Case Report: Gastrointestinal Basidiobolomycosis with Multi-Organ Involvement Presented with Intussusception.

Gastrointestinal basidiobolomycosis (GIB) is a rare, life-threatening fungal infection affecting immunocompetent individuals in tropical and subtropical regions. A diverse presentation of GIB has been reported, but no report has yet been published on intussusception. We describe a 23-month-old immunocompetent boy from a subtropical area in Iran who presented with intussusception. Prolonged fever, an abdominal mass, hepatomegaly, high erythrocyte sedimentation rate, and peripheral eosinophilia strongly suggested GIB. Accordingly, GIB was diagnosed based on the characteristic histopathology (the Splendore-Hoeppli phenomenon) detected in a liver sample taken via biopsy. Exploratory laparotomy showed several organs, including the colon, gall bladder, liver, and abdominal wall, were involved. Antifungal therapy with trimethoprim/sulfamethoxazole, liposomal amphotericin B, a saturated solution of potassium iodide, and surgical resection of involved tissues were used with improved outcome. The presence of non-septate fungal hyphal elements in the colonic mucosa led to the thickening of the bowel wall, leading to secondary intussusception.

Concurrent Campylobacter jejuni bacteremia and intussusception in an immunocompetent five-year-old child.

Bacterial enteritis has been reported to be a risk factor for childhood intussusception. However, no case report concerning children with concurrent Campylobactor bacteremia and intussusception were found in the literature. Herein, we report a 5-year-old male preschool child who presented symptoms of infectious diarrhea, and ileocecal type intussuscepton and Campylobactor jejuni bacteremia were found after a series of investigations.

First Reported Case of Intussusception Caused by Escherichia coli O157:H7 in an Adult: Literature Review and Case Report.

BACKGROUND: Intussusception is the process by which one segment of intestine "telescopes" into another segment. Escherichia coli O157:H7 is a rare cause of intussusception that uncommonly requires a surgical procedure. METHODS: Case report and literature review. RESULTS: We reviewed 25 cases of infection with E. coli O157:H7 that resulted in intussusception, all of which involved minors. Our case identifies the first reported adult with intussusception secondary to E. coli infection necessitating surgical intervention. In total, two (8%) required operation. Hemolytic uremic syndrome did not develop in any patient, and there were no deaths. CONCLUSIONS: E. coli O157:H7-associated intussusception is rare and does not commonly require operation. If conservative management fails, a surgical procedure may be necessary to resect the pathologic lead point.

[A rare case of suppurative mesenteric adenitis associated with intussusception in a child: a clinical case]. / Un cas rare d'adénite mésentérique suppurée associée à une invagination intestinale chez un enfant: cas Clinique.

Some cases of suppurative mesenteric adenitis have already been described in the literature but not associated with intussusception. We describe the case of a 3-year-old boy presenting to the department of surgery at the University Hospital of Lubumbashi with bowel obstruction. He was visited elsewhere, in the previous 12 days, for diarrhea, vomiting, fever, coma and treated for cerebral malaria and blackwater fever. Surgery revealed an ileal intussusception and a suppurative mesenteric adenitis whose pyoculture revealed the presence of Enterobacter cloacae, sensitive to norfloxacin. We performed desinvagination, sucked the pus out into a syringe and excized completely the site of suppurative adenitis. The evolution of patient was good. The clinician must know that the association between suppurative mesenteric adenitis and intussusceptions exists. The diagnosis is not easy and there is the risk of developing acute peritonitis due to its fistulation in the abdominal cavity.

Invaginación intestinal en un lactante con diagnóstico de botulismo: Reporte de un caso / Intussusception in infant with diagnostic botulism: A case report

El botulismo es un importante problema de salud pública en Argentina. Es una enfermedad potencialmente letal y de difícil diagnóstico. Existen casos de presentación infrecuente de dicha enfermedad, como el abdomen agudo. Exponemos el caso de un niño de 4 meses, que consultó por constipación de 3 días de evolución, asociada a decaimiento y regular actitud alimentaria de 12 horas de evolución. Presentaba tono muscular conservado, sin alteraciones en la succión ni deglución, según la referencia materna. Se constató sensorio alternante y abdomen agudo, por lo que ingresó a quirófano con sospecha de invaginación intestinal, la cual fue confirmada mediante desinvaginación neumática. Durante la internación, el paciente evolucionó desfavorablemente y presentó llanto débil, hipotonía progresiva e insuficiencia respiratoria, por lo que requirió cuidados intensivos. Se aisló Clostridium botulinum en la muestra de materia fecal y toxina botulínica tipo A en el suero. Recibió toxina antibotulínica equina como tratamiento, con recuperación total a los 25 días de haber ingresado.

