Carsote, Mara

Nistor, Claudiu

Via Medica Journals

641CLINICAL VIGNETTEing hypercalcemia through intravenous hydration and di-uretics, a right inferior parathyroidectomy was performed (pathological exam: parathyroid adenoma of 3.4×3.2 cm, with principal cells, areas of haemorrhage and oste-oid metaplasia, intact capsule, no atypia). She was early released with high-normal total calcium of 10.2 mg/dL and offered oral calcium (1 g/day) and vitamin D (alpha-calcidol 1 µg/day and cholecalciferol 2000 UI/day). She was readmitted a few days later after experiencing diffuse bone pain, and paraesthesia which correlated with acute hypo-A 56-year-old female was admitted as an emergency for acute hypercalcaemia. Extremely high parathormone (PTH) with intact kidney function were consistent with primary hyperparathyroidism (PHP) (Tab. 1). She described bone pain and chronic asthenia for 2 years. She had arterial hypertension and entered menopause at 53. The family medical history was irrelevant. Imaging assessments pointed out a 4-cm inferior right parathyroid tumour (Fig. 1). No elements of multiple endocrine neo-plasia were identified, nor kidney stones. After manag-From acute hypercalcaemia to post-parathyroidectomy hungry bone syndrome in a patient with primary hyperparathyroidism-associated brown tumours Mara Carsote 1, Claudiu Nistor 21Department of Endocrinology, Carol Davila University of Medicine and Pharmacy & C.I. Parhon National Institute of Endocrinology, Bucharest, Romania2Department 4, Cardio-Thoracic Pathology, Thoracic Surgery II Discipline, Carol Davila University of Medicine and Pharmacy & Thoracic Surgery Department, Dr. Carol Davila Central Emergency University Military Hospital, Bucharest, RomaniaCliniCal vignetteEndokrynologia PolskaDOI: 10.5603/ep.96134ISSN 0423–104X, e-ISSN 2299–8306Volume/Tom 74; Number/Numer 6/2023Submitted: 21.06.2023Accepted: 24.07.2023Early publication date: 05.12.2023Mara Carsote, Aviatorilor Ave 34-38, 011863, Bucharest, Sector 1, Romania, +40 744 851 934; e-mail: carsote_m@hotmail.comThis article is available in open access under Creative Common Attribution-Non-Commercial-No Derivatives 4.0 International (CC BY-NC-ND 4.0) license, allowing to download articles and share them with others as long as they credit the authors and the publisher, but without permission to change them in any way or use them commerciallyKey words: hungry bone syndrome; brown tumours; primary hyperparathyroidism; parathyroidectomy; anaemia; acute hypercalcaemiaTable 1. Biochemistry and hormonal assaysParameter Units On admission Second admission*1-month check-up Normal values UnitsTotal serum Ca mg/dL 14.76 5.6 (6.9**) 7.29 8.5–10.2 mg/dLTotal ionic Ca mg/dL 5.63 2.82 3 3.9–4.9 mg/dLSerum P mg/dL 2.3 4 3.18 2.5–4.5 mg/dLSerum Mg mg/dL 1.9 1.6 1.9 1.6–2.4 mg/dLPTH pg/mL 1109 70 71 15–65 pg/mL25OHD ng/mL 11 12.8 15.9 30–100 ng/mL24-h Ca ng/mL 0.9 NA 0.01 0.07–0.3 ng/mLAP U/L 660 492 309 38–105 U/LOsteocalcin ng/dL > 300 > 300 160 15–46 ng/dLP1NP ng/dL NA 408.8 400 20.25–76.3 ng/dLCrossLaps ng/mL 5.07 0.96 1.4 0.33–0.782 ng/mLHaemoglobin g/dL 8.3 (9.3***) 7.3 6.7 12–15.5 g/dLAP — total alkaline phosphatase; Ca — calcium; 24-h — 24-hour urinary calcium; Mg — magnesium; P — phosphorus; PTH — parathyroid hormone; 25OHD — 25-hydroxyvitamin D; NA — not available; blood bone turnover markers of formation (AP, osteocalcin, and P1NP) and of resorption (CrossLaps); blue — diagnosis HBS: symptomatic hypocalcaemia starting from the 4–5th post-operatory day and confirmed at lab assays, mild hypomagnesaemia, but normal phosphorus with  high-normal PTH that excluded post-operatory hypoparathyroidism; probably with a mild component of hypovitaminosis D-related secondary hyperparathyroidism; *10 days since parathyroidectomy;**the second value was achieved after a 10-day hospitalization; ***after 2 U of red blood cells mass transfusion before surgery642Hungry bone syndrome and brown tumours Mara Carsote, Claudiu NistorCLINICAL VIGNETTElonger disease