Pseudogout – a rare manifestation of hungry bone syndrome after focused parathyroidectomy

Tai, Chia Hui; Oh, Han Boon; Seet, Ju‐Ee; Ngiam, Kee Yuan

doi:10.1308/rcsann.2018.0028

Cited by 7 publications

(7 citation statements)

References 9 publications

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“…In these cases, in most patients, the presence of chondrocalcinosis, and painful crisis were correlated with a decline in calcium and magnesium levels 24–48 hours after surgery. This finding matches the patient cohort described earlier that had chondrocalcinosis with acute or chronic arthritis and most developed a pseudogout crisis in the postoperative period [ 10 ]. Additionally, this patient presented with calcification of the cruciform ligament in a neck CT, also known as Crowned Dens syndrome ( Figure 5 ), a finding that has been described in patients with chondrocalcinosis [ 12 ].…”

Section: Discussionsupporting

confidence: 90%

“…In a cohort of 74 patients with hyperparathyroidism, 40% presented with musculoskeletal complaints such as pseudogout, back pain, vertebral fracture, and arthralgias. Furthermore, in 20.2% of the patients, OFC was the most common radiological finding [ 10 ]. Until 2018, 30 cases of pseudogout after parathyroidectomy have been described [ 11 ].…”

Section: Discussionmentioning

confidence: 99%

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Giant Parathyroid Adenoma-Associated Fracture, Not All Lytic Bone Lesions are Cancer: A Case-Based Review

Álvarez-Payares

Ribero

Ramírez-Urrea

et al. 2022

Case Reports in Medicine

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Introduction. Due to the early diagnosis of primary hyperparathyroidism the musculoskeletal manifestations of this disease are becoming less frequent. When this disease manifests secondary to a giant adenoma, it presents with more aggressive symptoms and can have important repercussions such as the hungry bone syndrome after parathyroidectomy. There are few reported cases of hyperparathyroidism secondary to a giant adenoma in the literature, as the presence of a brown tumor is often misinterpreted as a metastatic lesion from an unknown primary tumor. Methods. We describe a case and performed a literature review to identify all case reports. A literature search was carried out on PubMed/MEDLINE and EMBASE bibliographic databases. All available studies from May 2009 to May 2021 were included. Data were tabulated, and outcomes were cumulatively analyzed. Results. Twenty-four cases of primary hyperparathyroidism due to giant adenoma have been described; the majority were women, with a mean age of 52 years. They presented with heterogeneous symptoms such as palpable nodules (45%), bone pain (33%), brown tumor (12.5%), asymptomatic (12.5%), metabolic profile with a mean calcemia of 13.8 mg/dL, PTH 1109 ng/L, and mean tumor weight of 47.24 g. Conclusion. Primary hyperparathyroidism due to giant adenoma increases the risk of developing potentially serious postoperative complications such as hungry bone syndrome. This implies the need of implementing preventive measures comprising administration of intravenous zoledronic acid and early supplementation of oral calcium to prevent complications after resection.

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Section: Discussionsupporting

confidence: 90%

Section: Discussionmentioning

confidence: 99%

Giant Parathyroid Adenoma-Associated Fracture, Not All Lytic Bone Lesions are Cancer: A Case-Based Review

Álvarez-Payares

Ribero

Ramírez-Urrea

et al. 2022

Case Reports in Medicine

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“…In cases with PHP, PG has been defined in the postoperative period following a decrease in serum calcium levels. [ 4 ] Dissolution of CPPD by the inhibition of alkaline BP, rapid decrease in serum calcium, crystal formation in joints, and release of cytokines that cause a flu-like syndrome after IV BP infusion are the causes of PG development. [ 5 ] There are limited cases of PG triggered by BP treatment given for osteoporosis in the literature,[ 5 ] and three of such cases developed after ZA.…”

Section: Discussionmentioning

confidence: 99%

A case of pseudogout attack after zoledronic acid treatment in primary hyperparathyroidism

Coskun¹

2023

Turk J Phys Med Rehab

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Pseudogout (PG) is an inflammatory arthropathy that develops due to the accumulation of calcium pyrophosphate dihydrate crystals in synovial structures. Herein, we present a 59-year-old male patient with PG developed as a result of zoledronic acid (ZA) infusion, which was administered due to primary hyperparathyroidism. The patient with parathyroid adenoma was given ZA since the calcium level did not decrease despite intravenous saline and loop diuretic. One day after ZA administration, the patient had severe pain, fever, and swelling in joints. The radiograph showed chondrocalcinosis. Calcium pyrophosphate deposition were observed in the arthrocentesis fluid under polarized light. The patient’s symptoms regressed after anakinra and colchicine treatment. To the best of our knowledge, this is the first case report of a PG attack after ZA treatment for primary hyperparathyroidism. Additionally, there have been few cases of PG after bisphosphonate treatment for osteoporosis in the literature, signifying that more care should be taken when administering bisphosphonate therapy in patients with risk factors.

