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syndrome of reconstitution immune in Children

Isabelle Viel-Thériault, Roseline Thibeault, Francois D Boucher, Jean-Philippe Drolet
Paradoxical immune reconstitution inflammatory syndrome is a well-described entity even in immunocompetent children, principally in association with Mycobacterium tuberculosis infections. Central nervous system involvement is a potential life-threatening form, sometimes refractory to standard treatment. We report the case of an HIV-negative refugee teenager, who presented with brain tuberculomas and pseudoabscesses responsive only to thalidomide.
November 2016: Pediatric Infectious Disease Journal
Noella Maria Delia Pereira, Ira Shah, Shilpa Kulkarni
Opsoclonus-myoclonus-ataxia (OMA) syndrome typically presents with chaotic eye movements and myoclonus with some patients exhibiting ataxia and behavioural disturbances. The pathogenesis may be inflammatory with an infectious or paraneoplastic trigger. We present a 13-year-old HIV-infected girl who was initially started on highly active antiretroviral therapy (HAART) in March 2013 with a CD4 count of 79 cells/cumm. Initially, the patient did not comply with treatment, resulting in a CD4+ count of 77 cells/mm(3) in November 2015 and prompting a new HAART scheme comprising lamivudine, tenofovir and ritonavir-boosted atazanavir...
October 2016: Oxford Medical Case Reports
Claire Dossier, Agnès Jamin, Georges Deschênes
Steroid sensitive nephrotic syndrome is marked by a massive proteinuria and loss of podocytes foot processes. The mechanism of the disease remains debated but recent publications suggest a primary role of Epstein-Barr Virus (EBV). EBV replication in the peripheral blood is found in 50% of patients during the first flare of the disease. The genetic locus of steroid sensitive nephrotic syndrome was also identified as influencing antibodies directed against EBNA1. EBV is able to establish, latent benign infection in memory B cells that display phenotypes similar to antigen-selected memory B cells...
January 2017: Pediatric Research
Isabelle Viel-Thériault, Roseline Thibeault, Francois D Boucher, Jean-Philippe Drolet
Paradoxical immune reconstitution inflammatory syndrome (IRIS) is a well described entity even in immunocompetent children, principally in association with Mycobacterium tuberculosis infections. Central nervous system involvement is a potential life-threatening form, sometimes refractory to standard treatment. We report the case of an HIV-negative refugee teenager, who presented with brain tuberculomas and pseudo-abscesses responsive only to thalidomide.
June 21, 2016: Pediatric Infectious Disease Journal
Sharon Nachman, Carmelita Alvero, Edward P Acosta, Hedy Teppler, Brenda Homony, Bobbie Graham, Terence Fenton, Xia Xu, Matthew L Rizk, Stephen A Spector, Lisa M Frenkel, Carol Worrell, Edward Handelsman, Andrew Wiznia
BACKGROUND: IMPAACT P1066 is a Phase I/II open-label multicenter trial to evaluate safety, tolerability, pharmacokinetics (PK), and efficacy of multiple raltegravir (RAL) formulations in human immunodeficiency virus (HIV)-infected youth. METHODS: Dose selection of the oral suspension formulation for each cohort (IV: 6 months to <2 years and V: 4 weeks to <6 months) was based on review of short-term safety (4 weeks) and intensive PK evaluation. Safety data through Weeks 24 and 48 and Grade ≥3 or serious adverse events (AEs) were assessed...
December 2015: Journal of the Pediatric Infectious Diseases Society
Dmitry Balashov, Anna Shcherbina, Michael Maschan, Pavel Trakhtman, Yulia Skvortsova, Larisa Shelikhova, Alexandra Laberko, Anna Livshits, Galina Novichkova, Alexei Maschan
The transplantation of stem cells from a matched unrelated donor (MUD) or a haploidentical mismatched related donor (MMRD) is a widely used variant of curative treatment for patients with primary immunodeficiency (PID). Currently, different strategies are used to reduce the risk of post-transplant complications and enhance immune reconstitution. We report the preliminary results of MUD and MMRD transplantation with TCRαβ/CD19 depletion in patients with PID (trial registered at as NCT02327351)...
November 2015: Biology of Blood and Marrow Transplantation
Annelies Van Rie, Shobna Sawry, Ruth Link-Gelles, Shabir Madhi, Lee Fairlie, Charl Verwey, Nasreen Mahomed, David Murdoch, Harry Moultrie
BACKGROUND: Paradoxical tuberculosis (TB)-associated Immune Reconstitution Inflammatory Syndrome (IRIS) is a common complication of combination antiretroviral treatment (cART) initiation in adults residing in resource-limited regions. Little is known about the burden and presentation of TB-IRIS in children initiating cART while receiving TB treatment. METHODS: Prospective cohort study of South African children initiating cART while on TB treatment. Children were assessed clinically and by chest x-ray before starting cART and at 2, 4, 6, and 12 weeks post cART initiation...
