Nonfunctioning
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Prezentujeme případ 39-letého muže, který v roce 2013 prodělal thyreoidektomii s parathyreoidektomií následně s mylně diagnostikovaným medulárním karcinomem štítné žlázy. Štítnou žlázu i příštítná tělíska bylo obtížné exstirpovat pro obezitu pacienta a pro atypickou lokalizaci příštítného tělíska. Obě tyto struktury byly při operaci arteficiálně porušené. Tři roky po výkonu se u pacienta objevily v krčních měkkých tkáních uzly v lymfatických uzlinách suspektní z metastáz. Mikroskopický obraz však odpovídal uzlům parathyreomatózy a invazivnímu nádoru, který byl po doplnění klinických údajů a imunohistochemického vyšetření překlasifikován na nefunkční karcinom parathyreoidey. V článku diskutujeme morfologické i imunohistochemické znaky parathyreomatózy a karcinomu parathyreoidey a možné diagnostické omyly.
We report on the case of a 39-year old man who underwent a thyroidectomy and a parathyroidectomy with misdiagnosed medullary carcinoma of the thyroid in 2013. During the operation the thyroid gland and parathyroid glands were artificially damaged due to the complicated surgical access to the glands because of the obesity of the patient as well as the deep placement of the enlarged parathyroid glands. Three years later, the neck ultrasound showed bilateral nodules on the neck, suspected to be metastases of the medullary carcinoma. Microscopically, the nodules were found to be focuses of parathyromatosis, and there was also an infiltrating carcinoma. This lesion was reclassified after clinico-pathological correlation and immunohistochemical examination as nonfunctioning parathyroid carcinoma. This article discusses morphological and immunohistochemical features of parathyromatosis and parathyroid carcinoma and its separation from lesions with which it may be misdiagnosed.
- MeSH
- chybná diagnóza MeSH
- dospělí MeSH
- imunohistochemie MeSH
- krk diagnostické zobrazování patologie MeSH
- lidé MeSH
- lymfatické uzliny anatomie a histologie chirurgie patologie MeSH
- metastázy nádorů MeSH
- nádory příštítného tělíska * diagnóza terapie MeSH
- nádory štítné žlázy diagnóza terapie MeSH
- obezita MeSH
- paratyreoidektomie MeSH
- tyreoidektomie MeSH
- Check Tag
- dospělí MeSH
- lidé MeSH
- mužské pohlaví MeSH
- Publikační typ
- kazuistiky MeSH
BACKGROUND AND OBJECTIVES: Stereotactic radiosurgery (SRS) represents an effective treatment for nonfunctioning pituitary adenomas (NFPAs). However, no data have yet been published regarding results of SRS on NFPAs after multiple previous resections. METHODS: Retrospective multicentric data of patients diagnosed with NFPA and who underwent multiple resections (≥2) before SRS were reviewed and analyzed. The treatment interval spanned the period of 1992 to 2022. Cox regression and Kaplan-Meier curves were used to assess predictive factors and the probability of tumor control and hypopituitarism. RESULTS: Among the 311 patients (median age: 50.2 [IQR: 18.0] years), 226 (72.7%) had undergone ≥2 previous resections. The median margin dose was 14 Gy (IQR: 4.0 Gy), and the median tumor volume 3.6 cm 3 (IQR: 4.8). Overall, the probability of tumor control after SRS was 93.3% (CI 95%: 89.9-96.9) and 86.7% (CI 95%: 81.1-92.6) at 5 and 10 years, respectively. A margin dose >14 Gy was associated with a decreased risk of tumor progression (hazard ratio = 0.33, CI 95% = 0.15-0.75, P = .008). At a last clinical follow-up of 4.1 (IQR 6.1) years, 10.1% (30/296) developed at least 1 new hormone deficiency after SRS. The cumulative probability of new hormone deficiency was 6.1% (95% CI: 3.0-9.1), 10.3% (95% CI: 5.8-14.6), and 18.9% (95% CI: 11.5-25.8) at 3, 5, and 10 years after SRS, respectively. The average latency between SRS and development of new hormone deficiencies was 3.3 years (IQR 4.1). A maximum point dose to the pituitary stalk >10 Gy was associated with a new deficiency (hazard ratio = 4.06, CI 95% = 1.57-10.5, P -value = .004). CONCLUSION: For patients with NFPA with multiple previous resections, SRS offers effective local tumor control and a low risk of delayed hypopituitarism for managing these challenging adenomas. SRS should be strongly considered in patients with NFPA with 2 previous resections compared with considering a third resection.
