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THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY
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Vol.124, No.6, June 2020
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Original Article
Title
Effectiveness of Rotavirus Vaccines in Preventing Rotavirus Gastroenteritis Analyzed by a Test-Negative Design Case-Control Study and Genotype Diversity of Rotavirus after Introduction of Vaccines
Author
Shuji Nakata1) Toshihiko Mori2) Mayumi Ono3) and Takeshi Tsugawa3)
1)Nakata Pediatric Clinic
2)Department of Pediatrics, NTT East Sapporo Hospital
3)Department of Pediatrics, Sapporo Medical University School of Medicine
Abstract
To estimate the efficacy of rotavirus vaccines in reducing office visits for rotavirus gastroenteritis (RVGE) using a test-negative design case-control study, 744 stool samples were collected from the patients aged mainly younger than five years old visiting a pediatric clinic in Sapporo between October 2011, and September 2018. Samples were tested for the RV antigen by immunochromatography. The RV positive samples were analyzed for GPI genotypes by reverse transcription-polymerase chain reaction (RT-PCR). Vaccine effectiveness (VE) was calculated as (1-OR [odds ratio]) × 100 in a logistic regression model adjusting for age, sex, and month of onset.
Among 744 patients, 46 of 248 (19%) RV positive cases were vaccinated, whereas 231 of 496 (47%) RV negative controls were vaccinated. VE against RVGE was 73% (95% confidence interval [CI]: 61 to 81), at 69% (95% CI: 50 to 81) for monovalent RV vaccine (RV1) and 78% (95% CI: 64 to 87) for pentavalent RV vaccine (RV5), respectively. VE was recognized up to two years old, against severe RVGE. Although RV vaccines were effective against G1P[8] and G9P[8] RV, there could be a difference between the two vaccines regarding VE against non-G1P[8] RV. During the 2017-2018 seasons, uncommon genotypes of RV (equine G3P[8], G8P[8], G12P[8]) were prevalent. High vaccine uptake rates could affect the judgment of VE.
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Original Article
Title
A Questionnaire Survey for Pediatric Patients with Chronic Constipation Treated with Polyethylene Glycol
Author
Ayako Suda1)2) Yoshiko Nakayama2) Ei Shimazaki1) Yuta Maruyama1)2) Jyun Shimizu1) Yu Furui1) and Sawako Kato2)
1)Division of Pediatrics, Shinshu Ueda Medical Center
2)Department of Pediatrics, Shinshu University School of Medicine
Abstract
Background
Polyethylene glycol (PEG) is recommended as a first-line laxative in Europe and North America. In Japan, PEG 3350 plus electrolyte (PEG3350+E) was newly approved in 2018 for the treatment of chronic constipation.
Subjects and Methods
The study population consisted of 20 pediatric patients with chronic constipation treated with PEG3350+E at our hospital between March and July 2019. We conducted a questionnaire survey to assess the improvement of defecation status, satisfaction, and adverse events.
Results
Completed questionnaires were collected from 18 patients (90.0%) at 2 weeks and 17 patients (85.0%) at 4 weeks after the prescription of PEG3350+E. The reasons for dropout were bad taste or difficulty in daily use. Of the 17 patients who were able to continue administration for 4 weeks, all showed improvements in general defecation status and were satisfied with the treatment. Before administration, eight patients reported defecating more than once every 2 days, which improved to 17 after 2 and 4 weeks of treatment.
Bristol stool foam scale type 4 to 5 increased from 4 (22.2%) to 17 (100%) patients at 4 weeks. Diarrhea, as an adverse event, was observed in two (11.1%) cases, but treatment could be continued.
Conclusion
This study suggested that PEG3350+E is effective and well tolerated with high satisfaction among Japanese pediatric patients with chronic constipation.
