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THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY
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Vol.120, No.1, January 2016
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Original Article
Title
One-dose Varicella Vaccine Effectiveness in an Elementary School Population and Breakthrough Varicella
Author
Masahiro Watanabe
Suzuka Pediatrics
Abstract
We conducted a retrospective cohort study to clarify the details of a varicella outbreak that occurred between December 2013 and January 2014 in an elementary school (number of students: 215) and determine varicella vaccine effectiveness. Immediately before the outbreak, 113 (52.6%), 82 (38.1%), and 20 (9.3%) students were identified as having a history of varicella infection, having a history of one dose varicella vaccination, and those who were susceptible to varicella, respectively. In the first wave of the epidemic, 5 students were infected through unknown routes of transmission. The varicella outbreak ended after a second and third wave that affected 7 and 2 students, respectively. Since thirteen out of 14 students who were infected with varicella were diagnosed with breakthrough varicella (BV), vaccine effectiveness could not be evaluated due to the low attack rate among unvaccinated students. Vaccine efficacy before the outbreak was low, at 48.5% (95% CI: 33.8-59.9), and varicella outbreaks occurred at a median of 3.4 years (95% CI: 2.9-4.7) after vaccination. These results suggest that a single dose of vaccination is not sufficient to decrease the occurrence rate of BV or reduce the risk of infection. In Japan, the administration of 2 doses of varicella vaccine was initiated for children under 3 years old in October 2014, and so the number of varicella cases is expected to decrease; however, the lack of booster vaccination means that there is still a risk of varicella outbreaks in elementary schools. The establishment of diagnostic criteria for BV and a survey of trends in varicella outbreaks are needed.
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Case Report
Title
Permanent Remission of Severe Cholestasis and Thrombocytopenia Associated with Acquired Cytomegalovirus Infection in Response to Glucocorticoid but not Ant-viral Therapy in a Preterm Infant
Author
Yasuto Maeda1) Junichiro Okada1) Hiroki Saitsu1) Yuhei Tanaka2) Tadahiro Yanagi2) Kenji Masunaga3) Osuke Iwata2) Hiroyuki Moriuchi4) and Tadashi Hisano1)
1)Division of Neonatology, St. Mary's Hospital
2)Department of Pediatrics and Child Health, Kurume University School of Medicine
3)Division of Infectious Disease, Department of Infectious Medicine, Kurume University School of Medicine
4)Department of Pediatrics, Nagasaki University School of Medicine
Abstract
We here report a preterm infant with acquired cytomegalovirus infection, whose prolonged thrombocytopenia and cholestasis showed a marked response to anti-cytokine therapies. A male neonate was born at a gestational age of 28 weeks (birth weight, 990 g). He developed septic shock (Klebsiella pneumoniae) on day 19, which was successfully treated by intravenous injections of antibiotics and hydrocortisone. After the remission of septicemia, he developed severe, refractory thrombocytopenia and cholestasis. On day 30, elevated copies of cytomegalovirus DNA (8.0×104 copies/mL) confirmed the diagnosis of active, acquired cytomegalovirus infection of the neonate. Administration of antiviral therapy significantly reduced the viral copy number to 4.0×102 copies/mL, however, clinical symptoms deteriorated, requiring daily platelet transfusion. Based on elevated cytokines, such as IL-6 and IL-10, supplementation of betamethasone and immunoglobulin was commenced, leading to dramatic and permanent remission of thrombocytopenia and cholestasis. In children and adults, an excessive immune response has been associated with the pathogenesis of cytomegalovirus disease, however, little evidence is available in younger patients. Our experience supports the concept that immuno-pathological mechanisms play an important role in acquired cytomegalovirus infections in preterm infants. Further studies are needed to confirm the potential benefit of immunosuppressive therapies for acquired cytomegalovirus infection in addition to antiviral therapies.
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Case Report
Title
A Fetus with Congenital Complete Atrioventricular Block and Atrial Flutter Associated with Maternal Sjögren Syndrome
Author
Yuka Tanabe1) Masaki Nii2) Jun Yoshimoto2) Tao Fujioka2) Kumiyo Matsuo2) Mayumi Nagasawa3) Norie Mitsushita2) Sung-Hae Kim2) Yasuhiko Tanaka3) and Yasuo Ono2)
1)Department of Pediatrics, Shimane University Faculty of Medicine
2)Department of Cardiology, Shizuoka Children's Hospital
3)Department of Neonatology, Shizuoka Children's Hospital
Abstract
The anti SS-A/SS-B antibody, known to be trans-placentally transmitted from a mother with Sjögren syndrome or systemic lupus erythematosus to the fetus, causes congenital atrioventricular block (AVB) in approximately 0.2-1.0% of fetuses exposed to these antibodies. It is reported that about 1.0-2.5% women of childbearing age are positive for anti SS-A/SS-B antibody even though they lack any symptoms. Although the precise mechanism causing AVB by the anti SS-A/SS-B antibody remains to be determined, anti SS-A antibody to 52-kDa epitope is reported to further increase the risk of AVB.
