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THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY
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Vol.116, No.7, July 2012
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Original Article
Title
Clinical Aspects of 65 Pediatric Patients with Pulmonary Dysfunction Caused by H1N1 Influenza 2009 Infection
Author
Katsuji Kuwakado Mariko Ishihara Yoshio Arakaki Hisako Iida Jun Ishizuka Yasunobu Miki Norihisa Miyashita Daisuke Yoshinaga Saki Ookubo Keiji Tsuchimoto Yousuke Hayama Takeo Mukai Kanako Kawamura Yoshiyuki Hanaoka Kayo Ogino Kanako Yoshizaki Aya Mima Tamako Maeba Mariko Sawada Tomohiro Hayashi Noriko Tanaka Masamichi Kubota Akihito Takahashi Akiko Ishihara Yoshinobu Nishida Mitsuhiro Fujiwara Shin-ichi Watabe and Kenji Waki
Department of Pediatrics, Kurashiki Central Hospital
Abstract
The number of pediatric outpatients with influenza A/H1N1 2009 was 2.2-fold that of patients with seasonal influenza in the two preceding seasons (September 2007 to May 2008 and December 2008 to June 2009) and the ratio of inpatients to outpatients was 2.6-fold higher. This ratio was particularly high among children aged between 3 and 9 years.
From among these patients, sixty-five 65 patients required oxygen support due to SpO2 levels of ≤93% and 6 of them required mechanical respiratory support. All patients survived after receiving antiviral drugs. We initiated treatment in 66% of them within 48 hours of illness onset and 71% developed a cough before fever. Although 52% of them had a history of asthma or wheezing, neither of these underlying conditions seemed to have any notable influence including on the duration of oxygen support. Leukocyte counts and CRP levels became elevated in a substantial proportion of the patients within 48 hours of illness onset, and those with levels of ≥12,000 /μL required a longer duration of oxygen support (p<0.01). Microorganisms were undetectable in either blood samples or bronchial sputum cultures. Chest X-ray films upon admission revealed hyperinflation in 82% of the patients and opacity in 58%. Four of 11 children with extensive lobular opacities upon admission required oxygen support for over five days. Bronchoscopic sputum aspiration relieved the respiratory disturbances in intubated patients. The removal of mucus plugs with a bronchoscope resulted in rapid recovery of respiratory condition for intubated patients. On initial X-rays films, consideration of opacities as atelectasis rather than pneumonia is considered appropriate. The effects of corticosteroids at dosages used to treat asthma were administered to 35% (23 cases) of the patients with mainly a history of asthma or who were wheezing on admission, but the specific effects were unclear because antiviral drugs seemed highly effective. Among 30 patients with wheezing on admission, the group given steroid required oxygen support for a longer period (p=0.02). This suggests that in individuals infected with influenza A/H1N1 2009 in the group of patients requiring oxygen support for more than 72 hours, there is a possibility corticosteroids may prolong convalesce in such instances.
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Original Article
Title
Two Cases of Pediatric Gastrolitiasis with Trichologia and Trichophagia and a Review of 95 Pediatric Cases of Gastrolithiasis in the Japanese Literature
Author
Hiroyuki Yamada1) Naoki Watanabe1)2) Tomoko Kurashige1)2) Ayako Kamata1) Yoshiyuki Ohtomo1) Taizo Sato3) Masahiko Urao4) Shinichi Niijima1) and Toshiaki Shimizu5)
1)Department of Pediatrics, Juntendo University Nerima Hospital
2)Department of Pediatrics, Tokyo Metropolitan Health and Medical Corporation Toshima Hospital
3)Sato Mental Clinic
4)Department of Pediatric Surgery, Juntendo University Nerima Hospital
5)Department of Pediatrics, Juntendo University Faculty of Medicine
Abstract
We reported 2 cases of gastrolithiasis in grade-school girls with trichologia and trichophagia. The first case was an 11-year-old girl who underwent abdominal surgery because of bezoar ileus following endoscopic extraction. The second case was a 9-year-old girl whose bezoar was extracted successfully after two endoscopy procedures. Then we summarized 95 cases of pediatric gastrolithiasis in Japan. Of these, 93 were girls and 93% had trichobezoar. Abdominal surgery was performed on 80 cases, including 11 cases who had undergone unsuccessful endoscopic extraction, and six cases were treated successfully by endoscopic extraction.
Treatment of gastrolithiasis should be considered on the basis of age and the size of the bezoar, and when its diameter is less than 3 cm endoscopic extraction is recommended. However, we encountered bezoar ileus following endoscopic extraction. Therefore, we strongly recommend that endoscopic extraction should be performed in facilities which can offer abdominal surgery when needed.
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Original Article
Title
Clinical Characteristics of Neonatal Arterial Cerebral Infarction during 25 Years
Author
Kenjiro Kikuchi1)2) Shin-ichiro Hamano1) Ryuki Matsuura1)2) Masaki Shimizu3) Ryoichi Sakuta4) and Hiroyuki Ida2)
1)Division of Neurology, Saitama Children's Medical Center
2)Department of Pediatrics, Jikei University School of Medicine
3)Division of Neonatology, Saitama Children's Medical Center
4)Center for Child Development and Psychosomatic Medicine, Dokkyo Medical University
Abstract
Objectives: Clinical investigations of patients with neonatal cerebral infarction have rarely been reported in Japan. We set out to determine the clinical characteristics of neonatal cerebral infarction.
