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Background: Malrotation is a congenital condition that predisposes individuals to midgut volvulus, which can result in significant bowel resection. While most cases of malrotation are diagnosed by the age of 1 year, typically presenting with symptoms related to volvulus or bowel obstruction, some cases remain asymptomatic. In children with visceral malposition, gastroschisis, omphalocele, or diaphragmatic hernia, malrotation may be suspected before symptoms manifest. However, isolated malrotation without midgut volvulus diagnosed prenatally is rare. We herein present a case of isolated malrotation without midgut volvulus that was prenatally diagnosed and successfully treated with laparoscopic surgery., Case Presentation: A 30-year-old woman (gravida 3, para 1) underwent routine obstetric ultrasound, which revealed increased blood flow in the lower uterine segment and abnormal placental attachment. To rule out placenta percreta, magnetic resonance imaging was performed at 34 weeks of gestation. Incidentally, abnormal fetal intestinal arrangement was noted, with the colon localized in the left hemi-abdomen and the small intestine distributed in the right hemi-abdomen, raising suspicion of malrotation. Postnatal contrast studies confirmed the diagnosis of malrotation without midgut volvulus. Given the risk of midgut volvulus, a laparoscopic Ladd's procedure was performed on day 6 of life. The postoperative course was uneventful, and the patient was still symptom-free 1 year postoperatively., Conclusions: This case illustrates that malrotation can be prenatally diagnosed using fetal magnetic resonance imaging. Considering the risk of midgut volvulus, prophylactic Ladd's procedure should be performed in neonatal period. In cases where malrotation is not complicated by midgut volvulus, a laparoscopic Ladd procedure can be safely performed in neonates., (© 2024. The Author(s).)

Background: Although many recent studies have scientifically verified the efficacy of the traditional herbal medicine daikenchuto (DKT) for postoperative gastrointestinal function, its efficacy has not been established in children. We retrospectively evaluated the effect of DKT in pediatric patients with panperitonitis associated with perforated appendicitis (PaPA) who underwent laparoscopic appendectomy., Methods: Among 34 children with PaPA who underwent laparoscopic appendectomy from May 2012 to May 2021, 19 received DKT (group D) and 12 did not (group C). We compared postoperative gastrointestinal function, complications, and improvement in the inflammatory response between the two groups., Results: Of the evaluation parameters for postoperative gastrointestinal function, the mean ± standard deviation time to first flatus was significantly shorter in group D than in group C (1.21 ± 0.42 and 2.17 ± 0.94 days respectively; p = 0.0005). The time to ingestion of half a meal was also significantly shorter in group D than in group C (8.42 ± 3.69 and 12.50 ± 4.96 meal occasions respectively; p = 0.01). There was no significant difference in complication rates between the two groups., Conclusion: Daikenchuto rapidly and safely improved postoperative gastrointestinal symptoms in children with PaPA. To the best of our knowledge, this is the first study to evaluate the effect of DKT on postoperative symptoms in laparoscopic appendectomy and in children., (© 2023 Japan Pediatric Society.)

Toll-like receptors (TLRs) sense microbial infection through recognition of pathogen-associated molecular patterns. For example, TLR4 responds to the lipopolysaccharide of gram-negative bacteria, whereas TLR2 recognizes a broad range of microbial ligands. Both receptors are, therefore, compelling targets for treating sepsis. Here, we developed a TLR2xTLR4 tetravalent bispecific antibody designated ICU-1, which inhibits both receptors. The inhibitory activity of ICU-1 was comparable to that of the parental antibodies in neutralization assays using a human monocyte cell line. Moreover, ICU-1 completely blocked stimulation of human peripheral blood mononuclear cells by clinically relevant bacterial species. These findings provide convincing evidence that ICU-1 offers a novel approach for treating bacterial sepsis.

Toll-like receptor 4 (TLR4) plays a critical role in the innate immune system and is involved in the pathogenesis of multiple diseases. Here, we report the antagonistic and ratized antibody, 52-1H4 e2 (e2), which completely inhibited lipopolysaccharide-induced interleukin-6 secretion in vitro . The average serum drug concentration was above 10 μg/mL for 28 days in rats injected with e2. The novel anti-rat TLR4/myeloid differentiation factor 2 antibody, e2, may be a useful tool for investigating the role of TLR4 in rat disease models.

Purpose: Our aim was the longitudinal assessment of cyst size in fetuses with biliary cystic malformation (BCM) to explore its etiology and the possibility of antenatal differentiation between biliary atresia (BA) and congenital biliary dilatation (CBD)., Methods: We conducted a retrospective review of all patients diagnosed antenatally with BCM from 1994 to 2014 at our institutions., Results: The study cohort comprised of three patients with BA and six with CBD. There were no significant differences in the gestational age and cyst size at the first detection of BCM between the two groups. In fetuses with CBD, the cyst size steadily increased as the gestational age advanced, while it fluctuated around 1.5 cm and remained below 2.1 cm in those with BA. However, the ratio of cystic area to fetal trunk area was approximately constant due to linear fetal growth in fetuses with CBD., Conclusion: Fetuses with BCM <2.1 cm in the late gestation period were more likely to have BA than CBD. Our observation of cyst enlargement with advancing gestational age in the CBD group was attributed solely to fetal growth. Biliary dilatation in fetuses with CBD and BA might be completed at the onset of BCM., Competing Interests: Conflict of Interest: The authors have no financial conflicts of interest., (Copyright © 2020 by The Korean Society of Pediatric Gastroenterology, Hepatology and Nutrition.)

Background: Torsion of the vermiform appendix is a rare disease with symptoms very similar to those of acute appendicitis. We herein report a case of torsion of the vermiform appendix diagnosed by intraoperative findings., Case Presentation: A 4-year-old boy presented to our hospital because of abdominal pain and vomiting. Laboratory data revealed a C-reactive protein level of 0.08 mg/dL and white blood cell count of 19,300/μL (neutrophils, 88.9%). Abdominal ultrasound showed a target sign-like finding in the ileocecal region. A computed tomography scan showed swelling of the appendix. We performed an emergency operation under suspicion of acute appendicitis. Laparoscopic examination showed that the appendix had twisted 720° in the clockwise direction. Appendectomy was performed, and the postoperative course was uneventful., Conclusions: Although torsion of the vermiform appendix is a very rare disease and difficult to differentiate from appendicitis, an inappropriate treatment plan could lead to necrosis and perforation of the appendix. It is important to consider this disease as a differential diagnosis in patients with right lower abdominal pain.