We report a fetus with congenital complete AVB and atrial flutter associated with maternal Sjögren syndrome. The complete AVB was diagnosed at 26 weeks of gestation, and the association of atrial flutter was noticed at 28 weeks. The mother was positive for anti SS-A/SS-B antibodies with high titers. Because the association of atrial flutter with congenital AVB is very rare, inflammation of the atrial myocardium was suspected and the dexamethasone was started from 28 weeks. The baby was born at 36 weeks by cesarean section and atrial flutter was electively stopped by direct current cardioversion. Although his heart rate was approximately 90 bpm at discharge, the heart rate gradually decreased to around 60 bpm and a permanent pacemaker (DDD) was implanted at 3 months of age. Because of recurrence of atrial flutter at 6 months of age, sotalol was started and the pacing mode was changed to VVI. The association of atrial flutter with congenital AVB is extremely rare and only one case has been reported in the literature.
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Case Report
Title
Airway Obstructing Infantile Subglottic Hemangioma Treated with Propranolol
Author
Satoshi Miyagaki1) Tomoko Iehara1) Mitsuru Miyachi1) Yasumichi Kuwahara1) Makoto Kita2) and Hajime Hosoi1)
1)Department of Pediatrics, Graduate School of Medical Science, Kyoto Prefectural University of Medicine
2)Department of Pediatrics, National Hospital Organization Kyoto Medical Center
Abstract
Infantile subglottic hemangioma (ISH) is rare, but proper treatment is necessary due to the risk of airway obstruction. Recently, propranolol has been recommended as a first-line therapy for infantile hemangioma (IH). We herein report a case of airway-obstructing ISH successfully treated by propranolol. A four-month-old boy demonstrated recurrent upper airway obstruction for a month. We diagnosed pseudocroup and treated with oral dexamethasone intermittently, however, he ultimately required tracheal intubation due to severe lack of oxygenation. The patient was diagnosed with ISH based on the laryngoscopy and magnetic resonance imaging (MRI) findings and started propranolol (2 mg/kg/day). According to previous reports, ISH was shown to diminish and the airway obstruction to improve as early as a week after initiating propranolol therapy, thus, we decided to extubate at 6 days after starting propranolol. Laryngoscopy indicated that his tumor was diminishing. Oral dexamethasone was then tapered and halted during his hospitalization. Following discharge at 22 days from the initiation of the propranolol therapy, he has finished propranolol at the age of 18 months, and his subglottic lesion does not show regrowth. Propranolol may sufficiently reduce IH and has few side effects compared with other therapeutic strategies, such as oral steroids, vincristine, laser treatment, tracheostomy, and open surgery. However, the optimal treatment duration of propranolol is unknown. In reference to the previous observation that IH rapidly grows during the first year of life, sometimes until the age of 18 months, we believe propranolol therapy should be continued beyond the age of 18 months.
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Case Report
Title
A Case of Multiple Brain Abscesses Indistinguishable from Brain Tumors on Imaging Studies
Author
Soichiro Kawata1) Nobuhiro Ito1) Tasuku Kitajima1) Yasutomo Funakoshi1) Masahiko Okada1) Reiko Ideguchi2) and Hiroyuki Moriuchi1)
1)Department of Pediatrics, Nagasaki University Hospital
2)Department of Radioisotope Medicine, Atomic Bomb Disease Institute, Nagasaki University
Abstract
Typically, brain abscesses show ring enhancement on post-contrast T1-weighted images of magnetic resonance imaging (MRI) and hyperintensity on diffusion-weighted imaging (DWI). Infrequently, however, these lesions may present with hypointensity on DWI, making the diagnosis quite challenging. We experienced a pediatric case of multiple brain abscesses with hypointensity on DWI.
Case: A previously healthy 5-year-old boy with a past medical history of febrile convulsions six weeks prior to admission presented with a two-day history of vomiting and gradually deteriorating consciousness. Computed tomography detected large low-density areas in the left cerebrum, strongly suggestive of a brain tumor. The patient had no obvious febrile episodes, and the C-reactive protein level was minimally elevated. Contrast-enhanced MRI revealed multiple lesions, some of which had ring enhancement. DWI identified these masses as hypointense lesions. Under the tentative diagnosis of a brain tumor, the initiation of radiation therapy was considered; however, a craniotomy biopsy revealed the lesions to be brain abscesses. Cultures of the abscess aspirates and blood yielded sterile findings, and the source and route of infection remained undetermined despite extensive investigations. He was empirically treated with meropenem for six weeks and discharged with a sequela of minimal verbal disability.
Our experience illustrates two pitfalls that we should be avoided in the diagnosis of brain abscesses: (1) such lesions may be quite indistinguishable from brain tumors on imaging studies and (2) they can develop in children with no apparent risk factors.
