Yersinia enterocolitica in a Child with Intussusception

Abstract

Intussusception is a common cause of bowel obstruction in infants and young children. In all, 75-90% of the cases of ileocolic intussusception are idiopathic (1). The remainder have a pathological lead point, such as a hamartomatous polyp, lymphoma, Meckel's diverticulum, juvenile vascular malformation, duplication cyst, or bowel wall hematoma (2,3). A role for viral and bacterial enteric infection in idiopathic intussusception has been proposed. Patients with intussusception have been reported to shed adenovirus, rotavirus, enterovirus, herpes simplex, and human herpes 6 virus in their stools (4-10). The association of adenovirus and rotavirus has been addressed in several studies (4-8). Adenovirus was isolated in 39% of cases when pharyngeal and rectal swabs were obtained from patients with intussusception (4). When appendixes removed from children with intussusception were examined, adenovirus was found in a majority by the presence of typical intranuclear inclusions and viral DNA by in situ hybridization (7). Montgomery and Popek found intranuclear inclusions in 10 of 32 surgical specimens obtained from children with intussusception and lymphoid hyperplasia (11). Immunohistochemistry for adenovirus was positive in five of 16 specimens. Konno et al. found rotavirus in one-third of patients with intussusception (5); however, in a separate study, rotavirus was found in only two of 24 cases (8). Bacterial infection in intussusception has been described only sporadically (10-13). Salmonella typhimurium has been cultured in an adult with intussusception (13). Yersinia has been the only bacterial agent incriminated in children. Yersinia has been reported in three previous cases in the North American literature and seven previous cases worldwide (10-12,14,15). Of these cases, three in North America and five worldwide have specifically involved Yersinia enterocolitica. We report a case of intussusception occuring in association with a gastroenteritis-like illness in which Yersinia enterocolitica was ultimately cultured from the stools. CASE REPORT A 29-month-old infant boy was transferred to Vanderbilt University Medical Center with a 1-week history of vomiting, diarrhea, and abdominal pain. The patient was healthy until 7 days before transfer, when he developed abdominal pain and nonbloody diarrhea. Over the next 24 h, he had severe crampy abdominal pain and two episodes of nonbloody, nonbilious emesis. A contrast enema was obtained at an outlying hospital. Intussusception was considered unlikely, although reflux into the terminal ileum was not observed. He was admitted to the outlying hospital 5 days before transfer because of continued vomiting and diarrhea. Stool cultures were obtained on 3 successive days. He was noted to have blood-streaked stools and a temperature of 38.5°C. The serum sodium declined from 131 mmol/L to 122 mmol/L. Vomiting increased on the morning of transfer, and a nasogastric tube was placed to suction mucus. There was no history of travel away from home, no exposure to well water, and no recent antibiotic use. Both parents were healthy, but a sibling was admitted to the outlying hospital with vomiting and diarrhea. Her symptoms resolved, and she was discharged home after 48 h. The patient's temperature was 38.5°C, pulse was 131/min, respiration was 24 min, and blood pressure was 122/71 mm Hg. The patient was sleeping but was awakened intermittently by abdominal pain. The nasogastric tube drained bilious secretions. The abdomen was mildly distended, and bowel sounds were hypoactive. There was no abdominal tenderness, mass, or organomegaly. There was no stool, mass, or tenderness on rectal exam. A flat and upright abdominal film showed distended loops of small bowel with air-fluid levels (Fig. 1). The patient was taken for laparotomy, at which time a 2-cm ileocolic intussusception was manually reduced. Mesenteric adenitis was noted. There was no evidence of vascular compromise to the bowel. The appendix, which appeared grossly normal, was resected. The patient's postoperative course was unremarkable. Histological examination of the appendix showed lymphoid hyperplasia (Fig. 2). Brown and Hopps stain revealed gram-negative rods characteristic of Yersinia. Three stool cultures obtained 3, 4, and 5 days before transfer grew Yersinia enterocolitica. The patient was treated with trimethoprim/sulfamethoxazole. The patient has had no recurrence of symptoms. DISCUSSION Most cases of intussusception are idiopathic and arise in previously healthy children <2 years of age. In ≈5% of all cases of