PMC7519974_01
(a) Facial appearance of the patient at presentation showing slight lateral displacement of the left eye. (b) Anterior segment photograph shows lymphatic malformation cysts in the inferonasal conjunctiva (arrow). (c) Lesion similar to lymphatic malformation affecting the left hard palate (arrow).
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The left eye was mildly displaced laterally although the patient did not complain of double vision (Figure 1(a)). Anterior segment biomicroscopic examination of the left eye revealed that the plica semilunaris and inferonasal bulbar conjunctiva had a multicystic appearance (Figure 1(b)). There was a lesion similar to LM on the patient's left hard palate (Figure 1(c)).
(a) Anterior segment swept source optical coherence tomography of the left eye demonstrates dilated lymphatic channels manifesting as hyporeflective spaces with different sizes demarcated by hyperreflective septae (arrows). (b) T1-weighted coronal MR image shows the inferonasally located intraconal tumor that is isointense with respect to muscle (arrow) in the left orbit. (c) T2-weighted axial MR image shows that the left orbital tumor is hyperintense (arrow).
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Anterior segment swept-source optical coherence tomography (AS-SSOCT) (DRI OCT Triton Plus, Topcon, Tokyo, Japan) showed there was elevation of the plica semilunaris and bulbar conjunctiva comprising variable-sized hyporeflective spaces separated by hyperreflective tissue in the left eye (Figure 2(a)). T1-weighted images, hyperintense on T2-weighted images, and demonstrated contrast enhancement (Figures 2(b) and 2(c)). There was no vascular malformation affecting the brain and meninges.
(a) Gross photograph of the excised left orbital tumor showing the reddish well-circumscribed mass measuring approximately 1.5x1.5 cm in base dimensions. (b) Histopathological examination of the orbital tumor reveals enlarged vascular channels with thick walls, the lumens of which are filled with erythrocytes embedded in a fibrous stroma (arrows), consistent with orbital cavernous venous malformation (H.E. x100). (c) Histopathological examination of the conjunctival lesion shows several ectatic bloodless lymphatic channels, lined with flattened endothelium (arrows). There is prominent lymphoid infiltration and aggregates in the stroma, consistent with the diagnosis of conjunctival lymphatic malformation (H.E. x100).
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The basal diameters of the excised reddish, smooth-edged, capsulated orbital mass were approximately 1.5 x 1.5 cm (Figure 3(a)). The histopathological diagnosis of the orbital mass was established as VM (Figure 3(b)). Histopathological examination of the conjunctival lesion revealed a vascular structure containing lymphatic fluid surrounded by endothelium and conjunctival epithelial cells on the surface, consistent with LM (Figure 3(c)).
Facial photograph of the patient 3 months after surgery shows excellent outcome with an orthophoric appearance.
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The conjunctiva looked devoid of any major lymphangioma cysts (Figure 4).