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Preliminary data revealing efficacy of Streptococcus salivarius K12 (SSK12) in Periodic Fever, Aphthous stomatitis, Pharyngitis, and cervical Adenitis (PFAPA) syndrome: A multicenter study from the AIDA Network PFAPA syndrome registry

OBJECTIVE: To evaluate the potential role of Streptococcus salivarius K12 (SSK12) in controlling febrile flares in patients with Periodic Fever, Aphthous stomatitis, Pharyngitis, and cervical Adenitis (PFAPA) syndrome. Further aims were to assess the impact of SSK12 on (i) flare duration, (ii) varia...

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Autores principales: La Torre, Francesco, Sota, Jurgen, Insalaco, Antonella, Conti, Giovanni, Del Giudice, Emanuela, Lubrano, Riccardo, Breda, Luciana, Maggio, Maria Cristina, Civino, Adele, Mastrorilli, Violetta, Loconte, Roberta, Natale, Marco Francesco, Celani, Camilla, Romeo, Mery, Patroniti, Serena, Gentile, Cristina, Vitale, Antonio, Caggiano, Valeria, Gaggiano, Carla, Diomeda, Federico, Cattalini, Marco, Lopalco, Giuseppe, Emmi, Giacomo, Parronchi, Paola, Gentileschi, Stefano, Cardinale, Fabio, Aragona, Emma, Shahram, Farhad, Marino, Achille, Barone, Patrizia, Moscheo, Carla, Ozkiziltas, Burcugul, Carubbi, Francesco, Alahmed, Ohoud, Iezzi, Ludovica, Ogunjimi, Benson, Mauro, Angela, Tarsia, Maria, Mahmoud, Ayman Abdel-Monem Ahmed, Giardini, Henrique Ayres Mayrink, Sfikakis, Petros P., Laskari, Katerina, Więsik-Szewczyk, Ewa, Hernández-Rodríguez, José, Frediani, Bruno, Gómez-Caverzaschi, Verónica, Tufan, Abdurrahman, Almaghlouth, Ibrahim A., Balistreri, Alberto, Ragab, Gaafar, Fabiani, Claudia, Cantarini, Luca, Rigante, Donato
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9977796/
https://www.ncbi.nlm.nih.gov/pubmed/36873863
http://dx.doi.org/10.3389/fmed.2023.1105605
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author La Torre, Francesco
Sota, Jurgen
Insalaco, Antonella
Conti, Giovanni
Del Giudice, Emanuela
Lubrano, Riccardo
Breda, Luciana
Maggio, Maria Cristina
Civino, Adele
Mastrorilli, Violetta
Loconte, Roberta
Natale, Marco Francesco
Celani, Camilla
Romeo, Mery
Patroniti, Serena
Gentile, Cristina
Vitale, Antonio
Caggiano, Valeria
Gaggiano, Carla
Diomeda, Federico
Cattalini, Marco
Lopalco, Giuseppe
Emmi, Giacomo
Parronchi, Paola
Gentileschi, Stefano
Cardinale, Fabio
Aragona, Emma
Shahram, Farhad
Marino, Achille
Barone, Patrizia
Moscheo, Carla
Ozkiziltas, Burcugul
Carubbi, Francesco
Alahmed, Ohoud
Iezzi, Ludovica
Ogunjimi, Benson
Mauro, Angela
Tarsia, Maria
Mahmoud, Ayman Abdel-Monem Ahmed
Giardini, Henrique Ayres Mayrink
Sfikakis, Petros P.
Laskari, Katerina
Więsik-Szewczyk, Ewa
Hernández-Rodríguez, José
Frediani, Bruno
Gómez-Caverzaschi, Verónica
Tufan, Abdurrahman
Almaghlouth, Ibrahim A.
Balistreri, Alberto
Ragab, Gaafar
Fabiani, Claudia
Cantarini, Luca
Rigante, Donato
author_facet La Torre, Francesco
Sota, Jurgen
Insalaco, Antonella
Conti, Giovanni
Del Giudice, Emanuela
Lubrano, Riccardo
Breda, Luciana
Maggio, Maria Cristina
Civino, Adele
Mastrorilli, Violetta
Loconte, Roberta
Natale, Marco Francesco
Celani, Camilla
Romeo, Mery
Patroniti, Serena
Gentile, Cristina
Vitale, Antonio
Caggiano, Valeria
Gaggiano, Carla
Diomeda, Federico
Cattalini, Marco
Lopalco, Giuseppe
Emmi, Giacomo
Parronchi, Paola
Gentileschi, Stefano
Cardinale, Fabio
Aragona, Emma
Shahram, Farhad
Marino, Achille
Barone, Patrizia
Moscheo, Carla
Ozkiziltas, Burcugul
Carubbi, Francesco
Alahmed, Ohoud
Iezzi, Ludovica
Ogunjimi, Benson
Mauro, Angela
Tarsia, Maria
Mahmoud, Ayman Abdel-Monem Ahmed
Giardini, Henrique Ayres Mayrink
Sfikakis, Petros P.
