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Giant desmoplastic cutaneous squamous cell carcinoma of the gluteal region
World Journal of Surgical Oncology volume 15, Article number: 121 (2017)
Cutaneous squamous cell carcinoma (cSCC) is the most common type of skin tumour with the ability of metastatic spread. It represents about 20% of all malignancies diagnosed worldwide each year. Despite increased knowledge regarding the causes of skin cancer, the incidence of cSCC rises. The disease originates from epidermal keratinocytes, but it may occur on all areas of the body. It has an invasive nature and the potential to metastasise. We report unusual case of a giant metastatic desmoplastic cSCC of the gluteal region in a patient with previously resected desmoplastic cSCC presenting 8 months later with multiple liver and lung metastases.
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A 55-year-old man presented with gradually enlarging pigmented skin lesion since many months. The lesion was located on his left hip measuring 5 × 5 cm and was surgically excised with a free margin of healthy tissue. Histopathological examination of the incisional biopsy showed the entire thickness of the epithelium filled with atypical CK5+, TTF−, CK7− and napsin A− keratinocytes. The tumour cells also demonstrated hyperchromasia, nuclear pleomorphism and increased number of mitosis, all consistent with diagnosis of desmoplastic cutaneous squamous cell carcinoma (cSCC) (Fig. 1a). The patient underwent regular examinations, and his condition remained stable for 8 months. After that period, he presented with growing, inflamed mass in the gluteal region. The metastatic work-up was positive and confirmed a diagnosis of stage IV desmoplastic cSCC with liver and lung metastases (Fig. 1b, c). Clinical and laboratory examinations did not reveal any signs of depleted immune system. The patient refused further treatment and unfortunately died after 2 months as a result of tumour progression (Fig. 1d, e). The ultimate cause of death was acute hepatic failure due to organ invasion by neoplastic cells.
cSCC is the second most common form of non-melanoma skin cancers and accounts for approximately 20% of all cutaneous malignancies. cSCC occurs in men two to three times more frequently than it does in women. Its global incidence varies geographically, from 0.03 to 3.5 cases per 100,000 people per year and seems to have increased over the past 30 years by 50 and up to 200% [1, 2]. In contrast to basal cell carcinoma, cSCC is aggressive malignancy and easily metastasise via hematogenous and lymphogenous routes. cSCC usually occurs on the upper portions of the body, and the main symptom is a wart-like growth that may have a rough, scaly surface with flat reddish patches which occasionally bleeds or it has elevated growth with central depression that persists for weeks and may rapidly increase in size. After the treatment, the overall prognosis for the majority of patients is excellent with an overall 5-year cure rate of greater than 90 and 4.6% rate of recurrence [1, 2]. The metastatic potential has been estimated to range from 2 to 5%, but this estimation should be considered with caution. The presence of distant metastatic disease is associated with median survival of less than 2 years [1, 2]. The most prominent risk factors include exposure to sun or artificial ultraviolet radiation, leukoplakia, Bowen’s disease, infection with human papillomavirus, genetic factors such as mutations in the MC1R gene, genetic defects in DNA repair, therapy with immunosuppressive agents and advanced age [3,4,5]. Mutations in the suppressor gene p53 and RAS gene as well as activation of EGFR are the most common genetic abnormalities found in cSCCs [2, 4,5,6]. The differential diagnosis of cSCC may include keratoacanthoma, basal cell carcinoma, amelanotic melanoma, fibroxanthoma, pseudoepitheliomatous hyperplasia or HPV-induced papillomas . Histopathologically, the subtypes of cSCC include verrucous, spindle, desmoplastic, acantholytic and adenosquamous form . A biopsy or excision of the lesion and the histologic examination including immunohistochemistry, especially cytokeratin AE1/AE3 immunostaining and 3D histology, should be performed in all clinically suspicious lesions .
