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THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY
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Vol.120, No.4, April 2016
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Original Article
Title
The Efficacy of Bright Light Therapy for Children with Circadian Rhythm Sleep Disorder Who Exhibit Poor Morning Awakening
Author
Miki Inutsuka and Katsuhiko Yamada
Department of Pediatrics, Sasebo Chuo Hospital
Abstract
We investigated the effects of administering bright light therapy (BLT) to nine children being treated at our department who met the diagnostic criteria for circadian rhythm sleep disorder (CRSD) and exhibited markedly poor morning awakening. The subjects were administered BLT for 2 hrs. in the early morning for 7 consecutive days while being hospitalized. We compared bedtime and waking up time, physical symptoms, changes in heart rate and systolic blood pressure during the orthostatic test, and diurnal variation in salivary melatonin and cortisol concentrations before and after the treatment. After BLT, advanced sleep phase effects were observed, with all subjects going to sleep between 21-23 p.m. and waking up between 6 : 30-8 : 30 a.m., and physical symptoms improved compared with before the treatment. During the orthostatic test, heart rate, except for 7 min after standing and systolic blood pressure, after standing for 1 min, significantly elevated after BLT. This suggested that sympathetic nerve activity might be activated after BLT. The time at which peak salivary cortisol concentration was exhibited became earlier after BLT, which appears to have been involved in the improvement observed in morning awakening. However, the efficacy of BLT alone could not be proved because there was a possibility that the correction of the living environment due to hospitalization contributed to the improvement of the physical symptoms and laboratory findings. Results indicated that BLT with hospitalization is useful as non-pharmacological therapy for poor morning awakening but the high number of relapse cases suggested that the one-week treatment period was too short.
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Original Article
Title
GENECUBE for the Rapid Detection of Macrolide-resistant Mycoplasma Pneumonia
Author
Hideko Nakajima1) Eizaburo Ishii1) Daisuke Matsuoka1) Kesashi Aonuma2) Tetsuo Kubo3) Kazuo Arakawa4) Toshimi Kobayashi5) and Toshio Matsuura6)
1)Department of Pediatrics, Nagano Prefectural Suzaka Hospital
2)Department of Pediatrics, Nagano Municipal Hospital
3)Department of Pediatrics, Hokushin General Hospital
4)Arakawa Children's Clinic
5)Kobayashi Children's Clinic
6)Matsuura Children's Clinic
Abstract
[Background] The prevalence of macrolide-resistant Mycoplasma pneumoniae has increased in recent years, prompting the need for methods allowing rapid diagnosis of an infection and proper use of antibiotics in the treatment of outpatients.
[Methods] We enrolled 230 patients who underwent examinations at our hospital and nearby hospitals and clinics, between December 2011 and March 2013, for respiratory tract infections. M. pneumoniae infection was diagnosed by clinical examinations, chest radiography, white blood cell counts, C-reactive protein levels, rapid immunoglobulin M tests, passive agglutination (PA), and quenching probe polymerase chain reaction (QP-PCR, GENECUBE) using nasopharyngeal swab specimens. We analyzed the patients' age, and symptom duration before consulting hospitals, initiating antibiotics, and deciding on hospitalization. Sensitivity to macrolide therapy was also examined with melting curve analysis. M. pneumoniae infection was diagnosed in 156 patients using loop-mediated isothermal amplification (LAMP).
[Results] QP-PCR revealed that 43.5% patients were PCR positive, while 56.5% were PCR negative. No significant differences were detected in rapid immunoglobulin M antibody detection results between the QP-PCR-positive and negative group. Additionally, 91.0% patients in the QP-PCR-positive group had macrolide-resistant M. pneumoniae. No significant differences were observed in clinical symptoms and examination results between the macrolide-resistant M. pneumonia group and macrolide-sensitive group. QP-PCR results were consistent with LAMP results, but had a lower positive conformity ratio than PA.
[Conclusions] QP-PCR enabled rapid, simple, and noninvasive detection of M. pneumoniae, and was as effective as LAMP. Additionally, QP-PCR was more effective than methods compared with our examination used for the detection of macrolide-resistant M. pneumoniae.
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Original Article
Title
Epidemiology of Pneumococci Obtained from the Nasopharynx of Infants and Young Children before and after Transition to 13-valent Pneumococcal Conjugate Vaccine: Change of Serotypes
Author
Akiyoshi Nariai Takanori Yanai and Taichi Kanetaka
Department of Pediatrics, Yokohama Minami Kyosai Hospital
Abstract
After the 7-valent pneumococcal conjugate vaccine (PCV7) introduction in 2010, we have determined the serotypes of pneumococci obtained from the nasopharynx of infants and young children with lower respiratory tract infections. This report presents the results from 2013 (between January and October 2013, before the transition to the 13-valent pneumococcal conjugate vaccine [PCV13]), to 2014 (between November 2013 and October 2014 after the transition to PCV13), in addition to previously reported results from 2010 to 2012.
