Funding source: This work was supported by program for Changjiang Scholars and Innovative Teams in Universities, Ministry of Education, China (IRT0760).
Multiple cutaneous malignancies and cherry hemangiomas in a vitiligo patient treated with topical nitrogen mustard
Article first published online: 10 APR 2013
© 2013 Wiley Periodicals, Inc.
Volume 27, Issue 1, pages 52–54, January/February 2014
How to Cite
Zhu, L.-l., Zheng, S., Wei, H., Hong, Y.-x., Zhang, L., Zhang, L., Chen, H.-D. and Gao, X.-H. (2014), Multiple cutaneous malignancies and cherry hemangiomas in a vitiligo patient treated with topical nitrogen mustard. Dermatologic Therapy, 27: 52–54. doi: 10.1111/dth.12045
Conflict of interest: None declared.
- Issue published online: 6 FEB 2014
- Article first published online: 10 APR 2013
- program for Changjiang Scholars and Innovative Teams in Universities, Ministry of Education, China. Grant Number: IRT0760
- Innovative Teams in Universities, Bereau of Education, Liaoning Province, China. Grant Number: LT2011012
- nitrogen mustard;
Topical nitrogen mustard used to be a treatment option for vitiligo as it accentuates pigmentation by disaggregating melanosomes and increasing their numbers within melanocytes . However, chronic exposure to nitrogen mustard increases the risk of cutaneous malignancies. Topical nitrogen mustard was reported to induce basal cell carcinoma (BCC), squamous cell carcinoma (SCC), keratoacanthoma, actinic keratosis, and lentigo maligna . In addition, topical nitrogen mustard was also found to be associated with cherry hemangiomas [3, 4].
Here, the present authors report that a vitiligo patient developed multiple cutaneous malignancies and cherry hemangiomas after over 20 years of applying topical nitrogen mustard.
A 40-year-old man presented with a 30-year history of depigmented skin on the face, neck, trunk, genitalia, and extremities. He was diagnosed with universal vitiligo and had been treated with topical nitrogen mustard (50 mg nitrogen mustard dissolved into 100 mL alcohol) since 1990. Although certain repigmentation was provoked, the depigmentation continued to spread during the course of treatment. Erythematous papules and plaques within or around the pigmented lesions, as well as ruby-red papules on the trunk, were first observed in 2006 and progressively increased in number. The patient denied any other past medical history. His family and social histories were noncontributory except that his only daughter was diagnosed to suffer from localized vitiligo in 2005.
Physical examination revealed mottled depigmented and hyperpigmented macules and patches on his neck, trunk, genitalia, and extremities. Over 40 red papules and plaques with a diameter of 0.5–2 cm were distributed within or around the pigmented skin, and most of the skin lesions were covered with keratin scales. There were more than 200 ruby-red, dome-shaped papules with a diameter of 1–3 mm scattered within or around the depigmented patches of the trunk. Five biopsies were taken from the suspicious lesions (FIG. 1D–F). The biopsy from the abdomen revealed interwoven strands of basaloid cells with peripheral palisading emanating from the epidermis, suggesting of superficial BCC (FIG. 1D). The remaining four specimens from the flexor aspects of lower legs showed similar features, manifested as slight epidermal hyperplasia with sharp demarcation from neighboring normal epidermis, numerous disoriented squamous cells with irregular nuclear outline, and hyperchromatic nuclei in the lower half of the epidermis. The upper epidermis consisted of bland-looking squamous cells with relatively pale cytoplasm compared with those of the surrounding normal keratinocytes. Overlying compact hyperkeratosis with focal parakeratosis was also present. These features were consistent with SCC in situ (FIG. 1E). A ruby-red papule on the trunk was also biopsied and historically confirmed to be cherry hemangioma (FIG. 1F).
The patient was informed of the malignant nature of his skin lesions and asked to discontinue the application of nitrogen mustard. He was treated with imiquimod (three times weekly use) for his biopsy-proved and suspicious neoplastic lesions, and followed up monthly at the outpatient clinic.
Topical nitrogen mustard was known to induce epithelial cancers in both sun-exposed and -protected areas because of its direct damaging effect on DNA . Grandinetti and Fowler  and Ma et al.  reported that topical application of nitrogen mustard was associated with the development of cherry hemangioma . Our patient presented concurrently with three types of skin lesions: BCC, SCC, and cherry hemangiomas after a 20-year use of nitrogen mustard. To our best knowledge, this is the first case reported of such kind with various types of skin tumors in a single patient treated with nitrogen mustard. Our case further confirmed the risk of long-term use of nitrogen mustard in induction of cutaneous malignancies and cherry hemangioma as well.
Financial support: This work was supported by a program for Innovative Teams in Universities, Bereau of Education, Liaoning Province, China (LT2011012).