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Figure 1. (a) A slightly elevated tumor with a black-to-brown pigmented macule on the left sole. (b) An electrocardiogram (ECG) at initial visit to our hospital. No obvious abnormal findings except for left atrial enlargement were observed. (c) ECG after the fourth injection of interferon-b. Atrial fibrillation was observed.

been no reports of cardiovascular complications due to natural IFN-b in the English-language published work. It is unclear why IFN-b rarely causes cardiac complications in spite of its similarities with IFN-a; further investigations are warranted. In conclusion, we report an extremely rare case of atrial fibrillation due to IFN-b. Although cardiovascular complications are uncommon, regular cardiologic examinations, such as ECG, should be performed before and after IFN-b treatment in melanoma.

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CONFLICT OF INTEREST:

The authors have no conflict of

interest.

Jun ASAI, Makoto WADA, Yukiko UEDA, Yukiko KATAOKA, Maiko TAURA, Hideya TAKENAKA, Norito KATOH Department of Dermatology, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, Kyoto, Japan doi: 10.1111/1346-8138.12874

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induced by IFN may trigger cardiac complications.3 However, we have found only one reported case of atrial fibrillation induced by recombinant IFN-b.5 To our knowledge, there have

REFERENCES 1 Kirkwood JM, Strawderman MH, Ernstoff MS et al. Interferon alfa-2b adjuvant therapy of high-risk resected cutaneous melanoma: the Eastern Cooperative Oncology Group Trial EST 1684. J Clin Oncol 1996; 14: 7–17. 2 Matsumoto T, Yokota K, Sawada M et al. Postoperative DAV-IFNbeta therapy does not improve survival rates of stage II and stage III melanoma patients significantly. J Eur Acad Dermatol Venereol 2013; 27: 1514–1520. 3 Sonnenblick M, Rosin A. Cardiotoxicity of interferon. A review of 44 cases. Chest 1991; 99: 557–561. 4 Fujiwara T, Kiura K, Ochi K et al. Giant negative T waves during interferon therapy in a patient with chronic hepatitis C. Intern Med 2001; 40: 105–109. 5 Sarna GP, Figlin RA, Pertcheck M. Phase II study of betaseron (beta ser17-interferon) as treatment of advanced malignant melanoma. J Biol Response Mod 1987; 6: 375–378.

Primary cutaneous adenoid cystic carcinoma arising from folliculitis decalvans Dear Editor, Primary cutaneous adenoid cystic carcinoma (PCACC) is a rare adnexal tumor, which appears often on the head and neck,

especially the scalp.1 It tends to recur locally but has a low metastatic potential. Folliculitis decalvans (FD) is a chronic deep folliculitis that usually occurs on the scalp as oval

Correspondence: Hideki Nakajima, Ph.D., M.D., Department of Dermatology, Kochi Medical School, Kochi University, Okoh-cho, Nankoku 783-8505, Japan. Email: [email protected]

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patches of scarring alopecia. We report a case of PCACC that arose from FD. A 65-year-old man presented a yellowish nodule on the vertex of 1-month duration around the atrophic skin with alopecia (Fig. 1a). He had been aware of the alopecic lesion on the scalp for 2 years, but no medical treatment was given. He had a medical history of diabetes mellitus and hypertension. Physical examination revealed a firm, peau d’orange nodule with a diameter of 1 cm at the edge of the atrophic, erosive, hyperkeratotic lesion of the scalp. A biopsy of the nodule revealed various masses of basaloid cells in a cystic, tubular or cribriform pattern located in the dermis and subcutis (Fig. 1b,c) with perineural invasion (Fig. 1d). Characteristic features of basal cell carcinoma, including palisading arrangement of cells at the

periphery of tumor nodules or peritumor clefting were not present. Cystic spaces contained basophilic mucin and eosinophilic hyaline material (Fig. 1c). Immunohistochemical analysis revealed diffuse positive staining for both AE1/AE3 and cytokeratin (CK)7 (Fig. 1e) in the tumor lobules, strong expression of epithelial membrane antigen (EMA) in the luminal cells of true tubular structure (Fig. 1f) and focal expression of smooth muscle actin, S100 and Ber-EP4 in the tumor cells of cribriform pattern. Carcinoembryonic antigen and CK20 were not expressed. A diagnosis of PCACC was made, and it can be distinguished from adenoid basal cell carcinoma by immunohistochemical positive staining for sweat gland-associated antigens, such as CK7, EMA and S100.2 Histological examination of the chronic alopecic lesion showed dermal fibrosis and

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Figure 1. (a) Scalp showing atrophic alopecia, slight erosion and focal hyperkeratosis. Arrow indicates yellowish nodule on the vertex at the upper-right corner of the hair loss lesion. (b) Histopathology of the nodule. Tumor lobules composed of basaloid cells arranged in a cribriform pattern and true lumina with eosinophilic hyaline material located in the dermis. Authentic tubular structures are abundant in the lower dermis (hematoxylin–eosin [HE], original magnification 940). (c) Higher magnification indicates aggregations of cribriform tumor nests containing mucin and hyaline material (HE, 9200). (d) Higher magnification highlights perineural invasion of basaloid tumor nests with cribriform pattern (HE, 9400). (e) Diffuse cytokeratin staining of tumor cells (cytokeratin 7, 9200). (f) Strong expression of epithelial membrane antigen in the luminal cells of true tubular structure (9200). (g) Histopathology of the chronic alopecic area. Fibrosing dermis adjacent to destroyed follicles contains a mixed inflammatory infiltrate composed of lymphocytes and multinucleated foreign body giant cells (HE, 9100).

