scholarly journals SAT-LB42 Bilateral Large Calcified Adrenal Leiomyoma Mimicking Adrenal Malignancy: A Rare Case Report With Literature Review

2020 ◽  
Vol 4 (Supplement_1) ◽  
Author(s):  
Mohammad Bilal M Jajah ◽  
Asim Hassan ◽  
Moutaz Haiba ◽  
Tohamy Elkhouly ◽  
Mohammed M Haj Elamin ◽  
...  

Abstract Background: Adrenal leiomyoma is a very rare benign soft tissue tumor, it is even more unusual if presenting bilaterally; 21 cases have been reported in the literature and only six had bilateral involvement; 5 in the pediatric population and only one in an adult patient. Radiological appearance may frequently be confused with malignancy especially if large, calcified and with central necrosis. We report a rare case of bilateral, large, calcified, non-functioning adrenal leiomyoma in a 20-year-old female, who was suspected for a malignancy preoperatively. Clinical Case: A 20 year-old-female presented with chronic abdominal discomfort, fatigue, and inability to gain weight. On examination, she was normotensive, underweight with BMI of 15.6 kg/m2, and there were no stigmata of Cushing’s syndrome, Addison’s disease or pheochromocytoma. A contrast CT scan of the abdomen revealed the presence of bilateral well-defined suprarenal lesions measuring 8.5 x 8.5 x 7.2 cm and 4.7 x 4.2 x 3.5 cm on the right and left side, respectively. The lesions showed large central areas of necrosis with multiple punctate calcifications and heterogenous peripheral enhancement. The radiological differential diagnosis included adrenal cortical carcinoma, adrenal metastasis, infectious etiology, and bilateral pheochromocytoma. Her hormonal assays showed normal free cortisol and catecholamine metabolites in the urine and normal serum androgens. Thus, the tumors were concluded to be non-functioning. Adrenal insufficiency was ruled out after a short Synacthen test. The patient underwent a successful right adrenalectomy. Resected specimen measured 10 x 9.5 x 7.5 cm. Histology revealed a well-circumscribed and pseudo-encapsulated smooth muscle tumor comprised of bland, spindle-shaped cells. The panel of immunohistochemical stains supported the diagnosis of leiomyoma. Postoperatively, the symptoms improved, she gained 4 kg weight over the following 4 months, and short Synacthen test confirmed an intact adrenal function. To avoid lifelong adrenal insufficiency and after discussion with the patient, we agreed to leave the left adrenal mass and follow it by serial imaging. There was only a minimal increase in the size over the following 4 years (5.5 x 4.5 x 3.8 cm). Conclusion: Adrenal leiomyoma is an extremely rare adrenal tumor and can be confused with adrenal malignancy. Therefore, it should be considered in the differential diagnosis of adrenal incidentalomas. In the case of bilateral etiology, permanent adrenal insufficiency and longterm replacement therapy can be avoided in certain population by removing the larger tumor and continuous follow-up for the other side.

2019 ◽  
Vol 96 (1138) ◽  
pp. 467-472
Author(s):  
Rajeev Kumar ◽  
Peter Carr ◽  
Kimberley Moore ◽  
Zeeshan Rajput ◽  
Louise Ward ◽  
...  

ObjectiveThe short synacthen test (SST) is widely used across the UK to assess adrenal reserve but there remains no consensus on the timing of cortisol sampling to help diagnose adrenal insufficiency. The main objective of our study was to see if both 30 and 60 min sample are required following administration of synacthen to investigate suspected adrenal insufficiency (AI).DesignThis was a single-centre retrospective study of 393 SSTs measuring 0, 30 and 60 min cortisol levels after administration of 250 µg of synacthen.Patients and methodsAll the SSTs for patients suspected of primary or secondary AI between April 2016 and October 2018 were included in this study. The tests were performed as per our hospital protocol. A post-adrenocorticotropic hormone (ACTH) cortisol response of 420 nmol/L at any time point was considered adequate to rule out AI. The data were analysed to ascertain the proportion of patients who achieved this level at 30 and/or 60 min.ResultsA total of 393 SST results were included in this study. Patients were divided into two groups depending on whether (group A) or not (group B) they were on steroids. Overall, a total of 313 (79.6%) subjects achieved cortisol level of ≥420 nmol/L at 30 and 60 min while 19 (4.8%) had late response (ie, insufficient 30 min cortisol levels, rising to ≥420 nmol/L at 60 min). Another 61 subjects (15.5%) showed insufficient response at both 30 and 60 min (ie, failed to achieved level of ≥420 nmol/L). Importantly, there was no patient in either group who had adequate response at 30 min and then failed at 60 min. Patients in group A were more likely to have inadequate response at both 30 and 60 min while patients in group B were more likely to have normal response at both time points.ConclusionsOur results suggest that about 5% of people undergoing SST may be inappropriately diagnosed as having AI (and subjected to long-term unnecessary steroid treatment) if the 60 min sample is not maintained. We suggest that 30 min sample does not add any additional diagnostic utility and can be omitted thus simplifying SST even further and saving on cost and resources. We propose that single measurement after 60 min of administration of synthetic ACTH is a sufficient screening test for AI.


Medicine ◽  
2020 ◽  
Vol 99 (43) ◽  
pp. e22621
Author(s):  
Muhammad Imran Butt ◽  
Nouf Alzuhayri ◽  
Lama Amer ◽  
Muhammad Riazuddin ◽  
Hadeel Aljamei ◽  
...  

