Bullous keratopathy treated with honey

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L etters to the Editor A pilot experiment using a network camera in ophthalmic teleconsultation Juha Hagman, Pauli Hyytinen and Anja Tuulonen Department of Ophthalmology, University of Oulu, Oulu, Finland doi: 10.1111/j.1600-0420.2004.00266.x Sir, W e carried out a study to test the feasibility of a network camera in a real time teleconsultation in eye care. The Sony SLC-VL10 network camera (Sony Corp., Shinagawa-ku, Tokyo, Japan) became commercially available in Finland in 2002 and was chosen for the study because of the possible cost-effectiveness related to its reasonable price (e1695 including 22% VAT, June 2003). The SLC-VL10 is capable of transmitting live video with a resolution of 720 576 pixels at up to 25 frames per second. To decrease the bandwidth requirements the Sony camera has a built-in compression system. It uses the Wavelet compression algorithm instead of the more common Joint Photo- graphic Experts Group (JPEG) algorithm (Bashshur et al. 2000; Eikelboom et al. 2000; Murdoch et al. 2000; Tuulonen & Ohinmaa 2000). The real time consultation link was established via the university hospital’s local area network (LAN). The images produced by the network camera were viewed side by side with those produced by a piece of videoconferencing equip- ment that is widely used in telemedicine in Finland, the Tandberg 880 (Tandberg Inc., New York, USA; e12 760 includ- ing 22% VAT, June 2003). The video image provided by the network camera can be visualized on any PC at the hos- pital network while the videoconfer- encing system allows for contact between only two sites at any one time. A total of 22 unselected patients from the Oulu University Eye Depart- ment were examined during a 3-day testing period. Of these, 12 were male (55%) and 10 were female (45%). Their ages ranged from 7 years to 82 years (mean 52 SD 19 years). The patients were examined face to face by two residents in a routine outpatient service while the teleconsultant viewed the examinations via a computer screen and a TV monitor, showing the images acquired with the Sony and Tandberg systems, placed side by side. The spe- cialist took notes of the lesions visible on each of the systems and compared the images transmitted by the two pieces of equipment using the principle of the preferential looking test as used in infant eye examination. Thereafter, the consultant also examined the patients face to face and evaluated the lesions that had possibly been missed on the tele-examination. The majority of the clinical findings could be detected through the network. The teleconsultant was able to assess flare and cells in the anterior chamber, cornea guttata, folding of the descemet membrane, cataract, pupillary adhesions and reduced break-up time. Fundus examination appeared to be more difficult via real time teleconsultation, especially when using the Volk lens. However, visi- bility improved when a 3-mirror contact lens was used. The consultant was able to assess the optic nerve head, laser scars on the retina, macular bleeding, macular hole, pigment displacement in the macula, retinal exudates and haemorrhages, vit- reous opacities and retinal drusen through the network. Lesions that were not seen in the anterior segment of the eye via telecon- sultation included very subtle punctate lesions of the corneal epithelium (in two patients), tiny follicles of the conjunc- tiva (in one patient) and in one case cells in the anterior chamber. In add- ition, the consultant was unable to assess the posterior segment of the eye for macular oedema and a tiny vascular anomaly on the retina (one patient). When comparing the Sony SLC-VL10 and the Tandberg 880 videoconferen- cing system, the quality of the Sony video image was preferred in six of 21 cases (29%), and that of the Tandberg image in two of 21 cases (9%). In 13 of 21 cases (62%) the images were rated equal in quality. In one case (5%) the PC malfunctioned and the consultant was unable to see any of the images broadcast by the Sony camera. When the transmission rate decreased due to other electronic traffic in the LAN, the image broke up and became more difficult to analyse. All unneces- sary movements of the patient and the slit-lamp also decreased the diagnostic value of the video images. The use of an independent additional light source improved the image quality. The main problems with real time telemedicine consultations have been lack of sufficient image resolution, network transmission speed and the high costs of equipment. In our small pilot patient series we were able to diag- nose most of the various, unselected ophthalmological lesions via telecon- sultation. The current technology seems to be sufficient for real time tele- ophthalmology consultation, especially when compared to verbal descriptions conveyed by telephone between pri- mary health care settings and eye hospitals. The new network camera offers a much more cost-effective method of real time telemedicine com- pared to the videoconferencing system. Further research with larger numbers of patients and longer distances between the two consulting sites is warranted. ACTA OPHTHALMOLOGICA SCANDINAVICA 2004 311

Transcript of Bullous keratopathy treated with honey

Letters to the Editor

A pilot experiment using a network camera in ophthalmic

teleconsultation

Juha Hagman, Pauli Hyytinen and Anja Tuulonen

Department of Ophthalmology, University of Oulu, Oulu, Finland

doi: 10.1111/j.1600-0420.2004.00266.x

Sir,

W e carried out a study to test thefeasibility of a network camera

in a real time teleconsultation in eyecare. The Sony SLC-VL10 networkcamera (Sony Corp., Shinagawa-ku,Tokyo, Japan) became commerciallyavailable in Finland in 2002 and waschosen for the study because of thepossible cost-effectiveness related to itsreasonable price (e1695 including 22%VAT, June 2003). The SLC-VL10 iscapable of transmitting live video witha resolution of 720� 576 pixels at up to25 frames per second. To decrease thebandwidth requirements the Sony camerahas a built-in compression system. Ituses the Wavelet compression algorithminstead of the more common Joint Photo-graphic Experts Group (JPEG) algorithm(Bashshur et al. 2000; Eikelboom et al.2000; Murdoch et al. 2000; Tuulonen &Ohinmaa 2000).

The real time consultation link wasestablished via the university hospital’slocal area network (LAN). The imagesproduced by the network camera wereviewed side by side with those producedby a piece of videoconferencing equip-ment that is widely used in telemedicinein Finland, the Tandberg 880 (TandbergInc., New York, USA; e12 760 includ-ing 22% VAT, June 2003). The videoimage provided by the network cameracan be visualized on any PC at the hos-pital network while the videoconfer-encingsystemallowsforcontactbetweenonly two sites at any one time.

A total of 22 unselected patientsfrom the Oulu University Eye Depart-ment were examined during a 3-daytesting period. Of these, 12 were male(55%) and 10 were female (45%). Theirages ranged from 7 years to 82 years(mean 52� SD 19 years). The patients

were examined face to face by tworesidents in a routine outpatient servicewhile the teleconsultant viewed theexaminations via a computer screenand a TV monitor, showing the imagesacquired with the Sony and Tandbergsystems, placed side by side. The spe-cialist took notes of the lesions visibleon each of the systems and comparedthe images transmitted by the twopieces of equipment using the principleof the preferential looking test as usedin infant eye examination. Thereafter,the consultant also examined thepatients face to face and evaluated thelesions that had possibly been missedon the tele-examination.

The majority of the clinical findingscould be detected through the network.The teleconsultant was able to assessflare and cells in the anterior chamber,cornea guttata, folding of the descemetmembrane, cataract, pupillary adhesionsand reduced break-up time. Fundusexamination appeared to be more difficultvia real time teleconsultation, especiallywhen using the Volk lens. However, visi-bility improved when a 3-mirror contactlens was used. The consultant was able toassess the optic nerve head, laser scars onthe retina, macular bleeding, macularhole, pigment displacement in the macula,retinal exudates and haemorrhages, vit-reous opacities and retinal drusen throughthe network.

