Granuloma inguinale (donovanosis) presenting as a neck mass in an infant

A case of granuloma inguinale (GI) presenting as a lateral neck mass in a 4-month-old, HIV-positive infant is described. The histological features of the mass were typical of GI, with numerous macrophages containing intracellular organisms with a “closed-safety-pin” appearance. This is a rare occurrence, and the mode of transmission of infection is discussed. An awareness of GI in infants by both clinicians and pathologists is important to prevent morbidity and allow for prompt institution of appropriate treatment. Accepted: 24 March 1998     

宫内生长迟缓对新生幼鼠胃肠发育的影响

为了解宫内生长迟缓（ＩＵＧＲ）新生儿胃肠发育状况，分别采用妊娠第１（Ｓ１）、第１７（Ｓ１７）日母鼠饥饿饲养，直至分娩以及正常妊娠第１７日子宫动脉结扎（ＵＡＬ）建立ＩＵＧＲ模型，运用生物体视学技术对新生幼鼠的胃肠组织切片定量分析。结果显示，小于胎龄（ＳＧＡ）新生幼鼠胃肠重量、长度及组织结构多项指标明显低于正常新生幼鼠；Ｓ１组胃肠发育受损程度轻于Ｓ１７组，两组小肠粘膜吸收面积无显著性差异；ＵＡＬ组胃肠重量、胃肠粘膜体积、小肠粘膜吸收面积低于Ｓ１７组；直线相关分析发现，ＳＧＡ新生幼鼠胃、肠重量与体重呈正相关，相关系数分别为０．６９及０．７３；而小肠长度以及胃肠相对重量与身长呈正相关，相关系数分别为０．６４及０．６８。提示：（１）ＩＵＧＲ对胎鼠胃肠发育产生了不良影响；（２）作为推测ＳＧＡ新生幼鼠胃肠发育状况的指标，身长优于体重；（３）ＩＵＧＲ发生时期、原因不同的ＳＧＡ新生幼鼠，可以出现不同的胃肠发育损害。     

A case of proximal jejunal ectopic pancreas causing sporadic vomiting

Aberrant rests of pancreatic tissue can be found throughout the gastrointestinal system and are known as pancreatic heterotopia or ectopic pancreas (EP). Authors report a 12-year-old girl with jejunal EP with a long-lasting history of sporadic bilious vomiting. Upper gastrointestinal (GI) study showed delayed passage beyond duodeno-jejunal junction. During laparotomy a 2x2 cm mass was encountered on the mesenteric border of the jejunum, 3 cm distal to the ligament of Treitz. Histopathologic examination revealed pancreatic tissue. The mass was excised and end-to-end anastomosis was performed. Postoperative course of the patient was uneventful and she is doing well after 10 months. Intestinal obstruction due to EP has been reported to occur only if it causes intussusception. Intestinal obstruction without intussusception due to jejunal EP has not been reported. In our case, the EP tissue was located just beneath the mucosa and involved the muscular layer. The foreign body effect of the EP tissue involving the muscular layer may cause dysmotility and/or local spasm, which we think were responsible for the long-lasting sporadic bilious vomiting in our patient.     

Enteral administration of hematopoietic growth factors in the neonatal intensive care unit

GI trophic factors have been identified that influence the prenatal and postnatal growth and development of the GI tract. Systemically as well as enterally administered growth factors can stimulate GI growth and maturation, suggesting that trophic factors in the serum of neonates can modulate GI growth via receptors on the serosal membranes of enterocytes. GI trophic factors can be synthesized endogenously or provided prenatally in amniotic fluid and postnatally in human milk.
GI trophic factors in human milk play an important role in regulating the adaptive functional changes that accompany the transition to postnatal enteral feedings. Although human milk growth factors are not essential for infant survival, the elevated risk of GI-related illnesses in formula-fed compared with human milk-fed infants suggests that bioactive compounds in human milk contribute to the protective effects of human milk feedings (13).
GI trophic factors have the potential to be used therapeutically to enhance GI maturation and repair following injury. These applications may be particularly useful in the premature or postsurgical infant. Several issues require further research, including: (i) the efficacy of oral versus systemic administration; (ii) characterization of the complex interactions among the various growth factors; (iii) the effect of exogenously administered growth factors on endogenous production of that factor, its receptor or other growth factors; (iv) the effect of growth factors upon tissues not directly associated with the GI tract; and (v) the determination of safe and effective limits. Significant advances in feeding strategies to reduce feeding intolerance in the neonate are likely to occur with the application of these principles in clinical neonatology.     

