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THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY
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Vol.125, No.3, March 2021
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Original Article
Title
Survey on Internal Medicine Institutions for Adults in Regard to the Transitional Care Given to Patients with Special Health Care Needs
Author
Asuka Nagahama1) Seigo Korematsu2) Keisuke Sato3) Mutsumi Akaishi4) and Koichi Iida4)
1)Sakanoichi Hospital
2)Nakatsu Municipal Hospital
3)The Holly Mother of Grace Hospital Home for Children and Persons with Severe Motor and Intellectual Disabilities
4)Oita Prefectural Hospital
Abstract
A survey on the state of acceptance of patients with special health care needs (PSHCN) was conducted at internal medicine institutions for adults in Oita prefecture. The survey was conducted using an anonymous self-completed questionnaire. Answers were collected from 201 institutions (47% response rate). While there were only 9 institutions in which treatment for PSHCN could be provided by a general practitioner, 73 institutions responded that providing medical treatment was possible in some way. The reasons given for why PSHCN cannot be treated were doctors' lack of knowledge and skills (81%), and nurses' lack of knowledge and skills (66%). Moreover, if the individuals have severe motor and intellectual disabilities, the staff are inexperienced when it comes to dealing with parents (41%). As a result, a certain number of medical institutions responded that outpatient treatment and visits are possible for limited treatment. In order to structure the home medical care system for transitional patients, it is necessary to recognize the way of transition based on situations such as asking to consult a pediatrician and establishing a support system. Moreover, it is ideal to provide the guardians with mental care in terms of transitional medical care, therefore holding lectures for physicians in accordance with their needs should be considered.
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Original Article
Title
Incidence of Bone Fractures in Medically Dependent Children with Severe Motor and Intellectual Disabilities during Short-stay Admission
Author
Yasuhiko Takahashi Miwa Yoshino Ryoko Nakamura Toru Yoneda Jun Muneuchi and Junko Yamamoto
Department of Pediatrics, Japan Community Healthcare Organization Kyushu Hospital
Abstract
In Japan, the number of medically dependent children with severe motor and intellectual disabilities (SMID), who have severe respiratory insufficiencies and require tube feeding, is increasing. They have an increased risk of limb bone fracture caused by low energy traumas, due to bone fragility and reduced mobility.
The aim of this study was to assess the ratio of limb bone fracture in medically dependent children with SMID during short-stay admission at an acute care hospital.
Between 2010 and 2019, 84 medically dependent children with SMID were admitted to our pediatric ward for a total of 1,905 times and 13,328 person-days. During this period, we encountered 15 incidental limb bone fractures in 10 medically dependent children with SMID.
Specific fracture sites were 3 upper limbs and 12 lower limbs and the most common site was supracondylar fracture of the femur.
Thus, the incidence of a limb bone fracture occurring as a complication for medically dependent children with SMID who were admitted for a short-stay at a pediatric ward was about 0.8%. Our study findings indicate a need for more gentle care and subsequent preventive services for children with SMID.
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Original Article
Title
Effectiveness of Official Telephone Consultation Services for the Management of Pediatric Emergency Care
Author
Tsuneo Igarashi1)2) Fumiya Ohyama1)3) Makoto Shigeta1)4) Kanji Nagashima1)5) and Tsutomu Taguchi1)6)
1)Gunma Pediatric Association
2)Takasaki General Medical Center, Department of Pediatrics
3)Oyama Pediatric Clinic
4)Shigeta Pediatric and Allergic Clinic
5)Nagashima Pediatric Clinic
6)Taguchi Pediatric Clinic
Abstract
Gunma Prefecture has an official telephone consultation service that provides parents with advice on how to cope with emergency situations involving children at night or during holidays. To evaluate whether this service can adequately guide parents to visit emergency hospitals when their children experience an emergency situation, we reviewed the official records on the number of telephone consultations, number of visits to emergency hospitals, and the ratio of admissions to emergency hospitals among children aged <15 years. We also reviewed the official data on the population of children aged <15 years in Gunma Prefecture.