Lesions that were not seen in theanterior segment of the eye via telecon-sultation included very subtle punctatelesions of the corneal epithelium (in twopatients), tiny follicles of the conjunc-tiva (in one patient) and in one casecells in the anterior chamber. In add-ition, the consultant was unable toassess the posterior segment of the eye

for macular oedema and a tiny vascularanomaly on the retina (one patient).

When comparing the Sony SLC-VL10and the Tandberg 880 videoconferen-cing system, the quality of the Sonyvideo image was preferred in six of 21cases (29%), and that of the Tandbergimage in two of 21 cases (9%). In 13 of21 cases (62%) the images were ratedequal in quality. In one case (5%) thePC malfunctioned and the consultantwas unable to see any of the imagesbroadcast by the Sony camera.

When the transmission rate decreaseddue to other electronic traffic in theLAN, the image broke up and becamemore difficult to analyse. All unneces-sary movements of the patient and theslit-lamp also decreased the diagnosticvalue of the video images. The use ofan independent additional light sourceimproved the image quality.

The main problems with real timetelemedicine consultations have beenlack of sufficient image resolution,network transmission speed and thehigh costs of equipment. In our smallpilot patient series we were able to diag-nose most of the various, unselectedophthalmological lesions via telecon-sultation. The current technologyseems to be sufficient for real time tele-ophthalmology consultation, especiallywhen compared to verbal descriptionsconveyed by telephone between pri-mary health care settings and eyehospitals. The new network cameraoffers a much more cost-effectivemethod of real time telemedicine com-pared to the videoconferencing system.Further research with larger numbersof patients and longer distancesbetween the two consulting sites iswarranted.

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AcknowledgementThe study described in this letter waspresented in part at the Meeting of theFirst Science Day of the Medical Faculty,University of Oulu, February 18th, 2003.

ReferencesBashshur RL, Reardon TG & Shannon GW

(2000): Telemedicine: a new health care

delivery system. Ann Rev Public Health 21:

613–637.

Eikelboom RH, Yogesan K, Barry CJ,

Constable IJ, Tay-Kearney M, Jitskaia L &

House PH (2000): Methods and limits of

digital image compression of retinal images

for telemedicine. Invest Ophthalmol Visual

Sci 41: 1916–1924.

Murdoch I, Bainbridge J, Taylor P, Smith L,

Burns J & Rendall J (2000): Postoperative

evaluation of patients following ophthalmic

surgery. J Telemed Telecare 6: 84–86.

Tuulonen A & Ohinmaa A (2000): Telemedi-

cine, essential for maintaining centres of

excellence? Ophthalmol Clin North Am 13:

151–162.

Correspondence:

Professor Anja Tuulonen MD, PhD

Department of Ophthalmology

University of Oulu

Box 5000

90014 University of Oulu

Oulu

Finland

Tel:þ 358 8 315 32 99

Fax:þ 358 8 330 122

Email: [email protected]

Bullous keratopathy treated with honey

Ahmad M. Mansour, Wadih Zein, Randa Haddad and Johnny Khoury

Department of Ophthalmology, American University of Beirut, Beirut, Lebanon

doi: 10.1111/j.1600-0420.2004.00258.x

Sir,

H oney was used by ancientEgyptians 5000 years ago to

treat inflammations and burns of thecornea and conjunctiva (Nunn 1996).This practice continued through theGreco-Roman period and the MiddleAges, right up to the modern era(Nunn 1996). Honey is mentioned as aremedy in the Old Testament (I Samuel14 : 27), the Talmud (Shabbath 77b�78a)and the Koran (Nahl 96). It is describedin the Talmud as having a propitiouseffect on the eyes: ‘Honey enlightensthe eyes of man’ (Yoma 83b). Thisprompted a prospective study of topicalhoney as a hyperosmotic and healingagent in the medical therapy of bullouskeratopathy.

We carried out a prospective studyof 24 consecutive patients with bullouskeratopathy, who were not immediatesurgical candidates and who attended aprivate clinic from January 2000 to Sep-tember 2003. One drop of honey wasapplied to the cornea after informedconsent had been obtained. Patientswere instructed to use topical honey3–4 times daily.

All corneas had clearing of epithelialoedema with collapse of corneal bullae.Median Snellen best corrected visualacuity (VA) improved from fingercounting at 50 cm to finger counting at

2 metres (p¼ 0.001) (Fig. 1). After top-ical application of honey, anterior seg-ment details could be seen and fundusevaluation was possible in two cases.Patients experienced stinging but withrepeated usage the discomfort becameminimal. Visual acuity improvementand corneal clearing lasted for around1 hour after topical honey.

Bullous keratopathy is a major com-plication of cataract surgery. In thepast, penetrating keratoplasty was con-sidered the most effective therapy forthe symptomatic stage of the disease.Other surgical options have included

conjunctival flaps, enucleation (reservedfor blind, painful eyes), and, morerecently, deep phototherapeutic kera-tectomy and amniotic membrane trans-plantation. Medical therapy of bullouskeratopathy using hypertonic saline(NaCl 5%) has been of marginal bene-fit due to its relatively weak osmoticeffect. We have described a topical medi-cation that can collapse corneal bullae(theosmoticpressureofhoney isextreme-ly high, exceeding 2000 milliosmols/kg)(Mansour 2002), prevent bacterialsuperinfection (honey does not supportthe vegetative life stage of any bacterialspecies) (Cooper et al. 2001), andenhance the healing of corneal defects(Mansour; unpublished data), therebydisplaying properties similar to itsunique healing facilities in recalcitrantulcers or burns (Bergman et al. 1983;Cooper et al. 2001; Topham 2002). Top-ical honey improved VA in eyes withepithelial corneal oedema (Mansour2002) and in eyes with bullous kerato-pathy (Fig. 1), and allowed anterior andposterior segment visualization. Givenits advantages in terms of its antimicro-bial, antioxidant, anti-inflammatoryand hygroscopic properties, furtherevaluation of the longterm effects ofhoney on bullous keratopathy iswarranted.

0

0.5

1

1.5

2

2.5

3

3.5

4

4.5

0 0.5 1 1.5 2 2.5 3 3.5 4 4.5VA Before

VA

Afte

r

Fig. 1. Scatter plot of LogMAR visual

acuity before and after topical application of

honey.

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ReferencesBergman A, Yanai J, Weiss J et al. (1983):

Acceleration of wound healing by topical

application of honey: an animal model. Am

J Surg 14: 374–376.

Cooper RA, Molan PC, Krishnamoorthy L

et al. (2001): Manuka honey used to heal a

recalcitrant surgical wound. Eur J Clin

Microbiol Infect Dis 20: 758–759.

Mansour AM (2002): Epithelial corneal

oedema treated with honey. Clin Experiment

Ophthalmol 30: 149–150.

Nunn JF (1996): Ancient Egyptian Medicine.

London: University of Oklahoma Press

198–202.

Topham J (2002): Why do some cavity wounds

treated with honey or sugar paste heal with-

out scarring? J Wound Care 11: 53–55.

