Literature DB >> 22897717

Clinical and immunological correction of DOCK8 deficiency by allogeneic hematopoietic stem cell transplantation following a reduced toxicity conditioning regimen.

Heidrun Boztug1, Cäcilia Karitnig-Weiß, Bernd Ausserer, Ellen D Renner, Michael H Albert, Julie Sawalle-Belohradsky, Bernd H Belohradsky, Georg Mann, Ernst Horcher, Alexandra Rümmele-Waibel, Rene Geyeregger, Karoly Lakatos, Christina Peters, Anita Lawitschka, Susanne Matthes-Martin.   

Abstract

Dedicator of cytokinesis 8 protein (DOCK8) deficiency is a combined immunodeficiency disorder characterized by an expanding clinical picture with typical features of recurrent respiratory or gastrointestinal tract infections, atopic eczema, food allergies, chronic viral infections of the skin, and blood eosinophilia often accompanied by elevated serum IgE levels. The only definitive treatment option is allogeneic hematopoietic stem cell transplantation (HSCT). We report a patient with early severe manifestation of DOCK8 deficiency, who underwent unrelated allogeneic HSCT at the age of 3 years following a reduced toxicity conditioning regimen. The transplant course was complicated by pulmonary aspergilloma pretransplantation, adenovirus (ADV) reactivation, and cytomegalovirus (CMV) pneumonitis 4 weeks after transplantation. With antifungal and antiviral treatment the patient recovered. Seven months after transplantation the patient is in excellent clinical condition. Eczematous rash, chronic viral skin infections, and food allergies have subsided, associated with normalization of IgE levels and absolute numbers of eosinophils. Chimerism analysis shows stable full donor chimerism. DOCK8 deficiency can be successfully cured by allogeneic HSCT. This treatment option should be considered early after diagnosis, as opportunistic infections and malignancies that occur more frequently during the natural course of the disease are associated with higher morbidity and mortality.

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Year:  2012        PMID: 22897717     DOI: 10.3109/08880018.2012.714844

Source DB:  PubMed          Journal:  Pediatr Hematol Oncol        ISSN: 0888-0018            Impact factor:   1.969


  21 in total

1.  Hematopoietic Stem Cell Transplantation as Treatment for Patients with DOCK8 Deficiency.

Authors:  Susanne E Aydin; Alexandra F Freeman; Waleed Al-Herz; Hamoud A Al-Mousa; Rand K Arnaout; Roland C Aydin; Vincent Barlogis; Bernd H Belohradsky; Carmem Bonfim; Robbert G Bredius; Julia I Chu; Oana C Ciocarlie; Figen Doğu; Hubert B Gaspar; Raif S Geha; Andrew R Gennery; Fabian Hauck; Abbas Hawwari; Dennis D Hickstein; Manfred Hoenig; Aydan Ikinciogullari; Christoph Klein; Ashish Kumar; Marianne R S Ifversen; Susanne Matthes; Ayse Metin; Benedicte Neven; Sung-Yun Pai; Suhag H Parikh; Capucine Picard; Ellen D Renner; Özden Sanal; Ansgar S Schulz; Friedhelm Schuster; Nirali N Shah; Evan B Shereck; Mary A Slatter; Helen C Su; Joris van Montfrans; Wilhelm Woessmann; John B Ziegler; Michael H Albert
Journal:  J Allergy Clin Immunol Pract       Date:  2018-11-02

2.  Matched related and unrelated donor hematopoietic stem cell transplantation for DOCK8 deficiency.

Authors:  Jennifer Cuellar-Rodriguez; Alexandra F Freeman; Jennifer Grossman; Helen Su; Mark Parta; Heardley Murdock; Nirali Shah; Catherine Bollard; Heidi H Kong; Niki Moutsopoulos; Kelly Stone; Juan Gea-Banacloche; Steven M Holland; Dennis D Hickstein
Journal:  Biol Blood Marrow Transplant       Date:  2015-01-27       Impact factor: 5.742

Review 3.  DOCK8 deficiency: Insights into pathophysiology, clinical features and management.

