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THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY

Vol.126, No.5, May 2022


Original Article
1. Clinical Characteristics of Children with Enterobacter Bacteremia at a Pediatric Hospital
Case Report
1. Isolated Unilateral Absence of Left Pulmonary Artery Diagnosed at Birth Triggered by Fetal Echocardiography
2. Use of a Token Economy System in Behavioral Therapy for Factitious Disorder (Munchausen Syndrome)
3. A Typical Case of COVID-19-associated Multisystem Inflammatory Syndrome in Children
4. Pediatric Canine Brucellosis, Diagnosed from Blood Culture, Presenting with Prolonged Rash and Fever
5. A 13-year-old Patient with Empyema Successfully Treated with Intrathoracic Fibrinolytic Therapy Using Urokinase: A Case Report
6. A Pediatric Case of Supragastric Belching with Frequent Belching as The Main Complaint
Brief Report
Group Vaccination against Influenza for Nursery Children during the COVID-19 Pandemic
Children with False-positive SARS-CoV-2 Antigen Test Results: A Retrospective Single-center Study


Original Article
Title
Clinical Characteristics of Children with Enterobacter Bacteremia at a Pediatric Hospital
Author
Moeko Aoki1) Kenji Kishimoto1) Shogo Otake2) Sayaka Nakamura1) Daiichiro Hasegawa1) Masashi Kasai2) and Yoshiyuki Kosaka1)
1)Department of Hematology and Oncology, Hyogo Prefectural Kobe Children Hospital
2)Department of Infectious Disease, Hyogo Prefectural Kobe Children Hospital
Abstract
This case series study was conducted to describe the clinical features in Enterobacter bacteremia in a tertiary Japanese pediatric care hospital. Patients diagnosed with Enterobacter bacteremia at our hospital between January 2006 and December 2020 were analyzed. A total of 22 patients were identified. Enterobacter cloacae (E. cloacae) was isolated in 17 (77%) patients and Enterobacter aerogenes (E. aerogenes) in five (23%) patients. The median patient age was 2 (interquartile range [IQR], 0−7) years, with 9 patients (41%) aged <1 year. All patients had underlying diseases, and the most common conditions were hematological and gastrointestinal diseases. Susceptibilities to cefotaxime and cefepime were 73% and 91%, respectively. The 28-day all-cause mortality was 14% (3/22). The median age at onset was higher in patients with E. cloacae bacteremia than in patients with E. aerogenes bacteremia (3 [IQR, 1−10] years vs. 0 [IQR, 0−0] years old, p = 0.016). There was a marked difference in the proportion of underlying hematological disease or solid tumors between patients with E. cloacae bacteremia and those with E. aerogenes bacteremia (71% vs. 0%, p = 0.010). The results suggest the difference in characteristics of patients with E. cloacae bacteremia and those with E. aerogenes bacteremia.




Case Report
Title
Isolated Unilateral Absence of Left Pulmonary Artery Diagnosed at Birth Triggered by Fetal Echocardiography
Author
Takeshi Kumagai1) Nobuyuki Kakimoto1) Masashi Satoh1) Shunji Uchita2) and Hiroyuki Suzuki1)
1)NICU Division, Department of Perinatal Medicine, Wakayama Medical University
2)First Department of Surgery, Wakayama Medical University
Abstract
We report a female infant born at full term who was hospitalized at our NICU as having right aortic arch (RAA) without cardiac defect by fetal ultrasound cardiography. Echocardiography detected RAA, absence of any intra-cardiac defect, and the main pulmonary artery (PA) flow to the right PA branch, but the detail of the left PA was not obvious. More meticulous inspection demonstrated left patent ductus arteriosus (PDA) flow from the left brachiocephalic artery was drained into the left proximal portion of the PA. We diagnosed the patient as having RAA with left unilateral absence of the pulmonary artery (UAPA) and bilateral PDA. To maintain the left PDA, infusion of lipo-prostaglandin E1 was started. However, heart failure due to bilateral PDA shunt developed gradually, necessitating modified Blalock-Taussig shunt and bilateral PDA closure on day 13. At seven months, she underwent anastomosis of the left PA to the main PA duct. The patient is currently 7 years old and attending elementary school without any physical limitation.
UAPA is a rare congenital cardiovascular malformation. In cases of RAA or PA branch dilation by fetal echocardiography, isolated UAPA should be distinguished, and further inspection conducted after birth. Neonatologists should be aware of this cardiovascular anomaly and confirm the PA branch using echocardiography when respiratory symptoms are evident.




