Posterior reversible encephalopathy syndrome associated with mild COVID-19 infection in a 9-year-old child: A case report and literature review.

The neurologic complications associated with the coronavirus disease 2019 (COVID-19) is becoming more prevalent in children after the appearance of the Omicron strain. An association between COVID-19 and posterior reversible encephalopathy (PRES) has been consistently reported in adults, but little information is available in the pediatric age group. There are only few case reports of COVID-19-related PRES in children, and all of these patients were either on some type of immunomodulatory medications or whose general condition was severe. The present case, a 9-year-old Japanese boy, who had no fever but vomited several times from days 1-4 of a COVID-19 infection had an afebrile seizure on the 8th day of his illness. The patient had no history of hypertension, and had not previously been administered any immunosuppressive drugs before or during the period of his COVID-19 infection. On admission, his physical findings were unremarkable, except for a high blood pressure. The results obtained by brain computed tomography and magnetic resonance imaging were consistent with PRES. The patient recovered with no sequelae after treatment with antihypertensive drugs. Further investigations did not suggest any underlying disease that could have caused the transient hypertension. Although PRES is relatively rare in children, pediatricians should keep in mind that this syndrome can be complicated, even in children with mild COVID-19 infections.

Efficacy of sodium-glucose cotransporter 2 inhibitor with glucagon-like peptide-1 receptor agonist for the glycemic control of a patient with Prader-Willi syndrome: a case report.

Prader-Willi syndrome (PWS) is often related to severe obesity and diabetes mellitus (DM). Clinical findings suggesting the benefits of glucagon-like peptide-1 (GLP-1) receptor agonists for glycemic control of DM in PWS have been recently increasing. However, there are only a few reports describing the effects of sodium-glucose cotransporter 2 (SGLT2) inhibitors for PWS. We present a diabetic female with PWS, whose glycemic control was deteriorated at the age of 19 but improved to a certain extent by introducing the GLP-1 analog liraglutide. At the age of 20, the SGLT2 inhibitor empagliflozin was administered. Subsequently, her HbA1c level and body weight markedly decreased. Improvement in both insulin resistance and secretion was observed during the subsequent six months. In addition to GLP-1 receptor agonists, SGLT2 inhibitors may be a potential approach for the management of DM in PWS, especially in young patients whose pancreatic insulin secretion capabilities are still preserved.

Increased serum high-sensitivity C-reactive protein is related to hypoxia and brain natriuretic peptide in congenital heart disease.

BACKGROUND: Studies of adults have shown a direct association between increased serum concentrations of high-sensitivity C-reactive protein (hs-CRP) and atherosclerotic cardiovascular disease, diabetes, and chronic heart failure. Some studies have documented elevated hs-CRP in obese children and adolescents, and in patients with a history of Kawasaki disease, but there are few data on its clinical significance in congenital heart disease. METHODS: Measurements of hs-CRP, brain natriuretic peptide (BNP), hemoglobin, and percutaneous oxygen saturation (SpO2) were done in the following 70 patients: 18 controls; 11 with hypoxia (SpO2 /= 40 pg/mL and SpO2 > 85%); and 10 patients with hypoxia-BNP (SpO2 /= 40 pg/mL). Correlations between hs-CRP, and age, body mass index (BMI), hemoglobin, SpO2, and BNP were analyzed using single and multiple regression analysis. RESULTS: Hs-CRP in the hypoxia-BNP group was higher than in the other three groups, while in the hypoxia and the BNP groups it tended to be higher than in the controls. Although single regression analysis showed significant correlations between hs-CRP and, age, BNP, hemoglobin, and SpO2, multiple regression analysis showed that only BNP and SpO2 had a significant correlation with hs-CRP. CONCLUSIONS: In preoperative or postoperative congenital heart disease, patients who had hypoxia or increased BNP had a higher hs-CRP level than controls. Serum concentration of hs-CRP significantly correlated with SpO2 and plasma BNP levels, but not with age or BMI.

Utility of three-dimensional transesophageal echocardiography for diagnosis of unroofed coronary sinus.

