|Ahead of print publication
Solitary humerus metastasis in a young endometrial cancer patient
SV S. Bala, Deepika Malik, Rajeev K Seam
Department of Radiation Oncology, Maharashi Markandeshwar Institute of Medical Sciences and Research, Mullana, Ambala, Haryana, India
|Date of Submission||09-Dec-2020|
|Date of Decision||10-Sep-2021|
|Date of Acceptance||24-Nov-2021|
|Date of Web Publication||15-Jun-2022|
Department of Radiation Oncology, Maharishi Markandeshwar Institute of Medical Sciences and Research, Mullana, Ambala - 133 207, Haryana
Source of Support: None, Conflict of Interest: None
Solitary bone metastasis in endometrial cancer is very rare. We report a young 29-year-old nulliparous female of endometrial cancer who developed solitary humerus metastasis after 8 months of primary treatment of surgery and adjuvant radiotherapy and chemotherapy. She was treated with local radiotherapy and combination chemotherapy and bisphosphonates. At 6 months follow-up the patient is doing well and is asymptomatic. Even though rare, keeping a high index of suspicion and sincere evaluation in patients on follow-up presenting with bone pains can detect early recurrences. Timely start of multimodality treatment helps relieve symptoms and improves quality of life.
Keywords: Humerus metastasis, metastatic endometrial cancer, solitary bone metastasis
| > Introduction|| |
Endometrial cancer is the second most common genital tract malignancy in women. Majority of the cases are diagnosed in Stage I as postmenopausal bleeding the commonest symptom prompts women to take early medical advice leading to an early diagnosis and treatment. Evaluation includes gynecological and ultrasonographical examination followed by endometrial biopsy. Computed tomography (CT) scan and/or magnetic resonance imaging is usually advocated as a part of preoperative evaluation. Treatment includes a Total hysterectomy with bilateral salpingo-oophorectomy with pelvic and/or para-aortic lymphadenectomy or sampling followed by radiotherapy, chemotherapy, and hormonal therapy depending upon pathological stage and grade.
Recurrence after primary treatment is mostly locoregional i.e., pelvic nodes. Distant metastasis is rare and the mostly occurs in nonregional lymph nodes, omentum, lungs, and liver. Bone metastasis is reported to occur in <1% of all metastatic endometrial cancer, spine, and hip being the most common sites. Peripheral skeleton is rarely involved. Few authors have reported metastasis to tibia, calcaneum, and metatarsal bone.
| > Case Report|| |
A 29-year-old young nulliparous married female reported as a proven case of Endometrial Cancer in January 2019 to our center. She had undergone Radical Hysterectomy with bilateral salpingo-oophorectomy and was staged as FIGO Stage IIIC1 (Pelvic nodes + Peritoneal deposits in pouch of Douglas) with Grade III. Immunohistochemistry on the surgical specimen showed it to be estrogen receptor positive and progesterone receptor positive. She gave a history of treatment for infertility for more than 5 years. After all the baseline investigations and metastatic workup including CA-125, she was treated with sequential combination chemotherapy (Paclitaxel 175 mg/m2 Day 1+ Carboplatin AUC 5 Day 1 for 6 cycles every 21 days) with sandwiched 3-D conformal external beam radiotherapy to the whole pelvis 50 Gray/25 fractions/5wks. The patient completed the planned treatment in July 2019. CT Abdomen and Pelvis on first follow-up in October 2019 showed no evidence of residual disease and she was kept on follow up.
After 8 months of uneventful follow-up, in March 2020 she presented with complaints of pain in left shoulder pain. X-ray [Figure 1] revealed Left upper-end metaphyseal humerus metastasis with large soft tissue mass and pathological fracture. To rule out disseminated metastases positron emission tomography CT [Figure 2] was done which revealed fluorodeoxyglucose avid uptake only in the proximal end of humerus. She was diagnosed as solitary humerus metastasis with fracture. Orthopedic consultation was taken and her left upper arm was immobilized. Palliative radiotherapy 30 Gray/10 fractions/2 weeks at 3 Gray per fraction was given to the bony lesion in the left humerus which was followed by combination chemotherapy (paclitaxel 175 mg/m2 Day 1, cisplatin 50 mg/m2 Day 2, doxorubicin 45 mg/m2 Day 2) every 21 days for 6 cycles along with bisphosphonate-zoledronic acid. The patient tolerated all chemotherapy cycles well and was kept on follow-up after that with continuation of zoledronic acid every 28 days. The patient is doing well with relief in pain and no restriction of movement. We report this case at 6-month follow-up, the patient is symptom free. Hormonal therapy has been kept in reserve for any future recurrences or metastases.
