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Atypical Parathyroid Adenoma Complicated with Protracted Hungry Bone Syndrome after Surgery: A Case Report and Literature Review.

Juárez-León ÓA, Gómez-Sámano MÁ, Cuevas-Ramos D, Almeda-Valdés P, López-Flores A La Torre MA, Reza-Albarrán AA, Gómez-Pérez FJ - Case Rep Endocrinol (2015)

Bottom Line: Bone densitometry showed decreased Z-Score values (total lumbar Z-Score of -4.2).Therefore we propose the term "Protracted HBS" in patients with particularly long recovery of 1 year.We present a literature review of the diagnosis, pathophysiology, and treatment of HBS.

View Article: PubMed Central - PubMed

Affiliation: Endocrinology and Metabolism Department, Instituto Nacional de Ciencias Médicas y Nutrición Salvador Zubirán, 14080 Mexico City, Mexico.

ABSTRACT
Hungry Bone Syndrome refers to the severe and prolonged hypocalcemia and hypophosphatemia, following parathyroidectomy in patients with hyperparathyroidism. We present the case of an eighteen-year-old woman with a four-year history of hyporexia, polydipsia, weight loss, growth retardation, and poor academic performance. The diagnostic work-up demonstrated primary hyperparathyroidism with hypercalcemia of 13.36 mg/dL, a PTH level of 2551 pg/mL, bone brown tumors, and microcalcifications within pancreas and kidneys. Neck ultrasonography revealed a parathyroid adenoma of 33 × 14 × 14 mm, also identified on (99)Tc-sestamibi scan. Bone densitometry showed decreased Z-Score values (total lumbar Z-Score of -4.2). A right hemithyroidectomy and right lower parathyroidectomy were performed. Pathological examination showed an atypical parathyroid adenoma, of 3.8 g of weight and 2.8 cm in diameter. After surgery she developed hypocalcemia with tetany and QTc interval prolongation. The patient required 3 months of oral and intravenous calcium supplementation due to Hungry Bone Syndrome (HBS). After 42 months, she is still under oral calcium. Usually HBS lasts less than 12 months. Therefore we propose the term "Protracted HBS" in patients with particularly long recovery of 1 year. We present a literature review of the diagnosis, pathophysiology, and treatment of HBS.

No MeSH data available.


Related in: MedlinePlus

(a) Corrected serum calcium during hospitalization and as outpatient. (b) Serum phosphate values during hospitalization and as outpatient. (c) Serum magnesium values during hospitalization and as outpatient. ∗Gray area represents reference values. ∗∗Vertical dotted line represents treatment beginning, which continued beyond last medical assessment at our institution.
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fig4: (a) Corrected serum calcium during hospitalization and as outpatient. (b) Serum phosphate values during hospitalization and as outpatient. (c) Serum magnesium values during hospitalization and as outpatient. ∗Gray area represents reference values. ∗∗Vertical dotted line represents treatment beginning, which continued beyond last medical assessment at our institution.

Mentions: Postsurgical laboratory analysis showed PTH values of 31.3 pg/mL, serum calcium of 8.5 mg/dL, phosphate of 1.9 mg/dL, and magnesium of 1.8 mg/dL; on physical examination she presented upper extremity distal contractures, oromandibular dystonia, Chvostek and Trousseau signs, and QTc interval prolongation. PTH levels reached up to 48.6 pg/mL after 1 month, coexisting with hypophosphatemia of 2.7 mg/dL. At last follow-up, PTH serum levels were between 80 and 90 pg/mL, Table 2. She developed a prolonged and severe HBS that required 3 months with oral and intravenous calcium supplementation. The calcium IV infusion was stopped three months later. High PTH levels with hypocalcemia but also hypophosphatemia ruled out hypoparathyroidism. “Protracted” HBS was therefore diagnosed. During her hospitalization she underwent two episodes of lithotripsy as treatment for the kidney stones. Serum calcium, phosphate, and magnesium levels during hospitalization and follow-up are shown in Figure 4.


