Comparison of long-term outcome according to involved aganglionic segments of total colonic aganglionosis.

ABSTRACT: Total colonic aganglionosis (TCA) is a rare form of Hirschsprung disease, with more severe symptoms than rectosigmoid Hirschsprung disease. We aimed to evaluate the surgical outcomes according to the involved segments of TCA.Patients with aganglionosis extending from the anus to at least the ileocecal valve were included. The medical records of 33 TCA patients from 1981 to 2014 were reviewed. Three groups were analyzed based on the involved segment (jejunum, jejunoileal junction, and distal ileum).The median age at the pull-through operation was 6.2 (3.3-114) months. The median follow-up duration was 216 (21-411) months. Transition zone in the jejunum, jejunoileal junction, and distal ileum was identified in 3, 5, and 25 patients, respectively. The most common method of operation was Duhamel pull-through. Perianal excoriation and enterocolitis were the most common postoperative complications. The complication rates were 45% to 51% and not different among the groups. The defecation frequency normalized 3 years postoperatively, and body weight started to recover after 2 years irrespective of the involved segment.Therefore, close monitoring with proper management of defecation and body weight for at least 2 to 3 years postoperatively is required.

Experiences of Rectovaginal Fistula in Anorectal Malformation.

BACKGROUND: Rectovaginal fistulas (RVFs) are very rare malformations in females with anorectal malformations (ARMs). Here, we share the clinical features of RVF and report the long-term outcomes. METHODS: RVF patients were classified using a retrospective analysis of ARM patients who underwent operations at Seoul National University Hospital between January 1999 and May 2017. The Krickenbeck continence scoring system was used to evaluate bowel function 5 and 10 years after surgery. RESULTS: Of the total 460 ARM patients, 203 were female, 7 of whom were diagnosed with RVF. The median age and weight at the time of anorectoplasty were 292 days (range, 140-617) and 8.2 kg (range, 5.5-12), respectively. Six patients had associated anomalies and three patients underwent redo-anorectoplasty. Voluntary bowel movements were observed in 6 out of 7 patients at 5 and 10 years of age. Soiling was observed in all patients at the age of five years and in 6 out of 7 patients at the age of ten years. Constipation was observed in 6 out of 7 patients at both five and ten years of age. CONCLUSIONS: An RVF is a very rare malformation, accounting for 1.5% of total ARMs and 3.4% of ARMs in females. Long-term counseling, education, and guidance are needed for effective management of patients' bowel movements. TYPE OF STUDY: Prognosis study LEVEL OF EVIDENCE: Level IV.

Association analysis of NOX5 polymorphisms with Hirschsprung disease.

BACKGROUND/PURPOSE: Hirschsprung disease (HSCR) is a developmental disease characterized by the absence of ganglion cells in the intestinal region. NADPH oxidase5 (NOX5) has been identified as one of the possible candidate genes for risk of Hirschsprung disease in our recent genome wide association study (GWAS). In this study, we performed a replication study to analyze the association of NOX5 polymorphisms with HSCR risk and conducted an extended analysis to investigate further associations for sub-groups and haplotypes. METHODS: A total of 23 NOX5 single nucleotide polymorphisms (SNPs) were genotyped in 187 HSCR patients and 283 unaffected controls. Statistical analysis was performed to examine the effects of genotype on risk of HSCR and HSCR subtype. RESULTS: Logistic regression analyses revealed that six SNPs (rs59355559, rs62010828, rs34990910, rs11856030, rs311905, and rs8024894) were associated with risk of HSCR (minimum p = 0.007 at rs62010828). Moreover, three SNPs (rs59355559, rs62010828, and rs8024894) were significantly associated with risk of long-segment HSCR (L-HSCR) subtype and 5 SNPs (rs59355559, rs62010828, rs34990910, rs11856030, and rs8024894) were found to be associated with risk of TCA subtype. CONCLUSION: Our results demonstrate that genetic variants in NOX5 have genetic effects on risk of HSCR, which may serve as useful preliminary information for further study. LEVELS OF EVIDENCE: Level III of prognosis study.

Why Do the Patients with Hirschsprung Disease Get Redo Pull-Through Operation?

