Ileostomy volvulus as an underreported problem causing small bowel obstruction in patients living with ostomy: a case report and literature review

Article information

Ann Coloproctol. 2023;.ac.2022.00976.0139

Publication date (electronic) : 2023 March 2

doi : https://doi.org/10.3393/ac.2022.00976.0139

Julianna Seo^1,*, Ishith Seth^,1,2,*, Dilshad Dooreemeah¹, Chun Hin Angus Lee¹

¹Department of Surgery, Bendigo Health, Bendigo, Australia

²Central Clinical School, Monash University, Mulgrave, Australia

Correspondence to: Ishith Seth, MD Central Clinical School, Monash University, 99 Commercial Road, Mulgrave 3004, Australia Email: ishithseth1@gmail.com

*Julianna Seo and Ishith Seth contributed equally to this study as co-first authors.

Received 2022 November 11; Revised 2022 December 23; Accepted 2023 January 9.

Abstract

INTRODUCTION

Volvulus results from torsion of the bowel around the axis of its mesentery, causing bowel obstruction and ischemia. It most often occurs at the sigmoid colon and caecum, and volvulus accounts for 5% of bowel obstructions in developed countries. Small bowel volvulus is an extremely rare complication, with a reported incidence of 1 to 5 cases per 100,000 in the developed world [1–3].

Ileostomy formation is a common intervention in elective and emergency settings, and the resulting stoma may be permanent or temporary. Currently, 120,000 ileostomies and colostomies are performed annually in the United States, carrying significant morbidity and altering patients’ quality of life. Harris et al. [4] found the most common stoma-related complications were herniation, necrosis, prolapse, stenosis, fistula, and small bowel obstruction (SBO). SBO following an ileostomy is usually the result of postoperative adhesions within the abdomen or pelvis, or at the site of ileostomy. A complication of stoma formation is stoma outlet obstruction (SOO), which occurs just below the site of the stoma. SOO may be caused by ileostomy volvulus; however, volvulus in the absence of adhesions following ileostomy formation is extremely rare.

Ulcerative colitis (UC) is a chronic inflammatory bowel disease, and stoma formation is required in 4% of UC patients within 5 years of diagnosis [2, 5]. This case report presents a 44-year-old woman with UC who had a recent ileostomy complicated by SBO secondary to volvulus with no adhesion formation. We also conducted a systematic literature review to investigate the causes, diagnosis, treatment, and preventative measures of ileostomy-related SBO.

METHODS

Case report

A 44-year-old woman with refractive UC presented to the emergency department with worsening abdominal pain, feculent vomiting, and high stoma output. Prior to presentation, she relayed no history of sick contacts or a precipitating cause. Her past medical history included Turner’s syndrome and medically refractory UC refractory to azathioprine, vedolizumab, and adalimumab. Surgical history included laparoscopic total colectomy and end ileostomy 11 months prior, with completion of proctectomy 5 months ago. On examination, she was afebrile and tachycardic (heart rate, 122 beats/min) with an abdomen that was soft and mildly distended. Generalized tenderness was noted on light palpation with absent bowel sounds. The stoma bag contained a small volume of feces but was unable to be digitated, and blood tests revealed mild hyponatremia but no other derangements. Abdominal computed tomography (CT) showed an SBO with a transition point at the level of the right iliac fossa ileostomy. A nasogastric tube (NGT) was inserted, and the patient was fluid-resuscitated. Diagnostic laparoscopy and flexible ileoscopy were performed, revealing small bowel dilatation and ileostomy stenosis to the level of the fascia. The small bowel remained viable and the ileoscopy showed no evidence of Crohn’s disease. The patient had a short postoperative admission and was discharged a few days later.