Non‐invasive biological quantification of acute gastrointestinal graft‐versus‐host disease in children by plasma citrulline

Clinical grading of GI involvement during acute GVHD remains a challenging issue, especially in children. Plasma citrulline, a non‐protein amino acid selectively produced and released by enterocytes, is a suitable surrogate endpoint for small intestinal epithelial cell mass, irrespective of the underlying cause of cell loss. Children referred for allogeneic bone marrow transplantation who were free from chronic malabsorption or constitutional disease involving the GI tract were consecutively included in this prospective study. Plasma citrulline and albumin concentration was measured every week between day 7 and day 28 of BMT until resolution of the aGVHD or occurrence of chronic GVHD. In total, 31 children were included between 2008 and 2011. After a CR, citrulline levels fell to a minimum level on day 7 and then increased to reach the initial value on day 28. After day 28, plasma citrulline but not albumin was strongly linked to the occurrence of GI GVHD, the threshold being set at 10 μmol/L. The correlation with clinical grade of GI‐aGVHD now needs to be assessed in larger populations. In pediatric patients, citrulline is valuable as a suitable non‐invasive marker of GI involvement in acute GVHD.     

Delayed presentation of congenital diaphragmatic hernia

The late-presenting congenital diaphragmatic hernia (CDH) represents a considerable diagnostic challenge. This study was undertaken to define various patterns of delayed presentation and to analyze pitfalls in the diagnosis and treatment of these patients. Thirty-three children with CDH were treated between 1993 and 2000; 15 of these (45.5%) who were diagnosed after the age of 2 months-14 years, median of 2.5 years are reported. Thirteen had a Bochdalek hernia and 2 had a Morgagni hernia. The diaphragmatic defect was right-sided in 6 cases and left-sided in 9. Five patients presented acutely, 3 with respiratory distress and 2 with gastrointestinal (GI) obstruction. The remaining 10 presented with chronic respiratory or GI complaints. Inappropriate insertion of a chest drain occurred in 3 patients misdiagnosed as having pleural effusion (2) and a pneumothorax (1). Two patients had previous chest radiographs reported normal. Plain radiographs were sufficient to make a definitive diagnosis in only 6 patients; GI contrast studies were necessary in the other 9. All patients were treated through an abdominal approach with primary closure of the diaphragmatic defect without a patch. A distinct hernia sac was present in 6 cases, and associated malrotation in 6. All except 1 patient survived the operation with rapid improvement of their GI and respiratory symptoms. We conclude that: (1) late-presenting CDH should be included in the differential diagnosis of any child with persistent GI or respiratory problems associated with an abnormal chest X-ray film; (2) nasogastric tube placement must be considered as an early diagnostic or therapeutic intervention when the diagnosis is suspected; and (3) GI contrast studies should be a part of the diagnostic work-up of these patients.     

Gastrointestinal stromal tumour of the duodenum in a 7-year-old boy

We report a 7-year-old boy presenting with an acute upper gastrointestinal (GI) haemorrhage subsequently diagnosed to have a very rare duodenal gastrointestinal stromal tumour (GIST). Endoscopy, pertechnetate and red cell scans were negative. Abdominal US detected a 17-mm mass lesion of the third part of the duodenum. This was confirmed on CT and shown to be hypervascular on selective angiography. At laparotomy, a 20-mm submucosal duodenal lesion was found associated with mucosal ulceration. Immunohistochemical analysis revealed it to be positive for CD117 (c-KIT protein) consistent with a GIST. We emphasize the importance of a thorough abdominal US examination in children with GI haemorrhage and the consideration of GIST in the diagnosis after the common causes have been excluded.     

Fetus in fetu: two case reports and review of literature

Fetus in fetu is a rare disorder. Its embryopathogensis and differentiation from teratoma has not been well established. It is a parasitic twin of a diamniotic monozygotic twin. Here we report, two cases of fetus in fetu with review of literature. In case report 1, a 2-year-old boy was referred for asymptomatic lump in abdomen since birth. X-ray showed the mass in the abdomen with some calcification and fluid inside. CT scan reported a heterogenous mass in the retroperitoneum with bony malformation. CT showed presence of three vertebrae in it. After surgically excising the mass and opening the sac it showed presence of trunk and two limbs with one of the limbs having a nail. Histopathology showed presence of GI tract. In case report 2, 4 month female was found to have lump in the abdomen by housemaid while bathing. X-ray showed a soft tissue shadow while ultrasonography revealed cystic mass arising from right kidney. CT suggested cystic mass with calcification not arising from kidney. During exploration whole mass was excised and there was frank fetus inside it. Histopathology confirmed presence of four vertebral bodies with germ layers. Although fetus in fetu is rare condition, correct diagnosis using imaging can be made before surgery. Complete excision is curative.     