INTRODUCTION: The treatment of Hirschsprung disease (HD) is pull-through (PT) surgery. Redo PT can be performed in 1 to 10% of patients after initial PT. In this study, we reviewed the causes and associated factors of redo PT. MATERIALS AND METHODS: We retrospectively reviewed medical charts of 657 patients with HD who underwent surgeries between September 1979 and January 2016. The indications for redo PT are as follows. First, there were persistent obstructive symptoms after the first operation, (1) with transition zone shown definitely on contrast study, (2) with anatomic problems, and (3) obstructive symptoms persist despite conservative or nonredo surgical treatment without (1) and (2). We analyzed the causes and associated factors of redo PT. RESULTS: A total of 49 (7.5%) patients underwent redo PT. Among them, 41 and 8 patients underwent PT twice and three times, respectively. Among 57 cases of redo, the causes of redo included pathologic problem (n = 28)-aganglionosis (n = 20), hypoganglionosis (n = 4), immature ganglion cell (n = 4)-or anatomic problem (n = 21)-stricture (n = 13), fistula and/or abscess (n = 8) at anastomosis. Comparing associated factors between the nonredo and redo groups, the redo group had longer initial PT operation time (p = 0.001), more postoperative complications (p < 0.001), and more transanal endorectal PT (TERPT) approach as initial PTs (p < 0.001). According to causes of redo, the anatomic problem group underwent more third PTs than the pathologic problem group (p = 0.010). CONCLUSION: Approximately 7.5% of patients experienced redo PT. The cause of redo included pathologic (n = 28) or anatomic problem (n = 21). Longer operation time, more complications, and TERPT were associated with redo. The anatomic problem group underwent more third PTs than the pathologic problem group.

Prospective evaluation of clinical outcomes and quality of life after gastric tube interposition as esophageal reconstruction in children.

Few studies on gastric tube interposition for esophageal reconstruction in children have assessed the long-term outcomes and quality of life (QoL). The aim of this study is to evaluate the long-term outcomes and QoL after a gastric tube interposition by reviewing our experiences with esophageal reconstruction.Twenty-six patients were included who underwent gastric tube interposition from 1996 to 2011 at our institution. We reviewed the medical records and conducted telephone surveys, prospectively performed esophagography, endoscopy, 24-hour pH monitoring, and esophageal manometry. The median follow-up period of 12 (range, 3-18) years.Median age at the time of surgery and survey were 9 (range, 2-50) months and 12.4 (range, 3.1-19.0) years, respectively. There were 14 cases of reoperation of gross type C and B esophageal atresia (EA) and 10 cases of long gap pure EA. The z scores of anthropometric data at the survey did not increase after the operation. Severe stricture in esophagography was observed in 20% of patients, but improved with balloon dilation with intact passage. Gastroesophageal reflux was able to be treated with medications. Esophageal peristalsis was observed in 1 of 8 patients in manometry. No Barrett esophagus or metaplasia was not found from endoscopy. QoL was similar to the general population and did not differ between age groups.Gastric tube interposition could be considered for esophageal reconstruction in pediatric patients when native esophageal anastomosis is impossible. Nutritional evaluation and support with consecutive radiological evaluation to assess the anastomosis site stricture are advised.

Inguinal hernia in preterms in neonatal intensive care units: Optimal timing of herniorrhaphy and necessity of contralateral exploration in unilateral presentation.

BACKGROUND: We sought to determine the optimal timing of IH repair in preterms and the need for routine contralateral exploration. METHODS: Medical records of 3690 pediatric patients who underwent unilateral IH repair between January 1998 and December 2009 were reviewed. We assessed medical record review and telephone interviews. In total, 1990 patients were enrolled in the study. Early, early-delayed, and late repair were defined as herniorrhaphy performed within 7 days of diagnosis, later than 7 days of diagnosis, and after discharge from the NICU, respectively. RESULTS: Of 1990 patients, 90 preterms and 1900 full-terms were included. Among these, 7, 11 and 72 preterm patients received early, early-delayed and late IH repairs, respectively. Preoperative incarceration and postoperative complication rates were not different, but the recurrence rate was higher in the early repair group. Two group analysis of early and early-delayed vs. late repairs indicated similar results. The rates of synchronous and metachronous bilateral IH (SBIH, MBIH) were observed to be higher and the diagnostic interval of MBIH was shorter in preterms than in full-terms (35.6% vs. 15.9%, P < 0.001; 12.2% vs. 6.3%, P < 0.001; 5.2 vs. 41.8 months, P = 0.003). CONCLUSION: Our results indicate that IH repair is safe to perform in preterm babies in the NICU at a delayed or late stage since the preoperative incarceration and recurrence rates were not different. Contralateral exploration could be considered in preterms because the rates of SBIH and MBIH were significantly higher and the MBIH diagnosis interval was shorter than in full-terms. LEVEL OF EVIDENCE: III, treatment study.

