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Table 4 Studies and case reports that discussed the use of Anti-TNF drugs in FMF

From: Update on the management of colchicine resistant Familial Mediterranean Fever (FMF)

Study Description Follow-up
Aldea et al. [100] 3 patients with fever, arthritis, abdominal attacks, and rash along with amyloidosis for 1 patient were treated with infliximab. The attacks were discontinued or significantly improved. Patients were followed-up for 12 months except for the amyloidosis patient for 3 months.
Metyas et al. [101] A 30-year-old presented with recurrent fever, weight loss, intermittent bilateral knee pain and swelling, abdominal pain with intermittent vomiting, and chest pain. Amyloidosis was confirmed by Congo red stain and immunohistochemical stains.
The patient was refractory to methotrexate treatment.
By the sixth dose of infliximab, the patient felt dramatically better. Arthritis, swelling, chest pain, and abdominal pain resolved. The patient was able to return to work after 2 years of disability secondary to arthritis. Laboratory markers of amyloidosis improved.
Kaya et al. [102] A 27-year-old male patient with FMF and Juvenile Idiopathic Arthritis (JIA) heterozygote for M694 V mutation presented for a relapse of his rheumatological diseases. He was started on menthotrexate and colchicine with steroid on board. Leflunomide was added later due to partial response to methotrexate. At the 7th month of MTX and leflunomide treatment, Etanercept was started as joint inflammation did not subside despite methotrexate and leflunomide. His FMF attacks were improved; he had only one attack of abdominal pain after the onset of etanercept treatment. Follow-up period was 2 months.
Ozgocmen et al. [103] A 35-year-old woman with FMF resistant to colchicine failed a combination of colchicine, sulfasalazine, and methotrexate. Infliximab was added to the treatment regimen. A 72-week follow-up of the patient yielded complete remission of the febrile abdominal episodes and spondylitis. The patient’s bilateral aseptic necrosis of the femoral head deteriorated and caused hip pain, discomfort, and disability.
Daysal et al. [104] A 21-year-old female homozygote for M694 V mutation presented for severe hip arthritis and elevated inflammatory markers which didn’t improve with colchicine, steroid, hydroxychloroquine and methotrexate. Infliximab was given intravenously. She had a prompt response. In 4–5 days, she was able to ambulate independently. Two weeks later her ESR and CRP were negative, and steroid therapy was discontinued. Follow-up period was 2 months.
Mor et al. [105] A 35-year-old man presented for episodes of articular pain, episodic testicular pain, recurrent oral ulcers, episodic abdominal pain, and episodic fever. He had elevated inflammatory markers. A single V726A mutation in the MEFV gene was identified. Colchicine was discontinued because of diarrhea. The patient was started on etanercept 25 mg twice a week, which immediately reduced the frequency, duration, and severity of the joint attacks for 3 years.
Nakamura et al. [106] A Japanese patient with FMF has a heterozygous mutations in the MEFV gene. Conventional treatment, such as colchicine and reserpine, failed to sufficiently control the FMF attacks. After starting infliximab and low-dose methotrexate, the frequency of the FMF attacks dramatically decreased and the clinical effect has remained unchanged for longer than 1 year.
Ozgocmen et al. [99] The patients were a 37-year-old man with concomitant Crohn’s disease and amyloidosis who was treated with infliximab and switched to adalimumab, and two female patients (a 24-year-old and a 31-year-old) with FMF who developed severe spondylitis. Adalimumab controlled FMF attacks quit effectively.
Bilgen et al. [107] The data for 10 FMF patients (5 male and 5 female patients) with chronic arthritis and/or sacroiliitis who were on anti-TNF agents. Frequency of FMF attacks before and after treatment with anti-TNF agents was recorded. The effects of the anti-TNF treatment were determined by using the number of tender and/or swollen joints, serum acute phase reactant levels, and Bath Ankylosing Spondylitis Disease Activity Index scores. Change in urine protein loss was also evaluated in patients with amyloidosis. After anti-TNF treatment, in 3 patients, FMF attack frequency decreased, and in the remaining 7 patients, no attack occurred. Serum acute phase reactant levels were decreased significantly at 3 and 6 months after anti-TNF treatment. After anti-TNF treatment Bath Ankylosing Spondylitis Disease Activity Index scores were also decreased significantly. In all 3 patients with amyloidosis, urine protein loss decreased without any increase in serum creatinine levels.
Erten et al. [108] A 35-year-old male patient presented with fever, abdominal pain, malaise, and low back and ankle pain. He was on colchicine and sulfasalazine for FMF with homozygous M694 V mutation. Infliximab treatment was discontinued due to allergic symptoms. The patient responded well to etanercept. Febrile abdominal attacks and joint symptoms did not recur. Urinary proteinuria and acute phase proteins returned to normal limits. He remained in an excellent condition for 4 years of follow-up.
Kosmidou et al. [109] A 33-year-old male patient heterozygote for M694 V mutation had been diagnosed with FMF since his adolescence. He presented for atypical abdominal pain to be diagnosed with Crohn’s disease. Steroid and azathioprine were started. 2 months after the initiation of immunosuppressive treatment, there was still no favorable response on his bowel inflammation. In a 6-month extensive follow-up after starting infliximab, both diseases were found to be inactive.