Recurrent Interstitial Pneumonitis in a Patient with Entero-Behçet's Disease Initially Treated with Mesalazine

Abstract

A 65-year-old man with entero-Behçet's disease (BD) being treated with mesalazine was presented to our hospital complaining of dyspnea. Computed tomography (CT) of the chest showed ground-glass opacities and he was initially diagnosed with mesalazine-induced interstitial pneumonitis (IP). Besides the discontinuation of mesalazine, a high dose of oral prednisolone was administered and the patient seemed to recover. However, four months later, dyspnea recurred and repeated CT revealed more extensive pulmonary infiltration despite steroid therapy. After the exclusion of infections, we suspected either a recurrence of mesalazine-induced IP or BD-related IP as a clinical manifestation of BD. The patient was treated with intravenous methylprednisolone and cyclophosphamide, followed by orally administered azathioprine, based on the assumption of underlying vasculitis. Thereafter, his condition improved. BD-related IP is an extremely rare condition with limited reports in the literature. Mesalazine-induced IP is also uncommon but the prognosis is generally good after discontinuation of mesalazine with or without steroid therapy. We discuss an extremely rare case, especially focusing on BD-related IP and mesalazine-induced IP as a potential cause of recurrent IP in a patient with entero-BD.

Figure 1

Chest X-ray images at the time of diagnosis with BD (a) and at the first time appearance of dyspnea (b).

Figure 2

An ulcer on the ileum identified by colonoscopy.

Figure 3

Computed tomography of the chest at the first time appearance of dyspnea (a), after treatment with prednisone 40 mg/day (b), at recurrence of dyspnea (c), and after treatment with methylprednisolone pulse and cyclophosphamide pulse therapy (d).

Figure 4

The result of transbronchial lung biopsy. Hematoxylin and eosin (HE) stain revealed diffuse lymphocytes infiltration without any findings of vasculitis (magnification: 100x).

Figure 5

Clinical course of the patient. mPSL: methylprednisolone; IVCY: intravenous cyclophosphamide; 5-ASA: mesalazine; AZA: azathioprine; EN: erythema nodosum.