February 2016: Pediatric Pulmonology
Alessia Scarselli, Silvia Di Cesare, Claudia Capponi, Simona Cascioli, Maria L Romiti, Gigliola Di Matteo, Alessandra Simonetti, Paolo Palma, Andrea Finocchi, Barbarella Lucarelli, Rita M Pinto, Ippolita Rana, Giuseppe Palumbo, Maurizio Caniglia, Paolo Rossi, Rita Carsetti, Caterina Cancrini, Alessandro Aiuti
PURPOSE: Hematopoietic cell transplantation (HCT) provides a curative therapy for severe forms of primary immunodeficiencies (PID). While the timing and extent of T-cell reconstitution following transplant for PID has been studied in depth, less is known about the kinetics of B-cell development and long-term restoration of humoral functions, which been often reported to be suboptimal after HCT. METHODS: We studied longitudinally B-cell development and function in a cohort of 13 PID patients transplanted between 1997 and 2010, with a follow-up ranging from 0...
May 2015: Journal of Clinical Immunology
Thomas Schneider, Verena Moos
Within the last years the understanding of infection with Tropheryma whipplei was significantly enhanced by improvement of molecular biology, microbiology and immunology. The following entities of infection or carriage, respectively, with T. whipplei must be differentiated: besides in the context of classical Whipple's disease (CWD), the rare chronic infection with T. whipplei (estimated incidence: 1 : 1,000,000), T. whipplei can be detected more frequently in stool specimens of children with acute gastroenteritis or asymptomatic carriers, or as a cause of isolated endocarditis...
March 2015: Deutsche Medizinische Wochenschrift
Janet Leath Alexander, Marijean Miller
Frosted branch angiitis is a rare vascular reaction believed to be a nonspecific immune response to an infective, neoplastic, or idiopathic insult. The clinical presentation is characteristic and typically affects children and younger adults, and the prognosis is good. We report a case of frosted branch angiitis during immune recovery in a 2-year-old boy with Langerhans cell histiocytosis on systemic immunosuppressive therapy.
February 2015: Journal of AAPOS: the Official Publication of the American Association for Pediatric Ophthalmology and Strabismus
Renate A Richardus, C Ruth Butlin, Khorshed Alam, Kallyan Kundu, Annemieke Geluk, Jan Hendrik Richardus
BACKGROUND: Although BCG is used as a vaccine against tuberculosis, it also protects against leprosy. Previous evaluation over 18 years of an intervention of two doses BCG for 3536 household contacts of leprosy patients showed that 28 (23%) out of 122 contacts diagnosed with leprosy, developed symptoms 2-10 months after vaccination. This study describes contacts of leprosy patients in Bangladesh who developed leprosy within 12 weeks after receiving a single BCG dose. METHODS: A cluster RCT in Bangladesh aims to study the effectiveness of the BCG vaccine versus BCG in combination with single dose rifampicin (SDR) given 2 to 3 months after BCG, in the prevention of leprosy among contacts of newly diagnosed leprosy patients...
March 24, 2015: Vaccine
M C Elze, O Ciocarlie, A Heinze, S Kloess, T Gardlowski, R Esser, T Klingebiel, P Bader, S Huenecke, M Serban, U Köhl, J L Hutton
DCs are potent APCs and key regulators of innate and adaptive immunity. After allo-SCT, their reconstitution in the peripheral blood (PB) to levels similar to those in healthy individuals tends to be slow. We investigate the age- and sex-dependant immune reconstitution of myeloid (mDC) and plasmacytoid DC (pDC) in the PB of 45 children with leukaemia or myelodysplastic syndrome (aged 1-17 years, median 10) after allo-SCT with regard to relapse, acute GVHD (aGVHD) and relapse-free survival. Low pDC/μL PB up to day 60 post SCT are associated with higher incidence of moderate or severe aGVHD (P=0...
February 2015: Bone Marrow Transplantation
Margot R Krauss, D Robert Harris, Thalita Abreu, Fabiana G Ferreira, Noris Pavia Ruz, Carol Worrell, Rohan Hazra
OBJECTIVE: To evaluate the occurrence, clinical presentations and diagnostic methods for tuberculosis in a cohort of HIV-infected infants, children and adolescents from Latin America. METHODS: A retrospective analysis of children with tuberculosis and HIV was performed within a prospective observational cohort study conducted at multiple clinical sites in Latin America. RESULTS: Of 1114 HIV-infected infants, children, and adolescents followed from 2002 to 2011, 69 that could be classified as having confirmed or presumed tuberculosis were included in this case series; 52...
