دورية أكاديمية
Multiple brown tumors: a bone complication due to long-term untreated pseudohypoparathyroidism.
| العنوان: | Multiple brown tumors: a bone complication due to long-term untreated pseudohypoparathyroidism. |
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| المؤلفون: | Gonnelli S; Université de Paris-APHP.Centre, Service de Rhumatologie, Hôpital Cochin, Centre de Référence des Maladies Rares du Métabolisme Phospho-calcique, Paris, France., Briot K; Université de Paris-APHP.Centre, Service de Rhumatologie, Hôpital Cochin, Centre de Référence des Maladies Rares du Métabolisme Phospho-calcique, Paris, France., Cormier C; Université de Paris-APHP.Centre, Service de Rhumatologie, Hôpital Cochin, Centre de Référence des Maladies Rares du Métabolisme Phospho-calcique, Paris, France., Teboul S; Université de Paris-APHP.Centre, Service de Rhumatologie, Hôpital Cochin, Centre de Référence des Maladies Rares du Métabolisme Phospho-calcique, Paris, France., Roux C; Université de Paris-APHP.Centre, Service de Rhumatologie, Hôpital Cochin, Centre de Référence des Maladies Rares du Métabolisme Phospho-calcique, Paris, France., Koumakis E; Université de Paris-APHP.Centre, Service de Rhumatologie, Hôpital Cochin, Centre de Référence des Maladies Rares du Métabolisme Phospho-calcique, Paris, France. eugenie.koumakis@aphp.fr.; INSERM UMR 1163, Imagine Institute, Paris, France. eugenie.koumakis@aphp.fr. |
| المصدر: | Osteoporosis international : a journal established as result of cooperation between the European Foundation for Osteoporosis and the National Osteoporosis Foundation of the USA [Osteoporos Int] 2024 Jan; Vol. 35 (1), pp. 195-199. Date of Electronic Publication: 2023 Aug 29. |
| نوع المنشور: | Case Reports; Journal Article |
| اللغة: | English |
| بيانات الدورية: | Publisher: Springer International Country of Publication: England NLM ID: 9100105 Publication Model: Print-Electronic Cited Medium: Internet ISSN: 1433-2965 (Electronic) Linking ISSN: 0937941X NLM ISO Abbreviation: Osteoporos Int Subsets: MEDLINE |
| أسماء مطبوعة: | Original Publication: London, UK : Springer International, c1990- |
| مواضيع طبية MeSH: | Osteitis Fibrosa Cystica*/complications , Pseudohypoparathyroidism*/complications , Hyperparathyroidism*/complications , Bone Diseases* , Neoplasms*, Choline/*analogs & derivatives, Humans ; Adult ; Female ; Child ; Adolescent ; Calcium/therapeutic use ; Positron Emission Tomography Computed Tomography ; Parathyroid Hormone ; Vitamins ; Vitamin D/therapeutic use |
| مستخلص: | Bone lytic lesions are a possible complication of pseudohypoparathyroidism type 1B, in undertreated adult patients. Whole body [18F] F-fluorocholine PET/CT is a useful imaging tool to assess brown tumor progression in this context. We describe the case of a 33-year-old woman, referred for the diagnostic evaluation of lytic bone lesions of the lower limbs, in the context of asymptomatic pseudohypoparathyroidism. She had been treated with alfacalcidol and calcium during her childhood. Treatment was discontinued at the age of 18 years old because of the lack of symptoms. A femur biopsy revealed a lesion rich in giant cells, without malignancy, consistent with a brown tumor. Laboratory tests showed a parathyroid level at 1387 pg/ml (14-50). Whole-body Fluorocholine PET/CT revealed hypermetabolism of bone lesions. The final diagnosis was brown tumors related to hyperparathyroidism complicating an untreated pseudohypoparathyroidism. Genetic testing confirmed PHP type 1B. Pseudohypoparathyroidism with radiographic evidence of hyperparathyroid bone disease, is a very rare condition due to parathyroid hormone resistance in target organs, i.e., kidney resistance, but with conserved bone cell sensitivity. It has been reported in only a few cases of pseudohypoparathyroidism type Ib. Long-term vitamin D treatment was required to correct bone hyperparathyroidism. With this rationale, the patient was treated with calcium, alfacalcidol, and cholecalciferol. One-year follow-up showed complete resolution of pain, improvement in serum calcium, and regression of bone lesions on [18F]F-fluorocholine PET/CT. This case illustrates the usefulness of [18F]F-fluorocholine PET/CT for the imaging of brown tumors in pseudohypoparathyroidism type 1B, and emphasizes the importance of calcium and vitamin D treatment in adult patients, to avoid the deleterious effects of high parathyroid hormone on skeletal integrity. (© 2023. International Osteoporosis Foundation and Bone Health and Osteoporosis Foundation.) |
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| فهرسة مساهمة: | Keywords: Brown tumor; Pseudohypoparathyroidism; fluorocholine; hyperparathyroidism |
| المشرفين على المادة: | SY7Q814VUP (Calcium) 6029HGL0QP (fluorocholine) 0 (Parathyroid Hormone) 0 (fluoromethylcholine) 0 (Vitamins) 1406-16-2 (Vitamin D) N91BDP6H0X (Choline) |
| تواريخ الأحداث: | Date Created: 20230829 Date Completed: 20240115 Latest Revision: 20240115 |
| رمز التحديث: | 20240115 |
| DOI: | 10.1007/s00198-023-06878-5 |
| PMID: | 37644196 |
| قاعدة البيانات: | MEDLINE |
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Full Text Finder | تدمد: | 1433-2965 |
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| DOI: | 10.1007/s00198-023-06878-5 |