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adipocyte tumors on imaging, however, 29% of SCLs show no radiological evidence of fat.4 Therefore, SCLs may be radiologically difficult to distinguish from, for instance, liposarcoma.4

the overlying skin. The patient gave his consent for possible need for re-resection and reconstruction in case of malignant histological findings which in the end proved unnecessary.

Because of the limitations of radiological differentiation, the diagnosis usually relies on histological examination. Nevertheless, biopsy alone is often insufficient for a correct diagnosis. Microscopically, SCLs are circumscribed lesions composed of mature fat cells mixed with spindle cells. On immunohistochemistry, the spindle cells express CD34 and are negative for S100 protein.2 Importantly, liposarcomas can have areas resembling SCL, and thus, these lesions should always be extensively sampled to rule out malignancy.1,3

Conclusion

In the patient, a diagnostic dilemma occurred because the lesion was recurrent and progressive, covered by irregular skin (likely caused by previous inconclusive biopsy) and MRI findings were aspecific. Although unlikely, a malignancy such as liposarcoma could not be ruled out.

1. de Bree E, Karatzanis A, Hunt JL, Strojan P, et al. Lipomatous tumours of the head and neck: a spectrum of biological behaviour. Eur Arch Otorhinolaryngol 2015;272:1061–77.

The therapeutic dilemma that followed was whether or not to include the overlying skin in the resection. Such an oncological approach would result in a partial-thickness defect in a functional and esthetic area of the nose requiring a secondary stage reconstruction, with for example, a full-thickness skin graft or nasofacial groove flap. This approach, however, believed as overtreatment, and it was therefore decided to attempt an excision of the lesion sparing

A spindle cell lipoma inside the nasal ala is very rare. Given its frequent presentation as a lesion with little or no fat, it is not easily recognized with biopsy and MRI. Total surgical excision is the treatment of choice. A vestibular incision provides excellent exposure for resection, without long-term functional and aesthetic sequellae.

References

2. Cheah A, Billings S, Goldblum J, Hornick J, et al. Spindle cell/ pleomorphic lipomas of the face: an under-recognized diagnosis. Histopathology 2015;66:430–7. 3. Khashper A, Zheng J, Nahal A, Discepola F. Imaging characteristics of spindle cell lipoma and its variants. Skeletal Radiol 2014;43:591–7. 4. Choi JW, Kim HJ, Kim J, Kim HJ, et al. Spindle cell lipoma of the head and neck: CT and MR imaging findings. Neuroradiology 2013;55:101–6.

Catharine A. Hellingman, MD, PhD Frank R. Datema, MD, PhD Department of Otolaryngology/Head and Neck Surgery, Erasmus Medical Center, Rotterdam

The authors have indicated no significant interest with commercial supporters.

Eruptive Keratoacanthomas After Photodynamic Therapy Photodynamic therapy (PDT) is being increasingly used by dermatologists because of its safety, efficacy, and excellent cosmetic results.1 PDT is currently approved by the Food and Drug Administration for the treatment of actinic keratosis (AK); however, it is

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often used off label for the treatment of superficial nonmelanoma skin cancers (NMSC), photoaging, acne, and verrucas.1 The therapeutic effect of PDT is achieved by interactions between a photosensitizing agent, a specific wavelength of light (blue or red

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light), and oxygen.1 Typical photosensitizers used are aminolevulinic acid (ALA) and methylaminolevulinate (mALA), which are applied to the skin and left to incubate before treatment with blue or red light. The result is selective destruction of premalignant atypical cells. Commonly reported side effects are pain, burning, erythema, erosion, crust formation, and ulceration.1 To date, there has been 1 case report of a single keratoacanthoma after PDT and 2 reports of melanoma after PDT and squamous cell carcinoma after PDT and 5-fluorouracil.2,3 To the authors’ knowledge, this is the first case of eruptive keratoacanthomas after the use of only PDT, illustrating a rare complication after PDT. This has not been previously reported as a side effect, and the etiology is unknown. A 71-year-old man with a history of seborrheic dermatitis, malignant melanoma in situ, numerous NMSC, and multiple AKs on the bilateral forearms was treated with PDT (ALA with blue light). At posttreatment follow-up, 3 weeks later, he had 20 to 30 painful and pruritic, heme-crusted, red papules, some with a central crater filled with keratin on the bilateral distal forearms, causing significant discomfort (Figure 1). The patient first noticed the papules 2 to 3 days after treatment and that some of the more proximal lesions had resolved spontaneously. All papules were well confined to the treatment area. Of note, he had 2 keratoacanthomas on his right forearm treated with electrodessication and curettage 2.5 weeks before his PDT. He denied any systemic symptoms such as fever, adenopathy, nausea, vomiting, diarrhea, fatigue, or unexplained weight loss. Biopsy confirmed the clinical diagnosis of keratoacanthoma. As some lesions were self-resolving, the authors elected to closely monitor the patient and watch for complete resolution. Five weeks after PDT, there were several scattered persistent keratoacanthomas noted on examination (Figure 2). Treatment options were discussed. He was treated with acitretin 25 mg orally daily and weekly intralesional 5-fluorouracil (5-FU) to the unresolving plaques and papules. The concentration of the 5-FU was 50 mg/mL with 0.1 to 0.4 mL injected into each lesion, depending on the

Figure 1. Pruritic, heme-crusted, red papules 3 weeks after PDT.

size. After several weeks of acitretin and 1 treatment of intralesional 5-FU, the papules and plaques had nearly resolved (Figures 3 and 4). Complete clearance was observed after 5 additional treatments with

Figure 2. Five weeks after PDT, the patient had a few remaining inflammatory papules and plaques on bilateral arms.