Spectrum of MNX1 Pathogenic Variants and Associated Clinical Features in Korean Patients with Currarino Syndrome.

BACKGROUND: The major genetic cause of Currarino syndrome (CS), a congenital malformation syndrome typically characterized by sacral agenesis, anorectal malformation, and presence of a pre-sacral mass, is known to be pathogenic variants in motor neuron and pancreas homeobox 1 (MNX1), which exist in almost all familial cases and 30% of sporadic cases. Less commonly, a large deletion or a complex rearrangement involving the 7q36 region is associated with CS. We investigated the spectrum of MNX1 pathogenic variants and associated clinical features in the Korean patients with CS. METHODS: We enrolled 25 patients with CS, including 24 sporadic cases and one familial case. Direct sequencing of MNX1 and multiplex ligation-dependent probe amplification were performed. We also analyzed clinical phenotypes and evaluated genotype-phenotype correlations. RESULTS: We identified six novel variants amongst a total of six null variants, one missense variant, and one large deletion. The null variants included four frameshift variants (p.Gly98Alafs* 124, p.Gly145Alafs*77, p.Gly151Leufs*67, and p.Ala216Profs*5) and two nonsense variants (p.Tyr186* and p.Gln212*). The missense variant, p.Lys295Gln, was located in the highly-conserved homeobox domain and was predicted to be deleterious. A large deletion involving the 7q36 region was detected in one patient. Pathogenic variants in MNX1 were detected in 28% of all CS cases and 25% of sporadic cases. The clinical phenotype was variable in patients with and without pathogenic variants; no significant genotype-phenotype correlation was observed. CONCLUSIONS: This study revealed the spectrum and phenotypic variability of MNX1 pathogenic variants in the Korean population.

Analysis of clinical features of lipoblastoma in children.

OBJECTIVE: A lipoblastoma is pathologically benign but often recurs. Due to its rarity, studies are scarce. The purpose of this study was to investigate the clinical characteristics of lipoblastoma occurring in children and to detect any correlations with the expression of Ki-67. PARTICIPANTS: From 1998 to 2010, 33 patients were diagnosed with lipoblastoma at Seoul National University Children's Hospital. METHODS: Ki-67 immunohistochemistry staining of the tumor tissue was performed. RESULTS: A total of 33 patients (64% males) were enrolled in the study, with a mean age of 28 month. Eleven and 22 lesions were deep and superficial, respectively. Complete excisions were performed for 30 patients, and three underwent incomplete excisions. Two patients who underwent incomplete excision subsequently underwent a second operation due to tumor regrowth, and one patient had a recurrence despite complete excision. There was no statistically significant correlation observed between the tumor size or recurrence and the expression of Ki-67. CONCLUSIONS: Lipoblastoma requires an accurate diagnosis and operative resection to alleviate the symptoms induced by its growth. Incompletely resected tumor may regrow; therefore, complete excision is the treatment of choice. Continuous follow-up is needed to monitor for recurrence of disease, even after a complete excision.

Clinical Study of Congenital Esophageal Stenosis: Comparison according to Association of Esophageal Atresia and Tracheoesophageal Fistula.

PURPOSE: Congenital esophageal atresia (CES) is a rare congenital disease. The severity of symptoms is variable; thus, diagnosis is difficult and tends to be delayed. CES is frequently accompanied by esophageal atresia (EA) with/without tracheoesophageal fistula (TEF). We investigated the characteristics of CES by reviewing our experience with CES patients and researched the differences between CES with EA-TEF and isolated CES. METHODS: A total of 31 patients underwent operations for CES were reviewed retrospectively. The patients were divided into two groups according to the association with EA-TEF, and compared the differences. RESULTS: Sixteen boys and 15 girls were included. The mean age at symptom onset was 8 months old, and the mean age at diagnosis was 21 months old. Nine patients with EA-TEF were included group A, whereas the other 22 patients were assigned to group B. There were no differences in sex, gestational age, associated anomalies and pathologic results between the groups. In group A, the age at diagnosis and age at surgery were younger than in group B despite the age at symptom occurrence being similar. Postoperative complications occurred only in group A. CONCLUSION: In this study, symptoms occurred during the weaning period, and vomiting was the most frequent symptom. CES patients with EA-TEF tended to be diagnosed and treated earlier despite the age at symptom occurrence being similar. CES patients with EA-TEF had more postoperative complications; therefore, greater attention should be paid during the postoperative period.

