Efficacy and safety of anakinra and canakinumab in PSTPIP1-associated inflammatory diseases: a comprehensive scoping review

IntroductionThis scoping review explores the effectiveness of IL-1 pathway inhibitors in managing PSTPIP1-associated inflammatory diseases (PAID). These diseases are marked by abnormal IL-1 pathway activation due to genetic mutations.MethodsOur methodology adhered to a pre-published protocol and inv...

Full description

Bibliographic Details
Main Authors: Juan Luis Sanz-Cabanillas, Francisco Gómez-García, Pedro Jesús Gómez-Arias, Ana Montilla-López, Jesús Gay-Mimbrera, Juan Ruano, Beatriz Isla-Tejera, Esmeralda Parra-Peralbo
Format: Article
Language:English
Published: Frontiers Media S.A. 2024-01-01
Series:Frontiers in Immunology
Subjects:
Online Access:https://www.frontiersin.org/articles/10.3389/fimmu.2023.1339337/full
_version_ 1797360335880978432
author Juan Luis Sanz-Cabanillas
Juan Luis Sanz-Cabanillas
Francisco Gómez-García
Francisco Gómez-García
Pedro Jesús Gómez-Arias
Pedro Jesús Gómez-Arias
Ana Montilla-López
Jesús Gay-Mimbrera
Juan Ruano
Juan Ruano
Juan Ruano
Beatriz Isla-Tejera
Beatriz Isla-Tejera
Esmeralda Parra-Peralbo
author_facet Juan Luis Sanz-Cabanillas
Juan Luis Sanz-Cabanillas
Francisco Gómez-García
Francisco Gómez-García
Pedro Jesús Gómez-Arias
Pedro Jesús Gómez-Arias
Ana Montilla-López
Jesús Gay-Mimbrera
Juan Ruano
Juan Ruano
Juan Ruano
Beatriz Isla-Tejera
Beatriz Isla-Tejera
Esmeralda Parra-Peralbo
author_sort Juan Luis Sanz-Cabanillas
collection DOAJ
description IntroductionThis scoping review explores the effectiveness of IL-1 pathway inhibitors in managing PSTPIP1-associated inflammatory diseases (PAID). These diseases are marked by abnormal IL-1 pathway activation due to genetic mutations.MethodsOur methodology adhered to a pre-published protocol and involved a thorough search of MEDLINE and EMBASE databases up to February 2022, following the Joanna Briggs Institute Reviewer’s Manual and the PRISMA Extension for Scoping Reviews. The review included studies reporting on IL-1 pathway inhibitor use in PAID patients.ResultsFrom an initial pool of 5,225 articles, 36 studies involving 43 patients were selected. The studies predominantly used observational designs and exhibited diversity in patient demographics, treatment approaches, and outcomes. Anakinra and canakinumab demonstrated promise in treating sterile pyogenic arthritis, pyoderma gangrenosum, and acne (PAPA) and PSTPIP1-associated myeloid-related-proteinemia inflammatory (PAMI) syndromes, with scant data on other syndromes. Notably, there was a paucity of information on the adverse effects of these treatments, necessitating cautious interpretation of their safety profile.ConclusionCurrent evidence on IL-1 pathway inhibitors for PAID is primarily from observational studies and remains limited. Rigorous research with larger patient cohorts is imperative for more definitive conclusions. Collaborative efforts among specialized research centers and international health initiatives are key to advancing this field.
first_indexed 2024-03-08T15:38:05Z
format Article
id doaj.art-bdaffb302326414e9a5c10d7da31ef1c
institution Directory Open Access Journal
issn 1664-3224
language English
last_indexed 2024-03-08T15:38:05Z
publishDate 2024-01-01
publisher Frontiers Media S.A.
