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舌骨巨细胞瘤:具有新颖治疗方法的诊断难题。

Giant cell tumor of hyoid bone: Diagnostic dilemma with a novel management.

机构信息

Department of ENT & Head & Neck Surgery, Medicare Hospital, Indore, Madhya Pradesh, India.

Department of Head and Neck Surgical Oncology, Tata Memorial Centre, Mumbai, Maharashtra, India.

出版信息

J Cancer Res Ther. 2022 Jan-Mar;18(1):282-285. doi: 10.4103/jcrt.JCRT_205_19.

DOI:10.4103/jcrt.JCRT_205_19
PMID:35381802
Abstract

Giant cell tumor of bone (GCTB) is locally aggressive tumor occurring in the epiphysis of long bones. GCTBs are uncommon tumors in the head-and-neck region and rarely involve hyoid bone. We report a case of GCTB of hyoid bone. The patient presented with swelling in left submandibular region. The tumor was surgically excised after initial denosumab therapy. Despite adequate resection and rehabilitation, he was tube dependent. Subsequently it was found that the patient had a coexisting myotonic dystrophy, unknown to exist with GCTB of hyoid. Eventually, the patient succumbed to respiratory failure secondary to myotonic dystrophy. GCTB hyoid is a rare presentation posing a diagnostic dilemma. Ours is the first case to report the use of denosumab for GCT in head-and-neck region. Myotonic dystrophy Type I and GCTB are both known to result from abnormality of closely situated foci on chromosome 19.

摘要

骨巨细胞瘤(GCTB)是一种局部侵袭性肿瘤,发生在长骨的骨骺。GCTB 在头颈部是罕见的肿瘤,很少累及舌骨。我们报告了一例舌骨骨巨细胞瘤。患者表现为左下颌区域肿胀。在初始地舒单抗治疗后,肿瘤被手术切除。尽管进行了充分的切除和康复,他仍需要依赖导管。随后发现患者同时患有肌强直性营养不良,这与舌骨骨巨细胞瘤并存,但此前未知。最终,患者因肌强直性营养不良继发呼吸衰竭而死亡。舌骨骨巨细胞瘤是一种罕见的表现,存在诊断难题。我们的病例是首例报告在头颈部使用地舒单抗治疗骨巨细胞瘤。肌强直性营养不良 I 型和骨巨细胞瘤都已知是由于染色体 19 上位置相近的焦点异常引起的。

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