Letters to the Editor

CD23( ), CD25( ), CD30( ), CD34( ), CD56( ), CD79a( ), CD117( ), CAM 5.2( ), AE1/AE3( ), CK7( ), CK20( ), desmin ( ), a-smooth muscle actin (a-SMA)( ), Mif4( ), myogenin( ), S-100( ), chromogranin A( ) and synaptophysin( ). Touch smear and frozen section for cytochemical stains were not available and cytogenetic study was not performed in this case. Bone marrow examination revealed no involvement of leukemia. Positron emission tomography detected no other involving lesions. No recurrence occurred until December 2014. Based on these clinicopathological features, we diagnosed the plaque as aleukemic solitary cutaneous myeloid sarcoma (CMS). Cutaneous involvement usually presents in the course of myeloid leukemia, but aleukemic cutaneous involvement is uncommon and may present multiple eruptions even if it occurs.3 We need to keep in mind that aleukemic solitary CMS can occur even in infancies, and aleukemic CMS mostly precede bone marrow involvement.3 The French-American-British classification of acute myeloid leukemia include acute myelomonocytic leukemia (M4), acute monoblastic leukemia (M5a) and acute monocytic leukemia (M5b).1 Results of immunophenotype examination suggested that aleukemic solitary CMS in our case was close to M4, M5a or M5b, although the figure of the peripheral leukocytes was normal. The histopathological diagnosis of CMS without synchronous bone marrow involvement may be difficult.3 Currently, we have no consensus recommendation for a panel of antibodies to reach a correct diagnosis, although a combination of those to CD43, CD68 and lysozyme, and another combination of those to CD68, CD33 and MPO have been shown to be highly sensitive.3–5 In this case, the diagnosis of CMS was supported by the expression of CD33, CD43, CD68, lysozyme and MPO

(focal). Previous and current cases indicate the usefulness of the five markers. Further accumulation of such cases would reveal the sensitivity and specificity of these markers.

CONFLICT OF INTEREST:

None declared.

Midori YANAGIHARA,1 Naoki OISO,1 Hirokazu TANAKA,2 Tomohiko NARITA,1 Eisuke ENOKI,3 Masatomo KIMURA,3 Hirokazu NAKAMINE,4 Itaru MATSUMURA,2 Akira KAWADA1 Departments of 1Dermatology, 2Hematology and Rheumatology, 3Pathology, Kinki University Faculty of Medicine, Osaka, and 4Department of Laboratory Medicine, The Japan Baptist Medical Foundation, Kyoto, Japan doi: 10.1111/1346-8138.12924

REFERENCES 1 Swerdlow SH, Campo E, Harris NL et al., eds. WHO Classification of Tumours of Haematopoietic and Lymphoid Tissures, 4th edn. International Agency for research on Cancer, Lyon, France, 2008. 2 Morgan EA, Murphy GF. Cutanrous lymphomas and leukemias. In: Elder DE, ed. Levers Histopathology of the Skin, 11th edn. Philadelphia: Lippincott Williams & Wilkins, 2014; 1116–1202. 3 Aboutalebi A, Korman JB, Sohani AR et al. Aleukemic cutaneous myeloid sarcoma. J Cutan Pathol 2013; 40: 996–1005. 4 Hurley MY, Ghahramani GK, Frisch S et al. Cutaneous myeloid sarcoma: natural history and biology of an uncommon manifestation of acute myeloid leukemia. Acta Derm Venereol 2013; 93: 319–324. net C, Gomez A, Aguilar C et al. Histologic and immunohistologic 5 Be characterization of skin localization of myeloid disorders: a study of 173 cases. Am J Clin Pathol 2011; 135: 278–290.

Multiple cutaneous squamous cell carcinoma in cutaneous sarcoidosis Dear Editor, A 90-year-old woman presented at our tertiary referral center with facial tumors arising in a “port wine stain”. A plastic surgeon had previously excised several cutaneous squamous cell carcinomas (cSCC) in this lesion, present for more than 10 years. Her medical history revealed hypertension. Clinical examination showed an extensive, sharply demarcated, erythematous, livid and partly sclerotic plaque on the left side of her face and neck, causing ear deformation. Within this plaque, four suspect lesions (temporal area and cheek) were identified (Fig. 1a). The opposite side of her face did not show skin abnormalities including actinic damage or rosacea. Histopathological examination showed two cSCC and two Bowen’s disease (BD) above a deep and superficial granu-

lomatous inflammation without necrosis or necrobiosis, suspect for infection or sarcoidosis (Fig. 1b–e). Similar granulomatous inflammation was found in a biopsy out of the neck region. The histopathological slides did not show multinucleated giant cells and total lysis of elastic tissue excluding actinic granuloma. An ultrasound test showed no pathological lymph nodes. Tuberculosis was ruled out by a QuantiFERON test. A biopsy taken for bacterial culture showed no actinomyces, nocardia or mycobacteria. Laboratorial tests showed normal levels of angiotensin-converting enzyme and soluble interleukin-2 receptor. Based on clinical, laboratorial and histological examination we diagnosed two cSCC and two BD in a cutaneous sarcoidosis plaque. There was no evidence for the previous diagnosis of port wine stain. The cSCC and BD

Correspondence: Annet Westers-Attema, M.D., Department of Dermatology, Medical Center Haaglanden, Postbus 432, 2501 CK Den Haag, the Netherlands. Email: [email protected] *Present address: Medical Center Haaglanden and Langeland skin clinic, the Hague region.

