Literature DB >> 25036278

CANDLE syndrome: a recently described autoinflammatory syndrome.

Özlem Tüfekçi1, ŞebnemYilmaz Bengoa, Tuba Hilkay Karapinar, Eda Büke Ataseven, Gülersu İrken, Hale Ören.   

Abstract

CANDLE syndrome (chronic atypical neutrophilic dermatosis with lipodystrophy and elevated temperature) is a recently described autoinflammatory syndrome characterized by early onset, recurrent fever, skin lesions, and multisystemic inflammatory manifestations. Most of the patients have been shown to have mutation in PSMB8 gene. Herein, we report a 2-year-old patient with young onset recurrent fever, atypical facies, widespread skin lesions, generalized lymphadenopathy, hepatosplenomegaly, joint contractures, hypertrglyceridemia, lipodystrophy, and autoimmune hemolytic anemia. Clinical features together with the skin biopsy findings were consistent with the CANDLE syndrome. The pathogenesis and treatment of this syndrome have not been fully understood. Increased awareness of this recently described syndrome may lead to recognition of new cases and better understanding of its pathogenesis which in turn may help for development of an effective treatment.

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Year:  2015        PMID: 25036278     DOI: 10.1097/MPH.0000000000000212

Source DB:  PubMed          Journal:  J Pediatr Hematol Oncol        ISSN: 1077-4114            Impact factor:   1.289


  6 in total

Review 1.  Dermatologic Manifestations of Monogenic Autoinflammatory Diseases.

Authors:  Kyawt Win Shwin; Chyi-Chia Richard Lee; Raphaela Goldbach-Mansky
Journal:  Dermatol Clin       Date:  2017-01       Impact factor: 3.478

2.  Novel proteasome assembly chaperone mutations in PSMG2/PAC2 cause the autoinflammatory interferonopathy CANDLE/PRAAS4.

Authors:  Adriana A de Jesus; Anja Brehm; Rachel VanTries; Pascal Pillet; Anne-Sophie Parentelli; Gina A Montealegre Sanchez; Zuoming Deng; Isabelle Koné Paut; Raphaela Goldbach-Mansky; Elke Krüger
Journal:  J Allergy Clin Immunol       Date:  2019-01-18       Impact factor: 10.793

3.  CANDLE syndrome: chronic atypical neutrophilic dermatosis with lipodystrophy and elevated temperature-a rare case with a novel mutation.

Authors:  Miria Paula V Cavalcante; Juliana B Brunelli; Clarissa C Miranda; Glaucia V Novak; Louise Malle; Nadia E Aikawa; Adriana A Jesus; Clovis Artur Silva
Journal:  Eur J Pediatr       Date:  2015-11-14       Impact factor: 3.183

Review 4.  CANDLE Syndrome As a Paradigm of Proteasome-Related Autoinflammation.

Authors:  Antonio Torrelo
Journal:  Front Immunol       Date:  2017-08-09       Impact factor: 7.561

Review 5.  Monogenic Autoinflammatory Diseases with Mendelian Inheritance: Genes, Mutations, and Genotype/Phenotype Correlations.

Authors:  Davide Martorana; Francesco Bonatti; Paola Mozzoni; Augusto Vaglio; Antonio Percesepe
Journal:  Front Immunol       Date:  2017-04-03       Impact factor: 7.561

6.  Molecular Insight Into the IRE1α-Mediated Type I Interferon Response Induced by Proteasome Impairment in Myeloid Cells of the Brain.

Authors:  Maja Studencka-Turski; Gonca Çetin; Heike Junker; Frédéric Ebstein; Elke Krüger
Journal:  Front Immunol       Date:  2019-12-18       Impact factor: 7.561

  6 in total

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