The rates of PCV7 serotypes were 61%, 6.4%, and 1.0% in 2010, 2013, and 2014, respectively; thus demonstrating nearly complete elimination of the PCV7 serotypes.
Of the six serotypes added to PCV13, only serotypes 3, 6A, and 19A were detected at rates of 23% and 19% in 2013 and 2014, respectively, essentially unchanged from 2010 at a rate of 20%. However, by focusing on the first and second halves of 2014, the rates of the six serotypes added to PCV13 declined from 25% in the first half of the year to 12% in the second half of the year.
In contrast, the rates of the non-PCV13 serotypes increased to 71% and 80% in 2013 and 2014, respectively, a substantial increase from 2010, at a rate of 19%. Because annual pneumococcal detection rates remain around 22%, and around 20 serotypes have been confirmed, which indicate no change since 2010, we believe that pneumococcal colonization will continue in infants and young children while replacing serotypes.
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Case Report
Title
Acute Magakaryoblastic Leukemia in a Child with Down Syndrome and Chronic Heart Failure
Author
Hiroki Takehara1) Masahiro Sekiguchi1) Motohiro Kato1)2) Ryosuke Shiozawa1) Taiyu Hayashi1) Nobutaka Shimizu1) Kentaro Watanabe1) Mitsuteru Hiwatari1) Junko Takita1) and Akira Oka1)
1)The Department of Pediatrics, University of Tokyo
2)The Department of Cell Therapy and Transplantation Medicine, University of Tokyo
Abstract
Myeloid leukemia in patients with Down syndrome (ML-DS) exhibits a favorable response to chemotherapy, especially to cytarabine. Considering the potential risk of excess toxicity, reduction of intensity of chemotherapy has been attempted in ML-DS cases. However, the appropriate intensity in ML-DS with severe heart failure caused by heart anomaly has not been established. We describe here, an ML-DS case with severe chronic heart failure caused by congenital heart anomalies. The patient tolerated palliative chemotherapy with reduced doses of cytarabine and etoposide well, and survived for 10 months from diagnosis. Accumulation of similar cases is needed to establish appropriate therapy.
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Case Report
Title
Carbamazepine-induced Severe Drug Eruption Developed in a Boy with HLA-A*31 : 01
Author
Ryota Igarashi Takayuki Hoshina Masahiro Ishii Masayuki Shimono and Koichi Kusuhara
Department of Pediatrics, School of Medicine, University of Occupational and Environmental Health
Abstract
The difference of human leukocyte antigen (HLA) genotype is associated with the development of carbamazepine (CBZ)-induced adverse drug reaction. We herein report a 15-year-old boy found to have CBZ-induced severe drug eruption. The patient presented with continuous fever from 3 weeks after the initiation of CBZ as a treatment for temporal lobe epilepsy, and skin rash appeared 4 weeks later. Serum chemistries showed the elevation of aspartate aminotransferase and alanine aminotransferase. In addition, congestion of bilateral bulbar conjunctiva, petechiae of the soft plate and cervical lymph node swelling appeared. Discontinuation of CBZ and initiation of prednisolone promptly improved symptoms. We diagnosed CBZ-induced severe drug eruption because of liquefaction degeneration and infiltration of inflammatory cells of the basal layer of the epidermis in skin tissue specimens and positive results of skin patch test and drug-induced lymphocyte stimulation test for CBZ. Histopathological findings showed erythema multiforme major because no severe mucosal lesions observed in Stevens-Johnson syndrome or toxic epidermal necrolysis appeared. An accurate diagnosis of drug-induced hypersensitivity syndrome was not provided because serum IgG antibody titres to human herpesvirus 6 were not elevated. The patient carried the HLA-A*31 : 01 allele, which has been reported to be associated with the development of adverse drug reactions in the Japanese population. In Japanese, the evaluation of HLA typing is not recommended before the initiation of CBZ. The accumulation of cases throughout Japan is needed to analyze the involvement of HLA-A*31 : 01 in each type of severe drug eruption.
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Case Report
Title
A Case of DDAVP-Effective Nephrogenic Diabetes Insipudus
Author
Nao Akiyama1) Sachiko Kaburagi2) Kentaro Ohmi1) Yoshihiro Takasato2) Mio Kohno1) Mizue Tomita1) Nobuaki Shikoro1) and Isamu Kamimaki1)
1)Department of Pediatrics, National Hospital Organization, Saitama National Hospital
2)Department of Pediatrics, Keio University, School of Medicine
Abstract
We report a case of an 11-year-old boy who was diagnosed as having nephrogenic diabetes insipidus (NDI) in his neonatal period. He had a history of fever with hypernatremia, low urine osmolality and increased serum concentration of arginine vasopressin (AVP). DNA analysis of the V2 receptor gene identified a missense mutation of L90P.
