Calcifying fibrous tumor (CFT) is a rare, benign proliferation of fibroblasts and inflammatory cells that is non-invasive and usually arises in deep tissue structures. Due to its overall paucity, no accurate incidence has been reported yet. We understand this disease via a handful of case studies published in the medical literature, first of them being from 1988. Earlier known as 'pseudotumor', it was recently given its name due to the potential of recurrence and multifocal involvement. We describe the case of a 43-year-old Hispanic male who presented with a large symptomatic pleural-based mass which turned out to be CFT and was later complicated by empyema. Our aim is to increase awareness about this rare disease and throw light upon its benign nature, despite an alarming and suspicious appearance on imaging. Due to the large size of our patient's mass (largest reported yet), he needed extensive chest wall reconstruction, leading to complications requiring additional invasive procedures. This underscores the importance of early diagnosis and treatment which can reduce the need for aggressive surgical manipulation and avoid postoperative complications, thereby providing high-value care. Treating physicians should be mindful of this, in order to prompt early recognition to ensure effective patient care.
We have previously reported that there is a strong association between unnecessarily ordered thyroid USGs and unnecessarily requested Endocrinology (Endo) consultations. Unnecessary consults consume time and resources, delay appropriate consults, and have even been proposed as a factor in the over-diagnosis of clinically innocuous thyroid cancers. We have examined the impact on the consumption of clinical resources. The database consisted of 201 new Endo consults, each accompanied by a pre-consult thyroid USG. The consult requests were graded as appropriately requested (APPROP), optionally requested (OPT), or unnecessarily requested (UNNEC). The USG requests were likewise graded as APPROP, OPT, or UNNEC. The USGs were also graded on their degree of contribution to the request for a consultation, specifically, as having a significant role (SIGNIF), a minor role (MIN), or little or no role (NONE). The impact of the UNNEC consults was categorized as (a) the initial Endo consult, a resource that would not have been utilized were an UNNEC consult not submitted, and (b) resources that probably would have been utilized were the UNNEC consults not submitted but would have been managed by and costed to the referring provider instead of to the Endo provider. Such resources included follow-up Endo visits and relevant USGs and blood tests, Of the 201 consults with associated USGs, 156 (77.6%) consults were APPROP, 23 (11.4%) were OPT, and 22 (10.9%) were UNNEC. Conversely, 157 (78.1%) of the USGs were APPROP, 11 (5.5%) were OPT, and 33 (16.4%) were UNNEC. With respect to the association of consults with their accompanying USGs, Among APPROP requested consults, 87.8% of the associated USGs were also APPROP while only 7.7% were associated with UNNEC USGs. Among UNNEC requested consults, 31.8% of the associated USGs were APPROP while 68.2% were associated with UNNEC USGs. Regarding the resource utilization borne by an Endo clinic as a consequence of the submission of 21 UNNEC consults for the two years after the initial consultation, each UNNEC consult had consumed, on average, 5.9 (1 + 2.6 + 2.3) [initial + Yr 1 + Yr 2 follow-ups] Endo clinic visits, 0.9 (0.5 + 0.4) USGs [excludes the USG associated with initial consultation] and 4.1 (2.2 + 1.9) blood work orders and reviews. No follow-up FNAs or thyroid surgeries were performed. Endocrine care from any source would be best served by reducing both the unnecessary utilization and the unnecessary assignment of relevant resources.
