A congenital melanocytic nevus of the scalp is occa-
sionally associated with intracranial pigmentation, which
can be a benign asymptomatic coexisting melanomatosis or an invasive extension of the cutaneous lesion or a paracrinopathy from the overlying nevus. 6 Although the differential diagnosis of pigmentation and bone abnormality includes Albright syndrome, it could be excluded based on pathologic diagnosis of pigmentary lesion. In Albright syndrome, pigmentation should be of type caf-au-lait spots, not melanocytic nevus. 10 To our knowledge, this is the rst case of congenital melanocytic nevus of the scalp with underlying skull defect. Although the association between a congenital melano- cytic nevus of the scalp, underlying skull defect, and the development of melanoma from the congenital melanocy- tic nevus of the scalp are unclear, we suggest that clini- cians should evaluate the skull in the patients who have congenital melanocytic nevi of the scalp and have regular follow-up examinations. In our case, the small size of the specimen obtained by a 3-mm punch biopsy instead of total excision might not be representative for histopathological examination and patient was informed and recommended regular visits at least every 6 months. Yeon Sook Kwon, MD Jin Mo Park, MD Hee Jung Kim, MD Min-Geol Lee, MD, PhD Department of Dermatology and Cutaneous Biology Research Institute Yonsei University College of Medicine Seoul Korea E-mail: mglee@yuhs.ac Funding sources: None. Conict of interest to disclosure: None declared. The authors have no relevant nancial interest in this article. References 1 Tannous ZS, Mihm MC Jr, Sober AJ, et al. Congenital melanocytic nevi: clinical and histopathologic features, risk of melanoma, and clinical management. J Am Acad Dermatol 2005; 52: 197203. 2 Reed WB, Becker SW Sr, Becker SW Jr, et al. Giant pigmented nevi, melanoma, and leptomeningeal melanocytosis. Arch Dermatol 1965; 91: 100118. 3 Ho N, Roig C, Diadori P. Epidermal nevi and localized cranial defects. Am J Med Genet 1999; 83: 187190. 4 Menter MA, Griessel PJC, de Klerk DJ. Giant blue naevus of the scalp with underlying skull defect. Br J Dermatol 1971; 85: 7375. 5 Silverberg GD, Kadin ME, Dorfman RF, et al. Invasion of the brain by a cellular blue nevus of the scalp. A case report with light and electron microscopic studies. Cancer 1971; 27: 349355. 6 Findler G, Hoffman HJ, Thomson HG, et al. Giant nevus of the scalp associated with intracranial pigmentation. Case report. J Neurosurg 1981; 54: 108 112. 7 Marano SR, Brooks RA, Spetzler RF, et al. Giant congenital cellular blue nevus of the scalp of a newborn with an underlying skull defect and invasion of the dura mater. Neurosurgery 1986; 18: 8589. 8 Madaree A, Ramdial PK, Du Trevou M. Giant congenital naevus of the scalp and cranium: case report and review of the literature. Br J Plast Surg 1997; 50: 2025. 9 Kousseff BG. Hypothesis: Jadassohn nevus phakomatosis: a paracrinopathy with variable phenotype. Am J Med Genet 1992; 43: 651661. 10 Albright F, Butler AM, Hampton AO, Smith P. Syndrome characterized by osteitis brosa disseminata, areas of pigmentation and endocrine dysfunction, with precocious puberty in females: report of ve cases. New Eng J Med 1937; 16: 727746. Erythema nodosum with eosinophilic panniculitis Erythema nodosum (EN) is manifested by the sudden onset of tender erythematous nodules and plaques on the legs. The lesions show septal panniculitis chiey inl- trated by lymphohistiocytes. Inltrated eosinophils are usually in a minority. We describe here a patient with EN who showed septal panniculitis with an admixture of numerous eosinophils. A 41-year-old Japanese woman visited our hospital with a 3-month history of a painful eruption affecting the left lower leg. She was in otherwise good health and used no medication. Examination showed a subcutaneous nod- ule, 9 10 cm, with an erythematous surface on the ante- rior of left lower leg (Fig. 1). A skin biopsy specimen showed the presence of lymphohistiocytic inltrate chiey in the septa of the subcutaneous tissues with minor inl- tration near the septa (Fig. 2a). The overlying dermis had a moderate, deep perivascular inltration. In the subcutis, the inltrate was admixed with a number of eosinophils, some of which showed degranulation (Fig. 2b). Clusters of eosinophils were also present. Staining for fungi or acid-fast bacilli was negative. Based on clinical and histo- pathologic ndings, we diagnosed this case as EN with eosinophilic panniculitis. Full blood count, electrolytes, 2010 The International Society of Dermatology International Journal of Dermatology 2010, 49, 960969 Correspondence 965 liver function tests, and antistreptolysin O titer were all within normal limits. No peripheral eosinophilia was found. An intradermal tuberculin test was positive (20 18 mm induration). However, there were no abnor- mal shadows on chest X-ray and no colonies of Mycobac- terium tuberculosis in the sputum culture. Two months later the lesion cleared spontaneously, and the patient has had no recurrence of the skin lesion over the following period of 12 months. The inltrates in EN chiey consist of lymphohistio- cytes, and eosinophils are usually in a minority. On the other hand, eosinophilic panniculitis shows prominent inltration with eosinophils in septa and/or lobules of the subcutis. As eosinophilic panniculitis is associated with a diverse range of local and systemic diseases including EN, parasitic infection, and hypereosinophilic syndrome, eosinophilic panniculitis is a histopathologic pattern rather than a distinct entity. 