The first known description of keloid was found in Smith Papyrus derived from ancient Egypt circa 3000 BC.12 The term keloid derives from the Greek word cheloid, chele (ηλη) meaning a crab’s claw and the suffix-oid, meaning like.4 Keloids are benign dermal fibroproliferative tumors13 that develop after the dermis experiences local trauma such as surgery, burns, laceration, tattoos, and infections.14 However, abnormal scarring remains poorly understood and is a consequence of surgical and traumatic wounds.15 Keloids are reported to be more frequent in certain ethnic groups and has an incidence of 15%–20% in the black population.16 Furthermore, these unique scars have been reported in patients with hereditary connective tissue disorders such as Ehlers–Danlos syndrome, in which keloids manifest as one of the clinical indicators.17 Keloid scar may develop anywhere; however, the ear is a common site for keloid formation, and the scar usually occurs after trauma or ear piercing,18although there is limited data available for the treatment of helical rim keloids.19 The ear is reported to be a region with a propensity for the development of keloidal BCCs and is a site that is prone to the development of keloids in certain individuals.10,18 This may be related to the frequent piercing of earlobes and the trauma and infection that can often ensue.
Requena et al5 described a keloidal BCC for its striking and distinctive features, and from a clinical pathological basis, as a variant of a BCC deemed to be rare.10 However, this description has been refuted by others10 with a study identifying that keloid BCCs are not as rare as originally stated and therefore do not characterize a distinctive clinicopathological variant.10 Jones et al10 state that keloid BCCs are found in different histological kinds of BCCs with varying appearances. They found 1.6% of all BCCs had keloidal collagen in the stroma.10 Misago9 who reported a case of a keloidal BCC, also found the stroma characteristically demonstrates the prominent keloidal, thickened collagen bundles and well-circumscribed keloidal collagen bundles that proliferated in a nodular form. Subsequently, it has been suggested that keloidal stromal reaction is due to local inflammatory changes secondary to necrosis or ulceration.10 Furthermore, it has been suggested there may be a correlation between keloid BCCs and the ear as a site for the development of keloidal stroma.10,20 However, the tumor described in this case, in contrast to a keloidal BCC, is rare and to our knowledge has never been reported in the literature. It is unique in that the common pathological process of a BCC developing necrosis and ulceration, which in turn cause inflammation and keloid scar formation, is reversed. In this case, we presented a keloid scar that has been dormant for 2 years, which improved under conservative treatment, and then underwent malignant transformation to a BCC. Since keloid scars can be considered a tumor, we in fact present a secondary tumor (BCC) which arises on top of a primary tumor (keloid scar). Furthermore, He et al13 and Meade et al21 demonstrate another example of unusual keloid behavior of eruptive keloids associated with cancer and the clinical importance of giving long-term dynamic consideration when following a keloid patient.
Our case study highlights the development of a BBC arising within a keloid scar to the auricle region after an ear piercing 2 years prior. Keloidal characteristics often occur on the ear; however, there remain no reports of such a case within the literature. Given the sparse amount of evidence available, it is important for clinicians to understand and identify key keloidal features in BCCs and to reinforce the association of morphoeiform patterns of growth, ulceration, and necrosis as described by Jones et al10 Therefore, the authors stress the importance of considering early biopsy in any rapidly growing or changing keloidal scar.
The authors would like to thank the patient for her cooperation.
The authors report no conflicts of interest in this work.
Maya Goder,1,* Rachel Kornhaber,2,* Daniele Bordoni,3 Eyal Winkler,1 Josef Haik,1 Ariel Tessone1
1Department of Plastic and Reconstructive Surgery, Sheba Medical Center, Tel Hashomer, Israel;2School of Health Sciences, Faculty of Health, University of Tasmania, Sydney, NSW, Australia;3Department of Senology, Ospedale Santa Maria della Misericordia Urbino, Urbino, Italy
*These authors contributed equally to this work
1. Chung S. Basal cell carcinoma. Arch Plast Surg. 2012;39(2):166–170.
2. Rao J, Deora H. Surgical excision with forehead flap as single modality treatment for basal cell cancer of central face: single institutional experience of 50 cases. J Skin Cancer. 2014;2014:1–5.
