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THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY

Vol.126, No.11, November 2022


Review
1. Asymptomatic Unilateral Pulmonary Artery Deficiency Diagnosed in Childhood: A Case Report and Literature Review
Original Article
1. Catheter Intervention for Severe Aortic Coarctation in Extremely Low Birth Weight Infants
2. A Questionnaire Survey on the Utilization of Touchscreen Devices in Young Infants and School Children
Case Report
1. Wolff-Parkinson-White Syndrome with Two Accessory Pathways Presenting with Fatal Arrhythmia as the First Symptom in a 3-Year-Old Child
2. Self-bloodletting Discovered during Workup for Heart Failure Accompanied by Severe Anemia


Review
Title
Asymptomatic Unilateral Pulmonary Artery Deficiency Diagnosed in Childhood: A Case Report and Literature Review
Author
Kenichi Iwata1) Yosuke Nishio1) Yuka Suenaga1) Fumie Kitai1) Arisa Sato1) Hirohide Tokunaga1) Michio Suzuki1) Taichiro Muto1) Makoto Morita1) Taichi Kato2) and Shinji Hasegawa1)
1)Department of Pediatrics, Nagoya Memorial Hospital
2)Department of Growth and Developmental Medicine, Nagoya University Graduate School of Medicine
Abstract
Unilateral absence of pulmonary artery (UAPA) is a congenital malformation in which the pulmonary artery branches only to one side, and no unilateral pulmonary artery is present. Some patients present with hemoptysis and wheezing associated with heart failure and pulmonary hypertension, while others remain asymptomatic and are diagnosed in adulthood. This case involves a 3-year-old girl diagnosed with acute exacerbation of bronchial asthma (BA). Chest X-ray showed rightward deviation of the mediastinum, left-dominant lung hyperinflation and attenuation of right pulmonary vascularity on admission and discharge. Echocardiography and contrast-enhanced computed tomography (CT) of the chest revealed right pulmonary artery defect, and she was diagnosed with UAPA. There were no symptoms of complications of UAPA. We only provided therapeutic management of BA, not UAPA.
We reviewed 22 reported cases of asymptomatic UAPA diagnosed in childhood. In 19 cases, the diagnosis was made by chest X-ray. In all cases, there were abnormal findings on chest X-ray. When mediastinal deviation or unilateral pulmonary hyperinflation is observed on chest X-ray, even if the patient is asymptomatic, close examination such as echocardiography or contrast-enhanced CT of the chest should be considered, given the possibility of UAPA.




Original Article
Title
Catheter Intervention for Severe Aortic Coarctation in Extremely Low Birth Weight Infants
Author
Mizuhiko Ishigaki Sung-Hae Kim Keisuke Sato Jun Yoshimoto Norie Mitsushita Masaki Nii and Yasuhiko Tanaka
Department of Cardiology, Shizuoka Children's Hospital
Abstract
The prognosis of congenital heart diseases other than patent ductus arteriosus is very poor in extremely low birth weight (ELBW) infants, except in cases where elective intervention after weight gain is possible. Coarctation of the aorta (CoA) without ventricular septal defect results in heart failure due to afterload mismatch in the left ventricle and pulmonary overflow. The primary treatment is aortoplasty, but surgical intervention is risky and uncommon in ELBW infants. An aggressive medical management including inotropic agents could be employed in infants with mild CoA, followed by surgical intervention after appropriate weight gain. On the other hand, severe CoA has been difficult to manage, leading to mortality.
Catheterization is an endovascular therapy that enables less invasive treatment, and its scope of activity is expanding rapidly with the advent of more advanced equipment. We have performed stenting in four ELBW infants with severe CoA since 2014. This is a novel treatment method that may save lives in previously impossible cases.




