Oral Lichen Planus in a Patient with Von Willebrand Disease: An Atypical Presentation with Persistent Tongue Bleeding

Background: Oral lichen planus (OLP) is a chronic inflammatory condition, commonly presenting with striae, plaques, or erosions. Significant oral bleeding is not a typical feature. This case highlights an unusual presentation of OLP in a patient with Von Willebrand Disease (VWD) where persistent tongue bleeding complicated both diagnosis and management.

Case Presentation: A 72-year-old male with VWD Type 2A presented to the National Coagulation Centre (NCC), Dublin, Ireland in May 2024 with soreness and episodes of prolonged bleeding of more than two hours from the tip of his tongue, unrelieved by topical antifibrinolytic therapy. Examination revealed a depapillated, red patch on the tip of the tongue, adjacent to calculus deposits and sharp incisal edges of the lower incisors. A provisional diagnosis of ‘glossitis’ was made by his haematologist. Tranexamic Acid (TxA) and WILATE, a plasma-derived von Willebrand factor (VWF) and factor VIII concentrate, were given to restore haemostasis and the patient referred to the special-care dental department of the NCC.

Despite smoothing sharp teeth, calculus removal, and nightguard fabrication, the patient experienced continued episodic bleeding and discomfort over a two month period. He was referred to oral-medicine and an incisional biopsy was performed with haemostatic support (TxA and WILATE), confirming OLP. The patient was started on prednisolone mouthwash and had no further episodes of bleeding. Given the risk of mucosal haemorrhage with OLP flares, long-term maintenance therapy (once-nightly or alternate-night use) was recommended. Additionally, the patient was advised to initiate TxA (1g three times daily) at onset of a flare to control any associated bleeding.

Conclusion: While OLP is well-documented, persistent oral bleeding is an unusual presentation and is undocumented in the VWD literature. This case highlights the need for multidisciplinary management of patients with VWD and the importance of maintaining good control of OLP to minimise significant mucosal/tongue bleeding .