We found that as the number of telephone consultations increased, the number of visits to emergency hospitals decreased; there was an inverse correlation among the variables. The rate of admission among children who visited emergency hospitals increased in proportion to the increase in telephone consultations. Thus, we concluded that telephone consultations adequately guided parents to visit an emergency hospital when their children underwent emergency situations. However, it was difficult to evaluate whether external factors such as the contribution of primary care units in the evening, spread of vaccination, and accurate knowledge about illnesses in children influenced the number of visits to emergency hospitals.
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Case Report
Title
An Extremely Low Birth Weight Infant Whose Patent Ductus Arteriosus Clip Moved into the Trachea
Author
Honoka Kotoku1) Junichi Arai1) Tae Kamakura1) Yoshiya Yukitake1) Daigo Kajikawa1) Yusuke Hoshino1) Rena Fuchino1) Masakazu Abe2) Yukiko Ban2) and Miki Toma3)
1)Department of Neonatology, Ibaraki Children's Hospital
2)Department of Cardiovascular Surgery, Ibaraki Children's Hospital
3)Department of Surgery, Ibaraki Children's Hospital
Abstract
We report a case of an extremely low birth weight infant whose titanium clip for patent ductus arteriosus (PDA) perforated the bronchi and moved into the trachea after surgical intervention.
At 22-weeks of gestational age, a premature boy underwent surgical intervention for PDA by titanium clip, at 20 days post-birth. High frequency oscillatory ventilation was performed from the day after the operation. The clip moved into the trachea on chest X-ray, at 18 days after the operation. The clip was moved into the endotracheal tube using a closed endotracheal suction catheter with his head down and removed with the tube. For extremely low birth weight infants, titanium clip that is used for surgical intervention for PDA is relatively large, and their tissues are fragile. Therefore, it is necessary to keep in mind that surgical intervention for PDA by titanium clip can cause unexpected complications in such patients.
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Case Report
Title
Infant with Acute Myocarditis the Day after the Sudden Death of Her Monochorionic Diamniotic Twin Sister
Author
Tomoya Tsuchihashi1) Masahiro Kamada1) Naomi Nakagawa1) Yukiko Ishiguchi1) Yuji Moritou1) Kengo Okamoto1) Noriko Kawata1) and Hiroaki Nomura2)
1)Department of Pediatric Cardiology, Hiroshima City Hiroshima Citizens Hospital
2)Department of Pediatrics, Kousei General Hospital
Abstract
In infants, acute myocarditis symptoms are generally nonspecific. Diagnosing acute myocarditis early is difficult; however, heart failure worsens rapidly in fulminant myocarditis. We report an infant who was given an early diagnosis of acute myocarditis owing to the sudden death of her twin sister.
The patient was an 8-month-old monochorionic diamniotic twin girl and had good growth and development. She presented to a previous hospital with only cold-like symptoms. She was observed closely because her twin sister died suddenly the previous day with a diagnosis of fulminant myocarditis. Blood tests showed abnormally high brain natriuretic peptide (BNP) levels and positive troponin T (TnT) results. She arrived at our hospital with acute myocarditis. A steroid pulse treatment and intravenous immunoglobulin were initiated. Her cardiac function improved. She was discharged after 15 days without any sequelae. Rhinovirus-induced acute myocarditis was suspected, based on polymerase chain reaction test results of a pharyngeal swab.
Acute myocarditis in infants is difficult to detect, but early diagnosis and treatment are important. Genetic factors may be involved in acute myocarditis. Blood tests such as (NT-pro) BNP and TnT are good first steps for early diagnosis, and early treatment may prevent fulminant myocarditis.
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Case Report
Title
A Case of Psychogenic Fever with Increased Sympathetic Activity on Holter Electrocardiography
Author
Aiko Kumamoto1)2) Yoshiyasu Ogata1) Shinji Nishimura2) and Muneaki Matsuo1)
1)Department of Pediatrics, Faculty of Medicine, Saga University
2)Department of Pediatrics, Saga-Ken Medical Centre Koseikan
Abstract
Psychogenic fever describes an increased body temperature associated with psychological stress. The physiological mechanism of psychogenic fever involves sympathetic hyperreactivity and thus differs from hyperthermia caused by infection or inflammation. We report a diagnosis of psychogenic fever based on a Holter electrocardiogram suggesting enhanced sympathetic nerve activity.