Atypical Parathyroid Adenoma Complicated with Protracted Hungry Bone Syndrome after Surgery: A Case Report and Literature Review.

Juárez-León ÓA, Gómez-Sámano MÁ, Cuevas-Ramos D, Almeda-Valdés P, López-Flores A La Torre MA, Reza-Albarrán AA, Gómez-Pérez FJ - Case Rep Endocrinol (2015)

(a) Corrected serum calcium during hospitalization and as outpatient. (b) Serum phosphate values during hospitalization and as outpatient. (c) Serum magnesium values during hospitalization and as outpatient. ∗Gray area represents reference values. ∗∗Vertical dotted line represents treatment beginning, which continued beyond last medical assessment at our institution.
© Copyright Policy
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4660009&req=5

fig4: (a) Corrected serum calcium during hospitalization and as outpatient. (b) Serum phosphate values during hospitalization and as outpatient. (c) Serum magnesium values during hospitalization and as outpatient. ∗Gray area represents reference values. ∗∗Vertical dotted line represents treatment beginning, which continued beyond last medical assessment at our institution.
Mentions: Postsurgical laboratory analysis showed PTH values of 31.3 pg/mL, serum calcium of 8.5 mg/dL, phosphate of 1.9 mg/dL, and magnesium of 1.8 mg/dL; on physical examination she presented upper extremity distal contractures, oromandibular dystonia, Chvostek and Trousseau signs, and QTc interval prolongation. PTH levels reached up to 48.6 pg/mL after 1 month, coexisting with hypophosphatemia of 2.7 mg/dL. At last follow-up, PTH serum levels were between 80 and 90 pg/mL, Table 2. She developed a prolonged and severe HBS that required 3 months with oral and intravenous calcium supplementation. The calcium IV infusion was stopped three months later. High PTH levels with hypocalcemia but also hypophosphatemia ruled out hypoparathyroidism. “Protracted” HBS was therefore diagnosed. During her hospitalization she underwent two episodes of lithotripsy as treatment for the kidney stones. Serum calcium, phosphate, and magnesium levels during hospitalization and follow-up are shown in Figure 4.

Bottom Line: Bone densitometry showed decreased Z-Score values (total lumbar Z-Score of -4.2).Therefore we propose the term "Protracted HBS" in patients with particularly long recovery of 1 year.We present a literature review of the diagnosis, pathophysiology, and treatment of HBS.

View Article: PubMed Central - PubMed

Affiliation: Endocrinology and Metabolism Department, Instituto Nacional de Ciencias Médicas y Nutrición Salvador Zubirán, 14080 Mexico City, Mexico.

ABSTRACT
Hungry Bone Syndrome refers to the severe and prolonged hypocalcemia and hypophosphatemia, following parathyroidectomy in patients with hyperparathyroidism. We present the case of an eighteen-year-old woman with a four-year history of hyporexia, polydipsia, weight loss, growth retardation, and poor academic performance. The diagnostic work-up demonstrated primary hyperparathyroidism with hypercalcemia of 13.36 mg/dL, a PTH level of 2551 pg/mL, bone brown tumors, and microcalcifications within pancreas and kidneys. Neck ultrasonography revealed a parathyroid adenoma of 33 × 14 × 14 mm, also identified on (99)Tc-sestamibi scan. Bone densitometry showed decreased Z-Score values (total lumbar Z-Score of -4.2). A right hemithyroidectomy and right lower parathyroidectomy were performed. Pathological examination showed an atypical parathyroid adenoma, of 3.8 g of weight and 2.8 cm in diameter. After surgery she developed hypocalcemia with tetany and QTc interval prolongation. The patient required 3 months of oral and intravenous calcium supplementation due to Hungry Bone Syndrome (HBS). After 42 months, she is still under oral calcium. Usually HBS lasts less than 12 months. Therefore we propose the term "Protracted HBS" in patients with particularly long recovery of 1 year. We present a literature review of the diagnosis, pathophysiology, and treatment of HBS.

No MeSH data available.


Related in: MedlinePlus