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Said Darawshi Department of Endocrinology, Rambam Health Care Campus, Haifa, Israel
The Faculty of Medicine, Technion, Haifa, Israel

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Mahmoud Darawshi Clalit Health Services, Northern District – Arrabah, Israel

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Deeb Daoud Naccache Department of Endocrinology, Rambam Health Care Campus, Haifa, Israel
The Faculty of Medicine, Technion, Haifa, Israel

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Severe hypocalcaemia in breast cancer with bone metastasis is a rare finding usually associated with an advanced stage of the disease. We report a case of a 45-year-old woman with a history of local ductal carcinoma in situ (DCIS) of the breast, who presented with muscle tremors and general weakness. Hypocalcaemia was evident, with a positive Chvostek sign and a serum calcium level of 5.9 mg/dL (1.47 mmol/L), phosphorus 5.9 mg/dL (normal range: 2.3–4.7 mg/dL) with normal levels of albumin, magnesium and parathyroid hormone. High oral doses of alpha calcitriol and calcium with i.v. infusion of high calcium doses were instituted, altogether sufficient to maintain only mild hypocalcaemia. A whole-body CT revealed bone lesions along the axial skeleton. A biopsy from a bone lesion revealed a metastasis of breast carcinoma. With this pathological finding, leuprolide (GNRH analogue) and chlorambucil (alkylating agent) were initiated, followed by prompt tapering of infused calcium down to full discontinuation. Serum calcium was kept stable close to the low normal range by high doses of oral alpha calcitriol and calcium. This course raises suspicion that breast metastases to the skeleton caused tumour-induced hypocalcaemia by a unique mechanism. We assume that hypocalcaemia in this case was promoted by a combination of hypoparathyroidism and bone metastasis.

Learning points

  • Severe hypocalcaemia can a presenting symptom for breast cancer relapse.

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Viviana Ostrovsky Diabetes, Endocrinology and Metabolic Disease Institute, Kaplan Medical Center, Hebrew University Medical School of Jerusalem, Rehovot, Israel

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Barak Bar-Zakai Surgery A Department, Kaplan Medical Center, Hebrew University Medical School of Jerusalem, Rehovot, Israel

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Rafael Miller Surgery A Department, Kaplan Medical Center, Hebrew University Medical School of Jerusalem, Rehovot, Israel

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Tal Schiller Diabetes, Endocrinology and Metabolic Disease Institute, Kaplan Medical Center, Hebrew University Medical School of Jerusalem, Rehovot, Israel

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Alena Kirzhner Diabetes, Endocrinology and Metabolic Disease Institute, Kaplan Medical Center, Hebrew University Medical School of Jerusalem, Rehovot, Israel

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Hilla Knobler Diabetes, Endocrinology and Metabolic Disease Institute, Kaplan Medical Center, Hebrew University Medical School of Jerusalem, Rehovot, Israel

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Taiba Zornitzki Diabetes, Endocrinology and Metabolic Disease Institute, Kaplan Medical Center, Hebrew University Medical School of Jerusalem, Rehovot, Israel

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Summary

Well-differentiated thyroid cancer (WDTC), including papillary, follicular, and Hurthle-cell types, is characterized by a slow course and usually remain localized to the thyroid. However, a minority of these cases develop distant metastases with the most common sites being lungs, bones, and lymph nodes. Liver metastases of WDTC are rare and are usually found along with other distant metastases sites and in a multiple or diffuse pattern of spread. The recognition of distant metastasis in WDTC has a significant impact on the treatment and prognosis. However, because of their low incidence and awareness, distant metastases are often diagnosed late. Herein, we describe a case of a 71 years old woman who during routine surveillance of a follicular variant of papillary thyroid cancer (FV-PTC), 5 years after being treated for her primary thyroid tumor, was found to have a single liver metastasis and underwent liver segmental resection. This case highlights the importance of maintaining vigilant surveillance of patients with WDTC, and illustrates the possibility of unique metastasis at unexpected sites. Further studies are needed to understand the organ tropism of some WDTC leading to distant metastases development and to better prediction of an aggressive course.

Learning points:

  • WDTC patients with distant metastases have a poor prognosis with a 10-year survival of about 50%. The most common sites of distant metastases are lung, bone and lymph nodes.

  • Liver metastases are rare in WDTC, are usually multiple or diffuse and are found along with other distant metastases sites.

  • Single liver metastasis of WDTC is an unexpected pattern of spread, and very few cases are reported in the literature.

  • Rare sites of distant metastases in WDTC can manifest many years after the primary tumor, stressing the importance of maintaining vigilant surveillance.

  • More studies are needed to predict which WDTC tumors may develop a more aggressive course, allowing clinicians to individualize patient management.

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Zaina Adnan Endocrinology and Metabolism Department, Zvulon Medical Center, Clalit Medical Health Care Services, Haifa, Israel

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David Nikomarov Orthopedic Surgery Department, Nuclear Medicine Department, Rambam Health Care Campus, Haifa, Israel

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Michal Weiler-Sagie Michal Weiler-Sagie, Nuclear Medicine Department, Rambam Health Care Campus, Haifa, Israel

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Noga Roguin Maor Clalit Medical Health Care and the Clinical Research Unit, Haifa and Western Galilee, Haifa, Israel

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Summary

Phosphaturic mesenchymal tumor (PMT) represents a rare cause of osteomalacia. The clinical signs and symptoms are vague and these lead to diagnosis delay. In the presence of hypophosphatemia and relatively high urine phosphate excretion, this entity should be taken into consideration in the deferential diagnosis of osteomalacia. In the present article, we report 81-year-old man presented to our clinic for evaluation due to osteopenia. His laboratory results disclosed hypophosphatemia, relatively increased urine phosphate excretion and increased level of intact fibroblast growth factor 23 (FGF23). A 68Gallium DOTATATE PET/CT revealed pathological uptake in the upper aspect of the left shoulder adjacent to the coracoid process. For suspected PMT a wide resection of the tumor was performed and pathological findings were consistent for PMT. Laboratory tests were normalized postoperatively. Reviewing the literature, we had identified 33 reported cases of PMTs among elderly patients age ≥70 years. Unlike previously reported data, where tumors predominantly localized in the lower extremities and pelvis, our search disclosed a high rate of tumor localization (10 cases – 33.3%) in the head with equal number of tumors (14 cases – 42.4%) localized in the head and upper extremity as well as in pelvis and lower extremity. The present case describes unique tumor localization in an elderly patient and our literature search demonstrated for the first time a high rate of tumor localization in the head among this group of patients.

Learning points:

  • PMTs represent a rare entity that should be considered in the differential diagnosis of elderly patients presented with persistent hypophosphatemia.

  • Unlike previously reported data, head and neck tumor localization is frequent among elderly patients.

  • 68Gallium-conjugated somatostatin peptide analogs, such as 68Ga-DOTATATE PET/CT demonstrated the greatest sensitivity and specificity for tumor localization in patients with phosphaturic mesenchymal tumors (PMTs).

  • Wide tumor resection using intraoperative ultrasound is of major importance in order to ensure long-term cure.

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