The cSCC is generally treated by surgical excision, electrodessication and curettage. Non-surgical options for the treatment include cryotherapy, topical chemotherapy, photodynamic therapy, topical immune response modifiers, radiotherapy and systemic chemotherapy . In the present case, the patient was initially diagnosed with desmoplastic cSCC and treated with surgical resection in combination with plastic reconstruction with preservation of function and satisfactory cosmetic results. The remission lasted 8 months. After that period, he was diagnosed with relapsed, poorly differentiated stage IV desmoplastic cSCC which is clinically characterised by a highly infiltrative growth and high metastatic potential (Fig. 1d, e). The probable cause for relapse in this case was incomplete clearence of the micrometastatic cancer cells that surrounded the primary tumour. The palliative radiotherapy and chemotherapy were not indicated according to patient’s own conditions and wishes. However, stage IV cSCC is responsive to a variety of different chemotherapeutic agents, but there is no established standard regimen. Among most common non-targeted agents used are platin salts, 5-fluorouracil or taxanes [2, 8, 9]. Polychemotherapy should be reserved for patients requiring more aggressive management, while otherwise, mono-chemotherapy should be considered as a first-line treatment [2, 8, 9]. A chimeric immunoglobulin G1 monoclonal antibody cetuximab has been reported in the literature as a successful second-line option [9, 10]. Some tyrosine kinase inhibitors (gefitinib, dasatinib, erlotinib) as well as different immunotherapy agents (anti-PD1 antibody, pembrolizumab) demonstrated encouraging results in the treatment of cSCC [11,12,13,14,15,16].
Finally, it is estimated that majority of all cSCC recurrences will develop within 2 years of the initial diagnosis . Therefore, all patients with cSCC should be followed closely with regular follow-up schedule such as every 6 months depending on aggressiveness of the tumour and clinical and histological criteria.
Cutaneous squamous cell carcinoma
Epidermal growth factor receptor
Melanocortin-1 receptor gene
Tumour suppressor gene p53
Thyroid transcription factor
Lomas A, Leonardi-Bee J, Bath-Hextall F. A systemic review of worldwide incidence of nonmelanoma skin cancer. Br J Dermatol. 2012;166:1069–80.
Stratigos A, Garbe C, Lebbe C, Malvehy J, del Marmol V, Pehamberger H, Peris K, Becker JC, Zalaudek I, Saiag P, Middleton MR, Bastholt L, Testori A, Grob JJ, European Dermatology Forum (EDF); European Association of Dermato-Oncology (EADO); European Organization for Research and Treatment of Cancer (EORTC). Eur J Cancer. 2015;51(14):1989–2007.
de Vries E, Trakatelli M, Kalabalikis D, Ferrandiz L, Ruiz-de-Casas A, Moreno-Ramirez D, Sotiriadis D, Ioannides D, Aquilina S, Apap C, Micallef R, Scerri L, Ulrich M, Pitkänen S, Saksela O, Altsitsiadis E, Hinrichs B, Magnoni C, Fiorentini C, Majewski S, Ranki A, Stockfleth E, Proby C, EPIDERM Group. Known and potential new risk factors for skin cancer in European populations: a multicentre case-control study. Br J Dermatol. 2012;167 Suppl 2:1–13.
Boukamp P. Non-melanoma skin cancer: what drives tumor development and progression? Carcinogenesis. 2005;26(10):1657–67.
Ratushny V, Gober MD, Hick R, Ridky TW, Seykora JT. From keratinocyte to cancer: the pathogenesis and modeling of cutaneous squamous cell carcinoma. J Clin Invest. 2012;122(2):464–72.
Forbes SA, Beare D, Boutselakis H, Bamford S, Bindal N, Tate J, Cole CG, Ward S, Dawson E, Ponting L, Stefancsik R, Harsha B, Kok CY, Jia M, Jubb H, Sondka Z, Thompson S, De T, Campbell PJ. COSMIC: somatic cancer genetics at high-resolution. Nucleic Acids Res. 2017;45(D1):D777–83.