Botulism is an important public health problem in Argentina. It is a potentially fatal disease, and its diagnosis may be difficult. There are rare presentation forms of the disease, such as acute abdomen. We present a 4-monthbaby with a 3-day constipation condition, associated with weakness and abnormal eating attitude in the last 12 hours. The baby presented preserved muscle tone, with no changes in sucking or deglutition according to the mother's observations. Altered sensorium and acute abdomen were found; the patient was entered into the operating room with presumptive diagnosis ofintussusception, which was confirmed by pneumatic desinvagination. During hospitalization, the patient did not make good progress and presented weak cry, progressive hypotonia and respiratory failure requiring intensive care. Clostridium botulinum was isolated from the stool sample and botulinum toxin type A was isolated from serum. The patient was treated with equine botulinum toxin. Twenty five days after admission, he was totally recovered.

[Intussusception in infant with diagnostic botulism: A case report]. / Invaginación intestinal en un lactante con diagnóstico de botulismo: Reporte de un caso.

Botulism is an important public health problem in Argentina. It is a potentially fatal disease, and its diagnosis may be difficult. There are rare presentation forms of the disease, such as acute abdomen. We present a 4-month baby with a 3-day constipation condition, associated with weakness and abnormal eating attitude in the last 12 hours. The baby presented preserved muscle tone, with no changes in sucking or deglutition according to the mother's observations. Altered sensorium and acute abdomen were found; the patient was entered into the operating room with presumptive diagnosis of intussusception, which was confirmed by pneumatic desinvagination. During hospitalization, the patient did not make good progress and presented weak cry, progressive hypotonia and respiratory failure requiring intensive care. Clostridium botulinum was isolated from the stool sample and botulinum toxin type A was isolated from serum. The patient was treated with equine botulinum toxin. Twenty five days after admission, he was totally recovered.

[Purulent pericarditis and colonic infiltrating to Salmonella enteritidis complicated by acute intussusception in a case of IL-12Rß1 deficiency]. / Péricardite purulente et infiltration colique à Salmonella enteritidis compliquée d'invagination intestinale aiguë dans un cas de déficit en IL-12Rß1.

IL-12 receptor ß1 deficiency (IL-12Rß1) predisposes patients to mycobacteria and Salmonella infections. We report a case of IL-12Rß1 deficiency with a fatal multi-resistant Salmonella enteritidis infection. This boy was born after from a consanguineous marriage, and diagnosed as having a IL-12Rß1 deficiency since the age of 3 months. He presented with recurrent Salmonella enteritidis essentially digestive localization, complicated by purulent pericarditis at the same germ at the age of two and a half years. At the age of 3, a colonic infiltration due to a Salmonella enteritidis resistant to antibiotics, was complicated by acute intussusception, and the child died. The IL-12Rß1 deficiency is considered as having a good prognosis, in contrast to what happened in our patient. We review therapeutic issues in these patients.

[Recurrent small-bowel intussusceptions revealing a disseminated BCG infection related to severe combined immunodeficiency]. / Invagination intestinale récidivante révélatrice d'une bécégite disséminée liée à un déficit immunitaire combiné sévère.

Infant small-bowel intussusceptions, most of the time idiopathic, may exceptionally reveal a severe digestive disease. We report the case of a 4-month-old infant who presented multiple and simultaneous ileal intussusceptions associated with severe acute gastroenteritis. Initially, the infant showed a protein-losing enteropathy with a clear alteration of the general state of health and undocumented fever, resistant to broad-spectrum antibiotic therapy. Skin and splenic nodules associated with hepatosplenomegaly and pancytopenia set in progressively. The etiologic evaluation led to the diagnosis of a Mycobacterium bovis BCG infection related to severe combined immune deficiency. The treatment consisted in anti-tuberculosis quadruple therapy in association with immunoglobin supplementation. Secondarily, the patient underwent gene therapy in a clinical trial. An early BCG vaccine in the first weeks of life, before the outbreak of infection revealing the immune deficiency, is a risk factor in triggering a disseminated BCG infection in immunodepressed infants. This clinical case is the first reported of severe combined immune deficiency revealed by small-bowel intussusceptions related to a disseminated BCG infection.

[Infectious etiology of acute idiopathic intussusception in children]. / Les étiologies infectieuses des invaginations intestinales aiguës idiopathiques chez l'enfant.

