Edited by: Eytan Wine, University of Alberta, Canada
Reviewed by: Amit Assa, Schneider Children’s Hospital, United States; Wael El Matary, University of Manitoba, Canada; Jason Silverman, University of Alberta, Canada
Specialty section: This article was submitted to Pediatric Gastroenterology, Hepatology and Nutrition, a section of the journal Frontiers in Pediatrics
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Crohn’s disease may be complicated by the development of penetrating (fistulizing) or structuring complications. The presentation of an intra-abdominal phlegmon or abscess with or without an associated fistula has traditionally required surgical intervention. This series of two cases illustrates a beneficial role of non-surgical management, with parenteral and then enteral nutrition playing central roles. This report further elaborates the potential adjunctive role of enteral nutrition in the management of this complication of CD.
Crohn’s disease (CD), one type of inflammatory bowel disease (IBD), may present at any age, with up to a quarter of cases diagnosed in childhood or adolescence (
Exclusive enteral nutrition (EEN) is a well-established therapy to induce remission in pediatric-onset CD (
We have previously reported the use of EEN in an adolescent with an entero-vesical fistula (
A 12-year-old boy presented to a peripheral hospital with a 12-month history of increasingly severe abdominal pain over a 4-week period prior to his hospital admission. He was noted to have had poor growth and anorexia for approximately 12 months, with recent weight loss (estimated at 2 kg) and lethargy. He had otherwise been well in the past, and there was no family history of note.
On initial examination, he was found to be mildly febrile but not tachycardic, with a tender mass palpable in the lower central abdomen. Weight was 26.7 kg with height of 143.7 cm and body mass index (BMI) of 12.9. Initial bloods showed a microcytic anemia, with normal white cell and platelet counts. CRP was elevated (237 mg/l) and albumin was at the lower range of normal (35 g/l). Abdominal CT scan with oral contrast showed circumferential wall thickening over the distal 17 cm of the ileum, with an adjacent loculated collection of fluid and gas measuring at least 4 cm in diameter. There was mild proximal distention in the ileum, but no other small bowel lesions were seen, although sigmoid colon wall thickening was also evident. The child was then transferred to a tertiary pediatric center (Christchurch Hospital) for further and ongoing care.
Upon arrival in Christchurch, he was commenced on intravenous cefuroxime and metronidazole (based on his weight). Oral intake was ceased and he was started on parenteral nutrition (PN), with progressive increases in fat and protein delivery over the following 3 days. A peripherally located central venous catheter (PICC) was placed on the second day. Given the recent completion of the CT scan, cross-sectional imaging was not repeated: however, an ultrasound scan (USS) was undertaken on the day of arrival. This imaging confirmed the phlegmon, with suggestion of a fistula from the involved ileum to the collection.
During the rest of his stay, he underwent serial USSs. On Day 13, the collection was resolving, with improved ileal wall thickening. By day 19, the collection was felt to have reduced to a volume of 1 ml, with normal peristalsis of the persistently mildly thickened ileal wall. By day 25, the USS showed resolution of the collection, with further reduction in mural thickening. No new changes were evident.
He continued PN and antibiotics for 25 days. During this time, he progressively improved clinically and biochemically. The course was, however, complicated by the development of a drug reaction on day 20 (thought most likely secondary to cefuroxime). On day 25, he commenced enteral nutrition with Fortisip (Nutricia), which was well tolerated. He progressively increased his oral intake with this polymeric formula, until day 29 at which time he was on full EEN (8 × 200 ml fortisips daily, providing 2400 kcal/day) whereupon his PN was ceased. On day 33, he was discharged home, with ongoing EEN for 8 weeks in total. He was also commenced on oral azathioprine prior to discharge following standard protocol. Approximately 2 months post presentation, he underwent diagnostic upper gastrointestinal endoscopy and ileocolonoscopy. Endoscopically, there were scattered colonic ulcers with mild chronic inflammatory changes seen histologically.
Subsequently, he was seen on a regular basis with serial measurement of inflammatory markers. Three months after diagnosis, he was noted to be well, with no symptoms, had no positive examination findings, and had gained approximately 6 kg. CRP at this time was 2, along with normal albumin, platelets, and ESR. Sixteen months post diagnosis, he was well on maintenance azathioprine, with no symptoms, satisfactory growth, normal examination findings, and with normal inflammatory markers (CRP 3, albumin 36, platelets 326, and hemoglobin of 119). Ongoing follow-up continues.
This girl presented at 12 years of age with a history of several months of diarrhea and abdominal pain. She had anorexia with weight loss of 3 kg in the preceding weeks. At presentation, she weighed 49.25 kg and was 158.1 cm tall (with BMI of 19.7). Abdominal examination was unremarkable with no mass evident. Initial blood tests showed elevated CRP but normal platelet count. Abdominal USS showed ileal wall thickening with no extra-intestinal abnormalities. She proceeded to undergo an endoscopic assessment, with no specific abnormalities seen. Histologically, focal active gastritis and colonic eosinophilia was seen (unfortunately, ileal biopsies were not obtained).
