We report three autopsy cases of congenital cytomegalovirus (CMV) infection in fetuses with a review of literature. The clinical manifestations in these cases of congenital CMV infection include intrauterine fetal death, hydrops fetalis, and CMV pneumonia associated with cardiovascular defect. The pathological characteristics were as follows: 1) the kidney was the most frequently involved organ, followed by lung and liver, 2) CMV inclusions were found predominantly in epithelial cells and to a lesser degree in endothelial cells, 3) intrahepatic bile duct epithelial cells were frequently involved, and 4) inflammatory reaction around CMV inclusions was not prominent in the early stage of pregnancy. Diagnostic confirmation was obtained by in situ hybridization (ISH) using a biotinylated CMV-DNA probe, which demonstrated intranuclear inclusions and sometimes recognized cells that did not show intranuclear inclusion.
Autopsy ; Case Report ; Cytomegalovirus Infections/virology ; Cytomegalovirus Infections/pathology* ; Cytomegalovirus Infections/congenital* ; Female ; Fetal Diseases ; Human ; Male ; Pregnancy ; Pregnancy Complications, Infectious

We report three autopsy cases of congenital cytomegalovirus (CMV) infection in fetuses with a review of literature. The clinical manifestations in these cases of congenital CMV infection include intrauterine fetal death, hydrops fetalis, and CMV pneumonia associated with cardiovascular defect. The pathological characteristics were as follows: 1) the kidney was the most frequently involved organ, followed by lung and liver, 2) CMV inclusions were found predominantly in epithelial cells and to a lesser degree in endothelial cells, 3) intrahepatic bile duct epithelial cells were frequently involved, and 4) inflammatory reaction around CMV inclusions was not prominent in the early stage of pregnancy. Diagnostic confirmation was obtained by in situ hybridization (ISH) using a biotinylated CMV-DNA probe, which demonstrated intranuclear inclusions and sometimes recognized cells that did not show intranuclear inclusion.
Autopsy ; Case Report ; Cytomegalovirus Infections/virology ; Cytomegalovirus Infections/pathology* ; Cytomegalovirus Infections/congenital* ; Female ; Fetal Diseases ; Human ; Male ; Pregnancy ; Pregnancy Complications, Infectious

PURPOSE: To observe changes in audiology, intellectual development, behavior development, and physical growth during systematic follow-up of infants with asymptomatic congenital human cytomegalovirus (HCMV) infection. MATERIALS AND METHODS: Fifty-two infants diagnosed with asymptomatic congenital HCMV infection from July 2003 to July 2007 served as the infection group, and 21 healthy infants served as the control group. All infants were confirmed to have HCMV infection by Fluorescent Quantative polymerase chain reaction (FQ-PCR). In both the infection and control groups, the neonates and infants at 3 months, 6 months, and 1 year of age underwent examinations. RESULTS: 1) 20 items of National Black Nurses Association (NBNA) scores of neonates 12-14 days after birth in 2 groups were 38.3 +/- 1.95 and 38.5 +/- 2.29, without significant differences. 2) Auditory test: 50 ears of 25 cases in the infection group showed abnormal auditory thresholds in V waves with an abnormal rate of 14%, while no abnormalities were found in 21 cases in the control group. 3) Mental and psychomotor development index scores in the control group (107.49 +/- 11.31 and 107.19 +/- 10.98) were compared with those in 41 asymptomatically infected infants at 1 year of age (107.21 +/- 9.96 and 108.31 +/- 11.25), and no statistically significant difference was noted. CONCLUSION: 1) An elevated threshold in the V wave was present in asymptomatically infected infants, but could not be detected through otoacoustic emission (OAE) screening. 2) Either in the neonatal or infant periods, asymptomatic congenital HCMV infection did not have a significant influence on nervous behavior or on physical and intellectual development.
Auditory Threshold ; *Child Development ; Cytomegalovirus Infections/*complications/congenital ; Female ; Humans ; Infant ; Infectious Disease Transmission, Vertical ; Male ; Neuropsychological Tests ; Psychomotor Performance

OBJECTIVETo evaluate the efficacy and safety of ganciclovir therapy for congenital cytomegalovirus (CMV) infection in newborn infants.

METHODSThe randomized controlled trials (RCTs) and quasi-RCTs on ganciclovir therapy for congenital CMV were reviewed in the following electronic databases: PubMed (January 1988 to January 2009), EMbase (January 1988 to January 2009), the Cochrane library (Issue 3, 2003 and Issue 1, 2009), the Chinese Journals Full-text Database (January 1994 to January 2009), the Chinese Biological Medical Disc (January 1994 to January 2009) and the Chinese Medical Current Contents (January 1994 to January 2009). Quality assessment, data extraction, and meta analysis were performed.

RESULTSTen papers were included. Meta analysis showed that the ganciclovir therapy increased the improvement rate (91.4% vs 34.0%; p<0.01) and led CMV infection indexes to become negative in more patients (87.6% vs 15.3%; p<0.01) and decreased incidence of hearing disturbance (4.7% vs 37.2%; p<0.01) as compared with the non-ganciclovir therapy control group. The incidence of the ganciclovir-therapy-related side effects was low.

CONCLUSIONSGanciclovir treatment may increase the improvement rate and the rate of CMV infection indexes becoming negative, and decrease incidence of hearing disturbance, with few side effects, in newborn infants with CMV infection. However the supporting evidence is not strong due to few trials and more high-quality research is needed.