|Figure 1: Plain radiograph left shoulder anteroposterior view showing left upper end metaphyseal humerus metastasis with large soft tissue component and pathological fracture|
Click here to view
|Figure 2: Positron emission tomography computed tomography whole body showing fluorodeoxyglucose avid uptake in proximal end left humerus. No other areas of increased fluorodeoxyglucose uptake|
Click here to view
| > Discussion|| |
Endometrial cancer is the sixth most common cancer in females worldwide with 3,82,069 cases, age-standardized risk of 8.4 per 1,00,000 women, and cumulative risk of 1.01. In India, the projected incidence of endometrial cancer is 26,514 with a crude rate of 3.9 and a cumulative risk of 1 in 190. The rise in endometrial cancer in India is mainly attributed to changing trends in the lifestyle and reproductive profile of women. The mean age at diagnosis is 60 years with majority of cases presenting in the sixth and seventh decades of life. Although it is a cancer that affects predominantly postmenopausal women, 5%–30% of women are <50 years of age at the time of diagnosis. Nulliparity and infertility, like in our patient, is associated with increased risk of endometrial cancer due in part to anovulatory menstrual cycles and may be the use of long-term hormonal agents for the treatment of infertility. Cancer Endometrium is diagnosed in an early stage in 70% of the patients with 5-year overall survival of 90%. It recurs mostly in the pelvic nodes. Distant metastases are rare and seen in nonregional lymph nodes, lung, or liver. Bone metastasis is even rarer, the prevalence is reported as 0%–15%. In a retrospective analysis of 1632 Endometrial cancer patients, the authors concluded the incidence to be <1%, the most common sites being spine and hip.
This is a report of solitary humerus metastasis in a young treated patient of FIGO Stage IIIC Grade III endometrioid adenocarcinoma. Literature search revealed only 2 such cases to have been reported by Devdas et al. and Schöls et al., Dursun et al. also reported humerus metastasis from endometrial cancer but was associated with extraosseous spread to axillary nodules.
The treatment for bone metastasis in endometrial cancer should be individualized. Solitary metastasis has a good prognosis and aggressive treatment with a multimodality approach should be followed. Surgical resection of the accessible site with acceptable functional status, radiotherapy, and systemic chemotherapy is the management approaches mentioned in literature. The treatment should be based on patient performance status, site, and number of metastasis. The optimal elective treatment for bone metastasis in endometrial cancer is unknown probably due to a small number of cases described and a variety of bony sites involved.
Our patient was managed by local radiotherapy with immobilization of the arm for fracture along with systemic therapy with combination chemotherapy and bisphosphonates and it achieved good symptom control and acceptable quality of life.
Further studies should be aimed to predict survival and designing treatment protocol for such patients with rare presentation.
| > Conclusion|| |
Solitary humerus metastasis in endometrial cancer is a rare entity and this is the first such case to be reported in a young female. Although bone metastasis in endometrial cancer is rare, a high index of suspicion and sincere evaluation of bone pains helps in early diagnosis and treatment. Timely start of multimodality management with chemoradiation and bisphosphonates provides good symptom control and improves the quality of life.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient (s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initial s will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| > References|| |
Uccella S, Morris JM, Bakkum-Gamez JN, Keeney GL, Podratz KC, Mariani A. Bone metastases in endometrial cancer: Report on 19 patients and review of the medical literature. Gynecol Oncol 2013;130:474-82.
Bray F, Ferlay J, Soerjomataram I, Siegel RL, Torre LA, Jemal A. Global cancer statistics 2018: GLOBOCAN estimates of incidence and mortality worldwide for 36 cancers in 185 countries. CA Cancer J Clin 2018;68:394-424.
Mathur P, Sathishkumar K, Chaturvedi M, Das P, Sudarshan KL, Santhappan S, et al.
Cancer statistics, 2020: Report From National Cancer Registry Programme, India. JCO Glob Oncol 2020;6:1063-75.
McMeekin DS, Yashar C, Campos SM, Zaino RJ. Corpus: Epithelial tumors. In: Barakat RR, Berchuk A, Markman M, Randall M, editors. Principles and Practice of Gynecologic Oncology. 6th
ed. Philadelphia: Lippincott Williams & Wilkins; 2013. p. 661-714.
Zucchetto A, Serraino D, Polesel J, Negri E, De Paoli A, Dal Maso L, et al.
Hormone-related factors and gynecological conditions in relation to endometrial cancer risk. Eur J Cancer Prev 2009;18:316-21.
Creasman WT, Odicino F, Maisonneuve P, Quinn MA, Beller U, Benedet JL, et al.
Carcinoma of the corpus uteri. FIGO 26th
Annual Report on the Results of Treatment in Gynecological Cancer. Int J Gynaecol Obstet 2006;95 Suppl 1:S105-43.
Fujimoto T, Nanjyo H, Fukuda J, Nakamura A, Mizunuma H, Yaegashi N, et al.
Endometrioid uterine cancer: Histopathological risk factors of local and distant recurrence. Gynecol Oncol 2009;112:342-7.
Devdas SK, Digumarti L, Digumarti R, Patro KC, Nutakki R. Isolated humeral recurrence in endometrial carcinoma. Indian J Med Paediatr Oncol 2016;37:199-201.
] [Full text]
Schöls WA, Kock HC, van Etten FH. Recurrent endometrial adenocarcinoma presenting as a solitary humeral metastasis. Gynecol Oncol 1995;59:148-50.
Dursun P, Gültekin M, Basaran M, Aydingöz U, Ayhan A. Bilateral bone metastasis in endometrial adenocarcinoma. Lancet Oncol 2003;4:547.
[Figure 1], [Figure 2]