- Email: [email protected]
Combined true and pseudoexfoliation in a Saudi patient with co-existing cataract and glaucoma
SJOPT 300
No. of Pages 3, Model NS
Saudi Journal of Ophthalmology (2014) xxx, xxx—xxx 1
Case Report
2
5
Combined true and pseudoexfoliation in a Saudi patient Q1 with co-existing cataract and glaucoma
8
Q2
6 4 7
Sami Alodhayb a,b,⇑; Deepak P. Edward a,c
9
Abstract
10
True exfoliation and pseudoexfoliation of the anterior lens capsule are different conditions, their coexistence is rare. We report a case with clinical findings of unilateral combined true exfoliation and pseudoexfoliation in a Saudi Bedouin that was confirmed histologically. We suggest that high levels of infrared radiation in the desert may have contributed to the capsular delamination.
11 12 13 14
Keywords: True exfoliation, Pseudoexfoliation, Cataract, Glaucoma
15 16
Ó 2014 Production and hosting by Elsevier B.V. on behalf of Saudi Ophthalmological Society, King Saud University. http://dx.doi.org/10.1016/j.sjopt.2014.05.002
17 18 19
Introduction
20
True exfoliation and pseudoexfoliation of the anterior lens capsule are two distinct entities. Their coexistence in the same individual is rare and has only been reported few times internationally and never been documented in Saudi or Arab patient.1–3 In this report we describe a patient with combined true and pseudoexfoliation of the anterior lens capsule in a Saudi patient that was confirmed histopathologically.
21 22 23 24 25 26 27
Case history
28
A 63 year old Saudi Male who is a nomad complaining of gradual decrease in vision of the left eye over 4 years. He had a history of poor vision of the right eye since childhood. There was no history of previous inflammation. His clinical findings on presentation are summarized in Table 1 and illustrated in Fig. 1. The pseudoexfoliation (PXF) materials were observed in the pupillary margin and over the iris surface of the left eye, while the capsular delamination formed outward rolled edges extending from 12:30 o’clock position to 4:00, and
29 30 31 32 33 34 35 36 37 38
then from 4:00 to 8:00 o’clock position. The patient’s pupil dilated poorly and hence we could not comment clinically whether pseudoexfoliative material was present on the anterior lens capsule and were not able to clearly define the extent of capsular delamination beyond the visible 4 mm pupillary area. The patient was put on a fixed combination of Dorzolamide 2.0%/Timolol 0.5% drops and then changed to Brimonidine 0.2%/Timolol 0.5% both of which failed to reduce the IOP below 22 mmHg, plus that the patient was poorly compliant to them, for those reasons he underwent extracapsular cataract extraction and posterior chamber intraocular lens implantation (ECCE + PC IOL) with endoscopic cyclophotocoagulation (ECP) of the left eye. ECCE was chosen over phacoemulsification due to the extensive PXF and delamination of the capsule and the former procedure was thought to be safer in the surgeon’s hand, and as the patient is nomad with a history of poor compliance there was a concern about the safety and efficacy of doing an incisional glaucoma procedure, and ECP was thought to be the safest in his condition as it needs no extra post operative care than the cataract surgery. Intra operatively the pupil was stretched using Kuglen
Received 24 February 2014; received in revised form 10 May 2014; accepted 26 May 2014; available online xxxx. a b c
Q3
Glaucoma Division, King Khaled Eye Specialist Hospital, Riyadh, Saudi Arabia Glaucoma Division, Bin Rushd Ophthalmic Center, Riyadh, Saudi Arabia Wilmer Eye Institute, Johns Hopkins University School of Medicine, Baltimore, USA
⇑ Corresponding author at: Glaucoma Division, King Khaled Eye Specialist Hospital, Riyadh, Saudi Arabia. Tel.: +966 568786232. e-mail address: [email protected] (S. Alodhayb). Peer review under responsibility of Saudi Ophthalmological Society, King Saud University
Access this article online: www.saudiophthaljournal.com Production and hosting by Elsevier www.sciencedirect.com