- MeSH
- adenom * chirurgie radioterapie MeSH
- dospělí MeSH
- hypopituitarismus MeSH
- lidé středního věku MeSH
- lidé MeSH
- nádory hypofýzy * chirurgie radioterapie MeSH
- radiochirurgie * metody MeSH
- retrospektivní studie MeSH
- senioři MeSH
- výsledek terapie MeSH
- Check Tag
- dospělí MeSH
- lidé středního věku MeSH
- lidé MeSH
- mužské pohlaví MeSH
- senioři MeSH
- ženské pohlaví MeSH
- Publikační typ
- časopisecké články MeSH
- multicentrická studie MeSH
OBJECTIVE Gamma Knife radiosurgery (GKRS) is frequently used to treat residual or recurrent nonfunctioning pituitary macroadenomas. There is no consensus as to whether GKRS should be used early after surgery or if radiosurgery should be withheld until there is evidence of imaging-defined progression of tumor. Given the high incidence of adenoma progression after subtotal resection over time, the present study intended to evaluate the effect of timing of radiosurgery on outcome. METHODS This is a multicenter retrospective review of patients with nonfunctioning pituitary macroadenomas who underwent transsphenoidal surgery followed by GKRS from 1987 to 2015 at 9 institutions affiliated with the International Gamma Knife Research Foundation. Patients were matched by adenoma and radiosurgical parameters and stratified based on the interval between last resection and radiosurgery. Operative results, imaging data, and clinical outcomes were compared across groups following early (≤ 6 months after resection) or late (> 6 months after resection) radiosurgery. RESULTS After matching, 222 patients met the authors' study criteria (from an initial collection of 496 patients) and were grouped based on early (n = 111) or late (n = 111) GKRS following transsphenoidal surgery. There was a greater risk of tumor progression after GKRS (p = 0.013) and residual tumor (p = 0.038) in the late radiosurgical group over a median imaging follow-up period of 68.5 months. No significant difference in the occurrence of post-GKRS endocrinopathy was observed (p = 0.68). Thirty percent of patients without endocrinopathy in the early cohort developed new endocrinopathies during the follow-up period versus 27% in the late cohort (p = 0.84). Fourteen percent of the patients in the early group and 25% of the patients in the late group experienced the resolution of endocrine dysfunction after original presentation (p = 0.32). CONCLUSIONS In this study, early GKRS was associated with a lower risk of radiological progression of subtotally resected nonfunctioning pituitary macroadenomas compared with expectant management followed by late radiosurgery. Delaying radiosurgery may increase patient risk for long-term adenoma progression. The timing of radiosurgery does not appear to significantly affect the rate of delayed endocrinopathy.
- MeSH
- čas zasáhnout při rozvinutí nemoci * MeSH
- časná lékařská intervence * MeSH
- dospělí MeSH
- endoskopie MeSH
- kohortové studie MeSH
- lidé středního věku MeSH
- lidé MeSH
- lokální recidiva nádoru diagnóza chirurgie MeSH
- nádory hypofýzy diagnóza chirurgie MeSH
- radiochirurgie normy MeSH
- reoperace MeSH
- retrospektivní studie MeSH
- reziduální nádor diagnóza chirurgie MeSH
- senioři MeSH
- sinus sphenoidalis chirurgie MeSH
- výsledky a postupy - zhodnocení (zdravotní péče) MeSH
- Check Tag
- dospělí MeSH
- lidé středního věku MeSH
- lidé MeSH
- mužské pohlaví MeSH
- senioři MeSH
- ženské pohlaví MeSH
- Publikační typ
- časopisecké články MeSH
- multicentrická studie MeSH
- srovnávací studie MeSH
