An Unusual Case of Meningioma Showing Increased CaSR Expression with Parathyroid Carcinoma

Article information

Endocrinol Metab. 2018;33(1):133-134

Publication date (electronic) : 2018 January 30

doi : https://doi.org/10.3803/EnM.2018.33.1.133

Jin Sae Yoo ¹, Hong Min Kim ¹, Sera Kim ¹, Tak Ho Kang ², Mee Yon Cho ³, Choon Hee Chung ¹

¹Department of Internal Medicine, Yonsei University Wonju College of Medicine, Wonju, Korea.

²Department of Medicine, Korea University College of Medicine, Seoul, Korea.

³Department of Pathology, Yonsei University Wonju College of Medicine, Wonju, Korea.

Corresponding author: Choon Hee Chung. Department of Internal Medicine, Yonsei University Wonju College of Medicine, 20 Ilsan-ro, Wonju 26426, Korea. Tel: +82-33-741-0506, Fax: +82-33-731-5884, cchung@yonsei.ac.kr

Received 2017 October 14; Revised 2017 November 27; Accepted 2017 December 06.

A 68-year-old woman with a history of parathyroidectomy of the left upper and right lower parathyroid gland for parathyroid carcinoma was admitted to our hospital for evaluation of fatigue and anorexia that lasted for 1 month. Blood chemistry was notable for hypercalcemia (14.1 mg/dL) and elevated parathyroid hormone (PTH) 1,652 pg/mL (0.35 to 5.50). Serum PTH-related peptide (PTHrP) level was measured at 33.2 pmol/L (13.8 to 55.3). T-score of spine and femur neck measured by dual energy X-ray absorptiometry was −3.12 and −4.15, suggesting osteoporosis. Computed tomography scan of the neck found a nodule sized 1×1.2 cm on the left neck (Fig. 1) [1]. She developed two episodes of myoclonic seizures at the 9th day on hospitalization. Magnetic resonance imaging of the brain found a 3×4 cm sized lesion on her left frontal lobe later reported as meningioma (Fig. 2A) [1]. Surgical resection of the meningioma and the left cervical mass was performed. Pathology of the cervical mass found hyperplasia of parathyroid gland, suggesting recurrence of parathyroid carcinoma. Thirteen years after the diagnosis of meningioma, we performed immunohistochemistry on the meningeal tissue for calcium-sensing receptor (CaSR) at the meninges and found strong CaSR expression (Fig. 2B).

Fig. 1

Computed tomography scan of the neck showing 1×1.2 cm sized left cervical mass with low density, consistent with recurrent parathyroid carcinoma (arrow). Adapted from Ju et al. [1].

Fig. 2

(A) Magnetic resonance imaging of the meningioma. A 3×4 cm sized extra-axial tumor along the left frontal convexity consistent with meningioma is noted (arrow). Adapted from Ju et al. [1]. (B) Immunohistochemical localization of calcium sensing receptor (CaSR) in meningioma. Strong CaSR-antibody (diluted 1:500, Novus Biologicals) reactivity in meningioma tissue is notable (×200).

It has been known that CaSR has oncogenic properties and over-expression of CaSR is observed in some metastasizing breast, prostate cancer, and meningioma [2 3]. Enhanced CaSR expression on meningeal cells and elevated calcium level due to parathyroid carcinoma would be a trigger for meningioma [4]. Enhanced PTHrP expression has also known to be observed in meningioma [4 5]; however, in this case PTHrP concentration in blood was within normal range. Additionally, the coincidence of parathyroid carcinoma and meningioma may infer the presence of multiple endocrine neoplasia type 1 (MEN1) cancer syndrome, whose diagnosis needs DNA testing [6].

Notes

CONFLICTS OF INTEREST: No potential conflict of interest relevant to this article was reported.

References

1. Ju HS, Koh JH, Hong TW, Shim MS, Kim MJ, Lee KG, et al. A case of parathyroid carcinoma with meningioma. Korean J Med 2003;65(Suppl 3):S767–S772.

2. Saidak Z, Mentaverri R, Brown EM. The role of the calcium-sensing receptor in the development and progression of cancer. Endocr Rev 2009;30:178–195. 19237714.

3. Brennan SC, Thiem U, Roth S, Aggarwal A, Fetahu ISh, Tennakoon S, et al. Calcium sensing receptor signalling in physiology and cancer. Biochim Biophys Acta 2013;1833:1732–1744. 23267858.

4. Chattopadhyay N, Evliyaoglu C, Heese O, Carroll R, Sanders J, Black P, et al. Regulation of secretion of PTHrP by Ca(2+)-sensing receptor in human astrocytes, astrocytomas, and meningiomas. Am J Physiol Cell Physiol 2000;279:C691–C699. 10942719.

5. Kim YL, Jang YW, Kim JT, Sung SA, Lee TS, Lee WM, et al. A rare case of primary hyperparathyroidism associated with primary aldosteronism, Hurthle cell thyroid cancer and meningioma. J Korean Med Sci 2012;27:560–564. 22563225.

6. Marini F, Falchetti A, Del Monte F, Carbonell Sala S, Gozzini A, Luzi E, et al. Multiple endocrine neoplasia type 1. Orphanet J Rare Dis 2006;1:38. 17014705.

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Fig. 1

Computed tomography scan of the neck showing 1×1.2 cm sized left cervical mass with low density, consistent with recurrent parathyroid carcinoma (arrow). Adapted from Ju et al. [1].

Fig. 2

(A) Magnetic resonance imaging of the meningioma. A 3×4 cm sized extra-axial tumor along the left frontal convexity consistent with meningioma is noted (arrow). Adapted from Ju et al. [1]. (B) Immunohistochemical localization of calcium sensing receptor (CaSR) in meningioma. Strong CaSR-antibody (diluted 1:500, Novus Biologicals) reactivity in meningioma tissue is notable (×200).