Q1 Please cite this article in press as: Alodhayb S., Edward D.P. Combined true and pseudoexfoliation in a Saudi patient with co-existing cataract and glaucoma. Saudi J Ophthalmol (2014), http://dx.doi.org/10.1016/j.sjopt.2014.05.002
39 40 41 42 43 44 45 46 47 48 49 50 51 52 53 54 55 56 57 58 59 60
SJOPT 300
No. of Pages 3, Model NS
2
S. Alodhayb, D.P. Edward
Table 1. Summary of pre-operative clinical findings. OD
OS
VA IOP (mm Hg) Cornea A/C Lens
Hand motion 14 Central corneal scarring Deep & quiet Mature cataract, no Pseudoexfoliative (PEX) deposits
A/C angle Fundus B-scan Endothelial cell count
Open grade III, Sampaolesi’s line No view Retina flat 1475
Uncorrected 20/100; pinhole: 20/80 31 Arcus senilis and mild diffuse haze Deep & quiet Dense cataract, dense PEX deposits, and delamination of anterior lens capsule Open grade III, Sampaolesi’s line No view Retina flat 1309
Figure 1. Left eye of patients showing rolled delaminated capsule in the pupillary area (arrowheads) and pseudoexfoliative material at the pupillary border (arrows).
75
Hooks, and the anterior capsulotomy was done with a disposable 30-gauge cystotome using a can opener technique away from the area of the capsular delamination. No difficulties or complications were encountered during this step, the anterior lens capsule was submitted for histopathology. We noted that during ECP an unusually high power of 0.7 Watts was required to elicit a response which was attributed to the presence of PEX material over the ciliary processes. Postoperatively his vision improved to 20/40 with an intraocular pressure of 9–11 mm Hg without medications at the last visit (16 months). The right eye maintained normal IOP throughout the follow up time, but with progressing cataract, which was eventually extracted uneventfully with mild visual improvement due to Amblyopia.
76
Pathology
61 62 63 64 65 66 67 68 69 70 71 72 73 74
82
Pathological examination of the anterior lens capsule was performed and changes described were observed away from the edge of the capsulotomy. The anterior capsule showed thickening and delamination in some areas and in other areas PAS positive fibrillar deposits consistent with pseudoexfoliative material were observed (Fig. 2).
83
Discussion
77 78 79 80 81
84 85 86 87
In this report we describe the patient with unilateral pseudoexfoliation deposits accompanied by delamination of the anterior lens capsule (true exfoliation) in one eye. Clinically the pseudoexfoliation material was visible only at the
pupillary margin but on pathologic examination it was also Q4 noted on the anterior lens capsule. The coexistence of both true exfoliation and pseudoexfoliation is rare and had been reported few times only, once by Küchle et al.,1 in an 81-year-old female with a long history of working with porcelain in an oven. Another case was reported in the Japanese literature in an 83 year old woman without any predisposing factors.2 Delamination of the anterior lens capsule has been reported in patients exposed to prolonged heat or infrared radiation,4–6 intraocular inflammation,7 trauma,8 pseudoexfoliation,9 and as an idiopathic entity.9–11 Our patient being a nomad spent prolonged periods of time in the desert which has high levels of infrared radiation.12 We suggest that such prolonged exposure to infrared radiation in the desert in this Bedouin nomad might have been a risk factor for capsular delamination. The condition however has not been previously reported in Saudi patients. The capsular delamination presented with rolled edges in multiple locations in the pupillary area. Based on the experience during the can opener capsulotomy, we believe that capsular delamination does not interfere with the anterior capsulotomy with a can opener technique. It is possible that the simultaneous presence of both conditions may potentially cause complications during capsulorhexis. Dense
Figure 2. Photomicrograph showing the anterior capsule Top. Showing capsular delamination (PAS; original magnification 20). Bottom: showing iron filing like pseudoexfoliation material on the anterior lens capsule (PAS; original magnification 20).
