic shock and acute respiratory failure requiring some days long respiratory therapy. After some clinical stabilization, due to the elevated troponin, in spite of lack of chest pain (for some days patient couldn’t speak) and typical abnormalities in ECG, the patient was transferred to the cardiology department with suspicion of acute myocardial infarction (at 11th of September 2012). However, after patient’s admitting in transthoracic and transoesophageal echocardiography the focal wall motion abnormalities or bacterial vegetation on valves cusps (endocarditis feature) were not found. Therefore, elevated troponin values were interpreted as secondary to patient’s condition and coronarography was not performed, especially that patient’s condition at that time was not good, and he presented a fever, breathless, abdominal pain, diarrhoea, but without blood. The laboratory tests showed normal activity of amylase and lipase in blood and urine, elevated C-reactive protein (CRP) level, neutrophilic leucocytosis with a left shift, and hypoalbuminemia. Finally, basing on clinical and radiological studies, pneumonia was diagnosed as a cause of patient’s state and antibiotic treatment with cefotaxime was started. However, the variability in patient’s state was observed; the clinical improvement was achieved when patient remained on the empty stomach due to abdominal symptoms exacerbation, and the early recurrence of fever, vomiting, abdominal distension and an increase in inflammatory markers were observed soon after oral nutrition introduction. In the context of repeated negative tests for pancreatitis and a recent history of septic shock, an acute bowel injury, probable ischemic in its course were suspected as a cause of clinical appearance. Diagnostic procedures to confirm clinical suspicion were performed. Sigmoidoscopy did not show features of ischemic or pseudomembranous colitis. However, angio-CT scan of the abdomen and pelvis with contrast shows, among other things, only thickening of the small bowel mucosa with no signs of ileus, preserved patency of both mesenteric artery and celiac trunk, and cyst of the pancreatic head without signs of abscess and the other signs of pancreatitis considered in CTSI. So, it was lacking of evident indications for surgical intervention. In this context and the progressive deterioration of patient’s nutritional status and risk of oral feeding the total parenteral nutrition through a central vein route and rehabilitation were recommended. In the same time, antibiotic therapy with vancomycin against sensitive strain of intestinal enterococci G+ (Enterococcus faecalis) cultured from blood, urine, sputum and pressure wound. Finally, the improvement of the patient clinical condition was gradually achieved and on 26th of October 2012 he was discharged home from cardiology ward.
As mentioned above, on 12th of December 2012 the patient required re-hospitalization. Antibiotic therapy with vancomycin was started, which improved patient’s state. Then, after surgical consultation and basing on patients history, previous and present microbiological cultures, and small bowel wall thickening in two CTs patient was qualified for diagnostic laparoscopy with an initial diagnosis of partial ileus. The extended loops of small bowel with several segmental stenosis and characteristic fibrous remodelling were found intra-operatively (with histological examination). However, as the cause of recurrent ileus the intestinal loops conglomerate with was recognized and immobilized in the pelvis. The loops were released from peritoneal adhesions and the entire small bowel was checked out. Unfortunately, in the postoperative period, patient’s general condition was not getting better, recurred abdominal pain, vomiting and abdominal distension with radiological signs of ileus, increased inflammatory markers level. After following surgical consultation patient was qualified for laparotomy, during which extensive intestine resection with residual 140 cm of functional small bowel and ileostomy were performed. The single, proximal intestine narrowing, rather irrelevant for bowel passage was left to save the already shortened jejunum and to avoid further short bowel syndrome. Histopathology of stenosis in resected bowel showed on fibrous tissue proliferation. Within post-operative period the antibiotic treatment with vancomycin of wound infection and severe pneumonia (sputum cultures revealed ‘enteric’ streptococci G+ sensitive to vancomycin). At the same time total parenteral nutrition and intensified physical and respiratory rehabilitation were carried out. In following days, patient’s state gradually improved, partial enteral nutrition with peptide diet was introduced. After 2 months of such therapy patient become self-serviced, but enteral nutrition was not sufficient to cover his needs due to short bowel syndrome. Due this reason patient was qualified to home parenteral nutrition and after family education was referred to ambulatory nutrition center (from the 17th of February 2013).
Initially, home therapy coursed without problems, however after 3 weeks appeared signs of catheter infection by the same enterococci sensitive to vancomycin. The patient was admitted to the internal ward for the antibiotic therapy, but during the hospitalization stroke occurred. The patient was referred for neurological rehabilitation, but after short time due to gastrointestinal bleeding he was admitted to surgical ward. Then, due to another stroke, he was referred to the neurological ward, on which he died on 28th of May 2013.
Discussion
In described patient with a history of chronic cardiovascular, pulmonary and gastrointestinal diseases, in whom in short period after septic shock in the course of pneumonia fast- progressive small bowel narrowing appearing initially as transient bowel passage disturbances associated with oral feeding and finally as ileus [1−3, 5, 6, 9] as well as recurrent sepsis and multi-site infections caused by enterococci G+. Resection of damaged intestine gave only temporary relief, probably due to spreading and periodic activation if infection foci, including infection of the catheter for parenteral nutrition. The relationships between onset of intestinal symptoms and prolonged hipotonia during shock, recurrent sepsis caused by intestinal bacteria, the association between beginning of oral feeding and septic symptoms exacerbation, in our opinion, justify suspicion that during septic shock intestinal injury in this patient appeared. The most probably pathomechanism of this injury seems to be intestinal ischemia sub form of NOMI due to prolonged hipotonia and use of high dose of catecholamines [1−3, 10−16].
