Bromadiolone, commonly known as super warfarin, is a long-acting coumarin dicoumarin rodenticide. The mechanism of bromadiolone is mainly to inhibit vitamin K1 epoxide reductase and affect the synthesis of coagulation factors Ⅱ, Ⅶ, Ⅸ and Ⅹ, which causes blood coagulation dysfunction and systemic multiple organ hemorrhage. Here, we report of a case of bromadiolone poisoning patient who had digestive tract, abdominal hemorrhage, as well as secondary paralytic ileus. After blood product transfusion and vitamin K1 supplementation, the patient was discharged after the physical condition was improved. It's suggestied that clinicians should pay attention to rare complications to prevent missed diagnosis when treating other bromadiolone poisoning.
4-Hydroxycoumarins ; Blood Coagulation Factors ; Dicumarol ; Hemorrhage ; Humans ; Intestinal Pseudo-Obstruction/chemically induced* ; Oxidoreductases ; Rodenticides ; Vitamin K 1 ; Warfarin

BACKGROUND/AIMS: Previous studies from Korea have described chronic intestinal pseudo-obstruction (CIPO) patients with transition zone (TZ) in the colon. In this study, we evaluated the pathological characteristics and their association with long-term outcomes in Korean colonic pseudo-obstruction (CPO) patients with TZ. METHODS: We enrolled 39 CPO patients who were refractory to medical treatment and underwent colectomy between November 1989 and April 2016 (median age at symptoms onset: 45 [interquartile range, 29–57] years, males 46.2%). The TZ was defined as a colonic segment connecting a proximally dilated and distally non-dilated segment. Detailed pathologic analysis was performed. RESULTS: Among the 39 patients, 37 (94.9%) presented with TZ and 2 (5.1%) showed no definitive TZ. Median ganglion cell density in the TZ adjusted for the colonic circumference was significantly decreased compared to that in proximal dilated and distal non-dilated segments in TZ (+) patients (9.2 vs 254.3 and 150.5, P < 0.001). Among the TZ (+) patients, 6 showed additional pathologic findings including eosinophilic ganglionitis (n = 2), ulcers with combined cytomegalovirus infection (n = 2), diffuse ischemic changes (n = 1), and heterotropic myenteric plexus (n = 1). During follow-up (median, 61 months), 32 (82.1%) TZ (+) patients recovered without symptom recurrence after surgery. The presence of pathological features other than hypoganglionosis was an independent predictor of symptom recurrence after surgery (P = 0.046). CONCLUSIONS: Hypoganglionosis can be identified in the TZ of most Korean CPO patients. Detection of other pathological features in addition to TZ-associated hypoganglionosis was associated with poor post-operative outcomes.
Cell Count ; Colectomy ; Colon ; Colonic Pseudo-Obstruction ; Cytomegalovirus Infections ; Eosinophils ; Follow-Up Studies ; Ganglion Cysts ; Humans ; Intestinal Pseudo-Obstruction ; Korea ; Male ; Myenteric Plexus ; Pathology ; Recurrence ; Ulcer

Intestinal failure (IF) is the critical reduction of the gut mass or its function below the minimum needed to absorb nutrients and fluids required for adequate growth in children. Severe IF requires parenteral nutrition (PN). Pediatric IF is most commonly due to congenital or neonatal intestinal diseases or malformations divided into 3 groups: 1) reduced intestinal length and consequently reduced absorptive surface, such as in short bowel syndrome (SBS) or extensive aganglionosis; 2) abnormal development of the intestinal mucosa such as congenital diseases of enterocyte development; 3) extensive motility dysfunction such as chronic intestinal pseudo-obstruction syndromes. The leading cause of IF in childhood is the SBS. In clinical practice the degree of IF may be indirectly measured by the level of PN required for normal or catch up growth. Other indicators such as serum citrulline have not proven to be highly reliable prognostic factors in children. The last decades have allowed the development of highly sophisticated nutrient solutions consisting of optimal combinations of macronutrients and micronutrients as well as guidelines, promoting PN as a safe and efficient feeding technique. However, IF that requires long-term PN may be associated with various complications including infections, growth failure, metabolic disorders, and bone disease. IF Associated Liver Disease may be a limiting factor. However, changes in the global management of IF pediatric patients, especially since the setup of intestinal rehabilitation centres did change the prognosis thus limiting “nutritional failure” which is considered as a major indication for intestinal transplantation (ITx) or combined liver-ITx.
Bone Diseases ; Child ; Citrulline ; Enterocytes ; Humans ; Intestinal Diseases ; Intestinal Mucosa ; Intestinal Pseudo-Obstruction ; Liver Diseases ; Micronutrients ; Parenteral Nutrition ; Parenteral Nutrition, Home ; Prognosis ; Rehabilitation ; Short Bowel Syndrome

