Refractory multisystem sarcoidosis involving pelvic bone responding to infliximab
© Karjigi and Paul; licensee BioMed Central Ltd. 2013
Published: 14 February 2013
Chronic progressive multisystem granulomatous disease is seen in 10–30% of patients with sarcoidosis and can result in end organ damage . Corticosteroids are the mainstay of treatment with the addition of cytotoxic agents in severe cases. Some patients are refractory to such treatment and therefore management is a challenge. There is currently limited evidence for biological agents such as infliximab, a monoclonal anti-tumor necrosis factor-α antibody in the treatment of multisystem sarcoidosis. Aim of this case report is to disseminate knowledge concerning successful treatment of multisystem sarcoidosis involving pelvis with the use of TNF-α blockers in refractory cases.
Intavenous zolendronic acid helped with bone pain and hypercalcemia. Commenced on azathioprine however unable to tolerate because of headaches. Continued to have active disease and started on infliximab and methotrexate. She improved on this combination treatment and managed to reduce steroids gradually. She is on this treatment for a year and the condition is well controlled.
Granulomatous bone involvement has an overall incidence of 1-13%. The small bones of hands and feet are the most common localizations, while skull, knee, rib, pelvic and sternal localizations are rarely reported . The diagnosis of the sarcoid is based on the clinical presentation, radiological manifestations and histopathological assay. Even though radiographic findings are characteristic, could be challenging to distinguish from bony metastasis. On literature review there are reported case of vertebral sarcoidosis treated with anti TNF agents, our case report suggests infliximab can be effective in treatment of refractory sarcoidosis involving pelvic bone [3, 4].
This is first case reported on successful use of anti-TNF in treatment of multisystem sarcoidosis involving pelvic bone. There is currently limited evidence for biological agents such as infliximab, a monoclonal anti–TNF α antibody in the treatment of multisystem sarcoidosis . Our case supports the need for randomized controlled clinical trials of anti-TNF therapy in refractory systemic sarcoidosis.
- Croft AP, Situnayake D, Khair O, Giovanni G, Carruthers D, Sivaguru A, Gordon C: . Clin Rheumatol. 2012, 31 (6): 1013-8. 10.1007/s10067-011-1933-9. Epub 2012 Jan 17Google Scholar
- Bargagli E, Olivieri C, et al: Rare localizations of bone sarcoidosis, two case reports and review of the literature. Rheumatol Int. 2011, 31 (11): 1503-6. 10.1007/s00296-009-1315-7. Epub 2009 Dec 15View ArticlePubMedGoogle Scholar
- Garg S, Garg K, Altaf M, Magaldi JA: Refractory vertebral sarcoidosis responding to infliximab. J Clin Rheumatol. 2008, 14 (4): 238-40. 10.1097/RHU.0b013e318181b45a.View ArticlePubMedGoogle Scholar
- Hasni SA, Kunz D, Finzel K, Gruber : Osseous sarcoidosis treated with tumor necrosis factor-inhibitors: case report and review of the literature. Spine (Phila Pa 1976). 2010, 35 (18): E904-7. 10.1097/BRS.0b013e3181dc9a54.View ArticleGoogle Scholar
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