Brown tumor of the jaw after pregnancy and lactation in a MEN1 patient
| Autor: | Casteràs, Anna, Darder, Lídia, Zafón, Carles, Hueto Madrid, Juan Antonio, Alberola Ferranti, Margarita, Caubet, Enric, Mesa Manteca, Jordi, Universitat Autònoma de Barcelona. Departament de Medicina, Universitat Autònoma de Barcelona. Departament de Cirurgia, Universitat Autònoma de Barcelona. Departament de Ciències Morfològiques |
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| Rok vydání: | 2016 |
| Předmět: |
Parathyroidectomy
Insight into Disease Pathogenesis or Mechanism of Therapy medicine.medical_specialty Hyperparathyroidism lcsh:RC648-665 business.industry Endocrinology Diabetes and Metabolism medicine.medical_treatment Urology 030209 endocrinology & metabolism medicine.disease lcsh:Diseases of the endocrine glands. Clinical endocrinology vitamin D deficiency Bone resorption Surgery 03 medical and health sciences Brown tumor 0302 clinical medicine Hypoparathyroidism Internal Medicine medicine business 030217 neurology & neurosurgery Primary hyperparathyroidism Parathyroid adenoma |
| Zdroj: | Dipòsit Digital de Documents de la UAB Universitat Autònoma de Barcelona Endocrinology, Diabetes & Metabolism Case Reports, Vol 1, Iss 1, Pp 1-5 (2016) Endocrinology, Diabetes & Metabolism Case Reports |
| ISSN: | 2052-0573 |
| DOI: | 10.1530/edm-16-0111 |
| Popis: | Summary Skeletal manifestations of primary hyperparathyroidism (pHPT) include brown tumors (BT), which are osteoclastic focal lesions often localized in the jaws. Brown tumors are a rare manifestation of pHTP in Europe and USA; however, they are frequent in developing countries, probably related to vitamin D deficiency and longer duration and severity of disease. In the majority of cases, the removal of the parathyroid adenoma is enough for the bone to remineralize, but other cases require surgery. Hyperparathyroidism in MEN1 develops early, and is multiglandular and the timing of surgery remains questionable. To our knowledge, there are no reports of BT in MEN 1 patients. We present a 29-year-old woman with MEN 1 who developed a brown tumor of the jaw 24 months after getting pregnant, while breastfeeding. Serum corrected calcium remained under 2.7 during gestation, and at that point reached a maximum of 2.82 mmol/L. Concomitant PTH was 196 pg/mL, vitamin D 13.7 ng/mL and alkaline phosphatase 150 IU/L. Bone mineral density showed osteopenia on spine and femoral neck (both T-scores = −1.6). Total parathyroidectomy was performed within two weeks, with a failed glandular graft autotransplantation, leading to permanent hypoparathyroidism. Two months after removal of parathyroid glands, the jaw tumor did not shrink; thus, finally it was successfully excised. We hypothesize that higher vitamin D and mineral requirements during maternity may have triggered an accelerated bone resorption followed by appearance of the jaw BT. We suggest to treat pHPT before planning a pregnancy in MEN1 women or otherwise supplement with vitamin D, although this approach may precipitate severe hypercalcemia. Learning points: Brown tumors of the jaw can develop in MEN 1 patients with primary hyperparathyroidism at a young age (less than 30 years). Pregnancy and lactation might trigger brown tumors by increasing mineral and vitamin D requirements. Early parathyroidectomy is advisable in MEN 1 patients with primary hyperparathyroidism, at least before planning a pregnancy. Standard bone mineral density does not correlate with the risk of appearance of a brown tumor. Removal of parathyroid glands does not always lead to the shrinkage of the brown tumor, and surgical excision may be necessary. |
| Databáze: | OpenAIRE |
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