Human intestinal spirochetosis, characterized by end-on attachment of densely packed spirochetes to the epithelial surface of the large intestines as a fringe has been associated with the weakly beta-hemolytic spirochetes Brachyspira aalborgi and Brachyspira (Serpulina) pilosicoli. In this study, fluorescent in situ hybridization with oligonucleotide probes targeting 16S or 23S rRNA of B. aalborgi, B. pilosicoli, and the genus Brachyspira was applied to 40 sections of formalin-fixed, paraffin-embedded intestinal biopsy specimens from 23 Danish and 15 Norwegian patients with histologic evidence of intestinal spirochetosis. Five biopsy specimens from patients without intestinal spirochetosis and three samples from pigs with experimental B. pilosicoli colitis were examined as well. In addition, the 16S ribosomal DNAs of two clinical isolates of B. aalborgi were sequenced, and a PCR procedure was developed for the identification of B. aalborgi in cultures. The genotypic characteristics of the two clinical isolates showed very high (99.5%) similarity with two existing isolates, the type strain of B. aalborgi and a Swedish isolate. Hybridization with the Brachyspira genus-specific probe revealed a brightly fluorescing fringe of spirochetes on the epithelia of 39 biopsy specimens, whereas 1 biopsy specimen was hybridization negative. The spirochetes in biopsy specimens from 13 Danish and 8 Norwegian patients (55.3%) were identified as B. aalborgi. The spirochetes in the biopsy specimens from the other 17 patients hybridized only with the Brachyspira probe, possibly demonstrating the involvement of as-yet-uncharacterized Brachyspira spirochetes in human intestinal spirochetosis.
Human intestinal spirochetosis (IS) is a condition defined histologically by the presence of spirochetal microorganisms attached to the apical cell membrane of the colorectal epithelium. Intestinal spirochetes comprise a heterogeneous group of bacteria. In humans, Brachyspira aalborgi and Brachyspira pilosicoli predominate. Prevalence rates of IS are low where living standards are high, in contrast to poorly developed areas where IS is common. Homosexuals and HIV-infected individuals are at high risk of being colonized. Clinical significance in individual cases has remained unclear up to now. A review of the literature assumes that invasion of spirochetes beyond the surface epithelium may be associated with gastrointestinal symptoms which respond to antibiotic treatment (metronidazole), whereas individuals lacking this feature may be mostly asymptomatic. Of unknown reason, homosexual and HIV-positive men as well as children are more likely to be symptomatic irrespective of invasion. Rare cases of spirochetemia and multiple organ failure have been reported in critically ill patients with IS.
human intestinal spirochetosis; microscopic colitis; commensals; intestinal bacterial invasion; HIV-infection; spirochetemia
Brachyspira pilosicoli is an etiological agent of human intestinal spirochetosis. Bloodstream infection due to this microorganism is rare. We report a case of B. pilosicoli bacteremia in a 70-year-old patient who presented with multiorgan failure.
Twenty-nine intestinal spirochetes isolated from Australian aboriginal children and six strains from Italian adults (HRM1, -2, -4, -5, -7, and -14) were genetically examined at 15 enzyme loci by using multilocus enzyme electrophoresis. Results were compared with those previously obtained for 188 porcine intestinal spirochetes. DNA from human strain HRM7 and porcine strain Serpulina hyodysenteriae P18A were also radioactively labeled and hybridized with DNA from 12 other human and porcine intestinal spirochetes. Both the multilocus enzyme electrophoresis and hybridization techniques demonstrated that the human spirochetes were not S. hyodysenteriae. They belonged to another distinct genetic group of spirochetes that included P43/6/78, the bacterium recovered from the first recorded case of porcine intestinal spirochetosis. Bacteria in this distinct group also differed from Serpulina spp. in possessing only four, five, or occasionally six axial filaments, being slightly thinner, and having more pointed ends. These findings add further weight to the possibility that human intestinal spirochetes may act as enteric pathogens.