BACKGROUND: Stereotactic radiosurgery (SRS) is used to treat recurrent or residual nonfunctioning pituitary neuroendocrine tumors (NFPA). The objective of the study was to assess imaging and development of new pituitary hormone deficiency. METHODS: Patients treated with single-session SRS for a NFPA were included in this retrospective, multicenter study. Tumor control and new pituitary dysfunction were evaluated using Cox analysis and Kaplan-Meier curves. RESULTS: A total of 869 patients (male 476 [54.8%], median age at SRS 52.5 years [Interquartile range (IQR): 18.9]) were treated using a median margin dose of 14Gy (IQR: 4) for a median tumor volume of 3.4 cc (IQR: 4.3). With a median radiological follow-up of 3.7 years (IQR: 4.8), volumetric tumor reduction occurred in 451 patients (51.9%), stability in 364 (41.9%) and 54 patients (6.2%) showed tumor progression.The probability of tumor control was 95.5% (95% Confidence Interval [CI]: 93.8-97.3) and 88.8% (95%CI: 85.2-92.5) at 5 and 10 years, respectively. A margin dose >14 Gy was associated with tumor control (Hazard Ratio [HR]:0.33, 95% CI: 0.18-0.60, P < 0.001). The probability of new hypopituitarism was 9.9% (95% CI: 7.3-12.5) and 15.3% (95% CI: 11-19.4) at 5 and 10 years, respectively. A maximum point dose >10 Gy in the pituitary stalk was associated with new pituitary hormone deficiency (HR: 3.47, 95% CI: 1.95-6.19). The cumulative probability of new cortisol, thyroid, gonadotroph, and growth hormone deficiency was 8% (95% CI: 3.9-11.9), 8.3% (95% CI: 3.9-12.5), 3.5% (95% CI: 1.7-5.2), and 4.7% (95% CI: 1.9-7.4), respectively at 10 years. CONCLUSIONS: SRS provides long-term tumor control with a 15.3% risk of hypopituitarism at 10 years.
- MeSH
- hypofyzární hormony MeSH
- hypopituitarismus * komplikace chirurgie MeSH
- lidé středního věku MeSH
- lidé MeSH
- nádory hypofýzy * radioterapie chirurgie MeSH
- následné studie MeSH
- radiochirurgie * metody MeSH
- retrospektivní studie MeSH
- výsledek terapie MeSH
- Check Tag
- lidé středního věku MeSH
- lidé MeSH
- mužské pohlaví MeSH
- ženské pohlaví MeSH
- Publikační typ
- časopisecké články MeSH
- multicentrická studie MeSH
OBJECTIVE: Stereotactic radiosurgery (SRS) is used for the treatment of residual/recurrent nonfunctional pituitary adenoma (NFPA). The aim of this study was to evaluate the factors related to long-term tumor control and delayed endocrinopathies following SRS. METHODS: This retrospective, multicenter study included patients with recurrent/residual NFPA treated with single-fraction SRS; they were then divided into two arms. The first arm included patients with at least 5 years of radiographic follow-up and all patients with local tumor progression. The second arm included patients with at least 5 years of endocrinological follow-up and all patients who developed endocrinopathy. Study endpoints were tumor control and new or worsening hypopituitarism after SRS and were analyzed using Cox regression and Kaplan-Meier methodology. RESULTS: There were 360 patients in the tumor control arm (median age 52.7 [IQR 42.9-61] years, 193 [53.6%] males) and 351 patients in the hypopituitarism arm (median age 52.5 [IQR 43-61] years, 186 [53.0%] males). The median follow-up in the tumor control evaluation group was 7.95 (IQR 5.7-10.5) years. Tumor control rates at 5, 8, 10, and 15 years were 93% (95% CI 90%-95%), 87% (95% CI 83%-91%), 86% (95% CI 82%-90%), and 69% (95% CI 59%-81%), respectively. The median follow-up in the endocrinopathy evaluation group was 8 (IQR 5.9-10.7) years. Pituitary function preservation rates at 5, 8, 10, and 15 years were 83% (95% CI 80%-87%), 81% (95% CI 77%-85%), 78% (95% CI 74%-83%), and 71% (95% CI 63%-79%), respectively. A margin dose > 15 Gy (HR 0.8, 95% CI 0.7-0.9; p < 0.001) and a delay from last resection to SRS > 1 year (HR 0.9, 95% CI 0.7-0.9; p = 0.04) were significant factors related to tumor control in multivariable analysis. A maximum dose to the pituitary stalk ≤ 10 Gy (HR 1.1, 95% CI 1.09-1.2; p < 0.001) was associated with pituitary function preservation. New visual deficits after SRS occurred in 7 (1.94%) patients in the tumor control group and 8 (2.3%) patients in the endocrinopathy group. Other new cranial nerve deficits post-SRS occurred in 4 of 160 patients with data in the tumor control group and 3 of 140 patients with data in the endocrinopathy group. CONCLUSIONS: SRS affords favorable and durable tumor control for the vast majority of NFPAs. Post-SRS hypopituitarism occurs in a minority of patients, but this risk increases with time and warrants long-term follow-up.
