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Diffuse siliconoma of the eyelid

Published:October 18, 2018DOI:https://doi.org/10.1016/j.jcjo.2018.08.003
      Silicone oil is widely used as a vitreous substitute in vitreoretinal surgeries.
      • Morphis G
      • Irigoyen C
      • Eleuteri A
      • Stappler T
      • Pearce I
      • Heimann H
      Retrospective review of 50 eyes with long-term silicone oil tamponade for more than 12 months.
      It is most frequently indicated in complex cases of retinal detachments and proliferative diabetic retinopathy associated with tractional retinal detachment, as well as ocular trauma to stabilize the retina and inhibit proliferative activity.
      • Morphis G
      • Irigoyen C
      • Eleuteri A
      • Stappler T
      • Pearce I
      • Heimann H
      Retrospective review of 50 eyes with long-term silicone oil tamponade for more than 12 months.
      Liquid silicone was first injected into the vitreous cavity of rabbit eyes in 1958, and then utilized for the treatment of retinal detachments in 1962 by Cibis et al.
      • Cibis P
      • Becker B
      • Okun E
      • Canaan S
      The use of liquid silicone in retinal detachment surgery.
      Several complications have been reported: including cataract formation; band keratopathy; rubeosis iridis; optic neuropathy; glaucoma; chronic uveitis; migration into the lateral ventricles of the brain; posterior ciliary artery occlusion; epiretinal membranes; and episcleral, subconjunctival, and orbital foreign body granulomas.
      • Morphis G
      • Irigoyen C
      • Eleuteri A
      • Stappler T
      • Pearce I
      • Heimann H
      Retrospective review of 50 eyes with long-term silicone oil tamponade for more than 12 months.
      • Cibis P
      • Becker B
      • Okun E
      • Canaan S
      The use of liquid silicone in retinal detachment surgery.
      • Shin H
      • Lemke BN
      • Stevens TS
      • Lim MJ
      Posterior ciliary-artery occlusion after subcutaneous silicone-oil injection.
      • Federman JL
      • Schubert HD
      Complications associated with the use of silicone oil in 150 eyes after retina-vitreous surgery.
      • Srinivasan S
      • Singh AK
      • Desai SP
      • Talbot JF
      • Parsons MA
      Foreign body episcleral granulomas complicating intravitreal silicone oil tamponade: a clinicopathological study.
      • Lee JH
      • Kim YD
      • Woo KI
      • Kong M
      Subconjunctival and orbital silicone oil granuloma (siliconoma) complicating intravitreal silicone oil tamponade.
      We report a rare case of silicone oil migration into the upper eyelid that was discovered during levator advancement surgery in a patient presenting with ptosis and an extensive history of retinal procedures.

      Case Report

      A 56-year-old man presented with ptosis and thickening of the left upper eyelid (Fig. 1A). Three years previously, he underwent numerous procedures in the U.K. for a recalcitrant retinal detachment of the left eye, including laser photocoagulation, cryotherapy, scleral buckling, pars plana vitrectomy and lensectomy, gas injection, infusion of intraocular silicone oil, retinotomies, and partial retinectomy, resulting in anatomic success with reattachment of the retina.
      Fig 1
      Fig. 1A. Thickening and ptosis of the left upper eyelid. B. Surgical incision disclosed an oily material (arrow) diffusely seeping from the left upper eyelid. Histopathologic examination showed clusters of variably sized clear vacuoles, presumably silicone oil. C. With surrounding foreign body multinucleated giant cells. D. Hematoxylin and eosin, original magnifications x 400 (C), x 250 (D). E. Positive brown staining with CD68, consistent with macrophages engulfing silicone oil (original magnifications x 500). F. Electron micrograph shows vacuoles of variable size and shape within the cytoplasm of macrophages (original magnification x 4000).
      Ocular examination disclosed a best corrected visual acuity of 20/20 in the right eye and count fingers vision in the left eye. The left upper and lower eyelids manifested what appeared to be xanthelasma-like changes associated with yellowish upper eyelid thickening. Four millimetres of ptosis was present in his left upper eyelid. Levator function was 15 mm bilaterally. He demonstrated a 20-prism diopter left hypertropia and a left enlarged, irregular postsurgical pupil. The retina was attached with evidence of laser retinopexy scars and mild inferior retinal scarring.
      A left levator advancement procedure was performed with repositioning of the lacrimal gland and graded excisions of prolapsed orbital fat. Intraoperatively, the entire left upper eyelid contained numerous clear fluid-filled cysts, 0.5 to 1.0 mm in diameter, encountered in the scarred pre-aponeurotic fat, and the levator and Muller's muscles (Fig. 1B). This vitreous-like appearance prompted intraoperative indirect ophthalmoscopy to assure integrity of the globe. The subcutaneous dermal layer had a striking yellow appearance and was grossly thickened to 4 to 5 times normal. The thickened skin was biopsied.
      Three weeks postoperatively, residual ptosis was evident. A revision of the levator advancement was performed, and additional biopsy specimens were obtained. Fifteen years postoperatively, eyelid height and contour were satisfactory, and the patient was pleased with the cosmetic improvement.

