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THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY

Vol.121, No.3, March 2017


Original Article
1. Divorce in Families of Children with Severe Motor and Intellectual Disabilities
2. Multidimensional Control of Catheter-related Blood Stream Infection
3. Comparison of the LAMP Method and the Rapid Antigen Test in the Detection of Mycoplasma Pneumoniae
4. Clinical Manifestation of Children Infected with an Cryptosporidium Outbreak
5. Extracorporeal Membrane Oxygenation for the Treatment of Children with Myocarditis: a Single-center Experience
Case Report
1. A 13-year-old Boy with Dilated Cardiomyopathy Diagnosed on Follow-up for Premature Ventricular Contractions
2. Atopic Myelitis with Acute Onset of Gait Disturbance and Dysuria
3. Liothyronine Was Effective in Two Cases of Consumptive Hypothyroidism Associated with Multiple Hepatic Hemangiomas


Original Article
Title
Divorce in Families of Children with Severe Motor and Intellectual Disabilities
Author
Chikako Ogawa1) Kiyokuni Miura2) Yuji Ito1) Hiroyuki Kidokoro1) Akihiro Hirakawa3) Tamiko Negoro4) and Jun Natsume1)5)
1)Department of Pediatrics, Nagoya University Graduate School of Medicine
2)Department of Pediatric Neurology, Toyota Municipal Child Development Center
3)Center for Advanced Medicine and Clinical Research, Nagoya University Hospital
4)Department of Clinical Psychology, Faculty of Child Development, Nihon Fukushi University
5)Developmental Disability Medicine, Nagoya University Graduate School of Medicine
Abstract
Objective: To examine the epidemiology and reveal risk factors for parental divorce (PD) in families of children with severe motor and intellectual disabilities (SMID).
Methods: We reviewed the medical records of 39 children with SMID in the Toyota Municipal Child Development Center. We determined the divorce rate among families of children with SMID and compared the background factors of patients who had and had not experienced PD.
Results: Six patients (5 boys) from six families experienced PD. Among the six patients who had experienced PD, the causes of SMID were varied, and five of the patients who had experienced PD had sleep disorders. There were no significant differences in medical care, scores for assessing the extreme intensive/extensive medical care among patients with SMID, parental age, siblings, and interventions among patients who had and had not experienced PD. Two families of patients who had experienced PD and three families of patients who had not experienced PD were dual-income families. Abuse, suspicion of abuse, or domestic violence was observed in four families of patients who had experienced PD (p< 0.01), and all of the abuse or violences was caused by fathers.
Conclusion: The rate of divorce is not abnormally high in parents of children with SMID. Abuse or domestic violence is a risk factor for PD in children with SMID. This study indicates that avoiding PD in families with children with SMID may require treatment of sleep disorders and the need for family support focused on the role of fathers.




Original Article
Title
Multidimensional Control of Catheter-related Blood Stream Infection
Author
Kentaro Watanabe1) Motohiro Kato1)2) Yutaka Harita1) Masahiro Sekiguchi1) Ryosuke Shiozawa1) Mitsuteru Hiwatari1) Junko Takita1) and Akira Oka1)
1)Department of Pediatrics, The University of Tokyo
2)Department of Cell Therapy and Transplantation Medicine, The University of Tokyo Hospital
Abstract
Background
Central venous catheters (CVCs) play an important role in the treatment of patients requiring long-term chemotherapy. However, CVCs are associated with an increased risk of developing catheter-related blood stream infection (CRBSI). In this study, we implemented practical measures to reduce the incidence of CRBSI and verified their effects.
Patients and Methods
The measures included changeover of closed system devices, reducing the frequency of catheter locks, shortening the distance from the connectors of the catheter to the T-shaped stopcocks where blood samples are taken, and improving the guidance provided on infection control for patients, families, and staff. Pediatric patients with hematologic/oncologic diseases in our department, treated with CVCs, were monitored for 4 end-points: the occurrence of positive blood culture, CRBSI, febrile neutropenia, and unscheduled removal of the CVC due to infection. We compared these end-points between the periods before (between April-August 2013) and after (between October 2013 and October 2014) the implementation of the measures using survival analyses and Cox proportional-hazards regression models adjusted for patient characteristics.
Results
Compared with the period before the implementation, the hazard ratios of positive blood cultures, CRBSI, and unscheduled removal of the CVC due to infection in the period after the implementation of the measures were 0.21 (p=0.0035), 0.21 (p=0.0080), and 0.10 (p=0.0032), respectively. As for the frequency of febrile neutropenia, there was no significant change between the two periods.
Conclusion
Our multidimensional approach to infection control may reduce the risk of CRBSI.