© 2015 Japanese Dermatological Association

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Letters to the Editor

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in the left temporal area were excised. The BD on her left cheek was treated with methyl aminolevulinate photodynamic therapy (PDT, 37 J/cm2; Aktilite, Galderma SA, Lausanne, Switzerland). A week after the first session, our patient showed a severe reaction with crusts and purulent exudate. Flucloxacillin 500 mg three times daily for 7 days was started and the second PDT session was cancelled. The patient refused treatment for sarcoidosis, because the lesion had been present for many years “without any complaints”. Cutaneous squamous cell carcinomas have been reported in chronic ulcers, cutaneous tuberculosis, burn scars and oral lichen planus.1,2 Our patient shows cSCC arising in cutaneous sarcoidosis. The chronic inflammation of sarcoidosis may be responsible for atypia of keratinocytes and tumor development. Sarcoidosis is a systemic disorder characterized by infiltration with non-caseating granulomas with skin involvement in 20–25% of patients.3 Isolated cutaneous sarcoidosis occurs in 9–30%.3 (Cutaneous) sarcoidosis is associated with cancer, especially in case of progressive sarcoidosis.3 Associated malignancies include lymphoma, and solid cancers such as lung and skin cancer.3 Interestingly, our patient only developed cSCC in her progressive sarcoidosis plaque. The chronic inflammation and/or the immunological deficiencies associated with sarcoidosis probably resulted in malignant transformation in our patient.3,4 Photodynamic therapy has been described as a treatment for sarcoidosis.5 We hypothesize that a synergistic effect of PDT on BD and sarcoidosis may explain the severe reaction in our patient. Alternatively, perhaps the immune response against tumor cells of the reactive oxygenated species is stronger in sarcoidosis lesions. This case illustrates that physicians should be aware of the possibility of tumor formation in chronic sarcoidosis plaques

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Figure 1. (a) Multiple cutaneous squamous cell carcinoma (in situ) in a cutaneous sarcoidosis plaque. (b,c) Hematoxylin–eosin stain of superficial and deep granulomatous infiltrate without necrosis or necrobiosis (green arrows), squamous cell carcinoma (SCC; blue arrow) and Bowen’s disease (red arrow), (d) close up sarcoidosis and (e) close up SCC (original magnifications: [b] 925; [c] 925; [d] 9100; [e] 950). and inform their patients about this risk if they refuse treatment for their cutaneous sarcoidosis.

CONFLICT OF INTEREST: There is no conflict of interest to report.

Annet WESTERS-ATTEMA,1,2,* Myrurgia ABDUL HAMID,3 Eline HAANS,1 Rene van der HULST,4 Nicole KELLENERS-SMEETS1,2 1

Department of Dermatology, Maastricht University Medical Center, GROW Research Institute for Oncology and Developmental Biology, Maastricht University, 3Department of Pathology, and 4Department of Plastic Surgery, Maastricht University Medical Center, Maastricht, the Netherlands 2

doi: 10.1111/1346-8138.12931

REFERENCES 1 Marcoval J, Alcaide F. Evolution of cutaneous tuberculosis over the past 30 years in a tertiary hospital on the European Mediterranean coast. Clin Exp Dermatol 2013; 38: 131–136. 2 Cocchetto V, Magrin P, de Paula RA, Aide M, Monte Razo L, Pantaleao L. Squamous cell carcinoma in chronic wound: Marjolin ulcer. Dermatol Online J 2013; 19: 7. 3 Alexandrescu DT, Kauffman CL, Ichim TE, Riordan NH, Kabigting F, Dasanu CA. Cutaneous sarcoidosis and malignancy: an association between sarcoidosis with skin manifestations and systemic neoplasia. Dermatol Online J 2011; 17: 2. 4 Askling J, Grunewald J, Eklund A, Hillerdal G, Ekbom A. Increased risk for cancer following sarcoidosis. Am J Respir Crit Care Med 1999; 160: 1668–1672. 5 Penrose C, Mercer SE, Shim-Chang H. Photodynamic therapy for the treatment of cutaneous sarcoidosis. J Am Acad Dermatol 2011; 65: e12–e14.

© 2015 Japanese Dermatological Association

Multiple cutaneous squamous cell carcinoma in cutaneous sarcoidosis.

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