Correspondence:

Ahmad Mansour MD

American University of Beirut

POB 113-6044

Beirut

Lebanon

Tel:þ 96 11 374 625

Fax:þ 96 11 744 464

Email: [email protected]

Hypermetropic refractive change after hyperbaric oxygen

therapy

Hans C. Fledelius1 and Erik Jansen2

1Department of Ophthalmology, Rigshospitalet, Copenhagen, Denmark2Unit of Hyperbaric Medicine, Rigshospitalet, Copenhagen, Denmark

doi: 10.1046/j.1600-0420.2004.00239.x

Sir,

S hort-term refractive fluctuationsoccurring in adults on a non-

traumatic basis are generally towardsmyopia. The myopic shifts have beenrelated to various causes such as dia-betes, infectious disease and certainmedications (Curtin 1985). Hyperbaricoxygen (HBO) therapy may alsocause a relative myopization. A dose-relationship to cumulated number ofhours under therapy has been suggested(Anderson & Farmer 1978). This correl-ation was not supported by a previousstudy from our clinic (Fledelius et al.2002), although when refractivechanges occurred in our material, theyinvariably tended towards less plus/more minus. Further, our follow-upshowed that all cases returned totheir original refractive level within1–2 months and no cases of refractiveovershooting were observed.

Recently, we followed a femalepatient who showed a hypermetropicchange in the wake of HBO therapy.As we have been unable to find anysimilar reports in the literature (Clark2003), we find it of interest to report onthe case.

A 41-year-old woman underwentradiotherapy for tonsillar carcinomain 2001. In June 2003 she received 30HBO sessions (each of 90 min at2.4 ATA) for osteonecrosis of the man-

dibular bone. As an adult her refrac-tion had been stable; she had habitualvisual acuity (VA) levels of 1.0 in theright eye (RE) and 0.5 in the left eye(LE), carried glasses of �0.5 D sph.(RE) and þ1.0 D sph. (LE), and hadno need of glasses for near vision.Before her treatment the subject hadbeen informed that she might experi-ence a slightly closer reading distancethan usual towards the end of the HBOseries, and this did in fact occur. How-ever, 10 days after the HBO series shewas suddenly unable to read text. Wesaw her on day 17. Her VA was still 1.0(RE) and 0.5 (LE) as before, but withþ1.5 D sph. and þ3.0 D sph., respect-ively, as best correction for distance.With this correction she was also ableto read text. Laboratory examinationsexcluded diabetes. As a temporarymeasure, the subject used ready-madesupermarket spectacles of þ1.5 D. Onday 31 refractive values were þ1.5 Dand þ2.25 D, respectively, undermydriasis. By day 88, her refractionand visual abilities had returned tonormal. Subjectively, this change hadbeen noticed about day 72. Slit-lampexamination and ophthalmosocopyshowed clear media and normal fundithroughout.

Keratometry values on the threeseparate examination days after termi-

nating the HBO series all averaged7.65–7.66 mm in corneal curvatureradius. Likewise, there was no changein the axial length values as determinedby IOL-Master equipment: 23.19 mmin the right eye, at its most hyper-metropic, and 23.21 mm when as usual.Corresponding left eye values were22.56 mm on both occasions.

The patient therefore experienced aperiod of 8–9 weeks with a 2 D hyper-metropic shift, quite parallel in the twoeyes despite the basic anisometropia,which clearly began 10 days after termin-ation of an HBO treatment series. Onceshe had acquired full correction fordistance, her ability for accommodationappeared unaffected.

All evidence from previous studies ofrefractive change during HBO therapyseems to indicate that changes in therefractive indices of the lens compart-ments are responsible for the relativemyopization often observed towardsthe end of longer treatment series(Palmquist et al. 1984; Fledelius et al.2002). Because there is a documentedlack of evidence of changes in cornealcurvature or axial length, we have toassume similar underlying mechanismsregarding the exceptional transitoryhypermetropic change reported here.

In previous studies of transitoryrefractive change in diabetes patients,

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some subjects experienced a short-termmyopic shift prior to a more longstand-ing swing towards hypermetropia(Riordan Eva et al. 1982; Fledelius1987). Typically, the initial changeappeared during acute dysregulationand was hypothesized to relate toosmotic changes in the lens associatedwith alternative pathways of carbo-hydrate metabolism. The subsequenthypermetropic phase, however, lastedmuch longer than the period whenextra insulin was required to returnthe patient to normal laboratoryvalues.

The reversible course of the refract-ive change under and after HBOtreatment in the present subjectdescribed a similar time curve. How-ever, we have no indication of anypathological biochemistry of the nor-mal lens, except that which might be

related to the oxidative stress possiblyassociated with hyperbaric oxygentreatment.

ReferencesAnderson B & Farmer JC (1978): Hyper-

oxic myopia. Trans Am Ophthalmol Soc

76: 116�124.

Clark J (2003): Side effects and complications.

In: Feldmeijer JJ (ed.) Hyperbaric Oxygen

2003, Medications and Results. Hyperbaric

Oxygen Committee Report. Kensington:

Undersea and Hyperbaric Medical Society.

137�141.

Curtin B (1985): Pseudomyopia. In: The Myo-

pias. Philadelphia: Harper & Row. 455�457.

Fledelius HC (1987): Refractive change in dia-

betes mellitus, around onset or when poorly

controlled. Acta Ophthalmol Scand 65:

53–57.

Fledelius HC, Jansen EC & Thorn J (2002):

Refractive change during hyperbaric

oxygen therapy. A clinical trial including

ultrasound oculometry. Acta Ophthalmol

Scand 80: 188�190.

Palmquist BM & Barr PO (1984): Nuclear

cataract and myopia during hyper-

baric oxygen therapy. Br J Ophthalmol 68:

113�11.

Riordan Eva P, Pascoe PT & Vaughan DG

(1982): Refractive change in hyperglycaemia:

hyperopia, not myopia. Br J Ophthalmol 66:

500�505.

Correspondence:

Dr Hans C. Fledelius

Rigshospitalet

Eye Dept E 2061

9 Blegdamsvej

DK-2100 Copenhagen

Denmark

Tel:þ 45 35 45 33 10

Fax:þ 45 35 45 22 98

Email: [email protected]

Displacement of a laser in situ keratomileusis flap during

retinal detachment surgery

Jost B. Jonas

Department of Ophthalmology, Faculty of Clinical Medicine Mannheim, University of

Heidelberg, Mannheim, Germany

doi: 10.1046/j.1600-0420.2003.00168.x

Sir,

L aser in situ keratomileusis(LASIK) has become the corneal

refractive surgery most often per-formed to correct mild to moderatemyopia. It includes preparation of asuperficial corneal flap, under whichthe corneal stroma is ablated using theexcimer laser. At the end of the proce-dure, the flap is repositioned. Althoughtraumatic dislocation of the flap hasbeen reported (Melki et al. 2000;Recep et al. 2000; Geggel & Coday2001), an iatrogenic subluxation of theflap has only rarely been encountered.It was therefore the purpose of the pre-sent study to report the late-onsetintraoperative iatrogenic dislocation ofa laser in situ keratomileusis flap.

A 37-year-old patient noticed adefect of his lower nasal visual fieldextending close to the centre of visionin his right eye. Four years earlier, he

had undergone LASIK for treatment ofmoderate myopia of � 6 dioptres. Aftercorneal refractive surgery, uncorrectedvision had increased to 20/20. At thetime of hospital admission, the fundusshowed a rhegmatogenous retinaldetachment in the temporal upperquadrant of the fundus extending intothe foveal region. Visual acuity (VA)measured 0.25 without glasses. Ascleral buckling procedure was per-formed under general anaesthesia.When the ophthalmoscopic visibilityof the fundus structures decreased dueto an opacification of the cornealepithelium, the surgeon, who wasaware of the previous LASIK proce-dure, carefully performed a cornealabrasion using a blunt hockey knife.The hockey knife was used mostly in acentrifugal direction starting close tothe centre of the cornea and extending

to the limbal region. A few strokes ofthe hockey knife went in a centripetaldirection, returning from the limbalzone to the corneal centre. During oneof these actions, the corneal flapbecame detached. The detached cornealflap was still connected to the cornea byits hinge; it was carefully rinsed, theexposed corneal stroma was cleansedof any cell debris, the detached cornealflap was repositioned and surgery con-tinued. At the first postoperative day,the corneal flap was in loco. Fine stri-ations of the flap could be detected in itsperiphery. In the optical centre, the cor-nea was clear. The retina was reat-tached. Four months after surgery,VA measured 0.60 without spectacles.Glasses did not improve vision. Finestriations of the flap were still presentin the peripheral region but did nottouch the optical centre.

