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.

Eribulin induces tumor vascular remodeling through intussusceptive angiogenesis in a sarcoma xenograft model.

Eribulin is a novel microtubule inhibitor that, similar to other types of microtubule inhibitors, induces apoptosis by inhibiting the mitotic division of cells. Besides this direct effect on tumor cells, previous studies have shown that eribulin has the potential to induce tumor vascular remodeling in several different cancers; however, the mechanisms underlying this phenomenon remain unclear. In the present study, we aimed to elucidate whether eribulin is effective against synovial sarcoma, a relatively rare sarcoma that often affects adolescents and young adults, and to histologically investigate the microstructure of tumor vessels after the administration of eribulin. We found that eribulin exhibits potent antitumor activity against synovial sarcoma in a tumor xenograft model and that tumor vessels frequently have intervascular pillars, a hallmark of intussusceptive angiogenesis (IA), after the administration of eribulin. IA is a distinct form of angiogenesis that is involved in normal developmental processes as well as pathological conditions. Our data indicate that IA is potentially involved in eribulin-induced vascular remodeling and thereby suggest previously unacknowledged role of IA in regulating the tumor vasculature after eribulin administration.

A Case of Kawasaki Disease with Intussusception.

Kawasaki disease (KD) is a systemic vasculitis of unknown cause and is associated with various digestive disorders, although only a few cases of intussusception associated with KD have been reported. We describe a case of intussusception followed by KD in a 3-year-old boy. The patient was admitted to our hospital for evaluation of severe abdominal pain. Because the target sign was seen on ultrasonography, intussusception was diagnosed and hydrostatic reduction was performed. On the second day after admission, he developed a high fever (38°C) and an irregular rash over his whole body. On the fourth day after admission, the high fever continued, and bilateral nonexudative conjunctivitis, erythema of the lips and oral mucosa, strawberry tongue, indurated edema of the dorsa of the hands and feet, and diffuse erythema of the palms and soles appeared, and KD was ultimately diagnosed. He was treated with intravenous immunoglobulin 2 g/kg, aspirin 30 mg/kg/day, and prednisolone 2 mg/kg/day. The high fever and other clinical symptoms resolved immediately after the start of treatment. There was no relapse of KD symptoms after initial treatment, and periungual desquamation was observed on the 10th day after admission. He was discharged on the 15th day, without abnormalities such as coronary dilatation, 3 months after the onset of KD symptoms. Patients with intussusception and KD were older (≥3 years vs <3 years) than those with intussusception alone. In addition, the site of intussusception in KD was mainly colonic rather than ileocolic. If intussusception precedes development of the characteristic clinical symptoms of KD, diagnosis of KD may be delayed. KD should be considered in children older than 3 years with intussusception at a colonic site.

Emergency Physician-Administered Sedation for Pneumatic Reduction of Ileocolic Intussusception in Children: A Case Series.

BACKGROUND: Ileocolic intussusception is a major cause for intestinal obstruction in early childhood. Reduction of intussusception, in the vast majority of institutions, is performed on awake children, without sedation. OBJECTIVE: The aim of this study was to report the course of the first patients who were sedated by emergency physicians for pneumatic reduction of intussusception (PRI). METHODS: We conducted a case series analysis of prospectively collected data on patients undergoing PRI, using a sedation recording tool. This tool uses standardized definitions from the Quebec guidelines for terminology and reporting adverse events in emergency medicine. Recording of time interval measurements and adverse events was performed by the emergency physician and nurse. RESULTS: Between February 2016 and March 2018, 11 males and 3 females, with a median age of 11 months (interquartile range [IQR] 6-20 months), underwent sedation for PRI by five attending pediatric emergency physicians. All of the reductions were successful and without complications. Eight patients received ketamine and propofol, 5 received ketamine, and 1 received ketamine and midazolam. Median times for the presedation phase, procedure, sedation, physiologic recovery and emergency department recovery were: 131 min (IQR 79-104 min), 10.5 min (IQR, 9-12 min), 21 min (IQR, 20-30 min), 25 min (IQR, 23-30 min), and 108 min (IQR, 82-161 min), respectively. Four respiratory adverse events that required intervention were recorded. All were effectively treated with airway repositioning, suctioning, oxygen administration, or bag-mask ventilation. CONCLUSIONS: The first series of patients sedated for PRI by emergency physicians is reported. Our initial findings suggest the feasibility of emergency physician-administered sedation for PRI.