record_format Article
series Frontiers in Immunology
spelling doaj.art-bdaffb302326414e9a5c10d7da31ef1c2024-01-09T17:47:46ZengFrontiers Media S.A.Frontiers in Immunology1664-32242024-01-011410.3389/fimmu.2023.13393371339337Efficacy and safety of anakinra and canakinumab in PSTPIP1-associated inflammatory diseases: a comprehensive scoping reviewJuan Luis Sanz-Cabanillas0Juan Luis Sanz-Cabanillas1Francisco Gómez-García2Francisco Gómez-García3Pedro Jesús Gómez-Arias4Pedro Jesús Gómez-Arias5Ana Montilla-López6Jesús Gay-Mimbrera7Juan Ruano8Juan Ruano9Juan Ruano10Beatriz Isla-Tejera11Beatriz Isla-Tejera12Esmeralda Parra-Peralbo13Inflammatory Immune-mediated Chronic Skin Diseases’ Laboratory, IMIBIC/Reina Sofia University Hospital/University of Cordoba, Córdoba, SpainDepartment of Dermatology, Reina Sofia University Hospital, Córdoba, SpainInflammatory Immune-mediated Chronic Skin Diseases’ Laboratory, IMIBIC/Reina Sofia University Hospital/University of Cordoba, Córdoba, SpainDepartment of Dermatology, Reina Sofia University Hospital, Córdoba, SpainInflammatory Immune-mediated Chronic Skin Diseases’ Laboratory, IMIBIC/Reina Sofia University Hospital/University of Cordoba, Córdoba, SpainDepartment of Dermatology, Reina Sofia University Hospital, Córdoba, SpainInflammatory Immune-mediated Chronic Skin Diseases’ Laboratory, IMIBIC/Reina Sofia University Hospital/University of Cordoba, Córdoba, SpainInflammatory Immune-mediated Chronic Skin Diseases’ Laboratory, IMIBIC/Reina Sofia University Hospital/University of Cordoba, Córdoba, SpainInflammatory Immune-mediated Chronic Skin Diseases’ Laboratory, IMIBIC/Reina Sofia University Hospital/University of Cordoba, Córdoba, SpainDepartment of Dermatology, Reina Sofia University Hospital, Córdoba, SpainSchool of Medicine and Nursing, University of Cordoba, Córdoba, SpainInflammatory Immune-mediated Chronic Skin Diseases’ Laboratory, IMIBIC/Reina Sofia University Hospital/University of Cordoba, Córdoba, SpainDepartment of Pharmacology, Reina Sofia University Hospital, Córdoba, SpainDepartment of Pharmacy and Nutrition, Faculty of Biomedical Science and Health, Universidad Europea, Madrid, SpainIntroductionThis scoping review explores the effectiveness of IL-1 pathway inhibitors in managing PSTPIP1-associated inflammatory diseases (PAID). These diseases are marked by abnormal IL-1 pathway activation due to genetic mutations.MethodsOur methodology adhered to a pre-published protocol and involved a thorough search of MEDLINE and EMBASE databases up to February 2022, following the Joanna Briggs Institute Reviewer’s Manual and the PRISMA Extension for Scoping Reviews. The review included studies reporting on IL-1 pathway inhibitor use in PAID patients.ResultsFrom an initial pool of 5,225 articles, 36 studies involving 43 patients were selected. The studies predominantly used observational designs and exhibited diversity in patient demographics, treatment approaches, and outcomes. Anakinra and canakinumab demonstrated promise in treating sterile pyogenic arthritis, pyoderma gangrenosum, and acne (PAPA) and PSTPIP1-associated myeloid-related-proteinemia inflammatory (PAMI) syndromes, with scant data on other syndromes. Notably, there was a paucity of information on the adverse effects of these treatments, necessitating cautious interpretation of their safety profile.ConclusionCurrent evidence on IL-1 pathway inhibitors for PAID is primarily from observational studies and remains limited. Rigorous research with larger patient cohorts is imperative for more definitive conclusions. Collaborative efforts among specialized research centers and international health initiatives are key to advancing this field.https://www.frontiersin.org/articles/10.3389/fimmu.2023.1339337/fullautoinflammatory diseasesPSTPIP1 gene mutationsPAIDinterleukin 1 pathwayIL-1 inhibitorsanakinra