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Due to delayed wound healing, cor-neal flap dehiscence may occur monthsor years after uneventful LASIK. Asreported in previous studies on trau-matic flap subluxation and singlereports on secondary intraoperativeflap dislocations, a dehiscence of theflap is a serious but manageable com-plication of LASIK (Melki et al. 2000;Recep et al. 2000; Geggel & Coday2001; Lombardo & Katz 2001; Sakuraiet al. 2002). If it is detected early, thevisual prognosis may be as good as thatin eyes without flap subluxation. Aslaser in situ keratomileusis increases inpopularity, especially in myopicpatients, extreme care should be takento avoid corneal trauma in retinaldetachment surgery, even when the sur-gery is taking place years after theinitial corneal refractive surgery (Ruiz-Moreno et al. 1999; Arevalo et al.2001). Furthermore, patients should

be informed of the potential fortraumatic or iatrogenic flap dehiscencefollowing laser in situ keratomileusis.

ReferencesArevalo JF, Ramirez E, Suarez E et al. (2001):

Rhegmatogenous retinal detachment in

myopic eyes after laser in situ keratomileu-

sis. Frequency, characteristics and mechan-

ism. J Cataract Refract Surg 27: 674–680.

Geggel HS & Coday MP (2001): Late-onset

traumatic laser in situ keratomileusis

(LASIK) flap dehiscence. Am J Ophthalmol

131: 505–506.

Lombardo AJ & Katz HR (2001): Late partial

dislocation of a laser in situ keratomileusis

flap. J Cataract Refract Surg 27: 1108–1110.

Melki SA, Talamo JH, Demetriades AM,

Jabbur NS, Essepian JP, O’Brien TP &

Azar DT (2000): Late traumatic dislocation

of laser in situ keratomileusis corneal flaps.

Ophthalmology 107: 2136–2139.

Recep OF, Cagil N & Hasiripi H (2000): Out-

come of flap subluxation after laser in situ

keratomileusis: results of 6 month follow-up.

J Cataract Refract Surg 26: 1158–1162.

Ruiz-Moreno JM, Perez-Santonja JJ & Alio JL

(1999): Retinal detachment in myopic eyes

after laser in situ keratomileusis. Am J

Ophthalmol 128: 588–594.

Sakurai E, Okuda M, Nozaki M & Ogura Y

(2002): Late-onset laser in situ keratomileu-

sis (LASIK) flap dehiscence during retinal

detachment surgery. Am J Ophthalmol 134:

265–266.

Correspondence:

Dr Jost B. Jonas

Universitats-Augenklinik

Theodor-Kutzer-Ufer 1–3

68167 Mannheim

Germany

Tel:þ 49 621 383 2652

Fax:þ 49 621 383 3803

Email:[email protected]

Efficacy and safety of limbal anaesthesia for clear cornea

phacoemulsification

Carlo Cagini, Giovan Battista Sbordone, Angela Luisa Ricci and Paola Menduno

Department of Ophthalmology, University of Perugia, Perugia, Italy

doi: 10.1111/j.1600-0420.2004.00259.x

Sir,

W e conducted a pilot study on 78consecutive eyes of 68 patients

(30 male, 38 female; mean age 69.9�12.3 years, range 54–89 years), whowere undergoing elective cataract sur-gery and intraocular lens (IOL) implant-ation, to verify the effectiveness andsafety of a new anaesthesia technique.This technique consists of applying acellulose ophthalmic sponge soakedin preservative-free lidocaine hydro-chloride 4% to the temporal perilimbalarea for 45 seconds. We have named thisprocedure limbal anaesthesia (LA).

Patients were excluded if they had hadprevious ocular surgery, inflammationor injury, or demonstrated barriersto co-operation and communicationduring surgery (dementia, hearingimpairment, etc.). All patients receivedidentical preoperative topical prepara-tion with tropicamide 0.5%, cyclopento-

late 1%, cyprofloxacine 0.3% anddiclofenac drops at 10-minute intervalsfour times before surgery. None of thepatients received systemic sedatives. Onedrop of benoxinate 0.4% and povidone-iodine 5% was instilled outside theoperating room immediately before thepatient was brought in for surgery. Skinpreparation was then performed withpovidone-iodine 10%. The patient wasdraped and the lid speculum wasinserted. Limbal anaesthesia was admi-nistered and the lidocaine and povidone-iodine 5% were then rinsed from theconjunctiva. No adjunctive anaestheticdrops were given. In order to minimizebias, one surgeon (CC) performed all thesurgical procedures. Surgery was identi-cal in all eyes and comprised: a three-plane, 3.2-mm, temporal clear cornealtunnel; two side-port incisions; injectionof 0.2–0.4 cc preservative-free lidocaine

1% in the anterior chamber; continuouscurvilinear capsulorhexis; hydrodis-section with balanced salt solution(BSS1, Allergan Inc., Irvine, CA,USA), bimanual phacoemulsifications,and bimanual irrigation/aspiration ofthe residual lens cortex. A foldable sili-cone IOL (Allergan SI40NB1, AllerganInc., Irvine, CA, USA) was implantedwith the injector in the capsular bag. Asingle stitch of 10–0 nylon (AlconLaboratories Inc., Forth Worth, Texas,USA) was placed and the visco-elastic removed. During all surgicalprocedures, we used high molecularweight sodium hyaluronate viscoelastic(Provisc1, Alcon Laboratories Inc.,Forth Worth, Texas, USA). Mean effec-tive phacoemulsification time was0.51� 0.03 min (range 0.15–2.18 min).Between 30 and 60 min after surgery, ina postoperative area, the patients were

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asked by a nurse to grade the pain feltduring the operation using a scale of 0–5,where 0¼no pain, 1¼minimal discom-fort, 2¼ low pain, 3¼moderate pain,4¼ intense pain and 5¼ severe pain. Ofthe 78 surgical procedures carried out,68 (87.2%) were described by patients aspain-free. Ten patients reported pain. Infour cases (5.1%) the pain score was 1(minimal discomfort) and in six cases(7.7%) the pain score was 2 (low pain).Postoperative evaluation was performedon days 1, 7, 20, 60 and 90. In this seriesof patients, we found no intraoperativeor postoperative surgical complications,and no epithelial defects at the end ofsurgery or at postoperative follow-up.

