Ophthal Plast Reconstr Surg, Vol. 31, No. 1, 2015

Case Reports

madarosis. The lesion appeared cystic with yellowish content. There was no evidence of tarsal or conjunctival involvement. The lesion was completely excised, and the defect was repaired with an anterior lamellar pedicle flap. His postoperative course was uncomplicated, and he had no recurrence at his 1-month follow-up appointment. Histopathology showed a nodular proliferation of cells with moderate-to-abundant granular eosinophilic cytoplasm and small round nuclei (Fig. 2). There was positive staining for S100 and vimentin but negative smooth muscle actin immunostain. There was no evidence of malignancy, and the margins were clear.

DISCUSSION Although granular cell tumors were first described in 1926, there has been debate on the cellular origin. Recently, it is thought that they most likely originate from Schwann cells.11,22 The theory of Schwann cell origin is supported by the presence of S100 protein, intracytoplastmic myelin, and basement membrane. Previous reviews have shown that they can occur in the lacrimal sac, conjunctiva, uvea, eyelids, and orbits11; however, they are still more common in other areas throughout the body. Many lesions classically masquerade as chalazion; however, physicians often do not routinely biopsy these lesions. Though this tumor was benign, granular cell tumors can rarely be malignant. Muqit et al.23 recently showed that chalazion was the most common cause of misdiagnosis and delay in diagnosis of eyelid sebaceous cell carcinoma. Lesions that were diagnosed within 6 months had a 14% 5-year mortality, whereas those that were >6 months had a 38% 5-year mortality. Ozdal et al.24 compared clinical and histological diagnosis and reported that 6.4% of lesions diagnosed as chalazion are misdiagnosed lesions. Most of these lesions are benign; however, 0.2% were found to be malignant. This patient had a 2-year delay in diagnosis and treatment. One could argue that given the relative rarity of malignant lesions misdiagnosed as chalazion at the time of excision in the Ozdal et al.24 study, routine biopsy is not warranted. However, the Muqit et al.23 study did show that 43% of sebaceous cell carcinomas were diagnosed as chalazion, and chalazion was the most common cause of delay in diagnosis. Although this patient was not found to have a malignant disease, if the lesion had previously been completely excised and evaluated by pathologic study, the patient could have avoided an additional operation and the potential for further morbidity.

REFERENCES 1. González-Almaraz G, de Buen S, Tsutsumi V. Granular cell tumor (myoblastoma) of the orbit. Am J Ophthalmol 1975;79:606–12. 2. Müller W, Dahmen HG. Granular cell tumor of the optic nerve. Albrecht Von Graefes Arch Klin Exp Ophthalmol 1978;207:181–8. 3. Drummond JW, Hall DL, Steen WH Jr, et al. Granular cell tumor (myoblastoma) of the orbit. Arch Ophthalmol 1979;97:1492–4. 4. Karcioglu ZA, Hemphill GL, Wool BM. Granular cell tumor of the orbit: case report and review of the literature. Ophthalmic Surg 1983;14:125–9. 5. Singleton EM, Nettleship MB. Granular cell tumor of the orbit: a case report. Ann Ophthalmol 1983;15:881–3. 6. Ueda N, Yoshida A, Yokota T, et al. Granular cell tumor of the orbit. Appl Pathol 1986;4:179–85. 7. Morgenstern C, Lipman H, Grüntzig J. [Granular cell tumor of the orbits. Diagnosis and therapy]. Laryngol Rhinol Otol (Stuttg) 1986;65:691–2. 8. Dolman PJ, Rootman J, Dolman CL. Infiltrating orbital granular cell tumour: a case report and literature review. Br J Ophthalmol 1987;71:47–53. 9. Jaeger MJ, Green WR, Miller NR, et al. Granular cell tumor of the orbit and ocular adnexae. Surv Ophthalmol 1987;31:417–23. 10. Kocks W, Mohr C, Richter HJ, et al. [Granular cell tumor of the orbit]. Neurochirurgia (Stuttg) 1988;31:213–5.