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mixed inflammatory infiltrate consisting of lymphocytes and multinucleated foreign body giant cells around hair follicles (Fig. 1g). These findings were compatible with FD. After metastatic disease was excluded by positron emission tomography/ computed tomography, the lesion was successfully resected. No recurrence or metastasis was detected at 1 year postoperation. Tumor in conjunction with FD is rare, and only one case of squamous cell carcinoma has been reported.3 Although the relation of PCACC to FD might have been coincidental, our case suggests the potential risk of developing malignant tumor which may arise from chronic erosive FD in the sun-exposed scalp. Primary cutaneous adenoid cystic carcinoma exhibits a clinical indolent course in contrast to adenoid cystic carcinoma of the salivary glands, showing greater mortality.4 Frequent recurrence of PCACC have been reported within several years after initial diagnosis, however, the overall 5-year survival of patients with PCACC on the head and neck is excellent (99.1%).5 Because of frequent PCACC’s perineural involvement, which was also seen in our case and considered a cause of high local recurrence, wide resection is recommended. In summary, this is the first case of PCACC that arose from pre-existing FD.

CONFLICT OF INTEREST:

None declared for all authors.

Hideki NAKAJIMA, Tomoko MATSUSHIMA, Shigetoshi SANO Department of Dermatology, Kochi Medical School, Kochi University, Nankoku, Japan doi: 10.1111/1346-8138.12880

REFERENCES 1 Naylor E, Sarkar P, Perlis CS, Giri D, Gnepp DR, Robinson-Bostom L. Primary cutaneous adenoid cystic carcinoma. J Am Acad Dermatol 2008; 58: 636–641. 2 Rocas D, Asvesti C, Tsega A, Katafygiotis P, Kanitakis J. Primary adenoid cystic carcinoma of the skin metastatic to the lymph nodes: immunohistochemical study of a new case and literature review. Am J Dermatopathol 2014; 36: 223–228. 3 Yip L, Ryan A, Sinclair R. Squamous cell carcinoma arising within folliculitis decalvans. Br J Dermatol 2008; 159: 481–482. 4 Gondivkar SM, Gadball AR, Chole R, Parikh RV. Adenoid cystic carcinoma: a rare clinical entity and literature review. Oral Oncol 2011; 47: 231–236. 5 Dores G, Huycke M, Devesa S, Garcia CA. Primary cutaneous adenoid cystic carcinoma in the Unites States: incidence, survival, and associated cancers, 1976 to 2005. J Am Acad Dermatol 2010; 63: 71–78.

Incidental finding of multiple Pacinian neuroma in hand Dear Editor, The Pacinian corpuscle is a rapidly adapting mechanoreceptor, mediating the perception of vibration stimuli. It consists of an elongated axon terminal surrounded by a lamellar inner core of Schwann cell origin that is encased in a multilayered outer core of concentric lamellae of cells of perineurial origin.1,2 Pacinian corpuscles in the human hand normally consist of 13– 15 lamellae; the largest longitudinal diameter of a standard corpuscle is 1.6 mm.3 The Pacinian neuroma is characterized by formation of typical painful nodules in connective tissue of the hand. The etiopathogenesis is still not clear, however, all nodules are of non-malign nature and after surgical removal, they do not develop again.4 The Pacinian neuroma is a rare clinical entity with only several reports published. We report a case of 24 Pacinian corpuscles in one hand, characterized by the multiplicity of corpuscles and their distribution through both median and ulnar nerves (Fig. 1a). Nodules have been found accidentally during standard anatomical dissection of an adult male. Unfortunately, no anamnesis data of the patient were available. The cadaveric tissue was perfused with fixative (mixture of ethanol, formal, glycerol and distilled water) and stored for 2 years for the purpose of following standard anatomical dissec-

tion. During the dissection, an operating microscope with 92.5 and 915–30 operating loupes was used for preparation of the left hand. All branches of median and ulnar nerves and ulnar and radial arteries were found and meticulously dissected, all veins were removed. Found nodules were measured and then carefully excised for histological analysis (Fig. 1b). The microdissection study of contralateral palmar tissue was then performed. The samples of all found nodules underwent histological analysis during which the largest transverse and longitudinal diameters in serial sections were measured. A total of 24 nodules were collected from deep palmar tissue of the left hand. All nodules were found in relation with the perineurium of palmar digital branches of median and ulnar nerve; no nodules which were not associated with the nerve were found. No nodules were found on the dorsal surface of the hand in relation with branching of the radial nerve or dorsal branches of the ulnar nerve or in any other location in the body. In all cases, the longest diameter was parallel to the course of the nerve. The mean diameter of nodules measured macroscopically was 4.50  0.31 mm (range, 2–7 mm). Histological analysis showed similar density of such nodules, and hypertrophic onion-like multilamellated structures located in the connective perineurial tissue (Fig. 1c,d) were found, surrounding

Correspondence: Marketa Skalna, Ph.D., Department of Anatomy, Faculty of Medicine, University of Ostrava, Syllabova 19, Ostrava 703 00, Czech Republic. Email: [email protected]

© 2015 Japanese Dermatological Association

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Primary cutaneous adenoid cystic carcinoma arising from folliculitis decalvans.

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