Hungry bone syndrome following parathyroidectomy for primary hyperparathyroidism in a developed country in the Asia Pacific. A cohort study

Chandran, Manju; Bilezikian, John P.; Salleh, Nurshazwani Mat; Ying, Hao; Lau, Joel; Lee, James; Jong, Mechteld C. de; Maung, Aye Chan; Parameswaran, Rajeev

doi:10.1016/j.afos.2022.03.004

Cited by 9 publications

(18 citation statements)

References 27 publications

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“…Post-operatively, HBS developed in 16.6% of patients, which is similar to other reports from Europe and the Middle East [ 35 – 37 ], but higher than rates observed in a recent report from Singapore [ 38 ]. The effects of preoperative bisphosphonate therapy are unclear, however, several reports have suggested that it may prevent or reduce HBS [ 14 , 39 – 42 ].…”

Section: Discussionsupporting

confidence: 87%

Characteristics, management and outcomes of primary hyperparathyroidism from 2009 to 2021: a single centre report from South Africa

Govind,

Paruk,

Motala

2024

BMC Endocr Disord

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Background There has been a notable shift towards the diagnosis of less severe and asymptomatic primary hyperparathyroidism (PHPT) in developed countries. However, there is a paucity of recent data from sub-Saharan Africa (SSA), and also, no reported data from SSA on the utility of intra-operative parathyroid hormone (IO-PTH) monitoring. In an earlier study from Inkosi Albert Luthuli Central Hospital (IALCH), Durban, South Africa (2003–2009), majority of patients (92.9%) had symptomatic disease. The aim of this study was to evaluate the clinical profile and management outcomes of patients presenting with PHPT at IALCH. Methods A retrospective chart review of patients with PHPT attending the Endocrinology clinic at IALCH between July 2009 and December 2021. Clinical presentation, laboratory results, radiologic findings, surgical notes and histology were recorded. Results Analysis included 110 patients (87% female) with PHPT. Median age at presentation was 57 (44; 67.5) years. Symptomatic disease was present in 62.7% (n:69); 20.9% (n:23) had a history of nephrolithiasis and 7.3% (n:8) presented with previous fragility fractures. Mean serum calcium was 2.87 ± 0.34 mmol/l; median serum-PTH was 23.3 (15.59; 45.38) pmol/l, alkaline phosphatase 117.5 (89; 145.5) U/l and 25-hydroxyvitamin-D 42.9 (33.26; 62.92) nmol/l. Sestamibi scan (n:106 patients) identified an adenoma in 83.02%. Parathyroidectomy was performed on 84 patients with a cure rate of 95.2%. Reasons for conservative management (n:26) included: no current surgical indication (n:7), refusal (n:5) or deferral of surgery (n:5), loss to follow-up (n:5) and assessed as high anaesthetic risk (n:4). IO-PTH measurements performed on 28 patients indicated surgical success in 100%, based on Miami criteria. Histology confirmed adenoma in 88.1%, hyperplasia in 7.1% and carcinoma in 4.8%. Post-operative hypocalcaemia developed in 30 patients (35.7%), of whom, 14 developed hungry bone syndrome (HBS). In multivariate analysis, significant risk factors associated with HBS included male sex (OR 7.01; 95% CI 1.28, 38.39; p 0.025) and elevated pre-operative PTH (OR 1.01; 95% CI 1.00, 1.02; p 0.008). Conclusions The proportion of asymptomatic PHPT has increased at this centre over the past decade but symptomatic disease remains the dominant presentation. Parathyroidectomy is curative in the majority of patients. IO-PTH monitoring is valuable in ensuring successful surgery.

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Section: Discussionsupporting

confidence: 87%

Characteristics, management and outcomes of primary hyperparathyroidism from 2009 to 2021: a single centre report from South Africa

Govind,

Paruk,

Motala

2024

BMC Endocr Disord

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“…The study showed significant correlation between adenoma volume and preoperative iPTH levels and demonstrated that the pre-operative iPTH and ALP levels were significantly associated with the risk of developing HBS. 13 Our patient had significantly higher pre-parathyroidectomy iPTH and corrected calcium levels, lower 25-hydroxyvitamin D levels and larger adenoma size compared to patients from the same population. This further emphasizes the atypical nature of our patient’s clinical presentation in a developed country and the risk for development of HBS.…”

Section: Discussionmentioning

confidence: 46%

An Atypical Presentation of Primary Hyperparathyroidism With Multiple Spontaneous Tendon Ruptures: A Case Report and Literature Review on the Management of Primary Hyperparathyroidism

Yew

Soh

2022

JAFES

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Primary hyperparathyroidism (PHPT) is a common endocrine condition, increasingly presenting asymptomatically and detected on routine laboratory examination in developed countries. Multiple spontaneous tendon ruptures as the initial presentation of PHPT is extremely rare. We present the case of a 28-year-old male diagnosed with severe hypercalcemia secondary to PHPT after presenting with complications of multiple spontaneous tendon ruptures,and discuss the management issues in PHPT for this patient.

