Literature DB >> 21537926

PFAPA syndrome in siblings. Is there a genetic background?

Pilar Antón-Martín1, Roberto Ortiz Movilla, Sara Guillén Martín, Luis M Allende, M Teresa Cuesta Rubio, M Fernanda López González, José Tomás Ramos Amador.   

Abstract

UNLABELLED: "PFAPA syndrome" is an autoinflammatory entity composed of periodic fever, aphthous stomatitis, pharyngitis, and cervical adenitis. There have been many reports of children with the disease, but only occasionally have been described in siblings, and no specific genetic mutation has been determined yet. Corticosteroids are the mainstay in the treatment of the acute attacks. The role of surgery in long-term follow-up (tonsillectomy with or without adenoidectomy) is controversial. We report two brothers affected with the syndrome, in whom corticosteroids as the only treatment led to an improvement. A genetic work-up was performed, making very unlikely other possible syndromes of recurrent fever.
CONCLUSION: PFAPA syndrome is the most common recurrent periodic fever disorder described in childhood. Its genetic background has not been elucidated yet. Our contribution with two siblings affected with PFAPA syndrome further support the genetic basis for the entity.

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Year:  2011        PMID: 21537926     DOI: 10.1007/s00431-011-1479-5

Source DB:  PubMed          Journal:  Eur J Pediatr        ISSN: 0340-6199            Impact factor:   3.860


  22 in total

1.  Two siblings with periodic fever, aphthous stomatitis, pharyngitis, adenitis (PFAPA) syndrome.

Authors:  Isabel Cristina Ramos Melo Sampaio; Maria João Rodrigo; José Gonçalo Duque Pereira Monteiro Marques
Journal:  Pediatr Infect Dis J       Date:  2009-03       Impact factor: 2.129

2.  Periodic fever syndrome in children.

Authors:  K T Thomas; H M Feder; A R Lawton; K M Edwards
Journal:  J Pediatr       Date:  1999-07       Impact factor: 4.406

3.  PFAPA syndrome.

Authors:  G S Marshall; K M Edwards; A R Lawton
Journal:  Pediatr Infect Dis J       Date:  1989-09       Impact factor: 2.129

Review 4.  Distinguishing among prolonged, recurrent, and periodic fever syndromes: approach of a pediatric infectious diseases subspecialist.

Authors:  Sarah S Long
Journal:  Pediatr Clin North Am       Date:  2005-06       Impact factor: 3.278

Review 5.  Periodic fever, apthous stomatitis, pharyngitis and adenitis syndrome.

Authors:  Roberta Caorsi; Maria Antonietta Pelagatti; Silvia Federici; Martina Finetti; Alberto Martini; Marco Gattorno
Journal:  Curr Opin Rheumatol       Date:  2010-09       Impact factor: 5.006

Review 6.  Pediatric hereditary autoinflammatory syndromes.

Authors:  Adriana Almeida Jesus; João Bosco Oliveira; Maria Odete Esteves Hilário; Maria Teresa R A Terreri; Erika Fujihira; Mariana Watase; Magda Carneiro-Sampaio; Clovis Artur Almeida Silva
Journal:  J Pediatr (Rio J)       Date:  2010 Sep-Oct       Impact factor: 2.197

7.  PFAPA syndrome is not a sporadic disease.

Authors:  Marie Cochard; Johanna Clet; Lan Le; Pascal Pillet; Xavier Onrubia; Thierry Guéron; Mohammed Faouzi; Michaël Hofer
Journal:  Rheumatology (Oxford)       Date:  2010-07-02       Impact factor: 7.580

8.  Syndrome of periodic fever, aphthous stomatitis, pharyngitis, and adenitis (PFAPA) in siblings.

Authors:  Patricia M Valenzuela; Daniela Majerson; Jose L Tapia; Eduardo Talesnik
Journal:  Clin Rheumatol       Date:  2009-07-11       Impact factor: 2.980