      Pathologic Findings

      Histopathological examination of the biopsy specimens obtained from the left upper eyelid disclosed a normal epidermis and the presence of large clusters of tightly packed, variably sized, clear vacuoles in association with numerous histiocytes (Fig. 1C). A fair number of multinucleated giant cells, which contained similar vacuoles, were evident (Fig. 1D). The vacuoles did not show birefringence under polarized light, nor did they stain with periodic acid-Schiff or special stains for glycosaminoglycans. Immunohistochemical studies with CD68 staining the histiocytes (Fig. 1E).
      Electron microscopic examination disclosed clear vacuoles of variable size and shape, localized within the cytoplasm of histiocytes (Fig. 1F). A number of the vacuoles, especially the smaller ones, were membrane-bound. Small irregular foci of an amorphous material were evident within the vacuoles. The vacuoles tended to mold and displace the nucleus of some histiocytes. These findings were consistent with a histiocytic response to foreign lipid material, presumably silicone oil.

      Discussion

      Silicone oil is an established treatment for achieving retinal tamponade to treat complex vitreoretinal diseases. Numerous complications have been associated with this treatment, as described above.
      A potential etiology of postoperative ptosis is thought to be due to a dehiscence or disinsertion of the levator aponeurosis during surgery.
      • Deady JP
      • Price NJ
      • Sutton GA
      Ptosis following cataract and trabeculectomy surgery.
      In our patient, the etiology was likely multifactorial with contributions from surgical manipulation, increased weight of the eyelid, and aponeurotic changes secondary to local infiltration of the silicone oil.
      With regard to the mechanism of silicone oil extravasation from the vitreous chamber into extraocular tissues, various routes have been reported. Federman and Schubert
      • Federman JL
      • Schubert HD
      Complications associated with the use of silicone oil in 150 eyes after retina-vitreous surgery.
      reported an outflow of silicone oil through the sclerotomy sites in 3% of eyes receiving this therapy. Lee et al.
      • Lee JH
      • Kim YD
      • Woo KI
      • Kong M
      Subconjunctival and orbital silicone oil granuloma (siliconoma) complicating intravitreal silicone oil tamponade.
      reported a case of silicone oil leakage through melted sclera in a patient with endophthalmitis and noted other cases with silicone oil leakage through an Ahmed glaucoma valve. We postulate that the silicone oil in our case may have escaped from the interior of the globe through previous scleral vitrectomy wounds and infiltrated the superior rectus-levator complex from the subconjunctival space, which caused the ptosis and thickened eyelid appearance.
      Within the English ophthalmic literature, we found 7 other cases of silicone oil migration to the eyelid from intraocular silicone oil use.
      • Lee JH
      • Kim YD
      • Woo KI
      • Kong M
      Subconjunctival and orbital silicone oil granuloma (siliconoma) complicating intravitreal silicone oil tamponade.
      ,
      • Deguchi Y
      • Maeno T
      • Hori Y
      • Hiruta N
      • Sasai D
      • Sato Y
      Migration of intraocular silicone oil from the vitreous cavity into the upper eyelid causing ptosis.
      • Dehghani A
      • Rezaei L
      • Tavallali A
      • Dastborhan Z
      Upper eyelid silicone oil migration after sutureless 23-gauge vitrectomy.
      • Donker DLT
      • Paridaens D
      • Mooy CM
      • van den Bosch WA
      Blepharoptosis and upper eyelid swelling due to lipogranulomatous inflammation caused by silicone oil.
      • Osaki TH
      • Osaki MH
      • Allemann N
      • Osaki T
      Silicone migration: an unusual eyelid complication following intraocular surgery.
      • Quintyn JC
      • Genevois O
      • Ranty ML
      • Retout A
      Silicone oil migration in the eyelid after vitrectomy for retinal detachment.
      Our patient showed more extensive disease than the others published, which was consistent with the diagnosis of diffuse siliconoma. We also are the first to illustrate the associated pathological intracytoplasmic, clear vacuoles engulfed by macrophages using electron microscopy.

      Conclusion

      In conclusion, we report a case of a diffuse siliconoma of the upper eyelid, secondary to extraocular silicone oil infiltration after retinovitreous surgery. In evaluating ptosis, patients with a history of intraocular silicone oil tamponade and the clinical appearance of a thickened eyelid should suggest silicone oil infiltration as a potential cause.

      Disclosure

      The authors have no proprietary or commercial interest in any materials discussed in this article.

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