Case Report
Title
Use of a Token Economy System in Behavioral Therapy for Factitious Disorder (Munchausen Syndrome)
Author
Yoichi Nishimura1) Emiko Hitomi1) Tomohiro Hitomi1) Yuichi Ookura2) Koumei Uetake3) and Kentaro Chonan4)
1)General Pediatric Center, Social Medical Corporation Hokuto, Hokuto Hospital
2)Clinical Psychologist, Social Medical Corporation Hokuto, Hokuto Hospital
3)Department of Pediatrics, Obihiro-Kosei General Hospital
4)Hikari Ophthalmology
Abstract
Factitious disorder or Munchausen syndrome (hereafter referred to as factitious disorder) is a disease concept characterized by physical and psychological symptoms that are unconsciously and intentionally created by the patients themselves, who are supposedly suffering from these symptoms, and is differentiated from malingering disorder in that there is no extrinsic incentive.
There are very few reported cases of factitious disorder in children, and diagnosis is difficult due to a combination of factors such as the absence of extrinsic incentives and the presentation of various physical and neurological findings, leading to excessive testing and treatments. Treatment is often difficult even after diagnosis.
This report describes a case of a child with factitious disorder who presented with a chief complaint of reduced vision acuity and color blindness. The mother underwent parent training and improvement was observed with behavioral therapy using a token economy system.




Case Report
Title
A Typical Case of COVID-19-associated Multisystem Inflammatory Syndrome in Children
Author
Yuusuke Shibata Kazunori Tagami Shunsuke Kato Toru Maeda Takae Kobayashi Takenori Adachi and Takashi Kawabe
Department of Pediatrics, Kasugai Municipal Hospital
Abstract
We treated a 12-year-old Japanese girl with pediatric COVID-19-related multisystem inflammatory syndrome (MIS-C / PIMS). Forty-three days before admission, the patient tested positive for SARS-CoV-2 on RT-PCR but was asymptomatic at the time. She was admitted to the hospital on day X with fever, cough, diarrhea and bilateral conjunctival redness lasting 5 days. Her blood test results showed lymphopenia and thrombocytopenia, elevated inflammatory markers, and coagulopathy. A chest X-ray and computed tomography revealed cardiac dilation, but the lung field was only a slight pneumonia image. She was diagnosed with MIS-C/PIMS based on Kawasaki disease-like symptoms and acute gastrointestinal symptoms after COVID-19, and a definitive diagnosis was made by referring to the diagnostic criteria. She had left heart failure, but she was treated with intravenous immunoglobulin 2 g/kg and made a full recovery without any residual cardiac dysfunction or coronary artery abnormalities.
Although only a few cases of MIS-C/PIMS have been reported in Japan, epidemiologically it has been reported that the number of cases will increase after the COVID-19 epidemic, and the number of patients may increase in Japan in the future. Although Kawasaki disease-like symptoms are important findings, it is desirable to be able to find this disease from non-specific symptoms for early diagnosis and treatment. It is important for all clinicians who attend to pediatric patients to familiarize themselves with the diagnostic criteria of MIS-C/PIMS.




Case Report
Title
Pediatric Canine Brucellosis, Diagnosed from Blood Culture, Presenting with Prolonged Rash and Fever
Author
Masazumi Miyahara1) Kyoko Osaki1) and Koichi Imaoka2)
1)Department of Pediatrics, Okanami General Hospital
2)Department of Veterinary Science, National Institute of Infectious Diseases
Abstract
Brucella canis (B. canis) is a zoonotic pathogen whose main host is dogs. Human brucellosis is rare, and therefore, poorly recognized by clinicians. It is difficult to diagnose as it is asymptomatic in many cases and presents with non-specific flu-like symptoms and non-characteristic physical findings. We report an 8-year-old child with B. canis infection who presented with rash and fever.
The boy was referred to our hospital on the 19th day of illness. Nodosum-like erythema was observed on the trunk and thighs, but other physical findings were inconclusive. We suspected viral infection due to the minimal inflammatory response on the blood test.
Dog-related zoonotic disease was suspected because of a positive blood culture and his parents being dog breeders. Due to a positive B. canis antibody test and the blood culture isolate being identified as B. canis by PCR, B. canis infection was diagnosed. The patient recovered after treatment with doxycycline and rifampicin.
Here, we report on the status of B. canis infection in Japan to raise awareness about brucellosis as a differential diagnosis, which is also an important cause of fever of unknown origin.




