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Ocul Immunol Inflamm 2022 Jun 13:1-4. Epub 2022 Jun 13.

Department of Rheumatology, Nizams Institute of Medical Services, Hyderabad, India.

Purpose: To report a challenging case of relapsing polychondritis with bilateral diffuse scleritis, with 17-year follow-up.

Methods: Case report.

Results: A 36-year-old female presented 17 years ago with bilateral diffuse scleritis and peripheral corneal infiltrates. Read More

Pediatr Dermatol 2022 Jun 11. Epub 2022 Jun 11.

Dermatology Unit and Genodermatosis Unit, Genetics and Rare Diseases Research Division, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy.

Cutaneous leishmaniasis (CL) is the most frequent form of leishmaniasis. The auricle is an extremely rare site for CL in the Old World. Auricular CL may be mistaken for other entities, such as relapsing polychondritis (RP). Read More

BMC Pulm Med 2022 Jun 8;22(1):222. Epub 2022 Jun 8.

Department of Respiratory and Critical Care Medicine, Beijing Institute of Respiratory Medicine and Beijing Chao-Yang Hospital, Capital Medical University, 8 Gongren Tiyuchang Nanlua, Chaoyang District, Beijing, 100020, China.

Background: Relapsing polychondritis (RP) patients with tracheal cartilage involvement are different from other patients. The objective of this study was to compare the clinical features and disease patterns between a respiratory involvement subgroup and a non-respiratory involvement subgroup according to chest computed tomography.

Method: We performed a retrospective cohort study collecting RP patients hospitalized at the Beijing Chao-Yang Hospital between January 2012 and August 2021. Read More

Am J Forensic Med Pathol 2022 May 30. Epub 2022 May 30.

Maricopa County Medical Examiner's Office, Phoenix, AZ.

Abstract: Relapsing polychondritis (RP) is a rare inflammatory disease process that affects cartilaginous tissues throughout the body. Although the pathogenesis remains unknown, RP is thought to be an autoimmune disorder in which host immune cells are conditioned to attack the body's cartilage, such as the ears, nose, eyes, joints, and airways, resulting in inflammation and destruction of otherwise healthy tissues. In rare and unusual cases, neurological involvement has been described. Read More

Arthritis Res Ther 2022 May 30;24(1):127. Epub 2022 May 30.

Department of Rheumatology and Clinical Immunology, Graduate School of Medicine, Kyoto University, Kyoto, Japan.

Background: Although the survival rates of patients with relapsing polychondritis (RP) have increased remarkably, the high recurrence rate remains a significant concern for physicians and patients. This retrospective study aimed to investigate the risk factors for RP recurrence.

Methods: Patients with RP who presented to Kyoto University Hospital from January 2000 to March 2020 and fulfilled Damiani's classification criteria were included. Read More

Br J Haematol 2022 May 20. Epub 2022 May 20.

Département de Médecine Interne, Hôpital Bichat, Université Paris Cité, Assistance Publique Hôpitaux de Paris, Paris, France.

Eur Rev Med Pharmacol Sci 2022 May;26(9):3061

Institute for Health Sciences from Federal University of Bahia, Salvador, BA, Brazil.

Orphanet J Rare Dis 2022 05 10;17(1):101. Epub 2022 May 10.

Laboratory for Statistical and Translational Genetics, RIKEN Center for Integrative Medical Sciences, Kanagawa, 230-0045, Japan.

Background: Relapsing polychondritis (RP) is a rare inflammatory disease characterized by recurrent inflammation and destruction of cartilaginous tissues. RP has characteristics of autoimmune disease and some reports have noted co-occurrence with autoimmune thyroid disease (AITD), consisting of Graves' disease (GD) and Hashimoto thyroiditis (HT). However, there have been no detailed studies on the co-occurrence of RP and AITD. Read More

Acta Neurol Belg 2022 May 5. Epub 2022 May 5.