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Original Article
Title
Clinical Practice and Problems of Critically Ill Pediatric Patients in Chiba Prefecture
Author
Tadashi Ishihara1)2)4) Hiromichi Hamada3)4) and Hiroshi Tanaka2)
1)Juntendo University Urayasu Hospital, Pediatric Emergency Center
2)Juntendo University Urayasu Hospital, Department of Emergency and Critical Care Medicine
3)Tokyo Women's Medical University Yachiyo Medical Center, Department of Pediatrics
4)Chiba Prefecture Pediatric Intensive Care Network
Abstract
[Background] In Chiba Prefecture, critically ill pediatric patients have been treated predominantly at the critical care center. Recently, two pediatric intensive care units (PICUs) have been constructed, which makes the total number of PICUs in Chiba equal to three. We assessed the clinical practice and problems of critically ill pediatric patients in Chiba Prefecture. [Patients and Methods] Data from critically ill pediatric patients aged < 15 years, who had been admitted to one of seven critical care centers or three PICUs in Chiba from January 2017 to December 2017, were analyzed. Characteristics of patients, disease categories, details of therapies, and outcomes were compared between the critical care centers and the PICUs. [Results] A total of 630 patients were registered in this study. Although the Pediatric Index of Mortality 2 score was significantly higher in the PICU group (1.7%) than in the critical care center group (0.8%), mortality was not significantly different between the two groups (both groups, 4%). More pediatric patients were admitted to the PICUs for respiratory failure and circulatory failure (57% and 22%, respectively), whereas more pediatric patients were admitted to the critical care centers for a dysfunction of the nervous system (54%). [Conclusion] Critically ill pediatric patients in Chiba tended to be admitted to PICUs than to critical care centers. There was a significant difference in severity but not in mortality between the PICUs and critical care centers. It is important to strengthen the collaboration between the PICUs and critical care centers to provide better medical care for critically ill pediatric patients in areas with limited medical resources in Chiba.
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Case Report
Title
Lactic Acidosis due to Relative Thiamine Deficiency Caused by Hyperglycemia in a Very-Low-Birthweight Infant
Author
Kei Uneoka Toshihiko Uchida Tatsuro Nawa Yuichiro Miura and Tatsuya Watanabe
Department of Neonatology, Miyagi Children's Hospital
Abstract
Thiamine is a crucial coenzyme of glycolysis. Thiamine deficiency causes beriberi and other symptoms. We encountered a case of lactic acidosis caused by thiamine deficiency in a very-low-birthweight infant, despite the continuous administration of thiamine. The patient was born at 31 weeks of gestation by an emergency cesarean section because of fetal growth restriction. Parenteral and enteral nutrition was initiated shortly after birth; lactic acidosis progressed in 23 hours after birth, for which aggressive thiamine administration was effective. In addition to lactic acidosis, complications of hyperglycemia, hypophosphatemia, and hypomagnesemia were present and were judged as conditions of refeeding syndrome (RS) accompanied by thiamine deficiency. Although 0.13 mg/kg of thiamine was added to the parenteral nutrition shortly after birth and the concentration of total blood thiamine just before aggressive thiamine administration was above the reference value, it is highly possible that a transient thiamine shortage occurred. This transient shortage might have been due to the relatively increased thiamine demand caused by the parenteral nutrition and steroid administration. The frequent occurrence of RS has recently been reported in very-low-birthweight infants with intrauterine growth restriction. Thus, if lactic acidosis of an unknown cause appears in a neonate with chronic malnutrition, as in this case, it is necessary to consider thiamine deficiency based on RS.
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Case Report
Title
An Infant with Catecholamine-producing Neuroblastoma Who Developed Junctional Ectopic Tachycardia during the Induction Course
Author
Yusuke Hayashi1) Jun Yoshimoto2) Takayuki Takachi3) Mitsunori Sato1) Kentarou Tomita1) Hiroyuki Kitamura1) Masahiro Tsubura1) Takuya Matsuda1) Saori Aiga1) Kenichiro Watanabe3) and Tatsuya Kawasaki1)
1)Department of Pediatric Critical Care, Shizuoka Children's Hospital
2)Department of Cardiology, Shizuoka Children's Hospital
3)Department of Hematology and Oncology, Shizuoka Children's Hospital
Abstract
Most cases of junctional ectopic tachycardia (JET) occur in the perioperative period of congenital heart disease. We report an infant without cardiac disease who developed JET during chemotherapy for neuroblastoma.
