Hiperparatireoidismo associado à osteomalacia hipofosfatêmica do adulto: relato de caso e revisão da literatura

Moreira, Rodrigo O.; Leal, Christianne Toledo Souza; Lacativa, Paulo Gustavo Sampaio; Figueiredo, João Guilherme; Lima, Maurício B.; Farias, Maria Lúcia Fleiuss

doi:10.1590/s0004-27302006000100021

Cited by 4 publications

(1 citation statement)

References 16 publications

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“…As queixas de fraqueza muscular, na OOH, são mais intensas, assim como a presença de fratura e a dor óssea são mais freqüentes e graves (15)(16)(17).…”

Section: Discussionunclassified

Osteomalácia oncogênica: cintilografia com sestamibi-99mTc na localização do tumor

BIAGINI

Coutinho

Jonasson

et al. 2008

Arq Bras Endocrinol Metab

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A osteomalácia oncogênica hipofosfatêmica (OOH) é uma síndrome paraneoplásica induzida por tumor, de tecidos mole ou ósseo. Apresenta-se com dor e fraturas, acompanhada de hipofosfatemia, hiperfosfatúria e concentrações plasmáticas de 1,25(OH) 2 D 3 inapropriadamente normais/diminuídas. Após a remoção do tumor, a completa resolução das anormalidades clínicas e bioquímicas é sua maior característica. Uma mulher de 44 anos de idade é descrita no caso com difi culdade para caminhar por causa de dores nos membros inferiores, fraqueza muscular generalizada e hipofosfatemia com relativa hiperfosfatúria. A cintilografi a de corpo total com sestamibi99m Tc mostrou acúmulo do radiofármaco no terço superior de coxa esquerda onde pequeno tumor foi detectado no exame pelo ultra-som. Com a retirada do tumor, um lipoma, os sintomas melhoraram após um mês, com recuperação completa ao redor do quarto mês. Neste caso, a cintilografi a de corpo inteiro com sestamibi99m Tc foi decisiva na localização do tumor causador da osteomalácia oncogênica. Oncogenic osteomalacia is a paraneoplastic syndrome usually induced by bone or soft tissue tumors. It is presented by the development of pain and fractures with hypophosphatemia, hyperphosphaturia, and inappropriate normal/low plasma 1,25(OH) 2 D 3 concentration. After the removal of the tumor the complete resolution of all biochemical and clinical abnormalities is the main characteristic. A case of a 44-year-old female with diffi culty in walking due to leg pain and generalized muscle weakness and hypophosphatemia, with relative hyperphosphaturia, is described. A whole-body 99m Tc-sestamibi scintigraphy showed accumulation in the left thigh region, and a small tumor was detected by ultrasound examination. By removal of the tumor, a lipoma, the symptoms improved signifi cantly after a month, with complete recovery by the fourth month. In this case, 99m Tc-sestamibi scintigraphy was useful in identifying the location of the tumor, which caused oncogenic osteomalacia.

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“…As queixas de fraqueza muscular, na OOH, são mais intensas, assim como a presença de fratura e a dor óssea são mais freqüentes e graves (15)(16)(17).…”

Section: Discussionunclassified

Osteomalácia oncogênica: cintilografia com sestamibi-99mTc na localização do tumor

BIAGINI

Coutinho

Jonasson

et al. 2008

Arq Bras Endocrinol Metab

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Tumor-induced osteomalacia

Yin

et al. 2018

Osteoporosis and Sarcopenia

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Tumor-induced osteomalacia (TIO), also known as oncogenic osteomalacia, is a rare paraneoplastic syndrome characterized by hypophosphatemia resulting from decreased tubular phosphate reabsorption, with a low or inappropriately normal level of active vitamin D. The culprit tumors of TIO could produce fibroblast growth factor 23 which plays a role in regulating renal Pi handling and 25-hydroxyvitamin D 1α-hydroxylase activity. Chronic hypophosphatemia could eventually lead to inadequate bone mineralization, presenting as osteomalacia. The diagnosis should be considered when patients manifest as hypophosphatemia and osteomalacia, or rickets and needs to be differentiated from other disorders of phosphate metabolism, such as the inhereditary diseases like X-linked hypophosphataemic rickets, autosomal dominant hypophosphataemic rickets, autosomal recessive hypophosphataemic rickets and acquired diseases like vitamin D deficiency. Localization of responsible tumors could be rather difficult since the vast majority are very small and could be everywhere in the body. A combination of thorough physical examination, laboratory tests and imaging techniques should be applied and sometimes a venous sampling may come into handy. The technology of somatostatin-receptor functional scintigraphy markedly facilitates the localization of TIO tumor. Patients undergoing complete removal of the causative neoplasm generally have favorable prognoses while a few have been reported to suffer from recurrence and metastasis. For those undetectable or unresectable cases, phosphate supplements and active vitamin D should be administrated and curative intended radiotherapy or ablation is optional.

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Adult-onset hypophosphatemic osteomalacia associated with Sjogren syndrome

Shen

Zhang²,

Hu³

et al. 2017

Medicine

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Rationale:Hypophosphatemic osteomalacia (HO) is a metabolic bone disease, exhibiting different etiologies such as genetic mutation, tumor induction, dysimmunity, or renal disease. Sjogren's syndrome (SS) is a connective tissue disorder commonly involving exocrine glands; however kidney involvement is also encountered, leading to abnormal phosphorus metabolism, even HO.Patient concerns:A 47-year-old female patient presented progressively worsening pain in the chest wall, back and bilateral lower extremities as well as muscle weakness was referred to our department.Diagnoses, interventions and outcomes:Due to the laboratory test results, radiographic findings and pathologic results, she was diagnosed with adult-onset HO associated with SS. She was then treated with alkalinization, steroids, neutral phosphate, calcium supplements together with activated vitamin D. So far, she recovered uneventfully with relieved pain and increased serum phosphorus level.Lessons:HO may be secondary to renal tubular acidosis of SS patients, and it might be a diagnostic challenge when the kidney involvement in SS is latent and precede the typical sicca symptoms.

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Hiperparatireoidismo associado à osteomalacia hipofosfatêmica do adulto: relato de caso e revisão da literatura

Cited by 4 publications

References 16 publications

Osteomalácia oncogênica: cintilografia com sestamibi-99mTc na localização do tumor

Osteomalácia oncogênica: cintilografia com sestamibi-99mTc na localização do tumor

Tumor-induced osteomalacia

Adult-onset hypophosphatemic osteomalacia associated with Sjogren syndrome

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