A Novel Surgical Treatment of Pediatric Fulminant Clostridium difficile Colitis

A 15-year-old White girl presented to the emergency department (ED) with one week of abdominal pain, diarrhea, and palpitations. She had a past medical history of autoimmune thyroiditis and gastritis. She was not on any medications and had no recent antibiotic therapy.

In the ED she was tachycardic, tachypneic, and tender in all 4 quadrants. Her white blood cell count (WBC) and C-reactive protein were elevated to 15.7 cells/mm³ and 33.8 mg/L, respectively. A computed tomography scan revealed pancolonic bowel wall thickening, hyperemia, and ascites (Fig 1). She was given supportive treatment with intravenous fluids, and pain control and stool cultures were sent. On her first hospital day (HD 1), she remained tachycardic, tachypneic, and diffusely tender and developed hematochezia. Stool cultures were positive for C difficile, and she was started on oral vancomycin. On HD 2, the patient had worsening abdominal distension and pain. She was transferred to the ICU where she was given additional intravenous fluids, metronidazole, and cefepime. On HD 3, the patient developed rectal prolapse with visible pseudomembranes (Fig 2). Her WBC was elevated to 34.6; she was thrombocytopenic and hypoalbuminemic (Table 1). Her daily abdominal radiographs revealed the progression of ascites (Fig 3). She was then diagnosed with severe, complicated C difficile pancolitis on the basis of criteria established by Neal et al⁵  (Table 2). A surgical consultation was obtained. After discussion of the options for operative management with the patient and her parents, it was decided to pursue a diverting loop ileostomy with antegrade colonic lavage in an attempt to preserve the patient’s colon, with the understanding that subsequent colectomy may be necessary if the patient did not improve.

A few hours later, she was taken to the operating room and underwent a diverting loop ileostomy with antegrade colonic lavage through a pezzar gastrostomy tube in the distal limb of the loop ileostomy. The colonic lavage was done intraoperatively, with 4 L of polyethylene glycol (PEG) and vancomycin 500 mg in 500 mL normal saline. A biopsy was also taken from the terminal ileum to evaluate for underlying IBD. The patient tolerated the procedure well.

Her postoperative treatment course followed the recommendations of Neal et al,⁵  which included antegrade vancomycin enemas 500 mg in 90 mL normal saline every 6 hours through the distal limb of the loop ileostomy and intravenous metronidazole for 10 days. On postoperative day (POD) 1, the patient had a dramatic improvement in her status, with her tachycardia, tachypnea, and abdominal pain improved. Her cefepime and oral vancomycin were discontinued, and she was transferred out of the ICU. She was discharged on POD 9, after completing her course of intravenous metronidazole. Her significant rectal prolapse and labial edema also delayed her discharge.

At her 1-week follow-up in clinic, the patient was doing well, and her labial edema and rectal prolapse resolved. The biopsy of the terminal ileum revealed attenuative villi, with reactive epithelium and acute inflammation. Three months later, she underwent colonoscopy, upper endoscopy, and ileoscopy, with biopsies that revealed chronic cecitis, chronic colitis continuous from the ascending colon to the sigmoid colon, and mild crypt architectural distortion in the rectum consistent with ulcerative colitis (Figs 4 and 5). She was initially treated with mesalamine but was changed to sulfasalazine because of associated pain. Her ileostomy was reversed 4 months after her initial operation.

C difficile is an anaerobic, Gram-positive, spore-forming, toxin-producing bacillus. Its colonization route is fecal-oral, and infections often present in the context of a disruption of the intestinal microbiome, such as antibiotics. The exotoxins produced by C difficile attack the intestinal epithelial cells, causing tissue damage and diarrhea.⁶  The incidence and severity of CDAD has been increasing, and there is more antimicrobial resistance.⁷  Risk factors for CDAD include hospitalization, antibiotic use, gastric acid suppression, and medical comorbidities, such as IBD, cystic fibrosis, and Hirschsprung disease.⁸  There is regional variation in the incidence of IBD in pediatric patients. The incidence is up to 32.6 per 100 000 in some areas.⁹ 