- MeSH
- adenom * chirurgie radioterapie diagnostické zobrazování MeSH
- dospělí MeSH
- hypopituitarismus etiologie MeSH
- lidé středního věku MeSH
- lidé MeSH
- lokální recidiva nádoru * MeSH
- nádory hypofýzy * chirurgie radioterapie diagnostické zobrazování MeSH
- následné studie MeSH
- radiochirurgie * metody škodlivé účinky MeSH
- retrospektivní studie MeSH
- reziduální nádor MeSH
- výsledek terapie MeSH
- Check Tag
- dospělí MeSH
- lidé středního věku MeSH
- lidé MeSH
- mužské pohlaví MeSH
- ženské pohlaví MeSH
- Publikační typ
- časopisecké články MeSH
- multicentrická studie MeSH
- MeSH
- adenom fyziologie chirurgie komplikace MeSH
- dospělí MeSH
- endokrinní žlázy fyziologie MeSH
- hypogonadismus etiologie patofyziologie MeSH
- hypotyreóza etiologie patofyziologie MeSH
- lidé středního věku MeSH
- lidé MeSH
- mladiství MeSH
- nádory hypofýzy chirurgie komplikace patofyziologie MeSH
- prolaktin analýza metabolismus MeSH
- senioři MeSH
- zrak genetika MeSH
- Check Tag
- dospělí MeSH
- lidé středního věku MeSH
- lidé MeSH
- mladiství MeSH
- mužské pohlaví MeSH
- senioři MeSH
- ženské pohlaví MeSH
Úvod: Karcinom příštítného tělíska je raritní endokrinní malignitou. Typicky se manifestuje závažnou hyperkalcemií při primárním hyperparathyreoidismu. Extrémně vzácně se může jednat o tzv. nefunkční karcinom s fyziologickými hladinami kalcia i parathormonu v séru pacienta. Pro karcinom příštítných tělísek je typický indolentní, avšak progresivní růst. Předoperační diagnostika onemocnění je velmi obtížná, definitivní diagnózu stanovuje histologické vyšetření. Za optimální léčbu je považována radikální chirurgická resekce s ipsilaterální lobektomií a blokovou krční disekcí. Účinnost adjuvantní onkologické léčby není prokázána a její indikace není definována. Recidivy onemocnění jsou časté. Kazuistika: Prezentujeme případ 26leté ženy, která na našem pracovišti podstoupila levostrannou hemithyreoidektomii pro růstovou progresi ultrasonograficky zjištěného ložiska za levým lalokem štítné žlázy. Předoperační cytologický nález ani závěry zobrazovacích vyšetření nevedly k podezření na malignitu. Hladina parathormonu a kalcia v séru pacientky byla ve fyziologických mezích. Na základě definitivní histologie byla stanovena diagnóza nefunkčního karcinomu příštítného tělíska. U pacientky nebyla indikována další chirurgická či onkologická léčba. Pacientka je 36 měsíců od operace bez známek recidivy či generalizace onemocnění. Závěr: Nefunkční karcinom příštítného tělíska je extrémně vzácný. V mnoha případech je diagnóza stanovena až v pokročilém stadiu onemocnění. V současnosti chybí formální klasifikace a také jednotný léčebný protokol. Stagingový systém onemocnění nově navrhuje osmé vydání AJCC/UICC. Včasná diagnostika spolu s kompletní primární chirurgickou resekcí a pečlivou dispenzarizací jsou zásadními faktory ovlivnění morbidity a mortality pacientů s touto malignitou.
Introduction: Parathyroid cancer is a rare endocrine malignancy. These tumors are typically functional, causing severe hypercalcemia due to primary hyperparathyroidism. Nonfunctional parathyroid cancer with normal serum calcium and parathyroid hormone levels is extremely rare. The disease is usually indolent but progressive with a tendency to metastasize. It is very difficult to diagnose this malignancy. The definitive diagnosis is made by histopathological examination. Radical surgery with ipsilateral lobectomy and en bloc neck dissection is considered to be the most appropriate therapeutic approach. There is no evidence of efficiency of adjuvant cancer therapy and its indication has not been defined. Disease recurrence is common. Case report: We report the case of a 26-year-old female patient who underwent left hemithyroidectomy for growth progression of a hypoechoic lesion behind the left thyroid lobe detected by ultrasonography. Preoperative cytology and imaging assessments were not suspicious for malignancy. Serum parathyroid hormone and calcium levels were normal. The diagnosis of nonfunctional parathyroid carcinoma was determined based on histopathological examination. No further surgery or adjuvant therapy was indicated. No signs of recurrence or generalization have been observed at 36 months after the surgery. Conclusion: Nonfunctional parathyroid cancer is extremely rare. In many cases, the diagnosis is made in advanced stages of the disease. No formal classification or treatment protocol has been established so far. A new staging system has been proposed in the 8th edition of AJCC/UICC. Early detection, radical surgery and close follow-up are crucial aspects to affect the mortality and morbidity of patients with this type of malignancy.
