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A Case Report: Ganciclovir Therapy of Cytomegalovirus Pneumonitis.

Jee Yeon MIN ; Soo Jong HONG ; Hyung Nam MOON ; Chang Yee HONG

Journal of the Korean Pediatric Society.1996;39(1):142-.

Ganciclovir is an antiviral agent that is effective for cytomegalovirus (CMV) infection in immunocompromised hosts. But the benefits of treatment for the congenital CMV diseases are still controversial. Cytomegalovirus pneumonitis is very rare. And so, a few cases of ganciclovir therapy in CMV pneumonitis were reported. We experienced a case of a 7 month-old male infant with prolonged pneumonitis and respiratory difficulty which were not improved with steroid and antibiotic therapy for 2 months. He was born at gestational age of 32 weeks and received oxygen therapy with mask for 2 days only. On physical examination, tachypnea, chest retracion, inspiratory wheezing and rales were present. PaCO2 was 84.0 mmHg on blood gas analysis and the anti-CMV-IgM, -IgG antibodies were positive. But anti-CMV-IgM, -IgG antibodies of his mother were negative. The open lung biopsy revealed CMV inclusion in alveolar interstitium. CMV shell vial assay of patient's urine and tracheal aspirate were positive. Ganciclovir (5-10 mg/kg, 10weeks) had been given without any significant complications. After treatment of ganciclovir for 4 weeks, shell vial assay of tracheal aspirate for CMV showed negative conversion and anti-CMV-IgM antibody was converted negatively. CMV pneumonitis of this patient had the possibility of secondary infection associated with immunosuppressive status after long-term steroid therapy or with blood transfusion. We report a first case of ganciclovir therapy of CMV pneumonitis with review of related literatures.
Antibodies ; Biopsy ; Blood Gas Analysis ; Blood Transfusion ; Coinfection ; Cytomegalovirus* ; Ganciclovir* ; Gestational Age ; Humans ; Immunocompromised Host ; Infant ; Lung ; Male ; Masks ; Mothers ; Oxygen ; Physical Examination ; Pneumonia* ; Respiratory Sounds ; Tachypnea ; Thorax

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A Case of Idiopathic Pulmonary Hemosiderosis.

Chang Sung LIM ; Seung Kyu PARK ; Won PARK ; Soon Jeong LEE ; Chul Zoo JUNG

Journal of the Korean Pediatric Society.1996;39(1):136-141.

Idiopathic pulmonary hemosiderosis is characterized by cough, hemoptysis, dyspnea, diffuse pulmonary infiltrates, and microcytic and hypochromic anemia. The cause of this illness is unknown. We experienced a case of idiopathic pulmonary hemosiderosis in a 2 year and 8 month-old boy. Hemosiderin-laden macrophages are demonstrated in smears of material obtained from tracheal aspirates. There were no specific causes for pulmonary hemorrhage. We report a case of idiopathic pulmonary hemosiderosis with brief review of related literatures.
Anemia, Hypochromic ; Cough ; Dyspnea ; Hemoptysis ; Hemorrhage ; Hemosiderosis* ; Humans ; Infant ; Macrophages ; Male

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A Case of Idiopathic Crescentic Glomerulonephritis.

Jung Keum PARK ; Sang Il GOO ; Woo Yeong CHUNG ; Chul Ho KIM

Journal of the Korean Pediatric Society.1996;39(1):131-135.

We experienced a case of idiopathic crescentic glomerulonephritis in a 10-year-old girl who was admitted to our hospital due to gross hematuria and oliguria for 2 months. The diagnosis was based on the rapidly progressive clinical course to chronic renal failure, positive p-ANCA test and light microscopic findings of diffuse crescents formation in about 80% glomeruli of renal tissue obtained by percutaneous renal biopsy. Our case was treated with high dose methylprednisolone ( 30mg/Kg) pulse therapy whenever its clinical course was aggravated for several times. After 2 years 6months later, she has been treated on peritoneal dialysis. A review of literatures was also presented briefly.
Antibodies, Antineutrophil Cytoplasmic ; Biopsy ; Child ; Diagnosis ; Female ; Glomerulonephritis* ; Hematuria ; Humans ; Kidney Failure, Chronic ; Methylprednisolone ; Oliguria ; Peritoneal Dialysis

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A Case of Neonatal Cholestasis with Arthrogryposis Multiplex Congenita and Renal Tubular Insufficiency(ARC Syndrome).

Hi Soo RHEE ; Soon Young KIM ; Nam Sun BAIK ; Il Soo HA ; Jeong Kee SEO

Journal of the Korean Pediatric Society.1996;39(1):126-130.

