Kentaro Ida

Ophthalmic Artery Embolization as Pretreatment of Orbital Exenteration for Conjunctival Squamous Cell Carcinoma

The aim of this study is to describe the effect of transarterial embolization from the ophthalmic artery as a pretreatment for orbital exenteration. A 75-year-old Chinese man with a 7-year history of gradual increase of the left eye swelling showed a massive conjunctival tumor growing outwardly from the interpalpebral fissure and had no light perception in the left eye. Magnetic resonance imaging showed orbital invasion of the tumor around the left eyeglobe. The initial surgery for the planned orbital exenteration was discontinued after skin incision around the orbital margin due to massive hemorrhage. The patient underwent transarterial embolization with gelatin sponge (Spongel) of the feeding arteries from the left ophthalmic artery and, the next day, had orbital exenteration with well-controllable bleeding and reconstruction with free vascularized anterolateral thigh cutaneous flap transfer. Pathologically, well-differentiated squamous cell carcinoma proliferated in exophytic, papillary, and nested fashions, arising from the bulbar conjunctiva. Tumor cells were also found in the conjunctival stroma around the vessels. The sclera at the equator had a perforated site with tumor cell invasion, but no intraocular invasion was found. Hematoxylin-positive gelatin sponges were found inside the orbital vessels and large choroidal vessels. In conclusion, transarterial embolization of feeding arteries arising from the ophthalmic artery is a useful pretreatment to control bleeding at orbital exenteration for malignancy.

Suspected early onset of congenital Langerhans cell histiocytosis involving ectopic cervical thymus and mediastinal thymus, simultaneously

To the Editor: Langerhans cell histiocytosis (LCH) is a rare histiocytic disorder characterized by accumulation of Langerhans cells [1,2]. Congenital LCH chiefly affects the skin and is selfhealing (Hashimoto–Pritzker disease), but rare involvements of the lung, bone, or liver have been reported [3]. Thymus is one of the uncommon organs involved by LCH [4]. Ectopic thymus is an extremely rare etiology of a neck mass in an infant which results from abnormal embryogenesis of the thymus [5,6]. We present a case of congenital LCH with simultaneous involvement of ectopic cervical thymus and mediastinal thymus. A 2-month-old female without any significant medical history through the prenatal and neonatal period was brought to a physician complaining of left submandibular swelling. On physical examination, there was an elastic, soft, and poorly movable mass in the left submandibular area. The mass was 3 cm in diameter and had a smooth surface. Computed tomography (CT) revealed cystic and solid components in the mass (Fig. 1A). An open biopsy of the mass was performed. The mass was well demarcated from surrounding tissues and was resected without macroscopic leaving residues. The pathological examination showed proliferation of CD1a positive cells with a round nuclear and rich cytoplasm in an eosinophil rich background (Fig. 1B–D). Diagnosis of LCH was made. Moreover, the resected mass contained ectopic thymic tissue whose architecture was partly effaced by infiltration of Langerhans cells (Fig. 1B and E). Reconfirmation of the CT image revealed the enlarged mediastinal thymus with multiple cysts and punctate calcifications which are specific findings of the thymic involvement of LCH (Fig. 1A) [7]. Although biopsy of the mediastinal thymic LCH