Intralesional injection of OK-432 for vision-threatening orbital lymphangioma.
BACKGROUND
Surgical excision of orbital lymphangiomas is difficult, and almost always incomplete due to the diffusely infiltrative pattern of these tumors. The present report describes the successful use of intralesional OK-432 administration to treat two patients with intractable hemorrhagic proptosis due to orbital lymphangiomas.


METHODS
A 3-year-old girl (case 1) presented with aggressive proptosis and ptosis, and a 1-year-old boy (case 2) presented with massive proptosis and exposure keratopathy, associated with recurrent intracystic bleeding of an orbital lymphangioma. In case 1, 0.02 mg OK-432 was intracystically injected in a volume of 2 ml of physiologic saline. Due to a lack of therapeutic response, a second injection of 0.05 mg OK-432 in 1 ml was administered. In case 2, a single dose of 0.05 mg OK-432 in 1 ml was administered.


RESULTS
In both cases, intracystic administration of 0.05 mg of OK-432 in a 1-ml volume resulted in a successful outcome. The adverse effects were minor (mild transient fever and lid swelling), and rebleeding and intraocular pressure elevation did not occur. Proptosis and eyelid swelling gradually improved over 1 month, and completely resolved within 3 months of treatment.


CONCLUSIONS
Intralesional administration of 0.05 mg/ml OK-432 (delivered in 1 ml) resulted in the successful treatment of two cases of orbital lymphangioma. Although this drug concentration is higher than in previous reports, there were no major adverse effects.
Yoon
Jin Sook
JS
Department of Ophthalmology, Yonsei Institute of Vision Research, Yonsei University College of Medicine, Seoul, South Korea.
Choi
Jung Bum
JB
Kim
Sung Joo
SJ
Lee
Sang Yeul
SY
D000970:Antineoplastic Agents / Q000008:administration & dosage*; D002675:Child, Preschool; D005260:Female; D006801:Humans; D007223:Infant; D015552:Injections, Intralesional; D008202:Lymphangioma / Q000188:drug therapy* / Q000473:pathology; D008279:Magnetic Resonance Imaging; D008297:Male; D009918:Orbital Neoplasms / Q000188:drug therapy* / Q000473:pathology; D010844:Picibanil / Q000008:administration & dosage*; D019233:Retreatment; D014057:Tomography, X-Ray Computed
D000970:Antineoplastic Agents; D010844:Picibanil