Infections in Infants with SCID: Isolation, Infection Screening, and Prophylaxis in PIDTC Centers,Journal of Clinical Immunology

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Infections in Infants with SCID: Isolation, Infection Screening, and Prophylaxis in PIDTC Centers
Journal of Clinical Immunology ( IF 7.2 ) Pub Date : 2020-10-02 , DOI: 10.1007/s10875-020-00865-9
Morna J Dorsey ₁ , Nicola A M Wright ₂ , Natalia S Chaimowitz ₃ , Blachy J Dávila Saldaña _{4,

5} , Holly Miller ₆ , Michael D Keller ₇ , Monica S Thakar ₈ , Ami J Shah ₉ , Rolla Abu-Arja ₁₀ , Jeffrey Andolina ₁₁ , Victor Aquino ₁₂ , J L Barnum ₁₃ , Jeffrey J Bednarski ₁₄ , Monica Bhatia ₁₅ , Francisco A Bonilla ₁₆ , Manish J Butte ₁₇ , Nancy J Bunin ₁₈ , Sharat Chandra _{19,

20} , Sonali Chaudhury ₂₁ , Karin Chen ₂₂ , Hey Chong ₁₃ , Geoffrey D E Cuvelier ₂₃ , Jignesh Dalal ₂₄ , Magee L DeFelice ₂₅ , Kenneth B DeSantes ₂₆ , Lisa R Forbes ₂₇ , Alfred Gillio ₂₈ , Fred Goldman ₂₉ , Avni Y Joshi ₃₀ , Neena Kapoor ₃₁ , Alan P Knutsen ₃₂ , Lisa Kobrynski ₃₃ , Jay A Lieberman ₃₄ , Jennifer W Leiding _{35,

36} , Benjamin Oshrine ₃₆ , Kiran P Patel ₃₇ , Susan Prockop ₃₈ , Troy C Quigg ₃₉ , Ralph Quinones ₄₀ , Kirk R Schultz ₄₁ , Christine Seroogy ₄₂ , David Shyr _{43,

44} , Subhadra Siegel ₄₅ , Angela R Smith ₄₆ , Troy R Torgerson ₈ , Mark T Vander Lugt ₄₇ , Lolie C Yu ₄₈ , Morton J Cowan ₁ , Rebecca H Buckley ₄₉ , Christopher C Dvorak ₁ , Linda M Griffith ₅₀ , Elie Haddad ₅₁ , Donald B Kohn ₅₂ , Brent Logan ₅₃ , Luigi D Notarangelo ₅₄ , Sung-Yun Pai _{55,