Original Article
Title
Comparison of the LAMP Method and the Rapid Antigen Test in the Detection of Mycoplasma Pneumoniae
Author
Mami Kuroda Fujio Kakuya Hitoshi Okubo and Hiroaki Fujiyasu
Departments of Pediatrics, Furano Kyokai Hospital
Abstract
Several diagnostic methods are utilized for the detection of Mycoplasma pneumoniae (MP), including serological tests, rapid antigen tests and genetic tests. The diagnostic reliability of the loop-mediated isothermal amplification (LAMP) method in the acute phase of MP infections was prospectively compared with that of two rapid antigen tests (Ribotest® and Prime check®) on the basis of the result of the culture method. Overall, 297 patients who were suspected of lower respiratory tract infections were enrolled. The LAMP method, the rapid antigen tests and culture method were performed in all cases. Thirty cases with positive culture were diagnosed to have definitive MP infections. The LAMP method had a sensitivity of 86.7% and a specificity of 100%, whereas Ribotest® had a sensitivity of 40.0% and a specificity of 91.4%, and Prime check® had a sensitivity of 7.7% and a specificity of 99.1%. Prime check® had a very low sensitivity. In addition, Ribotest® and Prime check® had low positive predictive values of 34.3% and 50.0%. The LAMP method is effective in the diagnosis of acute phase of MP lower respiratory tract infections in comparison with Ribotest® and Prime check®.




Original Article
Title
Clinical Manifestation of Children Infected with an Cryptosporidium Outbreak
Author
Hanako Yamanouchi1) Hiroshi Hataya1) Hiroshi Sakakibara1) Toshiro Terakawa1) Mihoko Isogai2) Kenta Ito2) and Yuho Horikoshi2)
1)Department of General Pediatrics, Tokyo Metropolitan Children's Medical Center
2)Department of Infectious Diseases, Tokyo Metropolitan Children's Medical Center
Abstract
Cryptosporidium spp. cause cryptosporidiosis, a form of zoonosis affecting the gastrointestinal epithelium cells and causing gastroenteritis. Cryptosporidium is usually pathogenic in an immunocompromised host, but occasionally affects immunocompetent hosts who have experienced travelers' diarrhea while in developing countries, swum in contaminated water, or ingested contaminated food or drink. Pediatric cases have rarely been reported in Cryptosporidium outbreaks in Japan. However, in June 2014, 118 primary school children in Tokyo experienced the largest recorded pediatric Cryptosporidium outbreak after engaging in farm activities. Fourteen of the affected children were seen at Tokyo Metropolitan Children's Medical Center, and the patients' clinical and laboratory information were reviewed at our hospital. The median incubation period was 7 days (interquartile range 6-7 days). All patients experienced diarrhea and abdominal pain, and 6 experienced emesis. Only 4 children were febrile and 1 child developed bloody diarrhea. Nine patients required intravenous fluid administration at our emergency department. Two of them were hospitalized for intravenous fluid administration without antibiotics, and were discharged after 5 and 6 days, respectively. As Cryptosporidium cannot be detected by routine stool laboratory tests for common viral or bacterial infectious gastroenteritis, special staining or polymerase chain reaction (PCR) should be used for suspected cases. Cryptosporidium gastroenteritis has a longer incubation period of 1 week compared to common infectious gastroenteritis. In an outbreak setting or among returning travelers with diarrhea, a longer incubation period should raise suspicion of Cryptosporidium infection and prompt notification of the public health agency and specific testing to confirm infection.




Original Article
Title
Extracorporeal Membrane Oxygenation for the Treatment of Children with Myocarditis: a Single-center Experience
Author
Susumu Urata1) Taiyu Hayashi1) Nao Nishimura2) Satoshi Nakagawa2) and Hiroshi Ono1)
1)Division of Cardiology, National Center for Child Health and Development
2)Division of Critical Care Medicine, National Center for Child Health and Development
Abstract
Background: Although extracorporeal membrane oxygenation (ECMO) is used to provide a bridge to recovery in patients with myocarditis, reports on survival outcomes in Japan are limited. We aimed to elucidate the characteristics and survival outcomes of pediatric myocarditis patients treated with ECMO, which should help us develop strategies for improving the prognosis of myocarditis in children.
Methods: We reviewed medical records of 19 consecutive myocarditis patients younger than 18 years who were admitted to our institution. The clinical, laboratory and echocardiographic data were compared between patients treated with ECMO (n=11) and those without ECMO (n=8).
Results: In patients treated with ECMO, which was introduced 76 (44-96) minutes after the arrival to our institution, the median duration of ECMO support was 7 (5-8.5) days. Of those treated with ECMO, 2 patients died during hospitalization and 1 patient had severe neurological sequelae; those with poor prognosis had developed cardiopulmonary arrest or disseminated intravascular coagulation before the introduction of ECMO. Compared with patients treated without ECMO, patients treated with ECMO showed smaller left ventricular end-diastolic diameter z-score (+0.34 [-0.9-+1.1] vs+2.8 [+1.3-+4.7], p< 0.01) at presentation.
Conclusion: To improve the prognosis of myocarditis, ECMO needs to be initiated prior to the development of cardiopulmonary arrest and multi-organ failure. A minimal left ventricular dilatation despite the symptomatic heart failure would indicate a rapidly deteriorating clinical course, which might warrant ECMO support.