- Klíčová slova
- nefunkční karcinom příštítného tělíska,
- MeSH
- diferenciální diagnóza MeSH
- dospělí MeSH
- karcinom chirurgie diagnóza MeSH
- lidé MeSH
- nádory příštítného tělíska * chirurgie diagnóza MeSH
- paratyreoidea anatomie a histologie chirurgie patologie MeSH
- vzácné nemoci MeSH
- Check Tag
- dospělí MeSH
- lidé MeSH
- ženské pohlaví MeSH
- Publikační typ
- kazuistiky MeSH
- práce podpořená grantem MeSH
BACKGROUND: The purpose of our study was to determine whether renal function can be restored by early nephrostomy in patients who fail to develop renal function immediately after transplantation. METHODS: Between 2001 and 2010, we have performed external/internal nephrostomy in 13 patients during the early posttransplant period. The reason for the procedure was graft nonfunction with oliguria/anuria in the presence of normal renal perfusion, absence of signs of rejection, and/or renal collecting system dilation. The nephrostomy was created under ultrasound/fluoroscopic guidance. RESULTS: Nephrostomy was technically successful in all cases. On days 1-3 following nephrostomy creation, 10 of 13 patients showed an increase in diuresis and subsequent graft function development. Once the obstruction had been removed, the graft remained functional for months up to years posttransplantation. Three patients failed to respond to nephrostomy. There were no nephrostomy-related complications. CONCLUSIONS: Our study documents that, in patients who failed to develop graft function posttransplant for unknown reasons, nephrostomy may result in graft function development.
- MeSH
- časové faktory MeSH
- chirurgie s pomocí počítače metody MeSH
- dospělí MeSH
- hodnoty glomerulární filtrace fyziologie MeSH
- kohortové studie MeSH
- lidé středního věku MeSH
- lidé MeSH
- následné studie MeSH
- perkutánní nefrostomie metody MeSH
- pooperační období MeSH
- přežívání štěpu MeSH
- rejekce štěpu chirurgie ultrasonografie MeSH
- reoperace metody MeSH
- retrospektivní studie MeSH
- sběrací ledvinové kanálky chirurgie ultrasonografie MeSH
- senioři MeSH
- stenóza chirurgie MeSH
- transplantace ledvin škodlivé účinky metody MeSH
- ultrasonografie dopplerovská metody MeSH
- vyšetření funkce ledvin MeSH
- výsledek terapie MeSH
- Check Tag
- dospělí MeSH
- lidé středního věku MeSH
- lidé MeSH
- mužské pohlaví MeSH
- senioři MeSH
- ženské pohlaví MeSH
- Publikační typ
- časopisecké články MeSH
- práce podpořená grantem MeSH
Introduction: Nonfunctional pituitary adenomas are treated surgically, and even partial resection can improve or eliminate clinical symptoms. Notably, progression requires further intervention, which presents an increased risk, especially in older patients. This study investigated whether the histopathological characteristics of nonfunctional adenomas could predict recurrence. Materials and Methods: Data were obtained retrospectively from 30 patients who underwent surgery for the partial resection of pituitary adenomas. Remnant tumor growth was observed in 17 patients, while the residual tumor was unchanged more than 7 years after surgery in 13 patients. Statistical analysis was performed to investigate correlations between remnant tumor progression and tumor histopathological findings, including protein expression of p21, p27, p53, and Ki-67. Results and Discussion: Remnant tumors that demonstrated regrowth showed significantly higher protein expression of p21 and Ki-67. Expression of the p53 tumor suppressor was also higher in this group, but the difference was at the limit of statistical significance. Conclusion: Tumors with high expression of p21 and p53 and with a high Ki-67 index were more likely to show residual pituitary adenoma progression. Such cases should undergo frequent radiological examination and timely reoperation, and radiosurgery should be considered.