Upper and lower gastrointestinal endoscopy in children with Crohn's disease

Twenty-three children and adolescents with Crohn's disease had systematic upper and lower gastrointestinal (GI) endoscopy as part of the initial diagnostic assessment. Twenty (86%) had evidence of some involvement of the upper GI tract (oesophagus-4, stomach-19, duodenum-5). In 13 children, the upper GI findings were instrumental in making the diagnosis of Crohn's disease. The ileum was viewed in 18 and found to be abnormal in 14 (78%). The colon was affected to some extent in 16 (70%); the proximal colon was affected twice as frequently as the distal colon and rectum. Early upper and lower GI endoscopy is recommended as part of the assessment of suspected inflammatory bowel disease in children. Offprint requests to: D. J. S. Cameron     

Ranitidine administration in Henoch-Schönlein vasculitis

In this study, we discuss 12 patients with gastrointestinal (GI) bleeding who were diagnosed as having Henoch Schoenlein vasculitis (HSV) in Dr Behçet Uz Children's Hospital, Izmir, between January 1991 and January 1992. Seven male and five female patients were included in the study. Their ages ranged between 6–14 years. The patients were separated into two identical groups and were given ranitidine or a placebo. Both groups were followed up for abdominal pain and GI bleeding. In the group administered ranitidine the duration and severity of abdominal pain and gastrointestinal bleeding decreased significantly as compared to the group taking placebo (P <0.05). No side effects of ranitidine were observed. As a result, it was concluded that ranitidine could be used to treat HSV with GI symptoms.     

Gastrointestinal blood flow and oxygen consumption in the newborn lamb: effect of chronic anemia and acute hypoxia

The purpose of this study was to investigate the compensatory change in circulation and oxygenation of the newborn lamb gastrointestinal (GI) tract in response to anemic and hypoxic hypoxemia. Radiolabeled microspheres were used to measure blood flow. We subjected the newborn lamb to a 30-35% reduction in hematocrit 4 d before study and to a 10% oxygen environment for 30 min during the study to induce chronic anemic and acute hypoxic hypoxemia, respectively. The circulatory and oxygenation responses were measured 1 h after a standard milk feeding in all cases. During the experimental periods, no change in total GI blood flow was observed. Because of a failure to augment blood flow during hypoxemia, O2 delivery to the GI tract decreased significantly. Despite this, GI O2 consumption was not compromised because tissue O2 extraction by the GI tract rose significantly. The response of the newborn lamb GI tract to hypoxemia after feeding is augmentation of O2 extraction. The newborn's GI tract did not regulate local GI blood flow.     

The use of clinical score to estimate the severity of an acute attack of asthma

OBJECTIVE: To identify patients with acute asthma who require intensive care, using a clinical score.METHODS: Retrospective analysis of 299 patients who were admitted to the hospital for acute asthma, between 1993 and 1996. Two groups were selected: group I (GI) with 26 patients admitted to the Intensive Care Unit; group II (GII) with 52 patients admitted to the Emergency Room, matched by age and sex with those of GI. A clinical score to assess the groups had the following items: heart rate, respiratory rate, retractions, cyanosis, alertness level, previous treatment, previous hospitalization, and duration of attack. The score ranged from 0 to 2 points for each of those, summing up 11 points.RESULTS: The clinical parameters that discriminated well the 2 groups were: retractions (p<0.001), cyanosis (p<0.01) and alertness level (p<0.005). Through a discriminate analysis of the clinical score components, 22/26 patients were recognized as GI and 40/52 patients were recognized as GII, showing an accuracy of 85% and 77% respectively for GI and GII.CONCLUSION: The clinical score was useful to discriminate patients with severe attack of asthma who might require intensive care, and should be employed at hospital admission.     

Improved gastrointestinal symptom burden after conversion from mycophenolate mofetil to enteric-coated mycophenolate sodium in kidney transplanted children

Abstract:  It has been shown in adult kidney transplant recipients that a conversion from MMF to EC-MPS significantly reduced the GI related symptom burden. No such study exists on children with GI problems while receiving MMF therapy. Ten paediatric kidney transplant recipients (mean age 14.5 yr, s.d. 4.5) receiving triple immunosuppression (Cyclosporin A or Tacrolimus + MMF + Prednisolone) with severe GI symptoms were converted to an equimolar dose of EC-MPS. The GSRS was completed before and at four wk after the switch, and GFR was determined for a mean period of six months. Values were compared by the paired t-test. Mean GSRS improved significantly after the switch to EC-MPS in all but one patient, from 2.1 (s.d. 0.9) to 1.1 (s.d. 0.6). The differences could be found in all four subscales. Graft function did not change after conversion to EC-MPS. In children with moderate or severe GI symptoms while receiving MMF, conversion to EC-MPS led to significantly reduced GI symptoms.     