There is currently a trend towardsthe increased use of non-injectionforms of ocular anaesthesia for catar-act surgery. In fact, in a 1995 surveyonly 5% of responding members of theAmerican Society of Cataract andRefractive Surgery reported the rou-tine use of topical anaesthesia for cat-aract surgery, as compared to 45% in1999 (Leaming 2000). Because it iscommonly believed that topical anaes-thetics have a limited ability to pene-trate ocular tissues and reach painfibres, multiple administration (3–5times) is required to achieve analgesiaduring routine cataract surgery. Oneof the results of insufficient adminis-tration of anaesthesia is intraoperativepain and reduced safety during sur-gery. The topical administration ofanaesthesia can nevertheless have atoxic effect on the corneal epithelium,and keratopathy can make surgery

more difficult, cause discomfort dur-ing the early postoperative period,reduce lacrimation and, on rare occa-sions, lead to serious keratopathy(Liu et al. 1993). Alternative techni-ques of surface anaesthesia have beenproposed to avoid multiple adminis-tration of anaesthetics and thus anypossible toxic effects on the cornea(Koch 1999; Bloomberg & Pellican1995; Lanzetta et al. 2000).

Our goal in conducting this pilotstudy was to establish whether LAcould be used as an alternative to topicalanaesthesia in cataract surgery. In ourstudy, LA achieved a high level ofanalgesia, sufficient for performingcataract surgery safely, with patientsreporting no discomfort or pain in87.2% of procedures. This percentageis quite high and is comparable to theanalgesia values obtained with topicalanaesthesia (Patel et al. 1996). More-over, in our series we did not findany intraoperative complications, nordid we observe any increased surgicaldifficulties due to intraoperative painor toxic effects on the corneal epithe-lium. In our experience, LA simplifiesthe preoperative preparation of thepatient, does not require the surgeon tomodify his/her normal modus operandi,and the time needed to administer theanaesthesia can be used to accustom thepatient to the light of the microscopeand explain the various phases of theoperation. Consequently, we considerLA to be a viable alternative anaesthetictechnique for use in planned clear corneaphacoemulsification.

ReferencesBloomberg LB & Pellican KJ (1995): Topical

anaesthesia using the Bloomberg Super-

Numb Anaesthetic Ring. J Cataract Refract

Surg 21: 16–20.

Koch PS (1999): Efficacy of lidocaine 2%

jelly as a topical agent in cataract surgery.

J Cataract Refract Surg 25: 632–634.

Lanzetta P, Virgili G, Crovato S, Bandello F &

Menchini U (2000): Perilimbal topical

anaesthesia for clear corneal phacoemul-

sification. J Cataract Refract Surg 26:

1642–1646.

Leaming DV (2000): Practice styles and

preferences of ASCRS members: 1999

survey. J Cataract Refract Surg 26: 913–921.

Liu JC, Steinemann TL & McDonald MB

(1993): Topical bupivacaine and proparacaine:

a comparison of toxicity, onset of action and

duration of action. Cornea 12: 228–232.

Patel BCK, Burns TA, Crandal A, Shomaker

ST, Pace NL, van Eerd A & Clinh T (1996):

A comparison of topical and retrobulbar

anesthesia for cataract surgery. Ophthal-

mology 103: 1196–1203.

Correspondence:

Carlo Cagini MD

Dipartimento di Specialita Medico-Chirurgiche

Sezione di Oculistica

Policlinico Monteluce

Via Brunamonti 51

06100 Perugia

Italy

Tel:þ 39 075 57 20 357

Fax:þ 39 075 57 83 951

Email: [email protected]

Nd:YAG laser membranotomy for premacular

haemorrhage

Jørgen Krohn and Christian Bjune

Department of Ophthalmology, University of Bergen, Bergen, Norway

doi: 10.1111/j.1600-0420.2004.00244.x

Sir,

P remacular haemorrhages mayoccur in a variety of disorders,

including diabetic retinopathy, retinal

vein occlusion, valsalva retinopathy,retinal artery macroaneurysm andocular trauma. In these conditions the

blood is entrapped in the retrohyaloidspace or beneath the internal limitingmembrane, leading to acute and

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A

B

C

Fig. 1. Patient 1. (A) Right fundus at presentation, showing a

premacular haemorrhage with a glistening light reflex. (B) A

photographic montage reveals a stream of blood flowing inferiorly

into the vitreous cavity immediately after successful Nd:YAG laser

membranotomy. (C) Two years after treatment, the fundus is normal

and VA is 6/4.

A

B

C

Fig. 2. Patient 2. (A) Left fundus at presentation, showing a large

premacular haemorrhage. (B) Shortly after Nd:YAG laser

membranotomy, the premacular haemorrhage has drained into the

vitreous. (C) Two months after treatment, the premacular area has

cleared and VA is 6/4. A circinate line in the posterior pole demarcates the

extent of an empty retrohyaloid space (arrows).

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severe visual loss. Spontaneousresorbtion of the blood usually takesseveral months, and may occasionallyinduce epiretinal membrane formationor tractional macular detachment(Cleary et al. 1975; O’Hanley & Canny1985). The present report describes thedrainage of premacular haemorrhagesinto the inferior vitreous cavity throughNd:YAG laser membranotomies.

Patient 1 (a 43-year-old man) andPatient 2 (a 26-year-old man) noticeda sudden loss of vision in one eye.Visual acuity (VA) was reduced to fin-ger counting and hand movements,respectively, due to large premacularhaemorrhages (Figs 1A and 2A). ANidek ophthalmic Nd:YAG laser YC-1400 (Nidek Co. Ltd, Gamagori,Japan) was used to make membrano-tomies at the inferior margin of theanterior surface of the haematomas.In Patient 1 the photodisruption wasperformed 10 days after the initialsymptoms, with a single Nd:YAGlaser pulse of 5.0 mJ through a 25 mmPeyman wide-field YAG laser lens(Ocular Instruments Inc., Washington,District of Columbia, USA) (Fig. 1B).Patient 2 was treated 1 day after hisvisual loss, with a Nd:YAG laser pulseof 6.0 mJ through a Volk Area Centra-lis lens (Volk Optical Inc., Mentor, OH,USA) (Fig. 2B). In both patients thepremacular area cleared completelywithin 3 days, and VA improved to 6/18 and 6/6, respectively. No retinal orsystemic pathological condition couldbe identified during follow-up (Figs 1Cand 2C).

Patient 3 (a 52-year-old man) pre-sented with VA of finger counting inhis right eye. Fundoscopic examinationrevealed a non-ischaemic central veinocclusion with multiple intraretinalhaemorrhages, mild dilatation of theretinal veins and two preretinal haem-orrhages, one located in the maculararea and one located superiorly to theoptic disc (Fig. 3A). Eleven days later,Nd:YAG laser membranotomy wasperformed through a 25 mm Peymanwide-field YAG laser lens (OcularInstruments Inc.), with four pulses of3.5 mJ at the inferior margin of the pre-macular haemorrhage, leading to arapid dispersion of blood into the vit-reous. Visual acuity improved to 6/9after 3 days and to 6/4 after 3 months(Fig. 3B).

Several authors have shown thebeneficial effect of Nd:YAG laser

A

B

C

Fig. 3. Patient 3. (A) Right fundus at presentation, showing two preretinal haemorrhages, one in

the macular area and one superior to the optic disc. Note the multiple intraretinal haemorrhages

and the mild dilatation of the retinal veins. (B) Three months after Nd:YAG laser membranotomy,

the premacular haemorrhage has completely cleared and VA is 6/4. (C) Six months after the initial

symptoms, the preretinal haemorrhage superior to the optic disc has evolved into depositions of

altered blood.

