Fatal Hypocalcaemia Due to Hungry Bone Syndrome with Secondary Refractory HyperParathyroidism after Parathyroidectomy: A Case Report

Abstract

Introduction Hungry bone syndrome (HBS) refers to the rapid, profound, and prolonged hypocalcaemia associated with hypophosphatemia and hypomagnesaemia, and is exacerbated by suppressed parathyroid hormone (PTH) levels, which follows parathyroidectomy in patients with severe primary hyperparathyroidism (PHPT) and preoperative high bone turnover. [1] Case report This report concerns a dialysed patient who underwent surgical treatment for secondary refractory hyperparathyroidism. Haemodialysis was carried out pre-operatively, and subsequently, a total parathyroidectomy with auto-transplantation of parathyroid tissue in the sternocleidomastoid muscle (SCM) was performed. Rapid and progressive hypocalcaemia symptoms developed during the second day postoperatively. Acute cardiac symptoms with tachyarrhythmia, haemodynamic instability and finally asystole occurred, which required cardiopulmonary resuscitation (CPR). The ionic calcium level was 2.2 mg/dL being consistent with a diagnosis of HBS. A second cardiac arrest unresponsive to CPR followed an initial period of normal sinus rhythm. Death ensued shortly after. Before death, the ionic calcium was 3.1 mg/dL. Conclusion HBS, after parathyroidectomy in patients with secondary hyperparathyroidism (SHPT), may be severe, prolonged and sometimes fatal. Generally, HBS symptomatology is that of a mild hypocalcaemia. It can, however, include heart rhythm disturbances with haemodynamic alterations requiring intensive care measurements and even cardiopulmonary resuscitation. A close clinical and laboratory post-parathyroidectomy monitoring of dialysed patients is of the utmost importance.

Multiple Brown Tumors Caused by Primary Hyperparathyroidism as a Differential Diagnosis to Multiple Osteolytic Bone Metastases: A Case Report

“…Other reported cases of brown tumors had received similar treatment [4,6,8,19,21]. The brown tumors may completely regress after the normalisation of PTH level.…”

Forestalling Hungry Bone Syndrome after Parathyroidectomy in Patients with Primary and Renal Hyperparathyroidism

“…Teriparatide might represent an alternative to dramatic cases of HBS-associated hypocalcemia, but we still need consistent statistical evidence; yet, the drug is applied in hypoparathyroidismassociated hypocalcemia [138][139][140]. Of note, severe low total calcium levels (of 2-3 mg/dL) were associated with an increased risk of acute cardiac complications from tachyarrhythmia to asystole in association with consecutive hemodynamic instability and a potential fatal outcome [31].…”

“…Despite a generous level of statistical evidence in RHPT according to the mentioned studies, since the topic of HBS is sometimes a matter of individual decision, we analyzed the published case reports, and identified another 25 papers with post-operatory HBS (7 cases series that enrolled 2 to 10 patients per series, and 18 single case reports, a total of 48 patients with RHPT-related HBS, aged between 23 and 74 years (these articles were published between 1989 and 2022) [8,25,26,31,111,[119][120][121][122]136,137,141,[143][144][145][146][147][148][149][150][151][152][153][154][155] (Table 6). Table 6.…”

“…Table 6. Original case reports and series in patients diagnosed with RHPT referred to PTx and complicated with HBS; the display starts with the most recent publication timeline [8,25,26,31,111,[119][120][121][122]136,137,141,[143][144][145][146][147][148][149][150][151][152][153][154][155].…”

“…Over the decades, HBS was listed among the severe side of post-PTx complications, a heterogeneous picture that includes vocal cord palsy, local hemorrhage, transitory or permanent hypocalcemia/hypoparathyroidism, or even peri-operatory mortality [27][28][29][30][31][32][33].…”