Supranumerary or ectopic parathyroid glands are the main cause of persistent hyperparathyroidism (HPT) in patients with end stage renal disease (ESRD) submitted to parathyroidectomy (PTx). PURPOSE: To evaluate the prevalence and location of parathyroid glands in these patients. METHODS: Thirty-five patients with ESRD and severe secondary hyperparathyroidism (HPT2) had been submitted to total PTx at HUCFF from December 2001 to July 2005. Surgery was always performed by the same surgeon, who described in details the location of the glands. RESULTS: Sixteen patients (45.7%) had ectopic glands, which were also extranumerary in five of them (14.3%). The most common locations were the thyroid parenchyma (33.3%), thyroid-thymus conduit (18.5%), and thymus (14.8%). Before PTx, the sensibility of ultrasonography and scintigraphy with technetium-99m Sestamibi was low (48.3% and 35.3%, respectively). Moreover, 51.4% of the nodules found at US were thyroid nodules. However, 99mTc-Sestamibi was useful to identify ectopic glands in those two patients with persistent HPT after PTx. CONCLUSION: The presence of extranumerary and ectopic parathyroid glands in HPT2 is sufficiently important to justify their exhaustive search. As the preoperative image exams present low sensibility to locate them, it is necessary to develop an exploratory routine embracing the most common sites of location.
Brown tumors are relatively uncommon but they are serious complications of renal osteodystrophy. We describe a 31-year-old woman with end-stage renal disease who had undergone hemodialysis for nine years and developed severe secondary hyperparathyroidism and a maxilla brown tumor despite increasing doses of oral calcitriol and calcium carbonate. The fast increase of the right maxillary bone tumor led to indication of parathyroidectomy (PTx). Despite normalization of serum PTH there was a slow regression of the mass and the patient still complained about her appearance after two-years of follow-up. Excision of the maxillary mass followed by recontouring of the maxilla was then performed, with adequate masticator rehabilitation. Tumores marrons são relativamente incomuns mas constituem sérias complicações da osteodistrofia renal. Descrevemos o caso de uma paciente, 31 anos, com doença renal em estágio terminal, em hemodiálise há nove anos, que desenvolveu severo hiperparatireoidismo secundário com tumor marrom em maxila apesar de doses crescentes de calcitriol oral e carbonato de cálcio. O rápido aumento do tumor marrom em maxila levou à indicação de paratireoidectomia (PTx). Apesar da normalização dos níveis de PTH sérico, a regressão da massa tumoral foi lenta e a paciente questionava sobre sua aparência após dois anos de seguimento. A excisão da massa maxilar foi seguida da reconstrução com adequada função mastigatória. BROWN TUMORS ARE EROSIVE bony lesions caused by rapid osteoclastic activity and peritrabecular fibrosis due to hyperparathyroidism (HPT) resulting in a local destructive phenomenon. Actually they represent a reparative cellular process rather than a true neoplasia (1,2). They are known to occur only in the setting of HPT, and are considered the most pathognomonic skeletal changes that accompany this disease. For years, these lesions have been recognized in primary hyperparathyroidism (HPT1) (3). However, brown tumors have also been reported in patients with severe hyperparathyroidism secondary to chronic renal failure (HPT2), especially those on long-term hemodialysis (4,5).
RESUMOA osteomalacia hipofosfatêmica é uma doença rara caracterizada por hipofosfatemia, níveis elevados de fosfatase alcalina e diminuição da densidade óssea. O tratamento é realizado com suplementação oral com fosfato e vitamina D e, nos casos de osteomalacia oncogênica, com a ressecção do tumor. Relatamos o caso de uma paciente que apresentou quadro de osteomalácia hipofosfatêmica de causa indeterminada. Apesar de extensivamente procurado, nenhum tumor produtor de substância hipofosfatêmica foi localizado. A paciente foi tratada como suplementação de fosfato e vitamina D por longo período, evoluindo com quadro de hiperparatireoidismo terciário. A retirada de três paratireóides não normalizou os níveis de PTH e a paciente recusouse a continuar a investigação e o tratamento. Após dez anos de tratamento irregular, foi internada por insuficiência respiratória causada por colabamento do arcabouço costal e múltiplas fraturas, evoluindo para o óbito. Os autores discutem a relação entre osteomalácia e hiperparatireoidismo e o curso agressivo da doença. Adult-onset hypophosphatemic osteomalacia is a rare disease characterized by hypophosphatemia, increased levels of alkaline phosphatase and decreased bone mass. Oral supplementation with phosphate and vitamin D is the main treatment and, in cases of oncogenic osteomalacia, tumor resection is mandatory. We report the case of a patient with hypophosphatemic osteomalacia of an unknown cause. Despite extensive search, no tumor was found. The patient was treated with phosphate for a long period and developed tertiary hyperparathyroidism. Serum PTH levels did not return to normal after surgical excision of three parathyroids and the patient refused to continue clinical investigation and treatment. After ten years absent from the hospital, during which medications were used irregularly, she was admitted with multiple fractures and respiratory insufficiency caused by severe thoracic deformities, and died. The authors discuss the relationship between osteomalacia and hyperparathyroidism and the aggressive course of the disease.
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