Given her disease location, and presentation pattern, EEN was recommended and commenced on the day following her endoscopy. She started this therapy without concerns and was able to be discharged home 2 days later.
However, she re-presented with increased abdominal pain 3 days after discharge. She was found to have localized tenderness in the right iliac fossa, but no clear palpable mass evident. She proceeded to have an USS, which suggested a phlegmon. Repeat blood tests showed an elevated CRP (268 mg/l) and neutrophilia (15.1 × 109/l). After reviewing these results, she was commenced on intravenous antibiotics (metronidazole, gentamicin and amoxicillin) and placed on full intravenous fluids (nil by mouth). Magnetic resonance enterography (MRE) with oral and intravenous contrast was booked and PICC line placement was arranged for the following day. MRE confirmed the USS impressions of a phlegmon, with marked ileal wall thickening extending proximally from the ileocecal valve for 20 cm. The small phlegmon was seen lying anterior to the most severely involved area of small bowel with no fistulous tract evident and no other abnormality evident on the imaging.
She was managed with PN for a fortnight, during which time her inflammatory markers fell (CRP 4 g/l at day 11). Repeat USS imaging after 2 weeks of PN showed marked improvement of the phlegmon but showed continued ileal wall thickening. She then slowly recommenced EEN and was able to establish full EEN intake orally by day 17 (at which point PN was ceased). She was also commenced on azathioprine in standard fashion and discharged home on day 18.
Over the following period of time, she was followed as an outpatient. She completed 8 weeks of EEN and resumed normal diet subsequently. After this, she remained well, with maintenance EN and azathioprine (dose at 2.5 mg/kg/day with satisfactory 6-thioguanine nucleotide levels). Follow-up included subsequent USS imaging, which showed no recurrence of collection, and routine review of serum inflammatory markers (with no increase in CRP).
Unfortunately, 18 months following diagnosis, she represented with abdominal pain and was found to have a palpable tender mass in the right iliac fossa. She was admitted and underwent a further USS followed by a second MRE, along with routine blood testing. Imaging showed recurrence of phlegmon without evidence of fistula or any other changes. She was again commenced on PN, with prompt improvements (clinically, biochemically, and radiologically) in the following 2 weeks. She restarted EEN at this time orally and quickly reached required daily volumes (7–8 fortisips daily, providing 2,100–2,400 kcal daily).
Given her recurrent presentation, surgical review was requested during this admission and arrangements were made for elective ileocecal resection. In the interim, she continued EEN. Resection was completed approximately 2 months later, with 17 cm of ileum resected along 8 cm of colon and primary re-anastomosis completed. Post-operatively she was managed with 3 months of oral metronidazole and continued oral azathioprine. Routine surveillance colonoscopy 6 months after her operation showed >6 aphthous ulcers at and distal to the anastomosis, with no stricture or other changes. She was subsequently commenced on adalimumab (standard dosage) in combination with azathioprine. Subsequently, over the following 5 years, she has been well, with no recurrent fistulizing disease, or other complication of CD.
These two cases illustrate the potential adjunctive role of nutritional interventions in the management of ileal CD complicated by the development of phlegmon. Both children had radiological features of a collection adjacent to the distal ileum, with a fistula demonstrated in one instance. Both children were managed in the same fashion with initial gut rest and intravenous nutrition, antibiotics, and subsequent EEN. Although the second child had recurrence of disease many months later, the acute management on this second occasion was similar and again successful. Case 1 has, however, had no further disease complications over more than 18 months of further observation. In addition to the previous case reports of EEN having roles in the management of fistulizing CD in other locations (
The benefits of EEN in the induction of remission of active CD have been described over the last decades, with the first comparative data coming from O’Morain and colleagues in 1986 (
In addition to the roles of inducing remission, EEN also clearly leads to high rates of mucosal healing, along with macro- and micro-nutritional benefits and enhanced bone health (
The adult and pediatric studies evaluating the roles of EEN in active CD have typically included subjects with luminal disease, with exclusion of those with fistulizing complications (
Adult reports have also illustrated the benefits of EEN for fistulizing CD. A Chinese report showed that 30 of 48 adults with entero-cutaneous fistulae had complete healing after up to 3 months of EEN (
The management of a phlegmon or intra-abdominal abscess in the context of CD has variously included peripheral drainage, antibiotics, and early surgical intervention (
This case report does not include any identifying features throughout. Accordingly, formal approval from the University of Otago Ethics Committee was not required. Written informed consent was obtained from Patient 2 and the parents of Patient 1 with their agreement to the publication of this case report.
AD prepared the initial draft of the manuscript and participated in the drafting of the work. SB participated in the drafting of the manuscript. Both authors approved the final version of the manuscript.
The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. The reviewer JS and handling editor declared their shared affiliation.