Skip to main content
NCBI home page
BMJ Case Reports logoLink to BMJ Case Reports
. 2016 Oct 4;2016:bcr2016216486. doi: 10.1136/bcr-2016-216486

Massive iatrogenic orbital cysts following glaucoma drainage implant and strabismus surgery

Elif Demirkilinc Biler 1, Ayse Yagci 1, Onder Uretmen 1, Melis Palamar 1
PMCID: PMC5073670  PMID: 27702931

Abstract

We describe 2 paediatric patients who developed large orbital conjunctival epithelium-lined inclusion cysts postsurgery. Case 1 underwent Ahmed glaucoma valve implant surgery 8 months ago and case 2 underwent strabismus surgery 7 years ago. Both cases had either symptomatic strabismus or a mass causing some degree of proptosis or incomitance due to lesions in the intraorbital space. Following total excision of the cysts all associated symptoms were resolved. Conjunctival cysts following ocular surgery can develop anywhere in the anterior segment; however, large orbital cysts following anterior segment surgery are quite rare. We think that implantation cysts should be considered in the differential diagnosis of patients that have signs of orbital mass with a history of previous anterior segment surgery. Complete excision of the cysts results in resolution of all associated signs and symptoms.

Background

Conjunctival epithelial inclusion cysts are not uncommon and can develop following trauma or ocular surgeries such as strabismus surgery, oculopalpebral surgery, enucleation, and scleral buckling surgery. These cysts have been reported to originate from dislocated epithelium below the surface of the conjunctiva following trauma or during ocular surgery.1 2 The cysts are often located in the periocular region, including the conjunctiva, caruncle and eyelids, and can be detected via slit lamp examination;3 however, large epithelial inclusion cysts extending into the orbit rarely occur. These lesions cause symptoms similar to those associated with orbital tumours. Cases have been reported, primarily as a rare and late complication of strabismus surgery.3–7 The incidence of conjunctival cysts following an otherwise uncomplicated strabismus surgery was reported to be 0.25%.8 Moreover, formation of orbital conjunctival inclusion cysts following various oculopalpebral surgeries has been reported in adults.9–12 To the best of our knowledge orbital inclusion cyst formation following drainage implant (Ahmed glaucoma valve (AGV)) glaucoma surgery and posterior migration of the AGV have not been previously reported.

We report two paediatric patients with intraorbital epithelial inclusion cysts following strabismus surgery (n=1) and AGV implantation surgery (n=1) that caused mechanical restriction of eye movement due to mass effect and proptosis. Ocular history, ophthalmic findings, surgical management, clinical course, and presurgical and postsurgical orbital MRI findings are discussed in detail.

Case presentation

Case 1

A 2-year-old girl presented with proptosis and decreased vertical gaze in the right eye. She had a history of surgery for bilateral congenital cataract at the age of 3 months. During follow-up, right aphakic glaucoma had developed and both endoscopic cyclophotocoagulation and glaucoma surgery were performed 2 months apart at another ophthalmology clinic. An AGV implantation was performed in a glaucoma surgery performed 8 months ago elsewhere. Based on anamnesis, no peroperative or postoperative complications were reported.

During primary gaze the right globe exhibited downward dystopia. She had a steady fixation and good ocular pursuit of moving test objects with that eye. She had a low intraocular pressure (IOP) without any medication. Motility examination showed restriction in both upper and, particularly, lower gaze in her right eye. The tube of the AGV was not observed in the anterior chamber and via slit lamp examination no mass was observed in anterior ocular tissues, including the fornices. MRI showed a 19×16 mm extraconal cystic lesion involving the lateral and superior quadrants next to the globe. The cyst had a smooth wall and homogeneously reflective contents, and pushed the globe downwards. At the periphery of the cystic lesion there was a hyporeflective object considered to be the AGV (figure 1A–C).

Figure 1.

Figure 1

Open in a new tab

Preoperative and postoperative MRIs of case 1. (A,B,C) Right orbital mass is apparent in the superolateral side of the globe displacing it downwards into the coronal and sagittal sections in preoperative images. A hyporeflective object considered to be the Ahmed glaucoma valve adjacent to the lesion. (B and C) No residual lesion is seen in postoperative control MRI (D and E).