The intussusception (IIA) is an invagination of the immediate part of the intestine. She is responsible for a syndrome with an occlusive venous compression and swelling that can rapidly progress to intestinal necrosis. Most cases occur in children aged 6 to 18 months and occur more frequently in boys than girls. There are two types of IIA: the IIA idiopathic representing 90-95% of invaginations of the child and the IIA secondary to local injury of the gastrointestinal tract or occurring in a particular context whose frequency are between 5 and 10%. The pathogenesis of the IIA remains uncertain, but the infectious origin is criminalized in most idiopathic invaginations. This component is dominated by viral agents including adenovirus, rotavirus, enterovirus, human herpesvirus 6 and 7, cytomegalovirus and Epstein-Barrvirus. Bacterial agents are rather found and include Yersinia enterocolitica, Staphylococcus aureus, Escherichia coli O157, H7, Salmonella and Campylobacter. In a small proportion parasitic agents may be reported in the IIA, the most frequently found are Entamoeba histolytica, Trichuris trichuira, Ascaris lumbricoides, Ankylostoma and Giardia.

[CMV-enterocolitis as a cause for repeated intestinal intussusceptions in an adult patient after liver transplantation?]. / CMV-Enterokolitis bei einer erwachsenen lebertransplantierten Patientin als Ursache rezidivierender Invaginationen?

Intestinal intussusception in the adult is often idiopathic but also known to be associated with chronic inflammatory bowel disease, coeliac disease, tumours or previous abdominal operations. A 22-year-old women after liver transplantation due to Crigler Najar Syndrome suffered from repeated episodes of abdominal pain. The diagnosis of repeated self-limited intestinal intussusceptions was made by computed tomography and ultrasonography. A laparoscopy revealed no cause for the intussusceptions. During a new episode of abdominal pain caused again by an intussusception a colonoscopy was performed that showed aspects of a discreet colitis. In the biopsies CMV was detected by qualitative PCR, while blood tests for CMV pp65 antigen were negative. A therapy with gancyclovir was initiated which lead to remission of the patient's symptoms. A colonoscopy six weeks later showed a completely normal colon, while in the biopsies CMV was not detectable. After a follow-up of one year the patient has not suffered from any further episodes. This case demonstrates the role of chronic intestinal CMV infection as a possible causative factor for repeated intussusceptions in immunosuppressed patients. Whenever possible a PCR for CMV in colon biopsies should be carried out to detect an intestinal CMV infection because as shown in our case results for immunohistopathology and CMV pp65 can be negative despite a chronic infection.

Abdominal tuberculosis presenting as ileocolic intussusception in an infant.

Intussusception is the most common cause of intestinal obstruction in children, with a peak in children 5 to 7 months of age. Identifiable causes are found in 90% of adults, whereas in infants and young children the majority are idiopathic. We report a case of abdominal tuberculosis (ATB) presenting as an ileocolic intussusception in an infant. A 6-month-old infant presented with features of acute intestinal obstruction. Peroperatively, ileocolic intussusception was found. Histological examination revealed caseating epitheloid cell granulomas with positivity for acid-fast bacilli. Only 3 cases of ATB presenting as intussusception have been previously reported in the literature, with only 1 case presenting in a child. This appears to be the 2nd case of ATB presenting as an intussusception in a child and also in an infant. Tuberculosis should also be kept in the differential diagnosis of lead point of intussusception, even in the age group in which most cases are idiopathic, especially in endemic areas.

Intussusception due to Yersinia enterocolitica enterocolitis in a patient with beta-thalassemia.

Patients who are homozygous for thalassemia major are at risk for Yersinia enterocolitica infections. We present a case of a 4-year-old child with intussusception of the terminal ileum whose past medical history was significant for beta-thalassemia. His monthly blood transfusions for this condition may have put him at risk for Y enterocolitica enterocolitis. The pathogenesis of this disease relates to the role of iron as an essential growth factor for Yersinia, and this patient's transfusions left him in an iron-overloaded state, despite treatment with Desferal. Our patient's unusual presentation of intussusception was secondary to the mass effect caused by lymphoid hyperplasia, specifically hypertrophied Peyer's patches in the ileum caused by Y enterocolitica infection. To our knowledge, this is the first such case of intussusception caused by Yersinia to be reported.

Long-standing painless intussusception in adults.

Long-standing painless intussusception in adults is considered to be rare. We report three such cases with an emphasis on color Doppler results. In our three cases the indication for abdominal US was a palpable mass in all these cases, and intussusception was detected by US at a time when the patients had only very mild abdominal discomfort. Persistence of sufficient blood flow, as was suggested by the color Doppler results, was thought to be the most likely pathomechanism of long-standing painless intussusception. The underlying disease was tuberculosis in two of the three cases. Thus, when encountering patients with painless intussusception, tuberculosis must be kept in mind.

