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Department of Ophthalmology, Hospital de Santa Maria, Lisbon, PortugalDepartment of Ophthalmology, Faculty of Medicine, Universidade de Lisboa, Lisbon, PortugalMoorfields Eye Hospital NHS Foundation Trust, London, United Kingdom
Southampton Eye Unit, University Hospital Southampton Foundation Trust, Southampton, United KingdomMoorfields Eye Hospital NHS Foundation Trust, London, United Kingdom
Moorfields Eye Hospital NHS Foundation Trust, London, United KingdomOcular Oncology Service, St. Bartholomew’s Hospital, London, United KingdomUCL Institute of Ophthalmology, London, United Kingdom
related to vitreous loss, persistent hypotony, capsule rupture, multiple surgeries, delayed wound healing, wound fistulas, and iris incarceration. According to the literature, 82% of cases presented within the first year after intraocular surgery
Epithelial cells that gain access to the anterior chamber can grow and present in 3 classical forms: (i) opaque sheet over the cornea, iris, and angle structures; (ii) epithelial anterior chamber clear cyst; (iii) rarely as a pearl-white solid cyst, also called as iris pearl tumor.
Serous cysts are transparent and often form in a wall of conjunctiva covered by epithelium, whereas pearl cysts are solid, opaque, and most commonly associated with cilia that enter the anterior chamber.
We report a case of ED presenting as an iris pearl tumour 2 years after uncomplicated clear cornea phacoemulsification.
Case Report
A 71-year-old male presented with a 3-month history of a red, painful, and photophobic left eye. His ocular history was significant for bilateral uncomplicated clear corneal phacoemulsification with posterior chamber intraocular lens (PC-IOL) implantation 2 years previously, and his family and medical history were unremarkable. There was no history of trauma. Visual acuity (VA) was 6/9 OD and 6/18 OS. OS biomicroscopy showed cilioconjunctival hyperemia, +4 anterior chamber cells, and a vascularized dome-shaped amelanotic iris mass at the 12 o’clock position, with a splashing of pigment over the surface with a slither hypopyon (Fig. 1A–D). There was also a PC-IOL and no evidence of ED from the section site of his previous cataract surgery. No vitritis was identified, and fundoscopy did not reveal any signs of posterior inflammation. Ultrasound B-scan detected an iris lesion at 12 o’clock with medium internal echogenecity and an elevation of 3.0 mm (Fig. 2A). Anterior segment optical coherence tomography confirmed these measurements (Fig. 2B). Treatment with topical steroids led to an improvement in inflammation but not in the mass dimensions. Uveitis work-up was negative, so an excisional biopsy by broad iridectomy was performed. Microbiology analysis of the aqueous aspirate was negative. Histopathology findings were of iris tissue partly covered by stratified squamous epithelium reminiscent of ocular surface epithelium.
Fig. 1External photograph of the left eye. (A) Image shows a vascularized dome-shaped amelanotic iris mass at the 12 o’clock position, with a splashing of pigment. (B) Magnified image of the lesion and cornea, where mild edema and keratic precipitates can also be identified. (C) Image shows cilioconjunctival hyperemia with a discrete hypopyon. (D) Gonioscopic view of lesion showing an elevated lesion with no corneal contact. These features suggested a neoplastic process.
Fig. 2(A) Ultrasonography of left eye detected an elevated iris lesion at the 12 o’clock position with medium internal echogenecity, a transverse base of 4.8 mm, a longitudinal base of 3.2 mm, and an elevation of 3.0 mm. (B) Anterior segment optical coherence tomography confirmed ocular ultrasonography measures.
The overall appearance was suggestive of ED (Fig. 3). There was no evidence of neoplasia.
Fig. 3Hematoxylin and eosin staining (×10 objective): cystic space containing keratin flakes and debris, lined by stratified squamous epithelium. The wall includes iris stroma and pigment epithelium.
The eye resolved well with topical medication. At 24 months’ follow-up, VA was 6/9, and there was no evidence of inflammation or recurrence (Fig. 4A–C).
Fig. 4Photographs of the left eye after broad iridectomy using different slit-lamp illumination techniques: (A) diffuse illumination, (B) tangential illumination, and (C) retroillumination, which show a clear cornea, residual scar tissue at the capsulorhexis margin and no recurrence of the lesion.
when intracapsular and extracapsular extraction techniques were used. With advances in cataract surgery, sutureless phacoemulsification has become the procedure of choice for most surgeons. It allows for a smaller wound with better closure that results in better visual acuity with a lower rate of complications,
Phacoemulsification with posterior chamber intraocular lens versus extracapsular cataract extraction (ECCE) with posterior chamber intraocular lens for age-related cataract.
reported the cystic form, namely, the serous cyst, presenting 3 years after cataract surgery and managed successfully by surgical excision. To the best of our knowledge, there have been no reports of pearl cyst formation after uncomplicated clear cornea phacoemulsification. In general, the cystic forms have been considered more easily managed because of prompt detection and better surgical eradication.
Other reported successful treatment options for implantation anterior chamber cysts have included aspiration of cyst contents and endolaser photocoagulation of the residual cyst wall, aspiration and intralesional administration of mitomycin C, or more invasive vitrectomy and local cryoablation of adjacent cyst wall.
but uniquely now has been found after uncomplicated phacoemulsification. It presented as a solid iris lesion, as documented by imaging, suggesting a neoplastic process. The natural sequence of events has been described after implantation of cilia into the iris after trauma: formation of the pearl cyst, followed by iridocyclitis, and then by enlargement of the cyst to cause secondary glaucoma.
reported a case of pseudophakic chronic aggressive inflammation that later was associated with the development of a vascularized infiltrative iris mass, which was proved to be a metaplastic ED of a limbal squamous cell carcinoma with secondary intraocular inflammation. Therefore, malignant and other etiologies of iris cysts, such as congenital cysts, gummas, and tubercles, should be included in the differential diagnosis.
Surgical treatment was the treatment of choice, and during follow-up, no recurrence was identified. This is in accordance with previous reports that show that in patients with ED, surgical treatment led to fewer enucleations than in those treated medically or not treated.
In summary, ED is a very rare complication of modern cataract surgery. This case shows that, in this setting, in addition to the previously described serous cystic and sheet-like growth forms, epithelial growth can also present as an implantation pearl cyst. Clinicians should have a high index of suspicion for this condition in pseudophakic patients when more usual conditions have been excluded. Appropriate treatment includes early surgical excision, which may be associated with a more favourable prognosis.
Disclosure
The authors have no proprietary or commercial interest in any materials discussed in this article.
References
Weiner M.J.
Trentacoste J.
Pon D.M.
Albert D.M.
Epithelial downgrowth: a 30-year clinicopathological review.
Phacoemulsification with posterior chamber intraocular lens versus extracapsular cataract extraction (ECCE) with posterior chamber intraocular lens for age-related cataract.
☆This work was carried out at the Oncology Clinic, Moorfields Eye Hospital NHS Foundation Trust, London, United Kingdom.
☆☆Presented at the 5th EUCORNEA Congress, London, UK, September 12–13, 2014, and at the International Society of Ocular Oncology’s 2015 Biennial Meeting, June 18, 2015, Paris, France.
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