Laskari, Katerina
Więsik-Szewczyk, Ewa
Hernández-Rodríguez, José
Frediani, Bruno
Gómez-Caverzaschi, Verónica
Tufan, Abdurrahman
Almaghlouth, Ibrahim A.
Balistreri, Alberto
Ragab, Gaafar
Fabiani, Claudia
Cantarini, Luca
Rigante, Donato
author_sort La Torre, Francesco
collection PubMed
description OBJECTIVE: To evaluate the potential role of Streptococcus salivarius K12 (SSK12) in controlling febrile flares in patients with Periodic Fever, Aphthous stomatitis, Pharyngitis, and cervical Adenitis (PFAPA) syndrome. Further aims were to assess the impact of SSK12 on (i) flare duration, (ii) variation in the degree of the highest body temperature during flares, (iii) steroid-sparing effect, and (iv) change of PFAPA accompanying symptoms before and after SSK12 introduction. PATIENTS AND METHODS: The medical charts from 85 pediatric patients with PFAPA syndrome (49 males and 36 females) enrolled in the AIDA registry and treated with SSK12 for a median period of 6.00 ± 7.00 months in the period between September 2017 and May 2022 were examined. Children recruited had a median time of disease duration of 19.00 ± 28.00 months. RESULTS: The number of febrile flares significantly decreased comparing the 12 months before [median (IQR), 13.00 (6.00)] and after SSK12 initiation [median (IQR), 5.50 (8.00), p < 0.001]. The duration of fever was significantly reduced from 4.00 (2.00) days to 2.00 (2.00) days [p < 0.001]. Similarly, the highest temperature in°C was found significantly lower in the last follow-up assessment [median (IQR), 39.00 (1.00)] compared to the period prior to SSK12 start [median (IQR), 40.00 (1.00), p < 0.001]. Steroid load (mg/year) of betamethasone (or any equivalent steroid) significantly decreased between 12 months before treatment with SSK12 [median (IQR), 5.00 (8.00) mg/year] and the last follow-up visit [median (IQR), 2.00 (4.00) mg/year, p < 0.001]. The number of patients experiencing symptoms including pharyngitis/tonsillitis (p < 0.001), oral aphthae (p < 0.001) and cervical lymphadenopathy (p < 0.001) significantly decreased following SSK12. CONCLUSION: SSK12 prophylaxis given for at least 6.00 months was found to reduce febrile flares of PFAPA syndrome: in particular, it halved the total number per year of fever flares, shortened the duration of the single febrile episode, lowered body temperature by 1°C in the febrile flare, provided a steroid-sparing effect, and significantly reduced the accompanying symptoms related to the syndrome.