Q1 Please cite this article in press as: Alodhayb S., Edward D.P. Combined true and pseudoexfoliation in a Saudi patient with co-existing cataract and glaucoma. Saudi J Ophthalmol (2014), http://dx.doi.org/10.1016/j.sjopt.2014.05.002
88 89 90 91 92 93 94 95 96 97 98 99 100 101 102 103 104 105 106 107 108 109 110 111 112
SJOPT 300
Q1 Combined true and pseudoexfoliation in a Saudi patient
113 114 115 116 117 118 119 120 121 122 123 124 125 126 127 128 129
130
131
132
133 134 135
pseudoexfoliation deposits might mask capsular delamination and following initiation of capsulorhexis, multiple layers of anterior capsule might be observed causing difficulty in completion of the capsulorhexis. The pathologic features seen in our patient were quite typical of what has been previously described in patients with capsular delamination9–11 and pseudoexfoliation13 as separate entities. It was interesting to note that pseudoexfoliation deposits were not observed in the areas where capsular delamination was prominent. In summary, this case report highlights the presence of pseudoexfoliation and true exfoliation in an eye of a Saudi Bedouin. We suggest that exposure to high levels of infrared radiation in the desert may play a role in capsular delamination. In addition, possible chronic and extended exposure to fire in such individuals who are directly exposed to wood burning fires may play a role in capsular delamination.
Conflict of interest The authors report no conflicts of interest in this work.
References 1. Küchle M, Iliff WJ, Green WR. Combined true exfoliation and pseudoexfoliation of the anterior lens capsule. Klin Monbl Augenheilkd 1996 Feb;208(2):127–9.
No. of Pages 3, Model NS
3 2. Oharazawa H, Suzuki H, Matsui H, Shiwa T, Takahashi H, Ohara K. Two cases of true exfoliation of the lens capsule after cataract surgery. J Nippon Med Sch 2007;74(1):55–60. 3. Yamamoto Y, Nakakuki T, Nishino K, Yagyu K, Kishi S, Ueno H, et al. Histological and clinical study of eyes with true exfoliation and a double-ring sign on the anterior lens capsule. Can J Ophthalmol 2009 Dec;44(6):657–62. 4. Elschnig A. Detachment of the zonular lamellae in glassblowers. Klin Monatsbl Augenheilkd 1922;69:732–4. 5. Burde RM, Bresnick G, Uhrhammer J. True exfoliation of the lens capsule: an electron microscope study. Arch Ophthalmol 1969;82:651–3. 6. Callahan A, Klien BA. Thermal detachment of the anterior lamella of the anterior lens capsule. Arch Ophthalmol 1958;59:73–80. 7. Elschnig A. Uber die Ablosung der Zonulalamelle. Klin Monatsbl Augenheilkd 1926;76:66–70. 8. Butler TH. Capsular glaucoma. Trans Ophthalmol Soc UK 1938;68:575–89. 9. Brodrick JD, Tate Jr GW. Capsular delamination (true exfoliation) of the lens. Report of a case. Arch Ophthalmol 1979;97:1693–8. 10. Radda TM, Klemen UM. Idiopathische echte Exfoliation. Klin Monatsbl Augenheilkd 1982;181:276–7. 11. Cashwell Jr LF, Holleman IL, Weaver RG, van Rens GH. Idiopathic true exfoliation of the lens capsule. Ophthalmology 1989;96:348–51. 12. Al-Khalaf AK. Results of outgoing longwave radiation and Albedo over Saudi Arabia from earth radiation budget experiment (ERBE) data. Met., Env. & Arid Land Agric. Sci. 2006;17(1):77–99. 13. Kincaid M.C. Pathology of the lens. In: Tasman W, Jaeger E.A. (Eds.), Duane’s Foundations of Clinical Ophthalmology, vol. 3. Lippincott Q5 Williams & Wilkins, Philadelphia, PA (Chapter 12).