We report a case of unroofed coronary sinus (URCS) in a 42-year-old female. At an outpatient clinic, she was found to have an atrioventricular septal defect and mitral regurgitation with pulmonary hypertension, and she was transferred to our institute for surgical treatment. Both atrioventricular valves were located at the same level, and both the right atrium (RA) and right ventricle were enlarged on two-dimensional transthoracic echocardiography. Color Doppler imaging demonstrated severe mitral and tricuspid regurgitation and a left-to-right shunt from the left atrium (LA) to the RA. Although an ostium primum defect of the atrial septum was suspected, the exact position of the shunt flow was unclear. Two-dimensional transesophageal echocardiography (2D-TEE) could visualize a direct communication between the LA and coronary sinus. Three-dimensional transesophageal echocardiography (3D-TEE) clearly visualized the entire route from the coronary sinus into the LA and RA. The utility of 3D-TEE as a modality complementary to 2D-TEE in diagnosis of URCS was confirmed.

Coil occlusion of PDA in patients younger than 1 year: risk factors for adverse events.

BACKGROUND: Transcatheter occlusion of infantile patent ductus arteriosus (PDA) challenges the interventionist. PURPOSE: To analyze the risk factors for adverse events from this procedure in patients younger than 12 months. SUBJECTS: We retrospectively analyzed data on 32 patients younger than 12 months in whom transcatheter coil occlusion of a PDA was attempted. Ages ranged from 1 to 11 (median, 7) months and body weight from 1.2 to 10 (6.0) kg. The minimum ductal diameter ranged from 1.0 to 4.6 (3.3) mm and pulmonary to systemic flow ratio from 0.7 to 12.5 (2.2). Major adverse events were defined as those requiring surgery, while minor adverse events included transient hemolysis not needing treatment, coil migration with successful transcatheter retrieval, and mild left pulmonary artery (PA) stenosis. We determined whether any factors such as age, body weight, minimum PDA diameter, angiographic type, operator, and use of a 0.052-in. Gianturco coil related to the occurrence of adverse events. RESULTS: In two patients, coils could not be deployed in the appropriate position. They underwent surgery following transcatheter retrieval of coils. Coils were successfully deployed in the other 30 patients, however, one patient developed persistent hemolysis that required surgical retrieval of the coils and closure. PDA was completely closed in the other 29 patients (clinical success rate, 91%). Thus, there were three major adverse events, while minor adverse events occurred in five patients. Univariate analysis did not identify any single factor that contributed to either major, minor, or total adverse events. However, all major adverse events occurred in patients under 6 months and less than 6 kg body weight with a minimum duct diameter of more than 3.5 mm. CONCLUSION: Transcatheter coil occlusion of PDA is feasible in selected patients younger than 1 year. A minimum diameter more than 3.5 mm in patients under 6 kg may be a risk factor for major adverse events.

Genesis stent implantation without using a long sheath in two children.

A Genesis stent was implanted in two children, one with superior vena caval (SVC) stenosis and one with pulmonary artery branch stenosis. Case 1 was a 2-month-old baby with SVC stenosis following intracardiac repair for total anomalous pulmonary venous connection (TAPVC) and case 2 was a 2-year-old child with left lower pulmonary artery stenosis following one-stage unifocalization for dextrocardia, double outlet right ventricle, ventricular septal defect, pulmonary atresia and major aortopulmonary collateral arteries. Both procedures resulted in immediate clinical and hemodynamic improvement. The Genesis stent has a closed-cell design with sigma hinges interpositioned between each cell. With improved deliverability and expandability of the stent, we can easily deliver it through smaller sheaths, which will facilitate its use in infants and smaller children with vascular stenosis.

Static balloon atrial septostomy with buddy wire technique: a case report.

The buddy wire technique, i.e. the use of a second 0.014" guidewire placed alongside the one employed to advance balloons, stents or other devices, improves the balloon or stent support and also provides guiding catheter stability. Furthermore, it concentrates forces transferred from the balloon along the line of the wire. We used a second 0.014" Platinum PLUS guidewire as a buddy wire during static balloon atrial septostomy in a 19-day-old baby with hypoplastic left heart syndrome, in whom standard balloon atrial septostomy was inadequate even with a fully dilated balloon. This procedure effectively dilated the atrial septal defect.

Efficacy of ticlopidine for preventing migraine after transcatheter closure of atrial septal defect with Amplatzer septal occluder: a case report.

Transcatheter closure of an atrial septal defect using the Amplatzer septal occluder is a current treatment option in Japan, but is occasionally associated with transient exacerbation or new onset migraine. Clopidogrel is effective in such a situation, but the efficacy of ticlopidine, an analog of clopidogrel, on migraine remains unclear. A 15-year-old girl presented with typical migraine attacks with aura 11 days after transcatheter closure of an atrial septal defect with an Amplatzer septal occluder. All examinations excluded thromboembolic origin of the migrain. Her symptoms disappeared completely after medication with ticlopidine.