Meckel diverticulum in exomphalos minor.

A congenital hernia into the base of the umbilical cord is known as an exomphalos and when the size of the defect is 5 cm or less and containing only bowel, it is called as exomphalos minor. We present a case of a newborn with an exomphalos minor within a Meckel diverticulum. He underwent surgical resection of the Meckel diverticulum and repair of the abdominal wall defect. To our knowledge, this is the first reported case of Meckel diverticulum in an exomphalos minor in Korea.

Currarino triad with Müllerian duct anomaly in mother and daughter without MNX1 gene mutation.

The Currarino triad is a unique complex of congenital caudal anomalies, including anorectal malformation, sacral bony defect and presacral mass. This triad may be associated with Müllerian duct anomalies, such as duplication of the vagina and uterus. Each of these diseases has a familial tendency and sometimes coexist within families. But, when coexisting in familial cases, nearly all reported cases revealed mutations of the motor neuron and pancreas homeobox 1 (MNX1) gene. Familial cases of Currarino triad combined with Müllerian duct anomaly without MNX1 gene mutation are very rare. Here we report cases of mother and daughter, who had Currarino triad and Müllerian duct anomaly without MNX1 gene mutation, along with a brief literature review.

Long-term functional outcomes of PPPD in children--Nutritional status, pancreatic function, GI function and QOL.

PURPOSE: The purpose of this study was to analyze the long-term outcomes, such as nutritional status, pancreatic function, gastrointestinal (GI) function, and quality of life (QOL), in children who underwent pylorus-preserving pancreaticoduodenectomy (PPPD). METHODS: Between 1992 and 2013, there were 15 children who underwent PPPD at Seoul National University Children's Hospital, and 10 of them participated in this study. A retrospective review of the patients' medical records and follow-up was done. Their nutritional statuses were estimated by height, body weight, weight for age Z-score, body mass index (BMI), and serum protein, albumin levels. The endocrine and exocrine functions of the pancreas were estimated by diabetes mellitus (DM), steatorrhea, and Bristol stool chart. The GI function and QOL were evaluated via questionnaires. The follow-up period ranged from 3 to 18years. RESULTS: There were no severe growth disturbances, 6 patients experienced mild steatorrhea and 3 showed above the category 6 in Bristol stool chart. All the patients experienced mild GI symptoms. As for the QOL, there were no significant negative answers, except for one patient with DM. CONCLUSIONS: Almost all the study subjects, who underwent PPPD in their childhood, did not present significant problems except for one patient with DM.

Association Analysis of SLC6A20 Polymorphisms With Hirschsprung Disease.

PURPOSE: Hirschsprung disease (HSCR) is a congenital and heterogeneous disorder, which is caused by no neuronal ganglion cells in part or all of distal gastrointestinal tract. Recently, our genome-wide association study has identified solute carrier family 6, proline IMINO transporter, member 20 (SLC6A20) as one of the potential risk factors for HSCR development. This study performed a replication study for the association of SLC6A20 polymorphisms with HSCR and an extended analysis to investigate further associations for subgroups and haplotypes. METHODS: For the replication study, a total of 40 single nucleotide polymorphisms (SNPs) of SLC6A20 were genotyped in 187 HSCR subjects composed of 121 short-segment HSCR, 45 long-segment HSCR (L-HSCR), 21 total colonic aganglionosis, and 283 unaffected controls. Imputation was performed using genotype data from our genome-wide association study and this replication study. RESULTS: Imputed meta-analysis revealed that 13 SLC6A20 SNPs (minimum P = 0.0002 at rs6770261) were significantly associated with HSCR even after correction for multiple comparisons using false discovery rate (FDR) (minimum PFDR =  .005). In further subgroup analysis, SLC6A20 polymorphisms appeared to have increased associations with L-HSCR. Moreover, haplotype analysis also showed significant associations between 2 haplotypes (BL3_ht2 and BL4_ht2) and HSCR susceptibility (PFDR <  .05). CONCLUSIONS: Although further replications and functional evaluations are required, our results suggest that SLC6A20 may have roles in HSCR development and in the extent of aganglionic segment during enteric nervous system development.

