Letters to the Editor

one on the head. No other systemic disorders such as diabetes and thyroiditis were found. All nevi were surgically removed under local anesthesia. Histological examination of an achromic halo revealed absence of melanocytes in the basal layer and intense inflammatory infiltrates of mononucleated cells in the upper dermis (Fig. 1b), where nests of nevus cells were seen sporadically. Immunohistochemistry revealed high CD8-positive T-cell infiltration into the nevoid cells (Fig. 1c). The halo nevus usually occurs alone or in limited numbers, and multiple occurrences are rare. The mechanism behind the occurrence of the halo phenomenon is still uncertain, but the main hypothesis involves a cellular immune response targeting the melanocytes. During the formation and regression of halo nevi, infiltrates composed of T cells, in particular CD8-positive cytotoxic suppressor cells, have been observed.1 In the present case, a large number of predominantly CD8 T cells infiltrated into the nevoid cells. Dereure et al. 2 reported a case of multiple halo nevi in a patient with a benign carcinoid tumor of the ileum, suggesting a cross-reaction between tumoral cells and melanocytes enabled by their common embryonic origin. Also, the onset of multiple of halo nevi has been triggered by treatment with interferon-b, infliximab, imatinib and tocilizumab.3,4 In our patient, the halo nevi might have been caused by severe sunlight exposure, which suggests that ultraviolet (UV) light may trigger the induction of halo nevi as a result of photo-Koebner’s phenomenon. One hypothesis is that a cellular immune response elicited by long-term UV exposure may be responsible for the occurrence of multiple halo nevi. However, Sutton’s phenomenon was not induced in all of the nevi on the sun-exposed sites (trunk). The reason is unknown, but it may be just due to more or less of the induction of local immune effect, or the external triggers (e.g. mechanical traumas) may be involved. Another possibility is that UV irradiation

induces an apoptotic pathway. Recent studies suggest that short-term UV induces apoptosis in various types of cells including melanocytes.5 One of the apoptotic mechanisms for cytotoxic T cells is the granule exocytosis pathway, mediated by perforin and granzymes. We speculate that induction of these molecules may be involved in local cell immunemediated melanocyte regression in our case.

CONFLICT OF INTEREST:

None.

Atsuko KAWAGUCHI,1 Toshiyuki YAMAMOTO,2 Yukari OKUBO,1 Yoshihiko MITSUHASHI,1 Ryoji TSUBOI1 1 Department of Dermatology, Tokyo Medical University, Tokyo, and Department of Dermatology, Fukushima Medical University, Fukushima, Japan

2

doi: 10.1111/1346-8138.12814

REFERENCES 1 Akasu R, From L, Kahn HJ. Characterization of the mononuclear infiltrate involved in regression of halo nevi. J Cutan Pathol 1994; 21: 302–311. 2 Dereure O, Guillon F, Guillot B, Guilhou JJ. Multiple halo naevi associated with carcinoid in a young man. Acta Derm Venereol 2004; 84: 495–496. 3 Kuet K, Goodfield M. Multiple halo naevi associated with tocilizumab. Clin Exp Dermatol 2014; 39: 717–719. 4 Tronnier M, Smolle J, Wolff HH. Ultraviolet irradiation induces acute changes in melanocytic nevi. J Invest Dermatol 1995; 104: 475–478. 5 Kadekaro AL, Kavanagh RJ, Wakamatsu K, Ito S, Pipitone M, AbdelMalek ZA. Cutaneous photobiology. The melanocyte vs. the sun: who will win the final round? Pigment Cell Res 2003; 16: 434–447.

Bronchogenic cyst in the chin region Dear Editor, Bronchogenic cysts (BC) are congenital pulmonary anomalies and are related to abnormal budding of the tracheobronchial tree during embryologic development.1 The most common sites of BC are the lungs and mediastinum. Extra-thoracic cutaneous BC are occasionally observed, mainly located in the suprasternal notch area, presternal area, neck and scapula.1 However, cutaneous BC of the face are extremely rare. Here, we report a case of cutaneous BC of the chin. An 8-month-old boy presented with an asymptomatic congenital cyst with punctum in the chin region (Fig. 1a,b). We excised the lesion, which was 22 mm in length and terminated in the periosteum of the mandible (Fig. 1c). Histologically, the

cyst was lined with squamous epithelium at the surface layer and contained ciliated columnar epithelium at the deep layer. Glandular tissue covered with pseudostratified ciliated epithelium was observed near the cyst wall, resembling those observed in the trachea and bronchi (Fig. 1d–h). These findings led to a diagnosis of BC. Bronchogenic cysts are an anomaly that results from migration and proliferation of misplaced buds of the tracheobronchial tree.2 The occurrence is rare, and in many cases observed in the mediastinum or intrapulmonary region.1 To the best of our knowledge, only four cases of facial BC have been reported to date.2,3 Interestingly, all facial BC, including our case, mainly appeared in the chin or lower lip region along the

