|Year : 2015 | Volume
| Issue : 1 | Page : 71-75
Bone metastasis in the tibia-fibula as a presenting sign of endometrial adenocarcinoma: A case report and review of the literature
Dhiraj Daga1, Rohitashwa Dana2
1 Department of Radiotherapy, RUHS College of Medical Sciences, Rukmani Devi Beni Prasad Jaipuria Hospital, Jaipur, India
2 Department of Radiotherapy, Dr. Sampurnanand Medical College, Mathura Das Mathur Hospital, Jodhpur, Rajasthan, India
|Date of Web Publication||13-Feb-2015|
7.M.13, R.C.Vyas Colony, Bhilwara - 311 001, Rajasthan
Source of Support: None, Conflict of Interest: None
Metastasis to the bone in endometrial carcinoma is very rare with a reported frequency of 0-8%. Metastasis to the extremities in endometrial carcinoma is an extremely rare entity, as well as presenting with it makes it rarest. We report here, the case of isolated metastasis in the tibia-fibula as a presenting feature of primary endometrial adenocarcinoma in a 75-year-old multiparous woman.
Keywords: Bone, endometrial carcinoma, metastasis
|How to cite this article:|
Daga D, Dana R. Bone metastasis in the tibia-fibula as a presenting sign of endometrial adenocarcinoma: A case report and review of the literature. Saudi J Health Sci 2015;4:71-5
|How to cite this URL:|
Daga D, Dana R. Bone metastasis in the tibia-fibula as a presenting sign of endometrial adenocarcinoma: A case report and review of the literature. Saudi J Health Sci [serial online] 2015 [cited 2022 Jan 22];4:71-5. Available from: https://www.saudijhealthsci.org/text.asp?2015/4/1/71/151413
| Introduction|| |
Endometrial carcinoma (EC) is one of the most common gynaecological cancer of female genital tract, is the sixth most common malignancy worldwide, most common in Western countries.  Metastasis of the endometrial adenocarcinoma in descending order is to the lymph nodes, lung and liver. Metastasis to the bone is very rare with a reported frequency of 0-8%. It usually metastases to the axial skeleton, metastasis to the extremities is extremely rare.  In the present study, we report the case of isolated metastasis in the tibia-fibula as a presenting feature of primary endometrial adenocarcinoma, which is a rare presentation and in clinical practice we have to see beyond the routine sites to reach the diagnosis.
| Case report|| |
A 75-year-old multiparous woman presented in orthopaedic department, complaining of pain and swelling in right lower leg, not controlled with analgesics, with no other complains. Physical examination did not reveal any abnormality, other than swelling in the right lower shaft of tibia. A radiograph of the lower shaft showed an osteolytic lesion in the distal shaft of tibia and fibula [Figure 1]. FNAC of the swelling suggested metastatic adenocarcinoma [Figure 2]. The haematological and biochemical profiles were normal. Chest X-ray did not reveal any abnormality. Ultrasonography of the whole abdomen showed uterus of size 10 × 6 × 5 cm, with large mass of size 5 × 5 × 5 cm in the uterine cavity, and computed tomography revealed an enlarged uterus measuring 98 × 68 × 50 mm with large homogeneously enhancing lesion measuring 55 × 50 × 58 mm along the uterine cavity, with an enlarged right external illac node of size 34 × 33 × 33 mm [Figure 3] and [Figure 4], prompted a gynaecological consultation. She did not had any gynaecological complains, CA-125 was found to be normal as well. She had no history of any carcinoma in her family. Biopsy of the mass revealed moderately differentiated endometroid adenocarcinoma [Figure 5]. A bone scan showed increased uptake in the lower cervical vertebrae and lower half of shaft of right tibia [Figure 6]. Palliative radiotherapy had been given to the right lower leg. Patient was put on monthly zoledronic acid and on daily medroxyprogestrone. Barther index  improved from 50 to 80 in 3 months and 85 by the end of 6 months, LASA Score  also improved from 20 to 32 and 36 by the end of 3 and 6 months, respectively, suggesting a good improvement in quality of life of the patient. She is continuing on all these with good pain relief and quality of life.
|Figure 1: Radiograph of the lower shaft showed an osteolytic lesion in the distal shaft of tibia and fibula|
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|Figure 2: FNAC of the bone lesions shows haemorrhagic smears with presence of atypical cells with ovoid to elongated hyperchromatic nuclei entrapped in fibrillary and hyalinestroma|
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|Figure 3: Transverse section of CT scan showing enlarged uterus with enhancing lesion|
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|Figure 4: Coronal section of CT scan showing enlarged uterus with enhancing lesion|
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|Figure 5: Biopsy shows moderately differentiate dendometroidadenocarcinoma with similar columnar cells having elongated nuclei|
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| Discussion|| |
Endometrial cancer is the most frequent malignancy of the female genital tract in the Western world and is diagnosed predominantly at stage I.  EC most commonly spreads by direct extension to adjacent tissue or lymphatic dissemination. The incidence of stage IV disease is approximately 5-10% with a 5-year overall survival of 0-10%. 