Background: The implantable cardioverter defibrillator (ICD) is a standard prevention therapy for patients at high risk for sudden cardiac death (SCD) due to life-threatening ventricular arrhythmia (VA), that is, ventricular fibrillation and ventricular tachycardia. However, clinical predictors of recurrent VA in secondary prevention ICD recipients with coronary artery disease (CAD) remain unknown. Methods and Results: We followed up 96 consecutive patients with CAD undergoing ICD implantation for secondary prevention of SCD. Long-term rates and clinical predictors of appropriate ICD therapy (ICD-Tx) for VA were analyzed. Appropriate ICD-Tx occurred in 41 (42.7%) patients during a median follow-up of 2.4 years (interquartile range, 0.9-6.1). These patients had significantly greater left ventricular end-diastolic diameter (62.3±1.3 vs. 54.6±1.1 mm, P<0.001), lower left ventricular ejection fraction (LVEF; 36.3±2.0% vs. 45.7±1.8%, P<0.001), and more incomplete revascularization (ICR; 70.7% vs. 45.5%, P=0.014) than those without appropriate ICD-Tx. Multivariable analysis showed that LVEF (hazards ratio [HR], 0.950; 95% CI: 0.925-0.975; P<0.001) and ICR (HR, 2.293; 95% CI: 1.133-4.637; P=0.021) were significant predictors of appropriate ICD-Tx for VA., Conclusions: Lower LVEF and ICR were independent predictors of recurrent VA in secondary prevention ICD recipients with CAD.

Aims: To confirm the presence of tachycardia-induced slur or notch in the terminal portion of the QRS complexes in a general patient population., Methods and Results: A tachycardia-induced J wave was defined as a slur or notch in the terminal portion of the QRS complexes newly induced at short RR intervals during atrial premature contractions (APCs) or atrial electrical stimulation in the electrophysiological study (EPS). Twenty-three out of 2000 patients with general diseases were involved. All patients with aborted sudden cardiac death, ventricular fibrillation or a family history of sudden cardiac death were excluded. The mean age was 72 ± 9 years, and 11 patients were male (47.8%). When the RR interval was shortened from 821 ± 142 ms to 464 ± 52 ms in the conducted APCs (P < 0.0001), J waves became diagnostic (0.02 ± 0.03 mV to 0.20 ± 0.07 mV, P < 0.0001). J waves were confined to the inferior leads in 22 (95.7%) patients and were notched in 11 (47.8%) and slurred in 12 (52.2%) patients. The induction of J waves was accompanied by visible changes of the QRS morphology. When the post-APC RR interval was prolonged to 992 ± 305 ms (P = 0.0154 vs. baseline), the J waves were similar to baseline levels. During the EPS, J wave induction was confirmed during atrial stimulation. There were no characteristic clinical or ECG features in the patients with tachycardia-induced J waves., Conclusions: J waves can be newly induced by short RR intervals in a general patient population, and a conduction delay is the likely mechanism causing such J waves.

Background: Glycosuria produced by sodium-glucose co-transporter-2 (SGLT-2) inhibitors is associated with weight loss. SGLT-2 inhibitors reportedly might reduce the occurrence of cardiovascular events. Epicardial adipose tissue (EAT) is a pathogenic fat depot that may be associated with coronary atherosclerosis. The present study evaluated the relationship between an SGLT-2 inhibitor (dapagliflozin) and EAT volume., Methods: In 40 diabetes mellitus patients with coronary artery disease (10 women and 30 men; mean age of all 40 patients was 67.2 ± 5.4 years), EAT volume was compared prospectively between the dapagliflozin treatment group (DG; n = 20) and conventional treatment group (CTG; n = 20) during a 6-month period. EAT was defined as any pixel that had computed tomography attenuation of - 150 to - 30 Hounsfield units within the pericardial sac. Metabolic parameters, including HbA1c, tumor necrotic factor-α (TNF-α), and plasminogen activator inhibitor-1 (PAI-1) levels, were measured at both baseline and 6-months thereafter., Results: There were no significant differences at baseline of EAT volume and HbA1c, PAI-1, and TNF-α levels between the two treatment groups. After a 6-month follow-up, the change in HbA1c levels in the DG decreased significantly from 7.2 to 6.8%, while body weight decreased significantly in the DG compared with the CTG (- 2.9 ± 3.4 vs. 0.2 ± 2.4 kg, p = 0.01). At the 6-month follow-up, serum PAI-1 levels tended to decline in the DG. In addition, the change in the TNF-α level in the DG was significantly greater than that in the CTG (- 0.5 ± 0.7 vs. 0.03 ± 0.3 pg/ml, p = 0.03). Furthermore, EAT volume significantly decreased in the DG at the 6-month follow-up compared with the CTG (- 16.4 ± 8.3 vs. 4.7 ± 8.8 cm 3 , p = 0.01). Not only the changes in the EAT volume and body weight, but also those in the EAT volume and TNF-α level, showed significantly positive correlation., Conclusion: Treatment with dapagliflozin might improve systemic metabolic parameters and decrease the EAT volume in diabetes mellitus patients, possibly contributing to risk reduction in cardiovascular events.

Purpose: The purpose of this study is to identify the current clinical features of neonatal gastrointestinal perforation in Japan., Methods: A questionnaire about cases of neonatal gastrointestinal perforation treated in recent 5 years was sent to participating institutions of the Japanese Society of Pediatric Surgeons (JSPS)., Results: Five hundred and thirty-six neonates with gastrointestinal perforation were treated. They consisted of 42 patients with gastric rupture/perforation (GR), 33 patients with intestinal atresia/stenosis (IA), 3 patients with malrotation (ML), 118 patients with necrotizing enterocolitis (NEC), 160 patients with focal intestinal perforation (FIP), 46 patients with meconium-related ileus (MRI), 77 patients with meconium peritonitis (MP), and 57 patients with other conditions. The total mortality rate was 20.5 %. The mortality rates of the patients with GR, IA, ML, NEC, FIP, MRI, and MP were 9.5, 9.1, 0, 33.1, 20.6, 28.2, and 9.1 %, respectively. In 263 cases involving extremely low-birth-weight neonates (ELBW), 108 died (mortality rate 41.1 %). The mortality rates for ELBW with GR, NEC, FIP, MRI, MP, and other conditions were 27.3 % (3/11), 58.5 % (48/82), 21.6 % (24/111), 70.6 % (24/34), 57.1 % (4/7), and 27.8 % (5/18), respectively., Conclusions: The mortality rates for ELBW decreased from 62.8 % in the previous survey to 41.1 % by the time of this survey.