Anterior transcutaneous orbitotomy was performed under general anaesthesia. Stallard-Wright incision was used to gain access to the subperiosteal space and the extraconally located cyst. The cyst's contents were stained with methylene blue and the walls were dissected from orbital tissues using blunt manoeuvres. Total excision of the cyst was performed and the AVG implant, which was in the cyst but not in contact with the globe, was also explanted (figure 2). Postsurgery proptosis resolved and there was no longer any gaze restriction or vertical deviation. Histopathological examination of the cyst showed that it was an epithelial cyst. IOP in the right eye was still normal with no medication and no cystic lesion was observed via postoperative follow-up MRI (figure 1C, D), and at 1 year postsurgery the eye remained cyst-free.

Figure 2.

Figure 2

Open in a new tab

Peroperative images of case 1. Anterior transcutaneous orbitotomy was performed to reach the subperiosteal space and the extraconally located cyst. Total excision of the cyst was performed. The AVG implant was observed above the cyst but not in contact with the globe (black arrow).

Case 2

An 11-year-old boy presented with proptosis, a mass lesion in the nasal conjunctiva of the left eye that was increasing in size, and a decrease in left eye motility over a period of 1 year. He had esotropia of +30 prism diopter (PD), hypotropia of 16 PD, and decreased adduction and upgaze in the left eye (figure 3A). Diplopia was not present, probably due to the slow growth of the lesion and development of suppression. There was prominent proptosis in the left eye based on Hertel exophthalmometry (right eye: 12 mm; left eye: 17 mm) and a cystic mass was observed in the left nasal area via gross and slit lamp examinations. Best-corrected visual acuity in both eyes was 1.0 via Snellen chart. Anamnesis was remarkable for esotropia and uneventful strabismus surgery (bimedial rectus recession) at age 4 years. Orbital MRI showed a well-defined 3.5×1.4×1.8 cm intraorbital cystic lesion that extended into the superonasal quadrant. The cyst was integrated with the left medial rectus, involved the area of insertion, and covered the globe superomedially, which caused temporal and downward displacement of the globe (figure 4A–C). Subconjunctival and intraorbital inclusion epithelial cyst was considered the most likely diagnosis, and excisional biopsy was performed under general anaesthesia. Following conjunctival incision, the cyst wall was bluntly dissected from the surrounding tissues towards the orbita, as much as possible and the cyst's contents were stained with methylene blue to make it more apparent. During surgery the superior portion of the medial rectus was observed to be partially attached to the cyst; as such, isolation of the medial rectus muscle was performed, no suture material was observed and following dissection the cyst was excised totally without any harm to the extraocular muscles (figure 5).

Figure 3.

Figure 3

Open in a new tab

Preoperative (A) and postoperative (B) photographs of case 2, showing ocular movements in nine diagnostic positions of gaze. The cystic mass was apparent in the superonasal portion of the left globe beneath the conjunctiva in the preoperative photographs.

Figure 4.

Figure 4

Open in a new tab

Preoperative and postoperative MRIs of case 2. (A and B) Left intraorbital mass causing temporal and downward displacement of the globe, observed in coronal sections. (C) The lesion was extended into the superonasal quadrant, covering the globe superomedially, seen in the transverse section. (D and E) No residual lesion is seen in post-operative control MRI.

Figure 5.

Figure 5

Open in a new tab

Peroperative images of case 2. (A) Conjunctival incision, (B) the cyst wall was bluntly dissected from the surrounding tissues towards the orbita. (C) Methylene blue was injected inside the cyst to stain it. (D) The superior portion of the medial rectus was observed to be partially attached to the cyst.

Postsurgery proptosis was completely resolved (13 mm based on Hertel exophthalmometry) and ocular movements had improved (figure 3B). Histopathological examination showed that the cyst was an epithelial cyst. Follow-up MRI in early postoperative period showed no residual cystic lesion (figure 4D, E), and at 1 year postsurgery his findings remained stable. A reoperation for residual esotropia has been planned.

