PMC5871368_01
Patient 1 at his first consultation. A - Evident
symmetrical lower lip swelling and angular cheilitis. B
- "Full-width" gengivitis
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f1
On physical examination, symmetrical lower lip swelling, cobblestoning of vestibular folds, "full width" gingivitis and angular cheilitis could be seen (Figure 1).
Patient 1. Histopathology of lip mucosa. A - Hematoxylin
& eosin stain with inflammatory infiltrate, non-caseating
granulomas and giant multinucleate cells (arrow) (X4).
B - Masson stain showing noncaseating granuloma and
giant multinucleate cells (arrow), adjacent to muscle fibers
(X40)
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Histopathology showed a chronic inflammatory infiltrate, with noncaseating granulomas and giant multinucleate cells, histiocytes and lymphocytes (Figure 2).
Patient 1 at 18-month follow-up. A: Asymmetrical lower
lip swelling, with soft tissue enlargement of right cheek.
B: Slight asymmetrical smile
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Eighteen months later, clinical evaluation showed improvement of the lip edema, but with slight cheek swelling, mainly towards his right side, with an asymmetric facial appearance (Figure 3).
PMC5871368_02
Patient 2 at his first consultation. A: Right upper lip
swelling, with mild lower lip edema. B: Fissured and
geographic tongue
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f4
No neurological symptoms were evident (Figure 4).
Patient 2 at 5-month follow-up. A: Mild lip swelling,
with dry lips. B: Fissured tongue persisted; geographic
tongue has a milder expression
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He continues to have a good clinical response to diet, with no systemic medication, with smaller orofacial swelling after two years of follow-up and a mild expression of geographic tongue (Figure 5).