Pediatric cholecystectomy for symptomatic gallstones unrelated to hematologic disorder.

BACKGROUNDS/AIMS: Gallstones are being increasingly diagnosed in pediatric patients. The purpose of this study was to determine characteristics of pediatric patients who underwent cholecystectomy because of symptomatic gallstone disease unrelated to hemolytic disorder. METHODS: We reviewed cases of pediatric patients (under 18 years old) who underwent cholecystectomy between May 2005 and December 2015. RESULTS: A total 20 pediatric patients (under 18 years old) underwent cholecystectomy during the study period. One patient was excluded because cholecystectomy was performed due to gall stones caused by hemolytic anemia. The 19 cases comprised 9 male (47.3%) and 10 female (52.7%) subjects. The mean age was 14.9 years (range, 5-18), and 66.7% of patients were older than 12 years of age. Mean body weight was 65.0 kg (range, 13.9-93.3), and mean body mass index was 21.7 kg/m2 (range, 12.3-35.1), with 26.37% of patients being overweight. All 19 patients underwent laparoscopic cholecystectomy. There were no postoperative complications and no mortality. Comparison between overweight and non-overweight patients indicated that significantly more overweight patients had cholesterol stones (5/5 vs. 7/14, p=0.036) and were classified as complicated disease (3/5 vs. 1/14, p=0.037). CONCLUSIONS: The more frequent occurrence of complications such as choledocholithiasis or gallstone pancreatitis, in overweight patients indicates the need for more careful evaluation and management in these patients. Pediatricians and surgeons should always consider gallstone disease in pediatric patients despite difficulty in suspecting symptomatic gallstones in cases who present with abdominal pain that is rarely clear-cut.

Potential Association of INMT Nonsynonymous Variant (His46Pro) with Hirschsprung's Disease.

BACKGROUND: Hirschsprung's disease (HSCR) is a congenital disorder which is characterized by the lack of ganglion cells in part of or the entire colon, resulting in intestinal obstruction and other related symptoms. Recently, our group has conducted a genome-wide association study in Korean HSCR cases and controls to identify novel markers in other genes. OBJECTIVES: The present research aimed to further study the potential association of INMT with HSCR by conducting a replication study. METHODS: A total of 15 INMT single nucleotide polymorphisms (SNPs) were analyzed for the association with HSCR in 187 HSCR patients and 283 controls. Analyses were also conducted for subtypes of HSCR (short-segment, long-segment, and total colonic aganglionosis). RESULTS: A nonsynonymous SNP rs77743549 (His46Pro) was significantly associated with the increased risk of HSCR (odds ratio = 1.77; corrected p = 0.002). Furthermore, this rs77743549 retained its association with all subtypes of HSCR (p = 0.006-0.002 under the codominant model). A global test showed that rs77743549 was associated with the length of aganglionosis (p = 0.00004). CONCLUSION: Although further replications and functional evaluations are needed, our study suggests that rs77743549 of INMT may be associated with the risk for HSCR and/or the development of the enteric nervous system.

How should the pyloric submucosal mass coexisting with hypertrophic pyloric stenosis be treated?: a case of pyloric ectopic pancreas with hypertrophic pyloric stenosis.

Co-existing pyloric submucosal masses with hypertrophic pyloric stenosis (HPS) are very rare and treating these lesions is always a problem. A 20-day-old boy presented with recurrent episodes of projectile non-bilious vomiting lasting for 5 days. HPS was suspected due to the presenting age and the symptoms. The sonography demonstrated not only circumferential wall thickening of the pylorus, but also a pyloric submucosal mass. At laparotomy, a 0.8 cm sized pyloric submucosal mass was identified along with a hypertrophied pylorus. Pyloric excision was performed due to the possibility of sustaining the symptoms and malignancy. The pathological report of the submucosal mass was ectopic pancreas. Coexisting pyloric lesions can be diagnosed along with HPS, and surgical excision, not just pyloromyotomy, should be considered in these circumstances. To the best of our knowledge, this is the first case report of pyloric ectopic pancreas and HPS to be diagnosed concurrently.