Human intestinal spirochetosis (HIS) is associated with overgrowth of the large intestine by spirochetes of the genus Brachyspira. The microbiological diagnosis of HIS is hampered by the fastidious nature and slow growth of Brachyspira spp. In clinical practice, HIS is diagnosed histopathologically, and a significant portion of cases may be missed. Fluorescence in situ hybridization (FISH) is a molecular method that allows the visualization and identification of single bacteria within tissue sections. In this study, we analyzed intestinal biopsy samples from five patients with possible HIS. All specimens yielded positive results by histopathological techniques. PCR amplification and sequencing of the 16S rRNA gene were performed. Sequences of two isolates clustered in the group of Brachyspira aalborgi, whereas in three cases, the sequences were highly similar to that of Brachyspira pilosicoli. Three phylotypes showed mismatches at distinct nucleotide positions with Brachyspira sp. sequences published previously. In addition, culture for Brachyspira was successful in three cases. On the basis of these data, we designed and evaluated a Brachyspira genus-specific 16S rRNA-directed FISH probe that detects all of the Brachyspira spp. published to date. FISH of biopsy samples resulted in strong, unequivocal signals of brush-like formations at the crypt surfaces. This technique allowed simultaneous visualization of single spirochetes and their identification as Brachyspira spp. In conclusion, FISH provides a fast and accurate technique for the visualization and identification of intestinal spirochetes in tissue sections. It therefore represents a valuable tool for routine diagnosis of HIS.
Acute appendicitis is a frequent pathology encountered by general surgeons. On the differential of aetiologies that can lead to this diagnosis lies the spectrum of infectious disease. We present a case report of a patient who presented to our institution as a classic acute appendicitis. Upon histological analysis it became evident that he had spirochetosis of the cecum and appendix. This paper reviews the literature on this rare infectious aetiology. A synopsis of the microbiology, epidemiology, clinical manifestations and treatments for intestinal spirochetosis is described. At large, the aim of this paper is to heighten awareness of this disease amongst clinicians and specifically surgeons.
The development of intramural duodenal haematoma (IDH) after small bowel biopsy is an unusual lesion and has only been reported in 18 children. Coagulopathy, thrombocytopenia and some special features of duodenal anatomy, e.g. relatively fixed position in the retroperitoneum and numerous submucosal blood vessels, have been suggested as a cause for IDH. The typical clinical presentation of IDH is severe abdominal pain and vomiting due to duodenal obstruction. In addition, it is often associated with pancreatitis and cholestasis. Diagnosis is confirmed using imaging techniques such as ultrasound, magnetic resonance imaging or computed tomography and upper intestinal series. Once diagnosis is confirmed and intestinal perforation excluded, conservative treatment with nasogastric tube and parenteral nutrition is sufficient. We present a case of massive IDH following endoscopic grasp forceps biopsy in a 5-year-old girl without bleeding disorder or other risk for IDH, which caused duodenal obstruction and mild pancreatitis and resolved within 2 weeks of conservative management. Since duodenal biopsies have become the common way to evaluate children or adults for suspected enteropathy, the occurrence of this complication is likely to increase. In conclusion, the review of the literature points out the risk for IDH especially in children with a history of bone marrow transplantation or leukaemia.
Intramural duodenal haematoma; Endoscopic biopsy; Children; Bone marrow transplantation; Leukaemia
Lipoma is relatively common in the colon but is less often in the small intestine. Most lipomas are incidentally detected at endoscopy and are usually small and asymptomatic. However, some of them can present with obstruction and/or intussusceptions. Surgical resection is commonly recommended to remove such significant lipomas with a limited pedicle and larger than 2 cm in size, as endoscopic resection may result in unfavorable complications such as intestinal perforations. We report a case of 62-year-old man presenting with hematochezia. Colonoscopy showed a submucosal tumor, about 50 mm in size, in the terminal ileum. A clinical diagnosis of lipoma was established based on the findings of colonoscopy and abdominal computed tomography (CT). As the patient complained of hematochezia and mild iron deficiency anemia associated with repeated tumor prolapse, we decided to remove his lipoma. Consequently, the lesion was completely removed en bloc. Although abdominal CT immediately after removal of the lesion showed a small amount of free air, conservative treatment was successfully carried out for the perforation. Histologically, the removed lesion was a lipoma.
Lipoma; Endoscopic submucosal dissection; Endoscopic unroofing; Perforation
Foreign body ingestion is associated with a variety of symptoms and complications, often mimicking various diseases. This case report describes an unusual presentation following foreign body ingestion.
A 56-year-old Greek Caucasian woman presented to a primary care setting, in rural Crete, Greece, with complaints of abdominal pain, cramping and bloating, for the last four months. Alternating constipation and diarrhea was reported. The patient had unknowingly ingested a foreign body that resulted in an irritable bowel syndrome-like presentation.
This case report emphasizes the need for a high index of suspicion from physicians for a wide differential in their approach to abdominal complaints, as well as the importance of an individualized approach to patients in the setting of clinical medicine.
