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THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY
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Vol.119, No.1, January 2015
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Original Article
Title
Analysis of Invasive Group B Streptococcal Disease in Children According to Onset of Disease
Author
Saori Deki1) Kensuke Shoji2) Takanori Funaki2) Hirokazu Sakai1) and Isao Miyairi2)
1)Department of General Pediatrics and Interdisciplinary Medicine, National Center for Child Health and Development
2)Division of Infectious Diseases, National Center for Child Health and Development
Abstract
Invasive Group B Streptococcus (GBS) infection is classified into early (day 0-6), late (7-89), and ultra-late onset (later than 3 months of age) disease according to the date of onset. There are few reports on ultra-late onset infection. We performed a retrospective review of patients who had GBS isolated from their blood or cerebrospinal fluid by culture at our institution from April, 2002 to March, 2013. Patients were categorized and characterized according to the date of onset into early, late, and ultra-late onset, with an emphasis on the ultra-late onset patients. There were 28 cases of invasive GBS infections with 9 early onset cases (32%), 11 late onset cases (39%), and with 8 ultra-late onset cases (29%). As previously reported, early onset patients tended to present with pneumonia (44%), bacteremia without an apparent focus (44%) and a few with meningitis (12%), whereas more late onset presented with meningitis (36%). The majority of ultra-late onset patients had an underlying disease and presented with bacteremia without an apparent focus (63%), followed by meningitis (25%), and cellulitis (13%). Interestingly, hypogammaglobulinemia was identified in 5 of the 7 ultra-late onset cases. The clinical manifestation of invasive GBS disease varied according to the timing of onset. Patients presenting with ultra-late onset infection tended to have underlying disease and bacteremia without a focus.
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Case Report
Title
A Boy and His Mother with Beta-thalassemia Minor Diagnosed with Iron-deficiency Anemia
Author
Junpei Hasumi and Hideo Ushiku
Department of Pediatrics, Saku Central Hospital
Abstract
A 15-year-old boy who consulted our department after a school physical health examination revealed a low mean corpuscular volume (MCV) of 60.2 fL. The boy had been examined at 10 years of age for anemia and after being diagnosed with iron-deficiency anemia, he was givenby iron supplements; however, he experienced little improvement. On examining his mother's medical records, we found that she also had microcytic anemia with a MCV of 60.9 fL. A thorough work-up was performed on suspicion of an abnormal hemoglobin gene, which revealed an abnormality in the gene that encodes beta-globin chains (codons 41/42 TTCTTT→ TT) in both the boy and his mother. This led to a diagnosis of beta-thalassemia minor. There are three types of beta-thalassemia, including minor, intermedia, and major. The minor type has relatively mild symptoms, whereas the major type requires regular blood transfusion and administration of iron-chelating agents. Patients requiring radical treatment have a severe disease that greatly affects daily life, such as diseases requiring bone marrow transplantation. Some minor cases, such as the present patient, are diagnosed with iron-deficiency anemia. However, if two individuals with the minor type marry, there is a chance that their child will be born with the major type. It is therefore important that patients fully understand their own disease through correct diagnosis and physicians' explanations.
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Case Report
Title
A Pediatric Case of Severe Heat Stroke Complicated by Rhabdomyolysis Requiring Long-term Hemodialysis
Author
Junji Shimizu Nao Nishimura and Takashi Muguruma
Pediatric Intensive Care Unit, National Center for Child Health and Development
Abstract
A 14-year-old boy lost consciousness and developed generalized tonic seizures during an outdoor soccer practice in late August, and was transported to the emergency department. On admission, the boy presented with evident shock and impaired consciousness, and an axillary temperature of 42°C. An initial diagnosis of severe heat stroke was established, and external cooling and fluid loading was administered. Continuous hemodialysis was initiated as renal replacement therapy for acute renal failure and also as internal cooling. He developed rhabdomyolysis on day 2; therefore, continuous hemodiafiltration (CHDF) was initiated. Long-term CHDF was continued due to persistent oliguria. CHDF was changed to intermittent hemodialysis from day 36 and was discontinued on day 38. He was discharged from the hospital on day 53 without any neurological sequelae.
We report a pediatric case of severe heat stroke resulting in central nervous system impairment, acute renal failure, and rhabdomyolysis. Intensive care, including systemic management such as respiratory and circulatory support is important for the treatment of severe heat stroke. Prompt transport to medical facilities capable of providing pediatric critical care should be considered at an early stage. Moreover, due to the paucity of heat stroke in the pediatric population, centralization of severe patients should be considered.