In presented case, we are not fully sure that the intestinal injury were caused by acute, focal, non-occlusive splanchnic ischemia. However, patient had no intestinal symptoms earlier, their beginning was associated with septic shock potential cause of multi-organ ischemia, which may also lead to increase intestinal barrier permeability, which explain intestinal type of bacteria cultured from blood, sputum, pressure wound and urine. Moreover, bowel injury had a progressive character, what suggested that prolonged hypotonia in the course of septic shock only started self-sustaining inflammatory process in the intestinal wall, for example sub form of focal ileitis, which rolled on till ileus appeared. It seems that these arguments are enough to justify ischemic pathogenesis of bowel injury in presented patient, especially that macroscopic and histological signs of the other potential causes of bowel narrowing such as Leśniowski-Crohn disease or neoplasm were not found. In the available literature we found only one description of similar case [4], but some papers suggest the possibility of intestinal complications in the course of septic shock [1−3, 11, 14]. According to Mitsuyoshi et al. [12] NOMI should be suspected in patients undergoing cardiac surgery (risk of low output syndrome) or chronic hemodialysis, when the three of the four following criteria are completed: abdominal symptoms, including abdominal pain, vomiting occur and ileus develop; catecholamine use (cause spasm of splanchnic artery); presence of hypotension episodes, which lead to ischemia per se, as well as are the cause of catecholamine use; and the slow serum transaminases increase. In our patients three of mentioned criteria were fulfilled, but transaminases were not determined in the ward of lung diseases. In such cases authors recommends performance of two-phase vascular examination (angio-CT, angio-NMR or classic abdominal angiography) [1]. If they show no abnormalities, the other symptoms causes should be investigated. Whereas, when mesenteric large vessel occlusion is found, it should be pursued to recanalization (thrombolysis, surgical thrombectomy or using Rotarex) [14−16]. In case of features of diffuse visceral artery spasm, which is a typical feature of NOMI, recommendations are as follows: catecholamine discontinuation or dose reduction, hydration and continuous infusion of vasorelaxants (papaverine, PGE1), intravenously or, preferably, directly to the superior mesenteric artery, under blood pressure control [1, 12, 14]. However, the confirmation of these abnormalities is only possible in acute phase of bowel ischemia. In our patient we could observe only complications of process starting in the course of septic shock, and two abdominal angio-CTs did not reveal significant disturbances in splanchnic blood in-flow and out-flow.
Presented patient after surgical treatment developed type 2 of intestinal failure [17], initially requiring the total, and then partial parenteral nutrition [18]. Because more than 30 cm of small bowel was left, enteral feeding with industrial peptide diet (Peptisorb) was applied, initially orally (after dilution with rice glue) and after the stroke-without dilution by the pump through the Flocare catheter [19]. Tolerance of enteral feeding with loperamide co-therapy have been improved over time. After a few weeks intestinal contents in the ileostomy was condensed and enteral nutrition become sufficient to cover full energy requirement. Thanks to them, after venous catheter infection and its removal, patient could be feed via enteral catheter in April and May 2013 [19]. Achieving the possibility of enteral nutrition in only four months after such an extensive resection of the bowel is not frequent [17].
In conclusion, in presented case the cause of progressive bowel injury and secondary fibrous narrowing as well as recurrent septic state is not fully clear and certain, however many clinical signs show on acute bowel ischemia with secondary focal ileitis as their most probable pathomechanism. Such type of complication are very rarely observed due to high mortality (80−100%) in the course of NOMI. In this case, despite the early clinical suspicion, it has failed to sufficiently document ABI complications in radiological imaging before contamination of the small intestine and infection spreading to many organs. These infection focuses were the most probable source of recurrent sepsis and subsequent nutritional catheter contamination, which, together with stroke, ultimately contributed to the patient’s death. The mystery remains, whether non-resected, proximal jejunal narrowing contributed to it as well. Although, it was reported that every digestive tract disorder, including colorectal cancer, may be potential source of sepsis or endocarditis [20].
The most important message linked to this case presentation is that mesenteric ischemia may appear both as acute and chronic disorder. The complications of acute bowel ischemia may be observed in survived patients for a long time, most common sub form some types of ischemic colitis, febrile states, and focal ileitis need to be to distinguish with stricturing Leśniowski-Crohn’s disease. The base for diagnosis of bowel ischemia is inclusion of this entity into differential diagnosis of symptoms as well as correlation of the symptomatology with different clinical conditions (risk factors) potentially leading to intestinal hypoperfusion, so not only atherosclerosis and atrial fibrillation, but also drugs, including OTC, and toxic substances (e.g. cocaine, ergotamine) taking by patients, history of vascular or endovascular procedures, and surviving of clinical conditions with cardiogenic or non-cardiogenic hypotension or hypovolaemia.
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Adres do korespondencji:
dr hab. n. med. Jacek Budzyński
Oddział Kliniczny Chorób Naczyń i Chorób Wewnętrznych Szpital Uniwersytecki nr 2 im. dr. Jana Biziela
ul. Ujejskiego 75, 85–168 Bydgoszcz
tel./faks: +48 52 36 55 347
e-mail: budz@cps.pl
Acta Angiol Vol. 20, No. 2 pp. 60–67
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