A 77-year-old woman developed dyspnea over three years which occurred during sitting, standing or walking. Her physical examination, chest X-ray, ECG and cardiac catheterization results were all normal. A marked fall in arterial oxygen saturation was observed on sitting or standing. Transesophageal echocardiography showed an increase of right to left shunt flow on sitting. The patient was diagnosed with platypnea-orthodeoxia syndrome and underwent the surgical closure of an atrial septal defect of 19 mm in diameter. After the surgery, the patient's POS symptoms were completely resolved. She was discharged and followed at the outpatient clinic. Her post-treatment course was uneventful.

Conscious sedation with propofol sometimes causes amnesia while keeping the patient awake. However, it remains unknown how propofol compromises the memory function. Therefore, we investigated the changes in brain activation induced by visual stimulation during and after conscious sedation with propofol using serial functional MRI. Healthy volunteers received a target-controlled infusion of propofol, and underwent functional MRI scans with a block-design paradigm of visual stimulus before, during, and after conscious sedation. Random-effect model analyses were performed using Statistical Parametric Mapping software. Among the areas showing significant activation in response to the visual stimulus, the visual cortex and fusiform gyrus were significantly suppressed in the sedation session and tended to recover in the early-recovery session of ∼20 min (P<0.001, uncorrected). In contrast, decreased activations of the hippocampus, thalamus, inferior frontal cortex (ventrolateral prefrontal cortex), and cerebellum were maintained during the sedation and early-recovery sessions (P<0.001, uncorrected) and were recovered in the late-recovery session of ∼40 min. Temporal changes in the signals from these areas varied in a manner comparable to that described by the random-effect model analysis (P<0.05, corrected). In conclusion, conscious sedation with propofol may cause prolonged suppression of the activation of memory-related structures, such as the hippocampus, during the early-recovery period, which may lead to transient amnesia.

The neuroradiological findings and its outcomes of intracerebral hemorrhage (ICH) were compared between the non-vitamin K antagonist oral anticoagulant (NOAC) therapy and warfarin therapy. In the latest 3 years, 13 cases of nonvalvular atrial fibrillation on NOAC therapy were admitted for ICH. For comparison, 65 age- and gender-comparable patients with ICH on warfarin therapy were recruited. Three NOACs had been prescribed: dabigatran (n = 4), rivaroxaban (n = 2), and apixaban (n = 7). The average ages were 76 ± 9 and 78 ± 8 years in the warfarin (n = 65) and NOAC groups (n = 13), respectively. There was no difference in the clinical features, including the CHADS2 score or HAS-BLED score: 2.62 ± 1.31 versus 2.62 ± 1.33, or 1.09 ± 0.43 versus 1.00 ± 0.41, for the warfarin and NOAC groups, respectively. The volume of ICH <30 ml was found in 84.6% of the patients on NOACs, but it was found in 53.8% of the patients on warfarin (p = 0.0106). The expansion of hematoma was limited to 7 patients (10.8%) of the warfarin group. A lower hospital mortality and better modified Rankin Scale were observed in the NOAC group than in the warfarin group: 1 (7.7%) versus 27 (41.5%; p = 0.0105) and 3.2 ± 1.4 versus 4.5 ± 1.6 (p = 0.0057), respectively. In conclusion, ICH on NOAC therapy had smaller volume of hematoma with reduced rate of expansion and decreased mortality compared with its occurrence on warfarin., (Copyright © 2016 Elsevier Inc. All rights reserved.)

A 21-year-old man developed ventricular fibrillation (VF) while drinking alcohol and was admitted to our hospital. An electrocardiogram (ECG) on admission revealed remarkably prominent slurs on the terminal part of QRS complexes in the left precordial leads and a coved type ST elevation at higher intercostal spaces. After hypothermia therapy, he underwent implantation of an implantable cardioverter-defibrillator (ICD). Standard twelve-lead follow-up ECGs revealed early repolarization pattern and an intermittent coved type ST elevation. When the coved type ST elevation appeared, the early repolarization pattern in the inferior and left precordial leads was attenuated. Prominent early repolarization pattern was the most likely trigger of the VF storm in this Brugada patient., (Copyright © 2016 Elsevier Inc. All rights reserved.)

Background: Electrical alternans (EA) has not been fully studied in the current percutaneous coronary intervention (PCI) procedure., Objective: The purpose of this study was to evaluate visible EA and the morphology of ST segment during PCI., Methods: The incidence of visible EA and ST-segment morphology were studied while the coronary artery was occluded for 20 seconds. When data were available, the relationship between EA and blood pressure was analyzed. The clinical and electrocardiographic data were compared with those of the age- and sex-matched controls., Results: During balloon inflation, visible EA was observed in 5 of 306 patients (1.6%) in the last 2 years. EA was limited to PCI in the proximal left anterior descending artery. The ST segment elevated to 10.1 ± 3.2 mm, followed by an alternating QRS complex with a lower ST segment (5.6 ± 1.9 mm; P = .0047) with characteristic ST-segment morphology, which is known as lambda pattern. The mean age of the 5 patients was 68 ± 20 years, and 4(80%). were men. After the release of inflation, the ST-segment level returned rapidly to baseline, followed by normalization of J point. Compared with controls, the maximal elevated ST segment was significantly higher in patients with EA (5.7 ± 2.7 mm; P = .0028). The occlusion of the proximal left anterior descending artery with more severe ischemia seemed to be a prerequisite for developing EA. A higher ST segment was associated with a lower blood pressure and vice versa., Conclusion: A short period of ischemia during PCI may induce visible EA and alternating QRS complexes with a characteristic ST-segment morphology. A higher ST segment was associated with a lower blood pressure and vice versa., (Copyright © 2015 Heart Rhythm Society. Published by Elsevier Inc. All rights reserved.)

