Multiple granulomatous inflammation in the minor salivary glands: A proposed new entity, allergic granulomatous sialadenitis

Abstract

We report a patient who presented with multiple small submucosal nodules with granulomatous inflammation in the minor salivary glands of the oral cavity. A 43-year-old woman presented with a 1-week history of multiple small submucosal nodules in her oral cavity after having taken medicine for abdominal pain. The patient did not have a history of fever, rectal bleeding, skin lesions or arthritis, but did have a history of drug allergy and bronchial asthma. Histopathological examination of the submucosal nodules showed sialadenitis with marked infiltration of lymphocytes, eosinophilic cells, macrophages and Langhans-type or foreign-body-type multinucleate giant cells. The macrophages tended to be aggregated and appeared to have caused immature granuloma formation without caseous necrosis. Degranulated eosinophilic cells were numerous. Sarcoidosis, Crohn's disease, tuberculosis and atypical mycobacterial infection were not identified by medical examination. Three weeks after discontinuing the medication the patient was seen again at a follow-up visit. Multiple submucosal small nodules and other symptoms were not evident at that time. This case report may represent a new entity of salivary gland disease that we tentatively refer to as 'allergic granulomatous sialadenitis'.

Eosinophilic sialodochitis: redefinition of ‘allergic parotitis’ and ‘sialodochitis fibrinosa’

“…These cases have been described in children, often in association with atopic illness, and in adults without atopy (Laudenbach et al , ). Similarly, it excluded cases of eosinophilic sialoadenitis marked by a different histopathology, including granulomatous inflammation of the minor salivary glands (Irie et al , ), Kimura's disease (Li et al , ), and angiolymphoid hyperplasia of the parotid gland (Hung et al , ).…”

“…These cases have been described in children, often in association with atopic illness, and in adults without atopy (Laudenbach et al, 1985). Similarly, it excluded cases of eosinophilic sialoadenitis marked by a different histopathology, including granulomatous inflammation of the minor salivary glands (Irie et al, 2004), Kimura's disease (Li et al, 1996), and angiolymphoid hyperplasia of the parotid gland (Hung et al, 2001). Table 2 summarizes the key clinical features of 59 cases of eosinophilic sialodochitis, including the three cases from our clinics and 56 cases from the literature (46 from Japan) who met our case definition (Pearson, 1935;Waldbott and Shea, 1947;Bookman, 1950;Serafini, 1951;Swinny, 1953;Riley, 1956;Cohen, 1965;Okuda et al, 1975;Miyazaki and Kumagami, 1981;Murakami et al, 1982;Yoshida et al, 1994;Ogawa et al, 1995;Watanabe et al, 1995;Irifune et al, 1996;Ishii et al, 1996;Kiuchi et al, 1996;Tanimoto et al, 1997;Yoshioka et al, 1997;Motosugi et al, 1998;Fujii et al, 2000;Yamashita et al, 2001Yamashita et al, , 2012Darling et al, 2002;Kishimoto et al, 2002;Yamamoto et al, 2002Yamamoto et al, , 2004Ikegami et al, 2003;Kinoshita and Sadanaga, 2003;Tsutsumi et al, 2003;Shino et al, 2005;Chikamatsu et al, 2006;…”

Review article: non‐malignant oral manifestations in inflammatory bowel diseases

“…In some areas, this ductal lesion is associated with rupture and mucocele formation. This condition has to be differentiated from multiple granulomatous inflammations in the minor salivary glands, an entity known as allergic granulomatous sialadenitis …”

“…This condition has to be differentiated from multiple granulomatous inflammations in the minor salivary glands, an entity known as allergic granulomatous sialadenitis. 94 Linear IgA disease. Association of linear IgA bullous disease with CD 95 and with UC has been reported, including cases of successful treatment with infliximab 96 or even with colectomy.…”

PI3K/AKT/PTEN pathway as a target for Crohn’s disease therapy (Review)

“…Crohn's disease is a chronic inflammatory disorder associated with mucosal inflammation of the bowel wall, which is characterized by repetitive active cycles of the disease state (5). It is histologically characterized by the massive transmural infiltration of lymphoctes and macrophages with granulomas (6). Crohn's disease can affect the entire gastrointestinal tract although the most common presentation is the ileum-colon junction.…”