Coil occlusion of patent ductus arteriosus.

BACKGROUND: Coils are the only devices available for transcatheter occlusion of patent ductus arteriosus (PDA) in Japan. Since April 1999, we have introduced a 0.052-inch Gianturco coil (0.052-inch coil) to close PDA >or=2.5 mm. METHODS AND RESULTS: A retrospective survey of the outcome of coil occlusions for PDA >or=2.5 mm before and after the 0.052-inch coil became available found that (1) the frequency of PDA >or=2.5 mm among all candidates for coil occlusion significantly increased after the availability of a 0.052-inch coil (p<0.01); (2) deployment complicated by migration (p<0.01), and prolonged procedure time (p<0.05) were significantly decreased after the introduction of the 0.052-inch coil. In a multivariate logistic regression model for uneventful deployment adjusted for age, pulmonary to systemic flow ratio, and use of a 0.052-inch coil, use of the 0.052-inch coil significantly decreased eventful deployment (p<0.05); and (3) successful deployment of a coil for PDA >or=4 mm significantly increased with the 0.052-inch coil (p<0.01). Complete occlusion was achieved once deployment was successful. CONCLUSION: Introduction of the 0.052-inch coil decreased complicated coil occlusion deployment for PDA >or=2.5 mm, and contributed to a better likelihood of coil occlusion for PDA >or=4 mm.

Coil occlusion for patent ductus arteriosus larger than 3 mm.

BACKGROUND: Coil occlusion of patent ductus arteriosus (PDA) is now widely accepted as the first-line treatment, but there are few reports of age-dependent differences in the complications associated with this technique. METHODS AND RESULTS: Sixteen patients (11 adults, 5 children) with a PDA larger than 3 mm, who underwent coil occlusion at Sapporo Medical University Hospital between September 1995 and August 2004, were enrolled. Immediate and intermediate outcomes and complications were analyzed. Procedural success rate was 72.7% (8/11) in the children and 100% (5/5) in the adults. Coil migration occurred in 4 children and 1 adult, and 3 adult patients had hemolysis. CONCLUSION: Hemolysis was more frequent in adults than in children even though the residual shunt was trivial.

Improvement of protein-losing enteropathy by coil embolization of the left azygos vein.

We report a 17-year-old boy with double-outlet right ventricle, atrioventricular septal defect, pulmonary stenosis, and persistent left superior vena cava who developed a protein-losing enteropathy, which was cured by coil embolization of the left azygos vein, thereby interrupting the vein-to-vein shunt.

Acute recoil of stents used for the relief of stenotic great vessels in the setting of congenital cardiac disease.

We implanted either large or medium Palmaz stents, or a Palmaz Corinthian stent, in various stenotic vessels, such as the pulmonary arteries, pulmonary veins, aorta, or superior caval vein. Using angiograms, we measured the diameter of the stenotic vessel before or after the implantation, the minimal diameter of the lumen, the minimal diameter of the largest fully expanded balloon used to expand the stent, and the diameter immediately after withdrawal of the balloon. The minimal diameter of the fully expanded balloon, and the minimal diameter of the lumen subsequent to expansion, were 8.2 +/- 2.4, and 7.7 +/- 2.3 mm, giving an absolute recoil of 0.5 +/- 0.4 mm, and a proportional recoil of 7 +/- 4%. There was no significant difference in either the absolute or proportional recoil for any of the stents, or for any of the different stenotic vessels. The proportional recoil correlated linearly with the minimal diameter of the lumen prior to the procedure, and with the ratio of the stenosis to the balloon, while the diameter of the stenotic vessels, the minimal diameter of the largest fully expanded balloon, the proportional stenosis prior to the procedure, and the ratio of the balloon to the diameter of the stenotic vessel, had no significant correlation with proportional recoil. The proportional recoil exceeded more than one-tenth when the minimal diameter of the lumen prior to the dilation was less than 3 mm, or the ratio of the balloon to the stenosis was greater than 3.0. An absolute recoil of around 1 mm is common when a large or medium Palmaz, or a Palmaz Corinthian stent, is implanted in great vessels. Balloons with a diameter of approximately one-tenth greater than that of the adjacent vessel may be needed if the minimal diameter of the lumen is small prior to the procedure.