Rationale: Cervical cancer primarily spreads through direct invasion or via local lymphatics, and hematogenous metastasis is infrequent. Previous reports have shown that lung, liver, and bone are the organs most frequently affected by hematogenous metastasis of cervical cancer, while skeletal muscle is very rarely involved. Patient concerns: A 75-year-old Japanese woman presented with a painful muscular mass in her right lower abdomen. Five years ago, she was treated for her International Federation of Gynecology and Obstetrics stage IB2 cervical adenocarcinoma with radical surgery plus adjuvant chemotherapy. Diagnoses: The patient was diagnosed with isolated oblique muscle metastasis from cervical adenocarcinoma as a first site of recurrence. Interventions: The patient was treated with salvage surgery consisting of partial resection of the oblique muscle and ilium. The tumor was completely excised with an adequate surgical margin by a partial resection of the oblique muscle and ilium Outcomes: The patient is currently free of disease at 10 months after the development of recurrent disease. Lessons: We describe a rare case of isolated oblique muscle metastasis as a first site of recurrence of the International Federation of Gynecology and Obstetrics stage IB2 cervical adenocarcinoma, which was successfully treated with surgery. Although skeletal muscle metastasis is rare, this condition should be considered during the follow-up period, especially when patients complain of muscular pain with insidious progression. The present case and our literature review highlighted the possibility that loco-regional treatment may be curative for selected recurrent cervical cancer developed in skeletal muscles.