In this study we report on the isolation and characterization of the intestinal spirochete Brachyspira aalborgi using human mucosal biopsy specimens taken from the colon of a young adult male with intestinal spirochetosis. A selective medium, containing 400 μg of spectinomycin/ml and 5 μg of polymyxin/ml was used for the isolation procedure. A high degree of similarity, in terms of phenotypic properties and 16S ribosomal DNA sequence, was observed between the isolated strain, named W1, and the type strain, 513A, of B. aalborgi. A similarity of 99.7% in the nucleotide sequence was found between W1 and 513AT, based on the almost-complete gene. A short segment of the 16S rRNA gene was amplified by PCR using genetic material enriched from paraffin-embedded biopsy specimens, which were taken from the patient on two occasions. The products showed 16S rRNA gene sequences virtually identical to that of strain 513AT in the actual region. Immunohistochemistry was performed on the colonic biopsy specimens with a polyclonal antibody raised against an intestinal spirochete isolated in a previous case of human intestinal spirochetosis. The antibody reacted strongly with the spirochete on the luminal epithelium. No immune reaction was seen within or below the surface epithelium. Routine histology did not reveal signs of colitis. Electron microscopy showed spirochetes attached end-on to the colonic mucosal surface. The isolate grew poorly on a commonly used selective medium for intestinal spirochetes, which may explain previous failures to isolate B. aalborgi.
Brachyspira pilosicoli is the etiologic agent of human and animal intestinal spirochetosis and is rarely implicated as a cause of bacteremia. Here, we describe the case of a B. pilosicoli spirochetemia in a 53-year-old male patient suffering from cardiogenic shock. This fastidious bacterium was isolated from blood, likely after translocation from the intestinal tract. Blood cultures were positive after 5 days of incubation (one day after the patient's death), highlighting the problem of the recovery of such type of fastidious bacterium. Identification was achieved by molecular methods (16S rRNA sequencing). A review of the English literature found only 8 cases of bacteremia caused by B. pilosicoli, mostly in immunocompromised or critically ill patients. Finally, difficulties in rapid and accurate diagnosis of B. pilosicoli bloodstream infections, in vitro antimicrobial susceptibility of human clinical isolates, and therapeutic options are discussed.
PCR procedures amplifying portions of the 16S rRNA and NADH oxidase genes of Brachyspira aalborgi and Serpulina pilosicoli were applied to DNA extracted from paraffin-embedded human colonic or rectal tissues from 30 Norwegian, Australian, and U.S. patients, 16 of whom had histologic evidence of intestinal spirochetosis (IS). B. aalborgi-specific sequences were identified by PCR in 10 of the IS patients (62.5%) but none of the others, while S. pilosicoli sequences were not detected in tissues from any patient. Direct sequencing of products from three of the positive samples provided further confirmation of the presence of B. aalborgi. B. aalborgi may be a more common cause of intestinal spirochetosis than has been previously thought.
DNA from gastrointestinal biopsy specimens from 28 Australian patients with histologic evidence of intestinal spirochetosis (IS) was subjected to PCRs to amplify segments of the 16S rRNA and NADH oxidase genes of Brachyspira aalborgi and Brachyspira (Serpulina) pilosicoli. B. aalborgi was identified in specimens from 24 (85.7%) patients and B. pilosicoli in those from 4 (14.3%) patients (2 of whom were also positive for B. aalborgi). For two patients, no product was amplified. This study demonstrates that B. aalborgi is much more commonly involved in histologically identified IS in Australian patients than is B. pilosicoli. This is the first report of amplification of B. pilosicoli DNA from humans with IS.
The ultrastructure of spirochetes obtained from rectal biopsies of patients with intestinal spirochetosis was studied by means of negative staining and ultrathin sectioning. The cells were sigmoidal with tapered ends, 2 to 6 microns long, with a wavelength of 2 microns. Four flagella were inserted at each end of the cells. The maximal cell width was about 0.2 microns. The spirochetes were cultured on tryptose soy blood agar plates. They were anaerobic and grew, although very slowly, at 37 to 38.5 degrees C in an atmosphere of 5% CO2-95% H2. Two types of colonies could be distinguished. The growth characteristics and the morphology of the isolated spirochetes differ from those of previously isolated spirochetal strains. Consequently, it is proposed that the present strains constitute a new genus, Brachyspira, of the family Treponemataceae. The type species is Brachyspira aalborgi, the type strain of which is 513A (NCTC 11492).
