Peripheral nervous system involvement in primary burning mouth syndrome-results of a pilot study

• Published in: Oral diseases Vol. 22; no. 4; pp. 338 - 344
• Main Authors: Puhakka, A, Forssell, H, Soinila, S, Virtanen, A, Röyttä, M, Laine, M, Tenovuo, O, Teerijoki-Oksa, T, Jääskeläinen, SK
• Format: Journal Article
• Language: English
• Published: Denmark Blackwell Publishing Ltd 01.05.2016; Wiley Subscription Services, Inc
• Subjects: Aged; burning mouth syndrome; Burning Mouth Syndrome - etiology; Cadaver; Case-Control Studies; Diabetes Mellitus - pathology; Epithelium - innervation; Female; Humans; intraepithelial nerve fiber density; Middle Aged; Mouth Mucosa - innervation; Peripheral Nervous System - pathology; Peripheral Nervous System - physiopathology; peripheral neuropathy; Pilot Projects; Psychophysiology; small fiber neuropathy; Tongue - innervation; Trigeminal Nerve - pathology; Trigeminal Nerve - physiopathology; small fiber neuropathy; intraepithelial nerve fiber density; burning mouth syndrome; peripheral neuropathy
• ISSN: 1354-523X; 1601-0825; 1601-0825
• DOI: 10.1111/odi.12454

Objective The pathophysiology of primary burning mouth syndrome (BMS) has remained enigmatic, but recent studies suggest pathology within the nervous system at multiple levels. This study aimed to investigate in detail the contribution of either focal or generalized alterations within the peripheral nervous system (PNS) in the etiopathogenesis of BMS. Subjects and methods Intraepithelial nerve fiber density (IENFD) of tongue mucosa was assessed in 10 carefully characterized BMS, and the results were compared to 19 age‐ and gender‐matched cadaver controls, 6 with lifetime diabetes. Extensive neurophysiologic and psychophysical examinations of the trigeminal system and distal extremities were performed to profile PNS function in BMS. Results Patients with BMS had significantly fewer intraepithelial nerve fibers (0,27, s.e. 0,18 mm−1; P = 0.0253) than non‐diabetic controls (0,92, s.e. 0,15 mm−1). In the subepithelial space, the amount of nerve fibers did not differ between the groups. The majority (9/10) of patients with BMS showed neurophysiologic or psychophysical signs of a more generalized PNS dysfunction. Conclusions Our results in neurophysiologically optimally characterized BMS patients confirm that pure focal small fiber neuropathy of the oral mucosa has a role in the pathophysiology of primary BMS. Furthermore, BMS may be related to a more generalized, yet subclinical peripheral neuropathy.