3. Lim K-R, Cho K-H, Hwang S-M, Jung Y-H, Kim Song J. Basal cell carcinoma presenting as a hypertrophic scar.Arch Plast Surg. 2013;40(3):289–291.
4. Sand M, Sand D, Brors D, Altmeyer P, Mann B, Bechara FG. Cutaneous lesions of the external ear. Head Face Med. 2008;4(2):1–13.
5. Requena L, Martin L, Farina MC, Pique E, Escalonilla P. Keloidal basal cell carcinoma. A new clinicopathological variant of basal cell carcinoma. Br J Dermatol. 1996;134(5):953–957.
6. Balestri R, Misciali C, Zampatti C, Odorici G, Balestri JA. Keloidal basal cell carcinoma: should it be considered a distinct entity? J Dtsch Dermatol Ges. 2013;11(12):1196–1198.
7. Nagashima K, Demitsu T, Nakamura T, et al. Keloidal basal cell carcinoma possibly developed from classical nodulo-ulcerative type of basal cell carcinoma: report of a case. J Dermatol. 2015;42(4):427–429.
8. Misago N, Ogusu Y, Narisawa Y. Keloidal basal cell carcinoma after radiation therapy. Eur J Dermatol. 2004;14(3):182–185.
9. Misago N. Keloidal basal cell carcinoma. Am J Dermatopathol. 2008;30(1):87.
10. Jones M, Bresch M, Alvarez D, Böer A. Keloidal basal cell carcinoma: not a distinctive clinicopathological entity.Br J Dermatol. 2009;160(1):127–131.
11. Tanaydin V, Beugels J, Piatkowski A, et al. Efficacy of custom-made pressure clips for ear keloid treatment after surgical excision. J Plast Reconstr Aesthet Surg. 2016;69(1):115–121.
12. Wilkins RH. Neurosurgical classic. XVII. J Neurosurg. 1964;21:240–244.
13. He Y, Merin MR, Sharon VR, Maverakis E. Eruptive keloids associated with breast cancer: a paraneoplastic phenomenon? Acta Derm Venereol. 2011;91(4):480–481.
14. De Sousa RF, Chakravarty B, Sharma A, Parwaz MA, Malik A. Efficacy of triple therapy in auricular keloids. J Cutan Aesthet Surg. 2014;7(2):98–102.
15. Del Toro D, Dedhia R, Tollefson TT. Advances in scar management: prevention and management of hypertrophic scars and keloids. Curr Opin Otolaryngol Head Neck Surg. 2016; Epub 2016 May 7.
16. Viera MH, Vivas AC, Berman B. Update on keloid management: clinical and basic science advances. Adv Wound Care. 2012;1(5):200–206.
17. Halim AS, Emami A, Salahshourifar I, Kannan TP. Keloid scarring: understanding the genetic basis, advances, and prospects. Arch Plast Surg. 2012;39(3):184–189.
18. Shin JY, Lee JW, Roh SG, Lee NH, Yang KM. A comparison of the effectiveness of triamcinolone and radiation therapy for ear keloids after surgical excision: a systematic review and meta-analysis. Plast Reconstr Surg. 2016;137(6):1718–1725.
19. Park TH, Rah DK. Successful eradication of helical rim keloids with surgical excision followed by pressure therapy using a combination of magnets and silicone gel sheeting. Int Wound J. 2015. Epub 2015 November 23.
20. Slemp AE, Kirschner RE. Keloids and scars: a review of keloids and scars, their pathogenesis, risk factors, and management. Curr Opin Pediatr. 2006;18(4):396–402.
21. Meade C, Smith S, Makhzoumi Z. Eruptive keloids associated with aromatase inhibitor therapy. JAAD Case Rep. 2015;1(3):112–113.
Source: OncoTargets and Therapy
Originally published August 2, 2016.