Discussion

Epithelial inclusion cysts are filled with clear serous fluid that contains shed cells or gelatinous mucous materials. The cyst wall in most cases includes several layers of non-keratinising epithelium and connective tissue.13 Epithelial inclusion cysts are categorised as primary or secondary/acquired, according their aetiology. The secondary/acquired type usually occurs as a result of detachment of a part of conjunctival epithelium due to surgery or trauma.14 These acquired cysts are benign and can be observed even years after surgery.4 5 15

We presented two paediatric patients with large, intraorbital conjunctival epithelium-lined inclusion cysts, and their MRI findings and clinical management. Both patients developed the cysts as a complication of ophthalmic surgical procedures that included conjunctival incision. We think the cysts in the presented cases most likely developed as a result of epithelial remnants captured by sutures or the suture needle. It was reported that such intraorbital cysts can also develop due to viable epithelial cells being dragged into the subdermal, subconjunctival, or episcleral space, and the associated inflammation.3–5 In some cases the cystic mass may be undetectable, even when carefully performing slit lamp examination.15 In suspected cases, orbital imaging is useful for diagnosing cysts located in the orbital space, differentiating cystic lesions from solid tumours, and planning and performing surgical excision.5 16 Management varies from local excision to observation, based on symptoms, localisation, and type of cyst.16 There are also thermal cautery and marsupialisation methods described in the literature in the management of these cysts particularly the smaller ones.17 18 A mass that causes disfigurement, strabismus, restriction of ocular movement, pressure exerted on the globe, proptosis, or any combination of these (as in the presented patients) is an indication for surgery. Complete excision of such cysts is curative.4–6 15 19 20 In cases with involvement of the rectus muscles special care and extensive dissection are necessary, so as not to cause any muscle damage.15

In addition to following strabismus surgery, conjunctival epithelial inclusion cyst formation can also occur following glaucoma surgery; however, intraorbital cyst formation is rare in paediatric patients. Although AGV implantation is reported to be relatively safe,21 case 1 is unusual, as the AGV migrated into the cyst towards the orbita. To the best of our knowledge case 1 is the first patient to be reported with intraorbital epithelial inclusion cyst formation combined with displacement of an AGV following glaucoma surgery.

In conclusion, although rare, intraorbital implantation cysts can cause significant complications and should be considered among space-occupying lesions in orbita, particularly in patients with a history of ocular surgery. In cases in which the posterior border of the cyst cannot be observed via anterior segment examination, orbital imaging can facilitate detailed assessment of the borders and surgical planning. Based on the two cases presented here, we think all ophthalmologists must be made aware of postoperative intraorbital cyst formation in paediatric patients.

Learning points.

  • Intraorbital implantation cysts can cause significant complications and should be considered among space-occupying lesions in orbita, particularly in patients with a history of ocular surgery.

  • In cases in which the posterior border of the cyst cannot be observed via anterior segment examination, orbital imaging can facilitate detailed assessment of the borders and surgical planning.

  • All ophthalmologists must be made aware of postoperative intraorbital cyst formation in paediatric patients.

Footnotes

Twitter: Follow Melis Palamar at @melispalamar

Contributors: EDB contributed to the data collection, surgery, and writing of the manuscript. AY contributed to the data supervision, surgery, and writing of the manuscript. OU contributed to the writing of the manuscript, and surgery. MP contributed to the study supervision and writing of the manuscript.

Competing interests: None declared.

Patient consent: Obtained.

Provenance and peer review: Not commissioned; externally peer reviewed.

