Diffuse mucocutaneous hyperpigmentation related to hydroxyurea

Sharma, Vishnu; Mahajan, Sidharth; Bagrodia, Vansh

doi:10.1016/j.htct.2024.03.009

Case presentation

A 63-year-old woman developed diffuse mucocutaneous hyperpigmentation post-hydroxyurea treatment for myelofibrosis. Hyperpigmentation appeared on eyelids, nasal ala, tongue, and palms without discomfort. Extensive investigations ruled out common causes; the patient had no prior dermatological conditions. Hydroxyurea was deemed causative due to temporal correlation. Counselling emphasized benign nature, recommending continued therapy with monitoring.

Hydroxyurea-induced skin changes include ulcerations, melanonychia, and hyperpigmentation.¹ Mechanism involves photosensitization, toxicity, genetic factors, possibly increased melanin, and iron deposition.², ³, ⁴ Management includes reassurance, differential evaluation, and discontinuation in severe cases.⁵ This case differs from typical singular-site presentations, presenting a rare, multifocal pattern.¹, ³, ⁴, ⁵, ⁶ Figures 1 and 2.

Figure 1
Bilateral periorbital, nasal ala, and lingual pigmentation.

Figure 2
Diffuse macular palmar pigmentation.

REFERENCES

¹ Veillet-Lemay G, Haber RM. Hydroxyurea-induced oral hyper-pigmentation: a case report and review of the literature. J Cutan Med Surg. 2019;23(1):111–3.
² Alawi F. Pigmented lesions of the oral cavity: an update. Dent Clin North Am. 2013;57(4):699–710.
³ Lee KP, Vangipuram R, Klimas NK, Sanyal S, Koshelev MV. Hydroxyurea-induced hyperpigmentation with iron deposition. Dermatol Online J. 2019 Oct 15;25(10). 13030/qt6q51c0p94.
⁴ Karanth SS, Gupta A, Prabhu M. Melanonychia and mucocutaneous hyperpigmentation from hydroxyurea use for the treatment of essential thrombocytosis. Singapore Med J. 2014;55:e7–8.
⁵ Sukhedo K, Yoon GH, Rothman L, Meehan SA, Levin MK, Kim RH. Mirtazapine-induced hyperpigmentation with type II histopathologic findings. JAAD Case Rep. 2018;4:1077–9. [PMID:30511003].
⁶ Alshammasi B, Albasry Z, Meshikhes F. Oral hyperpigmentation associated with hydroxyurea in a patient with polycythemia vera: a case report. Clin Case Rep. 2020;8(10):1904–9. https://doi.org/10.1002/ccr3.3012 PMID: 33088516; PMCID:PMC7562833.
» https://doi.org/10.1002/ccr3.3012

Publication Dates

Publication in this collection
03 Feb 2025
Date of issue
2024

History

Received
08 Mar 2024
Accepted
19 Mar 2024
Published
22 July 2024

This is an Open Access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

[1] ¹ Veillet-Lemay G, Haber RM. Hydroxyurea-induced oral hyper-pigmentation: a case report and review of the literature. J Cutan Med Surg. 2019;23(1):111–3.

[2] ² Alawi F. Pigmented lesions of the oral cavity: an update. Dent Clin North Am. 2013;57(4):699–710.

[3] ³ Lee KP, Vangipuram R, Klimas NK, Sanyal S, Koshelev MV. Hydroxyurea-induced hyperpigmentation with iron deposition. Dermatol Online J. 2019 Oct 15;25(10). 13030/qt6q51c0p94.

[4] ⁴ Karanth SS, Gupta A, Prabhu M. Melanonychia and mucocutaneous hyperpigmentation from hydroxyurea use for the treatment of essential thrombocytosis. Singapore Med J. 2014;55:e7–8.

[5] ⁵ Sukhedo K, Yoon GH, Rothman L, Meehan SA, Levin MK, Kim RH. Mirtazapine-induced hyperpigmentation with type II histopathologic findings. JAAD Case Rep. 2018;4:1077–9. [PMID:30511003].

[6] ⁶ Alshammasi B, Albasry Z, Meshikhes F. Oral hyperpigmentation associated with hydroxyurea in a patient with polycythemia vera: a case report. Clin Case Rep. 2020;8(10):1904–9. https://doi.org/10.1002/ccr3.3012 PMID: 33088516; PMCID:PMC7562833.
» https://doi.org/10.1002/ccr3.3012