A case of adolescent giant parathyroid adenoma presenting multiple osteolytic fractures and postoperative hungry bone syndrome

Ebina, Kosuke; Miyoshi, Yuji; Izumi, Shigeki; Hashimoto, Jun; Naka, Norifumi; Tsukamoto, Yasunori; Kashii, Masafumi; Kaito, Takashi; Yoshikawa, Hideki

doi:10.1002/ccr3.360

Cited by 10 publications

(7 citation statements)

References 17 publications

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“…PTH levels fell by 90.43% intraoperatively and serum Ca levels fell by 38.88% postoperatively. Studies have shown that a decrease in intraoperative PTH levels of 80% or more is a risk factor for the development of postoperative hungry bone syndrome (HBS) [11]. However, our patient had stable serum Ca levels and did not develop HBS postoperatively.…”

Section: Discussioncontrasting

confidence: 51%

Minimally Invasive Parathyroidectomy in a Child With Acute Pancreatitis

Ali¹,

Seetahal-Maraj²,

Roop³

et al. 2022

Cureus

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Acute pancreatitis (AP) is rarely seen in the paediatric population and is typically not associated with those aetiologies seen in adult pancreatitis. This case describes a 12-year-old female who presented with acute abdominal pain and constipation, with biochemical evidence of elevated serum amylase, calcium (Ca) and parathyroid hormone (PTH) levels. A diagnosis of AP was made, which was settled with conservative management. Further investigations, namely CT and technetium 99m (Tc-99m) sestamibi scans, revealed a solitary parathyroid adenoma. She subsequently underwent minimally invasive parathyroidectomy (MIP), following which Ca and PTH levels normalized postoperatively.

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Section: Discussioncontrasting

confidence: 51%

Minimally Invasive Parathyroidectomy in a Child With Acute Pancreatitis

Ali¹,

Seetahal-Maraj²,

Roop³

et al. 2022

Cureus

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“…According to these parameters, the present patient could be diagnosed as PTCA prior to surgery. The rapid postoperative normalization of PTH levels and prolonged hypocalcemia of the present patient was most likely due to 'hungry bone syndrome', which is characterized by prolonged (>4 days following parathyroidectomy) hypocalcemia (serum calcium levels, <8.4 mg/dl) or severe primary hyperparathyroidism as a result of extensive remineralization of bone tissue (18). In consequence, the patient received medical treatment of calcium gluconate, vitamin D3 and calcitriol.…”

Section: Discussionmentioning

confidence: 71%

An unusual mediastinal parathyroid carcinoma coproducing PTH and PTHrP: A case report

et al. 2016

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Abstract. Parathyroid carcinoma (PTCA) is a rare disease, and ectopic PTCA is particularly rare. Parathyroid hormone-related protein (PTHrP) expression in PTCA has not been previously described in the relevant literature to the best of our knowledge. The present study reports a unique case with a mediastinal parathyroid carcinoma producing parathyroid hormone (PTH) and PTHrP. A 53-year-old man presented with hyperparathyroidism symptoms, including fatigue, chest pain, dizziness, muscular soreness, polyuria, night sweats and renal stones. However, neck ultrasound revealed no significantly abnormal thyroid or parathyroid nodules. Tc99m methoxyisobutylisonitrile (Tc99m-MIBI) scintigraphy scanning indicated an ectopic mediastinal parathyroid adenoma.

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“…The case series of 10 patients included 4 participants with HBS (a rate of 4/10), the one of 5 patients reported one case of HBS (a rate of 1/5), and another case series of 2 patients only identified one individual with HBS; thus, we conclude that 19 children and teenagers were diagnosed with HBS (on case report level of statistical evidence). In addition to the mentioned pediatric studies in PHPT which analyzed the issue of post-operatory HBS, there were a total of 251 young patients, the youngest being a 6-year-old [86][87][88][89][90][91][92][93][94][95][96][97][98][99][100][101][102][103] (Table 4).…”

Section: Discussionmentioning

confidence: 99%

“…Original case reports/series of pediatric PHPT analyzing post-PTX HBS; the display starts with the most recent publication[86][87][88][89][90][91][92][93][94][95][96][97][98][99][100][101][102][103].…”

mentioning

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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A case of adolescent giant parathyroid adenoma presenting multiple osteolytic fractures and postoperative hungry bone syndrome

Cited by 10 publications

References 17 publications

Minimally Invasive Parathyroidectomy in a Child With Acute Pancreatitis

Minimally Invasive Parathyroidectomy in a Child With Acute Pancreatitis

An unusual mediastinal parathyroid carcinoma coproducing PTH and PTHrP: A case report

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

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