One-day-old chicks were infected orally with two strains of weakly hemolytic spirochetes isolated from a human and a pig with intestinal spirochetosis. These spirochetosis both colonized birds, attached end-on to their cecal enterocytes, induced watery diarrhea, and significantly depressed growth rates. Cultures of Serpulina innocens failed to colonize the chicks.
Colo-duodenal fistula is a rare complication of malignant and inflammatory bowel disease. Cases with malignant colo-duodenal fistulae can present with symptoms from the primary, from the fistula or from metastatic disease. The fistula often results in diarrhoea and vomiting with dramatic weight loss. Upper abdominal pain is usually present as is general malaise both from the presence of the disease and from the metabolic sequelae it causes. The diarrhoea relates to colonic bacterial contamination of the upper intestines rather than to a pure mechanical effect. Vomiting may be faeculant or truly faecal and eructation foul smelling but in the case reports this 'classic' symptomatology was often absent despite a fistula being present and patent enough to allow barium through it. Occasionally patients will present with a gastro-intestinal bleed.
We present an unusual case of colorectal carcinoma, where a 65 year old male patient presented with diarrhoea and vomiting secondary to a malignant colo-duodenal fistula near the hepatic flexure. Adenocarcinoma was confirmed on histology from a biopsy obtained during the patient's oesophageogastroduodenoscopy, and the fistula was demonstrated in his barium enema. Staging computed tomography showed a locally advanced carcinoma of the proximal transverse colon, with a fistula to the duodenum and regional lymphadenopathy. The patient was also found to have subcutaneous metastasis. Following discussions at the multidisciplinary meeting, this patient was referred for palliation, and died within 4 months after discharge from hospital.
We present the case, discuss the management and review the literature. Colo-duodenal fistulae from colonic primaries are rare but early diagnosis may allow curative surgery. This case emphasises the importance of accurate staging and repeated clinical examination.
We report the first known case of a retroperitoneal leiomyosarcoma that presented with an endoscopically defined source of gastrointestinal bleeding in the colon. A 68-year-old male with a history of diverticulosis, hypertension, and hypercholesterolemia who complained of a 3-month history of abdominal pain, nausea, and intermittent hematochezia presented for evaluation of large volume hematochezia and lightheadedness. Colonoscopy revealed left-sided diverticulosis and rectal varices without stigmata of recent bleed. CT scan showed a 26 × 20 × 13 cm heterogeneous retroperitoneal mass and multiple hypodense hepatic lesions. Liver biopsy revealed leiomyosarcoma. In summary, although surgery is the mainstay of treatment, resectability has not improved significantly. Early recognition and aggressive surgery are keys to long-term survival.
The motor patterns and luminal capacity of the human intestine should affect symptoms and resorption during pathological, massive small intestinal flow. Little is known of human intestinal motility in this situation. This study aimed at mimicking secretory diarrhoea (experimentally) in healthy volunteers by intrajejunal infusion of a non-absorbable iso-osmotic solution at 20 ml/min. During the infusion intraluminal jejunal pressures and small intestinal transit times were measured. The infusion initially caused jejunal contractile activity similar to that of the fed state but this was replaced by discrete clusters of contractions (DCCs) after 29.1 ((SEM) 8.2) minutes. DCCs each lasted 38 ((SEM) 0.8 seconds) and were associated with colicky abdominal discomfort. Later, after 1400-1800 ml had been infused, distal jejunal pressure waves fell to 10 mm Hg or less. Frequent fasting DCCs predicted earlier onset and more frequent DCCs during the infusion. Thus, the rate and volume of flow during simulated secretory diarrhoea determine the pattern of the small bowel pressure profile; eventually, a volume load is reached in which the small bowel acts as a poorly segmenting conduit resulting in very fast transit rates.
The association between Cryptosporidium, chronic diarrhoea and a proximal small intestinal mucosal enteropathy was reviewed over a six and a half year period. One hundred and twenty three children with cryptosporidiosis and no clinical evidence of immune deficiency were identified. 50% of children excreting only Cryptosporidium had chronic diarrhoea. Most cases (63%) of chronic diarrhoea occurred in the first two years of life. A mild to moderate enteropathy was present in all nine children undergoing a small intestinal biopsy and seven showed the presence of Cryptosporidium adhering to villous epithelium. All patients eventually recovered spontaneously. Cryptosporidium is a cause of chronic diarrhoea and a proximal small intestinal mucosal enteropathy in children without immune deficiency. Screening for the parasite should be part of the investigative procedures in children with chronic diarrhoea.
