Case Report: Necrotizing Stomatitis as a Manifestation of COVID-19-Associated Vasculopathy

Abstract

Necrotizing stomatitis is a rare, acute-onset disease that is usually associated with severely malnourished children or diminished systemic resistance. We describe a 1-year-old girl who developed necrotizing stomatitis, vasculitic rash, skin desquamation on the fingers and toes, and persistent hypertension after serologically confirmed SARS-CoV-2 infection. Her laboratory investigations revealed positive IgG anticardiolipin and IgG anti-β2 glycoprotein antibodies, and biopsy of the mucosa of the lower jaw showed necrosis and endothelial damage with mural thrombi. Swollen endothelial cells of small veins in the upper dermis were confirmed also by electron microscopy. As illustrated by our case, necrotizing stomatitis may develop as a rare complication associated with SARS-CoV-2 infection and can be considered as a part of the clinical spectrum of COVID-19 vasculopathy. The pathogenic mechanism could involve a consequence of inflammatory events with vasculopathy, hypercoagulability, and damage of endothelial cells as a response to SARS-CoV-2 infection.

Keywords: COVID-19 associated vasculopathy; SARS-CoV-2; case report; damage of endothelial cell; hypertension; necrotizing stomatitis; vasculitis.

Figure 1

Necrosis of the frontal region of the mandibular alveolar ridge and three missing incisors.

Figure 2

Magnetic resonance imaging of the girl's mandible (T1 TSE fat sat contrast medium sequence) showed areas of bone necrosis (star) with peripheral reactive bone contrast enhancement (arrows) and enhancement of the soft tissue edema (arrowheads).

Figure 3

Histopathology of the biopsy specimens taken from the lower jaw (first biopsy). (a) Inflamed gingival mucosa with superficial erosions, with granulation tissue and dystrophic calcifications. Retrospectively, calcifications were “reinterpreted” as remains of destructed alveolar bone. HE, original magnification, ×4. (b) Recanalization of the small vessels in the inflamed gingival mucosa (mark). Note the intense mixed inflammatory infiltrate in the background and the swollen endothelial cells. HE, original magnification, ×20.

Figure 4

Histopathology of the skin efflorescence in the sacral area. (a) Focal epidermal necrosis is present in the center. Note the pronounced underlying small vessels with swollen endothelial cells in the papillary dermis. HE, original magnification, ×6. (b) Electron microscopy of swollen endothelial cells obliterating the lumen of the aforementioned small veins.