Abstract
Background
Local application of fluorouracil (Efudix, 5-FU) induces sclerosis in patients with sinonasal tumors and superficial basocellular skin carcinoma. As a ‘back against the wall’ treatment, we investigated the local effect of nasally applied 5-FU and whether this could decrease the burden of severe epistaxis in patients with hereditary hemorrhagic telangiectasia (HHT).
Methods
HHT patients with severe and frequent epistaxis, subsequent anemia and a necessity for blood and/or iron infusions were treated with a nasal tampon with 5-FU. This tampon was placed unilaterally in the nasal cavity on the side of the most severe epistaxis and replaced once weekly during 4 weeks. Outcome measures were safety and side effects, the aspect of the nasal mucosa measured with the mucosal HHT score, the epistaxis severity score (ESS), hemoglobin and ferritin plasma levels, and quality of life assessment pre-treatment, one and three months post-treatment.
Results
Six HHT patients participated. During treatment and follow-up, the nasal mucosa turned more pale and sclerotic and the number of telangiectases diminished. The mucosal HHT score improved and the ESS declined (p = 0.01). The decline of ESS persisted up to 3 months post-5-FU treatment. Moreover, mean hemoglobin levels increased from 6.0 pre-5-FU to 6.8 after one month post-5-FU.
Conclusion
Unilateral application of 5-FU on a nasal tampon diminished the severity and frequency of epistaxis in all HHT patients. This effect sustained up to three months post-treatment, despite the fact that the contralateral side remained untreated. Subsequently, hemoglobin levels increased. Intranasal 5-FU is a promising entity for further research on epistaxis treatment in HHT patients.
References
Shovlin CL (2010) Hereditary haemorrhagic telangiectasia: pathophysiology, diagnosis and treatment. Blood Rev 24:203–219. https://doi.org/10.1016/j.blre.2010.07.001
Mahoney EJ, Shapshay SM (2006) New classification of nasal vasculature patterns in hereditary hemorrhagic telangiectasia. Am J Rhinol 20:87–90. https://doi.org/10.1177/194589240602000116
Geirdal AØ, Dheyauldeen S, Bachmann-Harildstad G, Heimdal K (2012) Quality of life in patients with hereditary hemorrhagic telangiectasia in Norway: a population based study. Am J Med Genet Part A 158A:1269–1278. https://doi.org/10.1002/ajmg.a.35309
Chin CJ, Rotenberg BW, Witterick IJ (2016) Epistaxis in hereditary hemorrhagic telangiectasia: an evidence based review of surgical management. J Otolaryngol Head Neck Surg 45:3. https://doi.org/10.1186/s40463-016-0116-8
Bulstrode NW, Mudera V, McGrouther DA et al (2005) 5-fluorouracil selectively inhibits collagen synthesis. Plast Reconstr Surg 116:209–221. https://doi.org/10.1097/01.prs.0000169701.16509.d6. discussion 222-3
Prince GT, Cameron MC, Fathi R, Alkousakis T (2018) Topical 5-fluorouracil in dermatologic disease. Int J Dermatol 57:1259–1264. https://doi.org/10.1111/ijd.14106
Knegt PP, Ah-See KW, Vd Velden L-A, Kerrebijn J (2001) Adenocarcinoma of the ethmoidal sinus complex. Arch Otolaryngol Neck Surg 127:141. https://doi.org/10.1001/archotol.127.2.141
Almeyda R, Capper J (2008) Is surgical debridement and topical 5 fluorouracil the optimum treatment for woodworkers’ adenocarcinoma of the ethmoid sinuses? A case-controlled study of a 20-year experience. Clin Otolaryngol 33:435–441. https://doi.org/10.1111/j.1749-4486.2008.01790.x
Hoag JB, Terry P, Mitchell S et al (2010) An epistaxis severity score for hereditary hemorrhagic telangiectasia. Laryngoscope. https://doi.org/10.1002/lary.20818
Funding
No funding was secured for this treatment, and there is no financial disclosure.
Author information
Authors and Affiliations
Corresponding author
Ethics declarations
Conflict of interest
The authors declare no conflict of interest.
Additional information
Publisher's Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Electronic supplementary material
Below is the link to the electronic supplementary material.
Rights and permissions
About this article
Cite this article
de Jel, D.V.C., Disch, F.J.M., Kroon, S. et al. Intranasal Efudix reduces epistaxis in hereditary hemorrhagic telangiectasia. Angiogenesis 23, 271–274 (2020). https://doi.org/10.1007/s10456-020-09712-2
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s10456-020-09712-2