Metastatic disease to the bone is most commonly from lung, prostate, kidney and breast cancers  Distant metastases in EC are less common and usually involve lung and liver and infrequently to the bones. Mariani et al. in a cohort of 612 endometrial cancer patients identified a 7.5% incidence of lung and 1.9% of liver metastasis. Bone metastasis at the time of diagnosis is extremely rare.  We systematically searched Medline (1950- present) for published data. To ensure completeness, we cross-referenced our search results and hand searched for additional titles. We excluded patients who had already been diagnosed and treated for endometrial cancer and presented with metastatic recurrent disease. Our search revealed 36 cases reported either as case reports or included in small cases series [Table 1]. In all cases, diagnosis was made due to osseous symptoms followed by bone biopsy and extensive workup to identify the primary site. Gynaecological symptoms may have occurred at a later time. Some cases had been managed as ''unknown primary''. Usually, bone metastases in EC are observed as disease relapses during the course of follow-up after the initial treatment. The vertebrae were the most common metastatic sites.  Furthermore, bone metastases involving only the lower limbs are extremely rare.  Bone involvement in endometrial cancer has been described in less than 15% of the patients with metastatic disease. Bone metastatic disease is often associated with a poor prognosis. Median survival time after bone metastases associated with endometrial cancer is about 16 months. 
|Table 1: Case reports of endometrial cancer presenting with bone metastasis|
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Recently, Uccella et al. have published report on bone metastasis in endometrial carcinoma treated at Mayo clinic between 1984 and 2001.  Of the 1632 patients with endometrial cancer during this period, 19 had bone metastasis at single or multiple sites, suggesting an overall incidence of less than 1%. Three (15.8%) of these 19 had bone metastases at presentation, which is uncommon, representing only 0.12% of all patients. Nine of them had with single bone involvement and no extraosseous spread. The most common sites were the spine and hip; only two of them had metastasis to tibia. Kehoe et al. published a clinical series in 2010 on bone metastases of EC diagnosed between 1990 and 2007 at Memorial Sloan-Kettering Cancer Center.  They reported 21 cases of osseous dissemination of EC, the median age of diagnosis of primary endometrial cancer was 60 years. Six patients (29%) had a bone metastasis at the time of diagnosis while 15 patients (71%) had a bone metastasis after primary treatment. Fourteen patients (67%) had FIGO stage III/IV disease. The overall survival of those patients with bone metastases at primary diagnosis was 17 months (95% CI: 2-32) compared to 32 months (95% CI: 14-49) for those with a recurrent bone metastasis. The spine was again the most commonly involved site, followed by the pelvic bones. Retrospective study done by Yoon et al. in 2013  for the patients reported between October 1994 and May 2012 at Samsung Medical Centre, Korea having endometrial carcinoma. Of the 1185 patients with endometrial carcinoma, 22 (1.8%) were identified with bone metastasis, and 21 patients were analyzed in the study. Four (19.0%) patients had a bone lesion at the diagnosis of endometrial cancer. The median overall survival (OS) and survival after bone metastasis of the entire cohort were 33 months (range, 9-57 months) and 15 months (range, 12-17 months), respectively. The patients with bone metastasis at recurrence had significantly longer OS than those patients with bone metastasis at diagnosis of endometrial cancer (36 vs. 13 months). Metastasis to extrapelvic bone was significantly associated with longer OS (46 vs. 19 months) and longer survival after bone metastasis (25 vs. 12 months). The patient reported in our literature has primary presentation of an endometrial carcinoma as bony metastasis to tibia-fibula which is a rare presentation in terms of primary presentation as well as site of metastasis.
The optimal elective treatment for bone metastasis of EC is unknown. This uncertainty is probably the result not only of the small number of cases described in the medical literature but also of the different osseous sites involved. Moreover, identifying the extraosseous sites of dissemination is crucial in determining possible therapeutic strategies. As per the report published by Uccella et al., although median survival seemed higher with multimodal therapy in terms of absolute numbers (33 vs. 20 months), statistical significance was not reached and no clear advantage over radiotherapy alone was evident.  Surgery was not related to dramatic improvement in oncologic outcomes. Conversely, hormonal therapy with megestrol has been associated with good results, particularly in combination with other treatment modalities. Again, we must emphasise that data are too scant to support a strong recommendation on the best therapeutic approach for bone metastases of EC.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]