January 2015: Brazilian Journal of Infectious Diseases
Gilad D Birnbaum, Lucila Marquez, Kevin M Hwang, Andrea T Cruz
Clinical deterioration while receiving antituberculosis (anti-TB) therapy can be due to a number of etiologies, including drug resistance, disease progression despite effective therapy, or alternative diagnoses. We present the case of a 22-month-old girl diagnosed with TB meningitis 4 months prior to presentation. At time of her initial diagnosis, computed tomography showed hydrocephalus and basilar meningitis with some evidence of ischemic damage. She required placement of a ventriculoperitoneal shunt and was discharged on multidrug anti-TB therapy and corticosteroids...
August 2014: Pediatric Emergency Care
Haseena Hassan, Mark F Cotton, Helena Rabie
We report 7 HIV-infected children with cryptococcosis. Median age and CD4 counts were 9.3 years and 12 cells/mm, respectively. Two children died early. Of 4 children requiring prolonged amphotericin B and fluconazole at a dosage above 12 mg/kg/d, 3 presented with meningitis and 1 with fever. Immune reconstitution inflammatory syndrome contributed to morbidity through exacerbations at the primary site and elsewhere.
January 2015: Pediatric Infectious Disease Journal
Elisabetta Walters, Joanie Duvenhage, Heather R Draper, Anneke C Hesseling, Susan S Van Wyk, Mark F Cotton, Helena Rabie
BACKGROUND: Early initiation of antiretroviral therapy (ART) in HIV-infected infants reduces mortality and opportunistic infections including tuberculosis (TB). However, young HIV-infected children remain at high risk of TB disease following mycobacterial infection. We document the spectrum of TB disease in HIV-infected children <2 years of age on ART. METHODS: Retrospective cohort study; records of children <2 years of age initiating routine ART at Tygerberg Children's Hospital, Cape Town, January 2003-December 2010 were reviewed...
November 2014: Archives of Disease in Childhood
Ruth Link-Gelles, Harry Moultrie, Shobna Sawry, David Murdoch, Annelies Van Rie
BACKGROUND: People with HIV initiating combination antiretroviral therapy are at risk for tuberculosis-associated immune reconstitution inflammatory syndrome (TB-IRIS). While this syndrome has been well researched in adults, little is known about the incidence, case fatality, underlying immunopathology and treatment approaches in children. METHODS: Major databases were searched for articles related to TB-IRIS in children. Data were abstracted using standardized forms...
May 2014: Pediatric Infectious Disease Journal
Hayden Schwenk, Lynn Ramirez-Avila, Shu-Hsien Sheu, Christian Wuthrich, Jeff Waugh, Adam Was, Umberto Degirolami, Sandra Burchett, Igor J Koralnik, Asim Ahmed
Progressive multifocal leukoencephalopathy is a rare, demyelinating disease of the central nervous system caused by JC virus. Fewer than 30 cases have been reported in HIV- and non-infected children. We report the case of a 15-year-old girl with progressive multifocal leukoencephalopathy and AIDS who presented with nystagmus, dysarthria and ataxia. Following combined antiretroviral therapy, she developed immune reconstitution inflammatory syndrome, which proved fatal.
April 2014: Pediatric Infectious Disease Journal
Despoina Gkentzi, Marc Tebruegge, Gareth Tudor-Williams, Sam Walters, Hermione Lyall, Mike Sharland, Katja Doerholt
BACKGROUND: Immune reconstitution syndrome (IRS) is a relatively common complication in HIV-infected adults starting combination antiretroviral therapy (cART). Data on IRS in HIV-infected children remain limited and are largely restricted to resource-limited settings. This study investigated the incidence, spectrum and outcome of IRS in a pediatric cohort in the United Kingdom. METHODS: Retrospective analysis of clinical events during the first 12 months after initiation of cART in 135 treatment-naïve, HIV-infected children in the United Kingdom over a 5-year period...
September 2014: Pediatric Infectious Disease Journal
Elisabetta Venturini, Anna Turkova, Elena Chiappini, Luisa Galli, Maurizio de Martino, Claire Thorne
UNLABELLED: HIV is the top and tuberculosis is the second leading cause of death from infectious disease worldwide, with an estimated 8.7 million incident cases of tuberculosis and 2.5 million new HIV infections annually. The World Health Organization estimates that HIV prevalence among children with tuberculosis, in countries with moderate to high prevalence, ranges from 10 to 60%. The mechanisms promoting susceptibility of people with HIV to tuberculosis disease are incompletely understood, being likely caused by multifactorial processes...
2014: BMC Infectious Diseases
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