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• W2012032142 abstract "INTRODUCTION Spirochetes were first identified in colonic biopsies over 100 years ago and have since been found in patients of various ages, both asymptomatic and symptomatic (1). It has been suggested that colonization with these organisms is linked to a variety of nonspecific gastrointestinal complaints including, but not limited to, watery diarrhea, abdominal pain, and rectal bleeding. The possibility of their pathogenicity remains to be thoroughly investigated, so debate continues. It is clear that many patients are colonized and asymptomatic, and at least some symptomatic cases resolve without therapy. However, strong anecdotal evidence indicates that some patients have persistent symptoms that respond to therapy. Several reports (2,3) have demonstrated invasive infection by light and electron microscopy in symptomatic patients. Reports of colorectal spirochetosis in children are scarce. Three relatively recent studies suggest that these organisms may cause symptoms in children (3-5). We report a 9-year-old patient who presented with rectal bleeding and diarrhea and was found to have a heavily colonized colon, but whose symptoms resolved without therapy. The patient remained symptom free at 1 year follow-up. CASE REPORT A 9-year-old boy was referred to our clinic for a 3 to 4 month history of blood mixed in his stool. The blood was noticed by his mother after almost every bowel movement. The patient reported occasional episodes of diarrhea during that time. He described occasional tenesmus and frequent flatulence and bloating but denied pain. Three hemoccult tests performed by his primary pediatrician were positive, and three examinations of the stool for ova and parasite were negative. His pediatrician also documented a 2 pound weight loss in the 3 to 4 months before referral. Review of systems, medical history, and family history were unremarkable. He had not traveled outside the United States and had no pet reptiles. There was nothing in history or physical examination to suggest sexual abuse. Physical examination was completely normal. Complete blood count, prothrombin time, partial thromboplastin time, and erythrocyte sedimentation rate were normal. Colonoscopy revealed a normal-appearing terminal ileum, colon, and rectum. Biopsies of the cecum, ascending, transverse, descending colon, and rectum were obtained. The biopsies were fixed in 4% zinc formalin, processed overnight, embedded in paraffin blocks, sectioned at 3 to 4 μm thickness, and then stained with hematoxylin-eosin. Periodic Acid Schiff (PAS) and silver impregnation (Steiner) histochemical stains were performed on the biopsy from transverse colon. Microscopic review of biopsies from the cecum, ascending, transverse, descending colon, and rectum showed mild epithelial reactive changes. These included mild loss of goblet cell mucin, nuclear enlargement and hyperchromasia, and prominent nucleoli. There was no architectural distortion or active inflammation. Examination of the surface epithelium at high power showed a fuzzy basophilic fringe along the luminal aspect consistent with intestinal spirochetosis (Fig. 1). PAS and Steiner stains helped highlight the presence of spirochetes along the luminal surface (Figs. 2 and 3). The terminal ileum was unremarkable.Fig. 1: High-power examination showing the organisms adherent to the luminal surface as diffuse basophilic fringe (arrow). Hematoxylin-eosin, magnification ×600.Fig. 2: Spirochetes along the surface epithelium of transverse colon (arrow). Silver impregnation (Steiner stain), magnification ×600.Fig. 3: Spirochetes along the surface epithelium of transverse colon (arrow). Periodic Acid Schiff, magnification ×600.We elected not to initiate immediate therapy, having reviewed cases that resolved spontaneously and recognizing that the pathologic nature of these organisms had not been firmly established. One month after colonoscopy, the patient was symptom-free, rapid plasma reagin was nonreactive, and three stools were negative for occult blood. One year later, the patient remained symptom free. DISCUSSION A case report by Harland and Lee (6) describing spirochetes on the mucosal surface of a rectal biopsy taken from a homosexual man generated interest recently in these organisms. A heterogeneous group of spirochetes have been discovered in the appendix, colon, and rectum and suggested as pathogens including (but not limited to) Brachyspira aalborgi, Serpulina pilosicoli, and Borrelia eurygyrata. These spirochetes are genetically unrelated to nonintestinal spirochetes (e.g., Treponema species) (1), although their classification has not been