Home
|
Inbox
|
Search
|
View Abstract
A Case of Neurologic and Parotid Sarcoidosis After Treatment with Tumor Necrosis Factor Alpha (TNF-α) Inhibitor Infliximab
Description
.abstract img { width:300px !important; height:auto; display:block; text-align:center; margin-top:10px } .abstract { overflow-x:scroll } .abstract table { width:100%; display:block; border:hidden; border-collapse: collapse; margin-top:10px } .abstract td, th { border-top: 1px solid #ddd; padding: 4px 8px; } .abstract tbody tr:nth-child(even) td { background-color: #efefef; } .abstract a { overflow-wrap: break-word; word-wrap: break-word; }
A6477 - A Case of Neurologic and Parotid Sarcoidosis After Treatment with Tumor Necrosis Factor Alpha (TNF-α) Inhibitor Infliximab
Author Block: L. Bajpayee, J. S. Berman, P. Govender, F. J. Hawkins; The Pulmonary Center, Boston University School of Medicine, Boston, MA, United States.
Introduction:
Tumor necrosis factor-alpha (TNF-α) inhibitors are often used to treat advanced or refractory sarcoidosis. Paradoxically, cases of new onset sarcoidosis-like diseases are increasingly being reported in patients receiving TNF-α inhibitors. We report a case of neurologic sarcoidosis emerging in a patient being treated for psoriasis with infliximab.
Case Report:
A 42-year-old man presented with acute onset of fatigue, weight loss, bilateral parotid gland swelling, right-sided facial droop, unsteady gait, dysgeusia, blurry vision, dry eyes and dry mouth. Medical history was notable for refractory psoriasis treated with ongoing infliximab infusions for the past seven years (with previous treatment trials of methotrexate and etanercept). Ophthalmic exam was significant for optic nerve swelling. Neurologic exam was notable for gait abnormality and right sided weakness. Brain MRI identified enhancement of cranial nerves VII and VIII without central lesions. Lumbar puncture revealed 183 WBC (95% lymphocytes), glucose 42, protein 163, with negative culture and cytology, consistent with aseptic meningitis. A right-sided parotid gland biopsy revealed non-caseating granulomas with negative culture and cytology, consistent with sarcoidosis. We postulated a paradoxical sarcoid-like reaction to infliximab and recommended treatment with mycophenoloate mofetil 1 gram twice a day, prednisone 10 milligram daily, and cessation of the TNF-α inhibitor. The patient developed clinical and radiographic improvement within three weeks and had complete resolution of disease within 7 months. Following symptom resolution, he was maintained on dual therapy for an additional 12 months. Subsequently, his immunosuppression was slowly stopped without recurrence of disease.
Discussion:
There is limited understanding of the early pathogenesis of sarcoidosis. Although TNF-α plays an important role in pathogenesis of sarcoidosis, the development of a sarcoid-like reaction with TNF-α inhibitor is paradoxical and the mechanism of this response remains unknown. To date, there are less than 50 cases of sarcoidosis-like granulomatosis in patients treated with TNF- α inhibitors, and only 4 cases of neurosarcoidosis from a TNF- α inhibitor. Like this case, all reported cases have resolved with treatment and withdrawal of the TNF-α inhibitor without further recurrence of the disease. With the ever-increasing use of TNF-α inhibitors, it is important to recognize this as a rare but treatable complication.
Author Block: L. Bajpayee, J. S. Berman, P. Govender, F. J. Hawkins; The Pulmonary Center, Boston University School of Medicine, Boston, MA, United States.
Introduction:
Tumor necrosis factor-alpha (TNF-α) inhibitors are often used to treat advanced or refractory sarcoidosis. Paradoxically, cases of new onset sarcoidosis-like diseases are increasingly being reported in patients receiving TNF-α inhibitors. We report a case of neurologic sarcoidosis emerging in a patient being treated for psoriasis with infliximab.
Case Report:
A 42-year-old man presented with acute onset of fatigue, weight loss, bilateral parotid gland swelling, right-sided facial droop, unsteady gait, dysgeusia, blurry vision, dry eyes and dry mouth. Medical history was notable for refractory psoriasis treated with ongoing infliximab infusions for the past seven years (with previous treatment trials of methotrexate and etanercept). Ophthalmic exam was significant for optic nerve swelling. Neurologic exam was notable for gait abnormality and right sided weakness. Brain MRI identified enhancement of cranial nerves VII and VIII without central lesions. Lumbar puncture revealed 183 WBC (95% lymphocytes), glucose 42, protein 163, with negative culture and cytology, consistent with aseptic meningitis. A right-sided parotid gland biopsy revealed non-caseating granulomas with negative culture and cytology, consistent with sarcoidosis. We postulated a paradoxical sarcoid-like reaction to infliximab and recommended treatment with mycophenoloate mofetil 1 gram twice a day, prednisone 10 milligram daily, and cessation of the TNF-α inhibitor. The patient developed clinical and radiographic improvement within three weeks and had complete resolution of disease within 7 months. Following symptom resolution, he was maintained on dual therapy for an additional 12 months. Subsequently, his immunosuppression was slowly stopped without recurrence of disease.
Discussion:
There is limited understanding of the early pathogenesis of sarcoidosis. Although TNF-α plays an important role in pathogenesis of sarcoidosis, the development of a sarcoid-like reaction with TNF-α inhibitor is paradoxical and the mechanism of this response remains unknown. To date, there are less than 50 cases of sarcoidosis-like granulomatosis in patients treated with TNF- α inhibitors, and only 4 cases of neurosarcoidosis from a TNF- α inhibitor. Like this case, all reported cases have resolved with treatment and withdrawal of the TNF-α inhibitor without further recurrence of the disease. With the ever-increasing use of TNF-α inhibitors, it is important to recognize this as a rare but treatable complication.
|
Home
|
Inbox
|
Search
|