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spelling pubmed-99777962023-03-03 Preliminary data revealing efficacy of Streptococcus salivarius K12 (SSK12) in Periodic Fever, Aphthous stomatitis, Pharyngitis, and cervical Adenitis (PFAPA) syndrome: A multicenter study from the AIDA Network PFAPA syndrome registry La Torre, Francesco Sota, Jurgen Insalaco, Antonella Conti, Giovanni Del Giudice, Emanuela Lubrano, Riccardo Breda, Luciana Maggio, Maria Cristina Civino, Adele Mastrorilli, Violetta Loconte, Roberta Natale, Marco Francesco Celani, Camilla Romeo, Mery Patroniti, Serena Gentile, Cristina Vitale, Antonio Caggiano, Valeria Gaggiano, Carla Diomeda, Federico Cattalini, Marco Lopalco, Giuseppe Emmi, Giacomo Parronchi, Paola Gentileschi, Stefano Cardinale, Fabio Aragona, Emma Shahram, Farhad Marino, Achille Barone, Patrizia Moscheo, Carla Ozkiziltas, Burcugul Carubbi, Francesco Alahmed, Ohoud Iezzi, Ludovica Ogunjimi, Benson Mauro, Angela Tarsia, Maria Mahmoud, Ayman Abdel-Monem Ahmed Giardini, Henrique Ayres Mayrink Sfikakis, Petros P. Laskari, Katerina Więsik-Szewczyk, Ewa Hernández-Rodríguez, José Frediani, Bruno Gómez-Caverzaschi, Verónica Tufan, Abdurrahman Almaghlouth, Ibrahim A. Balistreri, Alberto Ragab, Gaafar Fabiani, Claudia Cantarini, Luca Rigante, Donato Front Med (Lausanne) Medicine OBJECTIVE: To evaluate the potential role of Streptococcus salivarius K12 (SSK12) in controlling febrile flares in patients with Periodic Fever, Aphthous stomatitis, Pharyngitis, and cervical Adenitis (PFAPA) syndrome. Further aims were to assess the impact of SSK12 on (i) flare duration, (ii) variation in the degree of the highest body temperature during flares, (iii) steroid-sparing effect, and (iv) change of PFAPA accompanying symptoms before and after SSK12 introduction. PATIENTS AND METHODS: The medical charts from 85 pediatric patients with PFAPA syndrome (49 males and 36 females) enrolled in the AIDA registry and treated with SSK12 for a median period of 6.00 ± 7.00 months in the period between September 2017 and May 2022 were examined. Children recruited had a median time of disease duration of 19.00 ± 28.00 months. RESULTS: The number of febrile flares significantly decreased comparing the 12 months before [median (IQR), 13.00 (6.00)] and after SSK12 initiation [median (IQR), 5.50 (8.00), p < 0.001]. The duration of fever was significantly reduced from 4.00 (2.00) days to 2.00 (2.00) days [p < 0.001]. Similarly, the highest temperature in°C was found significantly lower in the last follow-up assessment [median (IQR), 39.00 (1.00)] compared to the period prior to SSK12 start [median (IQR), 40.00 (1.00), p < 0.001]. Steroid load (mg/year) of betamethasone (or any equivalent steroid) significantly decreased between 12 months before treatment with SSK12 [median (IQR), 5.00 (8.00) mg/year] and the last follow-up visit [median (IQR), 2.00 (4.00) mg/year, p < 0.001]. The number of patients experiencing symptoms including pharyngitis/tonsillitis (p < 0.001), oral aphthae (p < 0.001) and cervical lymphadenopathy (p < 0.001) significantly decreased following SSK12. CONCLUSION: SSK12 prophylaxis given for at least 6.00 months was found to reduce febrile flares of PFAPA syndrome: in particular, it halved the total number per year of fever flares, shortened the duration of the single febrile episode, lowered body temperature by 1°C in the febrile flare, provided a steroid-sparing effect, and significantly reduced the accompanying symptoms related to the syndrome. Frontiers Media S.A. 2023-02-16 /pmc/articles/PMC9977796/ /pubmed/36873863 http://dx.doi.org/10.3389/fmed.2023.1105605 Text en Copyright © 2023 La Torre, Sota, Insalaco, Conti, Del Giudice, Lubrano, Breda, Maggio, Civino, Mastrorilli, Loconte, Natale, Celani, Romeo, Patroniti, Gentile, Vitale, Caggiano, Gaggiano, Diomeda, Cattalini, Lopalco, Emmi, Parronchi, Gentileschi, Cardinale, Aragona, Shahram, Marino, Barone, Moscheo, Ozkiziltas, Carubbi, Alahmed, Iezzi, Ogunjimi, Mauro, Tarsia, Mahmoud, Giardini, Sfikakis, Laskari, Więsik-Szewczyk, Hernández-Rodríguez, Frediani, Gómez-Caverzaschi, Tufan, Almaghlouth, Balistreri, Ragab, Fabiani, Cantarini and Rigante. https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Medicine
La Torre, Francesco
Sota, Jurgen
Insalaco, Antonella
Conti, Giovanni
Del Giudice, Emanuela
Lubrano, Riccardo
Breda, Luciana
Maggio, Maria Cristina
Civino, Adele
Mastrorilli, Violetta
Loconte, Roberta
Natale, Marco Francesco
Celani, Camilla
Romeo, Mery
Patroniti, Serena
Gentile, Cristina
Vitale, Antonio
Caggiano, Valeria
Gaggiano, Carla
Diomeda, Federico
Cattalini, Marco
Lopalco, Giuseppe
Emmi, Giacomo
Parronchi, Paola
Gentileschi, Stefano
Cardinale, Fabio
Aragona, Emma
Shahram, Farhad
Marino, Achille
Barone, Patrizia
Moscheo, Carla
Ozkiziltas, Burcugul
Carubbi, Francesco
Alahmed, Ohoud
Iezzi, Ludovica
Ogunjimi, Benson
Mauro, Angela
Tarsia, Maria
Mahmoud, Ayman Abdel-Monem Ahmed
Giardini, Henrique Ayres Mayrink
Sfikakis, Petros P.