Department of Neurology, Samsung Medical Center, Sungkyunkwan University School of Medicine, 81 Irwon-ro, Gangnam-gu, Seoul, 06351, Republic of Korea.

Joint Bone Spine 2022 Apr 28;89(4):105387. Epub 2022 Apr 28.

Division of Allergy, Immunology and Rheumatology, Department of Medicine, Taipei Veterans General Hospital, Taipei, Taiwan; Faculty of Medicine, National Yang Ming Chiao Tung University, Taipei, Taiwan; Division of Allergy, Immunology and Rheumatology, Department of Internal Medicine, School of Medicine, College of Medicine, Taipei Medical University, Taipei Veterans General Hospital. No. 201, Sec.2, Shih-Pai Rd, Taipei 11217, Taiwan. Electronic address:

Zhonghua Nei Ke Za Zhi 2022 May;61(5):525-530

Department of Rheumatology and Clinical Immunology, Peking Union Medical College Hospital, Peking Union Medical College, Chinese Academy of Medical Sciences, National Clinical Research Center for Dermatologic and Immunologic Diseases, State Key Laboratory of Complex Severe and Rare Diseases, Key Laboratory of Rheumatology and Clinical Immunology, Ministry of Education, Beijing 100730, China.

Relapsing polychondritis is an immune mediated systemic inflammatory disease, involving the cartilaginous and proteoglycan rich structures. The characteristic manifestations were inflammation and deformity of ear and nasal cartilage. Here, Chinese Rheumatology Association summarized manifestations, diagnosis and disease activity index evaluation of relapsing polychondritis, standardized treatment regimens, to improve disease prognosis. Read More

JAAD Case Rep 2022 May 2;23:15-19. Epub 2022 Mar 2.

Department of Dermatology, Brigham and Women's Hospital, Boston, Massachusetts.

Hell J Nucl Med 2022 Jan-Apr;25(1):108-110. Epub 2022 Apr 8.

Department of Nuclear Medicine, Anhui Provincial Hospital, Cheeloo College of Medicine, Shandong University, Jinan, Shandong, 250021, P.R.China.

Biologics 2022 31;16:17-19. Epub 2022 Mar 31.

Department of Respiratory Medicine and Clinical Immunology, Graduate School of Medicine, Osaka University, Osaka, Japan.

TNF-alpha-targeted therapies during pregnancy is a topic of interest in rheumatology. Etanercept (ETN) is expected to have lower transplacental transfer, however, clinical evidence is lacking on the usefulness and safeness of continuing etanercept throughout pregnancy. We here described the first reported case of relapsing polychondritis where continuous use of ETN throughout pregnancy was required. Read More

Clin Ter 2022 Apr;173(2):97-98

Department of Rheumatology, Basaksehir Cam and Sakura City Hospital, University of Health Sciences, Istanbul, Turkey.

Abstract: Relapsing polychondritis (RP) is a rare autoimmune disease char-acterized by multi-systemic involvement characterized by recurrent and progressive inflammation of the cartilaginous tissue. Auricular inflammation is a characteristic finding of RP. Anti-tumor necrosis fac-tor alpha (anti-TNF) is a highly effective drug used in the treatment of inflammatory arthritis. Read More

Eur Rev Med Pharmacol Sci 2022 03;26(6):1789

Department of Cardiology, Zhuzhou City Central Hospital, Zhuzhou, China.

Front Immunol 2022 10;13:812927. Epub 2022 Mar 10.

Pediatric Rheumatology and Immunology, Wilhelmina Children's Hospital, University Medical Center Utrecht, Utrecht, Netherlands.

Background: Autologous hematopoietic stem cell transplantation (autoHSCT) is increasingly being recognized as a treatment option for severe refractory autoimmune diseases (AD). However, efficacy is hampered by high relapse rates. In contrast, allogeneic HSCT (alloHSCT) has high potential to cure AD, but is associated with significant morbidity and mortality, and data in AD are limited. Read More

Clin Exp Rheumatol 2022 May 29;40 Suppl 134(5):3-11. Epub 2022 Mar 29.