Neonatal metrizamide gastrointestinal series in suspected necrotizing enterocolitis

The diagnosis of necrotizing enterocolitis (NEC) in neonates may be made by clinical presentation, roentgenographic findings, or a combination of both. Diagnosis leads to immediate treatment including nasogastric suction, parenteral antibiotics, plasma, and close monitoring of clinical, roentgenographic, and laboratory findings. Occasionally, neither the clinical nor plain roentgenographic appearance of an infant allows the diagnosis of NEC to be made or excluded with confidence. In such infants portable isotonic metrizamide gastrointestinal (GI) series were used to help make the decision of whether to begin treatment for NEC or to continue feeding the patient. Of 15 patients examined, two exhibited signs of NEC and were successfully treated medically without GI (tract) sequelae. Twelve neonates had normal results of metrizamide GI series and ten were immediately fed with no GI complication. One of these 12 infants had feedings withheld for several days as a result of a positive blood culture. One infant with severe cardiac and pulmonary disease had profound adynamic ileus and could not be fed. We have found the metrizamide GI series to be a useful study in neonates suspected of having NEC.     

Enteropathy in premature newborn infants. Prospective study over one year]

BACKGROUND. In neonatal units, there is a tendency to assume that any acutely sick infant with gastro-intestinal symptoms has necrotizing enterocolitis (NEC). This prospective study was conducted to find a better definition of enteropathy in preterm neonates and their risk factors. MATERIAL AND METHODS. All the 351 preterm neonates admitted to a neonatal unit from 1 August 1988 to 31 July 1989 were included in the study. A chart including 45 items was established for each infant, with special attention to data on the pregnancy, delivery, any early ischemic and/or infectious problem, nutrition and any gastro-intestinal (GI) problem. All the neonates were fed similarly, depending their maturation, gestational age and GI status. Each infant was assigned to one of 5 categories: 1) no GI problem; 2) transient obstruction; 3) NEC with pneumatosis; 4) hemorrhagic colitis without obstruction or pneumatosis; 5) other GI disease. RESULTS. 267 infants had no GI problem during their stay in the neonatal unit. 53 developed GI symptoms: 23 transient obstructions, 6 NEC, and 24 hemorrhagic colitis. The mean age at onset of symptoms in these last 3 categories was 7 days, 14 days and 23 days, respectively. Ten risk factors were found to be significantly correlated with GI disturbances: umbilical venous catheter, benzodiazepines, birth weight < 1,500 g, patent ductus arteriosus, ventilatory assistance, abnormal amniotic fluid, gestational age < 32 weeks, early antibiotic treatment, passage of meconium > 48 hours, episodes of apnoea and/or bradycardia. CONCLUSION. This follow-up shows that the GI disturbances of preterm neonates admitted to a neonatal unit, specially those having one or more risk factors, can be separated into 3 groups: 1) isolated intestinal obstruction, seen in the most immature babies during the first week of life with the risk of developing NEC; 2) frank blood in the stool, indicating colitis and possibly minor forms of NEC; 3) combined obstructive and hemorrhagic symptoms, typical of NEC.     

Vedolizumab for pediatric patients with gastrointestinal acute graft-versus-host-disease

Vedolizumab, an anti-α4β7 integrin monoclonal antibody, impairs homing of T-cells to the gastrointestinal (GI) endothelium and acts as a gut-selective anti-inflammatory agent. Recent reports of the efficacy of vedolizumab in treating lower GI acute graft-versus-host disease (aGVHD) are promising, but experience in children is scarce. We present a cohort of 13 pediatric patients who were treated with vedolizumab for GI aGVHD. Ten of the patients were treated for steroid-refractory disease, out of which, six suffered from severe (stage 3 or 4) GI disease before the first dose of vedolizumab. In the other three patients, vedolizumab was introduced early in the disease course. Median time between GI GVHD onset to vedolizumab treatment was 23 days (range 7–59 days), with a median of 3 doses (range 1–5) per patient. GI GVHD staging was evaluated at various time points after the first vedolizumab dose, showing improvement in nine of the 13 patients. After a median follow-up time of 13 months (range 6–34 months), eight patients completely recovered, two had ongoing chronic colitis, and three patients died. During the vedolizumab treatment period, 38 infectious episodes were noted, most of them GI related. The unique activity profile of vedolizumab makes it an appealing treatment option for lower GI aGVHD, but caution for concurrent infections is warranted.     