Purpose: Lidocaine has a biphasic action on smooth muscle of peripheral blood vessels, with vasoconstriction at low concentrations and vasodilation at higher concentrations. Many in vivo studies have demonstrated the effects of lidocaine on aortic or coronary arteries in several animals, but there are few reports about the effect on peripheral vessels. This study was designed to investigate the direct effects of lidocaine on peripheral vessels, namely swine lingual and pulmonary arterial rings., Methods: Swine lingual artery and pulmonary artery segments, about 2-3 mm in diameter, were cut into 3-mm-long rings, and the lumen surface was gently rubbed to remove the endothelium. Isometric tension was measured using a displacement transducer and recorded. After a stable constriction was developed with 5 µM noradrenaline, 5 µM noradrenaline containing lidocaine (0.5, 1.0, 10, 20, 50 or 100 µg/ml) was perfused for 5 min, and then all drug perfusion was stopped. The strength of any isometric tension during an experiment was normalized to the strength of the isometric tension immediately before lidocaine perfusion, and expressed as a percentage., Results: Lidocaine elicited a concentration-dependent biphasic response of lingual and pulmonary arterial rings. The lidocaine concentration at 1 µg/ml caused mild contraction. Dilation occurred at 10 µg/ml and increased with increasing dose., Conclusions: Lidocaine-induced vasoconstriction of swine lingual and pulmonary smooth muscle may occur at low concentration when lidocaine is infiltrated into the oral submucosa or administered intravenously for the treatment of ventricular arrhythmia.

Background: Although mepivacaine has a known biphasic action on the aortic and coronary artery in several animal species, its effects on the lingual and pulmonary artery are not well understood and it is not yet known whether mepivacaine produces vasoconstriction in these vessels. The present study aims to investigate the direct effects of mepivacaine on swine lingual, pulmonary and coronary arterial endothelium-denuded rings., Methods: Artery rings were perfused with isotonic 40 mM KCl until a stable constricted plateau was reached. The rings were then perfused with isotonic 40 mM KCl plus a particular concentration of mepivacaine (0.4 μM, 4.0 μM, 40 μM, 0.4 mM and 4.0 mM). The isometric tension strengths in each experiment were normalized to the strength of the isometric tension immediately before mepivacaine perfusion and expressed as a percentage., Results: Mepivacaine at 0.4 to 40 μM did not significantly alter 40 mM KCl-induced contraction in the lingual, pulmonary and coronary arterial rings. In contrast, mepivacaine at 4 mM produced attenuated vasoconstriction in the lingual, pulmonary and coronary arterial compared with isotonic 40 mM KCl., Conclusions: Mepivacaine produced vasoconstriction at lower concentrations, followed by attenuated vasoconstriction at higher concentrations on swine lingual, pulmonary and coronary arterial endothelium-denuded rings. Mepivacaine (4 μM) appeared to increase isotonic 40 mM KCl-induced contraction, followed by attenuated vasoconstriction at 4 mM. Dentists using 3 % mepivacaine should take into consideration that the risk of complications may be increased if more than six mepivacaine cartridges are used in dental treatment or minor surgery, or if over 15 ml of mepivacaine is administered to a patient with cardiovascular complications during general anesthesia for oral maxillofacial surgery.

Background: J waves can be observed in individuals of the general population, but electrocardiographic characteristics are poorly understood., Objective: The purpose of this study was to examine the J-wave dynamicity in a general patient population., Methods: The responses of J waves (>0.1 mV above the isoelectric line in 2 contiguous leads) to varying RR intervals were analyzed. Patients with aborted sudden cardiac death, documented ventricular fibrillation, or a family history of sudden cardiac death were excluded. The J-wave amplitude was measured at baseline, in beats with short RR intervals in conducted atrial premature beats (APBs) or atrial stimulation during the electrophysiology study, and in the beats next to APBs with prolonged RR intervals., Results: Mainly notched J waves were identified in 94 of 701 (24.5%) general patients (13.4%), and APBs were present in 23 of 94 (24.5%) patients. The mean baseline amplitude of J waves was 0.20 ± 0.06 mV at the baseline RR interval of 853 ± 152 ms, 0.25 ± 0.11 mV at the RR interval in the conducted APB of 545 ± 133 ms (P = .0018), and 0.19 ± 0.08 mV at the RR interval of 1146 ± 314 ms (P = .3102). The clinical characteristics were not different between patients with and without tachycardia-dependent augmentation of J waves. Augmentation of J waves was confirmed by the electrophysiology study: 0.28 ± 0.12 mV vs 0.42 ± 0.11 mV at baseline and in the beats of atrial stimulation, respectively (P = .0001). However, no bradycardia-dependent augmentation (>0.05 mV) was observed. Such tachycardia-dependent augmentation can represent depolarization abnormality rather than repolarization abnormality., Conclusion: J waves in a general patient population were augmented at shorter RR intervals, but not at prolonged RR intervals. Mechanistically, conduction delay is most likely responsible for this., (Copyright © 2015 Heart Rhythm Society. Published by Elsevier Inc. All rights reserved.)

A 66-year-old man was admitted for congestive heart failure with tachycardiac atrial fibrillation (AF). Heart failure was improved by diuretics, but control of heart rate by verapamil was insufficient, and bisoprolol was prescribed. After taking 2.5 mg of bisoprolol, he developed a general malaise followed by sinus bradycardia and shock. In addition to catecholamines, the patient was treated with intra-aortic balloon pumping and a pacemaker. With intensive therapy, the general condition was improved, and acute elevation of liver enzymes after bisoprolol was normalized by the 17th hospital day. The blood sample taken 30 h after the intake of bisoprolol showed abnormally high levels. Although the patient was CYP2D6*10 heterozygote, the precise mechanism for excess accumulation of bisoprolol and refractory shock remains unknown. < Learning objective: Bisoprolol has been used for heart failure, but it may need caution to avoid hemodynamic deterioration. In our case, refractory shock and acute liver injury were induced by bisoprolol. The blood concentration was excessively high. The patient was CYP2D6*10 heterozygote, but the precise mechanism and shock are to be studied.>.

A 41-year-old man developed sustained monomorphic ventricular tachycardia (VT) with a left bundle branch block and inferior axis pattern during treadmill exercise concomitantly with unmasking of the typical Brugada electrocardiography (ECG) pattern. The typical ECG phenotype was provoked by a class IC drug. VT was not inducible with programmed electrical stimulation, but premature ventricular beat and non-sustained VT with the same morphology increased in frequency with isoproterenol treatment. Additionally, atrioventricular nodal reentrant tachycardia (AVNRT) was induced by electrical stimulation and VT and AVNRT were treated by radiofrequency catheter ablation.