Waardenburg syndrome (WS) type IV is characterized by pigmentary abnormalities, deafness and Hirschsprung's disease. This syndrome can be triggered by dysregulation of the SOX10 gene, which belongs to the SOX (SRY-related high-mobility group-box) family of genes. We discuss the first known case of a SOX10 frameshift mutation variant defined as c.895delC causing WS type IV without Hirschsprung's disease. This female patient of unrelated Kuwaiti parents, who tested negative for cystic fibrosis and Hirschsprung's disease, was born with meconium ileus and malrotation and had multiple surgical complications likely due to chronic intestinal pseudo-obstruction. These complications included small intestinal necrosis requiring resection, development of a spontaneous fistula between the duodenum and jejunum after being left in discontinuity, and short gut syndrome. This case and previously reported cases demonstrate that SOX10 gene sequencing is a consideration in WS patients without aganglionosis but with intestinal dysfunction.
Cystic Fibrosis ; Deafness ; Duodenum ; Female ; Fistula ; Frameshift Mutation ; Hirschsprung Disease ; Humans ; Ileus ; Intestinal Pseudo-Obstruction ; Jejunum ; Meconium ; Necrosis ; Parents ; Waardenburg Syndrome

The mammalian intestine contains many different cell types but is comprised of 2 main cell types: epithelial cells and smooth muscle cells. Recent in vivo and in vitro evidence has revealed that various alterations to the DNA methylation apparatus within both of these cell types can result in a variety of cellular phenotypes including modified differentiation status, apoptosis, and uncontrolled growth. Methyl groups added to cytosines in regulatory genomic regions typically act to repress associated gene transcription. Aberrant DNA methylation patterns are often found in cells with abnormal growth/differentiation patterns, including those cells involved in burdensome intestinal pathologies including inflammatory bowel diseases and intestinal pseudo-obstructions. The altered methylation patterns being observed in various cell cultures and DNA methyltransferase knockout models indicate an influential connection between DNA methylation and gastrointestinal cells' development and their response to environmental signaling. As these modified DNA methylation levels are found in a number of pathological gastrointestinal conditions, further investigations into uncovering the causative nature, and controlled regulation, of this epigenetic modification is of great interest.
Apoptosis ; Cell Culture Techniques ; Cell Differentiation ; DNA Methylation ; DNA ; Epigenomics ; Epithelial Cells ; In Vitro Techniques ; Inflammatory Bowel Diseases ; Intestinal Mucosa ; Intestinal Pseudo-Obstruction ; Intestines ; Methylation ; Muscle, Smooth ; Myocytes, Smooth Muscle ; Pathology ; Phenotype

Pediatric chronic intestinal pseudo-obstruction is a rare disorder characterized by a severe impairment of gastrointestinal motility leading to intestinal obstruction symptoms in the absence of mechanical causes. The diagnosis is usually clinical and diagnostic work is usually aimed to rule out mechanical obstruction and to identify any underlying diseases. Treatment is challenging and requires a multidisciplinary effort. In this manuscript we describe the youngest child successfully treated with the orally administrable, long-acting, reversible anti-cholinesterase drug, pyridostigmine. Like other drugs belonging to cholinesterase inhibitors, pyridostigmine enhances gut motility by increasing acetylcholine availability in the enteric nervous system and neuro-muscular junctions. Based on the direct evidence from the reported case, we reviewed the current literature on the use of pyridostigmine in severe pediatric dysmotility focusing on intestinal pseudo-obstruction. The overall data emerged from the few published studies suggest that pyridostigmine is an effective and usually well tolerated therapeutic options for patients with intestinal pseudo-obstruction. More specifically, the main results obtained by pyridostigmine included marked reduction of abdominal distension, reduced need of parenteral nutrition, and improvement of oral feeding. The present case and review on pyridostigmine pave the way for eagerly awaited future randomized controlled studies testing the efficacy of cholinesterase inhibitors in pediatric severe gut dysmotility.
Acetylcholine ; Child ; Cholinesterase Inhibitors ; Diagnosis ; Enteric Nervous System ; Female ; Gastrointestinal Motility ; Humans ; Intestinal Obstruction ; Intestinal Pseudo-Obstruction ; Parenteral Nutrition ; Pyridostigmine Bromide

The mechanism of medication-induced gastrointestinal hypomotility is primarily caused by muscarinic cholinergic antagonism. This effect may cause constipation and paralytic ileus, which may lead to fatal complications. A 51-year-old woman was admitted due to manic episode recurrence. She developed paralytic ileus under quetiapine use and treated successfully under low dose amisulpride use. The related mechanism, associated risk factors, and the rationale for medication switch are discussed.
Bipolar Disorder ; Cholinergic Antagonists ; Constipation ; Female ; Humans ; Intestinal Pseudo-Obstruction* ; Middle Aged ; Quetiapine Fumarate* ; Recurrence ; Risk Factors

