하직근 이상을 동반한 선천 한눈상전장애 1예
A Case of Congenital Monocular Elevation Deficiency Associated with Inferior Rectus Anomaly
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목적: 하직근 이상을 동반한 congenital monocular elevation deficiency (MED)를 경험하여 보고하고자 한다. 증례요약: 특이병력 없는 3세남아가 좌안 하사시를 주소로 내원, 제1안위와 외전 시만 상전부전을 보이며, 제1안위에서 좌안 내사시와60PD 하사시 및 가성안검하수를 보였고 Bell현상 음성, 상전충동운동속도 정상, 안와CT검사상 얇고 코쪽으로 치우친 하직근이 안와첨근처에서 비대를 보였다. 수술 시 견인검사상 강양성, 근부착점 및 주행이 코쪽으로 변위된 뻣뻣하고 짧고 섬유화된 하직근이 관찰되어 좌안 하직근후전술 5.5 mm, 이측전이술 2 mm, 록우드인대와의 근막부착 조정, 결막후전술을 시행하였다. 술후 가성안검하수와상전부전은 호전되었으나 속발외사시 및 하전제한이 발생, 하사시의 잔류로 Knapp 근이동술을 외직근후전술 5.5 mm, 내직근절제술4.5 mm와 동시에 시행하고, 우안 상직근후전술 9.5 mm를 추가 시행하였다. 턱올림과 안검하수는 소실되었고, 18개월 후 제1안위에서좌안 외사시 6PD, 하사시 10PD로 호전되었으나 외전 시 상전제한은 잔류되었다. 결론: MED에서 견인검사상 강양성 소견은 동반된 선천 하직근 이상을 고려해 보아야 한다.
Abstract
Purpose: To report a congenital monocular elevation deficiency (MED) associated with inferior rectus anomaly. Case summary: A 3-year-old, otherwise healthy boy presented with left hypotropia. He showed chin-up and left head turn with left pseudoptosis. On examination, there was a left hypertropia of 60 prism diopters (PD) and esotropia in primary position. Upgaze -4 limitation in abduction and -3 from primary position, poor Bell’s phenomenon, and normal vertical saccadic velocity until midline were observed. On orbital CT, thin and nasally displaced left inferior rectus showing enlargement at its posterior segments near the orbital apex was observed. Forced duction test at surgery revealed a strong positive restriction and thin, taut, fibrotic inferior rectus was inserted and displaced nasally. Conclusions: Left inferior rectus recession of 5.5 mm with 2.0 mm of temporal transposition improved elevation deficiency,but consecutive exotropia and hypotropia remained. Knapp procedure combined with left lateral rectus recession of 5.5mm and left medial rectus resection of 4.5 mm, and right superior rectus recession of 9.5 mm were additionally performed. Pseudoptosis and chin elevation resolved but left hypotropia 10PD, and exotropia 6PD remained. Elevation restriction in primary position and abduction were improved but still persisted. This case indicated that MED may accompany congenital inferior rectus anomaly. If severe inferior rectus restriction is observed during the forced duction test, a structural anomaly of the inferior rectus causing motility defects of MED.
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