Q1 Please cite this article in press as: Alodhayb S., Edward D.P. Combined true and pseudoexfoliation in a Saudi patient with co-existing cataract and glaucoma. Saudi J Ophthalmol (2014), http://dx.doi.org/10.1016/j.sjopt.2014.05.002
136 137 138 139 140 141 142 143 144 145 146 147 148 149 150 151 152 153 154 155 156 157 158 159 160 161 162 163 164 165 166
No. of Pages 3, Model NS
Saudi Journal of Ophthalmology (2014) xxx, xxx—xxx 1
Case Report
2
5
Combined true and pseudoexfoliation in a Saudi patient Q1 with co-existing cataract and glaucoma
8
Q2
6 4 7
Sami Alodhayb a,b,⇑; Deepak P. Edward a,c
9
Abstract
10
True exfoliation and pseudoexfoliation of the anterior lens capsule are different conditions, their coexistence is rare. We report a case with clinical findings of unilateral combined true exfoliation and pseudoexfoliation in a Saudi Bedouin that was confirmed histologically. We suggest that high levels of infrared radiation in the desert may have contributed to the capsular delamination.
11 12 13 14
Keywords: True exfoliation, Pseudoexfoliation, Cataract, Glaucoma
15 16
Ó 2014 Production and hosting by Elsevier B.V. on behalf of Saudi Ophthalmological Society, King Saud University. http://dx.doi.org/10.1016/j.sjopt.2014.05.002
17 18 19
Introduction
20
True exfoliation and pseudoexfoliation of the anterior lens capsule are two distinct entities. Their coexistence in the same individual is rare and has only been reported few times internationally and never been documented in Saudi or Arab patient.1–3 In this report we describe a patient with combined true and pseudoexfoliation of the anterior lens capsule in a Saudi patient that was confirmed histopathologically.
21 22 23 24 25 26 27
Case history
28
A 63 year old Saudi Male who is a nomad complaining of gradual decrease in vision of the left eye over 4 years. He had a history of poor vision of the right eye since childhood. There was no history of previous inflammation. His clinical findings on presentation are summarized in Table 1 and illustrated in Fig. 1. The pseudoexfoliation (PXF) materials were observed in the pupillary margin and over the iris surface of the left eye, while the capsular delamination formed outward rolled edges extending from 12:30 o’clock position to 4:00, and
29 30 31 32 33 34 35 36 37 38
then from 4:00 to 8:00 o’clock position. The patient’s pupil dilated poorly and hence we could not comment clinically whether pseudoexfoliative material was present on the anterior lens capsule and were not able to clearly define the extent of capsular delamination beyond the visible 4 mm pupillary area. The patient was put on a fixed combination of Dorzolamide 2.0%/Timolol 0.5% drops and then changed to Brimonidine 0.2%/Timolol 0.5% both of which failed to reduce the IOP below 22 mmHg, plus that the patient was poorly compliant to them, for those reasons he underwent extracapsular cataract extraction and posterior chamber intraocular lens implantation (ECCE + PC IOL) with endoscopic cyclophotocoagulation (ECP) of the left eye. ECCE was chosen over phacoemulsification due to the extensive PXF and delamination of the capsule and the former procedure was thought to be safer in the surgeon’s hand, and as the patient is nomad with a history of poor compliance there was a concern about the safety and efficacy of doing an incisional glaucoma procedure, and ECP was thought to be the safest in his condition as it needs no extra post operative care than the cataract surgery. Intra operatively the pupil was stretched using Kuglen
Received 24 February 2014; received in revised form 10 May 2014; accepted 26 May 2014; available online xxxx. a b c
Q3
Glaucoma Division, King Khaled Eye Specialist Hospital, Riyadh, Saudi Arabia Glaucoma Division, Bin Rushd Ophthalmic Center, Riyadh, Saudi Arabia Wilmer Eye Institute, Johns Hopkins University School of Medicine, Baltimore, USA
⇑ Corresponding author at: Glaucoma Division, King Khaled Eye Specialist Hospital, Riyadh, Saudi Arabia. Tel.: +966 568786232. e-mail address: [email protected] (S. Alodhayb). Peer review under responsibility of Saudi Ophthalmological Society, King Saud University
Access this article online: www.saudiophthaljournal.com Production and hosting by Elsevier www.sciencedirect.com
Q1 Please cite this article in press as: Alodhayb S., Edward D.P. Combined true and pseudoexfoliation in a Saudi patient with co-existing cataract and glaucoma. Saudi J Ophthalmol (2014), http://dx.doi.org/10.1016/j.sjopt.2014.05.002
39 40 41 42 43 44 45 46 47 48 49 50 51 52 53 54 55 56 57 58 59 60
SJOPT 300
No. of Pages 3, Model NS
2
S. Alodhayb, D.P. Edward
Table 1. Summary of pre-operative clinical findings. OD
OS
VA IOP (mm Hg) Cornea A/C Lens
Hand motion 14 Central corneal scarring Deep & quiet Mature cataract, no Pseudoexfoliative (PEX) deposits
A/C angle Fundus B-scan Endothelial cell count
Open grade III, Sampaolesi’s line No view Retina flat 1475
Uncorrected 20/100; pinhole: 20/80 31 Arcus senilis and mild diffuse haze Deep & quiet Dense cataract, dense PEX deposits, and delamination of anterior lens capsule Open grade III, Sampaolesi’s line No view Retina flat 1309
Figure 1. Left eye of patients showing rolled delaminated capsule in the pupillary area (arrowheads) and pseudoexfoliative material at the pupillary border (arrows).
