Untangling the Web of Systemic Autoinflammatory Diseases

Rigante, Donato; Lopalco, Giuseppe; Vitale, A.; Lucherini, Orso Maria; Caso, Francesco; Clemente, Caterina De; Molinaro, Francesco; Messina, Mario; Costa, Luisa; Atteno, Mariangela; Laghi-Pasini, Franco; Lapadula, Giovanni; Gargiulo, Mauro; Iannone, Florenzo; Cantarini, Luca

doi:10.1155/2014/948154

Cited by 26 publications

(14 citation statements)

References 125 publications

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“…Pazár et al have shown that, similar to MSU and CPP, hydroxyapatite crystals can also act as alarmins and trigger NLRP3 inflammasome activation [ 13 ]. The consequent overproduction of IL-1 β seems indirectly mediated by increased levels of other proinflammatory stimuli, including TNF- α or uric acid [ 17 , 18 ]. Pazár et al thus suggested that the IL-1 blockade may be clinically useful in acute HADD manifestations; this hypothesis was clinically confirmed for two of our patients [ 13 ].…”

Section: Discussionmentioning

confidence: 99%

Efficacy of Anakinra for Various Types of Crystal‐Induced Arthritis in Complex Hospitalized Patients: A Case Series and Review of the Literature

Aouba

Deshayes

Frenzel

et al. 2015

Mediators of Inflammation

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Background. There are few data on anakinra use after failure of conventional medications for crystal-induced peripheral arthritis and/or crowned dens syndrome among complex hospitalized patients. Methods. We retrospectively analyzed the outcome of six patients affected with subacute crystal-induced arthritis who had received anakinra in second or third line therapy, including three patients with crowned dens syndrome and three others with gouty arthritis. Patients' comorbidities, reasons for anakinra use and associated drugs, and outcomes were recorded. Results. All patients presented with elevated inflammatory syndrome, systemic symptoms with poly/oligoarthritis. Except for absolute contraindications, all patients were previously treated with full or decreased dose of NSAID, colchicine, and/or glucocorticoids, with unsatisfactory response. All three gouty patients exhibited complete responses in all acute involvements under anakinra within 3 to 5 days, including one of them who needed the reintroduction of colchicine treatment that was previously unsuccessful. Crowned dens syndrome patients, including two with pseudogout and one with subacute hydroxyapatite deposition disease, needed 9 to 11 days to achieve complete response. Tolerance to anakinra was good. Conclusion. In case series of complex hospitalized patients, anakinra showed good activity in crowned dens syndrome and associated crystal-induced peripheral arthritis, with longer treatment duration than in gouty arthritis.

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Section: Discussionmentioning

confidence: 99%

Efficacy of Anakinra for Various Types of Crystal‐Induced Arthritis in Complex Hospitalized Patients: A Case Series and Review of the Literature

Aouba

Deshayes

Frenzel

et al. 2015

Mediators of Inflammation

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“…The safety profile of treatment is of paramount importance when children with autoinflammatory disorders are managed, since it may affect adherence to treatment and overall clinical efficacy [ 23 , 24 , 25 ]. In this regard, in a review paper published in this issue, Bettiol and colleagues [ 26 ] aimed at providing current findings on the efficacy, safety, and tolerability of anti-IL-1 agents anakinra and canakinumab in multifactorial autoinflammatory diseases, focusing on the paediatric setting.…”

Section: Articles In This Special Issuementioning

confidence: 99%

Paediatric Formulation: Design and Development

Lopalco

Denora

2020

IJMS

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The development of medicines designed for children can be challenging since this distinct patient population requires specific needs. A formulation designed for paediatric patients must consider the following aspects: patient population variability; dose flexibility; route of administration; patient compliance; drug and excipient tolerability. The purpose of this Special Issue entitled “Paediatric Formulation: Design and Development” is to provide an update on both state-of-the-art methodology and operational challenges in the design and development of paediatric formulations. It aims at re-evaluating what is needed for more progress in the design and development of age-appropriate treatments for paediatric diseases, focusing on: formulation development; drug delivery design; efficacy, safety, and tolerability of drugs and excipients. This editorial, briefly, summarizes the objects of nine original research and review papers published in this Special Issue.

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“…Features of a typical attack include fever and the potential occurrence of either serositis or arthritis, lasting from 1 to 3 days, resolving spontaneously, but recurring with irregular periodism [8]. Abdominal pain is present in about 90% of patients and is the predominant clinical manifestation in half of them, with a clinical scenery simulating acute abdomen, as in appendicitis, cholecystitis, or urolithiasis [9]. …”

Section: Familial Mediterranean Fevermentioning

confidence: 99%

Lung Involvement in Children with Hereditary Autoinflammatory Disorders

Tarantino

Esposito

Andreozzi

et al. 2016

IJMS

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Short-lived systemic inflammatory reactions arising from disrupted rules in the innate immune system are the operating platforms of hereditary autoinflammatory disorders (HAIDs). Multiple organs may be involved and aseptic inflammation leading to disease-specific phenotypes defines most HAIDs. Lungs are infrequently involved in children with HAIDs: the most common pulmonary manifestation is pleuritis in familial Mediterranean fever (FMF) and tumor necrosis factor receptor-associated periodic syndrome (TRAPS), respectively caused by mutations in the MEFV and TNFRSF1A genes, while interstitial lung disease can be observed in STING-associated vasculopathy with onset in infancy (SAVI), caused by mutations in the TMEM173 gene. The specific pleuropulmonary diseases may range from sub-clinical abnormalities during inflammatory flares of FMF and TRAPS to a severe life-threatening disorder in children with SAVI.

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Untangling the Web of Systemic Autoinflammatory Diseases

Cited by 26 publications

References 125 publications

Efficacy of Anakinra for Various Types of Crystal‐Induced Arthritis in Complex Hospitalized Patients: A Case Series and Review of the Literature

Efficacy of Anakinra for Various Types of Crystal‐Induced Arthritis in Complex Hospitalized Patients: A Case Series and Review of the Literature

Paediatric Formulation: Design and Development

Lung Involvement in Children with Hereditary Autoinflammatory Disorders

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