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Case Report
Title
Familial Cases of Vivax Malaria with Severe Thrombocytopenia
Author
Yoshiki Kusama1)2) Ayako Ozawa1) Taichi Wakabayashi1) Naoe Akiyama1) Takaaki Segawa1) Hiroyuki Ida2) and Yasutaka Mizuno3)
1)Department of Pediatrics, Fuji City General Hospital
2)Depatment of Pediatrics, The Jikei University School of Medicine
3)Department of Infectious Diseases, Tokyo Medical University Hospital
Abstract
We report three familial cases of vivax malaria, including two pediatric cases. Although pediatric malaria cases are rare in Japan, with only one or two cases seen annually, early diagnosis and treatment of malaria are very important, because pediatric malaria sometimes causes severe and life-threatening conditions. As international interaction increases and diversifies, imported infectious diseases will become more common in Japan. Therefore, when we encounter a patient who has returned from a tropical country, we should recognize the importance of considering a diagnosis of malaria. Although our cases were diagnosed as vivax malaria, all patients had severe thrombocytopenia, as though they were suffering from falciparum malaria. Recently, many researchers have shown that vivax malaria causes more severe conditions than historically documented, and some studies have reported cases of fatality. Thus, we should not ignore the possibility that vivax malaria causes severe conditions, although it is suggested to be benign.
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Case Report
Title
Juvenile Dermatomyositis Secondary to Reactive Arthritis in a 4-year-old Boy
Author
Masaaki Hamada Naoki Hashimoto Yoshiko Uchida Kazufumi Izaki Yae Michinomae Taku Ueda and Ichiro Tanaka
Department of Pediatrics, Yao Municipal Hospital
Abstract
A 4-year-old boy complained of distal extremity pain for one month following a Salmonella infection. He subsequently developed progressive swelling and flexion difficulty in his finger joints and reactive arthritis was diagnosed. However, he thereafter had a facial heliotrope rash and erythema, several pruritic papules on the upper extremities, and proximal muscle weakness. Juvenile dermatomyositis (JDM) was diagnosed secondary to reactive arthritis and successfully treated with 2 cycles of methylprednisolone pulse therapy. To our knowledge, there have been no reports of JDM secondary to reactive arthritis associated with Salmonella infection. JDM is suspected to be associated with a variety of antecedent infections, this case may contribute to better understanding of the pathogenesis of JDM.
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Case Report
Title
Three Pediatric Cases of Cervical Cord Injury Caused by Unused Rear Seat Belts or an Unfastened Shoulder Belt of a Child Safety Seat
Author
Tomoko Ohhira Takahumi Obara Yuuichirou Muto Katsuki Hirai Takahiro Nishihara Nagisa Komatsu and Masahiro Migita
Japanese Red Cross Kumamoto Hospital
Abstract
Pediatric cervical cord injury is extremely rare. In many patients with cervical cord injury, managing the airway as well as respiratory and circulatory dynamics is difficult and they often cause severe after-effects. We encountered 3 cases of pediatric cervical cord injury caused by seat belt-related car accidents (unused rear seat belts in 2 cases and an unfastened shoulder belt of a child safety seat in 1 case). One patient died, and the other two patients (one had persisting paresis of the right arm and the other had complete paralysis of the legs) were taken off the ventilator. In conclusion, the appropriate use of seat belts, including those of child safety seats, is crucial.
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Case Report
Title
A Long-term Esophageal Foreign Body Related to Congenital Esophageal Diverticulum Caused by Respiratory Failure
Author
Ryo Imakiire1) Naohiro Ikeda1) Kanna Nakano1) Yumiko Ninomiya1) Shinsuke Maruyama1) Takayuki Tanabe1) Sho Hashiguchi2) Yuichiro Nuruki2) Toshiro Fukushige2) and Yoshifumi Kawano1)
1)Department of Pediatrics, Kagoshima University Hospital
2)Department of Pediatrics, Kagoshima Prefectural Hokusatsu Hospital
Abstract
A 1-year-old girl had complained of episodes of stridor soon after meals several months before admission. After she showed severe dyspnea, she was intubated, and an upper-mediastinum tumor, which was comprising the esophagus and trachea, was found by CT. Endoscopy showed a diverticulum of the esophagus, as well as a foreign body hanging by its edge on the diverticulum. This foreign body was a 3 cm long umbrella-shaped plastic object that was found to be part of a toy. After removing the foreign body, esophageal endoscopy showed no findings of stenosis, dilation, or mucosal defects. Since this removal, she has not showed any episodes of stridor after meals. CT at 1 month after removal of the foreign body showed the presence of esophageal diverticulum and deviation of the esophagus and trachea was normalized. The present case was diagnosed as having congenital esophageal diverticulum because the location of the diverticulum was not the anatomical narrow segment of the esophagus. The foreign body enlarged the diverticulum and then pressed on the trachea. We consider that this situation was enhanced by swelling from meals and caused her dyspnea. Because a chronic foreign body in the esophagus usually causes respiratory symptoms, the possibility of a foreign body should be considered in patients with refractory or recurrent respiratory symptoms. In conclusion, esophageal diverticulum might be related to long-term retention of a foreign body in the esophagus. Esophageal findings, including the diverticulum, should be observed in patients with a chronic foreign body in the esophagus.
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