definitively established (7). Although nontreponemal spirochetes are established agents of invasive disease and colitis in pigs and other animals, human spirochetes are believed to be related but distinct organisms (8). The prevalence of human colorectal spirochetes in developed countries varies from 2.5% to 16% (9,10) depending on the population sampled, with considerably higher prevalence in homosexual men and in developing countries (11,12). The mode of transmission is unclear. Oral-fecal contamination appears an obvious suggestion given the high prevalence in developing countries, but this has not been proven. The spirochetes appear on hematoxylin--eosin-stained sections as a densely packed basophilic band covering the colonic surface that may be highlighted by silver stains (13). Light and electron microscopy have demonstrated invasive disease (organisms found within epithelial cells, histiocytes, and Schwann cells) and reactive changes, including stunting of microvilli, in both children (3) and adults (2). There are reports of spirochetosis in asymptomatic adults (9,14), but reports of asymptomatic children are rare, perhaps reflecting that adults are more likely to undergo colonoscopy as a screening procedure. In at least some patients, mucosal colonization persists despite antibiotic therapy and resolution of symptoms (14). Colorectal spirochetosis has been reported in children from Australia (4), Scandinavia (3), the United Kingdom (7,15), the United States (5,13), and India (16). Three relatively recent retrospective analyses suggest that these organisms may cause disease in children. A Swedish study (3) of eight cases of spirochetosis in children 4 to 15 years old found the most common complaints to be rectal bleeding and persistent abdominal pain. A variety of antibiotic therapies were initiated including erythromycin, doxycycline, clarithromycin, amoxicillin, and metronidazole, and various combinations of these agents with or without the addition of omeprazole. In three of these patients, there was a correlation between treatment and the resolution of symptoms, four had partial resolution of symptoms, and one had no change in symptoms, although the organisms were found to be eradicated on light microscopic examination of rectal/colon biopsy after treatment with metronidazole. In all instances, treatment course was 7 to 10 days. A study of 14 cases of spirochetosis at Johns Hopkins Hospital (5) included four children (mean age 12 years), three of whom had abdominal pain, and one who had abdominal pain and rectal bleeding. Follow-up was available for only one child, who was treated with benzathine, after which symptoms resolved. In this series, the predominant pathogen was identified by way of polymerase chain reaction-based assays to be B. aalborgi. A study of four Australian children (4) ages 9 to 16 years, one of whom presented with vomiting, two with abdominal pain, and one with rectal bleeding and intermittent diarrhea, found a correlation between treatment and resolution of symptoms in all cases. Treatment in three of the four cases was with a combination of metronidazole and amoxicillin for 7 to 10 days and in one case with a week of metronidazole alone. In the one case for which histologic follow-up was available, all evidence of spirochetal colonization had vanished after treatment. These studies raise more questions than they answer about the relationship between colonization and disease, the necessity of treatment, and which antibiotic or combination of antibiotics should be chosen if treatment is initiated. Although the persistence of symptoms in a child colonized with colorectal spirochetes should not be ignored and may warrant a trial of antibiotics, our case points out that colonization does not mean that symptoms will persist without therapy even when reactive histologic changes are present. This report also suggests that a wide variety of nonspecific complaints may be linked to spirochetosis. Studies with long-term follow-up are needed to help determine whether similar symptoms recur years after spontaneous resolution and, indeed, whether colonization persists. Until the percentage of colonization in asymptomatic children can be more adequately assessed, it will be difficult to argue definitively that these organisms are more than harmless commensals. They may, however, become pathologic under certain as of yet ill-defined circumstances. Further investigation is warranted." @default.
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• W2012032142 title "Colorectal Spirochetosis in a Child with Rectal Bleeding: Case Report and Literature Review" @default.
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