Case Report
Title
A 13-year-old Boy with Dilated Cardiomyopathy Diagnosed on Follow-up for Premature Ventricular Contractions
Author
Shin Ono Etsuko Tsuda Shinsuke Hoshino Yuki Taniguchi Heima Sakaguchi and Isao Shiraishi
Department of Pediatric Cardiology, National Cerebral and Cardiovascular Center
Abstract
A 7-year-old asymptomatic boy was revealed by a school-based heart disease screening to have premature ventricular contractions (PVCs). He was revealed to have PVCs at 9-year old again and visited a previous hospital. Polymorphic PVCs were detected by 24-hour Holter electrocardiogram (ECG) however the absence of other organic diseases were confirmed by medical screening. He had undergone annual follow-up with 24-hour Holter ECG. The observation of 24-hour Holter ECG had become worse at age 13 and he developed dilated cardiomyopathy (DCM) after several months. Pediatric isolated PVCs are generally known as benign, at the same time PVCs are associated with DCM. It is best to evaluate other cardiac functions of PVC patients who need medical follow-up.




Case Report
Title
Atopic Myelitis with Acute Onset of Gait Disturbance and Dysuria
Author
Tomoko Uehara1) Ryo Imakiire2) Suzuka Toguchi1) and Asao Yara1)
1)Naha City Hospital
2)Kagoshima Prefectural Oshima Hospital
Abstract
Transverse myelitis is a rare disease in children. To identify its cause is often difficult because it can be caused by many factors. We report a 11-year-old-boy with acute transverse myelitis who presented with acute onset of gait disturbance, dysuria and atopic dermatitis. Blood tests showed hyper IgEemia and high titer of mite antigen specific IgE. Spinal cord MRI showed an abnormal intensity located at the cervical cord. Based on clinical, imaging, and laboratory findings, Atopic myelitis (AM) was diagnosed. He received steroid pulse therapy with methylprednisolone of 1 g/day for three days followed by oral steroids and intravenous immunoglobulin therapy. After pulse therapy symptoms have gradually improved and he made a complete recovery with no residual problems.
AM is a myelitis associated with atopic disorders, such as atopic dermatitis, bronchial asthma, allergic rhinitis, and hyperIgEemia. Although adult cases with AM have been reported in Japan, there are few reports of child case. AM should be considered in even children with atopic dermatitis and respiratory allergy in conjunction with neurological symptoms.




Case Report
Title
Liothyronine Was Effective in Two Cases of Consumptive Hypothyroidism Associated with Multiple Hepatic Hemangiomas
Author
Takehito Otani1) Masahiro Goto2) Ayumi Nogi1) Satoko Yanagihara1) Hiroshi Hataya1) Hiroshi Sakakibara1) Toshiro Terakawa1) Aya Shimada2) Shinji Higuchi2) Kentaro Miyai2) Masaki Takagi2) and Yukihiro Hasegawa1)2)
1)Department of General Pediatrics, Tokyo Metropolitan Children's Medical Center
2)Division of Endocrinology and Metabolism, Tokyo Metropolitan Children's Medical Center
Abstract
Introduction:
In multiple hepatic hemangiomas, the overexpression of type 3 iodothyronine deiodinase in the endothelial cells leads to the degradation of T4 to reversedT3 (rT3) and T3 to T2, thus eventually leading to consumptive hypothyroidism, which may render the treatment of hypothyroidism difficult. In this condition, known as consumptive hypothyroidism, administration of levothyroxine alone may be insufficient to maintain normal levels of TSH. We report herein the efficacy of liothyronine in two cases of consumptive hypothyroidism associated with hepatic hemangiomas.
Case 1:
An one-month-old male infant was referred to our institution with abdominal distention and jaundice. Ultrasonography and contrast-enhanced computed tomography revealed multiple hepatic hemangiomas. Levothyroxine 10μg/kg and liothyronine 2μg/kg were administered to treat his hypothyroidism successfully.
Case 2:
A two-month-old male infant came to our institution with jaundice. Ultrasonography and contrast-enhanced computed tomography revealed multiple hepatic hemangiomas, and laboratory analysis showed low free T3 and normal free T4 levels. His thyroid function normalized after administration of liothyronine 1μg/kg alone.
Discussion:
Based on the mechanism of consumptive hypothyroidism described above, our findings suggest that liothyronine is useful for consumptive hypothyroidism associated with hepatic hemangiomas. TSH level returned to the normal range in this patient in less than one month. The period was shorter than that required in most cases treated with levothyroxine.
Conclusion:
Administration of liothyronine can be effective against hypothyroidism caused by infantile hepatic hemangiomas in some patients.




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