Fibrofolliculomas in Birt-Hogg-Dubé Syndrome treated with a CO2 and dye laser combination: a case report and literature review


Submitted: 14 June 2024
Accepted: 28 September 2024
Published: 17 October 2024
Abstract Views: 168
PDF: 59
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Authors

The Birt-Hogg-Dubé Syndrome (BHDs) can cause benign skin lesions like fibrofolliculomas as well as additional systemic symptoms like pulmonary cysts, kidney tumors, and recurrent pneumothoraces.

We report the successful treatment of fibrofolliculomas in a patient with BHDs using a combination of fractionated ablative CO2 laser and a Flash Lamp Pulsed Dye Laser (FPDL). This combined method improves the CO2 healing process's functional and cosmetic results; while the first wavelength (ablative) has a greater capacity for excision and tissue regeneration, the second wavelength (non-ablative) provides more control over the tissue healing process. The lesions were successfully ablated up to the papillary dermis without leading to an enhancement in the overall textural appearance. Therefore, deeper laser ablations may be able to stop dermal residual lesion relapses.

In conclusion, fractionated CO2 laser and FPDL can be used safely and effectively to reduce the appearance of facial hamartomas related to BHDs.


Nickerson ML, Warren MB, Toro JR, et al. Mutations in a novel gene lead to kidney tumors, lung wall defects, and benign tumors of the hair follicle in patients with the Birt-Hogg- Dubè syndrome. Cancer Cell. 2002;2:157-64. DOI: https://doi.org/10.1016/S1535-6108(02)00104-6

Sattler EC, Steinlein OK. Delayed diagnosis of Birt–Hogg–Dubé syndrome due to marked intrafamilial clinical variability: a case report. BMC Med Genet. 2018;19:45. DOI: https://doi.org/10.1186/s12881-018-0558-0

Menko FH, van Steensel MA, Giraud , et al. European BHD Consortium. Birt-Hogg-Dubé syndrome: diagnosis and management. Lancet Oncol. 2009;10:1199-206. DOI: https://doi.org/10.1016/S1470-2045(09)70188-3

Reese E, Sluzevich J, Kluijt I, et al. Birt-Hogg-Dubé Syndrome. 2009. Available from: https://www.ncbi.nlm.nih.gov/books/NBK45326/

Toro JR, Glenn G, Duray P, et al. Birt–Hogg–Dubé syndrome: a novel marker of kidney neoplasia. Arch Dermatol. 1999;135:1195-202. DOI: https://doi.org/10.1001/archderm.135.10.1195

Daccord C, Good J-M, Morren M-A, et al. Birt-Hogg-Dubé syndrome. Eur Respir Rev. 2020;29:200042. DOI: https://doi.org/10.1183/16000617.0042-2020

Expert Consensus Group of the Expert Consensus on the Diagnosis and Management of Birt-Hogg-Dubé Syndrome, China Alliance for the Rare Lung Disease; Chinese Thoracic Society, et al. Expert consensus on the diagnosis and management of Birt-Hogg-Dubé syndrome. Zhonghua Jie He He Hu Xi Za Zhi. 2023;46:897-908.

Gijezen LM, Vernooij M, Martens H, et al. Topical rapamycin as a treatment for fibrofolliculomas in Birt-Hogg-Dube syndrome: a double-blind placebo-controlled randomized split-face trial. PLoS One. 2014;9:e99071. DOI: https://doi.org/10.1371/journal.pone.0099071

Vazirnia A, Schneider J, Ortiz A. Treatment of Benign Adnexal Tumors in Birt-Hogg-Dubé Syndrome With Surgical Debulking in Combination With Fractional and Fully Ablative Carbon Dioxide Laser Resurfacing. Dermatol Surg. 2020;46:1464-5. DOI: https://doi.org/10.1097/DSS.0000000000002103

Lodi G, Sannino M, Caterino P, et al. Fractional CO2 laser-assisted topical rifamycin drug delivery in the treatment of pediatric cutaneous leishmaniasis. Pediatr Dermatol. 2021;38:717-20. DOI: https://doi.org/10.1111/pde.14608

Gambichler T, Wolter M, Altmeyer P, Hoffman K. Treatment of Birt-Hogg-Dubé syndrome with erbium:YAG laser. J Am Acad Dermatol. 2000;43:856-8. DOI: https://doi.org/10.1067/mjd.2000.109294

Kim D, Wysong A, Teng JM, Rahman Z. Laser-Assisted Delivery of Topical Rapamycin: mTOR Inhibition for Birt-Hogg-Dube Syndrome. Dermatol Surg. 2019;45:1713-5. DOI: https://doi.org/10.1097/DSS.0000000000001778

Patel R, Wesenberg J, Brammeier J. Fibrofolliculomas in Birt-Hogg-Dubé syndrome treated with nonfractionated ablative CO2 laser. JAAD Case Rep. 2023;40:96-8. DOI: https://doi.org/10.1016/j.jdcr.2023.08.026

Wu J, Del Duca E, Espino M, et al. RNA Sequencing Keloid Transcriptome Associates Keloids With Th2, Th1, Th17/Th22, and JAK3-Skewing. Front Immunol. 2020;11:597741. DOI: https://doi.org/10.3389/fimmu.2020.597741

Seago M, Shumaker PR, Spring LK, et al. Laser Treatment of Traumatic Scars and Contractures: 2020 International Consensus Recommendations. Lasers Surg Med. 2020;52:96-116. DOI: https://doi.org/10.1002/lsm.23201

Nisticò SP, Bennardo L, Sannino M, et al. Combined CO2 and dye laser technique in the treatment of outcomes due to flap necrosis after surgery for basal cell carcinoma on the nose. Lasers Surg Med. 2022;54:523-9. DOI: https://doi.org/10.1002/lsm.23502

Lodi G, Sannino M, Cannarozzo G, et al. Pulsed dye laser prior to CO2 laser ablation to treat multiple cutaneous neurofibromas in von Recklinghausen's disease: a case report. Dermatol Ther. 2022;35:e15355. DOI: https://doi.org/10.1111/dth.15355

Domenico P, Giuliana C, Daniele B, et al. Ear keloids: An innovative 3-steps combined treatment. Skin Res Technol. 2023;29:e13506. DOI: https://doi.org/10.1111/srt.13506

Sannino M, Ambrosio AG, Lodi G, et al. A giant epidermal nevus of the face treated with a CO2 and dye laser combination: a case report and literature review. J Cosmet Laser Ther. 2021;23:59-64. DOI: https://doi.org/10.1080/14764172.2021.1990962

Cortonesi G, Donelli C, Orsini C, et al. Combination of CO2 laser therapy and pulsed dye laser therapy for the treatment of actinic cheilitis. J Cosmet Dermatol. 2023;22:715-6. DOI: https://doi.org/10.1111/jocd.15494

Lodi, G., Cannarozzo, G., Sannino, M., Pieri, L., Fusco, I., & Negosanti, F. (2024). Fibrofolliculomas in Birt-Hogg-Dubé Syndrome treated with a CO2 and dye laser combination: a case report and literature review. Dermatology Reports. https://doi.org/10.4081/dr.2024.10066

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