Background: In the revised diagnostic criteria for pancreaticobiliary maljunction (PBM), PBM can be diagnosed from a long common channel by magnetic resonance cholangiopancreatography (MRCP). However, it is necessary to differentiate from high confluence of pancreaticobiliary ducts (HCPBD) with a relatively long common channel (≥6 mm) and effect of the sphincter in the pancreaticobiliary junction. This multicenter study aimed to explore definite value of the length of an abnormally long common channel, which enables to distinguish PBM from HCPBD on MRCP., Methods: In 184 PBM patients and 22 HCPBD patients who were diagnosed by direct cholangiography and underwent MRCP, the length of the common channel was measured., Results: The length of the common channel was 16.2 ± 6.9 mm on direct cholangiography and 13.9 ± 6.2 mm on MRCP in PBM patients, and 7.7 ± 1.5 mm and 6.6 ± 1.4 mm in HCPBD patients. The optimal cut off value of the length of the common channel to distinguish PBM from HCPBD was identified to be 9 mm., Conclusions: Pancreaticobiliary maljunction can be diagnosed from an abnormally long common channel on MRCP, but in cases of the common channel ≤9 mm on MRCP, direct cholangiography is needed to confirm PBM., (© 2014 Japanese Society of Hepato-Biliary-Pancreatic Surgery.)

Background: The circadian pattern of ventricular fibrillation (VF) episodes in patients with idiopathic ventricular fibrillation (IVF) is poorly understood., Objective: The purpose of this study was to assess the circadian pattern of VF occurrence in patients with IVF., Methods: Excluding Brugada syndrome and other primary electrical diseases, the circadian pattern of VF occurrence was determined in 64 patients with IVF. The clinical and electrocardiographic characteristics were compared among patients with nocturnal (midnight to 6:00 AM) VF and nonnocturnal VF in relation to J waves. A J wave was defined as either notching or a slur at the QRS terminal >0.1 mV above the isoelectric line in contiguous leads., Results: The overall distribution pattern of VF occurrence showed 2 peaks at approximately 6:00 AM and around 8:00 PM. Nocturnal VF was observed in 20 patients (31.3%), and J waves were present in 14 of these 20 individuals (70.0%), whereas J waves were less frequent in the 44 nonnocturnal patients with VF: 16 (36.4%) (P = .0117). Among patients with J waves, nocturnal VF was observed in 46.7% with a peak at approximately 4:00 AM. Nocturnal VF was less common in patients without J waves, occurring in only 17.6% (P = .0124). Both the type and location of J waves and the pattern of the ST segment were similar between the nocturnal and nonnocturnal VF groups. J waves were associated with a VF storm and long-term arrhythmia recurrence., Conclusion: In IVF, the presence of J waves may characterize a higher nocturnal incidence of VF and a higher acute and chronic risk of recurrence., (Copyright © 2014 Heart Rhythm Society. Published by Elsevier Inc. All rights reserved.)

We present the first case report of laparoscopic Roux-en-Y duodenojejunostomy utilizing minimally invasive surgery to treat superior mesenteric artery syndrome in a child. A 6-year-old girl presented with a 3-year history of intermittent postprandial epigastric pain and anorexia. An upper gastrointestinal series revealed dilatation of the first and second parts of the duodenum and an abrupt vertical cut-off at the third portion. Despite conservative treatment for 7 weeks, there was no resolution of her symptoms, so the decision was made to proceed with a laparoscopic Roux-en-Y duodenojejunostomy. On follow-up 9 months later, there had been no recurrence of her symptoms., (© 2014 Japan Society for Endoscopic Surgery, Asia Endosurgery Task Force and Wiley Publishing Asia Pty Ltd.)

The patient was a 33-year-old male. Twenty years ago, he underwent radiofrequency catheter ablation for idiopathic sustained monomorphic ventricular tachycardia (VT) with an RBBB and superior axis pattern. The VT was inducible by programmed stimulation and entrained by rapid pacing. At this presentation, he developed palpitation and VT with the same morphology at the peak exercise on a treadmill with appearance of typical ECG pattern for Brugada syndrome (BrS). Pilsicainide induced the typical ECG pattern and premature ventricular beats (PVBs) of the same morphology as VT. The relationship between BrS and VT of left ventricular origin was discussed., (Copyright © 2014 Elsevier Inc. All rights reserved.)

Introduction: The aim of this study was to evaluate differences in postoperative pain between single-incision laparoscopic appendectomy (SLA) and conventional laparoscopic appendectomy (CLA) for uncomplicated appendicitis in children., Methods: In total, 30 patients underwent CLA, and 12 patients underwent SLA. Patients with perforated appendicitis or an abscess were excluded. We evaluated the length of hospital stay, the frequency of postoperative analgesic requirement, and the duration of postoperative pain., Results: The mean length of hospital stay was 3.67 ± 0.75 days for the CLA group and 4.0 ± 0.70 days for the SLA group. The mean frequency of postoperative analgesic requirement was 1.93 ± 1.63 times for the CLA group and 2.00 ± 1.00 times for the SLA group. The mean duration of postoperative pain was 52.63 ± 20.82 hours for the CLA group and 55.91 ± 18.45 hours for the SLA group. These postoperative outcomes were similar between the two groups., Conclusion: Our study suggests that SLA, which results in similar postoperative pain as CLA, is a feasible technique for uncomplicated appendicitis in children., (© 2014 Japan Society for Endoscopic Surgery, Asia Endosurgery Task Force and Wiley Publishing Asia Pty Ltd.)

Background: Anticoagulation therapy is essential in atrial fibrillation (AF), and in Japan, less intense control is popular., Purpose: To assess the efficacy and safety with a special reference to low intensity warfarin therapy., Subjects and Methods: In 488 out of 508 patients with non-valvular AF, prothrombin time-international normalized ratio (PT-INR) was kept at 1.6-2.59, and they were followed for 49.5 months: 2098 person-years. The mean age was 73.7±9.9 years and 62% were male. The patients were divided by age: ≥70 years and <70 years, and by the intensity of warfarin therapy: PT-INR at 1.6-1.99 and at 2.0-2.59, respectively. The clinical data and event rates, ischemic stroke and major bleeding, were compared among the subgroups., Results: Heart failure, previous stroke, and higher CHADS2 score were more often reported in patients ≥70 years while males were involved more often as younger patients. A total of 166 of 339 patients ≥70 years and 69 of 149 patients <70 years belonged to the low intensity group. Ischemic stroke and major bleeding occurred in 1.47%/year and 1.27%/year, respectively but there was no difference between the two age groups and between the two intensities of warfarin therapy. Time in therapeutic range was a predictor for ischemic stroke. A fall of PT-INR to <1.6 was found in 41.9% with ischemic stroke and a rise >2.61 in 40.0% with major bleeding at the time of the events. Blunt trauma and concomitant use of antiplatelets were risks for intracranial hemorrhage in the patients ≥70 years., Conclusions: The event rates were similar between the low- (1.6-1.99) and high- (2.0-2.59) intensity warfarin therapy groups in aged patients: <70 years and ≥70 years. Time in therapeutic range and a transient fall or rise in PT-INR were risks for clinical events. Blunt head trauma and concomitant use of antiplatelets were risks for intracranial hemorrhage., (Copyright © 2014. Published by Elsevier Ltd.)