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disruption of the posterior hyaloid andthe internal limiting membrane to allowdrainage of the haemorrhages into thelower vitreous cavity (Faulborn 1988;Ulbig et al. 1998; Rennie et al. 2001).Although macular hole and retinaldetachment have been described aftertreatment (Ulbig et al. 1998), complica-tions seem to be very rare. The presentcase series confirms that Nd:YAGlaser membranotomy is a safe andeffective procedure leading to a rapidrestoration of visual function. The posi-tive effect of preretinal blood drainage iswell illustrated in Patient 3, who had twouniform haemorrhages in his right eye.The premacular haemorrhage disap-peared completely within a few days ofthe membranotomy. On the other hand,the untreated preretinal haemorrhage

superior to the optic disc led to a long-lasting deposition of altered blood(Fig. 3C).

ReferencesCleary PE, Kohner EM, Hamilton AM &

Bird AC (1975): Retinal macroaneurysms.

Br J Ophthalmol 59: 355–361.

Faulborn J (1988): Behandlung einer diabe-

tischen pramacularen Blutung mit dem

Q-switched Neodym:YAG laser. Spektrum

Augenheilkd 2: 33–35.

O’Hanley GP & Canny CLB (1985): Diabetic

dense premacular haemorrhage. A possible

indication for prompt vitrectomy. Ophthal-

mology 92: 507–511.

Rennie CA, Newman DK, Snead MR &

Flanagan DW (2001): Nd:YAG laser treat-

ment for premacular subhyaloid haemor-

rhage. Eye 15: 519�524.

Ulbig MW, Mangouritsas G, Rothbacher HH:

Hamilton AMP & McHugh JD (1998);

Longterm results after drainage of pre-

macular subhyaloid haemorrhage into the

vitreous with a pulsed Nd:YAG laser. Arch

Ophthalmol 116: 1465–1469.

Correspondence:

Jørgen Krohn

Department of Ophthalmology

University of Bergen

Haukeland University Hospital

N-5021 Bergen

Norway

Tel:þ 47 55 97 41 00

Fax:þ 47 55 97 41 43

Email: [email protected]

Recurrent retinal haemorrhages after glaucoma surgery

Kyung-Chul Yoon,1 Man-Seong Seo,1 Yeoung-Geol Park1 and Kun-Jin Yang2

1Department of Ophthalmology, Chonnam National University Medical School,

Gwang-Ju, South Korea2Best Eye Clinic, Gwang-Ju, South Korea

doi: 10.1111/j.1395-3907.2004.00233.x

Sir,

R etinal haemorrhage followingglaucoma filtering surgery is a

rare condition, which was firstdescribed by Fechtner et al. (1992) as

ocular decompression retinopathy.Such haemorrhages at the posteriorpole or peripheral retina are diffuseand, when first observed, many have

white centres. Several reports haveconcerned this complication, but nocase of recurrence has been reported.Here we report a case of recurrent

A B

Fig. 1. Fundus photography. (A) Day 1 after trabeculectomy with mitomycin C. Multiple punctate haemorrhages are scattered throughout the

peripheral retina. (B) Day 1 after Amhed valve implantation. Retinal haemorrhages recur at the peripheral retina.

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diffuse retinal haemorrhages afterglaucoma surgery.

A 17-year-old man was admittedcomplaining of decreased left eye visionand a headache of 5 years duration. Atthe initial examination, corrected visualacuity (VA) was 20/20 in the right eyeand 20/160 in the left. Intraocular pres-sure (IOP) in the right and left eyeswere 18 mmHg and 43 mmHg, respect-ively, and cup : disc ratios were 0.4 and0.9, respectively. Visual field examina-tion showed inferior nasal step andcentral scotoma in the left eye. Despitefull medication, IOP remained uncon-trolled, and thus trabeculectomy withmitomycin C (40 mg/ml, applied for1 min) was performed in the left eye.This was repeated 1 year later.

Three years later, a third trabeculect-omy with mitomycin C was performeddue to uncontrolled IOP (> 40 mmHg).On the first postoperative day, IOP was4 mmHg and multiple scattered punc-tate haemorrhages were detectedthroughout the peripheral retina(Fig. 1A). On the 16th postoperativeday, fluorescein angiography showedmultiple scattered blocked hypofluor-escences and blurrings of the peripheralretinal vessels (Fig. 2). Three monthspostoperatively, the retinal haemor-rhages were found to have resolvedwithout complication.

Six months later, the subject’s left eyeIOP was >36 mmHg, despite the use ofthree or more antiglaucoma medica-tions. Amhed valve implantation wasperformed in the left eye. On the firstpostoperative day, IOP was 10 mmHg

and multiple punctate haemorrhageswere again observed at the peripheralretina (Fig. 1B), although they resolvedspontaneously 3 months postoperatively.

Fechtner et al. (1992) proposedmechanisms for ocular decompressionretinopathy based on two hypotheses.Their first hypothesis suggested thatacute lowering of IOP may reduceblood flow resistance, and the resultingblood flow increase causes multiplefocal endothelial leaks. Defects in theautoregulation of retinal blood flowmay contribute to haemorrhage devel-opment. Their second hypothesisviewed the condition as a variation ofcentral retinal vein occlusion.

In our case, the teenage patient had ahistory of several trabeculectomies withmitomycin C. In addition, fluoresceinangiography showed vascular injury atthe peripheral retina. In young patientsthe sclera is soft, and there is a likeli-hood that antimetabolite is absorbedby the ciliary body or vitreous andcan cause retinal vascular damage.Therefore, we propose that retinalhaemorrhages following glaucoma sur-gery may not be caused by an acutedecrease in IOP alone, but rather thatthe multiple use of mitomycin C mayhave contributed to the condition.

The prognosis is benign, and visualresults are usually unaffected after hae-morrhage resolution (Fechtner et al.1992; Danias et al. 2000; Karadimaset al. 2002). However, in the case offoveal involvement, VA may be affectedpermanently (Dudley et al. 1996). In ourcase, the retinal haemorrhages resolved

without complication, but we suspectthat longterm follow-up results may fea-ture retinal vasculitis.

This case demonstrates that retinalhaemorrhages can recur after glaucomasurgery. Moreover, we believe that theuse of mitomycin C in filtration surgeryin young subjects requires carefulconsideration.

ReferencesDanias J, Rosenbaum J & Podos SM (2000):

Diffuse retinal haemorrhages (ocular decom-

pression syndrome) after trabulectomy with

mitomycin C for neovascular glaucoma.

Acta Ophthalmol Scand 78: 468–469.

Dudley DF, Leen MM, Kinyoun JL & Mills RP

(1996): Retinal haemorrhage associated with

ocular decompression after glaucoma sur-

gery. Ophthalmic Surg Lasers 27: 146–150.

Fechtner RD, Minckler D, Weinreb RN,

Frangei G & Jampol LM (1992): Complication

of glaucoma surgery: ocular decompression

retinopathy. Arch Ophthalmol 110: 965–968.

Karadimas P, Papastathopoulos KI & Bouzas

EA (2002): Decompression retinopathy fol-

lowing filtration surgery. Ophthalmic Surg

Lasers 33: 175–176.

Correspondence:

Kyung-Chul Yoon MD

Department of Ophthalmology

Chonnam National University Hospital

8 Hak Dong, Dong Gu

Gwang Ju 501-757

South Korea

Tel:þ 82 62 220 6740

Fax:þ 82 62 227 1642

Email: [email protected]

A B

Fig. 2. Fluorescein angiography 16 days after trabeculectomy with mitomycin C. (A) Multiple scattered blocked hypofluorescences are observed. (B)

Blurrings of peripheral retinal vessels (arrows) suggest vasculitis.

