Solitary Epithelioid Histiocytoma Uveal Melanoma Is Not Clear

ated with osmium tetroxide, and re-embedded in Epon Correspondence: Dr Verdijk, Department of Pathol- (Hexion Specialty Chemicals, Inc, Danbury, Connecti- ogy, Erasmus MC University, PO Box 2040, 3000 CA, cut) showed cytoplasm densely packed with mitochon- Rotterdam, the Netherlands ([email protected]). dria and sparse melanosomes (eFigure, http://www Author Contributions: Drs Verdijk and van den Bosch .archophthalmol.com). No epithelial features were contributed equally to this article. observed. Financial Disclosure: None reported. Fluorescence in situ hybridization experiments on tu- Online-Only Material: The eFigure is available at http: mor tissue indicated 2 copies of chromosomes 1, 3, 6, //www.archophthalmol.com. and 8. Multiplex ligation-dependent probe amplifica- 1. Banerjee SS, Harris M. Morphological and immunophenotypic variations in tion testing with the Salsa P027 uveal melanoma kit (MRC- malignant melanoma. Histopathology. 2000;36(5):387-402. Holland, Amsterdam, the Netherlands) confirmed this 2. Jih DM, Morgan MB, Bass J, Tuthill R, Somach S. Oncocytic metaplasia oc- normal disomic state for a total of 31 different regions curring in a spectrum of melanocytic nevi. Am J Dermatopathol. 2002;24 (6):468-472. tested on multiple chromosomes. Single-nucleotide poly- 3. Gelman BB, Trier TT, Chaljub G, Borokowski J, Nauta HJ. Oncocytoma in morphism array analysis revealed no copy number al- melanocytoma of the spinal cord: case report. Neurosurgery. 2000;47(3): 756-759. terations or regions of heterozygosity on any of the chro- 4. Kacerovska D, Sokol L, Michal M, Kazakov DV. Primary cutaneous signet- mosomes. These investigations have been carried out ring cell melanoma with pseudoglandular features, spindle cells and onco- according to the tenets of the Declaration of Helsinki. cytoid changes. Am J Dermatopathol. 2009;31(1):81-83. 5. Jih DM, Morgan MB, Bass J, Tuthill R, Somach SC. Oncocytic metaplasia occurring in melanoma. Semin Cutan Med Surg. 2004;23(1):73-79. Comment. The many histologic faces of melanoma 6. Vancura RW, Thomas JH, Jewell WR, Damjanov I. Bilateral oncocytic ma- include primary and metastatic carcinoma, neuroendo- lignant melanoma in axillary lymph nodes without evidence of an extra- 1 nodal primary. Ultrastruct Pathol. 2005;29(5):399-404. crine tumors, sarcoma, leukemia, and germ cell tumors. 7. Schlo¨tzer-Schrehardt U, Ju¨ nemann A, Naumann GO. Mitochondria-rich epi- Intraocular oncocytoma has been considered in the dif- thelioid leiomyoma of the ciliary body. Arch Ophthalmol. 2002;120(1): 77-82. ferential diagnosis of mesectodermal leiomyoma of the 8. Cunha SL, Lobo FG. Granular-cell myoblastoma of the anterior uvea. Br J 7 ciliary body. A granular cell tumor of the iris and ciliary Ophthalmol. 1966;50(2):99-101. body has been described.8 The diagnosis of choroidal 9. Kilic E, van Gils W, Lodder E, et al. Clinical and cytogenetic analyses in uveal melanoma. Invest Ophthalmol Vis Sci. 2006;47(9):3703-3707. melanoma and exclusion of other cancers was based on 10. Hamperl H. Benign and malignant oncocytoma. Cancer. 1962;15:1019-1027. the tumor’s characteristic mushroom shape, positive immunohistochemical staining for HMB-45, Melan-A, and tyrosinase, and a 2-year follow-up without evidence of another primary cancer. The prognostic significance of oncocytic change in Solitary Epithelioid Histiocytoma uveal melanoma is not clear. Our case displayed unfa- (Reticulohistiocytoma) of the Eyelid vorable histological prognostic parameters in tumor size, epithelioid cell type, and vascular pattern. This olitary epithelioid histiocytoma of the dermis (and was not corroborated with cytonuclear negative para- rarely of the mucosae but not the viscera) was pre- meters as no cytogenetic aberrations were present. viously designated as a reticulohistiocytoma.1 S 2 Earlier, it was reported that cytogenetic aberrations There are reports of 2 solitary corneoscleral lesions and were detected in 80% (59 of 74 cases) of a series of 1 small recurrent lesion in an eyelid.3 Among the rarest uveal melanoma.9 In cutaneous melanoma, no prog- of histiocytic disorders, it typically affects the truncal re- nostic significance could be determined.5 Oncocytic gion of young men (uncommonly the face and digits) and change is generally proposed to be a reactive degenera- is unassociated with any systemic disease. Among 12 of tive adaptation10; however, the fact that no cytogenetic 44 noneyelid lesions of solitary epithelioid histiocy- changes were observed in this tumor poses the possi- toma with follow-ups, none recurred despite incom- bility of a distinct tumor variant as opposed to a plete excision.1 Multicentric reticulohistiocytoma, on the degenerative change. other hand, has a predilection for the face and digits of In conclusion, to our knowledge we give the first de- middle-aged women and displays an associated dis- scription of an oncocytic uveal melanoma that is not to abling multifocal arthropathy and internal carcinoma in be mistaken histologically for other tumors, including approximately 27% of patients.1,4,5 It has been described metastatic carcinoma. in all 4 eyelids.5 Current opinion holds that both uni- centric and multicentric epithelioid histiocytomas are non- Robert M. Verdijk, MD, PhD neoplastic, arising from the macrophagic rather than the Thomas van den Bosch, MD dendritic histiocytic compartment. Nicole C. Naus, MD, PhD Dion Paridaens, MD, PhD Report of a Case. A healthy 39-year-old woman was re- Cornelia M. Mooy, MD, PhD ferred for evaluation of a 3-month-old lesion on her left Annelies de Klein, PhD lower eyelid. The patient had no other skin lesions. The examination of the eyelids revealed a smooth, 10-mm- Author Affiliations: Departments of Pathology (Drs Verdijk elevated, flesh-colored, firm lesion (Figure,A)inthe and Mooy), Ophthalmology (Drs van den Bosch and Naus), lateral one-third of the eyelid. Most of the eyelashes were and Clinical Genetics (Dr de Klein), Erasmus MC Univer- missing. Visual acuity was 20/20 OU. There was no head sity, and Rotterdam Eye Hospital (Drs van den Bosch and and neck lymphadenopathy. Computed tomographic Paridaens), Rotterdam, the Netherlands. results of the neck, chest, and abdomen/pelvis were

