DIABETIC PAPILLOPATHY TWO CASE REPORTS

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1 Acta Ophthalmologica 2011, Vol.37(1-2) ISSN Prikaz slučaja UDK: : DIABETIC PAPILLOPATHY TWO CASE REPORTS Sonja Cekić 1,2, Zlatica Višnjić 2, Dragan Veselinović 1,2, Predrag Jovanović 1, Gordana Stanković Babić 1, Milena Vujanović 2 1 Faculty of Medicine University of Niš 2 Eye Clinic, Clinic Centre Niš, Serbia Diabetic papillopathy is a rare condition that occurs in diabetic patients (types 1 and 2). Its prevalence is 1.4% in diabetic patients. The exact pathogenesis is uncertain. The aim of our study is to present two patients with diabetic papillopathy. The study enrolled two patients aged 40 and 56 years. They were both suffering from diabetes mellitus, one from type 1 and the other from type 2. The diagnose of diabetes mellitus had been made 25 years and in other patients 12 years before the diagnose of diabetic retinopathy. A standard ophthalmic examination was performed, including visual acuity, anterior segment and posterior segment examination, applanation tomometry, photophundus and fluorescein angiography. Ultrasonography, A/B scan, CT and MRI as neuroimaging methods were preformed in both patients. The main symptom in both patients was sudden, painless loss of visual acuity in one eye. Anterior segment examination was unremarkable. Posterior segment showed a swollen left optic disc in one patient, and in the other one the right eye was affected. The diffuse macular edema was present in one patient, and mild-to-moderate nonproliferative diabetic retinopathy (NPDR) in both. The right eye fundus showed only mild NPDR. Fluorescein angiography was performed and revealed clinically significant macular edema and a hyperfluo-rescent optic disc. Neuroimaging of orbit and brain was normal in all patients. The spontaneous recovery occurred in both patients. Laser treatment was preformed according ETDRS protocol. Optic disc was partially or completely atrophic in the affected eye. Diabetic papillopathy is a rare condition. It is necessary to exclude anterior ischemic optic disc papillopathy, space occupying intra cranial and intra orbital lesions, as well as other inflammatory processes. Therapy, as well as using anti-vegf is a matter of debate. Acta Ophthalmologica 2011;37(1-2): Key words: diabetes mellitus, microvascular complications, optic disc Introduction Diabetic papillopathy (DP) is a rare condition that occurs in diabetic patients (types 1 and 2). Its prevalence is 1.4% in diabetic patients, and the exact pathogenesis is uncertain (1). Some risk factors include rapid improvement of the metabolic control and a small cup-to-disc ratio (2). One study suggested that DP could be a mild form of nonarteritic anterior ischemic optic neuropathy (AION) (3). DP is unilateral in 50% of the cases. Diabetic retinopathy in eyes with DP tends to progress with time (1). Visual prognosis is usually good in cases of DP (1). The disc swelling tends to resolve spontaneously within 3 4 months. However, in very few case reports treatment with intravitreal corticosteroids or antivascular endothelial growth factors (anti-vegf) was suggested. Literature 31