We report a case of ARC syndrome with arthrogryposis multiplex congenita, renal tubular insufficiency and cholestasis. The Patient presented in the early neonatal period with micrognathia, low set ears, high arched palate, multiple joint contracture, conjugated hyperbilirubinemia and failure to thrive. He died at the age of 1 month despite medical therapy. Findings of renal tubular insufficiency included persistent renal tubular acidosis, glucosuria, aminoaciduria, and proteinuria. Liver biopsy revealed intracellular and canalicular cholestasis, ballooning degeneration and giant cell formation of hepatocyte. Kidney sonography revealed medullary nephrocalcinosis. This association was first reported in 1973 by Lutz-Richner and Landolt and again in another family by Nezelof et al in 1979. Until now, 13 cases were reported worldwide. Except one case, all children died in infancy. Autosomal recessive inheritance is the most likely mode of transmission. We have experienced a case of ARC syndrome in a male neonate with signs and symptoms of lethargy, poor oral intake, direct hyperbilirubinemia, acidosis, and multiple joint contracture.
Acidosis ; Acidosis, Renal Tubular ; Arthrogryposis* ; Biopsy ; Child ; Cholestasis* ; Contracture ; Ear ; Failure to Thrive ; Giant Cells ; Hepatocytes ; Humans ; Hyperbilirubinemia ; Infant, Newborn ; Joints ; Kidney ; Lethargy ; Liver ; Male ; Nephrocalcinosis ; Palate ; Proteinuria ; Wills

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A Case of Eosinophilic Fasciitis.

Dong Hee LEE ; Jong Hyun KIM ; Won Bae LEE ; Jun Sung LEE

Journal of the Korean Pediatric Society.1996;39(1):120-125.

Eosinophilic faciitis is a recently recognized entity causing inflammation, thickening and fibrosis of the fascia, which is associated with hypergammaglobulinemia, peripheral eosinolhila. This case of 11 year-old male patient who complaint migrating arthralgia and flexion contration with nodule on left upper extremity for 6 month and suddenly developed proptosis had hypergammaglobulinemia and elevated ESR. Biopsy of the nodule demonstrated inflammatory infiltration of eosinophil in fascia and subcutis, which was consisted with eoainophilic fasciitis. We report a case of eosinophilic fasciitis with brief review who was treated with corticosteroid and had symptomatic improvement.
Arthralgia ; Biopsy ; Child ; Eosinophils* ; Exophthalmos ; Fascia ; Fasciitis* ; Fibrosis ; Humans ; Hypergammaglobulinemia ; Inflammation ; Male ; Upper Extremity

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A Case of Cerebral Infarction Associated with Mycoplasma pneumoniae Infection.

Young Jae KOH ; Dong Jun KIM ; In Joon SEOL ; Ha Baik LEE ; Ki Woong HONG

Journal of the Korean Pediatric Society.1996;39(1):115-119.

Mycoplasma pneumoniae has been shown to be of etiologic importance in cases of upper-and lower-respiratory tract infections, especially in children and young adults. It may cause a variety of extrapulmonary manifestations in multiple organ systems, most commonly the central nervous system. The extrapulmonary syndromes include meningitis, cerebral infarction, acute transverse myelitis, psychosis, cerebellar ataxia, Guillain-Barr syndrome and Reye syndrome. Cerebral infarction as a complication of mycoplasma infection in children has been rarely reported. We report the first documented case in Korea of cerebral infarction preceded by M. pneumoniae pneumonia in a 7-year-old boy, with a brief review of literatures.
Central Nervous System ; Cerebellar Ataxia ; Cerebral Infarction* ; Child ; Humans ; Korea ; Male ; Meningitis ; Mycoplasma Infections ; Mycoplasma pneumoniae* ; Mycoplasma* ; Myelitis, Transverse ; Pneumonia ; Pneumonia, Mycoplasma* ; Psychotic Disorders ; Reye Syndrome ; Young Adult

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Study on the Thyroid Function of Neonate Born to Mother with Hyperthyroidism.

Choong Ho SHIN ; Se Young KIM ; Sei Won YANG ; Jung Hwan CHOI ; Chong Ku YUN

Journal of the Korean Pediatric Society.1996;39(1):106-114.