The patient represented 2 months later with the same symptoms. On review, the patient was hemodynamically stable but experienced parastomal tenderness. The stoma appeared pink and healthy, and a digital examination revealed a superior parastomal hernia that was reducible. A clinical diagnosis of SBO with SOO and volvulus was made. Intraoperatively, a small bowel volvulus and a short segment of dusky small bowel along the ileostomy axis were found, but no parastomal hernia or adhesions (Fig. 1). The ileostomy volvulus was reduced laparoscopically (Supplementary Video 1); however, the distal small bowel appeared purple-grey and required resection (19 cm) (Fig. 2), so a local revision of the end ileostomy was performed. The admission was complicated by Enterobacter bacteremia, hyponatremia, and sinus tachycardia secondary to high stoma output. She was discharged 15 days postoperatively and has shown no recurrence of volvulus or SBO. Her stoma was fully functioning at 2-, 6-, and 12-week follow-up visits.

Fig. 1.

Ileostomy volvulus without the presence of any adhesions.

Fig. 2.

Resection of distal small bowel that appeared purple-grey in color and required resection.

Literature search

The PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analysis) guidelines were adhered to. This systematic review was registered on the PROSPERO (International Prospective Register of Systematic Reviews; No. CRD42022349075). PubMed (Medline), Embase, Google Scholar, Scopus, and Cochrane CENTRAL were searched for relevant studies published from January 1901 to August 2022 [6]. The search terms included different combinations of “ileostomy,” “stoma,” “small bowel obstruction,” “SBO,” and “volvulus.” The complete search strategy is shown in Supplementary Table 1, and the study selection is shown in Fig. 3. The titles and abstracts of the studies were then manually screened by 2 authors (IS and DD) to narrow down those relevant to this review. No study restrictions were imposed in terms of different populations, races, ethnicity, origin, and language. Finally, the full texts of the studies were assessed for eligibility. Any disparity in either selecting eligible studies or assessing findings between the 2 reviewers was resolved through consultation with a third reviewer (CHAL).

Fig. 3.

PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analysis) flow diagram of selected studies.

Inclusion criteria

The established inclusion criteria consisted of studies that reported the diagnosis of SBO in patients with ileostomy volvulus, affirming this diagnosis by different imaging modalities, or surgery with sufficient data to be reported individually. All full-text studies, including randomized controlled trials, observational studies, case series, and case reports, were included.

Exclusion criteria

The exclusion criteria consisted of reviews, meta-analyses, opinion articles, letters to the editor, cadaver studies, animal studies, and studies with insufficient clinical data or those not able to be translated into English.

Outcome measures

The outcomes of interest were patients’ demographics, the imaging modality used for diagnosis, the indication for initial surgery, the type and configuration of the stoma, surgical treatment, and recurrence of volvulus.

Data extraction

Two independent reviewers (IS and DD) evaluated the studies based on the selection criteria and extracted the relevant data into a standardized form. The data included the year of publication, publication language, publication format (full-text article, letter to the editor, abstract form), type of study (case report, case series), age, sex, medical history, the indication of treatment, the imaging modality, surgical treatment, duration of follow-up after therapy, and the recurrence of volvulus. Disagreements between the reviewers were settled by discussion and adjudication by the corresponding author (AL).

Quality assessment

The quality of included reports was determined using the tool designed by Murad et al. [7] for assessing the methodological quality and synthesis of case series and case reports (Supplementary Table 2). All included studies were single case reports, case series, or observational studies. According to this tool, each study is evaluated based on 4 domains: selection of study groups, ascertainment, causality, and reporting. The items related to the selection of cases, ascertainment, and reporting were kept, and 3 items were removed from the causality domain (challenge/rechallenge phenomenon, dose-response effect) as they were not relevant to the current study. This resulted in a 5-item tool to assess whether the methodological quality of included studies was good, unclear, or low based on 3 possible answers for each item (yes, cannot tell, no).

RESULTS

Demographics

Seven studies were included, comprising 967 patients [2, 8–13]. The mean age was 40.9 years (range, 7 months-60 years) and 21% of patients were male (Table 1) [2, 8–13]. The majority (70%) of the included patients presented to the emergency department with abdominal pain and distension, whereas no patients presented with hemodynamic instability or septic shock. The predominant cause (90%) of ileostomy in these patients was colorectal cancer or UC. SBO and SOO were reported in all patients. Twelve patients had ileostomy volvulus.