An unusual presentation of metastatic malignant melanoma causing jejuno-jejunal intussusception: a case report.

BACKGROUND: Small bowel intussusception in adults is rarely encountered. In most cases small bowel intussusception is caused by benign neoplastic lesions, but metastasis of cutaneous malignant melanoma causing small bowel intussusception is rare. We present such a case of jejuno-jejunal intussusception with an intraluminal metastatic lesion acting as a lead point. CASE PRESENTATION: We present a case of a 71-year-old Caucasian man who presented with small bowel obstruction. His medical history revealed that he had had a cutaneous malignant melanoma excised 7 years earlier and underwent total laryngectomy due to a metastasis 6 years later. The disease was classified as stage IV and he was receiving immunotherapy. An emergency abdominal computed tomography scan demonstrated small bowel obstruction, most probably caused by an intraluminal lesion. An emergency laparotomy revealed an intraluminal metastatic lesion causing jejuno-jejunal intussusception. Metastasectomy of the lesion was performed and 13 days later he was discharged. CONCLUSIONS: Jejuno-jejunal intussusception with a malignant melanoma metastasis acting as a lead point is very rare. With the gastrointestinal tract being a common location of distal metastases, a medical history of malignant melanoma treatment in cases of small bowel obstruction should raise a suspicion of possible metastatic disease. A computed tomography scan is the diagnostic modality of choice and surgery still remains the standard of care.

The aquarium in the right heart.

We presented a case with massive hepatic portovenous gas (HPVG) and gastric emphysema, probably due to increased intraluminal pressure in the stomach after bagging and noninvasive ventilation. There are multiple microbubbles in the inferior vena cava, right atrium and right ventricle. There has been only one case report ever published showing the similar features of the "aquarium sign" in the right heart in a patient with intussusception. We believe our case is a good illustration of this extremely rare entity in echocardiography.

Intussusception merged with systemic lupus erythematosus: one case report and retrospective analysis.

Intussusception is characterized by one segment of the gastrointestinal tract telescoping into the lumen of the adjacent segment; it is rarely reported in systemic lupus erythematosus (SLE), and the condition can be threatening. Only four cases of intussusception with SLE have been reported in literature. Here, we describe a new case of a patient with ileocecal intussusception merged with SLE, who was diagnosed using abdominal computed tomography and successfully treated with high-dose intravenous immunoglobulin (IVIG) and pulse methylprednisolone.

The role of dexamethasone in decreasing early recurrence of acute intussusception in children: A retrospective study.

BACKGROUND: Early recurrent intussusception (RI) is a concern after a successful pneumatic reduction. Steroids have been suggested as a treatment that decreases early RI. The purpose of this study was to examine the role of dexamethasone in decreasing early RI. METHODS: A retrospective review of 174 pediatric patients that underwent successful pneumatic reduction was conducted. Two groups were identified: group 1 that received intravenous dexamethasone (0.5mg/kg/dose) on diagnosis or immediately after the reduction maneuver, and group 2 that were not given dexamethasone. RESULTS: There were 113 boys and 61 girls ranging in age from 2 to 36months. There were no statistical differences between the groups except for younger age in the dexamethasone group (P=0.03). There was no significant difference (p=0.08) in the rate of early RI between the non-steroid group (5.4%, 4/74) and the steroid group 14% (14/100). Mean admission length was 30h. The majority of RIs occurred in the first 8h of admission. CONCLUSIONS: We found that premedication of children with idiopathic intussusception with dexamethasone did not decrease early RI. LEVEL OF EVIDENCE: Level III retrospective comparative study.

The Effect of Midazolam on Decreasing the Duration of Intussusception Hydrostatic Reduction in Children.