Case Report
Title
A 13-year-old Patient with Empyema Successfully Treated with Intrathoracic Fibrinolytic Therapy Using Urokinase: A Case Report
Author
Takehiro Homma1) Noriko Takeda1)2) Yuya Saito1) Yukiko Osawa1) and Toshimasa Obonai1)
1)Department of Pediatrics, Tama Hokubu Medical Center
2)Sagamihara Ryouikuen, Institute for Patients with Severe Motor & Intellectual 8 Disabilities
Abstract
Video-assisted thoracic surgery and open chest debridement are widely accepted treatments for pediatric patients with pleural empyema who are unresponsive to antimicrobial agents or drainage; however, these are invasive approaches. Both national and international guidelines suggest the administration of fibrinolytic agents into the pleural cavity through a chest drain tube as a less invasive therapeutic strategy with equivalent outcomes. However, Japanese guidelines do not offer detailed descriptions of drug selection and treatment duration. Among the fibrinolytic agents recommended by international guidelines, urokinase and alteplase are the only products available in Japan. Therapeutic outcomes and complications do not differ between these fibrinolytic agents. Neither drug is covered by health insurance in Japan. Nevertheless, in view of the invasiveness and cost of surgical treatment, fibrinolytic therapy is a promising alternative, and in our opinion, this therapy should be covered by the Japanese health insurance system. We report a case of pleural empyema treated with thoracic drainage, followed by fibrinolytic therapy using urokinase. We hope that our findings will contribute to the literature and serve as a prime example for the establishment of fibrinolytic therapy for empyema in Japan.




Case Report
Title
A Pediatric Case of Supragastric Belching with Frequent Belching as The Main Complaint
Author
Syunsuke Nagara1) Emi Tannaka1) Shinji Usui1) Miwa Kawashiri1) Tomomitsu Sado2) Shingo Kurasawa2) Yoshiko Nakayama2) Daisuke Masui3) and Atsushi Yamagishi1)
1)Department of Pediatrics, Takayama Red Cross Hospital
2)Department of Pediatrics, Shinshu University School of Medicine
3)Department of Pediatric Surgery, Kurume University School of Medicine
Abstract
A 15-year-old Japanese girl repeatedly presented with belching over six months. Burping persisted except during sleep. In some episodes, belching was observed every 10 s, resulting in poor drinking and acetonemic vomiting. She reported no abdominal distension, and abdominal plain X-ray revealed air retention in her intestinal tract was mild. Based on the clinical course and esophageal impedance pH monitoring examination, supragastric belching (SGB) was diagnosed. Cognitive-behavioral therapy reduced the frequency of belching. Overseas, there are reports of SGB mainly in adults but including children. However, there have been no such reports of children in Japan. The reason may be that since SGB is a newly acknowledged disease, the awareness is low. SGB is characterized by the absence of belching during sleep and when concentrating, suggesting psychological factors are involved. If clinical introduction of the esophageal impedance pH monitoring test accelerates, and awareness of the disease increases, the identification of SGB in adolescents, which increases with psychological stress, may be brought to attention.




Brief Report
Title
Group Vaccination against Influenza for Nursery Children during the COVID-19 Pandemic
Author
Shugo Misato1) Hiroaki Ito1) Naoko Furuya2) and Mayumi Ishii3)
1)Department of Pediatrics, Kameda Medical Center
2)Hospital Epidemiology and Infection Prevention Center
3)Nursing Administration Room, Kameda Medical Center
Abstract
In 2020, kindergarteners received group vaccination against influenza during child care in the absence of their parents to reduce the burden on parents at our medical-service-related facilities during the COVID-19 pandemic. Analysis of a questionnaire survey provided to parents after vaccination indicated that they found the vaccination convenient and that it provided them with a sense of security regardless of whether the parents were a part of the medical staff or not. It also suggests that there is a possibility of supporting child care and improving the immunization rate in kindergarteners. Furthermore, we hope that this study will help authorities consider group vaccination in child-care facilities against COVID-19 and other emerging infectious diseases.




Brief Report
Title
Children with False-positive SARS-CoV-2 Antigen Test Results: A Retrospective Single-center Study
Author
Kei Sakamoto1)2) Hiroyuki Iijima1) Takanori Funaki3) Kensuke Shoji3) Ken-Ichi Imadome4) Mitsuru Kubota1) and Akira Ishiguro2)
1)Department of General Pediatrics and Interdisciplinary Medicine, National Center for Child Health and Development (NCCHD)
2)Center for Postgraduate Education and Training, NCCHD
3)Division of Infectious Diseases, NCCHD
4)Division of Advanced Medicine for Virus Infections, NCCHD
Abstract
Japanese Guidelines for COVID-19 state that positive antigen test results for SARS-CoV-2 do not require further confirmatory testing. Among a total of 247 COVID-19 children who visited our hospital, 9, having no contact history with COVID-19 patients, received a COVID-19 diagnosis based on positive antigen test results. Confirmatory PCR tests for 6 of the 9 children revealed negative results in all six. Additional PCR tests should be considered in antigen-positive children without contact history with COVID-19 patients.




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