2 search was performed, and three reports of treatment with intravitreal injections were found: one with an intravitreal triamcinolone acetate injection and the other two with an intravitreal bevacizumab (Avastin) injection (5,6,7). In all three cases, visual acuity and disc swelling improved rapidly after the treatment. The use of periocular corticosteroid injection in treatment has been described, too (8). The aim of our study is to present two patients with diabetic papillopathy. The first case A 40-years old male presented with sudden, painless visual loss in the right eye. His best corrected visual acuity was (BCVA) 0,6 by Snellen chart, and 1,0 in the left eye. The anterior segment examination by slitlamp biomicroscopy was without any remarkable changes. Intraocular pressure was 16mmHg on both eyes, applanation tonometry. By indirect ophthalmoscopy, on dilated pupil, optic disc was prominent with hemorrhage split on edge, exudats in area of macula and small, punctiform hemorrhages and mycroaneurismas (Figure 1). Early leaking on optic disc, mycoaneurisma on posteror pole, and diabetic macular edema was present on fluorescein angiography (FA) (Figure 2). An ultrasonography B-scan (USG) showed prominent optic nerve. The so-called crescento sign, typical for intraorbial tumors, was absent. A magnetic resonance (MRI) and computed tomography (MSCT) of the orbit and brain were performed. Neuroimaging procedures were without abnormalities. The second case A 56-years old male presented with sudden, painless visual loss in the right eye. Best corrected visual acuity was (BCVA) 0,6 by Snellen chart, and 0,7 in the left eye. Anterior segment examination by slitlamp biomicroscopy was without any remarkable changes. Intraocular pressure was 14mmHg in the right eye, and 18mmHg in the left eye, by applanation tonometry. By indirect ophthalmoscopy, on dilated pupile, optic disc was prominent with hemorrhage split on edge, exudats in area of macula and small, punctiform hemorrhages and mycroaneurismas (Figure 3). Optic disc leakage, microaneurysms in the posteror pole, and diabetic macular edema were ide-ntified by fluorescein angiography (Figure 4). An ultrasonography B-scan (USG) showed prominent optic nerve, without echolucent circle within the optic nerve sheath (separating the sheath from the optic nerve). An MRI and MSCT of the orbit and brain were performed. No abnormalities were found. Discussion In our two case reports patients presented with an acute painless visual loss. Neuroimaging, MSCT and MRI did not show any abnormalities. Absence of any ocular inflammatory signs ruled out inflammatory causes. A conclusion of diabetic papillo-pathy was made due to the following reasons: young age, uncontrolled type 2 diabetes mellitus, absence of pain, presence of associated macular edema which is common in DP, early disc hyperfluo-rescence on FA, and good visual outcome posttreatment (9). Visual prognosis without treatment is also favorable in these cases, but it takes prolonged period of time to improve (9). Diabetic papillopathy is sometimes considered as a mild form of nonarteritic anterior ischemic optic neuropathy (AION) (10). On the other hand, it is also considered as a distinctive disorder. Differences between the two are as follows: first, visual loss in AION is worse than in DP(1). Second, visual field defect in AION is usually persistent, in contrast to DP, in which visual field defects are transient. Third, visual improvement is limited in AION cases to only 30% of the patients, but in DP usually patients will have spontaneous and slow visual acuity improvement, and around 85% of the cases have final visual acuity of 20/50 or better. Moreover, FFA may assist to differentiate between both conditions. In AION cases, FA shows early disc hypofluorescence due to hypoperfusion with late leakage around the affected segment (9). However, in DP cases the FA shows a very early disc leakage that increases throughout the study (11). The early hyperfluorescence is most likely due to telangiectasia of the optic disc. Finally, AION typically occurs in older patients, whereas DP occurs in younger ages (4). There are some literature findings about the use of anti-vegf, bevacizumab in treatment of DP (13). The main indication of using anti- VEGR in this case was to treat the macular edema. Consequently, the patient had a rapid 32

improvement of his vision postinjection along with resolution of the macular edema and disc swelling (13).

However, the good response to the treatment may suggest that vascular endothelial growth factor (VEGF) plays an important role in the pathogenesis of this condition.

As mentioned earlier, periocular corticosteroid was found also to be effective in accelerating improvement of DP.

3 improvement of his vision postinjection along with resolution of the macular edema and disc swelling (13). The mechanism of action of bevacizu-mab on DP is not well understood. However, the good response to the treatment may suggest that vascular endothelial growth factor (VEGF) plays an important role in the pathogenesis of this condition. Further studies are required to explore the role of VEGF in the pathogenesis of DP. As mentioned earlier, periocular corticosteroid was found also to be effective in accelerating improvement of DP. This suggests inflammatory elements could be involved in pathogenesis of this disorder (8). Recently reported, AION can be presented as an adverse effect of anti-vegf (12). The patients in these case reports will need follow-up of progression of diabetic retinopathy as well as of macular edema. Figure 1. Optic disc prominent with hemorrhage split on edge, exudats in area of macula Figure 2. FA a) laeking on optic disc b) laeking on optic disc and macular edema 33

Figure 3 Second case prominent optic disc on the right and left eye Conclusion Figure 4.

dyschromatopsia. The condition is Figure 5. USG B scan self-limiting and vision generally recovers to better than 20/30 in most patients. Occurrence is rare, with an incidence of 0.5%.

Bayraktar Z, Alacali N, Bayraktar S. Diabetic papillopathy in type II diabetic patients. Retina. 2002;22:752 8 2. Ostri C, Lund-Anderson H, Sander B, Hvidt-Nielsen D, Larsen M.

4 Figure 3 Second case prominent optic disc on the right and left eye Conclusion Figure 4. FF Diabetic papillopathy is characterized by optic disc edema in the absence of optic nerve dysfunction, elevated intracranial pressure, nerve infiltration or infection, afferent pupillary defect and dyschromatopsia. The condition is Figure 5. USG B scan self-limiting and vision generally recovers to better than 20/30 in most patients. Occurrence is rare, with an incidence of 0.5%. It is important for clinicians to be aware that this condition can mimic papilledema and optic nerve neovascularization. Therapy, as well as using anti-vegf is a matter of debate. Literature 1. Bayraktar Z, Alacali N, Bayraktar S. Diabetic papillopathy in type II diabetic patients. Retina. 2002;22: Ostri C, Lund-Anderson H, Sander B, Hvidt-Nielsen D, Larsen M. Bilateral diabetic papillo-pathy and metabolic control. Ophthalmology. 2010; 117: Hayreh SS, Zimmerman B. Nonarteritic anterior ischemic optic neuropathy: Clinical characteristics in diabetic patients versus nondiabetic patients. Ophthalmology 2008; 115: Vaphiades MS. The disk edema dilemma. Surv Ophthalmol. 2002; 47: Al-Haddad CE, Jurdi FA, Bashshur ZF. Intravitreal triamcinolone acetonide for the management of diabetic papillopathy. Am J Ophthalmol. 2004; 137: Ornek K, Oğurel T. Intravitreal bevacizumab for diabetic papillopathy. J Ocul Pharmacol Ther. 2010; 26: Al-Dhibi H, Khan AO. Response of diabetic papillopathy to intravitreal bevacizumab. Middle East Afr J Ophthalmol. 2011;18:

5 8. Mansour AM, El-Dairi MA, Shehab MA, Shahin HK, Shaaban JA, Antonios SR. Periocular corticosteroids in diabetic papillopathy. Eye 2005; 19: Regillo CD, Brown GC, Savino PJ, Byrnes GA, Benson WE, Tasman WS, et al. Diabetic papillopathy: Patient characteristics and fundus findings. Arch Ophthalmol. 1995; 113: Atkins EJ, Bruce BB, Newman NJ, Biousse V. Treatment of nonarteritic anterior ischemic optic neuropathy. Surv Ophthalmol. 2010; 55: Oto S, Yilmaz G, Cakmakci A, Aydin P. Indocyanine green and fluorescein angiography in nonarteritic anterior ischemic optic neuro-pathy. Retina. 2002; 22: Bodla A, Rao P. Non-arteritic ischemic optic neuropathy followed by intravitreal bevacizumab injection: Is there an association? Indian J Ophthalmol. 2010; 58: Al Hinai HS, Al-Abri MS, Al-Hajri RH. Diabetic papillopathy with macular edema treated with intravitreal bevacizumab. Oman J Ophthalmol. 2011; 4(3): DIJABETIČKA PAPILOPATIJA-PRIKAZ DVA SLUČAJA Sonja Cekić 1, Zlatica Višnjić 2, Dragan Veselinović 2, Predrag Jovanović 1, Gordana Stanković Babić 1, Milena Vujanović 2 1 Medicinski Fakultet, Univerzitet u Nišu 2 Očna klinika, Klinički Centar Niš, Srbija Dijabetička papilopatija je redak poremećaj koji se javlja kod bolesnika sa šećernom bolesti ( tip 1 i tip 2). Prevalenca pojave ovog poremećaja je 1,4% svih obolelih od šećerne bolesti. Tačna patogeneza nije poznata. Cilj studije je prikaz dva bolesnika sa dijabetičkom papilopatijom. Kod oba prikazana bolesnika postavljena je dijagnoza šećerne bolest, kod jednog tip 1 a kod drugog tip 2. Starost prikazanih bolesnika je 40 i 56 godina, a dijagnoza šećerne bolesti postavljena je kod prvog bolesnika 25 godina a kod drugog 12 godina. Načinjen je oftalmološki pregled koji je obuhvatio: određivanje vidne oštrine, pregled prednjeg segmenta i zadnjeg segmenta, aplanacionu tonometriju, fotodokumentaciju i fluoresceinsku angiografiju. Ultrazvuk oka, A i B sken, CT i MR, neuroimidžing metode su obavljene kod oba bolesnika. Naglo, bezbolno smanjenje vidne oštrine na jednom oku bio je osnovni simptom kod oba bolesnika. Nalaz prednjeg segmenta bio je uredan. Na zadnjem segmentu otok očnog diska kod jednog ispitanika bio je prisutan na desnom oku, a kod drugog na levom oku, blaga ka uznapredovaloj retinopatiji bila je prisutna kod oba ispitanika. Fluoresceinskom angiografijom prisutan je izliv na optičkom disku zahvaćenog oka, kao i klinički signifikantan edem u makuli. Neuroimidžing metode orbite i mozga bile su uredne. Spontani oporavak bio je prisutan kod oba bolesnika. Lasero tertman je primenjen u skladu sa ETDRS protokolom. Na zahvaćenom oku zahvaćen optički disk bio je delimično ili u potpunosti atrofičan. Dijabetička papilopatija je redak poremećaj. Značajno je postaviti diferencijalno dijagnostičku razliku u odnosu na prenje ishemičnu optikopatiju, ekspanzivne procese u orbiti i intraranijalne, kao i inflamatorne procese. Terapija je još uvek predmet diskusije. Acta Ophthalmologica 2011;37(1-2): Ključne reči: dijabetička papilopatija, edem, dijagnoza Kontakt: Sonja Cekić Medicinski fakultet u Nišu Bul.dr Zorana Đinđića Niš, Srbija sonjaziv@yahoo.com 35

The Prevalence of diabetic optic neuropathy in type 2 diabetes mellitus

The Prevalence of diabetic optic neuropathy in type 2 diabetes mellitus Received: 25/4/2016 Accepted: 8/12/2016 Introduction Diabetic papillopathy is an atypical form of non-arteritic anterior ischemic