Introduction To date, case reports have shown an association between paraganglioma and primary hyperaldosteronism, and primary hyperparathyroidism with primary aldosteronism, but not for all three of the aforementioned endocrinopathies combined. Clinical Case A 54-year-old woman with history of uncontrolled HTN, hypokalemia, DM2, CKD stage III, Takotsubo cardiomyopathy and primary hyperparathyroidism presented to the hospital with back pain. CT scan showed a 4.3 cm necrotic retroperitoneal mass. Due to her labile blood pressure, ranging from 194/102 mmHg to 106/60 mmHg, endocrinology was consulted for evaluation of secondary hypertension. Plasma normetanephrines were 3447 pg/mL (n ≤ 57 pg/mL), 24-hour urine normetanephrines 5954 mcg/24 hours (n 122-676 mcg/24 hours) and 24-hour urine norepinephrines 476 mcg/24 hours (n 15-100 mcg/24 hours). 24-hour urine cortisol and 24-hour urine metanephrines were within normal limits. Plasma aldosterone level and plasma renin activity (PRA) were 19 ng/dL and 0.78 ng/mL/h (n 0.25-5.82 ng/mL/h),. However, the patient was also taking spironolactone and telmisartan which interfered with results, and withdrawal of interfering medications was considered unsafe. 68Ga-DOTATATE PET/CT showed mild uptake in a left para-aortic mass. Genetic testing did not show pathogenic mutations of AP2S1, CASR, MEN1, NF1, RET, SDHA, SDHAF2, SDHB, SDHC, TMEM127 or VHL genes. The patient was started on doxazosin and underwent successful removal of the left retroperitoneal mass consistent with paraganglioma. Six months later, surveillance testing showed normalization of fractionated plasma metanephrines and of 24-hour urine metanephrines. However, due to blood pressure of 152/100 mmHg and K of 2.8 mmol/L (n 3.5-4.7 mmol/L), concern was raised for primary hyperaldosteronism. After potassium repletion, labs showed plasma aldosterone of 37 ng/dL, PRA of 0.2 ng/mL/h (n 0.25-5.82 ng/mL/h) and ARR of 185 with potassium of 4.1 mmol/L (n 3.5-4.7 mmol/L) consistent with primary hyperaldosteronism. MRI of adrenal glands showed a left adrenal gland that was thickened and nodular but without discrete nodules > 1 cm. The patient was referred for adrenal vein sampling. Five years previously, the patient had been diagnosed with primary hyperparathyroidism, with PTH of 188.5 pg/mL (n 14-64 pg/dL), Calcium of 10.6 mg/dL (n 8.5-10.1 mg/dL) and GFR of 39. Pre-operative imaging with MRI neck had shown enlargement of two parathyroid glands. She had undergone two-gland parathyroidectomy with intraoperative PTH decrease of > 50% and normalization of calcium. However two weeks later, her PTH returned to pre-operative levels (with normal 25(OH)D) and continues to remains elevated. This has been attributed to underlying secondary hyperparathyroidism from her chronic kidney disease. Conclusion To our knowledge, this is a unique case of combined paraganglioma, primary hyperaldosteronism and primary hyperparathyroidism, as thorough literature review has not shown such a coexistence in the past. Presentation: No date and time listed
e16113 Background: Thyroid gland is an uncommon site for metastases from clear cell renal cell carcinoma (CCRCC) and literature is scarce. Due to the variable and often long lag time before development of metastases in patients with CCRCC, thyroid nodules may be misdiagnosed initially as benign. This systematic review aims at a better understanding of the nature of these metastases. Methods: A bibliographic search was performed using PubMed (1990-2019), key words being “renal cell carcinoma, thyroid, kidney cancer, clear cell.” 147 cases were analyzed. The patient’s characteristics assessed were: age, sex, stage, size of metastases, lag time, diagnostic modality, initial symptoms, treatment and outcome in last documented follow up. Binary logistic regression, spearman and ANOVA were performed to identify differences between the existing variables. Results: The mean age (±SE) was 64± (10) years in males and 64 (±11) in females. The mean lag time to diagnosis of thyroid metastasis was 8.7 (±6.3) years. Gender distribution of the patients was 46.3% male, 52.4% female. There was a weak correlation between lag time and size of metastases, not statistically significant. Size of metastases was significantly higher in symptomatic patients (6.06±3.51cm) compared to those with painless mass (4.6±0.29cm) and asymptomatic ones (3.93±1.99cm) (p = 0.03). Fine Needle Aspiration (FNA) was diagnostic in 29.4% of cases, 47.1% were non diagnostic. Most patients (80.3%) underwent thyroid surgery. At 1 year follow up, 55.6% of patients operated were alive versus 35.3% who did not have surgery, though this was not statistically significant (p = 0.1). Conclusions: A larger size of thyroid metastasis was more likely to present with symptomatology. A high index of suspicion is warranted when evaluating thyroid nodules in CCRCC patients. There was no significant difference in outcome between patients who underwent surgery and those who did not. With the wider use of immune check-point and tyrosine kinase inhibitors in metastatic CCRCC, surgery may eventually be reserved only for palliative purposes.
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