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Hemophagocytic lymphohistiocytosis triggered by relapsing polychondritis:A case report

World Journal of Orthopedics 2024年第8期15卷 813-819页

作者： Mi-Ran Han Jeong-Hwan Hwang Seungah Cha So-Yeon Jeon Kyu Yun Jang Namsu Kim Chang-Hoon Lee Department of Internal Medicine Jeonbuk National University Medical SchoolJeonju 54907South Korea Department of Pathology Jeonbuk National University Medical SchoolJeonju 54907South Korea Department of Laboratory Medicine Jeonbuk National University Medical SchoolJeonju 54907South Korea

BACKGROUND Hemophagocytic lymphohistiocytosis(HLH)is a rare,life-threatening disorder caused by abnormal histiocytes and T cell *** adults,it is predominantly associated with infections,cancers,and autoimmune *** polychondritis(RP),another rare disease,is diagnosed based on symptoms without specific tests,featuring cartilage inflammation characterized by swelling,redness,and pain,rarely inducing *** SUMMARY A 74-year-old woman visited the emergency room with a fever of 38.6℃.Blood tests,cultures,and imaging were performed to evaluate *** showed increased fluorescent antinuclear antibody levels and mild cytopenia,with no other specific *** revealed lymph node enlargement was observed;however,biopsy results were *** re-evaluation of the physical exam,inflammatory signs suggestive of RP were observed in the ears and nose,prompting a tissue biopsy for ***,persistent fever accompanied by cytopenia prompted a bone marrow examination,revealing hemophagocytic *** finding no significant results in blood culture,viral markers,and tissue examination of enlarged lymph nodes,HLH was diagnosed by *** involved methylprednisolone followed by *** two months,bone marrow examination confirmed resolution of hemophagocytosis,with normalization of hyperferritinemia and *** Thorough physical examination enabled diagnosis and treatment of HLH trig gered by RP in patients presenting with fever of unknown origin.

关键词： Hemophagocytic lymphohistiocytosis relapsing polychondritis Autoimmune disease Fever of unknown origin Steroid Case report

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relapsing polychondritis causing breathlessness: Two case reports

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World Journal of Clinical Cases 2022年第23期10卷 8360-8366页

作者： Song-Yu Zhai Yu-Hao Zhang Ru-Yan Guo Jie-Wen Hao Shu-Xin Wen Department of Otorhinolaryngology Head and Neck Surgery Xi’an Fourth HospitalXi’an 710004Shaanxi ProvinceChina Department of Otorhinolaryngology Head and Neck Surgery Shanxi Bethune HospitalTaiyuan 030000Shanxi ProvinceChina Department of Otorhinolaryngology Head and Neck Surgery The First People's Hospital of NanyangNanyang 473000Henan ProvinceChina Department of Otorhinolaryngology Head and Neck Surgery The Third Clinical College of Shanxi Medical UniversityTaiyuan 030000Shanxi ProvinceChina

BACKGROUND relapsing polychondritis is a rare multisystem autoimmune disease that mainly involves systemic cartilage and proteoglycan-rich *** the larynx and trachea are involved,the patient’s condition deteriorates *** relapsing polychondritis becomes more advanced,the airways collapse and treatment is difficult,rendering a poor ***,the diagnosis method,treatment strategy and prognosis of relapsing polychondritis with larynx and trachea involvement need to be elucidated to improve clinicians’awareness of the *** SUMMARY A man and a woman were admitted because of *** polychondritis was diagnosed after a series of accessory *** were both treated with glucocorticoids and immunosuppressants,and underwent tracheotomy as their breathing difficulties could not be relieved by the *** The two cases highlight the importance of the timely diagnosis,full evaluation and initiating individualized treatment of relapsing polychondritis with larynx and trachea ***,bronchoscopy and pathological examination are helpful in diagnosis of this disease.

关键词： relapsing polychondritis Larynx Trachea Case report

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relapsing polychondritis with isolated tracheobronchial involvement complicated with Sjogren's syndrome:A case report

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World Journal of Clinical Cases 2022年第19期10卷 6563-6570页

作者： Jun-Yan Chen Xiao-Yan Li Chen Zong Department of Pulmonary and Critical Care Medicine The Second Hospital of Shanxi Medical UniversityTaiyuan 030000Shanxi ProvinceChina

BACKGROUNDrelapsing polychondritis (RP) is a rare, long-term, and potentially life-threateningdisease characterised by recurrent paroxysmal inflammation that can involve anddestroy the cartilage of the external ear, nose, larynx, and *** SUMMARYWe here report a case of RP involving solely the tracheobronchial cartilage ring(and not the auricular. nasal or articular cartilage) complicated by Sjögren's syndrome in a 47-year-old female whose delayed diagnosis caused a sharpdecline in pulmonary function. After corticosteroid treatment, her pulmonaryfunction *** such cases, our experience suggested that 18F-fluorodeoxyglucose positronemission tomography/computed tomography (18F-FDG PET/CT) and fiberopticbronchoscopy should be used to diagnose airway chondritis as relapsing polychondritisin the early phase of disease.