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Uncommon presentation of asymmetrical retinopathy in

diabetes type 1

Ansu Basu,1 Helen Palmer,2 Robert E. G. Ryder1 and Kenneth G. Taylor1

1Department of Diabetes, Endocrinology and Metabolism, City Hospital, Birmingham, UK2Department of Ophthalmology, Birmingham and Midland Eye Centre, Birmingham, UK

doi: 10.1111/j.1600-0420.2004.00267.x

Sir,

D iabetic retinopathy is oftenbilateral and symmetrical. Retinal

hypoperfusion, and incomplete retinalvein occlusion can also lead to changessimilar to and sometimes indistinguish-able from diabetic retinopathy. In thelatter situation, the findings result fromgeneralized retinal ischaemia as a stimu-lus for angiogenesis. In the case describedwe highlight the phenomenon of ‘retinalsparing’, where despite hypoperfusion,the ischaemic changes did not develop.

A 36-year-old woman with Down’ssyndrome, who had been diagnosed withdiabetes type 1 from the age of 12 years,was admitted following a left hemisphericischaemic stroke in December 1998.She had no previous history of hyper-tension or vascular disease. She had

treated hypercholesterolaemia. She was anon-smoker and a teetotaller. At presenta-tion, she was in sinus rhythm, normo-tensive (BP 123/70), without audiblecardiac murmurs or carotid bruits. Capil-lary blood glucose was 20.9 on admission,and her last measured (November 1998)glycated haemoglobin was 9.6%. Thediagnosis of ischaemic stroke was sus-pected on the basis of clinical findings ofconfusion, dysphasia and a right densehemiparesis associated with upper motorneurone right VII cranial nerve palsy, andlater confirmed with a computer tomo-graphy (CT) scan of the head.

Carotid Doppler studies demonstratednormal common carotid, internal carotidand external carotid arteries on the rightside, with a completely occluded internal

carotid artery on the left and normalcommon and external carotid arteries.Anti-nuclear antibody, smooth muscleantibody, anticytoplasmic antibody andcomplement levels were within theirreference ranges.

Echocardiography showed good leftventricular function with absence ofvalvular dysfunction or cardiac wallabnormalities.

At retinal screening the subject wasfound to have proliferative retinopathyin the right eye, with haemorrhages andcotton wool spots scattered over mostof the right retina and with new vesselson the disc (Fig. 1). The left eye showedonly minimal background retinopathy(Fig. 2). She subsequently received laserphotocoagulation with resolution of the

Fig. 1. Photograph of the right retina at presentation.

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proliferative changes. Unfortunately, shehad not received retinal screening priorto this hospital admission.

This case focuses on the phenomenonof retinal sparing and asymmetry notcommonly seen in diabetes patients. Ourpatient had developed proliferativeretinopathy in one eye, as might beexpected due to an early age of onset, along duration of diabetes, poor metaboliccontrol, and no previous retinal screening.These morphological changes in the retinawere confirmed by ophthalmoscopy andfound distinct from the changes seen inhypoperfusion retinopathy. The rightcarotid Doppler studies had been clear.

The absence of similar proliferativechanges in the fellow (left) eye was moreintriguing and could only be explained onthe basis of retinal sparing reportedpreviously (Slusher 1975; Neupert et al.1976; Browning et al. 1988) and believedto result from the development of acollateral circulation (Neupert et al.1976), or reduced metabolism of theretinal endothelial cells, thereby repre-senting an adaptive response to a gradualreduction in blood flow (Browning et al.1988). Further, a reduction in the net

retinal vascular pressure due to either arise in intraocular pressure or to reducedretinal blood flow could also protect theretina by preventing the development ofarteriovenous shunts, which couldpotentially haemorrhage or give rise toproliferation (Gay & Rosenbaum 1966;Slusher 1975).

For the ophthalmological standpoint,this case of retinal sparing leading tomarked asymmetry between the eyesmust be distinguished from other causesof asymmetrical retinopathy, especiallyincomplete retinal vein occlusion andhypoperfusion retinopathy as themanagement could be different. Hypo-perfusion retinopathy is characterizedby mid-peripheral location of micro-aneurysm and haemorrhages, sometimeswith neovascularization, whilst thesechanges, if present, are usually seen atthe posterior pole in diabetic retinopathy(Dahlmann et al. 2001). Fluoresceinangiography may help to distinguishthese conditions. In addition, hypo-perfusion may worsen pre-existingdiabetic retinopathy and therefore thetwo conditions might coexist (Ino-ueet al. 1999)

For the diabetologist, asymmetricalretinopathy in a diabetes patient shouldprompt further evaluation of bothcarotid arteries to detect vascular dis-ease. It is worth remembering in thiscontext that increased carotid intima-media thickness is positively correlatedwith insulin resistance (Bonora et al.1997) in the general population andwith dyslipidaemia and albuminuria intype 2 diabetes patients (Wiley et al.1995). Further, more recently, it hasbeen shown to be associated with anincreased risk of stroke and myocardialinfarction in older adults even withoutany prior history of cardiovascular dis-ease (O’Leary et al. 1999).

ReferencesBonora E et al. (1997): Intimal-medial thick-

ness of the carotid artery in non-diabetic and

NIDDM patients. Relationship with insulin

resistance. Diabetes Care 20: 627–631.

Browning DJ, Flynn HW & Blankenship G

(1988): Asymmetric retinopathy in patients

with diabetes mellitus. Am J Ophthalmol

105: 584–589.

Fig. 2. Photograph of the left retina at presentation.

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Dahlmann AH, McCormack D & Harrison RJ

(2001): Bilateral hypoperfusion retinopathy.

J R Soc Med 94: 298–299.

Gay AJ & Rosenbaum AL (1966): Retinal

artery pressure in asymmetric diabetic

retinopathy. Arch Ophthalmol 75: 758.

Ino-ue M et al. (1999): Ocular ischaemic syn-

drome in diabetic patients. Jpn J Ophthalmol

43: 31–35.

Neupert JR et al. (1976): Rapid resolution

of venous stasis retinopathy after carotid

endarterectomy. Am J Ophthalmol 81:

600–602.

O’Leary DH et al. (1999): The Cardiovascular

Health Study Collaborative Research Group.

Carotid-artery intima and media thickness as

a risk factor for myocardial infarction and

stroke in older adults. N Engl J Med 340:

14–22.

Slusher MM (1975): Retinal sparing in diabetic

retinopathy. Southern Medical Journal 68:

655–657.

Wiley KA et al. (1995): Albuminuria is an inde-

pendent predictor of carotid intima-media

thickness and atherosclerosis in NIDDM

patients. Diabetes Care 18: 1502–1503.

Correspondence:

Dr Ansu Basu

Department of Diabetes, Endocrinology and

Metabolism

City Hospital

Dudley Road

Birmingham B18 7QH

UK

Tel:þ 44 121 507 4988

Fax:þ 44 121 507 45

Email: [email protected]

Orbital involvement in multifocal fibrosclerosis

Maria Kyhn,1 Margrethe Herning,2 Jan Ulrik Prause1 and Steffen Heegaard1

1Eye Pathology Institute, University of Copenhagen, Copenhagen, Denmark2Danish Research Centre for Magnetic Resonance, University Hospital Hvidovre,

Hvidovre, Denmark

doi: 10.1046/j.1600-0420.2004.00238.x

Sir,

M ultifocal fibrosclerosis is char-acterized by fibrous lesions

occurring at a variety of sites. Clinicalvariants include retroperitoneal fibro-sis, Riedel’s thyroiditis, sclerosingcholangitis and mediastinal fibrosis(Aylward et al. 1995). In only a fewcases the sclerosing variant ofidiopathic non-specific orbital inflam-mation has been reported as a mani-festation of multifocal fibrosclerosis(Comings et al. 1967; Fruh et al. 1975;Schonder et al. 1985; Levine et al. 1993;Aylward et al. 1995; Oguz et al. 2002).

