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Guillaume Moulis, Grégory Pugnet, Nathalie Costedoat-Chalumeau, Alexis Mathian, Gaëlle Leroux, Jonathan Boutémy, Olivier Espitia, Laurence Bouillet, Sabine Berthier, Jean-Baptiste Gaultier, Pierre-Yves Jeandel, Amadou Konaté, Arsène Mékinan, Elisabeth Solau-Gervais, Benjamin Terrier, Daniel Wendling, Fanny Andry, Camille Garnier, Pascal Cathébras, Laurent Arnaud, Aurore Palmaro, Patrice Cacoub, Zahir Amoura, Jean-Charles Piette, Philippe Arlet, Maryse Lapeyre-Mestre, Laurent Sailler
OBJECTIVES: To assess the efficacy and the safety of biologics in a cohort of patients with relapsing polychondritis (RP). METHODS: We conducted a French multicentre retrospective cohort study including patients treated with biologics for RP. Efficacy outcomes were clinical response (partial or complete) and complete response during the first 6 months of exposure, plus daily corticosteroid dose at 6 months. Other outcomes were adverse drug reactions (ADRs), persistence of biologics and factors associated with a response...
March 13, 2018: Annals of the Rheumatic Diseases
Yuan He, Huifeng Liu, Zhi Ji, Binliang Zhu
Purpose: To report a case of surgical interventions for a patient with relapsing polychondritis who presented with late ocular complications. Observations: A 44-year-old male was diagnosed to have relapsing polychondritis on the basis of recurrent acute auricular chondritis, deformity of the ear, saddle nose deformity and painful nasal chondritis, acute ocular inflammation with conjunctivitis, episcleritis and keratouveitis, laryngotracheal chondritis, erythema nodosum in the skin, a history of polyarthritis, and abnormal blood examination findings...
April 2017: American Journal of Ophthalmology Case Reports
Hamdi Taşlı, Hakan Birkent, Mustafa Gerek
Relapsing polychondritis (RP) is a rare autoimmune and inflammatory disease, particularly characterized by recurrent inflammation of the hyaline cartilage. Laryngotracheal involvement in RP is the most serious complication that is observed in 50% of the patients and may lead to a life-threatening condition. The most common cause of death is laryngotracheal stenosis associated with lung infections or severe respiratory insufficiency that may be observed in 10%-50% of the patients. In this study, three RP patients comprising a child with isolated laryngotracheal stenosis have been presented...
June 2017: Turkish Archives of Otorhinolaryngology
Fernando Kemta Lekpa, Xavier Chevalier
Relapsing polychondritis is a severe systemic immune-mediated disease characterized by an episodic and progressive inflammatory condition with progressive destruction of cartilaginous structures. This disease has for nearly a century kept secrets not yet explained. The real incidence and prevalence of this rare disease are unknown. The multiple clinical presentations and episodic nature of relapsing polychondritis cause a significant diagnosis delay. No guidelines for the management of patients with relapsing polychondritis have been validated to date...
2018: Open Access Rheumatology: Research and Reviews
Yi-Yi Xuan, Tian-Fang Li, Lei Zhang, Sheng-Yun Liu
RATIOINALE: Relapsing polychondritis (RP) is a rare and heterogeneous disease complex of unknown origin which basically affects cartilaginous structures, 40% of which accompanied by rheumatic, hematologic, and endocrine disease. Among them, vasculitis is the most common accompanying type and usually presented with positive antineutrophilic cytoplasmic antibody (ANCA). The presence of ANCA could be primary or drug-induced like propylthiouracil (PTU). Central involvement of RP is very rare, and there is almost no report of cerebral vasculopathy manifested as moyamoya...
December 2017: Medicine (Baltimore)
M Anthony Albornoz, Michael Philip Baldassari, Christian Anthony Albornoz, Garen Boghosian
No abstract text is available yet for this article.