A 64-year-old woman underwent a coronary flow reserve evaluation using intracoronary-administered papaverine into the left anterior descending artery. Her baseline electrocardiogram (ECG) was normal, but toward the end of papaverine administration, the QTU intervals were excessively prolonged and torsade de pointes occurred, leading to ventricular fibrillation. Ten months previously, the patient's ECG showed mildly prolonged QTc (480 ms(1/2)), which normalized after the cessation of bepridil. This case report suggests that a history of drug-induced QT prolongation can be a risk factor for papaverine-induced fatal ventricular arrhythmia.

Dumping syndrome is infrequently reported, but known to occur after Nissen fundoplication in children. However, it may be difficult both to diagnose and manage. Here we presented four infants who received Nissen fundoplication for severe gastroesophageal reflux disease, two of whom developed dumping syndrome whilst the other two did not. Continuous glucose monitoring (CGM) was very useful to clearly detect large glycemic fluctuation around each feeding. CGM was also helpful to prove the effect of treatment to avoid abnormal glucose levels. We believe that dumping syndrome in children may be underdiagnosed if clinicians rely solely on the recognition of symptoms or limited frequency of blood samplings. CGM might be the most sensitive diagnostic tool., (© 2013 The Authors. Pediatrics International © 2013 Japan Pediatric Society.)

Objectives: This study sought to characterize patients with idiopathic ventricular fibrillation (IVF) who develop electrical storms., Background: Some IVF patients develop ventricular fibrillation (VF) storms, but the characteristics of these patients are poorly known., Methods: Ninety-one IVF patients (86% male) were selected after the exclusion of structural heart diseases, primary electrical diseases, and coronary spasm. Electrocardiogram features were compared between the patients with and without electrical storms. A VF storm was defined as VF occurring ≥3 times in 24 h and J waves >0.1 mV above the isoelectric line in contiguous leads., Results: Fourteen (15.4%) patients had VF storms occurring out-of-hospital at night or in the early morning. J waves were more closely associated with VF storms compared to patients without VF storms: 92.9% versus 36.4% (p < 0.0001). VF storms were controlled by intravenous isoproterenol, which attenuated the J-wave amplitude. After the subsidence of VF storms, the J waves decreased to the nondiagnostic level during the entire follow-up period. Implantable cardioverter-defibrillator therapy was administered to all patients during follow-up. Quinidine therapy was limited, but the patients on disopyramide (n = 3), bepridil (n = 1), or isoprenaline (n = 1) were free from VF recurrence, while VF recurred in 5 of the 9 patients who were not given antiarrhythmic drugs., Conclusions: The VF storms in the IVF patients were highly associated with J waves that showed augmentation prior to the VF onset. Isoproterenol was effective in controlling VF and attenuated the J waves, which diminished to below the diagnostic level during follow-up. VF recurred in patients followed up without antiarrhythmic agents., (Copyright © 2013 American College of Cardiology Foundation. Published by Elsevier Inc. All rights reserved.)

Background: The characteristic ECG of Brugada syndrome (BS) can be masked by complete right bundle-branch block (CRBBB) and exposed by resolution of the block or pharmacological or pacing maneuvers., Methods and Results: The study consisted of 11 patients who had BS and CRBBB. BS was diagnosed before the development of CRBBB, on the resolution of CRBBB, or from new characteristic ST-segment changes that could be attributable to BS. Structural heart diseases were excluded, and coronary spasm was excluded on the basis of a provocation test at catheterization. In 7 patients, BS was diagnosed before the development of CRBBB. BS was diagnosed when CRBBB resolved spontaneously (n=1) or by right ventricular pacing (n=3). The precipitating cause for the spontaneous resolution of CRBBB, however, was not apparent. On repeated ECGs, new additional upward-convex ST-segment elevation was found in V2 or V3 in 3 patients. In 2 patients, new ST-segment elevation was induced by class IC drugs. The QRS duration was more prolonged in patients with BS and CRBBB compared with age- and sex-matched controls: 170±13 versus 145±15 milliseconds in V1 and 144±19 versus 128±7 milliseconds in V5 (both P<0.0001). The amplitude of R in V1 was smaller [corrected] in the BS patients than in the control subjects (P=0.0323), but that of R' was similar (P=0.0560)., Conclusions: BS can coexist behind CRBBB, and CRBBB can completely mask BS. BS might be demonstrated by relief of CRBBB or by spontaneous or drug-induced ST-segment elevation. The prevalence, mechanism, and clinical significance of a combination of CRBBB and BS are yet to be determined.

Controversies concerning the association between obesity and acute myocardial infarction (AMI) are still ongoing in Japan. We investigated the association between obesity defined by body mass index of 25 kg/m(2) or higher and AMI by a case-control study using data from 1199 AMI cases and 4056 apparently healthy controls. The analysis was performed in age- and sex-matched samples of 621 case-control pairs younger than 80 years and in crude samples aged 40-79 years divided into 10-year age groups. Prevalence of obesity, diabetes, current smoking, hypertension, and hypercholesterolemia were compared between cases and controls, and a multivariable odds ratio (OR) of AMI was calculated for each risk factor in various age groups. The OR (95 % confidence interval (CI)) of AMI for obesity was 1.63 (1.23-2.17), P = 0.0008 in men younger than 80 years; 2.65 (1.41-5.00), P = 0.0025 in women younger than 80 years; 2.23 (1.46-3.41), P = 0.0002 in men aged 59 years or younger; 1.34 (0.90-2.01), P = 0.1510 in men aged 60-79 years; and 2.98 (1.56-5.71), P = 0.0010 in women aged 60-79 years using paired samples. The OR (95 % CI) of AMI for obesity was 4.92 (2.53-9.58), P < 0.0001 in men aged 40-49 years; 1.54 (1.07-2.21), P = 0.0197 in men aged 50-59 years; 1.07 (0.69-1.66), P = 0.7717 in men aged 60-69 years; 2.24 (1.20-4.20), P = 0.0118 in men aged 70-79 years; 2.48 (1.12-5.48), P = 0.0245 in women aged 60-69 years; and 3.05 (1.46-6.37), P = 0.0029 in women aged 70-79 years using crude samples. The association between obesity and AMI was age- and gender-dependent in a Japanese population.

