COMBINED CILIORETINAL ARTERY AND PARTIAL CENTRAL RETINAL VEIN OCCLUSION IN THE POSTPARTUM PERIOD Yasemin Ozdamar, MD,* Ugur Gurlevik, MD,* Golge Acaroglu, MD,† Mehmet Yasin Teke, MD,* Orhan Zilelioglu, MD*

Purpose: To report a combined cilioretinal artery and partial central retinal vein occlusion shortly after delivery in a twin-pregnant woman. Methods: A 25-year-old woman presented to our clinic with the complaint of blurred vision in the right eye 1 week after delivery. She underwent detailed ophthalmic and systemic investigations. Results: The patient had an uncomplicated twin pregnancy. Ocular examination showed combined cilioretinal artery and partial central retinal vein occlusion in the right eye. Systemic diseases were excluded. Laboratory evaluations revealed increased plasma D-dimer level (1.64 mg/mL). Spontaneous recovery occurred without treatment 1 month after delivery. Conclusion: Twin pregnancy may lead to exaggerated hypercoagulability and increased D-dimer level in pregnant women. This situation may be a risk factor for retinochoroidal vascular occlusions. RETINAL CASES & BRIEF REPORTS 4:99 –101, 2010

From the Departments of *Retinal Diseases and †Neuroophthalmology, Ankara Ulucanlar Eye Research Hospital, Ankara, Turkey.

retinal vein (CRVO) and cilioretinal artery occlusion (CLRAO) in our case is a rare phenomenon. Materials and Methods

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regnancy can cause multiple physiologic and nonphysiologic changes in women’s body. Alterations in systemic hemostatic systems occur in pregnancies, and also, they are excessive in multifetal pregnancies. The adaptation of the maternal hemostatic system to pregnancy predisposes women to an increased risk of thromboembolism.1,2 Pregnancy is associated with ocular changes that can be physiologic or pathologic or affect a preexisting condition. Pregnancy-induced hypercoagulability may lead to vascular occlusion of the retina and choroid.3 However, the combination of partial central

A 25-year-old woman presented with a sudden, painless, blurred vision in the right eye. The ophthalmic examination included slit-lamp biomicroscopy, ophthalmoscopy, and color fundus photography. Fluorescein angiography was not performed because she refused to abstain from nursing her babies. Laboratory and systemic evaluations were performed and a detailed medical history was noted. Results The patient had blurred vision in the right eye 1 week after delivery. Her visual acuity was 20/20 in both eyes. Anterior segment findings, pupils, and intraocular pressures were unremarkable in both eyes. Fundus examination of the right eye showed a small area of localized juxtafoveal retinal whitening. This

The authors have no proprietary or financial interest in any of the products used in this study. Reprint requests: Yasemin Ozdamar, MD, Ulucanlar cad. No:59 Altındag˘, Ankara 06100, Turkey; e-mail: [email protected]

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Fig. 1. Fundus appearance of the combined cilioretinal artery occlusion and incomplete central retinal vein occlusion at the initial examination.

finding was accompanied by dilated and tortuous veins and a few retinal hemorrhages (Figure 1). Fundoscopy of the left eye was normal. Ophthalmologic findings were felt to be consistent with the diagnosis of combined CLRAO and incomplete CRVO of the right eye. She had an uncomplicated twin pregnancy, elective caesarean section was performed, and normal twins were delivered 1 week before her presentation. She had no preeclampsia/eclampsia or abruption of placenta or clinically significant previous medical history. Blood pressure, full blood count, fasting blood sugar, and partial thromboplastin time were within normal limits. Serologic tests were negative for antiphospholipid antibody syndrome, dysproteinemia, and blood dyscrasias. There was no elevated homocysteine level, activated protein C resistance, protein S or C deficiency, antithrombin III deficiency, factor V Leiden mutation, or prothrombin A 20210 mutation. The analysis of the coagulation–fibrinolysis system revealed that the level of plasma D-dimer was higher (1.64 mg/mL) than normal limits (0.00 – 0.50 mg/mL). Carotid ultrasound and transthoracic echocardiogram were normal. The patient was not given any treatment, and she was followed-up regularly. Ocular and laboratory findings rapidly normalized in 1 month after delivery (Figure 2). Discussion Pregnancy may be associated with a variety of retinal findings in the mother, which may occur due to hemodynamic alterations. In normal pregnancies, the hemostatic balance is displaced toward hypercoagulability, which protects women from fetal hemorrhage



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Fig. 2. Disappearance of retinal findings 1 month after delivery.