75
Hooks, and the anterior capsulotomy was done with a disposable 30-gauge cystotome using a can opener technique away from the area of the capsular delamination. No difficulties or complications were encountered during this step, the anterior lens capsule was submitted for histopathology. We noted that during ECP an unusually high power of 0.7 Watts was required to elicit a response which was attributed to the presence of PEX material over the ciliary processes. Postoperatively his vision improved to 20/40 with an intraocular pressure of 9–11 mm Hg without medications at the last visit (16 months). The right eye maintained normal IOP throughout the follow up time, but with progressing cataract, which was eventually extracted uneventfully with mild visual improvement due to Amblyopia.
76
Pathology
61 62 63 64 65 66 67 68 69 70 71 72 73 74
82
Pathological examination of the anterior lens capsule was performed and changes described were observed away from the edge of the capsulotomy. The anterior capsule showed thickening and delamination in some areas and in other areas PAS positive fibrillar deposits consistent with pseudoexfoliative material were observed (Fig. 2).
83
Discussion
77 78 79 80 81
84 85 86 87
In this report we describe the patient with unilateral pseudoexfoliation deposits accompanied by delamination of the anterior lens capsule (true exfoliation) in one eye. Clinically the pseudoexfoliation material was visible only at the
pupillary margin but on pathologic examination it was also Q4 noted on the anterior lens capsule. The coexistence of both true exfoliation and pseudoexfoliation is rare and had been reported few times only, once by Küchle et al.,1 in an 81-year-old female with a long history of working with porcelain in an oven. Another case was reported in the Japanese literature in an 83 year old woman without any predisposing factors.2 Delamination of the anterior lens capsule has been reported in patients exposed to prolonged heat or infrared radiation,4–6 intraocular inflammation,7 trauma,8 pseudoexfoliation,9 and as an idiopathic entity.9–11 Our patient being a nomad spent prolonged periods of time in the desert which has high levels of infrared radiation.12 We suggest that such prolonged exposure to infrared radiation in the desert in this Bedouin nomad might have been a risk factor for capsular delamination. The condition however has not been previously reported in Saudi patients. The capsular delamination presented with rolled edges in multiple locations in the pupillary area. Based on the experience during the can opener capsulotomy, we believe that capsular delamination does not interfere with the anterior capsulotomy with a can opener technique. It is possible that the simultaneous presence of both conditions may potentially cause complications during capsulorhexis. Dense
Figure 2. Photomicrograph showing the anterior capsule Top. Showing capsular delamination (PAS; original magnification 20). Bottom: showing iron filing like pseudoexfoliation material on the anterior lens capsule (PAS; original magnification 20).