BACKGROUND AND OBJECTIVE: The use of sedative medications may be effective in the success and facilitation of the intussusception non-surgical treatment. Therefore, the purpose of this study was to examine the effect of midazolam on decreasing the duration of intussusception hydrostatic reduction in children. MATERIALS AND METHODS: In a double-blind clinical trial, 32 children were diagnosed with ileocolic intussusception based on sonographic findings, were studied and randomly divided into two groups. After obtaining written informed consent from the parents, 5 minutes before reduction, an intravenous Midazolam at the concentration of 1.0 mg/kg (up to 3 mg) was infused, and then barium reduction was performed under fluoroscopy guideline. In the control group, sterile water was injected as placebo and the remaining reduction steps were performed compared with the experiment group. RESULTS: Of 16 patients that received Midazolam, 15 patients demonstrated successful reduction; and of 16 patients that received distilled water, only 11 patients showed successful reduction (P=0.07). The mean duration of a successful reduction in the Midazolam group and placebo was 34.8±11.35 and 32.73±19.2 min, respectively (P=0.733). CONCLUSIONS: The use of Midazolam as a benzodiazepine with known sedative and muscle relaxant effects can increase the success rate of enema reduction in intussusception.

Reducing Antibiotic Use for Young Children with Intussusception following Successful Air Enema Reduction.

China introduced a new policy regarding the management of antibiotic use. We evaluated the reasonableness of antibiotic use among children suffering from intussusception before and after policy. A retrospective study was conducted involving 234 young children with intussusception who were treated between January 1, 2011 and December 30, 2013. Demographics and detailed antibiotics regimens were collected. χ2 test was used to evaluate differences between the phase I (preintervention, n = 68) and phase II (postintervention, n = 166). We determined that the overall antibiotic use rate following successful air enema reduction was 41% (97/234), which decreased from 99% (67/68) in phase I to 18% (30/166) in phase II. In phase I, prophylactic antibiotic usage reached up to 84% (56/67). The quantity of aztreonam for injection accounted for 63% (45/71), and cefamandole nafate for injection accounted for 25% (18/71). In phases II, prophylactic antibiotic usage were reduced to 13% (4/30). The quantity of aztreonam for injection was decreased to 12% (4/33) and cefamandole nafate for injection was 3% (1/33). Antibiotics' options were more diverse. In conclusion, policy intervention was effective in addressing some aspects of antibacterial drug usage among young children with intussusception. However, excessive drug use remains a public health problem. The guidelines for the antibiotic management of intussusception for children must be established in China.

A jejunal GIST presenting with obscure gastrointestinal bleeding and small bowel obstruction secondary to intussusception.

A 68-year-old man with episodes of overt obscure gastrointestinal (GI) bleeding was investigated with multiple upper and lower GI endoscopies, CT enterography and capsule endoscopy, but no cause was found. He then presented acutely with small bowel obstruction. A laparotomy revealed complete small bowel obstruction secondary to jejunal intussusception over a 4 cm intraluminal polyp. Following resection and primary anastomosis, histology revealed that the polyp was a GI stromal tumour (GIST). This is an exceptionally uncommon presentation of a rare tumour. It is surprising that this tumour was not detected by CT enterography and not seen on capsule endoscopy. Immunohistochemistry and mutation analysis of the GIST suggested that it had a low risk of metastatic disease, but a high risk of recurrence. Staging CT scans did not reveal evidence of distal spread. The patient is currently receiving 3 years of chemotherapy with imatinib.

Intussusception in gestational choriocarcinoma (not histologically proven), resolving spontaneously with chemotherapy.

We present a rare case of advanced gestational choriocarcinoma with small bowel metastatic involvement and intussusception, which presented acutely as a lower gastrointestinal bleed with symptomatic anaemia and haemoglobin 3.8 g/dL in a young woman. A diagnosis of gestational choriocarcinoma was made without biopsy, using a combination of clinical history, isolated elevated human chorionic gonadotropin markers of 77,000 IU/mL and radiological findings. Surgical intervention was too high risk due to the presence of active bleeding and increased vascularity surrounding the intussusception. Owing to the highly responsive nature of gestational choriocarcinoma to chemotherapy, frontline chemotherapy alone was used to reduce the size of the metastatic small bowel deposits, with subsequent resolution of the bleeding and intussusception. This is the first time chemotherapy alone has been used to successfully resolve small bowel intussusception secondary to metastatic choriocarcinoma that has been documented according to PubMed searches.