Correspondence: Tateki Kubo, M.D., Ph.D., Department of Plastic and Reconstructive Surgery, Osaka University Graduate School of Medicine, 2-2 C11 Yamadaoka, Suita-shi, Osaka 565-0871, Japan. Email: [email protected]

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Letters to the Editor

(a)

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system. Therefore, facial BC may be attributed to abnormal budding during the development of the trachea though the direct connection to the trachea has not been reported. As differential diagnoses, dermoid cysts, thyroglossal duct cysts and branchial cleft cysts should be considered. Dermoid cysts are often observed in the face of young children. However, the cyst wall is lined with keratinized squamous epithelium, rather than ciliated columnar epithelium. Thyroglossal duct cysts and branchial cleft cysts can present both squamous epithelium and ciliated columnar epithelium. However, the path of thyroid descent is well-defined. During the 4th week of fetal life, the thyroid gland develops as an invagination at the level of the foramen cecum at the base of the tongue. Over the next few weeks, the developing gland migrates inferiorly to the thyroid bed in the visceral space of the infrahyoid neck,4,5 which is incompatible with the location in the present case. The locations of branchial cleft cysts are also well known, including the lateral neck and pre- or posterior auricular regions.2 Malignant neoplasms arising from BC have been reported,1 suggesting the importance of surgical excision. In clinical practice, therefore, if a congenital cyst is found along the midline of the chin or lower lip, BC should be suspected, and complete excision should be considered.

CONFLICT OF INTEREST:

The authors report no conflicts

of interest.

Akimitsu NISHIBAYASHI,1 Tateki KUBO,1 Ken MATSUDA,2 Jun-ichiro IKEDA,3 Ko HOSOKAWA1 Figure 1. (a) An 8-month-old boy presented with a cyst in the midline of the chin. (b) Ultrasonography showing a subcutaneous, well-defined, hypoechoic lesion with posterior acoustic enhancement. (c) The lesion terminated in the periosteum of the mandible. (d,e) Histopathological findings with hematoxylin–eosin staining at the surface layer of the cyst (original magnifications: [d] 912.5; [e] 9100). The cyst wall was lined with squamous epithelium. (f–h) Deep layer of the cyst at (f) 912.5, (g) 9100 and (h) 9400. The cyst wall was lined with ciliated columnar epithelium. Glandular tissue covered with pseudostratified ciliated epithelium was observed near the cyst wall.

midline. Furthermore, in the previous reports, the majority of upper neck BC were also located along the midline.1 Ustundag et al.1 mentioned that BC located on the midline of the neck are due to abnormal budding from the trachea, and lateral BC in the lower neck region are due to budding of the bronchial

© 2015 Japanese Dermatological Association

1 Department of Plastic Surgery, Osaka University Graduate School of Medicine, Osaka, 2Division of Plastic and Reconstructive Surgery, Niigata University Graduate School of Medicine, Niigata, and 3Department of Pathology, Osaka University Graduate School of Medicine, Osaka, Japan

doi: 10.1111/1346-8138.12816

REFERENCES 1 Ustundag E, Iseri M, Keskin G, Yayla B, Muezzinoglu B. Cervical bronchogenic cysts in head and neck region. J Laryngol Otol 2005; 119: 419–423. 2 Magliocca KR, Ricalde P, Vincek V, Bhattacharyya I, Cohen DM. Bronchogenic cyst with intraoral and extraoral components. J Oral Maxillofac Surg 2011; 69: 2604–2607. 3 Kotsuji-Maruyama T, Umebayashi Y, Imakado S, Otsuka F. Cutaneous bronchogenic cyst of the chin. Dermatology 2001; 203: 192–193. 4 Koeller KK, Alamo L, Adair CF, Smirniotopoulos JG. Congenital cystic masses of the neck: radiologic-pathologic correlation. Radiographics 1999; 19: 121–146; quiz 52-3. 5 Shahin A, Burroughs FH, Kirby JP, Ali SZ. Thyroglossal duct cyst: a cytopathologic study of 26 cases. Diagn Cytopathol 2005; 33: 365– 369.

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Bronchogenic cyst in the chin region.

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