Steroid-resistant sarcoidosis: is antagonism of TNF-α the answer?

Denys, Bart G.; Bogaerts, Y; Coenegrachts, Kenneth; Vriese, An S. De

doi:10.1042/cs20060094

Cited by 46 publications

(26 citation statements)

References 63 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…However, the severity of extrapulmonary involvement varied considerably so the efficacy of infliximab for the treatment of severe multisystem refractory disease remains uncertain. The evidence for efficacy of other anti-TNF therapies is conflicting and limited to isolated case reports [11].…”

Section: Discussionmentioning

confidence: 99%

Refractory multisystem sarcoidosis responding to infliximab therapy

et al. 2012

View full text Add to dashboard Cite

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. We report outcomes of three patients with extensive multisystem sarcoidosis refractory to conventional treatment and treated at our center. Clinical assessment and radiographic imaging were used to assess the response to infliximab treatment. Infliximab therapy induced clinical remission in all three patients, and this clinical response correlated with radiographic evidence of the resolution of granulomatous disease. Serum ACE level was reduced in all cases, and daily steroid dosage was reduced. We propose that infliximab can be an effective treatment in patients with multisystem complex sarcoidosis refractory to conventional drug therapy and can result in sustained clinical remission. Our experience supports the urgent need for randomized controlled clinical trials of anti-TNF therapy in refractory systemic sarcoidosis.

show abstract

Section: Discussionmentioning

confidence: 99%

Refractory multisystem sarcoidosis responding to infliximab therapy

et al. 2012

View full text Add to dashboard Cite

show abstract

“…Most outcomes data in neurosarcoidosis are based on anecdotal reports and small series in the absence of prospective trials, with response to treatment being variable. Because of the suggested link between tumor necrosis factor-a (TNF-a) and disease, monoclonal antibodies to TNF-a have been studied for the treatment of refractory sarcoidosis [5,6,8,11,12], although this therapy remains experimental. Surgery (especially for intramedullary lesions) is reserved for diagnostic biopsy, as surgical resection is morbid and not beneficial [4].…”

Section: Discussionmentioning

confidence: 99%

Isolated intramedullary neurosarcoidosis of the thoracic spine: case report and review of the literature

2011

View full text Add to dashboard Cite

Sarcoidosis can involve the central nervous system in approximately 5-15% of cases. Any part of the nervous system can be involved, so presentation can be quite varied. Isolated disease of the spinal cord is even less common and reports are limited to single cases and small series. Although in the setting of systemic disease the diagnosis can be made with skin or lymph node biopsy, isolated disease of the spinal cord presents a diagnostic challenge. We present a case of isolated intramedullary neurosarcoidosis of the distal thoracic spinal cord presenting with posterior column dysfunction. Imaging demonstrated T2 changes in the patient's lower thoracic cord adjacent to disc herniation. Over time, however, his symptoms progressed despite decompression, and the abnormal region began to exhibit focal contrast enhancement. The persistence of symptoms as well as the new enhancement led us to perform a spinal cord biopsy, which demonstrated histopathological findings consistent with sarcoidosis. Further workup failed to reveal any evidence of systemic disease. Intramedullary sarcoidosis without systemic sarcoidosis is extremely rare. With its variable imaging appearance and inconsistent clinical manifestations, it can be difficult to diagnose. It should be considered in the differential diagnosis of a mass-like intramedullary lesion with progressive symptoms. Biopsy with histopathological correlation may be the only definite management option.

show abstract

“…A repeat biopsy showed persistent diabetic glomerulosclerosis, moderate chronic tubulointerstitial inflammation with complete resolution of interstitial epitheliod granulomas. Although adalimumab and infliximab are generally safe, some side effects include risk of lymphoma and reactivation of latent tuberculosis ( Denys et al 2007). These agents may hold promise for the future once large scale randomized studies are available to show consistent benefits with minimal side effects.…”

Section: Treatmentmentioning

confidence: 99%