Management of abdominal pain in children.

Abdominal pain (AP) is a very common complaint caused by a variety of conditions. Mild or moderate AP affects practically all children of all ages. The pain usually settles spontaneously without medical intervention. AP severe enough to require medical intervention has both surgical and non-surgical causes. It is responsible for considerable morbidity, missed school days, and significant use of health resources. Children usually present either with an acute or recurrent AP. In comparison, chronic AP with persistent symptoms, lasting days or weeks, is rare in children. Surgical conditions may be the underlying causes in acute AP, but non-surgical conditions are diagnosed more commonly in children with recurrent AP. Management can be difficult, time-consuming and often clinically challenging to diagnose and treat. In most instances, the cause of AP can be diagnosed through the history and physical examination. The main objective in managing an affected child is to differentiate between benign, self-limited conditions such as constipation or gastroenteritis, and more life-threatening surgical conditions such as intussusception or appendicitis. Irritable bowel syndrome (IBS) and Crohn's disease should be considered in any child presenting with recurrent AP.

Indications for repeated enema reduction of intussusception in children.

INTRODUCTION: Intussusception is a common abdominal emergency in early childhood. It is idiopathic in more than 90% of cases with incidence of 1.5-4 per 1,000 live births. The treatment of choice is nonoperative hydrostatic or air enema reduction. OBJECTIVE: The aim of the study was to evaluate the influence of clinical presentation and symptom duration in non-operative treatment, considering the indications for delayed enema reduction and its efficacy. METHODS: From the total number of 107 patients with intusussception, aged from 2 months to 14 years (median 9 months), 102 (95%) patients with ileo-colic intussusceptions were treated initially by ultrasound guided saline enema. Records were reviewed for patients with failed initial treatment and delayed repeated enemas or operative procedure. The predictor variable included duration of presenting symptoms. RESULTS: Successful treatment by hydrostatic saline enemas had 58/102 (57%) patients. Success in reduction was greater it symptom duration was < 24 hours (54/62 cases; 87%, p < 0.001), compared with > 24 hours, (4/45 cases; 9%). Despite failed initial attempts, enema reduction was reattempted in 12 patients, with success in 7/12 (60%) patients. Children with symptom duration > 24 hours had a greater risk of requiring surgery (41/45 cases; 91%, p < 0.001), including 5 (5%) patients with ileo-ileal intussusceptions. CONCLUSION: The accuracy of ultrasound guided saline enema in intussusception reduction is high. Delay in presentation decreases success of non-operative treatment. Delayed enema reduction is important therapeutic option for intussusceptions. Surgical treatment is indicated in cases of complications.

Diagnosis and management of pediatric appendicitis, intussusception, and Meckel diverticulum.

Three of the most common causes of surgical abdominal pain in pediatric patients include appendicitis, Meckel diverticulum, and intussusception. All 3 can present with right lower quadrant pain, and can lead to significant morbidity and even mortality. Although ultrasound is the preferred method of diagnosis with appendicitis and intussusception, considerable variety exists in the modalities needed in the diagnosis of Meckel diverticulum. This article discusses the pathways to diagnosis, the modes of treatment, and the continued areas of controversy.

The role of steroids in children with multiple recurrences of intussusception.

We describe the case of a boy who had 9 recurrences of intussusception, for which no pathological lesion at the leadpoint was identified. A contrast follow-through study revealed a follicular/nodular mucosal pattern, particularly prominent in the terminal ileum and caecum. Patients with multiple recurrences usually have an identifiable lesion at the leadpoint, but sometimes recurrences may be due to lymphoid hyperplasia, as presumed in our case. No further recurrences occurred after a two month tapering course of oral prednisolone.