Table 1.

Characteristics of included studies

Stoma outlet obstruction incidence

CT scans demonstrated the features of ileostomy volvulus in most cases. In particular, CT scans demonstrated a “cystic-like” structure compressing the ileum loop within the anterior abdominal loop just before the stoma opening. Utilizing the axial view of the preoperative CT scans, it was possible to measure the thickness of the subcutaneous fat from the skin to the rectus abdominis muscle surface at the umbilical level, as well as the thickness of the rectus abdominis muscle itself. Once ileostomy volvulus was diagnosed, low anterior resection with ileostomy was reported in 10.6% of patients, while ileal resection was reported in 3.6%. One case reported outlet obstruction after total colectomy [2]. Ileostomy and colostomy were surgically performed in most patients (90%), with ileostomy along with proctocolectomy performed in 2 patients.

Most included patients (95%) had loop ostomies, and the remainder had end ostomies. No cases of mortality were noted in the included study. The follow-up period ranged from 6 days to 6 months; no complications, other than recurrence of SBO in 13 patients, were reported at follow-up.

Table 2 shows the assessment of the methodological quality of included studies [2, 8–13]. Most included studies did not mention whether the reported cases represented the full experience of their centers; however, most other domains were consistently addressed. Nonetheless, the included studies reported good overall quality (93%). Most studies had adequate follow-up periods to evaluate long-term outcomes, but half of the included case reports/series were deemed to be good quality.

Table 2.

Assessment of the methodological quality of the included studies

DISCUSSION

The current study documents an unusual case of SBO following ileostomy volvulus and a systematic literature review. This review evaluated previous studies reporting volvulus as a cause of SBO in ostomy patients and found that the most common causes of ileostomy were colorectal cancer or UC, with abdominal CT being the most common diagnostic tool. Most patients had loop ostomies, and the second most common ostomy was endostomy. Only 13 individuals reported a recurrence of volvulus, and no mortality was reported during follow-up. Overall, the case reported herein is unusual, and ileostomy volvulus without the presence of adhesions has never been reported previously.

Normally, volvulus accounts for 2% of SBO; however, volvulus in the absence of adhesions in an ostomy patient has seldom been reported [14]. This may be due to difficulty in identifying and diagnosing ileostomy volvulus, as its etiology can be easily misattributed to subfascial adhesions. The patient documented in this case report presented to the emergency department with worsening abdominal pain, feculent vomiting, high stoma output, absent bowel sounds, and hemodynamic instability: this is congruent with the presentations described in the studies included in this systematic literature review, as well as current guidelines for clinically diagnosing obstruction in ostomy patients [15]. Features that would differentiate volvulus from other causes of SBO include the inability to easily insert a digit or NGT through the stoma, although this may also be present in stomal luminal stenosis. Considering that SBO as a complication of stomas is relatively common (4.6%–27.3%) and emergent management is needed to prevent gangrenous bowel resection and long-term morbidity, volvulus in ostomy patients should still be considered in the absence of adhesions [2, 16]. Inconsistencies in the definition of SOO within studies may also have limited their reporting. For example, Ohira et al. [12] defined outlet obstruction as intestinal obstruction after ileostomy with the part penetrating the abdominal wall, while Kitahara et al. [13] defined it as SBO symptoms with CT showing intestinal dilatation just below the penetrating part of the stoma site. These inconsistencies should be noted and unified in future studies.

Adhesions account for approximately 70% of SBO in adults, with up to 25% of patients who underwent abdominal-pelvic surgery subsequently developing adhesions. These are also the main cause of SOO [12, 17]. While laparoscopic surgery reduces the incidence and severity of adhesions compared to laparotomies, previous studies have commented that the absence of a fixation point in laparoscopic procedures would allow additional room for further twisting at the ileostomy site, increasing the risk of ileostomy volvulus [18]. Previous studies have also suggested loop stoma formation as a primary cause of SOO, identifying it as an independent risk factor compared to end ileostomy [10, 12]. Despite this risk, loop ileostomy also avoids life-threatening complications, such as the consequences of anastomotic leaks and perforation, and so should still be considered a viable ostomy option.