The patient was a 14-year-old schoolgirl who presented with hyperthermia lasting for 3 months. Physical findings, blood testing, and imaging results were normal. She developed a high body temperature up to 40°C on the days she went to school, and antipyretic drugs did not reduce her fever. Heart rate variability spectrum analysis by Holter electrocardiography (ECG) showed a marked increase in LF/HF, an index of sympathetic nerve activity, together with hyperthermia on the days she went to school. The LF/HF did not increase on non-school days. Malingering were ruled out and she was given a diagnosis of psychogenic fever due to school-induced psychosocial stress. Her temperature returned to normal following treatment using tricyclic antidepressants, psychotherapy and environmental control.
Psychogenic fever has no direct laboratory findings and is an exclusionary or therapeutic diagnosis that often takes time to verify. Evaluation by Holter ECG may be useful as a diagnostic aid for psychogenic fever.
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Case Report
Title
Neonatal Lethal Costello Syndrome with G12V Mutation
Author
Takashi Fujii1) Yosuke Sudo1) Ayako Sasaki1) Koki Nagai2) Yoko Aoki2) and Tetsuo Mitsui1)
1)Department of Pediatrics, Yamagata University
2)Department of Medical Genetics, Tohoku University School of Medicine
Abstract
Costello syndrome is a rare congenital disorder caused by heterozygous mutations in the HRAS. Symptoms of this syndrome vary, although hypertrophic cardiomyopathy and malignant tumors are key determinants of survival. Most patients are diagnosed during infancy to early childhood. However, severe cardiopulmonary complications and a lethal clinical course are rare in neonates. We report a case of a male newborn in which neonatally lethal Costello syndrome was diagnosed. The patient had nuchal edema, polyhydramnios, and perinatal tachycardia. Because of premature rupture of the membranes with fetal bradycardia indicating emergency cesarean section, he was born at 28 weeks 3 days of gestation. Myocardial hypertrophy and supraventricular extrasystoles, which caused marked edema, were already noted at birth. Refractory pleural effusion and ascites developed and required repeated paracenteses from 2 days of age. Supraventricular tachycardia and respiratory failure with pulmonary edema led to circulatory failure. He eventually died at 29 days of age. A heterozygous c.35_36delinsTT (G12V) mutation in HRAS was identified using targeted sequencing analysis. This pathogenic variant is rare and known as a neonatal lethal phenotype.
Costello syndrome has clinical features similar to Noonan syndrome, which is relatively common. There are exceptionally severe variants in Costello syndrome. HRAS analysis at the early postnatal period would be helpful for diagnosing and estimating the prognosis if these syndromes are suspected.
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Case Report
Title
A Girl with Trimethylaminura
Author
Junpei Hasumi1) and Hiroshi Yamazaki2)
1)Department of Pediatrics, Saku Central Hospital Advanced Care Center
2)Laboratory of Drug Metabolism and Pharmacokinetics, Showa Pharmaceutical University
Abstract
Trimethylaminuria (TMAU) is a disease characterized by emission of an unpleasant fishy smell from the body due to an unspecified increase in the biological concentration of trimethylamine. Although the disease is not life-threatening, its characteristics can potentially have negative effects on the social life of patients. Cases have been reported worldwide, but only few have been reported in Japan, and the awareness of the disease remains low.
Here, we report a case of TMAU that was initially suspected based on typical symptoms and course, and was then diagnosed by urine analysis and scraped oral mucus samples. The diagnostic approach guaranteed low invasiveness and high safety. The information obtained thus enabled us to classify the disease type and severity that proved to be incredibly useful.
We hope that TMAU and its diagnostic approaches will become more widely recognized in the future, and more cases will be accurately diagnosed.