Guillain-Barré syndrome (GBS) is characterized by ascending symmetric paralysis, paresthesia, and autonomic dysfunction. Autonomic dysfunctions develop in two-thirds of the patients, and urinary retention and paralytic ileus usually develop in severe and advanced cases. There has been no pediatric case with paralytic ileus as a presenting symptom of GBS. Reported herein is a case of GBS presenting vomiting as an initial symptom. A 28-month-old girl was brought to the emergency room due to a 2-day history of vomiting. She vomited multiple times 1 day before the visit, and had only single voiding on admission day. Her abdomen was distended, with decreased bowel sound. Intravenous fluid was given under the diagnosis with acute gastroenteritis with dehydration. The abdominal computed tomography revealed severe paralytic ileus and urinary bladder distention. After having two seizures, she developed respiratory failure necessitating ventilator care. On day 8 after admission, motor weakness with areflexia was noticed. The cerebrospinal fluid analysis showed elevated proteins (80 mg/dL) with no white blood cells (0/mm³). The nerve conduction study showed axonal-type peripheral polyneuropathy. GBS was diagnosed. During the follow-up, the patient was found to have motor weakness in the lower extremities. Paralytic ileus with protracted vomiting can be an initial presentation of GBS in children. Autonomic dysfunction with GBS can be considered in a differential diagnosis in vomiting patients with unclear etiologies.
Abdomen ; Cerebrospinal Fluid ; Child* ; Child, Preschool ; Dehydration ; Diagnosis ; Diagnosis, Differential ; Emergency Service, Hospital ; Female ; Follow-Up Studies ; Gastroenteritis ; Guillain-Barre Syndrome* ; Humans ; Ileus ; Intestinal Pseudo-Obstruction* ; Leukocytes ; Lower Extremity ; Neural Conduction ; Paralysis ; Paresthesia ; Polyneuropathies ; Respiratory Insufficiency ; Seizures ; Urinary Bladder ; Urinary Retention ; Ventilators, Mechanical ; Vomiting

BACKGROUND/AIMS: Chronic intestinal pseudo-obstruction (CIPO) is a serious, life-threatening motility disorder that is often related to bacterial overgrowth. Fecal microbiota transplantation (FMT) results in restoration of the normal intestinal microbial community structure. We investigated the efficacy of FMT in the treatment of CIPO patients. METHODS: Nine patients (age 18–53 years) with CIPO were enrolled in this prospective, open-label study. Patients received FMT for 6 consecutive days through nasojejunal (NJ) tubes and were followed up for 8 weeks after treatment. We evaluated the rate of clinical improvement and remission, feeding tolerance of enteral nutrition, and CT imaging scores of intestinal obstructions. Lactulose hydrogen breath tests were performed before FMT and 8 weeks after FMT to evaluate for the presence small intestinal bacterial overgrowth (SIBO). RESULTS: FMT significantly alleviated bloating symptoms, and symptoms of pain were relieved 2 weeks after FMT. Enteral nutrition administered through a NJ tube after FMT was well-tolerated by 66.7% (6/9) of patients. CT scores of intestinal obstructions were significantly reduced after FMT (P = 0.014). SIBO was eliminated in 71.0% (5/7) of patients. CONCLUSIONS: This pilot study demonstrated the safety of using FMT. FMT may relieve symptoms in selected patients with CIPO. FMT may also improve patient tolerance of enteral nutrition delivered via a NJ tube.
Breath Tests ; Enteral Nutrition ; Fecal Microbiota Transplantation* ; Humans ; Hydrogen ; Intestinal Obstruction ; Intestinal Pseudo-Obstruction* ; Lactulose ; Pilot Projects ; Prospective Studies

BACKGROUND/AIMS: Chronic intestinal pseudo-obstruction (CIPO) is a rare syndrome characterized by a failure of the propulsion of intraluminal contents and recurrent symptoms of partial bowel obstruction in the absence of mechanical obstruction. Regional variations of the intestinal compromise have been described. Intestinal manometry can indicate the pathophysiology and prognosis. Our objective is to establish the demographic and clinical characteristics of group Chilean patients and analyze the motility of the small intestine and its prognostic value. METHODS: Patients with symptoms of intestinal pseudo-obstruction with dilated bowel loops were included, in all of whom a manometry of the small intestine was performed using perfused catheters. RESULTS: Of the 64 patients included, 51 women (average age 41.5 ± 17.6 years), 54 primary and 10 secondary CIPO were included. Dilatation of the small intestine was the only finding in 38 patients; in the remaining, the compromise was associated with other segments, primarily the colon. Forty-nine patients underwent 65 surgeries, mainly exploratory laparotomies and colectomies. Intestinal manometry was performed on all patients; 4 “patterns” were observed: neuropathic (n = 26), myopathic (n = 3), mixed (n = 24), and a group without motor activity (n = 11). The most relevant findings were the complex migrating motor disorders and decreased frequency and propagation of contractions. The 9 patients who died had a severe myopathic compromise. CONCLUSIONS: In our series, isolated small bowel compromise was the most common disorder. Neuropathic motor compromise was observed in most of the patients. Mortality was associated with severe myopathic compromise.
Catheters ; Colectomy ; Colon ; Dilatation ; Female ; Humans ; Intestinal Obstruction ; Intestinal Pseudo-Obstruction* ; Intestine, Small ; Laparotomy ; Manometry ; Mortality ; Motor Activity ; Motor Disorders ; Prognosis