Methods: We retrospectively reviewed the medical records of patients in our institution for the following characteristics: sex, completed gestational weeks at birth, birth weight, Apgar scores, time of infarction onset, clinical presentations, neuroimaging findings, and neurological sequelae. The criteria for establishing a diagnosis of neonatal cerebral infarction were as follows: (1) infarctions found from birth to 28 days of age and (2) infarcted lesions found in the cerebral artery on neuroimaging, such as computed tomography and magnetic resonance imaging. Furthermore, we reviewed the literature on neonatal cerebral infarction in Japan.
Results: Twenty-one patients (14 boys and 7 girls) were enrolled. Eighteen patients were born at term. Low Apgar Scores (<7) at 1 minute was found in 6 patients, infection in 2, and maternal complications in 4. In 90% of patients, the onset of infarction was noted within 2 days after birth. As clinical presentations, seizures were found in 52.4% patients and respiratory disorder, in 33.3%. Infarctions occurred most commonly in the left hemisphere than in the right hemisphere, and the middle cerebral artery was the most common vessel involved (81.0%). The median follow-up duration was 6.9 years. Neurological sequelae were found in 10 patients: motor impairments, in 8; mental retardation, in 5; and epilepsy, in 3. All findings, except that of the right hemisphere being the dominant site of infarction, were similar to those reported previously.
Conclusions: Neonatal cerebral infarction was more frequent in boys within 2 days after birth and most commonly occurred in the middle cerebral artery. Neurological deficits were found in about half of the cases. These results indicate that the clinical characteristics of neonatal cerebral infarction are similar to those reported in other countries. The follow-up duration of our cases and other reports in Japan varied, and longer durations of follow-up, at least until the patients are of school age, are needed in order to evaluate the long-term neurological outcomes in Japan.
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Original Article
Title
A Hospital-based Study of Intracranial Hemorrhage Caused by Vitamin K Deficiency in Infants
Author
Nobuyuki Yotani1) Akira Ishiguro1)2) Tetsuya Mori3) Masaaki Kumagai3) Nobuhito Morota4) Mikiko Miyasaka5) and Hirokazu Sakai1)
1)Department of General Pediatrics and Interdisciplinary Medicine, National Center for Child Health and Development
2)Division of Hematology, National Center for Child Health and Development
3)Division of Pediatric Oncology, National Center for Child Health and Development
4)Division of Neurosurgery, National Center for Child Health and Development
5)Department of Radiology, National Center for Child Health and Development
Abstract
Since the introduction of vitamin K prophylaxis in the 1980's, the incidence of vitamin K deficiency bleeding (VKDB) has decreased remarkably. However, late VKDB is still reported. This is a retrospective analysis of children who were diagnosed with VKDB at our hospital from March 2002 to September 2010. Thirteen infants (8 male) were diagnosed with late VKDB. The median age was 62 days (41-121). The infants developed non-specific symptoms including "not doing well". In all, serum PIVKA-II levels were increased and coagulopathy was resolved within 24 hours after the administration of vitamin K. CT scan showed cerebral edema in 12 cases (92%) and mid-line shift in 9 cases (69%). Neurosurgical intervention was performed in 10 cases (77%): one patient died, 4 had neurological sequelae and 8 recovered without sequelae. Seven had underlying diseases, biliary atresia in 5 and hepatitis in 2 cases. In the remaining 6 cases diagnosed as idiopathic VKDB, 3 did not receive a complete series of the recommended vitamin K prophylaxis. While the current prophylaxis may not completely eradicate VKDB, we believe that full adherence to the recommended prophylaxis regimen will decrease the incidence of VKDB.
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Original Article
Title
Pneumococcal Meningitis in a 12-month-old Boy after the First Dose of Pneumococcal Conjugate Vaccine
Author
Yuji Ito Shinya Hara Naomi Otowa Takako Suzuki Kenji Aizu Hikaru Yamamoto Hajime Ushida Shinji Kido and Naoya Okumura
Department of Pediatrics, TOYOTA Memorial Hospital
Abstract
The patient received his first dose of pneumococcal conjugate vaccine at 9 months of age. His second dose was scheduled for 10 months of age, but was postponed because he had caught a cold. At 12 months of age, the patient suffered pneumococcal meningitis. He was started on antibiotics, and his clinical course was uneventful except that impaired hearing was detected later in the course of his illness. Blood and cerebrospinal fluid cultures were positive for serotype 6B pneumococcus. The serotype-specific anti-6B IgG antibody concentrations in the blood samples collected on admission and at the time of discharge were high enough to prevent invasive pneumococcal disease, but opsonophagocytic activity against serotype 6B was low. We hypothesized that the low level of opsonophagocytic activity contributed to the development of the meningitis. The opsonophagocytic activity remained low after the patient received his second and third dose of pneumococcal conjugate vaccine. We discovered that there is a certain population of patients who have low levels of opsonophagocytic activity after receiving routine pediatric vaccinations.