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Case Report
Title
A Boy with Recurrent Pneumococcal Meningitis Caused by a Transethmoidal Encephalocele: A Case Report
Author
Risako Notani1) Masashi Kasai2) Akari Mitsuboshi1) Yoshiaki Cho1) Yukiya Kurahashi1) and Yusuke Ito2)
1)Division of Pediatric Hospital Medicine, Department of Pediatrics, Hyogo Prefectural Kobe Children's Hospital
2)Division of Infectious Diseases, Department of Pediatrics, Hyogo Prefectural Kobe Children's Hospital
Abstract
We report a case of recurrent pneumococcal meningitis caused by a transethmoidal encephalocele. The patient had been admitted to the hospital 3 times, at 5, 8, and 10 years of age, because of pneumococcal meningitis. Standard treatment was successful in all cases, and he was referred to our department for further examination. Immune function did not appear compromised, such as decrease in immunoglobulins, neutrophils, lymphocytes, and antibodies for capsular polysaccharide antigen of Streptococcus pneumoniae. His spleen was normal. Since computed tomography showed an intranasal mass and a defect of the ethmoid bone, spinal drainage was performed, and it worked well. However, after stopping the drainage, the mass became enlarged. Therefore, we diagnosed transethmoidal encephalocele. Anatomical problems account for half of the cases with recurrent meningitis. However, most of them are related to trauma, and an encephalocele is rare. A transethmoidal encephalocele is difficult to diagnose because it presents with minimal symptoms, such as nasal discharge, other than recurrent meningitis. Therefore, transethmoidal encephalocele should be considered as a suspected cause of recurrent meningitis, and imaging should be reviewed.
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Case Report
Title
A Pediatric Case of Hepatitis A with Long-term Viral Shedding in the Stool
Author
Yuto Ochiai1) Hiroyuki Shimizu2) Kazushi Ichikawa1) Manri Kawakami3) Masaharu Takahashi4) and Hiroaki Okamoto4)
1)Department of Pediatrics, Fujisawa City Hospital
2)Department of Clinical Laboratory Medicine, Fujisawa City Hospital
3)Liver Disease Center, Okayama Saiseikai General Hospital
4)Division of Virology, Department of Infection and Immunity, Jichi Medical University School of Medicine
Abstract
We present a case of a 10-year-old girl with hepatitis A contracted via intrafamilial transmission after traveling to Indonesia. Despite being asymptomatic, the patient was referred to our hospital because her mother had developed hepatitis A. HAV-RNA was detected in the stool, but the general laboratory tests were unremarkable at the first visit. One month later, hepatitis A was diagnosed based on her symptoms and the elevated levels of serum transaminases, which became normal within a month following supportive treatment. However, HAV-RNA was detected in the plasma and stool until 3 and 5 months, respectively, after the onset of the disease.
The HAV strains recovered from the patient and her mother shared 100% nucleotide sequence identity and these strains were closest to those identified as epidemic strains in Japan in 2017. They were dissimilar to the Indonesian HAV strains. Recent globalization has made it difficult to specify the source of infection. Premature immunity has been suggested as the cause for prolonged viremia and fecal excretion of HAV after the onset of acute hepatitis A in children.
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Brief Report
Title
A Case of Pertussis in an Unvaccinated Infant Requiring Intensive Respiratory Care during the Coronavirus Disease 2019 Pandemic
Author
Hirotaka Koga1)2) Yoshitomo Motomura1)3) Wakato Matsuoka1)2) Sayaka Okuzono1) Kenichi Tetuhara1)2) Noriyuki Kaku1)2) and Shouichi Ohga1)
1)Department of Pediatrics, Graduate School of Medical Sciences, Kyushu University
2)Emergency and Critical Care Center, Kyushu University Hospital
3)Center for the Study of Global Infection, Kyushu University Hospital
Abstract
Pertussis is particularly severe in unvaccinated infants less than 6 months of age. In 2020, coronavirus disease 2019 (COVID-19) has spread and is affecting people worldwide. The Japanese government has requested the closure of schools and staying at home and many nosocomial COVID-19 infections have been reported. As a result, the majority of parents refrain from visiting clinics and the chance for vaccination is limited because of physical distancing requirements. We herein report a case of pertussis in an unvaccinated infant requiring intensive respiratory care during the COVID-19 pandemic. It is necessary to raise awareness that vaccination is essential and urgent to protect children from vaccine preventable diseases, and at the same time to take infection control measures to ensure safe vaccination.
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