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11. Moseley I. Granular cell tumour of the orbit: radiological findings. Neuroradiology 1991;33:399–402. 12. McNab AA, Daniel SE. Granular cell tumours of the orbit. Aust N Z J Ophthalmol 1991;19:21–7. 13. Rodríguez-Ares T, Varela-Durán J, Sánchez-Salorio M, et al. Granular cell tumor of the eye (myoblastoma): ultrastructural and immunohistochemical studies. Eur J Ophthalmol 1993;3:47–52. 14. Allaire GS, Laflamme P, Bourgouin P. Granular cell tumour of the orbit. Can J Ophthalmol 1995;30:151–3. 15. Ahdoot M, Rodgers IR. Granular cell tumor of the orbit: magnetic resonance imaging characteristics. Ophthal Plast Reconstr Surg 2005;21:395–7. 16. Golio DI, Prabhu S, Hauck EF, et al. Surgical resection of locally advanced granular cell tumor of the orbit. J Craniofac Surg 2006;17:594–8. 17. Sterker I, Hagert-Winkler A, Gradistanac T, et al. [Granular cell tumor of the orbit]. Ophthalmologe 2007;104:803–5. 18. Poyraz CE, Kiratli H, Söylemezoğlu F. Granular cell tumor of the inferior rectus muscle. Korean J Ophthalmol 2009;23:43–5. 19. Guerriero S, Giancipoli G, Sborgia A, et al. Orbital granular cell tumor in a patient with Churg Strauss syndrome: the importance of biopsy. Orbit 2011;30:30–3. 20. Ribeiro SF, Chahud F, Cruz AA. Oculomotor disturbances due to granular cell tumor. Ophthal Plast Reconstr Surg 2012;28: e23–7. 21. Fernandes BF, Belfort Neto R, Odashiro AN, et al. Clinical and histopathological features of orbital granular cell tumor: case report. Arq Bras Oftalmol 2012;75:137–9. 22. Bregman DK, Hodges T, La Piana FG. Granular cell tumor of the eyelid. Ann Ophthalmol 1991;23:106–7. 23. Muqit MM, Foot B, Walters SJ, et al. Observational prospective cohort study of patients with newly-diagnosed ocular sebaceous carcinoma. Br J Ophthalmol 2013;97:47–51. 24. Ozdal PC, Codère F, Callejo S, et al. Accuracy of the clinical diagnosis of chalazion. Eye (Lond) 2004;18:135–8.

Epiblepharon-Induced Head Tilt Masquerading as Torticollis Benjamin P. Erickson, M.D., and Wendy W. Lee, M.D. Abstract: A 10-month-old girl presented for ocular evaluation carrying a provisional diagnosis of torticollis. Her family reported that for the past 5 months, she consistently tilted her head to the left while twisting her chin toward the right shoulder. Her adnexal examination was notable for epiblepharon, with greater ciliary-corneal contact in the OS. It was therefore hypothesized that this posture was adopted to minimize ocular irritation. Her symptoms resolved immediately following a Hotz-Celsus procedure. To the best of the authors’ knowledge, this is the first report of asymmetric ciliary-corneal contact from epiblepharon, resulting in preference for a head position mimicking a musculoskeletal abnormality such as torticollis.

CASE REPORT The mother of a 10-month-old girl presented to our institution, seeking a second opinion. The girl had no other ocular history but had been diagnosed with torticollis at 5 months of age due to a strong preference for tilting her head to the left while twisting her chin toward the right shoulder. She was born fullterm via spontaneous vaginal delivery and had consistently met all developmental milestones. She had been otherwise healthy Accepted for publication August 21, 2013. Bascom Palmer Eye Institute, Miami, Florida, U.S.A. The authors have no financial or conflicts of interest to disclose. Address correspondence and reprint requests to Benjamin P. Erickson, M.D., Bascom Palmer Eye Institute, Miami, Florida 900 NW 17th St., Miami, FL. E-mail: [email protected]

© 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.

Ophthal Plast Reconstr Surg, Vol. 31, No. 1, 2015

Case Reports

DISCUSSION

FIG. 1. 

Epiblepharon is a congenital eyelid abnormality characterized by a redundant medial fold of skin and underlying orbicularis with inversion of the adjacent eyelashes. These abnormalities were traditionally thought to be cause and effect, but recent evidence suggests that tissue redundancy has a limited contribution to eyelash position.1 Recent histological studies have also failed to provide evidence of orbularis hypertrophy as was previously suggested.2 The fundamental deficit is now thought by many to be a weak attachment between the lower eyelid retractors and the anterior lamella.3 This condition tends to spontaneously resolve by 3 to 4 years of age as progressive growth of the facial skeleton exerts greater tension on the retractors.4 When inverted eyelashes come in contact with the ocular surface, this causes conjunctival and corneal irritation or even erosion.5 Symptoms may be particularly prominent in downgaze as this position maximizes ciliary-corneal contact.6 In severe cases, eyelid squeezing prompted by foreign body sensation can be powerful enough to induce significant astigmatism. To the best of their knowledge, this is the first report of asymmetric ciliary-corneal contact, resulting in preference for a head position mimicking a musculoskeletal abnormality such as torticollis. This highlights the need for ophthalmic evaluation of young patients with abnormal head position, with close attention to the ocular adnexa in addition to eye motility patterns.

