A case of sialodochitis fibrinosa (Kussmaul’s disease) involving multiple salivary glands

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  • MORI Keisuke
    Department of Oral and Maxillofacial Surgery, Faculty of Medicine, Saga University
  • MATSUO Yuumi
    Department of Oral and Maxillofacial Surgery, Faculty of Medicine, Saga University
  • AIJIMA Reona
    Department of Oral and Maxillofacial Surgery, Faculty of Medicine, Saga University
  • MORI Makiko
    Division of Dermatology, Department of Internal Medicine, Faculty of Medicine, Saga University
  • DANJO Atsushi
    Department of Oral and Maxillofacial Surgery, Faculty of Medicine, Saga University
  • YAMASHITA Yoshio
    Department of Oral and Maxillofacial Surgery, Faculty of Medicine, Saga University

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Other Title
  • 複数の唾液腺に生じた線維素性唾液管炎 (Kussmaul’s disease) の1例
  • フクスウ ノ ダエキセン ニ ショウジタ センイ スジョウ ダエキカンエン(Kussmaul's disease)ノ 1レイ

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Abstract

<p>Sialodochitis fibrinosa is a relatively rare disease characterized by repetitive salivary gland swelling and fibrinous plug discharge and was reported in 1879 by Kussmaul. Here, we describe a patient who had sialodochitis fibrinosa in multiple salivary glands.</p><p>　A 31-year-old woman was referred to our department for examination and treatment of swelling in the right submandibular region. At the initial visit, swelling and tenderness were observed in the right submandibular region. However, computed tomography and magnetic resonance imaging assessments revealed no abnormal findings. Therefore, we consulted an internist, an otolaryngologist, and a dermatologist, but the cause remained unknown. Gradually, the symptoms disappeared, and the patient discontinued medical follow-up. After 2 years, the symptoms recurred, and the patient returned to our hospital. The swollen area had spread beyond the right submandibular region into the bilateral subparotid regions. We again examined the patient, and noted characteristic findings such as fibrinous plug discharge; therefore, we diagnosed sialodochitis fibrinosa. We provided medical treatment with corticosteroids and antihistamine drugs, and her symptoms improved. The disease has remained stable for 3 years since the initiation of treatment.</p>

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