References

  • 1.Shields JA, Shields CL. Tumors of the conjunctiva and cornea. In: Smolin G, Thoft RA, eds. The cornea. Scientific foundations and clinical practice. 3rd edn Boston: Little, Brown, 1994:586–95. [Google Scholar]
  • 2.Ho VT, Rao VM, Flanders AE. Postsurgical conjunctival epithelial cysts. AJNR Am J Neuroradiol 1994;15:1181–3. [PMC free article] [PubMed] [Google Scholar]
  • 3.Cibis GW, Waeltermann JM. Muscle inclusion cyst as a complication of strabismus surgery. Am J Ophthalmol 1985;100:740–1. 10.1016/0002-9394(85)90637-3 [DOI] [PubMed] [Google Scholar]
  • 4.Kushner BJ. Subconjunctival cysts as a complication of strabismus surgery. Arch Ophthalmol 1992;110:1243–5. 10.1001/archopht.1992.01080210061025 [DOI] [PubMed] [Google Scholar]
  • 5.Song JJ, Finger PT, Kurli M et al. Giant secondary conjunctival inclusion cysts: late complication of strabismus surgery. Ophthalmology 2006;113:1049.e1 10.1016/j.ophtha.2006.02.031 [DOI] [PubMed] [Google Scholar]
  • 6.Metz HS, Searl S, Rosenberg P et al. Giant orbital cyst after strabismus surgery. J AAPOS 1999;3:185–7. 10.1016/S1091-8531(99)70065-0 [DOI] [PubMed] [Google Scholar]
  • 7.Ullrich CR, Garola RE, Cibis GW. Bilateral extraocular muscle epithelial inclusion cysts as a complication of strabismus surgery. J AAPOS 2003;7:366–7. 10.1016/S1091853103001745 [DOI] [PubMed] [Google Scholar]
  • 8.Guadilla AM, de Liaño PG, Merino P et al. Conjunctival cysts as a complication after strabismus surgery. J Pediatr Ophthalmol Strabismus 2011;48:298–300. 10.3928/01913913-20100818-02 [DOI] [PubMed] [Google Scholar]
  • 9.Hussin HM, Majid M, Spencer N et al. Large inclusion cyst after ptosis surgery. Acta Ophthalmol Scand 2007;85:121–2. 10.1111/j.1600-0420.2006.00830.x [DOI] [PubMed] [Google Scholar]
  • 10.Jünemann A, Holbach LM. Epithelial giant inclusion cyst 50 years after enucleation without orbital implant. Klin Monbl Augenheilkd 1998;212:127–8. 10.1055/s-2008-1034848 [DOI] [PubMed] [Google Scholar]
  • 11.De Potter P, Kunin AW, Shields CL et al. Massive orbital cyst of the lateral rectus muscle after retinal detachment surgery. Ophthal Plast Reconstr Surg 1993;9:292–7. 10.1097/00002341-199312000-00012 [DOI] [PubMed] [Google Scholar]
  • 12.Johnson DW, Bartley GB, Garrity JA et al. Massive epithelium-lined inclusion cysts after scleral buckling. Am J Ophthalmol 1992;113:439–42. 10.1016/S0002-9394(14)76169-0 [DOI] [PubMed] [Google Scholar]
  • 13.Grossniklaus HE, Green WR, Luckenbach M et al. Conjunctival lesions in adults. A clinical and histopathologic review. Cornea 1987;6:78–116. 10.1097/00003226-198706020-00002 [DOI] [PubMed] [Google Scholar]
  • 14.Jakobiec FA, Bonanno PA, Sigelman J. Conjunctival adnexal cysts and dermoids. Arch Ophthalmol 1978;96:1404–9. 10.1001/archopht.1978.03910060158012 [DOI] [PubMed] [Google Scholar]
  • 15.Curtis TH, Stout AU, Drack AV et al. Giant orbital cysts after strabismus surgery. Am J Ophthalmol 2006;142:697–9. 10.1016/j.ajo.2006.04.045 [DOI] [PubMed] [Google Scholar]
  • 16.Shields JA, Shields CL. Orbital cysts of childhood-classification, clinical features, and management. Surv Ophthalmol 2004;49:281–99. 10.1016/j.survophthal.2004.02.001 [DOI] [PubMed] [Google Scholar]
  • 17.Pereira LS, Hwang TN, McCulley TJ. Marsupialization of orbital conjunctival inclusion cysts related to strabismus surgery. J Pediatr Ophthalmol Strabismus 2009;46:180–1. 10.3928/01913913-20090505-13 [DOI] [PubMed] [Google Scholar]
  • 18.Hawkins AS, Hamming NA. Thermal cautery as a treatment for conjunctival inclusion cyst after strabismus surgery. J AAPOS 2001;5:48–9. 10.1067/mpa.2001.111779 [DOI] [PubMed] [Google Scholar]
  • 19.Başar E, Pazarli H, Ozdemir H et al. Subconjunctival cyst extending into the orbit. Int Ophthalmol 1998;22:341–3. 10.1023/A:1006369104483 [DOI] [PubMed] [Google Scholar]
  • 20.Al-Shehah A, Khan AO. Subconjunctival epithelial inclusion cyst complicating strabismus surgery: early excision is better. Saudi J Ophthalmol 2010;24:27–30. 10.1016/j.sjopt.2009.12.005 [DOI] [PMC free article] [PubMed] [Google Scholar]
  • 21.Kirwan C, O'Keefe M, Lanigan B et al. Ahmed valve drainage implant surgery in the management of paediatric aphakic glaucoma. Br J Ophthalmol 2005;89:855–8. 10.1136/bjo.2004.056143 [DOI] [PMC free article] [PubMed] [Google Scholar]

Articles from BMJ Case Reports are provided here courtesy of BMJ Publishing Group

RESOURCES