关键词： relapsing polychondritis Tracheobronchial involvement 18F-fluorodeoxyglucose positron emission tomography/computed tomography Fiberoptic bronchoscopy Case report

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The value of ^(99m)Tc methylene diphosphonate bone scintigraphy in diagnosing relapsing polychondritis

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Chinese Medical Journal 2006年第13期119卷 1129-1132页

作者： SHI Xu-hua ZHANG Feng-chun CHEN Li-bo OUYANG Meng Department of Rheumatology Department of Nuclear Medicine Peking Union Medical College Hospital Chinese Academy of Medical Sciences Beijing 100730 China

relapsing polychondritis （RP） is a recurrent disease involving cartilage mainly of the ear,nose, larynx, trachea, and bronchus. The typical manifestations of the disease in the ear and nose can be easily recognized, but the symptoms could be ignored or easily confused with those of other diseases when the cartilage of other sites is involved. Thus, it is necessary to develop a new technique for the diagnosis of this disease. Few cases of abnormal accumulation of radioactivity at cartilage shown by ^99mTc methylene diphosphonate （MDP） bone scintigraphy are described in the literature. In this report, we present 4 patients of whom 3 had positive findings on ^99mTc MDP bone scintigraphy with an assessment of ^99mTc MDP bone scintigraphy in the diagnosis of RP.

关键词： relapsing polychondritis ^99mTc methylene diphosphonate bone scintigraphy

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Eustachian tube involvement in a patient with relapsing polychondritis detected by magnetic resonance imaging:A case report

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World Journal of Clinical Cases 2022年第4期10卷 1441-1446页

作者： Daisuke Yunaiyama Akiko Aoki Hiroshi Kobayashi Miwako Someya Mitsuru Okubo Kazuhiro Saito Department of Radiology Tokyo Medical University HospitalShinjuku-ku 160-0023TokyoJapan Department of Radiology Tokyo Medical University Hachioji Medical CenterHachioji-shi 193-0998Japan Department of Rheumatology Tokyo Medical University Hachioji Medical CenterHachioji-shi 193-0998Japan Department of Otorhinolaryngology Head and Neck SurgeryTokyo Medical University Hachioji Medical CenterHachioji-shi 193-0998Japan

BACKGROUND relapsing polychondritis(RP)is a rare inflammatory disease involving the systemic cartilage,such as the auricle,trachea,and bronchiole,among others.A patient with RP shows variable symptoms based on the involved *** SUMMARY A 72-year-old Japanese woman with a history of redness of the bilateral auricles for 3 d was referred to a *** clinician prescribed antibiotics to the patient;however,the symptoms worsened;thus,she was referred to our *** and neck magnetic resonance imaging(MRI)showed edematous auricle with remarkable contrast,fluid collection in the bilateral mastoid cells,suggesting otitis *** eustachian tube(ET)on the right side was also edematous with contrast *** patient was suspected of RP according to the diagnostic criteria.A biopsy of the auricular cartilage was performed by an otorhinolaryngologist,confirming pathological proof of *** with steroids were immediately administered *** We highlight a rare case of RP with radiologically confirmed involvement of ET in the MRI.

关键词： relapsing polychondritis Magnetic resonance imaging Contrast enhancement Otitis media with effusion Eustachian tube Case report

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Rapid corneal thinning and perforated ulcerative keratitis in a patient with relapsing polychondritis

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Eye and Vision 2017年第1期4卷 37-40页

作者： Tracy Hiu Ting Lai Nikki Far Alvin Lerrmann Young Vishal Jhanji Department of Ophthalmology Tung Wah Eastern HospitalHong KongChina The Chinese University of Hong Kong Prince of Wales HospitalHong KongChina UPMC Eye Center University of Pittsburgh203Lothrop Street15231 PittsburghPAUSA

Background:To report rapid corneal thinning and perforation in a case with relapsing *** presentation:A 43 year-old male diagnosed with relapsing polychondritis suffered from bilateral scleritis,bilateral swelling of pinna,saddle nose and tracheal *** patient presented with right eye pain and redness for one *** lamp examination of the right eye showed 80%peripheral corneal thinning between 3 and 7 o’*** best-corrected visual acuity(BCVA)was 1.0 *** degree of corneal thinning worsened to 90%after one week of oral corticosteroid ***,topical cyclosporine 2%eye drops four times a day,oral doxycycline 100 mg twice a day and oral vitamin C 2 g daily were *** corneal thinning gradually improved to about 60%.However,the patient rapidly tapered oral prednisolone against medical advice and returned with an acute drop in vision in his right *** lamp examination of the right eye showed peripheral corneal perforation with iris *** emergency repair with cyanoacrylate glue was *** methylprednisolone 1 mg/kg body weight was administered for three days and 1 g/day intravenous immunoglobulin was administered every four *** 3 months postoperatively,BCVA in the right eye was *** lamp examination showed a well-formed anterior chamber with glue in ***:relapsing polychondritis may be associated with rapid corneal *** clinicians should be aware of the possibility of corneal perforation in these *** glue is a viable temporary management option in such scenarios.

关键词： relapsing polychondritis Corneal perforation Ulcerative keratitis

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