REFERENCES

FIG. 2. 

apart from infrequent sinusitis. Her family did report that an older sibling had experienced similar symptoms to a lesser degree. As a result of her abnormal posture, she was evaluated by a pediatric neurologist who prescribed physical therapy and ordered neuroimaging. MRI of the brain and cervical spine demonstrated age-appropriate myelination and gray-white differentiation, and several weeks of therapy had no appreciable benefit. During the initial ophthalmic examination, she was restless and uncomfortable. She could fix and follow but had a reduced attention span. Her ocular motility was full without nystagmus, and a Bruckner test revealed symmetric red reflexes. There were no other deficits observed to suggest congenital fourth nerve palsy. Her pupils were symmetric and reactive with no afferent pupillary defect, and her globes were soft to palpation. Her adnexal examination was notable for epiblepharon, and the lashes of her lower eyelids were noted to rub the central cornea, more so on the left side. The inferior tarsal plates were found to have a normal relationship to the globe, and downward distraction of the skin with finger pressure was observed to restore the cilia to a normal anatomical position. It was therefore hypothesized that her abnormal posture might serve to reduce ocular surface irritation by minimizing ciliary-corneal contact (Fig. 1). After careful discussion, the girl’s family elected a period of observation prior to surgical intervention. Her epiblepharon did not spontaneously resolve, and she underwent a Hotz-Celsus procedure at 18 months of age. After a standard ophthalmic preparation, subciliary incisions were made 2 mm below the eyelid margins, and skin ellipses were marked for excision, using the pinch technique. Skin-muscle flaps were then sharply dissected and removed. The resulting wounds were closed with 7-0 absorbable suture. The girl tolerated this procedure well, and her head tilt resolved immediately following extubation (Fig. 2). The clinical care and photographic documentation for this case was handled in compliance with Health Insurance Portability and Accountability Act and relevant institutional regulations.

1. Kakizaki H, Leibovitch I, Takahashi Y, et al. Eyelash inversion in epiblepharon: is it caused by redundant skin? Clin Ophthalmol 2009;3:247–50. 2. Chang M, Lee TS, Yoo E, et al. Surgical correction for lower lid epiblepharon using thermal contraction of the tarsus and lower lid retractor without lash rotating sutures. Br J Ophthalmol 2011;95:1675–8. 3. Hwang SW, Khwarg SI, Kim JH, et al. Lid margin split in the surgical correction of epiblepharon. Acta Ophthalmol 2008;86:87–90. 4. Kakizaki H, Takahashi Y, Kang H, et al. No histological evidence of orbicularis oculi muscle hypertrophy in congenital epiblepharon. Clin Experiment Ophthalmol 2013;41:167–71. 5. Naik MN, Ali MJ, Das S, et al. Nonsurgical management of epiblepharon using hyaluronic acid gel. Ophthal Plast Reconstr Surg 2010;26:215–7. 6. Sundar G, Young SM, Tara S, et al. Epiblepharon in East Asian patients: the Singapore experience. Ophthalmology 2010;117:184–9.

Pilomatrix Carcinoma of the Eyebrow Marian Pauly, M.B.B.S., M.S., D.N.B.*, Krishnakumar Subramanian, M.B.B.S., M.D.†, Giridhar Anantharaman, M.B.B.S., M.S.*, and Eliza Anthony, M.B.B.S.* Abstract: Pilomatrix carcinoma is an uncommon malignant neoplasm originating from the hair follicle matrix. It occurs more frequently in head and neck region and rarely on the eyelid and eyebrow. It is often clinically misdiagnosed. The diagnosis is confirmed by excision biopsy and histopathological examination. A 68-year-old woman presented with a mass on her left eyebrow mimicking sebaceous cyst. An excisional biopsy with 4-mm margin was performed under frozen section. Histopathology demonstrated pilomatrix carcinoma with cystic degeneration. Periocular malignan-

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Epiblepharon-induced head tilt masquerading as torticollis.

A 10-month-old girl presented for ocular evaluation carrying a provisional diagnosis of torticollis. Her family reported that for the past 5 months, s...
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