PURPOSE: In newborns from mothers with Graves' disease, neonatal transient hyperthyroidism or hypothyroidism may develop early in life. We evaluated the incidence and prognosis of neonatal thyroid dysfunction in neonates born to mothers with hyperthyroidism during pregnancy. METHODS: We measured blood T4, T3, TSH levels and TSH-R-Ab titer in 48 hyperthyroid mothers and their babies between 1988 and 1994. RESULTS: The mean birth weight was 3.21+/-0.49kg and gestational age was 39+5 weeks and three neonates(6%) were small for gestational age and two(4%) were premature whose mother had hyperthyroidism during pregnancy. Eight of the neonates showed high levels of thyroid hormone and four showed low levels of thyroid hormone but none had symptom or sign. The neonatal thyroid function was not affected by maternal antithyroid medication and maternal TSH-R-Ab level. The positive rate of TSH-R-Ab was 44 percent in neonates. In the neonates with low T4 level, the mean TSH-R-Ab was 38.1+/-19.6%, which was higher than those in euthyroid(14.7+/-9.7%) or hyperthyroid(8.4+/-6.2%) neonates(p<0.05). The neonatal blood TSH-R-Ab levels was correlated with maternal TSH-R-Ab levels(r=0.50, p<0.05). Neonatal T4 didn't show any correlation with maternal TSH-R-Ab but showed negative correlation with neonatal TSH-R-Ab(r=-0.43, p<0.05). Neonates with high T4 showed normal thyroid function within 35 days(10-54days) in average. Neonates with low T4 showed normal thyroid function within 29 days(7-61days) in average. One of neonate with low T4 was given thyroid hormone for 1 month. No one with initial abnoraml thyroid function showed clinical problem through follow-up. CONCLUSIONS: It is likely that hyperthyroid mothers have more tendency of small babies for gestational age or premature babies. Neonates born to hyperthyroid mothers should be closely monitored because of possible abnormal thyroid function, as shown by this study. It is likely that TSH-R-Ab from hyperthyroid mother usually consists of blocking antibody, rather than stimulating antibody, which might cause hypothyroidism in the neonates.
Birth Weight ; Follow-Up Studies ; Gestational Age ; Graves Disease ; Humans ; Hyperthyroidism* ; Hypothyroidism ; Incidence ; Infant, Newborn* ; Mothers* ; Pregnancy ; Prognosis ; Thyroid Gland*

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Allogeneic Bone Marrow Transplantation Experience for Children with Severe Aplastic Anemia and Refractory Leukemia.

Dae Chul JEONG ; Hack Ki KIM

Journal of the Korean Pediatric Society.1996;39(1):97-105.

PURPOSE: We reviewed the result of allogeneic bone marrow transplantation(BMT) from HLA-identical sibling donors in children with refractory stem cell disorder along with future implication. METHODS: Forty-two children with refractory stem cell disorder received BMT from HLA-identical sibling donors between Nov. 1983 and Feb. 1995. Out of 42 children, 23 cases were severe aplastic anemia(SAA) and 19 cases were refractory leukemias. There were 20 male and 22 female with median age of 13 years (range, 2-17) and median follow-up of 36 months (range, 4-139 months). RESULTS: 1) The overall survival rate of all patients was 73.8%. The survival rate for SAA cases was 87.0%, while that for leukemia was 57.9%. 2) Acute GVHD(> or = grade II) was observed in 16.7% of all patients, 8.7% of SAA patients and 26.3% of leukemia patients, respectively. Chronic GVHD developed in 9.5% of all patients, 4.8% of limited type and 4.8% of extended type. No death was directly attributable to GVHD. 3) The causes of death after allogeneic BMT were graft rejection(7.1%), relapse of leukemia(7.1%), thrombotic thrombocytopenic purpura(4.8%), veno-occlusive disease, sepsis and CMV pneumonia respectively 2.4%. 4) The most common complication except death after allogeneic BMT was herpes zoster(26.2%). The other complications were hemorrhagic cystitis(7.1%), bronchiolitis obliterans and measles respectively 2.4%. CONCLUSIONS: We confirmed that allogeneic BMT is the curable treatment for children with refractory stem cell disorder. The most important factors that influence the result of transplantation are interval between diagnosis and transplantation in severe aplastic anemia and remission state at transplantation in leukemia.
Anemia, Aplastic* ; Bone Marrow Transplantation* ; Bone Marrow* ; Bronchiolitis Obliterans ; Cause of Death ; Child* ; Diagnosis ; Female ; Follow-Up Studies ; Humans ; Leukemia* ; Male ; Measles ; Pneumonia ; Recurrence ; Sepsis ; Siblings ; Stem Cells ; Survival Rate ; Tissue Donors ; Transplants

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A Clinical Study of Febrile Convulsion and Factors Related to Recurrence.

Keun Wook LEE ; Eun Kyung CHEAN ; Young Taek JANG ; Wan Seob KIM

Journal of the Korean Pediatric Society.1996;39(1):88-96.