Antiviral Agents ; therapeutic use ; Cytomegalovirus Infections ; complications ; congenital ; drug therapy ; Follow-Up Studies ; Ganciclovir ; therapeutic use ; Hearing Disorders ; etiology ; Humans ; Infant, Newborn

OBJECTIVETo investigate the impact of congenital cytomegalovirus infection on the hearing ability in infants.

METHODSBy using the tools of distortion product otoacoustic emission (DPOAE) and auditory brain-stem response (ABR), the hearing ability of 38 infants with congenital cytomegalovirus infection and 16 cases of normal controls during neonatal periods was screened with a follow-up study at 6 and 24 months.

RESULTIn infants with congenital cytomegalovirus infection, 86.8% (66/76) ears at neonatal stage and 76.3% (58/76) ears at 6 months passed the tests; while in normal controls, 96.9% (31/32) ears passed the tests. The reaction threshold of ABR V in infants with congenital cytomegalovirus infection was higher than that in normal controls (P<0.005). Furthermore,in infants with congenital cytomegalovirus infection, 13 ears (17.1%) were extreme hearing loss, 5 ears (6.6%) were severe hearing loss, and 6 ears (7.9%) were moderately severe hearing loss. The incidence of hearing loss during the follow-up was 7.9% (3/38) at neonatal stage, 23.7% (9/38) at 3-4 months, and 7.9% (3/38) after 6 months.

CONCLUSIONThe congenital cytomegalovirus infection could cause the prompt and late-onset hearing loss. The combination of the laboratory evidence with the dynamic hearing screening may contribute to the early detection of hearing loss in infants with congenital cytomegalovirus infection.

China ; epidemiology ; Cytomegalovirus Infections ; complications ; congenital ; physiopathology ; Evoked Potentials, Auditory, Brain Stem ; Female ; Follow-Up Studies ; Hearing Loss, Bilateral ; epidemiology ; prevention & control ; Humans ; Infant, Newborn ; Male ; Neonatal Screening ; Otoacoustic Emissions, Spontaneous

OBJECTIVEHemorrhagic cystitis (HC) is one of the common complications of hematopoietic stem cell transplantation (HSCT), which causes significant pain, prolongs hospitalization, and occasionally results in renal failure and death. This study aimed at investigating the incidence, risk factors, and outcome of HC in children post umbilical cord blood transplantation (UCBT) and peripheral blood stem cell transplantation (PBSCT).

METHODSFrom October 1998 to June the clinical records of 53 pediatric patients (aged 2-18 years with median age of 7.5 years) in our HCST center who underwent UCBT (n = 37) and PBSCT (n = 16) were retrospectively analyzed. Thirty out of 53 patients were diagnosed as hereditary hemolytic anemia (56.6%), and the others as haematological malignancies (43.4%): of whom 8 had acute lymphoblastic leukemia, 12 acute myeloid leukemia, 2 chronic myeloid leukemia and 1 non-hodgkin lymphoma. Conditioning regimen varied according to disease and clinical status, however based on cyclophosphamide (CTX, 120-200 mg/kg) and busulphan (BU, 12-16 mg/kg) in the cohort. Total body irradiation (TBI) or total lymphoid irradiation was added in 7 patients respectively. The patients were divided into regular treatment group (RTG) with 15 cases who received hyperhydration, alkalinizing, diuresis and Mesna during CTX infusion and prostaglandin E1 (PGE1) group (PEG) with 38 cases who received hyperhydration, alkalinizing, diuresis and Mesna plus prostaglandin E1 (0.03 microg/kg.h). The risk factors of HC were examined by univariate and multivariate analysis.

RESULTSIn all, 11 of the 53 transplanted patients developed HC (21%) with a median onset time of day +15 (rage day +2 - +25). HC was classified as early in 4 (36%) and late in 7 (64%), and scored as grade Iin 2 cases (18%), grade II in 4 (36%) and grade III in 5 (46%). There was no significant difference between RTG and PEG in the incidence of HC, however, the incidence was much higher in the group of patients who were > or = 6 years old, positive group of graft-versus-host disease (GVHD) and group of cytomegalovirus (CMV) infection than that in the group of patients who were < 6 years of age (32% vs. 8%, P < 0.05), negative group of GVHD (35% vs. 7%, P < 0.05) and CMV non-infected group (62% vs. 13%, P < 0.05), respectively. Furthermore, by multivariate analysis, > or = 6 years old (OR = 3.53, P < 0.05) and CMV infection (OR = 4.31, P < 0.05) were significant risk factors for HC. Three of 11 patients were treated with bladder irrigation. All patients recovered from HC in a median 12.8 days (range 2-53 days).

CONCLUSIONOlder age (> or = 6 years) as well as CMV infection were the risk factors of HC. Both hyperhydration and Mesna were effective in preventing HC, while addition of PGE1 could not reduce the incidence of HC. The prognosis of HC in children post HSCT was satisfactory.

Adolescent ; Age Factors ; Anemia, Hemolytic, Congenital ; metabolism ; therapy ; Body Water ; metabolism ; Child ; Child, Preschool ; Cystitis ; epidemiology ; etiology ; prevention & control ; therapy ; Cytomegalovirus Infections ; complications ; physiopathology ; Female ; Fluid Therapy ; methods ; Hematologic Neoplasms ; metabolism ; therapy ; Hematopoietic Stem Cell Transplantation ; adverse effects ; methods ; Humans ; Incidence ; Male ; Mesna ; therapeutic use ; Multivariate Analysis ; Protective Agents ; therapeutic use ; Retrospective Studies ; Risk Factors ; Treatment Outcome