A genome-wide association study identifies potential susceptibility loci for Hirschsprung disease.

Hirschsprung disease (HSCR) is a congenital and heterogeneous disorder characterized by the absence of intramural nervous plexuses along variable lengths of the hindgut. Although RET is a well-established risk factor, a recent genome-wide association study (GWAS) of HSCR has identified NRG1 as an additional susceptibility locus. To discover additional risk loci, we performed a GWAS of 123 sporadic HSCR patients and 432 unaffected controls using a large-scale platform with coverage of over 1 million polymorphic markers. The result was that our study replicated the findings of RET-CSGALNACT2-RASGEF1A genomic region (rawP = 5.69×10(-19) before a Bonferroni correction; corrP = 4.31×10(-13) after a Bonferroni correction) and NRG1 as susceptibility loci. In addition, this study identified SLC6A20 (adjP = 2.71×10(-6)), RORA (adjP = 1.26×10(-5)), and ABCC9 (adjP = 1.86×10(-5)) as new potential susceptibility loci under adjusting the already known loci on the RET-CSGALNACT2-RASGEF1A and NRG1 regions, although none of the SNPs in these genes passed the Bonferroni correction. In further subgroup analysis, the RET-CSGALNACT2-RASGEF1A genomic region was observed to have different significance levels among subgroups: short-segment (S-HSCR, corrP = 1.71×10(-5)), long-segment (L-HSCR, corrP = 6.66×10(-4)), and total colonic aganglionosis (TCA, corrP>0.05). This differential pattern in the significance level suggests that other genomic loci or mechanisms may affect the length of aganglionosis in HSCR subgroups during enteric nervous system (ENS) development. Although functional evaluations are needed, our findings might facilitate improved understanding of the mechanisms of HSCR pathogenesis.

Pediatric split liver transplantation after Fontan procedure in left isomerism combined with biliary atresia: a case report.

LI is a subset of the heterotaxy syndrome and a rare birth defect that involves the heart and other organs. It can be combined with extracardiac abnormalities, especially BA. CHD can be associated with LI in up to 15% of cases, although it is rare in BA. Pediatric LT for a child with ESLD due to BA combined with LI and CHD is a challenging issue for a transplant surgeon. Herein, we report a successful split LT on a three-yr-old boy with LI who survived after a Fontan procedure due to single ventricle, but who suffered from HPS associated with BA.

Neonatal gastric perforation: A single center experience.

AIM: To determine the etiology and prognostic factors for neonatal gastric perforation (NGP), a rare but life-threatening disease. METHODS: Between 1980 and 2011, nine patients underwent surgical intervention for NGP at Seoul National University Children's Hospital. The characteristics and prognosis of the patients were retrospectively analyzed. RESULTS: Among the nine patients, three (33.3%) were preterm babies and five (55.5%) had associated anomalies, which included diaphragmatic eventration (n = 2), congenital diaphragmatic hernia, esophageal atresia with tracheoesophageal fistula, and antral web. Three (33.3%) patients were born before 1990 and three (33.3%) had a birth weight < 2500 g. Pneumoperitoneum was found on preoperative images in six (66.7%) patients, and incidentally in the other three (33.3%) patients. Surgery was performed within 24 h after the onset of symptoms in seven (77.8%) patients. The overall mortality rate was 22.2% (2/9). The time between symptoms and surgical intervention was the only prognostic factor for survival, whereas premature birth and birth weight were not. CONCLUSION: Early detection and advances in neonatal intensive care may improve the prognosis of NGP.

Gastrointestinal hemangioma in childhood: a rare cause of gastrointestinal bleeding.

Gastrointestinal (GI) hemangiomas are relatively rare benign vascular tumors. The choice of an appropriate diagnostic method depends on patient age, anatomic location, and presenting symptoms. However, GI hemangiomas are not a common suspected cause of GI bleeding in children because of their rarity. Based on medical history, laboratory results, and imaging study findings, the patient could be treated with either medication or surgery. Herein, we report 3 cases of GI hemangioma found in the small bowel, rectum, and GI tract (multiple hemangiomas). Better knowledge and understanding of GI hemangioma could help reduce the delayed diagnosis rate and prevent inappropriate management. Although rare, GI hemangiomas should be considered in the differential diagnosis of GI bleeding.