Q1 Please cite this article in press as: Alodhayb S., Edward D.P. Combined true and pseudoexfoliation in a Saudi patient with co-existing cataract and glaucoma. Saudi J Ophthalmol (2014), http://dx.doi.org/10.1016/j.sjopt.2014.05.002
88 89 90 91 92 93 94 95 96 97 98 99 100 101 102 103 104 105 106 107 108 109 110 111 112
SJOPT 300
Q1 Combined true and pseudoexfoliation in a Saudi patient
113 114 115 116 117 118 119 120 121 122 123 124 125 126 127 128 129
130
131
132
133 134 135
pseudoexfoliation deposits might mask capsular delamination and following initiation of capsulorhexis, multiple layers of anterior capsule might be observed causing difficulty in completion of the capsulorhexis. The pathologic features seen in our patient were quite typical of what has been previously described in patients with capsular delamination9–11 and pseudoexfoliation13 as separate entities. It was interesting to note that pseudoexfoliation deposits were not observed in the areas where capsular delamination was prominent. In summary, this case report highlights the presence of pseudoexfoliation and true exfoliation in an eye of a Saudi Bedouin. We suggest that exposure to high levels of infrared radiation in the desert may play a role in capsular delamination. In addition, possible chronic and extended exposure to fire in such individuals who are directly exposed to wood burning fires may play a role in capsular delamination.
Conflict of interest The authors report no conflicts of interest in this work.
References 1. Küchle M, Iliff WJ, Green WR. Combined true exfoliation and pseudoexfoliation of the anterior lens capsule. Klin Monbl Augenheilkd 1996 Feb;208(2):127–9.
No. of Pages 3, Model NS
3 2. Oharazawa H, Suzuki H, Matsui H, Shiwa T, Takahashi H, Ohara K. Two cases of true exfoliation of the lens capsule after cataract surgery. J Nippon Med Sch 2007;74(1):55–60. 3. Yamamoto Y, Nakakuki T, Nishino K, Yagyu K, Kishi S, Ueno H, et al. Histological and clinical study of eyes with true exfoliation and a double-ring sign on the anterior lens capsule. Can J Ophthalmol 2009 Dec;44(6):657–62. 4. Elschnig A. Detachment of the zonular lamellae in glassblowers. Klin Monatsbl Augenheilkd 1922;69:732–4. 5. Burde RM, Bresnick G, Uhrhammer J. True exfoliation of the lens capsule: an electron microscope study. Arch Ophthalmol 1969;82:651–3. 6. Callahan A, Klien BA. Thermal detachment of the anterior lamella of the anterior lens capsule. Arch Ophthalmol 1958;59:73–80. 7. Elschnig A. Uber die Ablosung der Zonulalamelle. Klin Monatsbl Augenheilkd 1926;76:66–70. 8. Butler TH. Capsular glaucoma. Trans Ophthalmol Soc UK 1938;68:575–89. 9. Brodrick JD, Tate Jr GW. Capsular delamination (true exfoliation) of the lens. Report of a case. Arch Ophthalmol 1979;97:1693–8. 10. Radda TM, Klemen UM. Idiopathische echte Exfoliation. Klin Monatsbl Augenheilkd 1982;181:276–7. 11. Cashwell Jr LF, Holleman IL, Weaver RG, van Rens GH. Idiopathic true exfoliation of the lens capsule. Ophthalmology 1989;96:348–51. 12. Al-Khalaf AK. Results of outgoing longwave radiation and Albedo over Saudi Arabia from earth radiation budget experiment (ERBE) data. Met., Env. & Arid Land Agric. Sci. 2006;17(1):77–99. 13. Kincaid M.C. Pathology of the lens. In: Tasman W, Jaeger E.A. (Eds.), Duane’s Foundations of Clinical Ophthalmology, vol. 3. Lippincott Q5 Williams & Wilkins, Philadelphia, PA (Chapter 12).
Q1 Please cite this article in press as: Alodhayb S., Edward D.P. Combined true and pseudoexfoliation in a Saudi patient with co-existing cataract and glaucoma. Saudi J Ophthalmol (2014), http://dx.doi.org/10.1016/j.sjopt.2014.05.002
136 137 138 139 140 141 142 143 144 145 146 147 148 149 150 151 152 153 154 155 156 157 158 159 160 161 162 163 164 165 166