Water restriction test and the 1-Desamino-8-D-arginine vasopressin (DDAVP) test at age 1 neither increased his urine osmolality nor decreased his urine output, revealing that he was completely resistant to antidiuretic effect of AVP. He was treated with thiazide diuretic (hydrochlorothiazide) and potassium-sparing diuretic (spironolactone), and his frequency of urination was controlled. At age 11, his urine volume was 5-6 liters per day. The control was not satisfactory. Water restriction test and DDAVP test at this time showed increased urine osmolality and decreased urine output, revealing that his sensitivity for AVP had improved. High volume intranasal DDAVP before bedtime allowed him to sleep longer at night.
Even if the clinical presentation of the patient is improving, his or her sensitivity for AVP may improve. Thus, we recommend performing the water restriction test repeatedly.
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Case Report
Title
Management of Respiratory Syncytial Virus Infection in Infants with Congenital Bronchial Stenosis: Two Case Reports
Author
Mai Fukui Madoka Sawada Takashi Soga and Yoh Umeda
Children's Medical Center, Showa University Northern Yokohama Hospital
Abstract
Lower respiratory tract infection due to respiratory syncytial virus (RSV) in infants may manifest as bronchiolitis and pneumonitis. Infants with chronic lung disease or congenital heart disease and preterm infants are at an increased risk of hospitalization and mechanical ventilation after RSV infection.
We describe our experience in managing RSV infection in 2 infants with tetralogy of Fallot and congenital bronchial stenosis: (1) a 20-day-old girl with no pulmonary valve and carinal compression and (2) a 1-month-old girl with right tracheal bronchus and anomalous origin of the left pulmonary artery from the right pulmonary artery, wrapping around the trachea. Both needed controlled mechanical ventilation. Determination of adequate expiration time, positive end-expiratory pressure for maintaining dilation of the bronchial stenosis, and peak inspiratory pressure for adequate inspiratory volume was difficult. We found that computed tomography, bronchoscopy, and pressure-volume loop were valuable for determining the optimal ventilator settings.
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Case Report
Title
A Case of Wilson Disease with Hepatocirrhosis Presenting with Lower Myalgia
Author
Yukari Sakurai1) Hironori Takahashi1) Tsunehisa Nagamori1) Shin Koyano1) Hiroshi Azuma1) and Koichi Mizuta2)
1)The Department of Pediatrics, Asahikawa Medical University
2)The Department of Transplant Surgery, Jichi Medical University
Abstract
Wilson disease is one of the few remediable inborn errors of metabolism. However, it can often be difficult to diagnose because of divergent initial symptoms. Here we describe a case of Wilson disease with cirrhosis initially presenting solely with myalgia of the lower limbs. A 12-year-old boy was referred to our hospital for continuous calf pain. His serum creatine kinase level was elevated at 682 U/L and T2-weighted magnetic resonance imaging showed a high intensity area in the muscles of the distal lower extremities. However, the clinical manifestations were not concordant with either polymyositis or dermatomyositis. On the other hand, initial laboratory examinations suggested liver dysfunction; therefore the boy underwent abdominal computed tomography, which clearly revealed cirrhosis. Subsequently he was diagnosed with Wilson disease based on decreased ceruloplasmin levels, increased urinary copper excretion, and the presence of a Kayser-Fleischer rings. After initiation of Trientin administration, myalgia of the lower extremity improved. He underwent living donor liver transplantation 6 months after the diagnosis and has shown favorable progress.
Previous descriptions of the musculoskeletal manifestations associated with Wilson disease have been limited to osteoarthritis and reports of muscular symptoms are rare. Although the pathophysiology was unclear, the present patient's myalgic pain was considered to be associated with Wilson disease because it resolved after drug treatment for this condition. This case report suggests that Wilson disease should be considered in patients with unexplained hepatic dysfunction, even if they only present with pain in the lower extremities.
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Brief Report
Title
Study of the New AZT Regimen for Neonates for Prevention of Mother-to-child Transmission
Author
Mari Honda1) Mizue Tanaka1) Shinichi Hosokawa1) Ei Kinai2) Moe Akahira1) Hiroyuki Shichino1) Noriko Sato1) and Takeji Matsushita1)
1)Pediatrics, National Center for Global Health and Medicine
2)Center for AIDS Research, National Center for Global Health and Medicine
Abstract
Zidovudine (AZT) at 2 mg/kg every 6 h for 6 weeks is given to human immunodeficiency virus (HIV)-exposed infants for prevention of mother-to-child transmission. A pilot prospective study was performed to examine the infection rate and safety of AZT at 4 mg/kg twice daily for 6 weeks, with the aim of reducing the side effects of AZT. Ten cases were included in our study. All mothers had received antiretroviral therapy since early pregnancy and their HIV RNA levels were kept low. All infants were delivered by caesarean section and breastfeeding was avoided. Results showed that none of the infants were infected, 4 had anemia after 1 month, and none had hyperlacticacidemia or leukopenia. Thus, the new postnatal AZT regimen appears to be safe and effective if mothers had achieved good control. A larger number of patients is needed to confirm the results of this study.
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