Case
A 20-month-old female infant was diagnosed with stage M primary neuroblastoma in the left adrenal gland with systemic metastasis. On the next day, she was admitted to the pediatric intensive care unit because of alveolar hemorrhage and respiratory failure. She required mechanical ventilation, and noradrenaline infusion was administered for hypotension. On the same day, narrow QRS tachycardia episodes were observed, which led to the diagnosis of paroxysmal supraventricular tachycardia. The sinus rhythm was restored with adenosine triphosphate (ATP). However, despite the cessation of noradrenaline, transient tachycardia repeated frequently, which was alleviated with ATP every time. These tachycardic episodes were well controlled with dexmedetomidine, induced hypothermia, and electrolyte correction. A retrospective review of the electrocardiograms revealed that these episodes were JET. The serum catecholamine level after the cessation of noradrenaline infusion was significantly elevated with the predominance of noradrenaline. Catecholamine release following neuroblastoma tumor lysis was considered as the main cause of the tachycardia episodes.
Conclusion
This case demonstrates that children without congenital heart disease may develop JET. Accurate diagnosis of arrhythmia is essential for prompt and strategic management.
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Case Report
Title
A Report of an Infant with Cerebellar and Brainstem Malformation Exhibiting Symptoms of Blepharoclonus
Author
Yoko Yamamoto1) Kyoko Hirasawa1) Ken Imai2) Yuya Sato1) Satsuki Totsu2) Masaki Wada2) Jun-ichi Takanashi3) and Satoru Nagata1)
1)Department of Pediatrics, Tokyo Women's Medical University
2)Maternal and Perinatal Center, Tokyo Women's Medical University
3)Department of Pediatrics, Tokyo Women's Medical University, Yachiyo Medical Center
Abstract
We report on a case of a 2-month-old girl who exhibited myoclonic movements of the eyelids immediately after birth and was diagnosed with blepharoclonus caused by cerebellar and brainstem hypoplasia. Although initially, the myoclonic movements of her eyelids were presumed to be a consequence of epilepsy, the video recording of the EEG undeniably disproved this phenomenon. Blepharoclonus was previously reported in patients with brain stem lesions in adulthood; however, there have been no reports about blepharoclonus in neonates or infants with cerebellar brainstem hypoplasia. The patient in this report had myoclonus of the eyelids immediately after birth. She exhibited neither abnormal eye movements nor other significant anomalies barring mild, self-limiting apnea attacks. No abnormal laboratory findings were detected. The simultaneous recording of video EEG showed no epileptic changes during the myoclonic movements of her eyelids without a remarkable response to any anticonvulsants. Prominent cerebellar and brainstem dysplasia was noted on imaging. Thus, we speculate that the movement of the eyelids (blepharoclonus) was caused by this abnormal structure. The frequency of symptoms decreased and disappeared when she was six months old. There have been few reports concerning infants with pontocerebellar hypoplasia, but no case with these symptoms. We thus conclude that the blepharoclonus, in this case, was caused by this anomaly of the brain, and the symptoms might improve with growth. However, the overall development might have been delayed without some intervention.
