Literature DB >> 31597133

Targeted Therapy with Biologicals and Small Molecules in Primary Immunodeficiencies.

Ottavia Maria Delmonte1, Luigi Daniele Notarangelo2.   

Abstract

Primary immunodeficiencies are disorders resulting from mutations in genes involved in immune defense and immune regulation. These conditions are characterized by various combinations of recurrent infections, autoimmunity, lymphoproliferation, inflammatory manifestations, and malignancy. In the last 20 years, newborn screening programs and next generation sequencing techniques have increased the ability to diagnose primary immunodeficiencies. Furthermore, an advanced understanding of the molecular basis of these inherited disorders has led to the implementation of targeted therapies that utilize small molecules and biologics to modulate the activity of impaired intracellular pathways. This article will discuss selected primary immunodeficiencies, the genetic defects of which have been recently studied and are amenable to targeted therapy as a reflection of the potential of precision medicine in the future.
© 2019 The Author(s) Published by S. Karger AG, Basel.

Entities:  

Keywords:  Combined immunodeficiency; Immunodysregulation; Monoclonal antibody; Precision medicine; Primary immunodeficiency diseases; Small molecule inhibitor

Mesh:

Substances:

Year:  2019        PMID: 31597133      PMCID: PMC7098309          DOI: 10.1159/000503997

Source DB:  PubMed          Journal:  Med Princ Pract        ISSN: 1011-7571            Impact factor:   1.927


  92 in total

1.  Abatacept alleviates severe autoimmune symptoms in a patient carrying a de novo variant in CTLA-4.

Authors:  Sangmoon Lee; Jin Soo Moon; Cho-Rong Lee; Hye-Eun Kim; Sun-Mi Baek; Solha Hwang; Gyeong Hoon Kang; Jeong Kee Seo; Choong Ho Shin; Hyoung Jin Kang; Jae Sung Ko; Sung Gyoo Park; Murim Choi
Journal:  J Allergy Clin Immunol       Date:  2015-10-21       Impact factor: 10.793

Review 2.  A 1-year-old girl with a gain-of-function STAT1 mutation treated with hematopoietic stem cell transplantation.

Authors:  Juan Carlos Aldave; Enrique Cachay; Luis Núñez; Ausberto Chunga; Sergio Murillo; Sophie Cypowyj; Jacinta Bustamante; Anne Puel; Jean-Laurent Casanova; Armando Koo
Journal:  J Clin Immunol       Date:  2013-10-09       Impact factor: 8.317

Review 3.  NLRC4 inflammasomopathies.

Authors:  Neil Romberg; Tiphanie P Vogel; Scott W Canna
Journal:  Curr Opin Allergy Clin Immunol       Date:  2017-12

4.  Hematopoietic stem cell transplant in patients with activated PI3K delta syndrome.

Authors:  Zohreh Nademi; Mary A Slatter; Christopher C Dvorak; Benedicte Neven; Alain Fischer; Felipe Suarez; Claire Booth; Kanchan Rao; Alexandra Laberko; Julia Rodina; Yves Bertrand; Sylwia Kołtan; Robert Dębski; Terence Flood; Mario Abinun; Andrew R Gennery; Sophie Hambleton; Stephan Ehl; Andrew J Cant
Journal:  J Allergy Clin Immunol       Date:  2016-11-12       Impact factor: 10.793

5.  Signal transducer and activator of transcription 1 (STAT1) gain-of-function mutations and disseminated coccidioidomycosis and histoplasmosis.

Authors:  Elizabeth P Sampaio; Amy P Hsu; Joseph Pechacek; Hannelore I Bax; Dalton L Dias; Michelle L Paulson; Prabha Chandrasekaran; Lindsey B Rosen; Daniel S Carvalho; Li Ding; Donald C Vinh; Sarah K Browne; Shrimati Datta; Joshua D Milner; Douglas B Kuhns; Debra A Long Priel; Mohammed A Sadat; Michael Shiloh; Brendan De Marco; Michael Alvares; Jason W Gillman; Vivek Ramarathnam; Maite de la Morena; Liliana Bezrodnik; Ileana Moreira; Gulbu Uzel; Daniel Johnson; Christine Spalding; Christa S Zerbe; Henry Wiley; David E Greenberg; Susan E Hoover; Sergio D Rosenzweig; John N Galgiani; Steven M Holland
Journal:  J Allergy Clin Immunol       Date:  2013-03-28       Impact factor: 10.793

6.  Efficacy and safety of rilonacept (interleukin-1 Trap) in patients with cryopyrin-associated periodic syndromes: results from two sequential placebo-controlled studies.

Authors:  Hal M Hoffman; Martin L Throne; N J Amar; Mohamed Sebai; Alan J Kivitz; Arthur Kavanaugh; Steven P Weinstein; Pavel Belomestnov; George D Yancopoulos; Neil Stahl; Scott J Mellis
Journal:  Arthritis Rheum       Date:  2008-08

7.  Familial chilblain lupus due to a gain-of-function mutation in STING.

Authors:  Nadja König; Christoph Fiehn; Christine Wolf; Max Schuster; Emanuel Cura Costa; Victoria Tüngler; Hugo Ariel Alvarez; Osvaldo Chara; Kerstin Engel; Raphaela Goldbach-Mansky; Claudia Günther; Min Ae Lee-Kirsch
Journal:  Ann Rheum Dis       Date:  2016-08-26       Impact factor: 19.103

8.  Ruxolitinib partially reverses functional natural killer cell deficiency in patients with signal transducer and activator of transcription 1 (STAT1) gain-of-function mutations.

