全部 标题 作者
关键词 摘要

OALib Journal期刊
ISSN: 2333-9721
费用:99美元

查看量下载量

相关文章

更多...

Sarcoidosis Presenting as L?fgren’s Syndrome with Myopathy

DOI: 10.1155/2013/125251

Full-Text   Cite this paper   Add to My Lib

Abstract:

A 34-year-old female patient, who had proximal muscle weakness for 8 months, presented with erythema nodosum lesions on the pretibial region in addition to pain, swelling, and movement restriction in both ankles for the last one month. Thoracic CT demonstrated hilar and mediastinal lymphadenopathy. She underwent mediastinoscopic lymph node biopsy; biopsy result was consistent with noncaseating granuloma. Serum angiotensin converting enzyme level and muscle enzymes have been elevated. Muscular MRI and EMG findings were consistent with myositis. Muscle biopsy was done, and myopathy was found. The patient was diagnosed with sarcoidosis, L?fgren's syndrome, and sarcoid myopathy. The patient displayed remarkable clinical and radiological regression after 6-month corticosteroid and MTX therapy. 1. Introduction Sarcoidosis is a systemic granulomatous disease of unknown origin and can involve many tissues and organs in the body. The disease most commonly presents with bilateral hilar lymphadenopathy, lung infiltrations, and skin and ophthalmic lesions [1]. L?fgren’s syndrome (LS) is an acute sarcoidosis presentation characterized by arthritis/arthralgia, erythema nodosum (EN), and bilateral hilar lymphadenopathy [2]. Musculoskeletal system involvement may usually be in the form of synovitis of the large joints of lower extremities. Sarcoidosis may involve proximal muscles and may mimic idiopathic inflammatory myositis [3]. Symptomatic muscular disease is rare and seen in 0.5–5% of the cases [4]. It has three different types: chronic myopathy, palpable nodules, and acute myositis [5]. In the present case, we have reported the presence of chronic sarcoid myopathy together with L?fgren’s syndrome. 2. Case Report A 34-year-old female patient consulted with several physicians for proximal muscle weakness, which had continued and worsened for 8 months. Her complaints became worse despite NSAIDs and she presented to our outpatient clinic of rheumatology because of pain, swelling, and restricted motion in both ankles, as well as painful erythematous lesions on the pretibial region, which started a month ago. On the inspection, erythema nodosum skin lesions were detected on the pretibial region (Figure 1). Musculoskeletal system examination revealed synovitis in both ankles and proximal muscle weakness in the proximal extremities. Results of laboratory analysis were as follows; SGOT: 152?U/L (normal 0–30?U/L), SGPT: 144?U/L (normal 0–40?U/L), alkaline phosphatase: 46?U/L (normal < 90?U/L), LDH: 318?U/L (normal 135–220?U/L), CK: 1213?U/L (normal 26–192?U/L), C-reactive

References

[1]  G. W. Hunninghake, U. Costabel, M. Ando et al., “ATS/ERS/WASOG statement on sarcoidosis,” Sarcoidosis Vasculitis and Diffuse Lung Disease, vol. 16, no. 2, pp. 149–173, 1999.
[2]  J. Grunewald and A. Eklund, “L?fgren’s syndrome: human leukocyte antigen strongly infl uences the disease course,” American Journal of Respiratory and Critical Care Medicine, vol. 179, no. 4, pp. 307–312, 2009.
[3]  W. Chatham, “Rheumatic manifestations of systemic disease: sarcoidosis,” Current Opinion in Rheumatology, vol. 22, pp. 85–90, 2010.
[4]  K. D. Torralba and F. P. Quismorio Jr., “Sarcoidosis and the rheumatologist,” Current Opinion in Rheumatology, vol. 21, pp. 62–70, 2009.
[5]  F. Fayad, F. Lioté, F. Berenbaum, P. Orcel, and T. Bardin, “Muscle involvement in sarcoidosis: a retrospective and followup studies,” Journal of Rheumatology, vol. 33, no. 1, pp. 98–103, 2006.
[6]  J. Mana, G. C. Vaquero, A. Montero, et al., “L?fgren’s syndrome revisited: a study of 186 patients,” The American Journal of Medicine, vol. 107, pp. 240–245, 1999.
[7]  A. Glennas, T. K. Kvien, K. Melby et al., “Acute sarcoid arthritis: occurrence, seasonal onset, clinical features and outcome,” British Journal of Rheumatology, vol. 34, no. 1, pp. 45–50, 1995.
[8]  J. M. Kremer, “Histologic findings with acute sarcoid arthritis: association with the B8, DR3 phenotype,” Journal of Rheumatology, vol. 13, pp. 593–599, 1979.
[9]  J. Mana, G. C. Vaquero, A. Salazar, et al., “Periarticular ankle sarcoidosis: a variant of L?fgren’s syndrome,” The Journal of Rheumatology, vol. 23, pp. 874–877, 1996.
[10]  D. L. Johnson, M. Yamakido, and O. P. Sharma, “Musculoskeletal involvement in sarcoidosis,” Seminars in Respiratory Medicine, vol. 13, pp. 415–419, 1992.
[11]  A. Glennas, T. K. Kvien, K. Melby et al., “Acute sarcoid arthritis: occurrence, seasonal onset, clinical features and outcome,” British Journal of Rheumatology, vol. 34, no. 1, pp. 45–50, 1995.
[12]  R. A. Prayson, “Granulomatous myositis: clinicopathologic study of 12 cases,” American Journal of Clinical Pathology, vol. 112, no. 1, pp. 63–68, 1999.
[13]  J. Barnard and L. S. Newman, “Sarcoidosis: immunology, rheumatic involvement, and therapeutics,” Current Opinion in Rheumatology, vol. 13, no. 1, pp. 84–91, 2001.
[14]  D. S. Tews and D. E. Pongratz, “Immunohistological analysis of sarcoid myopathy,” Journal of Neurology Neurosurgery and Psychiatry, vol. 59, no. 3, pp. 322–325, 1995.
[15]  G. W. Hunninghake, P. Broska, R. Naber, et al., “Correlation of lung T-cell and macrophage function with disease activity in pulmonary sarcoid,” Clinical Research, pp. 29550–29553, 1981.
[16]  R. L. Dawkins and F. L. Mastaglia, “Cell-mediated cytotoxicity to muscle in polymyositis,” The New England Journal of Medicine, vol. 288, no. 9, pp. 434–438, 1973.
[17]  P. Pinkston, P. B. Bitterman, and R. G. Crystal, “Spontaneous release of interleukin-2 by lung T-lymphocytes in active pulmonary sarcoidosis,” The New England Journal of Medicine, vol. 308, no. 14, pp. 793–797, 1983.

Full-Text

Contact Us

service@oalib.com

QQ:3279437679

WhatsApp +8615387084133