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“…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).…”

Section: Panel Of Investigations In Phpt and Rhptmentioning

confidence: 99%

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

Cârșote

Nistor

2023

Diagnostics

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Hungry bone syndrome (HBS), severe hypocalcemia following parathyroidectomy (PTX) due to rapid drop of PTH (parathormone) after a previous long term elevated concentration in primary (PHPT) or renal hyperparathyroidism (RHPT), impairs the outcome of underlying parathyroid disease. Objective: overview HBS following PTx according to a dual perspective: pre- and post-operative outcome in PHPT and RHPT. This is a case- and study-based narrative review. Inclusion criteria: key research words “hungry bone syndrome” and “parathyroidectomy”; PubMed access; in extenso articles; publication timeline from Inception to April 2023. Exclusion criteria: non-PTx-related HBS; hypoparathyroidism following PTx. We identified 120 original studies covering different levels of statistical evidence. We are not aware of a larger analysis on published cases concerning HBS (N = 14,349). PHPT: 14 studies (N = 1545 patients, maximum 425 participants per study), and 36 case reports (N = 37), a total of 1582 adults, aged between 20 and 72. Pediatric PHPT: 3 studies (N = 232, maximum of 182 participants per study), and 15 case reports (N = 19), a total of 251 patients, aged between 6 and 18. RHPT: 27 studies (N = 12,468 individuals, the largest cohort of 7171) and 25 case reports/series (N = 48), a total of 12,516 persons, aged between 23 and 74. HBS involves an early post-operatory (emergency) phase (EP) followed by a recovery phase (RP). EP is due to severe hypocalcemia with various clinical elements (<8.4 mg/dL) with non-low PTH (to be differentiated from hypoparathyroidism), starting with day 3 (1 to 7) with a 3-day duration (up to 30) requiring prompt intravenous calcium (Ca) intervention and vitamin D (VD) (mostly calcitriol) replacement. Hypophosphatemia and hypomagnesiemia may be found. RP: mildly/asymptomatic hypocalcemia controlled under oral Ca+VD for maximum 12 months (protracted HBS is up to 42 months). RHPT associates a higher risk of developing HBS as compared to PHPT. HBS prevalence varied from 15% to 25% up to 75–92% in RHPT, while in PHPT, mostly one out of five adults, respectively, one out of three children and teenagers might be affected (if any, depending on study). In PHPT, there were four clusters of HBS indicators. The first (mostly important) is represented by pre-operatory biochemistry and hormonal panel, especially, increased PTH and alkaline phosphatase (additional indicators were elevated blood urea nitrogen, and a high serum calcium). The second category is the clinical presentation: an older age for adults (yet, not all authors agree); particular skeleton involvement (level of case reports) such as brown tumors and osteitis fibrosa cystica; insufficient evidence for the patients with osteoporosis or those admitted for a parathyroid crisis. The third category involves parathyroid tumor features (increased weight and diameter; giant, atypical, carcinomas, some ectopic adenomas). The fourth category relates to the intra-operatory and early post-surgery management, meaning an associated thyroid surgery and, maybe, a prolonged PTx time (but this is still an open issue) increases the risk, as opposite to prompt recognition of HBS based on calcium (and PTH) assays and rapid intervention (specific interventional protocols are rather used in RHPT than in PHPT). Two important aspects are not clarified yet: the use of pre-operatory bisphosphonates and the role of 25-hydroxyitamin D assay as pointer of HBS. In RHPT, we mentioned three types of evidence. Firstly, risk factors for HBS with a solid level of statistical evidence: younger age at PTx, pre-operatory elevated bone alkaline phosphatase, and PTH, respectively, normal/low serum calcium. The second group includes active interventional (hospital-based) protocols that either reduce the rate or improve the severity of HBS, in addition to an adequate use of dialysis following PTx. The third category involves data with inconsistent evidence that might be the objective of future studies to a better understanding; for instance, longer pre-surgery dialysis duration, obesity, an elevated pre-operatory calcitonin, prior use of cinalcet, the co-presence of brown tumors, and osteitis fibrosa cystica as seen in PHPT. HBS remains a rare complication following PTx, yet extremely severe and with a certain level of predictability; thus, the importance of being adequately identified and managed. The pre-operatory spectrum of assessments is based on biochemistry and hormonal panel in addition to a specific (mostly severe) clinical presentation while the parathyroid tumor itself might provide useful insights as potential risk factors. Particularly in RHPT, prompt interventional protocols of electrolytes surveillance and replacement, despite not being yet a matter of a unified, HBS-specific guideline, prevent symptomatic hypocalcemia, reduce the hospitalization stay, and the re-admission rates.

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Pseudogout – a rare manifestation of hungry bone syndrome after focused parathyroidectomy

Cited by 7 publications

References 9 publications

Giant Parathyroid Adenoma-Associated Fracture, Not All Lytic Bone Lesions are Cancer: A Case-Based Review

Giant Parathyroid Adenoma-Associated Fracture, Not All Lytic Bone Lesions are Cancer: A Case-Based Review

A case of pseudogout attack after zoledronic acid treatment in primary hyperparathyroidism

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

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