Authors:  Alexander Vargas-Hernández; Emily M Mace; Ofer Zimmerman; Christa S Zerbe; Alexandra F Freeman; Sergio Rosenzweig; Jennifer W Leiding; Troy Torgerson; Matthew C Altman; Edith Schussler; Charlotte Cunningham-Rundles; Ivan K Chinn; Alexandre F Carisey; Imelda C Hanson; Nicholas L Rider; Steven M Holland; Jordan S Orange; Lisa R Forbes
Journal:  J Allergy Clin Immunol       Date:  2017-10-27       Impact factor: 10.793

9.  Dominant Splice Site Mutations in PIK3R1 Cause Hyper IgM Syndrome, Lymphadenopathy and Short Stature.

Authors:  Slavé Petrovski; Roberta E Parrott; Joseph L Roberts; Hongxiang Huang; Jialong Yang; Balachandra Gorentla; Talal Mousallem; Endi Wang; Martin Armstrong; Duncan McHale; Nancie J MacIver; David B Goldstein; Xiao-Ping Zhong; Rebecca H Buckley
Journal:  J Clin Immunol       Date:  2016-04-13       Impact factor: 8.317

10.  Disease Evolution and Response to Rapamycin in Activated Phosphoinositide 3-Kinase δ Syndrome: The European Society for Immunodeficiencies-Activated Phosphoinositide 3-Kinase δ Syndrome Registry.

Authors:  Maria Elena Maccari; Hassan Abolhassani; Asghar Aghamohammadi; Alessandro Aiuti; Olga Aleinikova; Catherine Bangs; Safa Baris; Federica Barzaghi; Helen Baxendale; Matthew Buckland; Siobhan O Burns; Caterina Cancrini; Andrew Cant; Pascal Cathébras; Marina Cavazzana; Anita Chandra; Francesca Conti; Tanya Coulter; Lisa A Devlin; J David M Edgar; Saul Faust; Alain Fischer; Marina Garcia-Prat; Lennart Hammarström; Maximilian Heeg; Stephen Jolles; Elif Karakoc-Aydiner; Gerhard Kindle; Ayca Kiykim; Dinakantha Kumararatne; Bodo Grimbacher; Hilary Longhurst; Nizar Mahlaoui; Tomas Milota; Fernando Moreira; Despina Moshous; Anna Mukhina; Olaf Neth; Benedicte Neven; Alexandra Nieters; Peter Olbrich; Ahmet Ozen; Jana Pachlopnik Schmid; Capucine Picard; Seraina Prader; William Rae; Janine Reichenbach; Stephan Rusch; Sinisa Savic; Alessia Scarselli; Raphael Scheible; Anna Sediva; Svetlana O Sharapova; Anna Shcherbina; Mary Slatter; Pere Soler-Palacin; Aurelie Stanislas; Felipe Suarez; Francesca Tucci; Annette Uhlmann; Joris van Montfrans; Klaus Warnatz; Anthony Peter Williams; Phil Wood; Sven Kracker; Alison Mary Condliffe; Stephan Ehl
Journal:  Front Immunol       Date:  2018-03-16       Impact factor: 7.561

View more
  6 in total

1.  Diagnostic Yield and Therapeutic Consequences of Targeted Next-Generation Sequencing in Sporadic Primary Immunodeficiency.

Authors:  Georgios Sogkas; Natalia Dubrowinskaja; Katharina Schütz; Lars Steinbrück; Jasper Götting; Nicolaus Schwerk; Ulrich Baumann; Bodo Grimbacher; Torsten Witte; Reinhold E Schmidt; Faranaz Atschekzei
Journal:  Int Arch Allergy Immunol       Date:  2021-10-07       Impact factor: 2.749

2.  Molecular diagnosis of childhood immune dysregulation, polyendocrinopathy, and enteropathy, and implications for clinical management.

Authors:  Sarah K Baxter; Tom Walsh; Silvia Casadei; Mary M Eckert; Eric J Allenspach; David Hagin; Gesmar Segundo; Ming K Lee; Suleyman Gulsuner; Brian H Shirts; Kathleen E Sullivan; Michael D Keller; Troy R Torgerson; Mary-Claire King
Journal:  J Allergy Clin Immunol       Date:  2021-04-20       Impact factor: 10.793

3.  Frequency and Manifestations of Autoimmunity Among Children Registered in the Kuwait National Primary Immunodeficiency Registry.

Authors:  Michel J Massaad; Mohammad Zainal; Waleed Al-Herz
Journal:  Front Immunol       Date:  2020-06-02       Impact factor: 7.561

Review 4.  Pathogenesis of Autoimmune Cytopenias in Inborn Errors of Immunity Revealing Novel Therapeutic Targets.

Authors:  Manuela Cortesi; Annarosa Soresina; Laura Dotta; Chiara Gorio; Marco Cattalini; Vassilios Lougaris; Fulvio Porta; Raffaele Badolato
Journal:  Front Immunol       Date:  2022-04-06       Impact factor: 8.786

Review 5.  Recent advances in primary immunodeficiency: from molecular diagnosis to treatment.

Authors:  Giorgia Bucciol; Isabelle Meyts
Journal:  F1000Res       Date:  2020-03-19

6.  Update in Primary Immunodeficiencies.

Authors:  Lucia Leonardi; Beatrice Rivalta; Caterina Cancrini; Elena Chiappini; Claudio Cravidi; Carlo Caffarelli; Sara Manti; Mauro Calvani; Alberto Martelli; Michele Miraglia Del Giudice; Marzia Duse; Gian Luigi Marseglia; Fabio Cardinale
Journal:  Acta Biomed       Date:  2020-09-15
  6 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.