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C D

E F

Figure. Solitary epithelioid histiocytoma of the eyelid. A, Frontal view reveals a rounded, projecting tumor with a smooth surface and 2 surviving eyelashes surmounting a flesh-colored mass involving the left lower lateral eyelid skin and margin of a 39-year-old woman. B, Full-thickness eyelid resection shows a tuberose mass (Masson trichrome stain, original magnification ϫ25). O indicates orbicularis muscle; S, skin; and T, tarsus. C, The epidermis displays surface parakeratosis (arrow) (Masson trichrome stain, original magnification ϫ100). Clusters of polygonal epithelioid cells (also depicted in the inset at higher magnification; Masson trichrome stain, original magnification ϫ200) compose the tumor and are separated by thin strands of collagen. D, Myriad lymphocytes, polymorphonuclear leukocytes, and eosinophilic leukocytes envelop individual epithelioid cells with abundant eosinophilic cytoplasm that fail to manifest peripolesis and emperipolesis (hematoxylin-eosin, original magnification ϫ200). The cells display clefts as they retract from their neighbors, a distinctive feature differing from the syncytial appearance of most tuberculoid histiocytic collections, as in sarcoidosis. Inset, Some cells have adopted irregular shapes and have a more deeply staining, eosinophilic cytoplasm (hematoxylin-eosin, original magnification ϫ200). E, Intense positive staining of epithelioid cells for vimentin, an intermediate cytoplasmic filament, is seen (immunoperoxidase reaction, diaminobenzidine chromogen, original magnification ϫ200). F, The marker CD163 is strongly positive and highly specific for histiocytes (immunoperoxidase reaction, diaminobenzidine chromogen, original magnification ϫ200). Inset, The marker CD68 is more weakly positive and less specific (immunoperoxidase reaction, diaminobenzidine chromogen, original magnification ϫ200).

