Saddle Nose Deformity as a Result of an Aseptic Nasal Abscess Accompanied by Ulcerative Colitis and Pyoderma Gangrenosum

Abstract

Ulcerative colitis (UC) is a chronic inflammatory bowel disease (IBD) that causes granulomatous inflammation in the mucous membrane of the intestinal tract, especially in the colon. Additionally, UC can be accompanied by extraintestinal manifestation (EIM). EIM of UC includes cutaneous lesions such as pyoderma gangrenosum, and rarely, systemically occurring aseptic abscesses (AA) have been reported in a few cases. Nasal involvement of UC as an extraintestinal manifestation of IBD is rare, and few reports of nasal mucosa and septal cartilage involvement have been documented in the literature. However, aseptic nasal abscess involving septal cartilage associated with UC and pyoderma gangrenosum resulting in saddle nose deformity have not been reported. The author presents a case of a 52 year-old woman with UC and pyoderma gangrenosum and an aseptic abscess in the nasal septal cartilage resulting in saddle nose deformity, which was corrected by augmentation rhinoplasty. KEY WORDS:Saddle nose deformityㆍAseptic abscessㆍUlcerative colitisㆍPyoderma gangrenosum.

Presence of Phlebitis in Aseptic Nasal Septal Abscess Complicated with Ulcerative Colitis; Possible Association with Granulomatosis with Polyangiitis: A Case Report

“…Representative extra-intestinal manifestations of UC include anemia, arthropathy, uveitis, hepatobiliary disease, and pyoderma gangrenosum (PG) (Magro et al 2017). Recently, aseptic nasal septal abscess has also been reported as a rare manifestation of PG in patients with UC (Yilmaz et al 2011;Chang 2018;Tomioka et al 2018;Yu et al 2020) (Table 1). Additionally, leukocytoclastic vasculitis (a form of small-vessel vasculitis) is one of the extra-intestinal manifestations of UC, and it has been reported that 41% of patients with UC complicated with leukocytoclastic vasculitis are positive for ANCA (Pantic et al 2022).…”

“…However, upper respiratory tract lesions of GPA often do not show typical pathological features (e.g., necrotizing granulation), posing a challenge to the diagnosis of GPA by pathology in the localized type (Harabuchi et al 2013;Sattui and Lally 2020). Also, nasal PG in patients with UC and IgA monoclonal gammopathy has been reported (Matsumura et al 1999;Isomura et al 2005;Yilmaz et al 2011;Chang 2018;Tomioka et al 2018;Yu et al 2020). However, unlike in this case, vasculitis or elevation of PR3-ANCA was not reported in any cases (Table 1).…”

“…Therefore, we diagnosed the patient with aseptic nasal septal abscess associated with UC. Although the aseptic nasal septal abscess had been reported as extra-intestinal manifestations of UC (Yilmaz et al 2011;Chang 2018;Tomioka et al 2018;Yu et al 2020), the presence of phlebitis indicated co-existing small-vessel vasculitis, possibly GPA in the context of PR3-ANCA-positivity (Jennette et al 2013;Robson et al 2022). The patient was diagnosed with probable GPA according to the diagnostic criteria from Japanese Ministry of Health, Labour and Welfare (MHLW) (Isobe et al 2020).…”

“…Classically, it can occur secondary to trauma, nasal surgery, dental infections, and sinusitis, particularly in immunocom-promised patients and those with uncontrolled diabetes mellitus (Ambrus et al 1981;Salam and Camilleri 2009;Cheng et al 2019). Moreover, recent studies showed that systemic diseases, such as ulcerative colitis (UC), could cause nasal septal abscesses (Yilmaz et al 2011;Chang 2018;Tomioka et al 2018;Yu et al 2020). In this article, we report a rare case of an aseptic nasal septal abscess in a patient with proteinase 3-antineutrophil cytoplasmic antibody (PR3-ANCA)-positive UC, in which phlebitis was observed and granulomatosis with polyangiitis (GPA) might co-exist.…”