during delivery. Maternal hypercoagulability may counteract a progressive increase in maternal thrombolytic factors, which persists during puerperium. Because human multifetal gestation requires greater physiologic changes, the instability in hemostasis may further be exaggerated in twin pregnancies.1,2 In normal hemostasis, fibrinogen is converted to fibrin monomer and fibrinopeptides, and fibrin polymer is formed by cross-linkage. When fibrin is degraded, its products such as D-dimer are released. Quantification of fibrinolysis is enabled by the measurement of plasma D-dimer levels. D-dimer is formed only by plasmin-mediated proteolysis of cross-linked fibrin and is the most specific marker of fibrinolysis, which is active in the presence of thrombotic disorders.4 An increase in maternal plasma D-dimer levels has been demonstrated in normal or complicated pregnancies and in the postpartum period.1,2 Coagulopathy observed in the laboratory is not clinically apparent in the majority of women with multifetal gestation. Thromboembolic events of the eye rarely occur unless pregnancy is complicated. These events include purtscherlike retinopathy or ocular changes associated with disseminated intravascular coagulation, thrombotic thrombocytopenic purpura, and retinal artery or retinal vein occlusions. These occlusions are commonly related to pregnancy-induced hypertension.3,5,6 Several studies have mentioned the combination of CLRAO and CRVO in nonpregnant patients.7–9 There are three forms of CLRAO: isolated CLRAO, its association with CRVO, or anterior ischemic optic neuropathy. The cilioretinal artery occlusion accounts for 5% of all retinal artery occlusions.10 The etiologic factors for a CLRAO combined with CRVO have not been clearly established, and several factors such as

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inflammation, thrombosis, atherosclerosis, arterial hypotension, and vasospasm have been suggested in various studies. The pathogenetic mechanism of combined CRVO and CLRAO still remains unclear probably because of the unawareness on the regulation of cilioretinal blood flow. The decrease in cilioretinal arterial perfusion may lead to CLRAO. Two hypotheses have been suggested with regard to the pathogenesis of decreased perfusion of CLRA in the combination of CLRAO and CRVO. The first suggests an increase in the venous pressure caused by CRVO. The second concerns a primary reduction in cilioretinal and retinal arterial perfusion pressure leading to decreased retinal circulation and consequent venous stasis and thrombosis. Deterioration of visual acuity depends on the precise area of macular supply or macular edema by CRVO, and in general, the outcome of a combined CRVO and CLRAO depends mainly on the sequelae of the venous occlusion.7–9 In our case, we thought that there was partial occlusion in the central retinal vein and the cause of CLRAO might be secondary to partial CRVO, because our patient was a postpartum woman with an increased level of D-dimer, which predisposes venous occlusion.11 It may be thought that exaggerated hypercoagulability in twin pregnancy may be a possible risk factor and may lead to CLRAO in conjunction with partial CRVO during the immediate postpartum period. To the best of our knowledge, no such association has been previously described shortly after delivery. In conclusion, increased D-dimer levels should be considered as a responsible factor even in the absence of complicated pregnancy. Further studies are neces-

sary to evaluate changes in D-dimer and to assess its relationship with thromboembolic events during or shortly after pregnancy. Key words: cilioretinal artery, central retinal vein, hypercoagulability, pregnancy. References 1. 2.

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Bar J, Blickstein D, Hod M, et al. Increased D-dimer levels in twin gestation. Thromb Res 2000;98:485– 489. Nolan TE, Smith RP, Devoe LD. Maternal plasma D-dimer levels in normal and complicated pregnancies. Obstet Gynecol 1993;81:235–238. Sheth BP, Mieler WF. Ocular complications of pregnancy. Curr Opin Ophthalmol 2001;12:455– 463. Wilde JT, Kitchen S, Kinsey S, Greaves M, Preston FE. Plasma D-dimer levels and their relationship to serum fibrinogen/fibrin degradation products in hypercoagulable states. Br J Haematol 1989;71:65–70. Rahman I, Saleemi G, Semple D, Stanga P. Pre-eclampsia resulting in central retinal vein occlusion. Eye 2006;20:955– 957. Lara-Torre E, Lee MS, Wolf MA, Shah DM. Bilateral retinal occlusion progressing to long-lasting blindness in severe preeclampsia. Obstet Gynecol 2002;100:940 –942. Brazitikos PD, Pournaras CJ, Othenin-Girard P, Borruat FX. Pathogenetic mechanisms in combined cilioretinal artery and retinal vein occlusion: a reappraisal. Int Ophthalmol 1993; 17:235–242. Wrigstad A. Central retinal vein occlusion combined with occlusion of a cilioretinal artery. A case report Acta Ophthalmol Scand 1998;76:503–505. Hayreh SS, Fraterrigo L, Jonas J. Central retinal vein occlusion associated with cilioretinal artery occlusion. Retina 2008;28:581–594. Brown GC, Moffat K, Cruess A, Magargal LE, Goldberg RE. Cilioretinal artery obstruction. Retina 1983;3:182–187. Rosenberg VA, Lockwood CJ. Thromboembolism in pregnancy. Obstet Gynecol Clin North Am 2007;3:481–500.

Combined cilioretinal artery and partial central retinal vein occlusion in the postpartum period.

To report a combined cilioretinal artery and partial central retinal vein occlusion shortly after delivery in a twin-pregnant woman...
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