The current standard for surgical management of severe, complicated CDAD is subtotal colectomy, which has been traditionally used to remove accumulated toxins in addition to the C difficile organisms.⁴  However, CDAD is rarely associated with colonic necrosis; thus, there is no primary reason to remove the colon.^4,10  Additionally, subtotal colectomy is associated with high morbidity and mortality.⁴  Recently, diverting loop ileostomy with colonic lavage has been suggested as an alternative to colectomy in the adult population.^5,10 

In their 2011 study on diverting loop ileostomy and colonic lavage as surgical management of severe, complicated CDAD, Neal et al⁵  showed a statistically significant decrease in postoperative death when compared with colectomy in 84 adult patients with severe, complicated CDAD who met indications for operative management (Tables 2 and 3). A total of 8 of 42 patients treated with diverting ileostomy and colonic lavage died, whereas 21 of the 42 patients treated with subtotal colectomy died, which was statistically significant. The surviving patients were followed for 6 months, and 79% of the patients receiving diverting ileostomy and colonic lavage had their ileostomy reversed, as opposed to 19% of the patients treated with subtotal colectomy.⁵ 

Currently, the medical management of pediatric patients is based on the medical management of adult CDAD.⁸  Pediatric patients with mild CDAD are medically managed with oral metronidazole, whereas the treatment of more severe infections is varied but generally includes oral vancomycin.^3,8  Fidaxomicin is a newer antibiotic option that can also be used for CDAD and is gaining traction, particularly in the treatment of recurrent CDAD.⁸  Fecal microbiota transplants have also been shown to be effective.⁸ 

In 1996, Liacouras and Piccoli successfully used whole-bowel irrigation with PEG in 2 pediatric patients with refractory CDAD.¹¹  Both patients had frequent recurrences of CDAD and failed treatment with vancomycin, metronidazole, bacitracin, rifampin, cholestyramine, and lactobacillus. Neither patient met criteria for severe, complicated CDAD. After the PEG infusion, the patients were both treated with 3 weeks of vancomycin, and 1 patient was also treated with lactobacillus to assist gut microbiota recolonization. Both patients were asymptomatic within 3 days of treatment and did not experience CDAD recurrence.¹¹ 

Likewise, our pediatric patient, despite being critically ill and meeting the criteria for severe, complicated CDAD, responded quickly and recovered fully after colon-preserving loop ileostomy and antegrade irrigations. This appears to be the first report using this approach to treat a critically ill pediatric patient with severe, complicated CDAD. This novel approach in a pediatric patient allowed us to minimize the long-term consequences of CDAD by preserving her colon. Colonic preservation should be considered in this population because many patients with Crohn disease or ulcerative colitis do not progress to colectomy.¹²  The use of biological agents to medically treat IBD has been found to reduce the number of patients requiring colectomies.¹³ 

In the future, clinicians may want to consider evaluation of the rectal function and defecation before ileostomy take down, given the risk for peristalsis after a bacterial infection and operation. Because our patient had a brief course diverted, we did not think it was necessary in her case. Colonic motility and anorectal manometry may be considered in the assessment of proper function before ileostomy take down.

In conclusion, diverting loop ileostomy and antegrade colonic lavage in a pediatric patient was an effective and safe way to manage this patient’s severe, complicated CDAD without requiring a colectomy. There are benefits for children to avoid total abdominal colectomy both in the short and long-term. Additionally, loop ileostomy with antegrade lavage is a simpler, less morbid operation on a critically ill patient. To our knowledge, this is the first reported case in the literature of this surgical approach in a critically ill pediatric patient with severe, complicated CDAD. More research should be done on the management of CDAD in pediatric patients, including the surgical management and the long-term outcomes.

We acknowledge Elizabeth Witkowski, MILS, from the Medical College of Wisconsin Libraries for her assistance in creating search criteria for the literature review.

CDAD

Clostridium difficile–associated disease

ED

emergency department

HD

hospital day

IBD

inflammatory bowel disease

PEG

polyethylene glycol

POD

postoperative day

WBC

white blood cell count