Parathyroid Crisis as Presentation of Atypical Parathyroid Adenoma: Two Diagnostically Challenging Cases

Abstract

Atypical parathyroid adenoma (APA) is a rare cause of primary hyperparathyroidism (PHPT) and represents a diagnostic challenge since it is an intermediate form of parathyroid neoplasm of uncertain malignant potential with atypical histological features that require differential diagnosis of parathyroid carcinoma (PC). We present 2 cases of parathyroid crisis as a presentation of APA. The first case was that of a 56-year-old man with parathyroid crisis, constitutional syndrome, and anemia, with evidence of APA after en bloc resection, evolving with hungry bone syndrome after surgery and curation criteria at 6 months after parathyroidectomy (PTX). The second case was a 64-year-old woman with acute chronic kidney disease and parathyroid crisis, with evidence of APA after selective PTX and >50% reduction in parathyroid hormone levels after surgery; however, persistent PHPT at 6 months post-surgery was observed. These cases represented a diagnostic challenge due to their rare clinical presentation (parathyroid crisis), with a heterogeneous spectrum of target organ damage and infrequent symptoms (constitutional syndrome and acute chronic renal disease), in turn caused by a rare pathology (APA). The presentation of these patients may be indicative of PC; however, histopathological diagnosis is a key to the diagnosis of APA. The differential diagnosis of APA vs. PC in clinical practice is indispensable.

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

“…This was a 56-year-old male (also associating anemia) who developed a 6-month HBS after en bloc resection. The authors reported a similar case of atypical PT adenoma on a 64-year-old female presented for a PT crisis and an acute form of a chronic renal damage as onset of PHPT; she underwent a selective PTx with persistent HPT and no HBS, suggesting that persistent HPT might be the opposite scenario for the patients who develop HBS [48]. Generally, a PT crisis is an extremely severe and rare complication of hormonal anomalies in PHPT.…”

“…Additionally, in this category damage or elevated blood urea nitrogen [3 calcium [46]. The second category comes from age for adults (yet, not all authors agree) [37,4 case reports) especially brown tumors 58,65,69,70,75,77,88,95,96]; less than convinc oporosis [43] or of a PT crisis [48,71,78]. The self; larger adenomas might have a higher ris [39,82], giant PT adenomas [50,51,78,88], ect…”

“…Additionally, in this category damage or elevated blood urea nitrogen [3 calcium [46]. The second category comes from age for adults (yet, not all authors agree) [37,4 case reports) especially brown tumors 58,65,69,70,75,77,88,95,96]; less than convinc oporosis [43] or of a PT crisis [48,71,78]. The self; larger adenomas might have a higher ris [39,82], giant PT adenomas [50,51,78,88], ect blood urea nitrogen Diagnostics 2023, 13, x FOR PEER REVIEW [120] Ohlrich 2005 [151] 38-y female with RHPT and severe malnutrition Savio 2004 [122] N = 6 p with X-linked dominant hypophosphatemic ric Miles 1997 [152] N = 1 with RHPT after 1 y since renal transplant Hardoff 1996 [153] 59-y male with RHPT and brown tumors…”

“…Generally, a PT crisis is an extremely severe and rare complication of hormonal anomalies in PHPT. This severe clinical complication is still an open issue if it represents an independent risk factor for HBS [48,49].…”

“…The timeline started from 1976, while the most recent paper was published in 2023. The youngest patient was of 20 years, the oldest of 72 years; a total number of 1582 adults were studied for HBS following surgery for PHPT [7,10,11,14,15,19,20,48,[50][51][52][53][54][55][56][57][58][65][66][67][68][69][70][71][72][73][74][75][76][77][78][79][80][81][82][83] ((Table 2).…”