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Original Article
Title
A Case of Transient Neonatal Hyperparathyroidism Secondary to Untreated Maternal Hypoparathyroidism
Author
Kei Takasawa1)2) Makoto Ono1) Kikuko Oku3) and Shuki Mizutani1)
1)Department of Pediatrics and Developmental Biology, Tokyo Medical and Dental University
2)Department of Pediatrics, Kawaguchi Municipal Medical Center
3)Department of Neonatal Intensive Care Unit, Kawaguchi Municipal Medical Center
Abstract
Neonatal hyperparathyroidism secondary to maternal hypocalcaemia is a rare disorder caused by intrauterine hypocalcemia and shows benign and transient clinical and radiological skeletal features with bone demineralization. Here, we report the case of a female neonate with severe bone demineralization whose mother was suffering from untreated hypoparathyroidism.
The patient was born after 38 weeks and 6 days of gestation by an uncomplicated caesarean section to a 33-year-old, gravida 2, para 2 mother, with a birth weight of 1,692 g, body length of 47 cm and head circumference of 29 cm. Her Apgar scores were 9 and 10 at 1 and 5 minutes, respectively. When she was referred to us at the age of 11 days, her whole-body X-ray showed chest wall deformity, marked bone demineralization and elevated serum calcium and parathyroid hormone (PTH) values. A few days later, the concentration of serum PTH and balance of Ca and P normalized and bone features improved spontaneously. Chemical pathology investigation and review of the mother's medical history indicated hypoparathyroidism. The mother was diagnosed with hypoparathyroidism at the age of 10 years and was treated for 2 years, but devided to discontinue treatment.
Typically, in infants with hyperparathyroidism secondary to maternal hypocalcemia, hypercalcemia is less well defined. Elevated serum values of PTH are normalized in 1-3 weeks and bone abnormalities are improved in 4-16 months (median 8.1 months).
It is very important that maternal hypoparathyroidism or hypocalcaemia during pregnancy is treated appropriately to prevent neonatal hyperparathyroidism. However, we must note that, as in the present case, maternal hypoparathyroidism may or may not be identified during pregnancy.
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Original Article
Title
Neonatal Onset Carbamoylphosphate Synthetase 1 Deficiency Treated with Living-related Liver Transplantation at Five Months after Birth
Author
Tomotaka Kono1) Katsuya Aizu1) Kenji Shimizu2) Hirofumi Ohashi2) Shuichiro Fujinaga3) Koichi Mizuta4) and Hiroshi Mochizuki1)
1)Division of Endocrinology and Metabolism, Saitama Children's Medical Center
2)Division of Genetics, Saitama Children's Medical Center
3)Division of Nephrology, Saitama Children's Medical Center
4)Department of Transplant Surgery, Jichi Medical University
Abstract
We report a Japanese girl with early neonatal onset carbamoylphosphate synthetase 1 (CPS1) deficiency. Extreme hyperammonemia was observed at two days after birth. Continuous hemodialysis was immediately introduced. As a result, plasma ammonia concentration was normalized within two days. Afterwards, plasma ammonia level was controlled well by medication and protein restriction. The living-related liver transplantation from her father was performed successfully at five months of age. Although her post-transplant developmental quotient (DQ) level was not sufficient, there were marked improvements in her quality of life after the transplantation. Neonatal onset CPS1 deficiency holds a high risk of severe neurological sequelae due to recurrence of acute elevation of plasma ammonia concentration. Therefore, early liver transplantation is needed for improvement of outcome.
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Original Article
Title
BCG Osteomyelitis of Radius in a Suspected Bone Tumor Case
Author
Naoki Hashimoto Katsumi Nishiya Taku Ishihara and Midori Shima
Nara Medical University
Abstract
A 1-year-old boy was admitted to a local hospital with a fracture of the left radius. He was transferred to our hospital for the evaluation of a suspected bone tumor due to an abnormal shadow on the X-ray of his left radius. He had received bacillus Calmette-Guérin (BCG) vaccination at the age of 4 months and had had no problem related to the vaccination Magnetic resonance imaging showed malformation of the bone and inflammation within the medullary cavity at the proximal lesion of the left radius. Detailed examinations indicated suspected osteomyelitis. Surgical drainage and biopsy were performed. The discharge was found to be positive for the Mycobacterium tuberculosis complex by polymerase chain reaction. Hence, antituberculosis therapy was started. Cultured bacteria from the discharge were identified as M. bovis by restriction fragment length polymorphism. BCG osteomyelitis is a rare disease, and only 5-10 cases of this disease are reported every year. In Japan, the incidence of this disease has been reported to have increased since 2005, when the vaccination time was changed to below 6 months of age. BCG osteomyelitis should be considered as a possible complication of BCG vaccination. Our patient had no abnormality related to interferon-gamma production, but clinicians should carefully evaluate the presence of congenital immunodeficiency in patients with severe BCG complications.
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