Uesugi Atsushi, Tsushima Fumihiko, Kodama Makoto, Kuroshima Takeshi, Sakurai Jinkyo, Harada Hiroyuki
Oral and Maxillofacial Surgery, Tokyo Medical and Dental University, Tokyo 113-8549, Japan.
Department of Oral Health Sciences, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo 1138510, Japan.
World J Clin Cases. 2020 Nov 26;8(22):5663-5669. doi: 10.12998/wjcc.v8.i22.5663.
Oral mucositis is often observed with graft-versus-host disease (GVHD); however, the occurrence of oral granuloma is rare. The rapid increase in granulomatous lesions should be distinguished from malignant tumors in patients with GVHD because malignant diseases can develop in those patients. This case is the youngest pediatric patient with granuloma associated with GVHD.
The patient was a 1-year and 5-mo-old girl who presented to our department for the management of oral nodules. At the age of 5 mo, she was diagnosed with primary immunodeficiency disease, cord blood transplant was performed at 11 mo and bone marrow transplant at 1 year of age. After transplantation, GVHD and oral mucositis developed, and tacrolimus was administered. Interestingly, nodules appeared on the lower lip and buccal mucosa, which spontaneously disappeared. Then, a new nodule appeared on the left lateral border of the tongue. Resection was performed and the histopathological diagnosis was granuloma. The origin of these nodules were considered to be the fibroblasts activated under inflammation caused by GVHD because the calcineurin inhibitor tacrolimus acted on their proliferation.
It is very important to distinguish oral granulomatous lesions from malignancies if GVHD is present at the base and if immunosuppressive agents and steroids are being administered.
口腔黏膜炎常与移植物抗宿主病（GVHD）同时出现；然而，口腔肉芽肿的发生较为罕见。在GVHD患者中，肉芽肿性病变的迅速增加应与恶性肿瘤相鉴别，因为这些患者可能会发生恶性疾病。该病例是最年轻的与GVHD相关的肉芽肿儿科患者。
患者为一名1岁5个月大的女孩，因口腔结节前来我科就诊。5个月大时，她被诊断出患有原发性免疫缺陷病，11个月时进行了脐血移植，1岁时进行了骨髓移植。移植后，发生了GVHD和口腔黏膜炎，并给予了他克莫司治疗。有趣的是，下唇和颊黏膜出现结节，这些结节自行消失。随后，舌左侧缘出现了一个新的结节。进行了切除，组织病理学诊断为肉芽肿。这些结节的起源被认为是由GVHD引起的炎症激活的成纤维细胞，因为钙调神经磷酸酶抑制剂他克莫司作用于它们的增殖。
如果存在GVHD且正在使用免疫抑制剂和类固醇，将口腔肉芽肿性病变与恶性肿瘤区分开来非常重要。