Authors:  Catherine M Biggs; Sevgi Keles; Talal A Chatila
Journal:  Clin Immunol       Date:  2017-06-15       Impact factor: 3.969

4.  DOCK8 deficiency: clinical and immunological phenotype and treatment options - a review of 136 patients.

Authors:  Susanne E Aydin; Sara Sebnem Kilic; Caner Aytekin; Ashish Kumar; Oscar Porras; Leena Kainulainen; Larysa Kostyuchenko; Ferah Genel; Necil Kütükcüler; Neslihan Karaca; Luis Gonzalez-Granado; Jordan Abbott; Daifulah Al-Zahrani; Nima Rezaei; Zeina Baz; Jens Thiel; Stephan Ehl; László Marodi; Jordan S Orange; Julie Sawalle-Belohradsky; Sevgi Keles; Steven M Holland; Özden Sanal; Deniz C Ayvaz; Ilhan Tezcan; Hamoud Al-Mousa; Zobaida Alsum; Abbas Hawwari; Ayse Metin; Susanne Matthes-Martin; Manfred Hönig; Ansgar Schulz; Capucine Picard; Vincent Barlogis; Andrew Gennery; Marianne Ifversen; Joris van Montfrans; Taco Kuijpers; Robbert Bredius; Gregor Dückers; Waleed Al-Herz; Sung-Yun Pai; Raif Geha; Gundula Notheis; Carl-Philipp Schwarze; Betül Tavil; Fatih Azik; Kirsten Bienemann; Bodo Grimbacher; Valerie Heinz; H Bobby Gaspar; Roland Aydin; Beate Hagl; Benjamin Gathmann; Bernd H Belohradsky; Hans D Ochs; Talal Chatila; Ellen D Renner; Helen Su; Alexandra F Freeman; Karin Engelhardt; Michael H Albert
Journal:  J Clin Immunol       Date:  2015-01-28       Impact factor: 8.317

Review 5.  Current Status of Dedicator of Cytokinesis-Associated Immunodeficiency: DOCK8 and DOCK2.

Authors:  Dimana Dimitrova; Alexandra F Freeman
Journal:  Dermatol Clin       Date:  2017-01       Impact factor: 3.478

6.  Flow cytometry biomarkers distinguish DOCK8 deficiency from severe atopic dermatitis.

Authors:  Erin Janssen; Erdyni Tsitsikov; Waleed Al-Herz; Gerard Lefranc; Andre Megarbane; Majed Dasouki; Francisco A Bonilla; Talal Chatila; Lynda Schneider; Raif S Geha
Journal:  Clin Immunol       Date:  2013-12-31       Impact factor: 3.969

7.  Hematopoietic stem cell transplant effectively rescues lymphocyte differentiation and function in DOCK8-deficient patients.

Authors:  Bethany A Pillay; Danielle T Avery; Joanne M Smart; Theresa Cole; Sharon Choo; Damien Chan; Paul E Gray; Katie Frith; Richard Mitchell; Tri Giang Phan; Melanie Wong; Dianne E Campbell; Peter Hsu; John B Ziegler; Jane Peake; Frank Alvaro; Capucine Picard; Jacinta Bustamante; Benedicte Neven; Andrew J Cant; Gulbu Uzel; Peter D Arkwright; Jean-Laurent Casanova; Helen C Su; Alexandra F Freeman; Nirali Shah; Dennis D Hickstein; Stuart G Tangye; Cindy S Ma
Journal:  JCI Insight       Date:  2019-04-25

8.  New Gene Variants Associated with the Risk of Chronic HBV Infection.

Authors:  Mengjie Fan; Jing Wang; Sa Wang; Tengyan Li; Hong Pan; Hankui Liu; Huifang Xu; Daria V Zhernakova; Stephen J O'Brien; Zhenru Feng; Le Chang; Erhei Dai; Jianhua Lu; Hongli Xi; Yanyan Yu; Jianguo Zhang; Binbin Wang; Zheng Zeng
Journal:  Virol Sin       Date:  2020-04-15       Impact factor: 4.327

Review 9.  Primary immunodeficiency update: Part I. Syndromes associated with eczematous dermatitis.

Authors:  Dominique C Pichard; Alexandra F Freeman; Edward W Cowen
Journal:  J Am Acad Dermatol       Date:  2015-09       Impact factor: 11.527

10.  Hematopoietic stem cell transplantation outcomes for 11 patients with dedicator of cytokinesis 8 deficiency.

Authors:  Waleed Al-Herz; Julia I Chu; Jet van der Spek; Raj Raghupathy; Michel J Massaad; Sevgi Keles; Catherine M Biggs; Lucinda Cockerton; Janet Chou; Ghassan Dbaibo; Scott A Elisofon; Rima Hanna-Wakim; Heung Bae Kim; Leslie E Lehmann; Douglas R McDonald; Luigi D Notarangelo; Paul Veys; Talal A Chatila; Raif S Geha; H Bobby Gaspar; Sung-Yun Pai
Journal:  J Allergy Clin Immunol       Date:  2016-04-06       Impact factor: 10.793

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