spellingShingle Juan Luis Sanz-Cabanillas
Juan Luis Sanz-Cabanillas
Francisco Gómez-García
Francisco Gómez-García
Pedro Jesús Gómez-Arias
Pedro Jesús Gómez-Arias
Ana Montilla-López
Jesús Gay-Mimbrera
Juan Ruano
Juan Ruano
Juan Ruano
Beatriz Isla-Tejera
Beatriz Isla-Tejera
Esmeralda Parra-Peralbo
Efficacy and safety of anakinra and canakinumab in PSTPIP1-associated inflammatory diseases: a comprehensive scoping review
Frontiers in Immunology
autoinflammatory diseases
PSTPIP1 gene mutations
PAID
interleukin 1 pathway
IL-1 inhibitors
anakinra
title Efficacy and safety of anakinra and canakinumab in PSTPIP1-associated inflammatory diseases: a comprehensive scoping review
title_full Efficacy and safety of anakinra and canakinumab in PSTPIP1-associated inflammatory diseases: a comprehensive scoping review
title_fullStr Efficacy and safety of anakinra and canakinumab in PSTPIP1-associated inflammatory diseases: a comprehensive scoping review
title_full_unstemmed Efficacy and safety of anakinra and canakinumab in PSTPIP1-associated inflammatory diseases: a comprehensive scoping review
title_short Efficacy and safety of anakinra and canakinumab in PSTPIP1-associated inflammatory diseases: a comprehensive scoping review
title_sort efficacy and safety of anakinra and canakinumab in pstpip1 associated inflammatory diseases a comprehensive scoping review
topic autoinflammatory diseases
PSTPIP1 gene mutations
PAID
interleukin 1 pathway
IL-1 inhibitors
anakinra
url https://www.frontiersin.org/articles/10.3389/fimmu.2023.1339337/full
work_keys_str_mv AT juanluissanzcabanillas efficacyandsafetyofanakinraandcanakinumabinpstpip1associatedinflammatorydiseasesacomprehensivescopingreview
AT juanluissanzcabanillas efficacyandsafetyofanakinraandcanakinumabinpstpip1associatedinflammatorydiseasesacomprehensivescopingreview
AT franciscogomezgarcia efficacyandsafetyofanakinraandcanakinumabinpstpip1associatedinflammatorydiseasesacomprehensivescopingreview
AT franciscogomezgarcia efficacyandsafetyofanakinraandcanakinumabinpstpip1associatedinflammatorydiseasesacomprehensivescopingreview
AT pedrojesusgomezarias efficacyandsafetyofanakinraandcanakinumabinpstpip1associatedinflammatorydiseasesacomprehensivescopingreview
AT pedrojesusgomezarias efficacyandsafetyofanakinraandcanakinumabinpstpip1associatedinflammatorydiseasesacomprehensivescopingreview
AT anamontillalopez efficacyandsafetyofanakinraandcanakinumabinpstpip1associatedinflammatorydiseasesacomprehensivescopingreview
AT jesusgaymimbrera efficacyandsafetyofanakinraandcanakinumabinpstpip1associatedinflammatorydiseasesacomprehensivescopingreview
AT juanruano efficacyandsafetyofanakinraandcanakinumabinpstpip1associatedinflammatorydiseasesacomprehensivescopingreview
AT juanruano efficacyandsafetyofanakinraandcanakinumabinpstpip1associatedinflammatorydiseasesacomprehensivescopingreview
AT juanruano efficacyandsafetyofanakinraandcanakinumabinpstpip1associatedinflammatorydiseasesacomprehensivescopingreview
AT beatrizislatejera efficacyandsafetyofanakinraandcanakinumabinpstpip1associatedinflammatorydiseasesacomprehensivescopingreview
AT beatrizislatejera efficacyandsafetyofanakinraandcanakinumabinpstpip1associatedinflammatorydiseasesacomprehensivescopingreview
AT esmeraldaparraperalbo efficacyandsafetyofanakinraandcanakinumabinpstpip1associatedinflammatorydiseasesacomprehensivescopingreview