PDF(Pubmed)
Abstract：
Xanthogranuloma is a benign histiocytic disorder that generally appears in infants and children and often called juvenile xanthogranuloma (JXG). Typical reddish-yellow cutaneous papules or nodules are the most common presentation of JXG. Extracutaneous JXG affects eyes, brain, lungs, liver, spleen, and other sites. Isolated ocular manifestation without skin lesion is rare, especially in adult patients. Here, we report a case of a 27-year-old man who presented with gradually growing yellowish mass at the corneoscleral area of the left eye for 5 months. The patient had worn soft contact lenses for more than 10 years. With atypical age of onset and the absence of skin lesion, total mass excision with lamellar corneoscleral graft and amniotic membrane transplantation was done, and the diagnosis of adult-onset limbal xanthogranuloma was made by histopathological and immunohistochemical examinations. Postoperatively, the patient had good vision with corrected distant visual acuity of 20/30, and the graft was clear. There was no evidence of recurrence at 4-year follow-up. We found that excision with lamellar corneoscleral graft in limbal xanthogranuloma shows good result with no recurrence. The same result occurred to other previous cases reported, so complete excision with graft could be an effective treatment of choice in patient with limbal xanthogranuloma.
摘要：
黄色肉芽肿是一种良性组织细胞性疾病，通常出现在婴儿和儿童中，通常称为青少年黄色肉芽肿（JXG）。典型的红黄色皮肤丘疹或结节是JXG的最常见表现。皮肤外JXG影响眼睛，大脑,肺,肝脏,脾,脾和其他网站。孤立的眼部表现没有皮肤损伤是罕见的，尤其是成人患者。这里,我们报告了一例27岁的男性，他在左眼的角膜巩膜区域逐渐变黄，持续5个月。患者佩戴软性隐形眼镜超过10年。发病年龄不典型，没有皮肤病变，全块切除联合板层角膜巩膜移植和羊膜移植，通过组织病理学和免疫组织化学检查诊断为成人发病的角膜缘黄色肉芽肿。术后,患者视力良好，远距矫正视力为20/30，移植物清晰。在4年的随访中没有复发的证据。我们发现角膜缘黄色肉芽肿的板层角膜巩膜移植物切除效果良好，无复发。以前报告的其他病例也出现了同样的结果，因此，对于角膜缘黄色肉芽肿患者，完全切除与移植物可能是一种有效的治疗方法。