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“…Studies have been reported currently that risk factors of occurrence of HBS include high PTH with a previous long-term elevated concentration, high calcium levels, and excessive ATP elevation, and so forth. 5,15,16 Active preoperative measures may reduce the incidence of postoperative HBS, Davenport and Stearns reported that preoperative pamidronate in combination with high doses of oral alfacalcidol and calcium supplements can prevent postoperative symptomatic hypocalcemia and help to prevent the occurrence of HBS. 17 In our case, the patient was given intravenous zoledronic acid combined with oral calcitriol before the operation in order to prevent the occurrence of HBS after parathyroid surgery since he had the above high-risk factors of postoperative HBS.…”

Section: Discussionmentioning

confidence: 99%

“…After radical resection of PC, the sudden decrease of PTH, which acts on bone resorption of osteoclasts does not affect the process of osteogenesis of osteoblasts; osteoclasts activity is replaced by osteoblasts activity, resulting in a rapid increase of calcium uptake, which leads to a large amount of serum calcium into the bone and deposit in the bone tissues, inducing severe symptomatic hypocalcemia such as numbness of perioral sites and fingertips, tetany, and even dyspnea. Studies have been reported currently that risk factors of occurrence of HBS include high PTH with a previous long‐term elevated concentration, high calcium levels, and excessive ATP elevation, and so forth 5,15,16 . Active preoperative measures may reduce the incidence of postoperative HBS, Davenport and Stearns reported that preoperative pamidronate in combination with high doses of oral alfacalcidol and calcium supplements can prevent postoperative symptomatic hypocalcemia and help to prevent the occurrence of HBS 17 .…”

Section: Discussionmentioning

confidence: 99%

Multiple pathological fractures and muscle atrophy caused by a parathyroid carcinoma with postoperative hungry bone syndrome: A case report

Liu,

Zhang,

et al. 2024

Cancer Reports

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BackgroundParathyroid carcinoma (PC) is a rare endocrine malignancy causing pathological changes such as abnormal bone metabolism, elevated serum calcium, and impaired renal function, and uncontrollable hypercalcemia is the main cause of death in PC patients. The diagnosis of PC is challenging and relying on postoperative histopathology. Radical surgery at the first time is the only effective therapy to cure PC. Hungry bone syndrome (HBS) is a relatively uncommon complication of parathyroidectomy characterized by profound and prolonged hypocalcemia, timely electrolyte monitoring and alternative interventional protocols can prevent symptomatic hypocalcemia.CaseA 57‐year‐old man presented with multiple pathological fractures and muscle atrophy as the main symptoms accompanied by bone pain, hypercalcemia, elevated parathyroid hormone (PTH), and an enlarged left‐sided neck mass. After consultation of multidisciplinary team, he was treated conservatively with plaster bandage fixation and infusion of intravenous zoledronic acid; and then complete resection of parathyroid mass + removal of involved tissue structures + left thyroid and isthmus lobectomy + lymph node dissection in the VI region in left neck were performed. The postoperative histopathology suggested a diagnosis of parathyroid carcinoma. Calcium and fluid supplementation and oral levothyroxine tablets were given postoperatively. Unexpectedly, the patient's PTH level decreased rapidly at 24 h postoperative, and serum calcium and phosphorus decreased continuously, and he felt numb around perioral sites and fingertips, which considered to be postoperative HBS complicated by parathyroidectomy. Then, a large amount of calcium supplementation and vitamin D were given timely and the patient got better at 1 month postoperatively. At 9‐month postoperative, his bone pain and fatigue were significantly relieved compared with before with calcium, phosphorus, and PTH levels at normal range.ConclusionThe possibility of parathyroid disease, particularly PC, should be considered in the presence of multiple pathological fractures, muscle atrophy, generalized bone pain, hypercalcemia, and clear neck mass. Radical resection of the tumor lesions at the first surgery is a key element affecting the prognosis of PC, and the effective management of preoperative hypercalcemia and postoperative HBS is also of great significance for improving prognosis.

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Hungry bone syndrome following parathyroidectomy for primary hyperparathyroidism in a developed country in the Asia Pacific. A cohort study

Cited by 9 publications

References 27 publications

Characteristics, management and outcomes of primary hyperparathyroidism from 2009 to 2021: a single centre report from South Africa

Characteristics, management and outcomes of primary hyperparathyroidism from 2009 to 2021: a single centre report from South Africa

An Atypical Presentation of Primary Hyperparathyroidism With Multiple Spontaneous Tendon Ruptures: A Case Report and Literature Review on the Management of Primary Hyperparathyroidism

Multiple pathological fractures and muscle atrophy caused by a parathyroid carcinoma with postoperative hungry bone syndrome: A case report

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