title: Simultaneous development of sarcoidosis and cutaneous vasculitis in a patient with refractory Crohn's disease during infliximab therapy.
abstract: BACKGROUND
  Paradoxical inflammations during anti-TNF-α therapy are defined as adverse effects such as psoriasiform skin lesions, uveitis and sarcoidosis-like granulomas induced by immune reactions, not by infectious agents. Here, we report a very rare case of the simultaneous development of sarcoidosis and cutaneous vasculitis in a patient with refractory Crohn's disease during infliximab therapy and both of which resolved spontaneously without the cessation of infliximab.
  
  
  METHODS
  In September 2000, 23-year old Japanese male was diagnosed with Crohn's disease. Prednisolone in combination with mesalazine was introduced at first and succeeded for almost one year. In June 2002, since his gastrointestinal symptoms relapsed and were refractory, infliximab (IFX) therapy 5 mg/kg was introduced. In February 2011, because he had repeated arthralgia almost every intravenous IFX administration, IFX was increased to 10 mg/kg under the diagnosis of a secondary failure of IFX. In December 2012, he complained of slight dry cough and an itchy eruption on both lower limbs, and he was referred to our hospital due to the appearance of bilateral hilar lymphadenopathy on chest X-ray examination. Chest computed tomogram revealed bilateral hilar lymphadenopathy and fine reticulonodular shadows on the bilateral upper lungs. Serum calcium, angiotensin-converting enzyme and soluble interleukin 2 receptor levels were not elevated, but the titer of antinuclear antibody was considerably elevated. Mycobacterium infection was carefully excluded. Trans-bronchial lung biopsy showed non-caseating epithelioid cell granulomas compatible with sarcoidosis. The skin biopsy of the right limb was diagnosed as leukocytoclastic vasculitis. The patient was diagnosed as having a series of paradoxical inflammations during anti-TNF-α therapy. Since his paradoxical inflammations were not severe and opportunistic infections were excluded, IFX was cautiously continued for refractory Crohn's disease. Nine months later, not only his intrathoracic lesions but also his cutaneous lesions had spontaneously resolved.
  
  
  CONCLUSIONS
  Physicians caring for patients with anti-TNF-α therapy should know that, based on a careful exclusion of infectious agents and thoughtful assessment of the patient's possible risks and benefits, paradoxical inflammations can be resolved without the cessation of anti-TNF-α therapy.
authors: Department of Respiratory Medicine, Tohoku University Graduate School of Medicine, 1-1 Seiryo-machi, Aoba-ku, Sendai, 980-8574, Japan. tnumakura@rm.med.tohoku.ac.jp.
authors: Tadahisa
authors: T
authors: Numakura
authors: Department of Respiratory Medicine, Tohoku University Graduate School of Medicine, 1-1 Seiryo-machi, Aoba-ku, Sendai, 980-8574, Japan. tamada@rm.med.tohoku.ac.jp.
authors: Tsutomu
authors: T
authors: Tamada
authors: Clinical Research, Innovation and Education Center, Tohoku University Hospital, Sendai, Japan. nara@hosp-tohoku.ac.jp.
authors: Masayuki
authors: M
authors: Nara
authors: Department of Respiratory Medicine, Tohoku University Graduate School of Medicine, 1-1 Seiryo-machi, Aoba-ku, Sendai, 980-8574, Japan. soshi@rm.med.tohoku.ac.jp.
authors: Soshi
authors: S
authors: Muramatsu
authors: Department of Respiratory Medicine, Tohoku University Graduate School of Medicine, 1-1 Seiryo-machi, Aoba-ku, Sendai, 980-8574, Japan. kmura@rm.med.tohoku.ac.jp.
authors: Koji
authors: K
authors: Murakami
authors: Department of Respiratory Medicine and Infectious Diseases, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan. kikuchi@med.niigata-u.ac.jp.
authors: Toshiaki
authors: T
authors: Kikuchi
authors: Department of Respiratory Medicine, Tohoku University Graduate School of Medicine, 1-1 Seiryo-machi, Aoba-ku, Sendai, 980-8574, Japan. m-kobayashi@rm.med.tohoku.ac.jp.
authors: Makoto
authors: M
authors: Kobayashi
authors: Department of Respiratory Medicine, Tohoku University Graduate School of Medicine, 1-1 Seiryo-machi, Aoba-ku, Sendai, 980-8574, Japan. miho0601ds@msn.com.
authors: Miho
authors: M
authors: Muroi
authors: Department of Respiratory Medicine, Tohoku University Graduate School of Medicine, 1-1 Seiryo-machi, Aoba-ku, Sendai, 980-8574, Japan. tmokazaki0808@gmail.com.
authors: Tatsuma
authors: T
authors: Okazaki
authors: Takagi Clinic, Sendai, Japan. shotakagi@yahoo.co.jp.
authors: Sho
authors: S
authors: Takagi
authors: Department of Human Pathology, Tokyo Medical and Dental University, Tokyo, Japan. eishi.path@tmd.ac.jp.
authors: Yoshinobu
authors: Y
authors: Eishi
authors: Department of Respiratory Medicine, Tohoku University Graduate School of Medicine, 1-1 Seiryo-machi, Aoba-ku, Sendai, 980-8574, Japan. ichinose@rm.med.tohoku.ac.jp.
authors: Masakazu
authors: M
authors: Ichinose
mesh_terms: D018501:Antirheumatic Agents / Q000627:therapeutic use*; D001706:Biopsy; D003424:Crohn Disease / Q000150:complications / Q000188:drug therapy*; D006801:Humans; D000069285:Infliximab / Q000627:therapeutic use*; D008168:Lung / Q000473:pathology; D008297:Male; D017565:Sarcoidosis, Pulmonary / Q000150:complications* / Q000473:pathology; D012867:Skin / Q000473:pathology; D018366:Vasculitis, Leukocytoclastic, Cutaneous / Q000150:complications* / Q000473:pathology; D055815:Young Adult
chemical_list: D018501:Antirheumatic Agents; D000069285:Infliximab