A 59-year-old Japanese man presented with a giant submucosal tumor with ulceration during follow-up of duodenal ulcer. Pancreaticoduodenectomy was undertaken, and subsequent histological examination of the tumor disclosed anaplastic carcinoma of the pancreas head. The carcinoma components contained adenocarcinoma and squamous cell carcinoma. Additionally, undifferentiated spindle or pleomorphic cells were seen in continuity with the carcinoma component. Undifferentiated neoplastic cells with rhabdoid features and with hyaline globule-like structures positive for PAS stain with diastase pretreatment were also observed. Immunohistochemically, cytoplasmic inclusions corresponding to rhabdoid features showed aggregates of vimentin. Ultrastructurally, hyaline globule-like structures corresponded to lysosomes. Finally, we report here the first case of anaplastic carcinoma of the pancreas with hyaline globule-like structures.