Aims: The prevalence, clinical significance, and pathogenesis of J-waves were studied in the patients with an ST-elevation myocardial infarction (MI) after percutaneous coronary intervention (PCI)., Methods and Results: One hundred and fifty-two consecutive patients with an acute ST-elevation MI were included. The mean age was 68.6 ± 13.5 years, and 78.3% of the patients were male. Following successful PCI, 12-lead electrocardiograms (ECGs) were monitored, and J-waves were measured 1 week after the MI and analysed in relation to the location of the MI and arrhythmias. Clinical and ECG parameters were compared between the groups with and without J-waves. The rate dependency of the J-wave amplitude was analysed in the conducted atrial premature beats (APBs). J-waves were present in 60.5% (≥0.1 mV) or 48.9% (≥0.2 mV) of the 152 patients. The J-waves were more often located in the inferior leads and more frequently in an inferior MI. The presence of J-waves was associated with ventricular arrhythmias, including ventricular fibrillation. The J-wave amplitude increased in the conducted APB, mechanistically suggesting a phase 3 block., Conclusion: Many patients in the early recovery phase after an acute MI had J-waves. This ECG phenomenon was associated with an increased incidence of ventricular arrhythmias. The tachycardia-dependent augmentation of the J-wave amplitude suggested a mechanistic role of conduction delay.

Single-incision laparoscopic surgery (SILS) has been performed on children for various procedures. However, few reports are available about SILS for small bowel resection, particularly involving conventional instruments in the pediatric population. Herein, we report four cases of small bowel resection with single umbilical incision and a three-trocar approach. From October 2010 to September 2011, we performed small bowel resection with SILS on four cases, including a boy with an intestinal duplication cyst and three children with Meckel's diverticulum. An intraumbilical arcuate incision was made to expose the abdominal wall fascia, and one 5-mm and two 3.5-mm trocars were inserted. It was not necessary to extend the initial incision to exteriorize the lesion except in one case in which we applied the so-called Y-V closure plasty. All procedures were successful and did not require conversion, and all patients recovered smoothly without any complications. Small bowel resection using the SILS approach is suitable for these diseases., (© 2012 Japan Society for Endoscopic Surgery, Asia Endosurgery Task Force and Wiley Publishing Asia Pty Ltd.)

A 69-year-old man without structural heart disease was admitted for syncope. His electrocardiogram displayed complete right bundle branch (CRBBB). A coved type ST elevation was observed with transient normalization of CRBBB giving rise to a normal QRS. These findings suggest that Brugada syndrome can be masked by CRBBB., (Copyright © 2012 Elsevier Inc. All rights reserved.)

Transumbilical surgical procedures have been reported to be a feasible, safe, and cosmetically excellent procedure for various pediatric surgical diseases. Umbilical loop colostomies have previously been created in patients with Hirschsprung's disease, but not in patients with anorectal malformations (ARMs). We assessed the feasibility and cosmetic results of temporal umbilical loop colostomy (TULC) in patients with ARMs. A circumferential skin incision was made at the base of the umbilical cord under general anesthesia. The skin, subcutaneous tissue, and fascia were cored out vertically, and the umbilical vessels and urachal remnant were individually ligated apart from the opening in the fascia. A loop colostomy was created in double-barreled fashion with a high chimney more than 2 cm above the level of the skin. The final size of the opening in the skin and fascia was modified according to the size of the bowel. The bowel wall was fixed separately to the peritoneum and fascia with interrupted 5-0 absorbable sutures. The bowel was opened longitudinally and everted without suturing to the skin. The loop was divided 7 days postoperatively, and diversion of the oral bowel was completed. The colostomy was closed 2-3 months after posterior saggital anorectoplasty through a peristomal skin incision followed by end-to-end anastomosis. Final wound closure was performed in a semi-opened fashion to create a deep umbilicus. TULCs were successfully created in seven infants with rectourethral bulbar fistula or rectovestibular fistula. Postoperative complications included mucosal prolapse in one case. No wound infection or spontaneous umbilical ring narrowing was observed. Skin problems were minimal, and stoma care could easily be performed by attaching stoma bag. Healing of umbilical wounds after TULC closure was excellent. The umbilicus may be an alternative stoma site for temporary loop colostomy in infants with intermediate-type anorectal malformations, who undergo radical anorectoplasty via a non-abdominal approach.

Background: Apart from the severity of myocardial infarction and coronary artery disease, several predictors of in-hospital death (In-HD) are suggested in patients with acute myocardial infarction (AMI)., Methods: We investigated predictors of In-HD and ventricular rupture (VR) including ventricular septal rupture (VSR) and free wall rupture (FWR) with stepwise multivariable logistic regressions in 1,042 patients admitted to our Cardiovascular Center within 48 hours from symptom onset for the first attack of AMI., Results: In-HD, VSR, and FWR were observed in 78 cases (7.5%), 14 cases of which 13 cases were In-HD, and 13 cases of which 6 cases were In-HD, respectively. Apart from the disease severity, age and renal dysfunction (RD) defined by estimated glomerular filtration rate of lower than 60 mL/min/ 1.73 m 2 were independent positive predictors of In-HD (the odds ratios (ORs) (95% confidence interval (CI)): 1.04 (1.01 - 1.06) P = 0.0069 and 5.75 (3.12 - 10.59) P < 0.0001, respectively) and hypercholesterolemia was an independent negative predictor for In-HD (OR (95% CI): 0.34 (0.17 - 0.67) P = 0.0017). After including the categories of coronary disease, ventricular rupture, and ejection fraction in predictors, RD remained an independent predictor of In-HD (OR (95% CI): 6.65 (2.67 - 16.60) P < 0.0001). Age (OR (95% CI): 1.07 (1.02 - 1.12) P = 0.0064), RD (OR (95% CI): 2.77 (1.18 - 6.49) P = 0.019), and diabetes (OR (95% CI): 2.52 (1.12 - 5.71) P = 0.026) were independent predictors of VR., Conclusions: RD was an independent predictor of In-HD and VR in patients with initial AMI.

We encountered two consecutive cases with spontaneous ST elevation due to right coronary spasms and subsequent ventricular fibrillation (VF). Their 12-lead ECGs on anterior chest pain showed elevation of ST-segments in the inferior leads, but coronary angiography (CAG) revealed no significant stenosis. Both cases showed dramatically evolving J waves in the inferior leads during the right CAG, but it was not observed during angiography of the left CAG. Neither Brugada-type ECG nor long-QT was evident. In summary, J waves can be produced without ST-segment elevation, and contrast media-induced J waves might be related to the arrhythmogenesis of subsequent VF evoked by right coronary spasms.