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Case Report
Title
Pleural Tuberculoma Developing during Treatment for Tuberculous Pleural Effusion
Author
Yoji Uejima1) Risa Tanaka1) Keiji Akamine1) Tadamasa Takano1) Toshiaki Saeki1) Tomoo Miyakawa2) Tsutomu Oh-ishi1) and Yutaka Kawano1)
1)Division of Infectious Diseases, Immunology, and Allergy, Saitama Children's Medical Center
2)Department of Respiratory Medicine, Tokyo Metropolitan Children's Medical Center
Abstract
Tuberculous pleurisy is an uncommon disease in children. A previously healthy, 12-year-old girl was referred to our hospital with fever and respiratory discomfort. CT scan showed a large left pleural effusion. The pleural effusion contained predominantly lymphocytes, and the pleural effusion ADA levels were elevated. The blood QuantiFERON-TB Gold in-Tube (QFT-3G) test was equivocal on admission, but the QFT-3G and the T-SPOT. TB (T-SPOT) obtained on the 17th day of hospitalization were positive. Tubercle bacilli were cultivated from pleural fluid obtained 2 weeks after starting antituberculous medication. Although the tuberculous pleural effusion responded to medication with resolution of the pleural fluid, new nodular opacities developed in the lung 4 months after starting antituberculous chemotherapy, but they finally decreased with continuation of the initial treatment. The new pulmonary lesions were diagnosed as pleural tuberculomas.
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Case Report
Title
Successful Management of Refractory Asthma with Allergic Laryngitis and Diffuse Aspiration Bronchiolitis Using Omalizumab
Author
Tatsuki Fukuie Toshiki Nakanishi Ryuhei Yasuoka and Tomohide Taguchi
Department of Pediatrics, Hamamatsu University School of Medicine
Abstract
Diffuse aspiration bronchiolitis (DAB) is a type of aspiration pneumonia that is characterized by chronic inflammation of bronchioles caused by recurrent aspiration, generally in the elderly, but it can also occur in childhood. We herein report an 11-year-old boy with a chief complaint of refractory asthma, with complications of obesity, obstructive sleep apnea syndrome and gastroesophageal reflux disease. DABi was diagnosed with DAB based on chest computed tomography and esophagograph which showed chronic occult aspiration. He received comprehensive therapies including inhaled corticosteroids, montelukast, proton pump inhibitors, respiratory and swallowing rehabilitation and exercise and nutrition therapy. However, his symptoms showed no clear improvement. Tube feeding was therefore recommended, but he and his family refused. Therefore, we began anti-immunoglobulin E (omalizumab) therapy and his symptoms and respiratory function tests showed dramatic amelioration. We confirmed resolution of the silent aspiration during re-esophagography, and thus concluded that allergic laryngitis had co-existed with DAB and that omalizumab had been effective for this allergic condition.
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Case Report
Title
Two Patients with Secondary Biotin Deficiency Caused by the Use of Ketogenic Formula in Japan
Author
Fumihito Nozaki Tomohiro Kumada Takashi Kusunoki Anri Hayashi Ikuko Hiejima Kanako Maizuru Atsushi Yokoyama Minoru Shibata and Tatsuya Fujii
Department of Pediatrics, Shiga Medical Center for Children
Abstract
Ketogenic diets (KDs) are effective for the treatment of medication-resistant epilepsy and can be used in the early course of treatment for epilepsy. Keton Formula® (817-B; Meiji, Tokyo, Japan) is a ketogenic formula available in Japan. Although the World Health Organization recommends including biotin at 1.5 μg/100 kcal in formula, 817-B contains only 0.1 μg/100 kcal of biotin. We encountered two patients with secondary biotin deficiency caused by long-term use of 817-B. Patient 1 was a severely disabled 5-year-old boy with cerebral palsy and West syndrome due to severe birth asphyxia. Patient 2 was a severely disabled 4-year-old girl with Gaucher disease type 2. Both patients had feeding difficulty and medication-resistant epilepsy. Use of 817-B had been continued for 2 years and 5 months in Patient 1 and for 1 year and 4 months in Patient 2, until alopecia had developed in both cases. Patient 1 showed temporal alopecia, while Patient 2 had frontal alopecia. Low urinary levels of biotin and elevated urinary levels of 3-hydroxyisovaleric acid were found in both patients, indicating secondary biotin deficiency. Alopecia improved within 3 months after starting daily supplementation of biotin (1 mg/day) in both patients. Laboratory data also improved. Long-term use of 817-B can cause secondary biotin deficiency because of insufficient levels of biotin in the formula. Biotin supplementation is warranted in patients on a ketogenic diet using 817-B, which should be fortified with biotin in the near future.
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