January 31, 2018: Journal of Clinical Rheumatology: Practical Reports on Rheumatic & Musculoskeletal Diseases
Chrong-Reen Wang, Sheng-Min Hsu, Ying-Chen Chen, Chung-Liang Ho, Sheen-Yie Fang
A 55-year-old woman with relapsing polychondritis had progressively enlarged right retro-orbital tumor invading the optic nerve, followed by left retrobulbar infiltrating lesions despite prescription of high-dose corticosteroids and pulse methylprednisolone. Repeated histopathologic analyses showed dense collagen fibers with scanty inflammatory cells, consistent with the diagnosis of idiopathic sclerosing orbital inflammation. This disorder has been recognized as a distinct entity with unique clinical features and coexisting rheumatologic disorders, requiring more focused diagnostic strategies and therapeutic regimens...
January 30, 2018: Journal of Clinical Rheumatology: Practical Reports on Rheumatic & Musculoskeletal Diseases
Marcela A Ferrada
Relapsing polychondritis is a rare autoimmune disease that can be fatal. This systemic condition with a predilection for cartilage can inflame the trachea, the distal airways, ear and nose, blood vessels, eyes, kidneys, and brain. If relapsing polychondritis is not diagnosed early and treated effectively, there can be irreversible damage to the cartilage that can lead to tracheal or bronchial collapse, blindness, and deafness. I am a physician and a clinical investigator. I have severe relapsing polychondritis with prominent involvement of my airways...
December 28, 2017: Annals of the American Thoracic Society
William E Monaco, Carey J Field, Thomas H Taylor
We describe a 69-year-old man who presented with a right sternocleidomastoid intramuscular abscess that grew Propionibacterium acnes. Despite initial improvement with antibiotics, he subsequently developed signs and symptoms fulfilling diagnostic criteria for hemophagocytic lymphohistiocytosis (HLH)-fevers, cytopenias, hyperferritinemia, splenomegaly, and low natural killer cell activity. The patient improved spontaneously but at outpatient follow-up complained of erythema over the bridge of his nose as well as bilateral ear redness sparing the earlobes at which point relapsing polychondritis (RP) was diagnosed...
December 27, 2017: Journal of Clinical Rheumatology: Practical Reports on Rheumatic & Musculoskeletal Diseases
Jean-Charles Piette, Jérémie Dion, Nathalie Costedoat-Chalumeau
In this issue of Arthritis Care & Research, the authors of Patient-Perception of Disease-Related Symptoms and Complications in Relapsing Polychondritis report for the first time the patient's view over the disease (1). Thanks go to them for this important step forward. The method used however, carries some limitations, as indicated very honestly by the authors. This article is protected by copyright. All rights reserved.
December 15, 2017: Arthritis Care & Research
Marcela A Ferrada, Peter C Grayson, Shubhasree Banerjee, Keith Sikora, Robert Colbert, Ninet Sinaii, James D Katz
OBJECTIVE: To assess patient-reported symptoms and burden of disease in relapsing polychondritis (RP). METHODS: Patients with RP completed a disease-specific online survey to identify symptoms attributed to illness. Patients were divided into subgroups based upon presence or absence of ear/nose, airway, or joint involvement. Pathway to diagnosis, treatment, and disease-related complications were assessed within each subgroup. RESULTS: Data from 304 respondents were included in this analysis...
December 15, 2017: Arthritis Care & Research
Pauline Baudart, Achille Aouba, Marie Beaufrère, Nicolas Aide
No abstract text is available yet for this article.
December 12, 2017: European Journal of Nuclear Medicine and Molecular Imaging
Stella Pak, Shaina Logemann, Christine Dee, Adam Fershko
Mouth and genital ulcers with inflamed cartilage (MAGIC) syndrome refers to a condition in which features of Behcet's disease (BD) and relapsing polychondritis (RP) occur in the same individual. The existence of MAGIC syndrome suggests a potential common etiology for BD and RP. However, connecting these two diseases and referring to this condition as MAGIC syndrome might have been premature, as there is currently insufficient knowledge on BD and RP. In this critical review, we argue that these two clinical entities could possibly be unique disease processes rather than two ends of the same disease spectrum...
October 4, 2017: Curēus
Francesca Wanda Rossi, Valentina Mercurio, Felice Rivellese, Antonio Lobasso, Letizia Barbieri, Carlo Gabriele Tocchetti, Amato de Paulis, Gianni Marone
No abstract text is available yet for this article.