Liga Portuguesa Contra as Doenças Reumáticas, Lisbon, Portugal.

In order to address the main challenges related to the rare diseases (RDs) the European Commission launched the European Reference Networks (ERNs), virtual networks involving healthcare providers (HCPs) across Europe. The mission of the ERNs is to tackle low prevalence and RDs that require highly specialised treatment and a concentration of knowledge and resources. In fact, ERNs offer the potential to give patients and healthcare professionals across the EU access to the best expertise and timely exchange of lifesaving knowledge, trying to make the knowledge travelling more than patients. Read More

Clin Exp Rheumatol 2022 May 28;40 Suppl 134(5):40-45. Epub 2022 Mar 28.

Policlinic and Hiller Research Unit for Rheumatology, ERN-ReCONNET member, University Clinic Duesseldorf, Heinrich-Heine-University Duesseldorf, Germany.

Objectives: To perform a systematic literature review (SLR) on the association of common variable immunodeficiency (CVID) and rare and complex connective tissue and musculoskeletal diseases, namely systemic lupus erythematosus (SLE), Sjögren's syndrome (SS), idiopathic inflammatory myopathies (IIM), systemic sclerosis (SSc), relapsing polychondritis, antiphospholipid syndrome, immunoglobulin (Ig) G4-related disease, as well as undifferentiated and mixed connective tissue disease.

Methods: An SLR on studies and cases about the association of CVID and rare and complex connective tissue and musculoskeletal diseases was performed. Animal studies were excluded. Read More

ACR Open Rheumatol 2022 Mar 17. Epub 2022 Mar 17.

The Jikei University School of Medicine, Kashiwa Hospital, Chiba, Japan.

Iran J Immunol 2022 Mar;19(1):10

Department of Rheumatology and Immunology, Tongji Hospital, Tongji University School of Medicine, Shanghai, People's Republic of China.

A male patient had suffered miscellaneous ocular symptoms for 20 years with auricular dysmorphosis and was diagnosed with Relapsing Polychondritis (RP) in the ear, nose, joints, and costal cartilage. The patient lost his vision owing to recurrent ocular symptoms for decades. He presented an increased IgA and was diagnosed with monoclonal gammopathy of undetermined significance (MGUS) and treated by prednisone and cyclophosphamide. Read More

Int J Mol Sci 2022 Feb 22;23(5). Epub 2022 Feb 22.

Respiratory Diseases Unit, Department of Medical and Surgical Sciences, University of Modena Reggio Emilia, University Hospital of Modena, 41121 Modena, Italy.

Laryngotracheal stenosis (LTS) is a complex and heterogeneous disease whose pathogenesis remains unclear. LTS is considered to be the result of aberrant wound-healing process that leads to fibrotic scarring, originating from different aetiology. Although iatrogenic aetiology is the main cause of subglottic or tracheal stenosis, also autoimmune and infectious diseases may be involved in causing LTS. Read More

Eur J Case Rep Intern Med 2022 4;9(2):003076. Epub 2022 Feb 4.

Department of Internal Medicine, Hospital Universitari de Bellvitge, L'Hospitalet de Llobregat, Barcelona, Spain.

Rosai-Dorfman-Destombes disease (RDD) or sinus histiocytosis with massive lymphadenopathy is a rare non-Langerhans cell histiocytosis of unknown cause. The disease often manifests as painless bilateral cervical lymphadenopathy associated with systemic symptoms such as fever and weight loss. Extranodal disease is also frequent and can involve any organ, mostly the skin, nasal cavity, bone, and retro-orbital tissue. Read More

EJVES Vasc Forum 2022 22;54:69-73. Epub 2022 Jan 22.

Department of Vascular Surgery, AZ Sint-Jan Brugge, Brugge, Belgium.

