Severe hypercalcemic hyperparathyroidism developing in a patient with hyperaldosteronism and renal resistance to parathyroid hormone

Abstract

We evaluated an African American woman referred in 1986 at age 33 years because of renal potassium and calcium wasting and chronic hip pain. She presented normotensive, hypokalemic, hypocalcemic, normophosphatemic, and hypercalciuric. Marked hyperparathyroidism was evident. Urinary cyclic adenosine monophosphate (cAMP) excretion did not increase in response to parathyroid hormone (PTH) infusion, indicating renal resistance to PTH. X-rays and bone biopsy revealed severe osteitis fibrosa cystica, confirming skeletal responsiveness to PTH. Renal potassium wasting, suppressed plasma renin activity, and elevated plasma and urinary aldosterone levels accompanied her hypokalemia, suggesting primary hyperaldosteronism. Hypokalemia resolved with spironolactone and, when combined with dietary sodium restriction, urinary calcium excretion fell and hypocalcemia improved, in accord with the known positive association between sodium intake and calcium excretion. Calcitriol and oral calcium supplements did not suppress the chronic hyperparathyroidism nor did they reduce aldosterone levels. Over time, hyperparathyroid bone disease progressed with pathologic fractures and persistent pain. In 2004, PTH levels increased further in association with worsening chronic kidney disease. Eventually hypercalcemia and hypertension developed. Localizing studies in 2005 suggested a left inferior parathyroid tumor. After having consistently declined, the patient finally agreed to neck exploration in January 2009. Four hyperplastic parathyroid glands were removed, followed immediately by severe hypocalcemia, attributed to ''hungry bone syndrome'' and hypoparathyroidism, which required prolonged hospitalization, calcium infusions, and oral calcitriol. Although her bone pain resolved, hyperaldosteronism persisted. ß

The chloride/phosphate ratio combined with alkaline phosphatase as a valuable predictive marker for primary hyperparathyroidism in Chinese individuals

“…Previous studies 25, 28–30 suggested that elevated ALP levels existed in the patients of primary hyperparathyroidism. Serum alkaline phosphatase originated principally from bone.…”

“…On the one hand, the pathophysiological mechanism of elevated Hb in PHPT patients has not yet known. On the other hand, as for serum sodium level was different in two groups, one possible pathophysiological mechanism was that patients with hyperparathyroidism have higher aldosterone levels, and elevated aldosterone levels have positive correlation with sodium intake 30 . Another possible pathophysiological mechanism was that the reabsorption of sodium was induced by PTH 25, 26 .…”

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

“…On a larger picture, we mention some aspects that are still a matter of debate. Park-Sigal et al [148] reported a case of PHPT with multiple complications that associated primary hyperaldosteronism; we do not have enough evidence to correlate it with HBS [148]. Some studies included patients with PHPT associated with renal damage, but not representing secondary (renal) or tertiary HPT, and this aspect might embrace the scenario of RHPT following PTx [43].…”

“…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].…”