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Figure 3. Resolving papules and plaques to the left arm after several weeks of oral acitretin and the 1st of 6 injections with intralesional 5-FU.

Figure 4. Resolving papules and plaques to the right arm after several weeks of oral acitretin and the 1st of 6 injections with intralesional 5-FU.

intralesional 5-FU. He continues on acitretin as chemoprophylaxis given his history of numerous NMSC and actinic damage.

PDT and laser ablation has been used as a treatment of typical keratoacanthoma.4

Discussion This patient has a long history of numerous AK to his bilateral arms. Previous treatments include topical 5fluorouracil (5-FU), chemical peels, and cryotherapy. Given the persistence of his AK, his arms were treated with PDT. PDT plays an important role when there are multiple and/or confluent AKs, sites of poor wound healing, and/or a poor response to other topical therapies, as was the case with this patient.2 In a Cochrane review, PDT demonstrated higher efficacy along with more cosmetically pleasing outcomes than with cryotherapy or topical 5-FU.1 PDT can cause photosensitivity side effects such as pain, edema, and erythema at the treatment sites.2 This case is unique in that the patient experienced multiple eruptive keratoacanthomas after PDT. Ironically,

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Keratoacanthoma is a squamoproliferative neoplasm that some consider a variant of squamous cell carcinoma.5 Keratoacanthomas appear as 1- to 2-cm dome-shaped crateriform papules with central keratin and have been associated with excessive sun exposure and other forms of trauma such as surgery, skin grafting, electrodessication and curettage, laser resurfacing, and chemical carcinogens.3,5 The incidence of developing keratoacanthomas peaks in middle age, typically occurring during the sixth and seventh decades of life.3 Keratoacanthomas have been noted to appear within lesions of hypertrophic lichen planus, discoid lupus erythematosus, psoriasis, herpes zoster, and epidermolysis bullosa dystrophica.3 Keratoacanthomas are also associated with familial syndromes such as familial keratoacanthomas of Ferguson-Smith, the multiple familial

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keratoacanthomas of Witten and Zak and Muir– Torre syndrome, a form of Lynch syndrome that is associated with sebaceous tumors, gastrointestinal malignancy, and keratoacanthomas.3–5

toacanthomas as an adverse reaction to PDT. This adverse event may be more likely if multiple treatment modalities have failed previously, although it is rare.

Generalized eruptive keratoacanthoma variant must also be considered. Although there is no set criteria to diagnose generalized eruptive keratoacanthoma, suggested proposed criteria include abrupt onset, older age, significant pruritus, hundreds to thousands of generalized follicular papules, and a progressive course.4

References

Exposure to UV light has been proven to cause cutaneous neoplasms, including keratoacanthoma, in humans.3 UV-induced mutations accumulate in the skin with chronic exposure; this, combined with the individual’s inability to repair the cellular damage, predisposes the mutated cells to become tumors.3 Keratoacanthomas have been known to develop after excessive exposure to ultraviolet light and in the irradiated sites of treated basal cell epitheliomas. Eruptive KAs are also a potential side effect secondary to CO2 laser resurfacing.5 The trauma that PDT can cause to the follicle and its development may represent a possible mechanism for the eruption of keratoacanthomas after PDT.3 The patient tried multiple therapeutic modalities before treatment with PDT. This supports the belief postulated by some authors that trauma may promote tumor development in actinically damaged skin. It is unclear whether these injurious treatments may have predisposed the patient to this response, or whether the PDT itself specifically created this unique reaction. Conclusion This case cautions dermatologists of a potential complication of PDT when treating AK. The authors propose that the patient developed eruptive kera-

1. Wan MT, Lin JY. Current evidence and applications of photodynamic therapy in dermatology. Clin Cosmet Investig Dermatol 2014;7:145–63. 2. Morton CA, Szeimies RM, Sidoroff A, Braathen LR. European guidelines for topical photodynamic therapy part 1: treatment delivery and current indications—actinic keratoses, Bowen’s disease, basal cell carcinoma. J Eur Acad Dermatol Venereol 2013; 27:536–44. 3. Mamelak AJ, Goldberg LH, Marquez D, Hosler GA, et al. Eruptive keratoacanthomas on the legs after fractional photothermolysis: report of two cases. Dermatol Surg 2009;35:513–8. 4. Nofal A, Assaf M, Nofal E, Alradi M. Generalized eruptive keratoacanthoma: proposed diagnostic criteria and therapeutic evaluation. J Eur Acad Dermatol Venereol 2014;28: 397–404. 5. Gewirtzman A, Meirson DH, Rabinovitz H. Eruptive keratoacanthomas following carbon dioxide laser resurfacing. Dermatol Surg 1999;25: 666–8.

Mary Ramirez, MS Texas Tech University Health Sciences Center School of Medicine Lubbock, Texas Sarah Groff, MD Division of Dermatology University of Texas Health Sciences Center at San Antonio San Antonio, Texas Catherine Kowalewski, DO Division of Dermatology University of Texas Health Sciences Center at San Antonio and South Texas Veterans Health Care System San Antonio, Texas

The authors have indicated no significant interest with commercial supporters.

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