Upper gastrointestinal bleeding in children in Southern Iran

Objective  To find out whether the causes of upper GI bleeding in our center in a developing country differed from developed countries. Methods  Children presenting to our center with upper GI bleeding from March 2002 to March 2007, were retrospectively evaluated. Informations were retrieved from patient’s history and physical examination and results of upper GI endoscopy regarding etiology of bleeding, managements, use of medications which might predispose patient to bleeding, and the mortality rate. Results  From 118 children (67 boys; with age of 7.7±4.7 yrs) who underwent upper GI endoscopies, 50% presented with hematemesis, 14% had melena and 36% had both. The most common causes of upper GI bleeding among all patients were gastric erosions (28%), esophageal varices (16%), duodenal erosions (10%), gastric ulcer (8.5%), Mallory Weiss syndrome tear (7.8%), duodenal ulcer (6.8%), esophagitis (1.7%) and duodenal ulcer with gastric ulcer (0.8%). The causes of bleeding could not be ascertained in 20.5% of cases. No significant pre-medication or procedure related complications were observed. Endoscopic therapy was performed in 13.5% of patients. In 14.4% of patients, there was a history of consumption of medications predisposing them to upper GI bleeding. Two deaths occurred (1.7%) too. Conclusion  The findings in the present study showed that half of upper GI bleedings in pediatric patients from south of Iran, were due to gastric and duodenal erosions and ulcers. This study concludes that the causes of upper GI bleeding in children in our center of a developing country, are not different from those in developed ones.     

Effects of hypoxemia on gastrointestinal blood flow and gastric emptying in the newborn piglet

The effects of severe hypoxemia on gastrointestinal (GI) blood flow and gastric emptying were studied in nine 2- to 4-day-old piglets which were mechanically ventilated while receiving nitrous oxide anesthesia. Each animal was studied during a control period of oxygenation (PaO2 91 +/- 8 torr), 35 min of hypoxemia (PaO2 29 +/- 1 torr), and a recovery period (PaO2 90 +/- 5 torr) (mean +/- SEM). During each study period, the animal received a 10% dextrose test meal with phenol red marker (22 ml/kg), gastric residual volumes were determined at 10-min intervals over 30-min study periods using a dye dilution double sampling technique, and GI blood flow (radionuclide-labeled microspheres), O2 delivery, O2 extraction, and O2 consumption were measured at the end of the 30-min period. Hypoxemia resulted in decreased blood flow to the following GI organs: stomach, jejunal and ileal mucosa-submucosa, and colon decreased 62, 31, and 35%, respectively (p less than 0.05). Jejunal and ileal muscularis blood flow remained unchanged. Oxygen delivery and consumption by GI tract decreased 79 and 58%, respectively; whereas oxygen extraction of GI tract increased 115%. Values returned toward baseline levels during the recovery period. The hypoxemic gastric emptying pattern showed significantly greater gastric residuals at 20 min compared to the 10-min value (p less than 0.05). This pattern was different than that observed during control and recovery periods. We conclude that severe hypoxemia results in decreased GI blood flow, tissue oxygenation, and an altered gastric emptying pattern. These observations may have clinical significance for feeding infants following an hypoxemic episode.     

Pancreas‐preserving duodenectomy after living donor liver transplantation for invasive cytomegalovirus disease

CMV infection plays an important role in the postoperative course following solid organ transplantation. We present the case of an 11‐year‐old male patient who underwent LDLT due to severe hepatopulmonary syndrome and biliary cirrhosis. Four weeks after LDLT, he developed persistent GI bleeding and was subjected to repeated endoscopic treatment and radiological arterial embolization to stop the bleeding from duodenal ulcers. Diagnostic workup was negative for CMV disease. Because the bleeding persisted, surgical treatment was indicated, and a pancreas‐preserving duodenectomy was performed. Immunohistochemical staining of the surgical specimen demonstrated diffuse endothelial infiltration by CMV. Despite ganciclovir treatment, the patient developed new erosions in the jejunal mucosa and melena; ganciclovir was discontinued, and foscarnet was started, resulting in clinical improvement and the cessation of bleeding. This case highlights the technical aspects of performing a complex upper GI resection in a patient recently subjected to LDLT, taking care to avoid injury to the previous liver graft anastomosis and restore GI continuity. Moreover, CMV tissue‐invasive disease compartmentalized in the GI tract may be difficult to diagnose, as indicated by the negative results of antigenemia and PCR assays and endoscopic superficial mucosal biopsies.