Abstract
Background: The incidence of skeletal muscle metastasis from oesophageal cancer is very low, and the treatment strategy has not been established. Case Report: A 77-year-old man underwent oesophagectomy following neoadjuvant chemotherapy for oesophageal squamous cell carcinoma (CT-pT3 N0 M0, CT-pStage II). Fourteen months after surgery, he became aware of a subcutaneous tumour in his left forearm. Computed tomography and fluorodeoxyglucose positron-emission tomography revealed a 65×75 mm intramuscular nodular lesion with a standardized uptake value of 8.5. Further examination by biopsy strongly suggested this was a solitary metastasis from oesophageal cancer. The patient received chemoradiotherapy with two cycles of 5-fluorouracil combined with cisplatin and radiation. Clinical complete response was confirmed by imaging 7 months after chemoradiation and no recurrence has occurred at 20 months since chemoradiation. Conclusion: Radiotherapy or chemoradiotherapy can be an alternative locoregional therapy to surgery for solitary skeletal muscle metastasis.
Oesophageal cancer is considered a serious malignancy with regard to mortality and prognosis. Oesophageal cancer is notorious for spreading by a variety of pathways including direct invasion, lymphatic spread, and hematogenous metastasis. The main organs to which oesophageal cancer metastasizes are the liver, lungs, bones, adrenal glands, kidney and brain, in order of decreasing frequency (1). Skeletal muscle is a rare organ for metastasis of oesophageal cancer, and only a few cases have been reported to date.
Here, we report a case of skeletal muscle metastasis after oesophagectomy for oesophageal cancer that was successfully treated with chemoradiotherapy. We also review 15 similar cases reported in the literature.
Case Report
A 77-year-old man was referred to our hospital for the treatment of lower thoracic oesophageal cancer diagnosed as stage III oesophageal squamous cell carcinoma (T3N0M0) according to the 11th edition of the Japanese Classification of Oesophageal Cancer (2). Following neoadjuvant chemotherapy with two courses of a 5-fluorouracil and cisplatin regimen, subtotal oesophagectomy with two-field lymphadenectomy and reconstruction using a gastric tube through the retrosternal route was performed. The pathological diagnosis of the tumour was stage II (T3 N0 M0) oesophageal squamous cell carcinoma. The patient refused adjuvant chemotherapy but did undergo follow-up examinations every 3 months.
Fourteen months after oesophagectomy, the patient became aware of a subcutaneous tumour in his left forearm. On physical examination, a hard painless subcutaneous nodule was palpated in the left forearm and computed tomography showed a 65×75 mm intramuscular nodular lesion. The patient underwent magnetic resonance imaging (MRI) and T2-weighted MRI of the left upper extremity revealed an intramuscular nodular lesion with high signal intensity located within the extensor digitorum communis muscle, long thumb abductor muscle, and ulnar extensor group (Figure 1a). Whole-body 18F-fluorodeoxyglucose positron-emission tomography demonstrated significant accumulation of 18F-fluorodeoxyglucose in the tumour of the left forearm with standardized uptake value of 8.5 (Figure 1b). The patient underwent biopsy of the subcutaneous nodular lesion. The histopathological findings of the lesion revealed squamous cell carcinoma, which was similar to the histology of the oesophageal cancer resected 14 months previously (Figure 2a and b). The carcinoma cells were positive for p63 and cytokeratin 903, and negative for cytokeratin 7 and cytokeratin 2 (Figure 2c-f). Based on the histological similarity between the biopsy specimen and the resected oesophageal cancer, we finally diagnosed the tumour as solitary skeletal muscle metastasis from oesophageal cancer.
a: T2-Weighted magnetic resonance image of the left upper extremity revealed an intramuscular nodular lesion with high signal intensity. b: Whole-body 18F-fluorodeoxyglucose positron-emission tomography demonstrated significant accumulation of 18F-fluorodeoxyglucose in the tumour of the left forearm with standardized uptake value of 8.5.
Literature review of cases of skeletal muscle metastasis of oesophageal carcinoma.