Introduction: Isolated iliac artery dissection (IAD) is a rare form of arterial dissection. The most commonly known causes of non-traumatic isolated iliac dissection are connective tissue diseases. Relapsing polychondritis (RP) is a rare inflammatory and multisystemic disease, typically affecting cartilage and connective tissue. Read More

Exp Ther Med 2022 Apr 9;23(4):272. Epub 2022 Feb 9.

Department of Pharmacy, Hebei Provincial Chest Hospital, Shijiazhuang, Hebei 050041, P.R. China.

Relapsing polychondritis (RP) is a clinical disease characterized by inflammation of cartilage tissue and chondrocytes. The principal curcuminoid curcumin is the most active component in turmeric and has been reported to have a chondroprotective effect, including anti-inflammatory activity, which is vitally important for mitigating RP symptoms and prognosis. However, the mechanisms underlying these actions have remained to be fully elucidated. Read More

Tidsskr Nor Laegeforen 2022 03 28;142(4). Epub 2022 Feb 28.

Background: VEXAS syndrome (Vacuoles, E1 enzyme, X-linked, Autoinflammatory, Somatic syndrome) first described in 2020, is caused by a limited repertoire of somatic mutations in UBA1, a gene involved in the initiation of ubiquitination. Ubiquitination, adding an ubiquitin protein to a substrate protein, can have various effects on the substrate. Disruption of UBA1 function results in diverse clinical manifestations, mimicking a variety of disorders. Read More

Clin Exp Rheumatol 2022 May 24;40 Suppl 134(5):86-92. Epub 2022 Feb 24.

Rheumatology Department, County Emergency Clinical Hospital Cluj, Centre for Rare Musculoskeletal Autoimmune and Autoinflammatory Diseases, Cluj-Napoca, and Iuliu Hațieganu University of Medicine and Pharmacy, Cluj-Napoca, Romania.

Objectives: Relapsing polychondritis (RP) evolves with variable and intermittent involvement of cartilage and proteoglycan-rich structures. Ocular manifestations are present in up to two-thirds of RP patients. Necrotising scleritis (NS) and peripheral ulcerative keratitis (PUK) may be inaugural and may lead to eye perforation and vision loss. Read More

Clin Exp Rheumatol 2022 May 28;40 Suppl 134(5):81-85. Epub 2022 Jan 28.

Service de Rhumatologie, Hôpitaux Universitaires de Strasbourg, Centre National de Référence des Maladies Systémiques et Auto-immunes Rares Grand-Est Sud-Ouest (RESO), Strasbourg, France.

Objectives: Due to the rarity of relapsing polychondritis (RP), no randomised clinical trial has been conducted to date and treatment remains empirical. We performed a systematic literature review to assess the efficacy of the main conventional immunosuppressants and biotherapies used in RP.

Methods: We searched MEDLINE for original articles without language restriction. Read More

Expert Opin Biol Ther 2022 05 6;22(5):661-671. Epub 2022 Mar 6.

Division of Rheumatology, Department of Medicine DIMED, University of Padova, Padova, Italy.

Introduction: Relapsing polychondritis (RP) is a rare systemic inflammatory disease of unknown etiology, primarily affecting cartilaginous tissue and proteoglycan-rich structures. Clinical manifestations vary from mild symptoms to occasional organ or life-threatening complications. Treatment can be challenging and is mostly based on experience or case reports/series. Read More

Cureus 2022 Jan 20;14(1):e21463. Epub 2022 Jan 20.

Department of Respiratory Medicine, Hamanomachi Hospital, Fukuoka, JPN.

We describe the case of a 60-year-old Japanese man with relapsing polychondritis (RP). The patient was referred to Hamanomachi Hospital due to mild elevation of C-reactive protein and mild anemia on medical checkup without any symptoms. Body CT imaging showed thickened tracheal and bronchial walls with no active lesions in the lung. Read More