J Korean Assoc Oral Maxillofac Surg.  2023 Apr;49(2):96-99. 10.5125/jkaoms.2023.49.2.96.

Oral spindle cell/sclerosing rhabdomyosarcoma on mandible with anaplastic lymphoma kinase expression mimicking inflammatory myofibroblastic tumor

• Lee JY
• Lee W
• Kim MY

• KMID: 2542216
• DOI: http://doi.org/10.5125/jkaoms.2023.49.2.96

Abstract

Oral spindle cell/sclerosing rhabdomyosarcoma (SCRMS) with anaplastic lymphoma kinase (ALK) expression is extremely rare, and its diagnosis is very challenging in the absence of clinical or pathological indicators. This case presented with gingival swelling and alveolar bone resorption and was suspected clinically to be periodontitis. A biopsy was performed and, due to immunoreactivity with ALK, the patient was misdiagnosed with inflammatory myofibroblastic tumor. However, based on the combined histological and immunohistochemical features, a revised diagnosis of SCRMS with ALK expression was finally concluded. We believe that this report makes a significant contribution to the precise diagnosis of this rare disease for proper treatment.

Keyword

Maxillofacial procedure; Pathology; Oral neoplasm

Figure

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  Fig. 1 Preoperative panoramic radiograph.

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  Fig. 2 A. Photomicrograph reveals proliferation of fascicular spindled tumor cells in the subepithelial portion (H&E staining, ×100). B. Higher magnification of tumor cells showing blunted, elongated nuclei and abundant eosinophilic cytoplasm with inconspicuous inflammation (H&E staining, ×200). C. Abundant inflammatory infiltrate associated with mucosal erosion (H&E staining, ×200). D. Tumor cells diffusely positive for desmin (immunohistochemistry staining, ×200). E. Tumor cells diffusely positive for myoD1 (immunohistochemistry staining, ×200). F. Tumor cells multifocally positive for anaplastic lymphoma kinase (immunohistochemistry staining, ×200).

Reference

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