intussusception, a pathological lead point is identified; the majority are noted in children >5 years of age. Symptoms include colicky abdominal pain, vomiting, and, less commonly, rectal bleeding. Diarrhea is seen in 7-30% of cases (16,17). Inasmuch as these symptoms are similar to those found in patients with infectious gastroenteritis, several authors have investigated a potential association between infectious diseases and idiopathic intussusception (4-13,16). The case reported herein shows an association between an infectious agent, Yersinia enterocolitica, and intussusception. Three cases of intussusception associated with Yersinia enterocolitica have been described in the North American literature (10-12). In one of these three cases, the resected specimen had lymphoid hyperplasia and abundant gram-negative organisms morphologically compatible with Yersinia species. Antibodies to Yersinia enterocolitica were detected in the acute postoperative period (11). The clinical aspects of this case were not further discussed. In the remaining two cases, the clinical course was discussed in more detail (10,12). Burchfield et al. described the case of an 8-month-old infant with an illness similar to that of our patient. Fever and diarrhea developed 6 days before the onset of bloody diarrhea and bilious emesis. At laparotomy a 15-cm ileocolic intussusception and enlarged mesenteric lymph nodes were found. Preoperative stool culture grew Yersinia enterocolitica. The patient was treated with antimicrobials and had an uneventful postoperative course (12). Hervas et al. reported the case of an 8-year-old boy with a 4-week history of abdominal pain, weight loss, and normal-appearing stools. An abdominal radiograph showed air-fluid levels and a mass effect in the transverse colon. The stool culture grew Yersinia enterocolitica. At surgery an ileocecocolic intussusception and mesenteric lymphadenitis were found. Examination of the appendix and a mesenteric lymph node showed marked nonspecific follicular hyperplasia. Culture of the mesenteric lymph node yielded Yersinia enterocolitica of the same serotype, biotype, and isotype as the organism isolated from stool (10). An additional five cases of Yersinia enterocolitica associated with intussusception have been reported in the German and Italian literature (14,15). The propensity for Yersinia to affect the terminal ileum and the occasional report of an association with ileocolic intussusception (10-12) invite speculation that there may be a pathogenic role. The development of lymphoid hyperplasia during the course of Yersinia enterocolitica infection may give rise to a lead point in intussusception. Lymphoid hyperplasia is common in the ileocecal area. Peyer's patches are circumferentially distributed in this region, in contrast to a linear distribution elsewhere; hence, enlargement is of greater consequence (11). In the first year of life, the incidence of ileocolic intussusception is high, and lymphoid tissue is prominent. In the second year of life, the incidence of intussusception begins to decline, as does the lymphoid tissue in the terminal ileum and cecum (18). In our case, bowel was not resected; however, examination of the appendix showed lymphoid hyperplasia and gram-negative rods morphologically compatible with Yersinia. Presumably the patient was infected with Yersinia, which localized to the terminal ileum, led to lymphoid hyperplasia, and then served as a lead point for the intussusception. In support of observations in humans, an animal model has shown localization of Yersinia to the terminal ileum (19). Yersinia enterocolitica, isolated from the blood of a patient who acquired the infection from stream water, gave rise to disease of the gastrointestinal tract after oral infection in mice. The initial lesions occurred in the distal ileum and cecum. This report emphasizes an association between ileocolic intussusception and Yersinia enterocolitica enteritis. When the predominant signs and symptoms of bacterial enteritis in children evolve toward a clinical picture of intestinal obstruction, intussusception and Yersinia enterocolitica should be considered. This association is attractive from an etiological viewpoint, because of the propensity for Yersinia to affect the terminal ileum. If stool cultures are performed more regularly when an intussusception is found in the context of a diarrheal illness, more cases may be identified.FIG. 1.: Abdominal radiograph shows a paucity of gas and a few distended loops of small bowel with air-fluid levels.FIG. 2.: Appendix removed from patient shows lymphoid hyperplasia characteristic of Yersinia.