Harsch Igor Alexander, Konturek Peter Christopher
Department of Internal Medicine II, Division of Endocrinology and Metabolism, Thuringia Clinic Saalfeld, Saalfeld/Saale, Germany.
Department of Internal Medicine II, Division of Gastroenterology, Thuringia Clinic Saalfeld, Saalfeld/Saale, Germany.
Am J Case Rep. 2019 Sep 7;20:1325-1330. doi: 10.12659/AJCR.917275.
BACKGROUND Hypercalcemia in cholangiocellular carcinoma is a highly uncommon event, mainly reported in Asian patients. In the absence of bone metastases, humoral hypercalcemia of malignancy (HHM) can be assumed. This is mostly the consequence of an elevated parathormone-related peptide (PTHrP) level. The standard therapeutic options in HHM are sometimes limited by the underlying disease or concomitant diseases. CASE REPORT We report the case of a 65-year-old Caucasian male. A syncope due to a hypercalcemia of 4.16 mmol/L (normal range, 2.19-2.54 mmol/L) was the initial symptom that eventually led to the diagnosis of cholangiocellular carcinoma. He had no metastatic bone disease; HHM was suspected. PTHrP was moderately elevated. Since there were contraindications for the standard therapeutic options, a therapy with 120 mg denosumab was initiated and proved effective, safe, and restored the patient's quality of life for 11 months. CONCLUSIONS The moderate elevation of parathyroid hormone-related peptide (PTHrP) in this case is addressed in context with the recent insights of a substantial underestimation of this parameter by many commercial assays which can explain our observation. Denosumab, a human monoclonal antibody which acts as a RANKL-inhibitor (receptor activator of nuclear factor kappaB ligand) was recently suggested as a therapeutic alternative. In this case, the therapy of the hypercalcemia with denosumab due to contraindications for other therapies led to an effective and long-standing remission of hypercalcemia. Its effectivity should be studied in larger case samples.
背景 胆管细胞癌中的高钙血症是一种非常罕见的情况，主要在亚洲患者中报道。在没有骨转移的情况下，可以假定为恶性肿瘤体液性高钙血症（HHM）。这主要是甲状旁腺激素相关肽（PTHrP）水平升高的结果。HHM的标准治疗选择有时会受到基础疾病或伴随疾病的限制。病例报告 我们报告了一名65岁白种男性的病例。最初的症状是因血钙水平为4.16 mmol/L（正常范围2.19 - 2.54 mmol/L）导致的晕厥，最终诊断为胆管细胞癌。他没有转移性骨病；怀疑为HHM。PTHrP中度升高。由于标准治疗选择存在禁忌证，开始使用120 mg地诺单抗进行治疗，结果证明该治疗有效、安全，并使患者的生活质量恢复了11个月。结论 结合近期许多商业检测对该参数严重低估的见解，讨论了本病例中甲状旁腺激素相关肽（PTHrP）的中度升高情况，这可以解释我们的观察结果。地诺单抗是一种人单克隆抗体，作为核因子κB受体激活剂配体（RANKL）抑制剂，最近被建议作为一种治疗选择。在本病例中，由于其他治疗存在禁忌证，用地诺单抗治疗高钙血症导致了高钙血症有效且长期缓解。其有效性应在更大的病例样本中进行研究。