We present a case of sclerosingidiopathic non-specific orbital inflam-mation in combination with retroperi-toneal fibrosis. In this report we areable to present the histopathologicalfindings in both locations.

A 40-year-old white woman with ahistory of unexplained hypertensionpresented in 1991 with diplopia, inter-mittent painful swelling and erythemaof her right eye region. There was nofamilial history of similar symptoms.Her visual acuity (VA) was 0.8/1.0. Anorbital computer tomography (CT)scan showed thickening of the lateralrectus muscle and of the lacrimalgland in the right orbit. During thefollowing 6 months the patient devel-oped pain in the right flank and abdo-men. Abdominal CT images revealed

retroperitoneal tumour masses shred-ding the aorta, inferior vena cava andthe ureteres, causing unilateral hydro-nephrosis. An explorative laparotomydisclosed retroperitoneal and para-metrical tumour masses. A peroperat-ively performed biopsy indicatedretroperitoneal fibrosis. Systemic treat-ment with oral prednisolone (30 mg/day)was initiated. On this treatment thepatient’s eye symptoms regressed, andthe medication was continued for aperiod of 3 years. Six months after pred-nisolone was discontinued the orbitalsymptoms recurred, with progressiveexophthalmus, diplopia and lid swelling.A magnetic resonance imaging (MRI)scan verified progression in the soft tis-sue changes in the right orbit (Fig. 1A),as well as retroperitoneally (Fig. 1B). Abiopsy from the tumour in the right orbitshowed orbital fibrosis. The patient wasnow started on oral prednisolone incombination with oral azathioprin.Despite this treatment the symptomscontinued to fluctuate and during thefollowing 4 years the orbital symptomsgradually progressed. In April 2001, alateral orbitotomy was performed. Thetumour had extended posteriorly to thesuperior orbital fissure and was onlypartly excised. Oral azathioprin treat-ment was continued postoperatively,this time for 2 years. On this treatment,

the orbital as well as the retroperitonealchanges showed no signs of activity.

Histopathological examination ofthe tissue from both the orbit (Fig. 1C)and the retroperitoneum (Fig. 1D)demonstrated the same morphology,with masses of dense, hyalinizedfibrous tissue. The fibrous tissue wasarranged in more or less concentricalwhorls around obliterated small bloodvessels. Areas with moderately severediffuse lymphocyte and plasma cellinfiltration were seen. A heterogeneouslymphocytic cell population was identi-fied with a pan-T-cell marker (CD 3), apan-B-cell marker (CD 79a), a mixedIgG, IgA and IgM immunoglobulinheavy chain and both kappa andlambda light chains.

Two male siblings, offspring of con-sanguine parents, were described byComings et al. (1967) as presentingwith different combinations of orbitalpseudotumour, retroperitoneal fibrosis,mediastinal fibrosis, sclerosing cholan-gitis and Reidel’s thyroiditis. The term‘multifocal fibrosclerosis’ was sug-gested for the first time, proposing asingle common disease. An autosomalrecessive or polygenic inheritance wassuggested (Comings et al. 1967).

The aetiology of multifocal fibro-sclerosis remains unknown. An infec-tious origin has been suspected, as the

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orbital changes have been observed fol-lowing upper respiratory tract infections(Mombaerts et al. 1996). The finding ofconcurrent autoimmune diseases in 10%of patients with idiopathic non-specificorbital inflammation has lead to thehypothesis of an autoimmune origin(Mombaerts et al. 1996). Rootmanet al. (1994) suggested a cell-mediatedpathogenesis in which cytokines withcertain fibroproliferative abilitiesdirectly stimulate fibroblast prolifera-tion and collagen production.

Lymphocytic infiltration withoutlymphoid follicles was found in thepresent case. Rootman et al. (1994)compared histological specimens of 16idiopathic sclerosing non-specific orbi-tal inflammations with six differentcases of retroperitoneal fibrosis. Afterspecific staining, the orbital specimensshowed T-cells in 94% of cases, B-cellsin 40%, tissue macrophages in 56%,HLA Dr positive antigen presentingcells and activated T-cells in 44%. Ofthe immunoglobulins, the orbital speci-mens showed kappa in 80%, lambda in63%, IgG in 73%, IgA in 44% and

IgM in 31% of cases. The results weresimilar for the six cases of retroperi-toneal fibrosis and accord with thepresent case.

Treatment of multifocal fibrosclero-sis is controversial (Mombaerts et al.1996). Based on the symptoms, treat-ment possibilities start with systemicprednisolone, possibly combined withimmunosuppressing agents. If thesymptoms do not regress on this treat-ment, surgical removal and/or radia-tion therapy should be considered(Mombaerts et al. 1996).

ReferencesAylward GW, Sullivan TJ, Garner A, Moseley

I, Wright JE (1995): Orbital involvement in

multifocal fibrosclerosis. Br J Ophthalmol

79: 246�249.

Comings DE, Skubi KB, Van Eyes J &

Motulsky AG (1967): Familial multifocal

fibrosclerosis. Ann Intern Med 66: 884�892.

Fruh D, Jaeger W & Kafer O (1975): Orbital

involvement in retroperitoneal fibrosis

(morbus Ormond). Mod Probl Ophthalmol

14: 651�656.

Levine MR, Kaye L, Mair S & Bates J (1993):

Multifocal fibrosclerosis. Arch Ophthalmol

111: 841�843.

Mombaerts I, Goldschmeding R, Schlingemann

RO & Koornneef L (1996): What is orbital

pseudotumour? Surv Ophthalmol 41: 66�78.

Oguz KK, Kirath H, Oguz O, Cila A, Oto A &

Gokoz A (2002): Multifocal fibrosclerosis: a

new case report and review of the litterature.

Eur Radiol 12: 1134�1138.

Rootman J, McCarthy M, White V, Harris G

& Kennerdell J (1994): Idiopathic sclerosing

inflammation of the orbit. Ophthalmology

101: 570�584.

Schonder AA, Clift RC, Brophy JW & Dane

LW (1985): Bilateral recurrent orbital inflam-

mation associated with retroperitoneal fibro-

sclerosis. Br J Ophthalmol 69: 783�787.

Correspondence:

Steffen Heegaard

Eye Pathology Institute

University of Copenhagen

Frederik V’s Vej 11

DK-2100 Copenhagen

Denmark

Tel:þ 45 35 32 60 70

Fax:þ 45 35 32 60 80

Email: [email protected]

Fig. 1. (A) MRI scan of the orbit. The right orbit shows massive fibrosis (asterisk). (B) MRI scan of the abdomen. Massive fibrosis (asterisk)

surround the aorta (white arrow) and vertebra body (C) Histopathological specimen from the orbit. Note the fibrosis and whorls around vessels

(black arrows) (original magnification � 70). (D) Histopathological specimen from the retroperitoneum. The morphology is similar to the orbital

biopsy. Higher magnification of the whorls (black arrow) showing fibrosis around the vessels and scattered inflammatory cells between the collagen

fibres (original magnification � 140).

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