Bacterial wall lipopolysaccharide as a cause of intussusception in mice.

BACKGROUND: There is evidence that intussusception is associated with bacterial infection. It was hypothesized that a component of the bacterial wall may induce the intussusception. This study was intended to determine whether lipopolysaccharide from Escherichia coli or Salmonella can initiate intussusception in mice. METHODS: Lipopolysaccharide was injected intraperitoneally in mice, and the animals were examined for the presence of intussusception from 2 to 192 hours after injection. Gastrointestinal transit was assessed by measuring the passage of charcoal in the small intestine. Transit index was defined as the ratio between the distance traveled by charcoal and the total length of the small intestine. RESULTS: Intussusceptions were found in as much as 25.9% of lipopolysaccharide-injected animals, whereas in control animals, the incidence was zero. The threshold for the lipopolysaccharide effect was at 4 mg/kg and incidence reached a plateau at 8 mg/kg to 16 mg/kg. The incidence of intussusception peaked 6 hours after injection of lipopolysaccharide and declined to zero after 15 hours. To test the possibility that lipopolysaccharide induces intussusception by altering motility, its effect on transit index was measured. A dose of 12 mg/kg lipopolysaccharide reduced the transit index from 56.2+/-1.4% to 37.7+/-2.1% (p < 0.05). No microscopic histologic changes were found in the bowels with intussusception. CONCLUSIONS: Intraperitoneal bacterial wall lipopolysaccharide causes intussusception in mice by disturbing gastrointestinal motility.

Intussusception associated with Yersinia pseudotuberculosis infection.

Intussusception associated with Yersinia pseudotuberculosis infection was developed in three boys; two of them had a history of drinking untreated water. All intussusceptions were localized at the ileocolic region, and all patients completely recovered with Gastrografin enema and supportive treatment without complication and operation.

[Yersinia enterocolitica: a cause of acute intestinal intussusception]. / Yersinia enterocolitica: une cause d'invagination intestinale aiguë.

OBJECTIVES: Yersinia enterocolitica infection is a rare cause of intestinal intussusception, especially in adults. We report here a case in a 29-year-old man and review the literature on diagnosis and therapy. CASE REPORT: A 29-year-old man presented with a 2-week history of diarrhea and weight loss. Ultrasonography revealed acute intestinal intussusception localized at the site of enlarged mesenteric nodes. At laparostomy, intestinal resection was not required. Histology examination of the mesenteric nodes showed follicular hyperplasia. Serology was positive for Yersinia enterocolitica. Outcome was favorable after treatment with tetracycline for 15 days. DISCUSSION: Yersinia enterocolitica are Gram negative bacilli that grow at low temperature. Food contamination is the most frequent source of infection in man, usually in children. Clinical manifestations include gastroenteritis or pseudoappendicular syndrome. Intestinal intussusception is rare. Operative reduction by taxis is generally sufficient. Histology examination of the lymph nodes excludes lymphoma. The diagnosis is confirmed by serology. A 10 to 15-day antibiotic regimen is needed.

[Isolation of verotoxin-producing Escherichia coli from pediatric patients suffering from enteritis with bloody stool and intussusception].

We tried to isolate verotoxin-producing Escherichia coli (VTEC) on sorbitol-MacConkey (SMAC) agar and in part by the polymerase chain reaction (PCR) method from sporadic enteritis patients with bloody stools and intussusception patients who came to three pediatric clinics in the Fukuoka area from October 1990 to September 1994. VTEC O157:H7 strains were isolated from 6 (10.5%) of 57 patients with bloody stools, Campylobacter spp. 15 (26.3%), Salmonella spp. 14 (24.6%) and Yersinia enterocolitica 2. We were not able to detect VT genes by PCR from 11 of 20 patients from whose stools no causative bacteria were isolated. Massive fresh bloody stools following frequent watery diarrhea were typical of the VTEC enteritis patients. Only 1 patient had fever and 2 had leukocytosis, but the C-reactive protein in all of them was below 1+. The VTEC strains were isolated during the summer season, 1 in June, 2 in July, and 3 in September. Since in the area O157:H7 appeared to be the most prevalent VTEC serotype, SMAC is very useful for screening VTEC in bloody stools. VTEC seems to be a rare pathogen of intussusception because the organisms were detected from none of the 30 patients.