Objective: Papaverine is used for the evaluation of functional status of the coronary arteries but it may provoke severe ventricular tachyarrhythmias (VTAs). This study compared the clinical and ECG characteristic of patients with papaverine-induced VTAs., Materials and Methods: The study involved 25 patients who underwent a fractional flow reserve (FFR) study. FFR was determined as the ratio of blood pressure at the distal and the proximal site of stenosis after intracoronary papaverine administration at 12 mg into the left and 8 mg into the right coronary artery. The QT and QTU intervals were measured manually in the limb leads and in the precordial leads, respectively and corrected by the R-R interval to obtain QTc and QTUc. The clinical and ECG data were compared between the patient groups with and without VTAs., Results: After papaverine administration into the left (20), right (3) or both coronary arteries (2), the RR interval shortened, but non-significantly however, the QT interval (and QTc) and the QTU interval (and QTUc) were significantly prolonged. VTAs developed in four women: torsade de pointes in 3 followed by ventricular fibrillation and ventricular premature beats in 1 patient. After papaverine administration, QTU and QTUc were more prolonged in women than men and in patients with VTAs compared to those without. Just prior to VTAs, giant T-U waves were observed., Conclusion: Intracoronary papaverine was used to determine FFR which may induce VTAs. VTAs developed only in women and they were closely related to prolongation of the QTU intervals with prominent T-U waves.

A 67-year-old woman was admitted for severe abdominal pain (stomach ache). Computed tomography (CT) revealed gas along the abdominal aortic wall. A blood culture was positive for Salmonella dublin, a gram-negative bacillus that is rare in humans. Treatment with an antibiotic improved the inflammatory signs; however, on the 11th hospital day, the patient complained of sudden severe abdominal pain. Enhanced CT revealed a pseudoaneurysm surrounded by a periaortic abscess. The infected aortic wall, including the aneurysm, was resected and an extra-anatomic bypass was constructed between the axillary artery and the external iliac arteries. The patient recovered fully and her course has been uneventful for the past two years since her discharge.

Background: Recently, we and others reported that early repolarization (J wave) is associated with idiopathic ventricular fibrillation. However, its clinical and genetic characteristics are unclear., Methods and Results: This study included 50 patients (44 men; age, 45 ± 17 years) with idiopathic ventricular fibrillation associated with early repolarization, and 250 age- and sex-matched healthy controls. All of the patients had experienced arrhythmia events, and 8 (16%) had a family history of sudden death. Ventricular fibrillation was inducible by programmed electric stimulation in 15 of 29 patients (52%). The heart rate was slower and the PR interval and QRS duration were longer in patients with idiopathic ventricular fibrillation than in controls. We identified nonsynonymous variants in SCN5A (resulting in A226D, L846R, and R367H) in 3 unrelated patients. These variants occur at residues that are highly conserved across mammals. His-ventricular interval was prolonged in all of the patients carrying an SCN5A mutation. Sodium channel blocker challenge resulted in an augmentation of early repolarization or development of ventricular fibrillation in all of 3 patients, but none was diagnosed with Brugada syndrome. In heterologous expression studies, all of the mutant channels failed to generate any currents. Immunostaining revealed a trafficking defect in A226D channels and normal trafficking in R367H and L846R channels., Conclusions: We found reductions in heart rate and cardiac conduction and loss-of-function mutations in SCN5A in patients with idiopathic ventricular fibrillation associated with early repolarization. These findings support the hypothesis that decreased sodium current enhances ventricular fibrillation susceptibility.

A male neonate was clinically diagnosed with congenital intestinal atresia. Surgical operation was performed and the ileum including the atretic portion was resected. Grossly, there was a plaque-like elevation of mucosa at the proximal side of the ileal atresia. Microscopic examination of this lesion revealed proliferation of severely atypical glands. Although dysplasia was a serious diagnostic concern, we concluded that ischemia due to the intestinal atresia induced this benign pseudodysplastic regenerative mucosa, judging from the pattern of coexisting inflammation and the literature review., (© 2011 The Authors. Pathology International © 2011 Japanese Society of Pathology and Blackwell Publishing Asia Pty Ltd.)

Purpose: In 2006, The Japanese Society of Pediatric Endoscopic Surgeons devised a plan to develop a pediatric endoscopic surgical skill qualification (ESSQ) system. This system is controlled by The Japan Society for Endoscopic Surgery. The standard requirement for skills qualification is the ability of each applicant to complete common types of laparoscopic surgery. The main goal of the system is to decrease complications of laparoscopic surgery by evaluating the surgical skills of each applicant and subsequently certify surgeons with adequate skills to perform laparoscopic operations safely., Methods: A committee of pediatric ESSQ created a checklist to assess the applicant's laparoscopic surgical skills. Skills are assessed in a double-blinded fashion by evaluating an unedited video recording of a fundoplication for pediatric gastroesophageal reflux disease., Results: The initial pediatric ESSQ system was started in 2008. In 2008 and 2009, respectively, 9 out of 17 (53%) and 6 out of 12 (50%) applicants were certified as expert pediatric laparoscopic surgeons., Conclusions: Our ultimate goal is to provide safe and appropriate pediatric minimally invasive procedures and to avoid severe complications. To prove the predictive validity of this system, a survey of the outcomes of operations performed by certified pediatric surgeons is required.

Choledochocele, type III choledochal cyst, is a rare abnormality. We report a rare case of choledochocele involving a 15-year-old girl who presented with intermittent episodes of upper abdominal pain. She underwent computed tomography (CT) and magnetic resonance cholangiopancreatography (MRCP), which revealed no abnormalities of biliary and pancreatic ducts. Endoscopic retrograde cholangiopancreatography (ERCP) initially suggested a diagnosis of pancreas divisum (PD), and so we failed to diagnose choledochocele at that time. Her condition improved and she was discharged, but she presented again with recurrent episodes of upper abdominal pain. Then, spiral computed tomography with intravenous infusion cholangiography (IVC-SCT) facilitated a diagnosis of choledochocele. The coexistence of choledochocele and pancreas divisum is extremely rare, and it is difficult to diagnose. However, this case suggests that the absence of pancreatitis does not rule out choledochal cysts, and modern imaging techniques, such as IVC-SCT, must be applied in difficult cases.