Combination Treatment of Surgery and Sirolimus in Lingual Lymphangioma -- A Case Report

使用斥消靈錠和手術合併治療舌部海綿狀淋巴管瘤──病例報告

Chi Lo(羅琪)；Chang-Cheng Chang(張長正)；Jian-Jr Lee(李建智)

lymphangioma ； sirolimus ； surgical treatment ； lingual lymphangioma

臺灣整形外科醫學會雜誌

33卷2期（2024 / 06 / 01）

232 - 242

英文;繁體中文

Background: Lymphangioma (LM) is an uncommon, benign vascular disorder, usually presents at birth and diagnosed in infancy or early childhood. It has spectrum of clinical manifestations and is often found in head and neck region. When LM occurs at tongue, life-threatening situation such as airway obstruction may happen. Aim and Objectives: Our objective is to treat lingual lymphangioma with combination therapy of sirolimus and surgical intervention without or with limited complications. Case presentation: A 23-year-old female had presented with an asymptomatic lingual tumor since childhood. At the age of nineteen, the tumor exhibited noticeable enlargement and MRI imaging revealed the presence of two lingual masses which suspected to be lymphangiomas. Following eleven months of sirolimus treatment, surgical intervention was undertaken to excise the lingual masses. Results: In the end of sirolimus treatment, both anterior and posterior tumors exhibited reduction of volume comparing to the initial image. However, the patient also experienced adverse effect of severe oral ulcers and acnes. Surgical intervention was undertaken to excise the shrunken lesions and the patient made a successful recovery and maintained full tongue functionality. Conclusion: In cases where patients exhibit a limited response to monotherapy, a combination approach with pharmaceutical agents and surgical intervention may be a more effective treatment strategy. More discussions are needed for adjusting the optimal medicine concentration and the timing of surgical intervention based on the location of the tumor and the drug reactivity.

背景：淋巴管瘤是一種為相對罕見的良性腫瘤，通常在嬰兒或幼兒時期被診斷。它具有多種臨床表現並時常出現在頭頸部。當淋巴管瘤發生在舌頭上時，可能會發生危及生命的狀況，如氣道阻塞，對於臨床醫師而言，治療淋巴管瘤是一具有挑戰的領域。目的及目標：結合斥消靈錠的藥物治療及手術切除以治療舌部淋巴管瘤並盡量降低併發症的產生。材料及方法：一位二十三歲女性自幼兒時期被診斷有無症狀的舌部腫瘤。十九歲開始腫瘤明顯增大並部分影響說話功能。核磁共振影像可看到兩處疑似為淋巴管瘤的舌腫瘤。在經過十一個月的斥消靈錠治療後，以手術切除縮小之舌部腫瘤。結果：在斥消靈錠治療下，前後兩處的舌腫瘤體積均比初始體積縮小。然而患者在治療過程中出現了嚴重的口腔潰瘍和痤瘡的藥物副作用。在藥物效果有限且腫瘤相對縮小的狀況下，後續以手術盡量切除兩處病灶，患者恢復良好並完整保持舌頭功能。結論：在淋巴管瘤患者對單一治療反應有限的狀況下，結合藥物和手術的治療方式可能是更有效的治療策略。根據腫瘤的部位和對藥物的反應，我們需要按照每位病患的狀況來調整最佳藥物濃度及手術介入的時機。