Laskari, Katerina
Więsik-Szewczyk, Ewa
Hernández-Rodríguez, José
Frediani, Bruno
Gómez-Caverzaschi, Verónica
Tufan, Abdurrahman
Almaghlouth, Ibrahim A.
Balistreri, Alberto
Ragab, Gaafar
Fabiani, Claudia
Cantarini, Luca
Rigante, Donato
Preliminary data revealing efficacy of Streptococcus salivarius K12 (SSK12) in Periodic Fever, Aphthous stomatitis, Pharyngitis, and cervical Adenitis (PFAPA) syndrome: A multicenter study from the AIDA Network PFAPA syndrome registry
title Preliminary data revealing efficacy of Streptococcus salivarius K12 (SSK12) in Periodic Fever, Aphthous stomatitis, Pharyngitis, and cervical Adenitis (PFAPA) syndrome: A multicenter study from the AIDA Network PFAPA syndrome registry
title_full Preliminary data revealing efficacy of Streptococcus salivarius K12 (SSK12) in Periodic Fever, Aphthous stomatitis, Pharyngitis, and cervical Adenitis (PFAPA) syndrome: A multicenter study from the AIDA Network PFAPA syndrome registry
title_fullStr Preliminary data revealing efficacy of Streptococcus salivarius K12 (SSK12) in Periodic Fever, Aphthous stomatitis, Pharyngitis, and cervical Adenitis (PFAPA) syndrome: A multicenter study from the AIDA Network PFAPA syndrome registry
title_full_unstemmed Preliminary data revealing efficacy of Streptococcus salivarius K12 (SSK12) in Periodic Fever, Aphthous stomatitis, Pharyngitis, and cervical Adenitis (PFAPA) syndrome: A multicenter study from the AIDA Network PFAPA syndrome registry
title_short Preliminary data revealing efficacy of Streptococcus salivarius K12 (SSK12) in Periodic Fever, Aphthous stomatitis, Pharyngitis, and cervical Adenitis (PFAPA) syndrome: A multicenter study from the AIDA Network PFAPA syndrome registry
title_sort preliminary data revealing efficacy of streptococcus salivarius k12 (ssk12) in periodic fever, aphthous stomatitis, pharyngitis, and cervical adenitis (pfapa) syndrome: a multicenter study from the aida network pfapa syndrome registry
topic Medicine
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9977796/
https://www.ncbi.nlm.nih.gov/pubmed/36873863
http://dx.doi.org/10.3389/fmed.2023.1105605
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AT balistrerialberto preliminarydatarevealingefficacyofstreptococcussalivariusk12ssk12inperiodicfeveraphthousstomatitispharyngitisandcervicaladenitispfapasyndromeamulticenterstudyfromtheaidanetworkpfapasyndromeregistry
AT ragabgaafar preliminarydatarevealingefficacyofstreptococcussalivariusk12ssk12inperiodicfeveraphthousstomatitispharyngitisandcervicaladenitispfapasyndromeamulticenterstudyfromtheaidanetworkpfapasyndromeregistry
AT fabianiclaudia preliminarydatarevealingefficacyofstreptococcussalivariusk12ssk12inperiodicfeveraphthousstomatitispharyngitisandcervicaladenitispfapasyndromeamulticenterstudyfromtheaidanetworkpfapasyndromeregistry
AT cantariniluca preliminarydatarevealingefficacyofstreptococcussalivariusk12ssk12inperiodicfeveraphthousstomatitispharyngitisandcervicaladenitispfapasyndromeamulticenterstudyfromtheaidanetworkpfapasyndromeregistry
AT rigantedonato preliminarydatarevealingefficacyofstreptococcussalivariusk12ssk12inperiodicfeveraphthousstomatitispharyngitisandcervicaladenitispfapasyndromeamulticenterstudyfromtheaidanetworkpfapasyndromeregistry