PURPOSE: To investigate the risk factors of recurrence after their first febrile convulsions. And to evaluate effect of anticonvulsant therapy with recurrent febrile convulsion infants and children. METHODS: We have studied the relationship of their clinical pictures and factors related to the risk of recurrence of 178 patients with primary or recurrent febrile convulsion, who were admitted to department of pediatrics, or visited to emergency room, chonju presbyterian medical center from january 1983 to december 1992. The 178 patients were followed up and consisted of the 97 patients with primary febrile convulsion and the others 81 patients with recurrent febrile convulsions. RESULTS: 1) In sex distribution, the boys(59%) outnumbered the girls(41%) and the ratio was 1.4:1. 2) 95% of overall patients were occurred febrile convulsion under 5 year-old and recurrent rate was 45.5%. In recurrent cases, the first episode under the 12 months was 50.6%(p<0.005). 3) There were family history of febrile convulsion in 28.4% of recurrent cases, compared to 10.9% of primary cases(p<0.001). 4) There was no significant difference with duration and type of convulsion in both groups. 5) Small proportion of 27 children were prescribed anticonvulsants (phenobarbital), but it's not reduced the recurrence and epilepsy significantly. CONCLUSIONS: The risk factor of recurrent febrile convulsion were the first episode under 12 month and familial history of febrile convulsion.
Anticonvulsants ; Child ; Child, Preschool ; Emergency Service, Hospital ; Epilepsy ; Humans ; Infant ; Jeollabuk-do ; Pediatrics ; Protestantism ; Recurrence* ; Risk Factors ; Seizures ; Seizures, Febrile* ; Sex Distribution

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Evaluation of Abnormal Coronary Artery Flow in Anomalous Coronary Artery Connections by Pulsed and Color Doppler Echocardiography.

Young Mi HONG

Journal of the Korean Pediatric Society.1996;39(1):78-87.

PURPOSE: Anomalous connection of coronary artery can result in damage of myocardial perfusion, myocardial infarction, or congestive heart failure. Echocardiography has been used to demonstrate coronary artery anomalies such as coronary artery fistula, anomalous origin of the left coronary artery from the pulmonary artery, and aneurysm of the coronary arteries in Kawasaki disease. Definitive diagnosis relies on cardiac catheterization The objectives of this study were to know the diagnostic sensitivity of echocardiography in detecting abnormal coronary artery connections, and identify the direction of blood flow by pulsed Doppler echocardiography in these patients. METHODS: Evidence of abnormal connections were found during echocardiographic examination in 25 patients(anomalous origin of left coronary artery from the pulmonary artery, coronary artery fistula, right ventricular to coronary artery sinusoids in pulmonary atresia with intact ventricular septum, and left ventricular to coronary artery sinusoids in aortic atresia with intact venticular septum). Two-dimensional imaging, color and pulsed Doppler echocardiography were performed in supine or left decubitus position from parasternal, apical, and subcostal views. The internal dimension of the coronary arteries and aortic root were measured at the level of aortic annulus during systole from the parastenal short axis view. RESULTS: 1) Diagnostic sensitivity by echocardiography in anomalous origin of the left coronary artery from the pulmonary artery was 85.7%(6/7). The direction of flow was from the left coronary artery into pulmonary artery during systole and reversed flow in diastole in 4 patients. There was continuous flow from the left coronary artery to the pulmonary artery in 1 patient. 2) Diagnostic sensitivity by echocardiography in congenital coronary artery fistula was 85.7% (6/7), and in acquired coronary artery fistula was 100%(4/4). There was continous systolic and diastolic flow from the coronary artery into the right ventricle or pulmonary artery by pulsed Doppler in all patients. 3) Diagnostic sensitivity in coronary sinusoid was 100% (7/7). Pulsed Doppler echocardiography revealed systolic flow from the right or left ventricle into coronary circulation during systole, and reversed flow during diastole. CONCLUSIONS: Color and pulsed Doppler echocardiography is extremely useful in detecting abnormal coronary artery connections. Although the sensitivity of echocardiography to coronary artery abnormalities is not 100%, there was no false positive. Cardiac catheterization may be necessary to confirm the diagnosis, but echocardiography is a helpful diagnostic tool in high risk infnats.
Aneurysm ; Axis, Cervical Vertebra ; Cardiac Catheterization ; Cardiac Catheters ; Coronary Circulation ; Coronary Vessels* ; Diagnosis ; Diastole ; Echocardiography ; Echocardiography, Doppler, Color* ; Echocardiography, Doppler, Pulsed ; Fistula ; Heart Failure ; Heart Ventricles ; Humans ; Mucocutaneous Lymph Node Syndrome ; Myocardial Infarction ; Perfusion ; Pulmonary Artery ; Pulmonary Atresia ; Systole ; Ventricular Septum

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