PMID- 18430790 OWN - NLM STAT- MEDLINE DCOM- 20080715 LR - 20161124 IS - 1479-683X (Electronic) IS - 0804-4643 (Linking) VI - 159 IP - 1 DP - 2008 Jul TI - Insulinoma cell calcium-sensing receptor influences insulin secretion in a case with concurrent familial hypocalciuric hypercalcemia and malignant metastatic insulinoma. PG - 81-6 LID - 10.1530/EJE-08-0069 [doi] AB - CONTEXT AND OBJECTIVE: Arterial stimulation and venous sampling (ASVS) is an important technique for localizing insulinoma. The principle behind ASVS is that insulin secretion is promoted from insulinoma cells by the injection of calcium into the insulinoma-feeding artery. However, the mechanism for ASVS-induced insulin secretion remains unclear. Both insulinoma and familial hypocalciuric hypercalcemia (FHH) are rare diseases. This study reports on a case in which both of these diseases occur concurrently. DESIGN AND PATIENT: The patient with FHH also suffered from insulinoma. We reasoned that insulin secretion for ASVS is dependent on the calcium-sensing receptor (CaSR). ASVS was performed on this patient. The expression of the CaSR protein and corresponding mRNA were confirmed. RESULTS: No significant changes in the plasma levels of insulin and C-peptide were observed during ASVS. The patient was clinically diagnosed as having FHH. We confirmed that a mutation in the CaSR gene was present in the genomic DNA of this patient and that there were no mutations in the multiple endocrine neoplasia type 1 gene. In addition, expression of both CaSR mRNA and CaSR protein was confirmed in the insulinoma samples. CONCLUSION: These results suggest that the CaSR gene is involved in ASVS-induced insulin secretion. FAU - Ono, Yasunaga AU - Ono Y AD - Division of Endocrinology and Metabolism, Department of Internal Medicine, Fujita Health University School of Medicine, Toyoake, Aichi 470-1192, Japan. FAU - Oda, Naohisa AU - Oda N FAU - Ishihara, Shin AU - Ishihara S FAU - Shimomura, Atsushi AU - Shimomura A FAU - Hayakawa, Nobuki AU - Hayakawa N FAU - Suzuki, Atsushi AU - Suzuki A FAU - Horiguchi, Akihiko AU - Horiguchi A FAU - Senda, Takao AU - Senda T FAU - Miyakawa, Shuichi AU - Miyakawa S FAU - Itoh, Mitsuyasu AU - Itoh M LA - eng PT - Case Reports PT - Journal Article DEP - 20080422 PL - England TA - Eur J Endocrinol JT - European journal of endocrinology JID - 9423848 RN - 0 (Insulin) RN - 0 (Receptors, Calcium-Sensing) SB - IM MH - Blotting, Western MH - Female MH - Humans MH - Hypercalcemia/diagnostic imaging/genetics/metabolism/*pathology MH - Immunohistochemistry MH - Insulin/metabolism MH - Insulinoma/genetics/metabolism/*pathology MH - Liver/diagnostic imaging/metabolism/pathology MH - Middle Aged MH - Pancreas/diagnostic imaging/metabolism/pathology MH - Radionuclide Imaging MH - Receptors, Calcium-Sensing/genetics/metabolism/*physiology MH - Reverse Transcriptase Polymerase Chain Reaction MH - Tomography, X-Ray Computed EDAT- 2008/04/24 09:00 MHDA- 2008/07/17 09:00 CRDT- 2008/04/24 09:00 PHST- 2008/04/24 09:00 [pubmed] PHST- 2008/07/17 09:00 [medline] PHST- 2008/04/24 09:00 [entrez] AID - EJE-08-0069 [pii] AID - 10.1530/EJE-08-0069 [doi] PST - ppublish SO - Eur J Endocrinol. 2008 Jul;159(1):81-6. doi: 10.1530/EJE-08-0069. Epub 2008 Apr 22.