14 We consider our case as EN with eosinophilic panniculitis, because the clinical and histopathologic features were consistent with those of EN except numerous eosinophil inltration. EN is thought to be a reactive phenomenon that may occur in response to antigen stimuli. In our case, the exact patho- mechanism is not understood, but the inltration may be the result of some antigen expression which induces eosinophils in the subcutis. In addition, eosinophilic inl- tration may develop in several diseases, including non- episodic type of angioedema with eosinophila, insect bite, drug eruption, and Wells syndrome. However, our case did not match that noted in them. Eosinophilic inltration in EN may be more common than has been thought. Teie Egawa, MD, PhD Ryuhei Okuyama, MD, PhD Hachiro Tagami, MD, PhD Setsuya Aiba, MD, PhD (a) (b) Figure 2 Histopathologic nding: inltration in and near the septa (a) and the admixture of eosinophils (b), some of which showed degranulation (hematoxylin and eosin stain: a 100; b 400) Figure 1 Clinical nding: a subcutaneous nodule with an erythematous surface on the lower leg International Journal of Dermatology 2010, 49, 960969 2010 The International Society of Dermatology Correspondence 966 Department of Dermatology, Tohoku University Graduate School of Medicine, Aoba-ku, Sendai, Japan Ryuhei Okuyama Department of Dermatology Tohoku University Graduate School of Medicine 1-1 Seiryo-machi Aoba-ku, Sendai 980-8574 Japan E-mail: rokuyama@mail.tains.tohoku.ac.jp References 1 Adame J, Cohen PR. Eosinophilic panniculitis: diagnostic considerations and evaluation. J Am Acad Dermatol 1996; 34: 229234. 2 Burket JM, Burket BJ. Eosinophilic panniculitis. J Am Acad Dermatol 1985; 12: 161164. 3 Winkelmann RK, Frigas E. Eosinophilic panniculitis: a clinicopathologic study. J Cutan Pathol 1986; 13: 112. 4 Wolff K, Goldsmith LA, Katz SI, Gilchrest BA, et al. Fitzpatricks Dermatology in General Medicine. 7th edn; New York: Mcgraw-Hill, 2008; 585. Primary cutaneous peripheral T-cell non-Hodgkin lymphoma, not otherwise specied, with cytotoxic features We have read with great interest the recently published case report by Yamamoto et al. 1 describing a patient with primary cutaneous aggressive T-cell lymphoma that exhibited cytotoxic phenotype, however, without the expression of CD8 antigen. In the light of this publica- tion, we would like to present a similar patient history, which, in our opinion, provides further data in regard of this unique type of cutaneous lymphoma. A 53-year-old man, with a 25-year history of tobacco smoking, presented with a large, slightly painful ulcera- tion in the right groin. The lesion started as a small, dark reddish nodule that progressed within 2 weeks into a rap- idly growing ulceration. About 6 months later, when the patient came to our department, a moist ulceration of about 35 cm diameter with a raised, inltrated border was found (Fig. 1a). Until then only topical and systemic antimicrobial agents were administered by the patient as treatment. The patient was in good general condition; peripheral lymph nodes were not enlarged and no other clinically relevant abnormalities were stated during physi- cal examination. The family history was irrelevant. Abdo- men sonography revealed only mild hepatomegaly. The patient also had slight leukocytosis (11 000 ll) with neu- trophilia (74%) and accelerated erythrocyte sedimentation rate (76 mm after 1 h). No other abnormalities were found in laboratory and imaging examination (including chest CT scan). To establish the diagnosis, a biopsy was performed from the ulcer edge. The histology showed a dense inltrate of medium- to large-sized lymphoid cells, involving mainly dermis and lower layers of epidermis (Fig. 2a,b). Majority of these cells were positive for CD3 (Fig. 2c), CD5, CD7, Ki-67, granzyme B (Fig. 2e), and TIA-1 (Fig. 2f), while only some of them (< 20%) showed expression of CD30. The tumor cells were also negative for CD4, CD8, CD20, CD25, CD34, CD56, CD57, CD68, CD117, pancytokeratin, HMB-45, Melan A, S100 protein, myeloperoxidase, perforin, ALK-1, and alfa- actin. The CD8 positive staining was only restricted to small cells representing a reactive inltrate (Fig. 2d). In (a) (b) Figure 1 (a) Primary cutaneous epidermotropic cytotoxic lym- phoma manifesting as an isolated, large ulceration with a raised border in the right groin. (b) Local relapse of the tumor 2010 The International Society of Dermatology International Journal of Dermatology 2010, 49, 960969 Correspondence 967