Three Rare Concurrent Complications of Tertiary Hyperparathyroidism: Maxillary Brown Tumor, Uremic Leontiasis Ossea, and Hungry Bone Syndrome

Bransky, Natalie; Iyer, Neena R.; Cannon, Sophie M; Tyan, Alexander; Mylavarapu, Praneet; Orosco, Ryan K.; Hom, David Brain; Moazzam, Alan A.

doi:10.11005/jbm.2020.27.3.217

Cited by 10 publications

(6 citation statements)

References 77 publications

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“…With maxillary bone brown tumour involvement, the bone lesion usually regresses after parathyroidectomy. However, in some instances, the bone brown tumours can continue to grow despite parathyroidectomy as was the case with our patient [16]. Radiographically, resorption of the lamina dura around the roots of the teeth and demineralisation of the medullary bones of the jaws causing a characteristic "ground glass" appearance [17] as we also observed.…”

Section: Discussionsupporting

confidence: 79%

A Rare Presentation of a Bi-Maxillary Brown Tumour due to Secondary Hyperparathyroidism

Cho-Fon

Ngokwe

Ines

2023

Case Reports in Dentistry

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Brown tumours are localized bone lesions, seen in patients with high parathyroid hormone levels. This can be due to primary hyperparathyroidism, which occurs more often in neoplasms of the parathyroid gland or due to secondary hyperparathyroidism more often seen as a result of renal impairment. Facial involvement is rare, with most reports focusing on long and axial bones. However, the mandibular bone is often the only bone affected. Here, we report a rare case of a bi-maxillary attainment of brown tumour in a patient with secondary hyperparathyroidism due to chronic kidney disease.

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Section: Discussionsupporting

confidence: 79%

A Rare Presentation of a Bi-Maxillary Brown Tumour due to Secondary Hyperparathyroidism

Cho-Fon

Ngokwe

Ines

2023

Case Reports in Dentistry

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“…According to the National Kidney Foundation's Kidney Disease Quality Outcomes Initiative (KDOQI), parathyroidectomy should be considered for patients with severe HPT (PTH >800 pg/mL) who are unresponsive to medical interventions and have concurrent hypercalcemia and/or hyperphosphatemia. (8) In cases where the brown tumor causes anatomic complications such as facial pain or compromised vision, hearing, chewing, speaking, and breathing, surgical excision of the lesion may be a viable treatment option to consider (8).…”

Section: Discussionmentioning

confidence: 99%

Brown tumor of multiple facial bones associated with tertiary hyperparathyroidism in a patient on maintenance hemodialysis

Nezha El-bouhi,

Halima Sobhi,

Meriem Chettati

et al. 2023

GSC Adv. Res. Rev.

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“…A part from specific peri-operatory protocols of electrolytes surveillance and intervention, we should mention that certain approaches are a matter of individual decision, especially unexpected dramatic situations as, for instance, uncontrolled HBS-associated hypocalcaemia; for example, the off label use of teriparatide [136,137]. A case of tertiary HPT-associated maxillary brown tumor in USA was reported in 2020.…”

Section: Study Design Studied Populationmentioning

confidence: 99%

“…A case of tertiary HPT-associated maxillary brown tumor in USA was reported in 2020. The 48-year-old female developed HBS with severe post-PTx hypocalcemia that turned out refractory to high calcium regimes (2.5 g every 3 h) and calcitriol (2 µg twice per day) regimes, and increased hemodialysis (a total of 12 g of intravenous calcium gluconate was necessary to maintain a total serum calcium between 7 and 8 mg/dL) and teriparatide was offered (20 µg twice per day) starting with post-operatory day 25 (that was continued for one month when she was discharged with a serum calcium of 11 mg/dL) [136]. A similar case (also published in 2020) is represented by a 35-year-old woman who developed HBS following PTx for RHPT; her severe hypocalcemia persisted for 8 months remaining refractory to a standard approach, thus teriparatide was introduced (20 µg/day for the first 7 days followed by 20 µg/day, 3 times/week after dialysis) and continued for one month [137].…”

Section: Study Design Studied Populationmentioning

confidence: 99%

“…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.…”

Section: Study Design Studied Populationmentioning

confidence: 99%

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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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Three Rare Concurrent Complications of Tertiary Hyperparathyroidism: Maxillary Brown Tumor, Uremic Leontiasis Ossea, and Hungry Bone Syndrome

Cited by 10 publications

References 77 publications

A Rare Presentation of a Bi-Maxillary Brown Tumour due to Secondary Hyperparathyroidism

A Rare Presentation of a Bi-Maxillary Brown Tumour due to Secondary Hyperparathyroidism

Brown tumor of multiple facial bones associated with tertiary hyperparathyroidism in a patient on maintenance hemodialysis

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

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