Original Article
Title
A Questionnaire Survey on the Utilization of Touchscreen Devices in Young Infants and School Children
Author
Yuri Ozawa1) Hiroshi Ozawa1) Nobuko Sugiura2) Yuka Shirakawa3)4)5) and Yosuke Kita3)6)
1)Department of Child Neurology, Shimada Ryoiku Medical Center for Challenged Children Hachioji
2)Department of Child Psychiatry, Shimada Ryoiku Medical Center for Challenged Children Hachioji
3)Keio University Faculty of Letters
4)Research Fellow of the Japan Society for the Promotion of Science
5)The Institute of Natural Sciences, College of Humanities and Sciences, Nihon University
6)Cognitive Brain Research Unit (CBRU), Faculty of Medicine, University of Helsinki
Abstract
To clarify the habits of children using touchscreen devices, we assessed 107 questionnaires completed by parents of children who consulted at Ryoiku Medical Center Hachioji. The surveys were completed between October and December 2018. The age groups were young (under 4), middle (from 5 to 8) and older (over 9). We found that the younger the children, the earlier they had first used touchscreen devices. The older the children, the longer the usage. Children using touchscreen devices for over 30 minutes went to sleep later than those who used for less than 30 minutes. The period of usage that puts children at risk of going to sleep later than usual is 30 minutes in the young and middle groups, and 60 minutes in the older group. Children who have developmental problems used touchscreen devices for a longer duration and more frequently. We need regulations regarding usage of touchscreen devices to protect children from sleep problems and addiction of using such devices. We suggest an appropriate usage duration of touchscreen devices.




Case Report
Title
Wolff-Parkinson-White Syndrome with Two Accessory Pathways Presenting with Fatal Arrhythmia as the First Symptom in a 3-Year-Old Child
Author
Mayuko Yamauchi1) Yuji Doi1) Yukie Okabe1) Kazuhisa Sato1) Kazutoshi Ueda1) Kayo Ogino1) Tomohiro Hayashi1) Kenji Waki1) Tsugutoshi Suzuki2) and Yoshio Arakaki1)
1)Department of Pediatrics, Kurashiki Central Hospital
2)Department of Pediatric Electrophysiology, Osaka City General Hospital
Abstract
The patient, a 3-year-old boy, was found unconscious by his mother after crying. She performed cardiopulmonary resuscitation (CPR) until the ambulance arrived. The initial waveform on the automated external defibrillator (AED) was pulseless electrical activity (PEA), and defibrillation was performed once due to ventricular fibrillation. CPR for PEA was continued until he had return of spontaneous circulation. Electrocardiogram on admission showed delta waves. Because the cause of the cardiac arrest remained unknown after etiology exploration, he was diagnosed with fatal arrhythmia due to Wolff-Parkinson-White syndrome. He underwent radiofrequency catheter ablation (RFCA), and two accessory pathways (APs) were found on the posterior wall of the mitral annulus and the lateral wall of the tricuspid annulus. Atrioventricular reciprocating tachycardia between the APs was an arrhythmia causing circulatory failure with a heart rate of 300 beats per minute. Bilateral APs were cauterized, but the right side was not completely transected. Because fatal arrhythmias no longer occurred, oral flecainide was started. After rehabilitation, his activities of daily living improved, and he is now waiting for repeat RFCA. Appropriate resuscitation and the use of AED at the civilian level at the onset may contribute to a good neurological prognosis. Medical professionals need to judge the appropriateness of the waveforms on the AED, which may lead to improved prehospital care.




Case Report
Title
Self-bloodletting Discovered during Workup for Heart Failure Accompanied by Severe Anemia
Author
Yoko Hirano Akiko Kinumaki Yuko Kajiho Chie Takahashi Shoichiro Kanda and Motohiro Kato
Department of Pediatrics, The University of Tokyo Hospital
Abstract
Recently, children's social environment is rapidly evolving. Widespread exposure to the Internet has enabled easy access to harmful information regarding self-destructive behaviors (non-suicidal self-injury/substance abuse). Therefore, wide varieties and early onset of these practices are significant concerns.
We describe a 15-year-old female adolescent who presented with heart failure and severe anemia secondary to self-bloodletting. She did not attend school and indulged in self-cutting and prescription/over-the-counter drug abuse since 12 years of age. She visited many psychiatric and pediatric centers but repeatedly failed to follow-up. She developed edema, pallor, and gradually worsening dyspnea 2 months before hospitalization. Rapidly worsening dyspnea necessitated emergency hospitalization. Laboratory investigations on admission showed severe anemia (serum hemoglobin 3.1 g/dL) complicated with high-output heart failure. However, comprehensive evaluation did not reveal the etiology of anemia. Eventually, the patient was admitted due to a history of self-bloodletting and was diagnosed with iron-deficiency anemia secondary to persistent self-bloodletting. She began this practice 6 months ago, which was unnoticed by her family (estimated blood loss 1,000-1,500 mL/month). Before her discharge, we arranged social and medical support following multidisciplinary coordination.
Pediatricians should consider intentional self-bloodletting as a differential diagnosis of unexplained anemia. Patient interviews including psychosocial patterns should be conducted politely. Pediatricians should understand the actual situation and provide multidisciplinary coordination-based holistic care for children and adolescents with self-destructive behaviors.




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