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Central airway stenosis in a patient with autoimmune pancreatitis

¹ First Dept of Internal Medicine, ² Second Dept of Internal Medicine, ⁴ Dept of Laboratory Medicine, ⁵ Dept of Radiology, Shinshu University School of Medicine, and ³ Center for Health, Safety and Environmental Management, Shinshu University, Matsumoto, Japan

CORRESPONDENCE: M. Yasuo, First Dept of Internal Medicine, Shinshu University School of Medicine, 3-1-1 Asahi, Matsumoto 390-8621, Japan. Fax: 81 263363722. E-mail: yasumasa{at}shinshu-u.ac.jp

Keywords: Airway, autoimmune disease, bronchoscopy, immunopathology

Received: April 3, 2008
Accepted September 17, 2008

Autoimmune pancreatitis is a unique form of chronic pancreatitis characterised by a high-serum immunoglobulin (Ig)G4 concentration involving various extra pancreatic lesions.

A 63-yr-old female with autoimmune pancreatitis complained of cough. Chest computed tomography revealed an irregular stenosis of the central airway, lung hilar and mediastinal lymph node swelling, and a marked thickness of the bronchovascular bundle. Bronchoscopic examination revealed an irregular tracheobronchial stenosis accompanied with an oedematous mucosa and engorged vessels. Lung hilar and mediastinal lymph node swelling, central airway stenosis and bronchoscopic findings remarkably resembled those of sarcoidosis.

Bronchial biopsy specimens demonstrated diffuse infiltrations of plasma cells, lymphocytes and eosinophils with fibrosis. Immunostaining showed infiltration of several IgG4-positive plasma cells. The patient was treated with oral prednisolone at 1 mg·kg^–1·day^–1 for pancreatic lesions. A month later, the lung lesions, including central airway stenosis, lung hilar and mediastinal lymph node swelling, and bronchovascular bundle thickness, had dramatically improved along with improvement of pancreatitis, thus indicating a close association between the two conditions.

This is the first report of a patient with autoimmune pancreatitis showing central airway stenosis similar to that of sarcoidosis.