Schweinzer K, Kofler L, Bauer J, Metzler G, Breuninger H, Häfner HM. Cytokeratin AE1/AE3 immunostaining and 3D-histology—improvement of diagnosis in desmoplastic squamous cell carcinoma of the skin. Arch Dermatol Res. 2017;309(1):43–6.
Breuninger H, Schaumburg-Lever G, Holzschuh J, Horny HP. Desmoplastic squamous cell carcinoma of skin and vermilion surface: a highly malignant subtype of skin cancer. Cancer. 1997;79:915–19.
Burtness B, Goldwasser MA, Flood W, Mattar B, Forastiere AA, Eastern Cooperative Oncology Group. Phase III randomized trial of cisplatin plus placebo compared with cisplatin plus cetuximab in metastatic/recurrent head and neck cancer: an Eastern Cooperative Oncology Group study. J Clin Oncol. 2005;23(34):8646–54.
Maubec E, Petrow P, Scheer-Senyarich I, Duvillard P, Lacroix L, Gelly J, Certain A, Duval X, Crickx B, Buffard V, Basset-Seguin N, Saez P, Duval-Modeste AB, Adamski H, Mansard S, Grange F, Dompmartin A, Faivre S, Mentré F, Avril MF. Phase II study of cetuximab as first-line single-drug therapy in patients with unresectable squamous cell carcinoma of the skin. J Clin Oncol. 2011;29(25):3419–26.
Lewis CM, Glisson BS, Feng L, Wan F, Tang X, Wistuba II, El-Naggar AK, Rosenthal DI, Chambers MS, Lustig RA, Weber RS. A phase II study of gefitinib for aggressive cutaneous squamous cell carcinoma of the head and neck. Clin Cancer Res. 2012;18(5):1435–46.
Read WL, Brumund KT, Weisman RA, Nguyen AQ. Squamous cell carcinomas of the skin responsive to erlotinib: 5 cases. JAAD Case Rep. 2015;1(3):153–6.
Farshchian M, Nissinen L, Grénman R, Kähäri VM. Dasatinib promotes apoptosis of cutaneous squamous carcinoma cells by regulating activation of ERK1/2. Exp Dermatol. 2017;26(1):89–92.
Winkler JK, Schneiderbauer R, Bender C, Sedlaczek O, Fröhling S, Penzel R, Enk A, Hassel JC. Anti-programmed cell death-1 therapy in nonmelanoma skin cancer. Br J Dermatol. 2016;176(2):498–502.
Seifert TY, Haddad RI, Gupta S, Mehra R, Tahara M, Berger R, Lee SH, Burtness B, Le DT, Heath K, Blum A, Dolles-Filhart M, Emancipator K, Pathiraja K, Cheng JD, Chow LQ. Antitumor activity and safety of pembrolizumab in patients (pts) with advanced squamous cell carcinoma of the head and neck (SCCHN): preliminary results from KEYNOTE-012 expansion cohort. J Clin Oncol. 2015;33:15.
Malaspina TS, Gasparoto TH, Costa MR, de Melo Jr EF, Ikoma MR, Damante JH, Cavasanni KA, Garlet GP, da Silva JS, Campanelli AP. Enhanced programmed death 1 (PD-1) and PD-1 ligand (PD-L1) expression in patients with actinic cheilitis and oral squamous cell carcinoma. Cancer Immunol Immunother. 2011;60(7):965–74.
Nuño-González A, Vicente-Martín FJ, Pinedo-Moraleda F, López-Estebaranz JL. High-risk cutaneous squamous cell carcinoma. Actas Dermo-Sifiliográficas (English Edition). 2012;103(7):567–78.
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Katalinic, D., Juretic, A. Giant desmoplastic cutaneous squamous cell carcinoma of the gluteal region. World J Surg Onc 15, 121 (2017). https://doi.org/10.1186/s12957-017-1191-7
- Cutaneous squamous cell carcinoma