56} , Jennifer Puck ₁ , Michael A Pulsipher ₃₁ , Jennifer Heimall ₅₇

Affiliation

Division of Pediatric Allergy, Immunology, & Bone Marrow Transplant, Benioff Children's Hospital, University of California San Francisco, San Francisco, CA, USA.
Division of Hematology/Immunology, Department of Pediatrics, Alberta Children's Hospital, University of Calgary, Calgary, AB, Canada.
Section of Immunology, Allergy and Retrovirology, Department of Pediatrics, William T. Shearer Center for Human Immunobiology, Texas Children's Hospital, Baylor College of Medicine, Houston, TX, USA.
Division of Blood and Marrow Transplantation, Children's National Medical Center, Washington, DC, USA.
Department of Pediatrics, George Washington University, Washington, DC, USA.
Center for Cancer and Blood Disorders, Phoenix Children's Hospital, Phoenix, AZ, USA.
Division of Allergy & Immunology, Children's National Health System, and Division of Pediatrics, George Washington University, Washington, DC, USA.
Fred Hutchinson Cancer Research Center, University of Washington, Seattle, WA, USA.
Division of Stem Cell Transplantation and Regenerative Medicine, Lucille Packard Children's Hospital, Stanford School of Medicine, Stanford, CA, USA.
Nationwide Children's Hospital, Columbus, OH, USA.
Department of Pediatrics, Golisano Children's Hospital, University of Rochester Medical Center, Rochester, NY, USA.
University of Texas, Southwestern, Dallas, TX, USA.
UPMC Children's Hospital of Pittsburgh, Pittsburgh, PA, USA.
Department of Pediatrics, Washington University in St. Louis School of Medicine, St. Louis, MO, USA.
Pediatric Stem Cell Transplant Columbia, University Irving Medical Center, New York, NY, USA.
Northeast Allergy, Asthma & Immunology (private practice), Leominster, MA, USA.
Division of Immunology, Allergy, and Rheumatology, Department of Pediatrics, University of California Los Angeles, Los Angeles, CA, USA.
Cellular Therapy and Transplant Section, Division of Oncology, Children's Hospital of Philadelphia, Philadelphia, PA, USA.
Division of Bone Marrow Transplantation and Immune Deficiency, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, USA.
Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, OH, USA.
Division of Pediatric Hematology, Oncology, Stem Cell Transplantation, Ann & Robert H Lurie Children's Hospital of Chicago, Northwestern University Feinberg School of Medicine, Chicago, IL, USA.
Division of Allergy and Immunology, Department of Pediatrics, University of Utah School of Medicine, Salt Lake City, UT, USA.
Pediatric Blood and Marrow Transplant Program, CancerCare Manitoba, Department of Pediatrics and Child Health, Rady Faculty of Health Sciences, University of Manitoba, Winnipeg, MB, Canada.
Pediatric Bone Marrow Transplant, Rainbow Babies and Children's Hospital, Cleveland, OH, USA.
Division of Allergy and Immunology, Nemours/AI duPont Hospital for Children, Wilmington, DE, USA.
Division of Hematology, Oncology and Bone Marrow Transplant, Department of Pediatrics, University of Wisconsin School of Medicine, Madison, WI, USA.
William T Shearer Center for Human Immunobiology, Texas Children's Hospital, Baylor College of Medicine, Houston, TX, USA.
Joseph M Sanzari's Childrens Hospital, Hackensack University Medical Center, Hackensack, NJ, USA.
Department of Pediatrics, Division of Hematology Oncology, University of Alabama at Birmingham, Birmingham, AL, USA.
Pediatric and Adult Allergy/Immunology, Mayo Clinic, Rochester, MN, USA.
Section of Transplantation and Cellular Therapy, Children's Hospital Los Angeles Cancer and Blood Diseases Institute, USC Keck School of Medicine, Los Angeles, CA, USA.
Pediatric Allergy and Immunology, Cardinal Glennon Children's Hospital, St. Louis, MO, USA.
Children's Healthcare of Atlanta, Emory University Department of Pediatrics, Allergy and Immunology, Atlanta, GA, USA.
Department of Pediatrics, The University of Tennessee Health Science Center, Memphis, TN, USA.
Division of Allergy and Immunology, Department of Pediatrics, University of South Florida, St. Petersburg, FL, USA.
Johns Hopkins All Children's Hospital, Cancer and Blood Disorders Institute, St. Petersburg, FL, USA.
Children's Healthcare of Atlanta, Atlanta, GA, USA.
Department of Pediatrics, Bone Marrow Transplant Service, Memorial Sloan Kettering Cancer Center, New York, NY, USA.
Pediatric Blood and Marrow Transplantation Program, Methodist Children's Hospital, San Antonio, TX, USA.
Pediatric Hematology, Oncology and Bone Marrow Transplant, Children's Hospital Colorado, Aurora, CO, USA.
Michael Cuccione Childhood Cancer Research Program, BC Children's Hospital and Research Institute, Vancouver, British Columbia, Canada.
University of Wisconsin School of Medicine and Public Health, Madison, WI, USA.
Division of Pediatric Hematology/Oncology, Department of Pediatrics, University of Utah School of Medicine, Primary Children's Hospital, Salt Lake City, UT, USA.
Division of Stem Cell Transplant, Department of Pediatrics, Stanford Medicine, Lucile Packard Children's Hospital, Palo Alto, CA, USA.
Division of Pediatric Pulmonology, Allergy and Immunology and Sleep Medicine, Westchester Medical Center, Valhalla, NY, USA.
Pediatric Blood and Marrow Transplantation, University of Minnesota, Minneapolis, MN, USA.
Blood and Marrow Transplant Program, University of Michigan, Ann Arbor, MI, USA.
Division of Heme-Onc/HSCT, Children's Hospital/LSUHSC, New Orleans, LA, USA.
Division of Allergy and Immunology, Department of Pediatrics and Department of Immunology, Duke University School of Medicine, Durham, NC, USA.
Division of Allergy, Immunology and Transplantation, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD, USA.
Pediatric Immunology and Rheumatology Division, CHU Sainte-Justine, Department of Pediatrics, Department of Microbiology, Immunology and Infectious Disease, University of Montreal, Montreal, QC, Canada.
Department of Pediatrics, David Geffen School of Medicine, University of California, Los Angeles, Los Angeles, CA, USA.
Division of Biostatistics, Medical College of Wisconsin, Milwaukee, WI, USA.
Laboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD, USA.
Division of Hematology-Oncology, Boston Children's Hospital, Boston, MA, USA.
Department of Pediatric Oncology, Dana-Farber Cancer Institute, Boston, MA, USA.
Division of Allergy and Immunology, Children's Hospital of Philadelphia, Wood 3301, 3401 Civic Center Blvd, Philadelphia, PA, 19104, USA.