The definitive cause of volvulus in ileostomy patients is an ongoing debate, and etiologies aside from adhesions have been previously considered. Uchino et al. [19] reported cross incision of the rectus abdominis muscle sheath as a risk factor for torsion of the mesentery, while Ohira et al. [12] and Kanazawa et al. [20] stated that rectus abdominis muscle thickness ≥ 10 mm on CT was associated with a higher rate obstruction, but that difference was not statistically significant [21]. A study by Kameyama et al. [10] also stated the thickness of the rectus abdominis muscle may be a risk factor for obstruction due to increased resistance of stoma output, with Kitahara et al. [13] reporting that the rectus abdominis was significantly thicker in patients who experienced recurrent SOO. Preoperative steroid dosing, high stoma output, and the presence of malignant tumors were also noted by studies as potential risk factors. Kitahara et al. [13] reported an association between a lowered corticosteroid dose a month prior to surgery and an increased risk of SOO, but this is also a common guideline for medically refractory UC preoperatively and may simply reflect the risk of patients with worsening UC [22]. Nonetheless, Kameyama et al. [10] found that the perioperative steroid dose did not affect SBO incidence. Otherwise, Kitahara et al. [13] noted high stoma output as a risk factor; if concomitant with intravascular depletion, this causes edematous small intestinal mucosa that is predisposed to obstruction. Neoplasms of the small intestine are also considered potential risk factors, as the associated poor systemic well-being and malnutrition increases the risk of SBO [23]. Although these factors are largely unpreventable, the orientation and fixation of ileostomy can assist in preventing obstructions. Lee et al. [11] and Anderson et al. [9] emphasize the tendency of ileostomy to twist on its axis and form an obstruction. Antimesenteric fixation for the widening of adjacent ileal loops may be considered for correcting ileostomy orientation and reducing risk of SOO.

The main treatment options for SOO identified in the literature are conservative and surgical management. Conservative management largely involves the insertion of an NGT or transstomal decompression tube, as well as intravenous therapy. When conservative management fails, or cause of obstruction could not be managed medically, such as in cases of adhesions or recurrent SOO, or if there is a sense of urgency (e.g., a risk of intestinal necrosis), surgical management is explored. Operative management includes early takedown of the temporary ileostomy, adhesiolysis, or mobilization of the stoma [2, 7, 10, 13]. The study by Ohira et al. [12] noted the use of interventional radiology to manage SOO; however, the majority of patients who underwent this ultimately required early closure of the stoma as definitive management. The literature review revealed 2 main treatment options for SOO, but the literature largely focused on preventative measures to reduce the risk of SOO, signifying the need for further research on the management of SOO.

The current study uniquely reports an unusual case of ileostomy volvulus in the absence of adhesions and presents a discussion about the current literature on SBO in ostomy patients. Despite this, some limitations should also be acknowledged. First, due to the paucity of case-control studies, a definitive recommendation through a meta-analysis was not possible. However, the included studies were of good quality, as per the quality assessment tool. Secondly, discrepancies in the definition of SOO may have resulted in underdiagnosis of the pathology and limited the studies available for inclusion in the systematic literature review. Lastly, a larger volume of patients with risk characteristics for SOO (such as loop ileostomies, increased rectus abdominis muscle thickness, or lower preoperative total glucocorticoid dosage) must be included in future studies before these can be stated as definitive risk factors.

In conclusion, this case demonstrates the need for high clinical suspicion of SOO in patients with loop ileostomy, and rapid management should be undertaken. Several factors are associated with SOO, including loop ileostomy, increased rectus abdominis muscle thickness, and lower preoperative total glucocorticoid dosage; however, future studies are needed to evaluate these as risk factors.

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