9.  MEFV, TNF1rA, CARD15 and NLRP3 mutation analysis in PFAPA.

Authors:  Efrat Dagan; Ruth Gershoni-Baruch; Ihab Khatib; Adi Mori; Riva Brik
Journal:  Rheumatol Int       Date:  2009-07-05       Impact factor: 2.631

10.  C-reactive protein in the periodic fever, aphthous stomatitis, pharyngitis and cervical adenitis (PFAPA) syndrome.

Authors:  Jostein A Førsvoll; Knut Oymar
Journal:  Acta Paediatr       Date:  2007-11       Impact factor: 2.299

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  8 in total

Review 1.  Periodic Fever, Aphthous Stomatitis, Pharyngitis, and Cervical Adenitis (PFAPA) Syndrome: a Review of the Pathogenesis.

Authors:  Katerina Theodoropoulou; Federica Vanoni; Michaël Hofer
Journal:  Curr Rheumatol Rep       Date:  2016-04       Impact factor: 4.592

2.  NLRP3 gene variants and serum NLRP3 levels in periodic fever, aphthous stomatitis, pharyngitis, and adenitis (PFAPA) syndrome.

Authors:  Damla Kaynak; Mehmet Yildiz; Sezgin Sahin; Fatih Haslak; Aybuke Gunalp; Amra Adrovic; Kenan Barut; Mehmet Guven Gunver; Ozgur Kasapcopur; Selcuk Dasdemir
Journal:  Clin Rheumatol       Date:  2022-09-10       Impact factor: 3.650

3.  Family History in Periodic Fever, Aphthous Stomatitis, Pharyngitis, Adenitis (PFAPA) Syndrome.

Authors:  Kalpana Manthiram; Emily Nesbitt; Thomas Morgan; Kathryn M Edwards
Journal:  Pediatrics       Date:  2016-09       Impact factor: 7.124

4.  Profile of inflammatory mediators in tonsils of patients with periodic fever, aphthous stomatitis, pharyngitis, and cervical adenitis (PFAPA) syndrome.

Authors:  Patricia M Valenzuela; Andrea Araya; Claudio I Pérez; Ximena Maul; Carolina Serrano; Constanza Beltrán; Paul R Harris; Eduardo Talesnik
Journal:  Clin Rheumatol       Date:  2013-07-23       Impact factor: 2.980

Review 5.  Basic Characteristics of Adults with Periodic Fever, Aphthous Stomatitis, Pharyngitis, and Adenopathy Syndrome in Comparison with the Typical Pediatric Expression of Disease.

Authors:  Marco Cattalini; Martina Soliani; Donato Rigante; Giuseppe Lopalco; Florenzo Iannone; Mauro Galeazzi; Luca Cantarini
Journal:  Mediators Inflamm       Date:  2015-08-18       Impact factor: 4.711

6.  Clinical features and genetic background of the periodic Fever syndrome with aphthous stomatitis, pharyngitis, and adenitis: a single center longitudinal study of 81 patients.

Authors:  Daša Perko; Maruša Debeljak; Nataša Toplak; Tadej Avčin
Journal:  Mediators Inflamm       Date:  2015-03-04       Impact factor: 4.711

7.  Synchronous disease onset and flares in siblings with PFAPA.

Authors:  Kristen L Dammeyer; Amanda Schneider; Max M April; Philip J Kahn
Journal:  Pediatr Rheumatol Online J       Date:  2022-10-05       Impact factor: 3.413

Review 8.  Clinical genetic testing of periodic fever syndromes.

Authors:  Annalisa Marcuzzi; Elisa Piscianz; Giulio Kleiner; Alberto Tommasini; Giovanni Maria Severini; Lorenzo Monasta; Sergio Crovella
Journal:  Biomed Res Int       Date:  2013-01-01       Impact factor: 3.411

  8 in total

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