Histopathological findings of the lesion revealed squamous cell carcinoma that was similar to the oesophageal cancer resected 14 months earlier. Pathological examination of the resected oesophageal cancer revealed squamous cell carcinoma (a and b: Hematoxylin-eosin stain). The carcinoma cells of the lesion were positive for p63 (c) and cytokeratin 903 (d), and negative for cytokeratin 7 (e) and cytokeratin 2 (f) (x 400 magnification).
As the recurrent lesion was confined only to the left forearm, we considered surgery or chemoradiation as locoregional therapy. Because of concerns about the possibility of postoperative dysfunction of motor function of the left forearm and left fingers, the patient refused surgery and received chemoradiotherapy: two cycles of 5-fluorouracil (700 mg/m2 on days 1 to 5) combined with cisplatin (70 mg/m2 on day 1) and radiation (total 70 Gy/35 fr) were administered. Clinical complete response was confirmed by MRI 7 months after chemoradiation. The patient has had no recurrence at 20 months since the chemoradiation therapy.
Discussion
Metastatic and recurrent oesophageal cancer is highly aggressive and, until recently, has almost always been associated with a dismal prognosis. The treatment and management of recurrent oesophageal cancer have evolved in recent years,with dramatic advances in multimodal therapy involving surgery with chemotherapy with/without radiation, but a therapeutic strategy for recurrence has not been established. Weinberg et al. reviewed 1,588 patients with oesophageal cancer and reported metastasis from oesophageal cancer as being to the lymph nodes in 830 patients (52%), the liver in 351 patients (22%), the lungs in 245 patients (15%), and bone or soft tissue in 138 patients (9%) (3). Skeletal muscle is a rare site for metastasis of malignant tumours, and the incidence of skeletal muscle metastasis is reported to be less than 1% among patients with all types of malignant tumours (1). The most common primary tumour sites were the lungs (35%), gastrointestinal system (23%), and the kidneys (19%) (4).
Our literature search revealed 15 reported cases of treated oesophageal cancer with metastasis to skeletal muscle (Table I) (5-17). The mean patient age was 62.1 (range=45 to 77) years and male patients were dominant (87%). The histology of oesophageal cancer was squamous cell carcinoma in 10 patients (67%) and adenocarcinoma in five (33%). The muscles to which the cancer metastasized were most commonly in the trunk (seven patients, 47%), followed by those of the lower limbs (five patients, 33%) and upper limbs (three patients, 20%). Recurrence was confined to skeletal muscle in only 5 out of the 15 patients; the other cases were accompanied by systemic metastases in multiple organs. The patients with solitary metastasis confined to skeletal muscle tended to be treated with surgery or radiotherapy, and those with systemic metastases tended to be treated with chemotherapy. Regardless of the spread of metastasis and differences in treatment modality, patients with skeletal muscle metastasis had an extremely poor prognosis.
We successfully performed chemoradiotherapy for solitary skeletal muscle metastasis after oesophagectomy for oesophageal cancer, and our patient has had the best outcome compared with cases reported in the literature. The patient is still alive with no recurrence during the 20-month period since the chemoradiation. However, further careful follow-up will be needed to ascertain the validity of this treatment. Considering the extremely poor outcomes of patients with skeletal muscle metastasis, surgical treatment that might be accompanied by loss of function should be avoided for these cases. In order to maintain the quality of life, radiotherapy or radiochemotherapy can be considered as an alternative locoregional therapy to surgery.
Conclusion
Skeletal muscle metastasis from oesophageal cancer is very rare, and the treatment strategy has not been established. Most cases of skeletal muscle metastasis from oesophageal cancer spread to multiple organs, and the prognosis is very poor, even in cases with solitary metastasis. Considering the poor treatment outcomes and the risk of loss of function, chemoradiotherapy is considered the best treatment modality for these patients.
Acknowledgements
The Authors thank Mary Derry, Ph.D. ELS, from Edanz Group (www.edanzediting.com/ac) for editing a draft of this article.
Footnotes
Conflicts of Interest
The Authors have no conflicts of interest.
- Received August 6, 2017.
- Revision received August 24, 2017.
- Accepted August 29, 2017.
- Copyright© 2017, International Institute of Anticancer Research (Dr. George J. Delinasios), All rights reserved
