Relative size of fungiform papillae in patients with neurofibromatosis Type 1

U S Speth; L Kluwe; M Gosau; R E Friedrich

doi:10.1016/j.jormas.2023.101394

Relative size of fungiform papillae in patients with neurofibromatosis Type 1

J Stomatol Oral Maxillofac Surg. 2023 Jun;124(3):101394. doi: 10.1016/j.jormas.2023.101394. Epub 2023 Jan 24.

Authors

U S Speth¹, L Kluwe², M Gosau², R E Friedrich²

Affiliations

¹ Department of Oral and Maxillofacial Surgery, University Medical Center Hamburg Eppendorf, Hamburg, Germany. Electronic address: ullispeth@gmail.com.
² Department of Oral and Maxillofacial Surgery, University Medical Center Hamburg Eppendorf, Hamburg, Germany.

PMID: 36702351
DOI: 10.1016/j.jormas.2023.101394

Abstract

Objective: To explore possible manifestations of neurofibromatosis on the tongue, especially, enlarged papillae fungiformes. According to the literature, enlarged fungiform papillae of the tongue are a very common oral finding in patients with Neurofibromatosis Type 1 (NF1).

Materials and methods: Firstly, photos of 18 NF1 patients' tongues, taken at different times were compared to rule of possible fluctuating papillae size. Secondly, 60 photos of 60 patients with NF1 were age/sex matched, blinded and compared to 60 photos of 60 healthy patients. Three independent medical doctors rated the photos in regard of the fungiform papillae as smaller/same/larger at two different times. The inter- and intraindividual results were compared. Thirdly, fungiform papillae of 11 NF1 patients were quantitatively measured using the Denver protocol.

Results: The fungiform papillae showed a stable size. The comparison of the healthy individuals to the NF1 patients suggests that the larger papillae are significantly more frequent in NF1 patients than in age- and gender-matched non-NF1 individuals. The agreement between two ratings of each of the 3 raters at different time points was two moderate and one substantial, the agreement among raters was only fair, since the Fleiss' Kappa value of all 6 ratings was 0.38.

Conclusion: Although this study confirms that the evaluation of the size of the fungiform papillae by visual inspection only is very subjective, the fungiform papillae in NF1 patients do seem to be enlarged at a statistically significant level. Nonetheless the statistical difference is not distinctive enough to establish this as a diagnostic tool in diagnosing NF1.

Clinical relevance: Facilitating the diagnosis of NF1 is an important factor in finding the correct treatment for these patients. A clinical inspection of the tongue is a simple and non-invasive procedure. This paper addresses the question if an inspection of the tongue, especially the fungiform papillae is a reliable indicator for the diagnosis of NF1.

Keywords: Fungiform papillae; Intraoral; Neurofibromatosis; Tongue.

MeSH terms

Humans
Neurofibromatosis 1* / complications
Neurofibromatosis 1* / diagnosis
Neurofibromatosis 1* / epidemiology
Taste Buds*
Tongue