March 2018: Journal of Clinical Rheumatology: Practical Reports on Rheumatic & Musculoskeletal Diseases
Hayato Kaida, Kazunari Ishii, Soichiro Hanada, Yuji Tohda, Takamichi Murakami
A 78-year-old man had fever, persistent wheezing, and serum C-reactive protein elevation. Malignant lymphoma was suspected because of mediastinal lymph nodes swelling on CT and soluble interleukin 2 receptor elevation. Symmetric F-FDG uptake in the tracheobronchial tree and bilateral auricles was observed on PET/CT. He was finally diagnosed as having relapsing polychondritis by auricular cartilage biopsy. F-FDG PET/CT may have crucial role in evaluating the extent of inflammation and deciding the biopsy site of relapsing polychondritis...
January 2018: Clinical Nuclear Medicine
Laurent Dormoy, Annette Belin, Fabien Labombarda
Relapsing polychondritis is a rare multisystem autoimmune disease characterized by recurrent inflammation and destruction of different cartilaginous and proteoglycan-rich structures like cardiovascular system. We reported a case of heart transplantation in a 20-year-old man having a relapsing polychondritis with severe multiple cardiovascular involvement. The immunosuppressive treatment used to prevent graft rejection allowed to turn out the inflammatory disease. Cardiac transplantation may be considered as last resort treatment option in relapsing polychondritis with intractable extensive heart lesions...
September 2017: Progress in Transplantation
Renata Dal-Prá Ducci, Francisco Manoel Branco Germiniani, Letícia Elizabeth Augustin Czecko, Eduardo S Paiva, Hélio Afonso Ghizoni Teive
No abstract text is available yet for this article.
November 2017: Revista Brasileira de Reumatologia
Wei Liu, Hongli Jiang, Han Jing, Bing Mao
INTRODUCTION: Fever of unknown origin (FUO) is a common initial presentation leading to a diagnostic challenge. PATIENT CONCERNS: A 3-month history of moderate-to-high fever was reported in an otherwise healthy 54-year-old man. Enhanced computed tomography (CT) scans of his chest showed a remarkable progressive enlargement of bilateral cervical, supraclavicular, hilar, and mediastinal lymph nodes within 2 weeks. Bronchofibroscopy manifested obvious luminal stenosis with swelling, thick pale mucosa, and disappearing of structures of trachea cricoid cartilage, followed by a 18F-fluorodeoxyglucose positron-emission tomography-computed tomography (18F-FDG PET/CT) with intense symmetric FDG uptake in larynx, tracheobronchial tree, and hilar, mediastinal, and axillary lymph nodes being demonstrated...
November 2017: Medicine (Baltimore)
Fatma Canbay, Aydın Yılmaz, Ayperi Öztürk, Zafer Aktaş, Ebru Tatcı
Relapsing polychondritis (RP) is a rare autoimmune disease, characterized by episodic inflammatory attacks on cartilaginous tissue. Elastic cartilage of the ears and nose, hyaline cartilage of the peripheral joints, vertebral fibrocartilage, tracheobronchial cartilage, and tissues rich in proteoglycan such as those in the eyes, heart, blood vessels, and inner ear are more likely to be affected. A 35-year-old male presented with complaints of hoarseness, tinnitus and dyspnea for 19 years, with a history of several diagnostic and therapeutic interventions for laryngeal and respiratory tract...
September 2017: Tüberküloz Ve Toraks
Jack Kingdon, Joseph Roscamp, Shirish Sangle, David D'Cruz
Relapsing polychondritis (RPC) is a rare autoimmune rheumatic disorder that is traditionally classified as a systemic vasculitis. It is characterized by inflammation of cartilage, and typical presenting features include chondritis of the nasal bridge, auricular chondritis, ocular inflammation and involvement of the bronchial tree. Its rarity often leads to considerable delay in establishing a diagnosis and poses a significant management challenge to clinicians, as no conventional guidelines exist. This review summarizes the clinical features of RPC and provides guidance for rheumatologists on making the diagnosis and assessing organ involvement...
November 6, 2017: Rheumatology
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