Purpose

The Primary Immune Deficiency Treatment Consortium (PIDTC) enrolled children with severe combined immunodeficiency (SCID) in a prospective natural history study of hematopoietic stem cell transplant (HSCT) outcomes over the last decade. Despite newborn screening (NBS) for SCID, infections occurred prior to HSCT. This study’s objectives were to define the types and timing of infection prior to HSCT in patients diagnosed via NBS or by family history (FH) and to understand the breadth of strategies employed at PIDTC centers for infection prevention.

Methods

We analyzed retrospective data on infections and pre-transplant management in patients with SCID diagnosed by NBS and/or FH and treated with HSCT between 2010 and 2014. PIDTC centers were surveyed in 2018 to understand their practices and protocols for pre-HSCT management.

Results

Infections were more common in patients diagnosed via NBS (55%) versus those diagnosed via FH (19%) (p = 0.012). Outpatient versus inpatient management did not impact infections (47% vs 35%, respectively; p = 0.423). There was no consensus among PIDTC survey respondents as to the best setting (inpatient vs outpatient) for pre-HSCT management. While isolation practices varied, immunoglobulin replacement and antimicrobial prophylaxis were more uniformly implemented.

Conclusion

Infants with SCID diagnosed due to FH had lower rates of infection and proceeded to HSCT more quickly than did those diagnosed via NBS. Pre-HSCT management practices were highly variable between centers, although uses of prophylaxis and immunoglobulin support were more consistent. This study demonstrates a critical need for development of evidence-based guidelines for the pre-HSCT management of infants with SCID following an abnormal NBS.

Trial Registration

NCT01186913

中文翻译：

SCID 婴儿的感染：PIDTC 中心的隔离、感染筛查和预防

目的

原发性免疫缺陷治疗联盟 (PIDTC) 招募患有严重联合免疫缺陷 (SCID) 的儿童参与一项关于过去十年造血干细胞移植 (HSCT) 结果的前瞻性自然史研究。尽管对 SCID 进行新生儿筛查 (NBS)，但感染发生在 HSCT 之前。本研究的目的是确定通过 NBS 或家族史 (FH) 诊断的患者在 HSCT 之前感染的类型和时间，并了解 PIDTC 中心采用的感染预防策略的广度。

方法

我们分析了 2010 年至 2014 年间由 NBS 和/或 FH 诊断并接受 HSCT 治疗的 SCID 患者的感染和移植前管理的回顾性数据。2018 年对 PIDTC 中心进行了调查，以了解他们的 HSCT 前管理实践和方案。

结果

通过 NBS 诊断的患者 (55%) 与通过 FH 诊断的患者 (19%) 相比，感染更为常见 ( p = 0.012)。门诊与住院管理对感染没有影响（分别为 47% 和 35%；p = 0.423）。PIDTC 调查受访者对 HSCT 前管理的最佳设置（住院患者与门诊患者）没有达成共识。虽然隔离做法各不相同，但免疫球蛋白替代和抗菌预防的实施更为统一。