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©2011 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 10/02/2021 negative. A full-thickness eyelid resection was per- School, Boston (Drs Jakobiec and Kirzhner); and De- formed with 2-mm margins. The patient has experi- partments of Ophthalmology (Drs Morales Tollett, Man- enced no recurrence after 9 months. cini, and Hogan) and Pathology (Dr Hogan), University The excised mass measured 0.4ϫ0.5 cm at its base and of Texas Southwestern Medical Center, Dallas. 1.0 cm in height (Figure, B). Microscopically, a mass of Correspondence: Dr Jakobiec, Department of Ophthal- grouped, large epithelioid histiocytes (Figure, C) began be- mology, David G. Cogan Laboratory of Ophthalmic Pa- neath a mildly parakeratotic epidermis and a narrow band thology, 243 Charles St, Room 321, Boston, MA 02114 of collagen; they were usually mononucleated but occa- ([email protected]). sionally multinucleated, subdivided by fine collagen strands, Author Contributions: Dr Jakobiec had full access to and accompanied by a mixture of lymphocytes, polymor- all of the data in the study and takes responsibility for phonuclear leukocytes, and eosinophilic leukocytes. The the integrity of the data and the accuracy of the data tumor evinced incursion into the orbicularis muscle and analysis. tarsus (Figure, B). The principal polygonal cells were en- Financial Disclosure: None reported. dowed with abundant eosinophilic or amphophilic cyto- 1. Miettinen M, Fetsch JF. Reticulohistiocytoma (solitary epithelioid histiocy- plasm (Figure, D) without periodic acid–Schiff–positive in- toma): a clinicopathologic and immunohistochemical study of 44 cases. Am clusions. Their nuclei were round and possessed small J Surg Pathol. 2006;30(4):521-528. nucleoli; mitotic figures were not identified. Acid fast and 2. Allaire GS, Hidayat AA, Zimmerman LE, Minardi L. Reticulohistiocytoma of the limbus and cornea: a clinicopathologic study of two cases. Ophthalmology. methenamine silver stains did not detect organisms. The 1990;97(8):1018-1022. large tumor cells were strongly positive for vimentin (Figure, 3. Bakri SJ, Carlson JA, Meyer DR. Recurrent solitary reticulohistiocytoma of the eyelid. Ophthal Plast Reconstr Surg. 2003;19(2):162-164. E), sporadically positive for S-100 protein, strongly posi- 4. Zelger B, Cerio R, Soyer HP, Misch K, Orchard G, Wilson-Jones E. Reticulo- tive for CD163, and more weakly positive for CD68 (Figure, histiocytoma and multicentric reticulohistiocytosis: histopathologic and im- F) and ␣1-antitrypsin. Staining results for lysozyme, fac- munophenotypic distinct entities. Am J Dermatopathol. 1994;16(6):577-584. 5. Eagle RC Jr, Penne RA, Hneleski IS Jr. Eyelid involvement in multicentric tor XIIIa, and AE1/AE3 (for keratin), HMB-45, melan A, reticulohistiocytosis. Ophthalmology. 1995;102(3):426-430. and Mitf (for melanocytes), desmin and smooth muscle ac- 6. Zak FG. Reticulohistiocytoma (“ganglioneuroma”) of the skin. Br J Dermatol. tin (for myoid cells), and CD1a (for Langerhans cells) were 1950;62(9):351-355. doi:10.1111/j.1365-2133.1950.tb15475.x. negative in the principal tumor cells. Ki-67 nuclear staining for cells in S-phase was absent in the round cells but present among the dispersed inflammatory cells. COMMENTS AND OPINIONS Comment. Zak6 called attention to the round ganglionlike aspect of the principal cells in solitary epithelioid histiocytoma. Their capacious, often partially xanthomatized cytoplasm with a deeply eosinophilic or amphophilic char- acter, their large nuclei with a distinct and sometimes large Obviating Endophthalmitis After nucleolus, the absence of a desmoplastic stroma, and fre- Cataract Surgery: Excellent Wound Closure quent surrounding lacunae permitting definition of the cell Is the Key borders set the lesion apart. Granulocytes and T lymphocytes (but not B lymphocytes) are admixed within the le- e congratulate Dr Raizman on his excellent, sion. The tumor cells are CD163, CD68, and ␣1-antitrypsin timely Editorial1 in the April 2011 issue of positive (histiocytic markers) with erratic and focal stain- W the Archives. In discussing the importance ing for S-100 protein and Mitf (the last feature being absent of adequate wound closure in his final paragraph, we in our case).1 They are negative for keratin, CD1a (Langer- believe he has highlighted the single most important hans cells), melan A, and HMB-45 (melanoma or Spitz factor in preventing endophthalmitis after cataract nevus) and have a very low Ki-67 index (Ͻ1%); mitotic surgery. figures can occasionally be observed.1,4 The differential di- We in the state of New South Wales, Australia, have agnosis should focus on sarcoidosis (which has a tubercu- repeatedly published that we unfortunately own the high- loid pattern), Rosai-Dorfman disease (strongly S-100 est documented endophthalmitis rate in the world.2,3 We protein positive) with emperipolesis, elevated tuberous xan- believe this is due to failure to achieve “excellent clo- thoma, juvenile and adult xanthogranulomas with Touton sure of the incisions.”1(p502) giant cells, Erdheim-Chester disease with Touton cells and There is overwhelming evidence that clear corneal in- marked fibrosis, necrobiotic xanthogranuloma, Langerhans cisions managed by stromal hydration alone are not self- cell histiocytosis, and epithelioid sarcoma.1 sealing.4,5 Moreover, anatomical closure of this nature does not endure beyond 20 minutes.3 It is also known that the Frederick A. Jakobiec, MD, DSc eye becomes more deformable with blinking after the de- Maria Kirzhner, MD velopment of the usual postoperative ocular hypotony 3 Marlene Morales Tollett, MD hours following surgery.6 We have previously described Ronald Mancini, MD this as the “sucking corneal wound.”3 This wound, when R. Nick Hogan, MD, PhD not adequately closed, provides repeated opportunities for the ingress of organisms into the eye, occurring with Author Affiliations: Department of Ophthalmology, Da- each blink. vid G. Cogan Laboratory of Ophthalmic Pathology, Mas- Future research comparing topical antibiotics and in- sachusetts Eye and Ear Infirmary and Harvard Medical tracameral antibiotics in preventing endophthalmitis will

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