Literature DB >> 25068252

Clinical course and outcome predictors of critically ill infants with complete DiGeorge anomaly following thymus transplantation.

Jan Hau Lee1, M Louise Markert, Christoph P Hornik, Elizabeth A McCarthy, Stephanie E Gupton, Ira M Cheifetz, David A Turner.   

Abstract

OBJECTIVES: To identify risk factors for PICU admission and mortality of infants with complete DiGeorge anomaly treated with thymus transplantation. We hypothesized that age at transplantation and the presence of congenital heart disease would be risk factors for emergent PICU admission, and these factors plus development of septicemia would increase morbidity and mortality.
DESIGN: Retrospective review.
SETTING: Academic medical-surgical PICU. PATIENTS: All infants with complete DiGeorge anomaly treated with thymus transplantation between January 1, 1993, and July 1, 2010.
INTERVENTIONS: None.
MEASUREMENTS AND MAIN RESULTS: Consent was obtained from 71 infants with complete DiGeorge anomaly for thymus transplantation, and 59 infants were transplanted. Median age at transplantation was 5.0 months (range, 1.1-22.1 mo). After transplantation, 12 of 59 infants (20%) required 25 emergent PICU admissions. Seven of 12 infants (58%) survived to PICU discharge with six surviving 6 months posttransplantation. Forty-two of 59 infants (71%) transplanted had congenital heart disease, and 9 of 12 (75%) who were admitted to the PICU had congenital heart disease. In 15 of 25 admissions (60%), intubation and mechanical ventilation were necessary. There was no difference between median ventilation-free days between infants with and without congenital heart disease (33 d vs 23 d, p = 0.544). There was also no correlation between ventilation-free days and age of transplantation (R, 0.17; p = 0.423). Age at transplantation and the presence of congenital heart disease were not associated with risk for PICU admission (odds ratio, 0.95; 95% CI, 0.78-1.15 and odds ratio, 1.27; 95% CI, 0.30-5.49, respectively) or PICU mortality (odds ratio, 0.98; 95% CI, 0.73-1.31 and odds ratio, 0.40; 95% CI, 0.15-1.07, respectively).
CONCLUSIONS: Most transplanted infants did not require emergent PICU admission. Age at transplantation and the presence of congenital heart disease were not associated with PICU admission or mortality.

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Year:  2014        PMID: 25068252      PMCID: PMC4156516          DOI: 10.1097/PCC.0000000000000219

Source DB:  PubMed          Journal:  Pediatr Crit Care Med        ISSN: 1529-7535            Impact factor:   3.624


  24 in total

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Journal:  Blood       Date:  2010-06-07       Impact factor: 22.113

2.  Complete DiGeorge syndrome: persistence of profound immunodeficiency.

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3.  Foetal thymic transplant in a case of Digeorge's syndrome.

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5.  Postnatal thymus transplantation with immunosuppression as treatment for DiGeorge syndrome.

Authors:  M Louise Markert; Marilyn J Alexieff; Jie Li; Marcella Sarzotti; Daniel A Ozaki; Blythe H Devlin; Debra A Sedlak; Gregory D Sempowski; Laura P Hale; Henry E Rice; Samuel M Mahaffey; Michael A Skinner
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7.  Survival in a recent cohort of mechanically ventilated pediatric allogeneic hematopoietic stem cell transplantation recipients.

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Authors:  Robert F Tamburro; Raymond C Barfield; Michele L Shaffer; Surender Rajasekaran; Paul Woodard; R Ray Morrison; Scott C Howard; Richard T Fiser; Jeffrey E Schmidt; Elaine M Sillos
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10.  Bone marrow transplantation (BMT) in Europe for primary immunodeficiencies other than severe combined immunodeficiency: a report from the European Group for BMT and the European Group for Immunodeficiency.

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