A Case of Intramuscular Hemangioma Presenting with Large-angle Hypertropia

Abstract

PurposeTo report the case of a patient with large-angle hypertropia of an intramuscular hemangioma of the right superior rectus muscle (SR).MethodsA 63-year-old man with progressive vertical deviation of the right eye for the past 6 months visited our strabismus department; his condition was not painful. An examination indicated that he had 60PD of right hypertropia at distance and near in primary gaze. Additionally, a significant limitation of his downgaze was noted. The right eye appeared mildly proptotic, and the upper and lower eyelids were slightly edematous. Corrected vision was 20/20 in both eyes.ResultsOrbital magnetic resonance imaging (MRI) studies revealed fusiform enlargement of the right superior rectus muscle, with prominent but irregular enhancement following gadolinium administration. Incisional biopsy revealed an intramuscular hemangioma in the superior rectus muscle with cavernous-type vessels.ConclusionsThis case demonstrates that intramuscular hemangioma should be considered in the differential diagnosis of isolated extraocular muscle enlargement and unusual strabismus.

Combined endoscopic and computed tomography guided trans-nasal embolisation of intramuscular hemangioma of the medial rectus muscle: a rare case

“…The first case of an intramuscular hemangioma affecting the extraocular muscles was reported by Christensen et al 9 Since then, a total of eight cases have been published in the literature. 3,[9][10][11][12][13][14] The ages ranged from 3 to 63 years, affecting both genders. Due to the rarity of this presentation, there is no standardised treatment protocol.…”

Intracranial extension of an intramuscular haemangioma of superior rectus: case report and literature review

“…Isolated IMHs of a single extraocular muscle are exceedingly rare [ 3 ]. The histological characteristics of IMH have been sub-classified by Allen and Enzinger to be capillary type (small vessel), cavernous type (large vessel) and mixed types, with frequencies of 50, 29, and 21%, respectively [ 1 ]. The exact aetiology is unknown, although congenital, traumatic and hormonal theories have been proposed [ 2 ].…”

“…Kim et al describe the IMH presented in a 63-year-old man presented with diplopia; they report the involvement of the right superior rectus with large-angle hypertropia, and confirmed the IMH diagnosis by biopsy. A right superior rectus recession and inferior recuts resection was made, and systemic corticosteroids administered to improve the swelling [ 1 ]. Mehta et al report on the case of an 11-year-old male child with a two-year history of a mass in the nasal aspect of the right eye and an associated decrease in EOM.…”

“…Imaging features of IMHs often show a sharp demarcation, with bright areas within the tumours that indicate a combination of large vessels with stagnant blood and nonvascular tissue. Images also show areas of septated-striated high signal intensity and curvilinear areas of low signal intensity [ 1 , 6 , 7 ].…”

“…Intramuscular haemangiomas (IMHs) are a rare type of tumour, accounting for less than 1% of the total number of haemangiomas; their occurrence in extraocular muscle is extremely rare [ 1 – 3 ]. IMHs are considered to be benign, congenital, slow growing neoplasms that can remain undetected for a long time, only becoming apparent when sudden growth causes symptoms.…”