In the great majority of patients treated with radiation, only transitory injury to the bowel occurs, but in five percent of patients, permanent damage to the small bowel or rectum is seen. Symptoms of radiation enteropathy may begin four to six months after the treatment is completed or may not present until several years later. Most often, the patient presents with abdominal pain, diarrhea, hematochezia, and signs of malnutrition. Others may present, initially, with intestinal obstruction, perforation, or fistulization. It is important to differentiate this clinical syndrome from recurrent cancer by appropriate radiological studies and biopsies.
This paper presents four patients who were treated with radiation for invasive carcinoma of the cervix and subsequently developed radiation enteritis. All were treated surgically and are surviving.
Leukemic and lymphomatous involvement of the appendix is rare and even rarer is its presentation as appendicitis. Burkitt’s lymphoma is a high grade B-cell neoplasm. Its non-endemic form typically presents as abdominal mass in children. This rapidly growing tumour may cause symptoms due to mass effect or direct involvement of the bowel. Clinical presentations like acute abdomen can be secondary to intestinal obstruction, intussusception or sometimes perforation.
We describe here a case of an adult male with an unusual presentation of appendiceal Burkitt’s lymphoma mimicking acute cholecystitis or appendicitis.
Lymphoma; Appendicitis; Cholecystitis; Burkitt’s lymphoma; Appendix; CT
Lipomas are the most common benign mesenchymal tumors of the gastrointestinal tract, with the colon being the most prevalent site. Intestinal lipomas are usually asymptomatic. Tumors >2 cm in diameter may occasionally cause nonspecific symptoms, including change in bowel habits, abdominal pain, or rectal bleeding, but with resection the prognosis is excellent. Herein, we describe the case of an elderly male who presented with painless hematochezia.
Both colonoscopy and computed tomography of the abdomen and pelvis confirmed the presence of a mass near the ileocecal valve. Because of continuing bleeding, the patient required laparoscopic-assisted right hemicolectomy to resect the mass.
Both gross and microscopic pathology were consistent with lipoma at the ileocecal valve.
Previous cases of ileocecal valve lipomas have been reported in the English literature, with the majority presenting as intussusception or volvulus. We present a rare case of an ulcerated ileocecal valve lipoma presenting as lower gastrointestinal bleeding that was treated successfully with laparoscopic resection.
Lipoma; Ileocecal valve; Rectal bleeding
We report a case of the intestinal lesion in Henoch-Schönlein purpura, presented with an acute abdomen in a 4 year old boy. Five days after sudden colicky abdominal pain, skin purpura and painful joint swelling developed. These manifestations were associated with abdominal distension, hematemesis, hematochezia and hematuria. Exploratory laparotomy revealed a marked bowel distension with edema and patchy dark reddish discoloration of the jejunum and ileum. These patchy areas showed transmural hemorrhage and necrosis associated with characteristic leukocytoclastic vasculitis in and around the hemorrhagic lesions. These vasculitis was thought to be related to Henoch-Schönlein purpura.
Peptic ulcer disease is uncommon in children and rarely suspected as a cause of abdominal complaints in this age group; the diagnosis is therefore made almost exclusively when complications develop. Peptic ulcer disease is usually not considered in the differential diagnosis of pediatric patients. We present the case of a 30-month-old boy with duodenal perforation due to a peptic ulcer without a known etiology. The patient was admitted through the emergency department due to severe hematochezia and ongoing anemia; he presented with neither abdominal pain nor abdominal distension. There were no medical problems, and no drugs, such as corticosteroids or nonsteroidal anti-inflammatory drugs, had been prescribed or administered recently. We tried to control the active bleeding by medical treatment including arterial embolization, but the active bleeding was not controlled. Finally, an exploratory laparotomy was performed. A discrete anterior perforation with active bleeding of the duodenal wall was found. After the operation, there were no complications and the patient recovered fully.
Duodenal ulcer; Peptic ulcer perforation; Children; Hematochezia; Hemorrhage
The diagnosis of hepatocellular carcinoma (HCC) became easier in relation to the improved radiological examinations; however, the neoplasm may occur under atypical presentations mimicking other benign or malignant processes. Multicystic HCC mimicking a liver abscess associated with septic-type fever and leukocytosis is rare, has a poor prognosis, and poses diagnostic and therapeutic dilemmas. We present the case of an 80-year-old patient, who presented with fever, leukocytosis, and large cystic masses involving right and left lobes of the liver initially considered abscesses and finally diagnosed as HCC after open drainage and liver biopsy. Although the patient died on the tenth postoperative day due to pulmonary oedema, the authors emphasize the high index of suspicion needed in the diagnosis of this unusual presentation of HCC.