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Case Report
Title
Invasive Pneumococcal Infection Following Bone Marrow Transplantation for Juvenile Myelomonocytic Leukemia
Author
Daichi Sajiki Daiki Yamashita Ryo Maemura Hirotoshi Sakaguchi Nao Yoshida and Asahito Hama
Department of Hematology and Oncology, Children's Medical Center, Japanese Red Cross Nagoya First Hospital
Abstract
Invasive pneumococcal infection (IPI) is a life-threatening infection in immunocompromised patients. The rates of mortality and limb amputation are particularly high in patients who develop acute infectious purpura fulminans (AIPF). Herein, we describe the case of a 4-year-old boy who underwent haploidentical bone marrow transplantation for relapsed juvenile myelomonocytic leukemia 3 years ago. He was administered mycophenolate mofetil for chronic graft-versus-host disease and received the 13-valent pneumococcal conjugate vaccine once. He was admitted to our hospital with a high-grade fever, disturbed consciousness, and lower limb pain. His physical, laboratory, and imaging findings led to the diagnosis of influenza-associated encephalopathy, bacteremia caused by Streptococcus pneumoniae (serotype 24B, not included in the pneumococcal vaccine), disseminated intravascular coagulation, and AIPF. His condition gradually improved with intensive care, including artificial ventilation, antibiotics, and anticoagulants. He completely recovered without any sequelae, including neurological deficits. The necrotic tissue in his distal extremities caused by AIPF was conservatively treated with plastic surgery, and amputation was avoided. However, the patient again developed IPI after eight months. Computed tomography revealed spleen atrophy and the presence of Howell-Jolly bodies in peripheral blood erythrocytes, suggesting hyposplenism. Despite the widespread use of pneumococcal vaccines, IPI caused by nonvaccine serotypes requires special attention in immunocompromised patients, including those who receive hematopoietic stem cell transplantation.
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Case Report
Title
Atypical X-linked Severe Combined Immunodeficiency with a Phenotype of T+/lowB+NK+
Author
Yuta Eguchi Takehiko Doi Kosuke Noma Takaki Asano Satoshi Okada and Masao Kobayashi
Department of Pediatrics, Hiroshima University Hospital
Abstract
X-linked severe combined immunodeficiency (X-SCID) is characterized by the absence of T and natural killer (NK) cells. However, X-SCID due to an R222C mutation in the IL-2 receptor γ chain (IL2RG) leads to an atypical phenotype without an obvious lack of T and NK cells. An 11-month-old boy presented with a recurrent respiratory infection and chronic diarrhea starting at 5 months of age. At the time of admission, he presented with intractable respiratory syncytial virus bronchiolitis, pneumocystis pneumonia, and persistent cytomegalovirus antigenemia. Immunological analysis revealed a decreased number of T cells, a normal number of B cells with hypogammaglobulinemia, and an increased number of NK cells with normal activity. A genetic study identified a hemizygous R222C mutation in IL2RG. The patient was diagnosed with atypical X-SCID. The patient underwent umbilical cord blood transplantation with reduced-intensity conditioning. We observed engraftment, but the patient developed respiratory infection 30 days after the transplantation. Consequently, uncontrollable infection led to fatal multiple organ failure on post-transplant day 101. Since lymphocyte subset screening is possibly insufficient for the detection of atypical X-SCID because of a mutation in IL2RG, this case suggests that early accurate analysis and diagnosis are necessary for the appropriate treatment of patients with SCID.
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Case Report
Title
Two Cases of Eosinophilic Gastroenteritis that Took a Long Time to Diagnose and Could Observe Changes in Histopathology
Author
Kasumi Asai1) Tsunehisa Nagamori1) Sorachi Shimada1) Yoichiro Yoshida1) Hironori Takahashi2) and Hiroshi Azuma1)
1)Department of Pediatrics, Asahikawa Medical University
2)Department of Pediatrics, Asahikawa-Kosei General Hospital
Abstract
Eosinophilic gastroenteritis (EGE) is caused by excessive infiltration of eosinophilic leukocytes into the alimentary canal mucosa, muscular layer, and serosa. Symptoms and laboratory findings of eosinophilic enteritis are nonspecific. Therefore, it is difficult to distinguish them from other inflammatory bowel diseases. We experienced a case that was first diagnosed with ulcerative colitis and later diagnosed with eosinophilic enteritis. It took 3 and 5 years, respectively, to confirm the diagnosis, and we could observe changes in the pathological images over time. In both pathological images, the eosinophil count was initially within the normal range but increased over time. We consider that it is useful to perform repeated endoscopy and evaluation of pathological images in cases in which EGE is suspected.
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