duration, increased pre-surgery PTH, large parathyroid adenomas, and D hypovitaminosis. Their adequate recognition in relationship with a para-thyroid condition avoids unnecessary bone biopsy, to exclude a malignancy, although a certain index of suspi-cion should be kept in mind and long-term surveillance is needed until their resolution. Remarkably, the patient presented severe hypochromic microcytic hyposider-emic anaemia that required 2 units of red blood cells before surgery and long-term oral iron supplementation. A complete haematological exam was done, which found no other cause; no suspected malignancies, such as mul-tiple myeloma that might mimic bone lesions, were con-firmed. The potential mechanisms of anaemia in PHP are related to the following pathogenic traits: it represents an element of chronic illness, including calcium-induced loss of appetite and stomach pain, PTH-induced focal fibrosis of the marrow space, direct PTH inhibition of erythrocytes, increased peripheral calcium-mediated destruction of red cells, and (not in this case) chronic kidney failure either as a complication of a primary or renal hyperparathyroidism. Take-home message: excep-tional presentations of PHP might be found; unusual complications and outcome make awareness mandatory even in the modern era, whereas the typical frame of the condition has shifted to a mild, rather asymptomatic disease with a good overall prognosis.Ethics statementThe research was conducted ethically in accordance with the World Medical Association Declaration of Helsinki. Data were collected retrospectively.Author contributionsConceptualization, methodology, writing, review and editing, and supervision: M.C. and C.N. AcknowledgmentsWe thank the patient and the entire medical and surgical teams.Conflict of interestThe authors declare no conflict of interest.calcaemia and hungry bone syndrome (HBS). Intensive intravenous calcium replacements (2–4 g/day) and oral calcitriol (1 g/day) were continued with cholecalciferol 2000 UI/day (total calcium increased from 5.6 to 6.9 mg/dL after a 10-day hospitalization). Due to bone complains an X-ray exam was done and confirmed by computed tomography (CT): 3 bone lesions at ribs and clavicle with cortical disruption (brown tumours) (Fig. 2). Osteopo-rosis was confirmed by DXA — lowest T score of –3.5. She was discharged with oral calcium and vitamin D replacements, with progressive clinical improvement as well as one-month blood assays, including previ-ously high bone turnover markers. Monthly risendronate (75 mg × 2/month) was added. Long-term surveillance is needed; a resolution of brown tumours is expected.  Post-parathyroidectomy HBS is reported in 15–20% (if any) of cases with PHP; a higher risk is described in cases with elevated pre-operatory serum calcium, PTH (especially >1000 pg/mL) and alkaline phosphatase (mostly, 6 times upper normal limits), and, probably, low 25-hydroxyvitamin D, brown tumours, anaemia, large parathyroid tumours, and osteoid metaplasia (functional aspect of long-standing disease). Brown tumours, part of severe traditional bone complications in PHP, represent an exceptional event nowadays, being associated with Figure 2. Thorax X-Ray: brown tumours at the level of 2nd and 3rd left ribs (1.8 cm), 4th, 5th, and 6th right ribs (4 cm) and 1/3 external left clavicle (3 cm)Figure 1. CT: right later-cervical tumour of 2.4 × 4 cm (oval, well-shaped parathyroid mass; heterogeneous consistency, next to the right thyroid lobe, with mass effect on the right jugular vein and carotid artery. Transversal plan (left). Coronal plan (right)

From acute hypercalcaemia to post-parathyroidectomy hungry bone syndrome in a patient with primary hyperparathyroidism-associated brown tumours

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From acute hypercalcaemia to post-parathyroidectomy hungry bone syndrome in a patient with primary hyperparathyroidism-associated brown tumours

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