Gut ◽  
2011 ◽  
Vol 60 (Suppl 2) ◽  
pp. A15-A15
Author(s):  
G. Fede ◽  
L. Spadaro ◽  
T. Tomaselli ◽  
G. Privitera ◽  
R. Scicali ◽  
...  

Rheumatology ◽  
2020 ◽  
Vol 59 (Supplement_2) ◽  
Author(s):  
Ruth Smith ◽  
Mohammed Akil

Abstract Background Many rheumatological conditions are treated with steroids. Patients on long-term steroids are known to be at risk of adrenal insufficiency especially when withdrawing them. Sheffield Teaching Hospital (STH) Endocrinologists advised the following protocol for patients reducing steroids to determine hypothalamic pituitary adrenal status and confirm safe onward withdrawal of steroids:- After 1 month on 3mg prednisolone measure 9am serum cortisol and ACTH (after 24 hours off steroid)- If cortisol <150nmol/L or < 200nmol/L and ACTH <10pg/mL - refer Endocrinology- If cortisol 200-430nmol/L (or > 150nmol/L and ACTH >10pg/mL) arrange short Synacthen test (SST). If 30-minute cortisol <430nmol/L - refer Endocrinology. If 30 minute cortisol >430nmol/L - stop steroids and monitor for adrenal insufficiency (give hydrocortisone 20mg TDS to cover illness for 3 months after stopping prednisolone). Methods We performed a retrospective review of records of rheumatology patients having morning cortisol level checked July 2017-January 2019 to determine how many tests were abnormal and to what degree the results changed management. Results 160 cortisol tests were performed. 36 were excluded from further analysis. Of the 124 cases reviewed: 80 females and 44 males. Age range 18-87; mean 64 years. Starting dose of prednisolone 5-60mg. Duration of steroid use 6 months to > 10 years; modal duration 13-24 months (49 patients/40%). 59 patients/48% were taking other immunosuppressants. Prednisolone dose at cortisol testing 1-5mg; modal value 3mg (89 patients/72%). 12 patients/10% had a normal morning cortisol. 9 patients/7% fell into the group ‘cortisol <150nmol/L refer Endocrinology.’ 103 patients/83% fell into the group requiring SST. 79 patients had an SST (out of an expected 112). 58/73% were normal. 17 patients/81% with abnormal SST were referred to Endocrinology. 11/65% were advised to wean prednisolone as per normal rheumatology practice of 1mg per month; 1/6% to continue prednisolone and repeat SST in 2-3 months; 3/18% to commence hydrocortisone. 2/12% had not yet been seen. Of the patients with abnormal SST: 8 females and 13 males. Age range 32-87; mean 67 years. Starting dose of prednisolone 5-40mg. Duration on steroids 6 months to > 10 years; modal duration 13-24 months in 9 patients/43%. 12 patients/57% were on other forms of immunosuppression. Conclusion Endocrinology guidance changed the management of 3 patients/2% by the introduction of hydrocortisone compared with previous rheumatology standard practice of weaning prednisolone by 1mg per month or similar. No distinctive characteristics were identified in patients with abnormal SST, except for a greater proportion of men (35% of whole group: 62% of abnormal SST group). Given the limited impact of the investigations, and the shortage of Synacthen, a new protocol has been implemented: if morning cortisol <300nmol/L, or 300-350nmol/L and symptoms - contact Endocrinology for SST; if 30 minute cortisol <430nmol/L - refer Endocrinology; otherwise wean prednisolone as normal. Disclosures R. Smith None. M. Akil Honoraria; AbbVie, Actelion, Celgene, Astra Zenica, Lilly and UCB.


2021 ◽  
Vol 10 (9) ◽  
pp. 1189-1199
Author(s):  
Filippo Ceccato ◽  
Elisa Selmin ◽  
Giorgia Antonelli ◽  
Mattia Barbot ◽  
Andrea Daniele ◽  
...  

Context The low-dose short synacthen test (LDSST) is recommended for patients with suspected central adrenal insufficiency (AI) if their basal serum cortisol (F) levels are not indicative of an intact hypothalamic–pituitary–adrenal (HPA) axis. Objective To evaluate diagnostic threshold for salivary F before and 30 min after administering 1 μg of synacthen, performed before 09:30 h. Design A cross-sectional study from 2014 to 2020. Setting A tertiary referral university hospital. Patients In this study, 174 patients with suspected AI, 37 with central AI and 137 adrenal sufficient (AS), were included. Main outcome measure The diagnostic accuracy (sensitivity (SE), specificity (SP)) of serum and salivary F levels measured, respectively, by chemiluminescence immunoassay and liquid chromatography-tandem mass spectrometry. Results Low basal serum or salivary F levels could predict AI. For the LDSST, the best ROC-calculated threshold for serum F to differentiate AI from AS was 427 nmol/L (SE 79%, SP 89%), serum F > 500 nmol/L reached SP 100%. A salivary F peak > 12.1 nmol/L after administering synacthen reached SE 95% and SP 84% for diagnosing central AI, indicating a conclusive reduction in the likelihood of AI. This ROC-calculated threshold for salivary F was similar to the 2.5th percentile of patients with a normal HPA axis, so it was considered sufficient to exclude AI. Considering AS those patients with salivary F > 12.1 nmol/L after LDSST, we could avoid unnecessary glucocorticoid treatment: 99/150 subjects (66%) had an inadequate serum F peak after synacthen, but salivary F was >12.1 nmol/L in 79 cases, who could, therefore, be considered AS. Conclusions Salivary F levels > 12.1 nmol/L after synacthen administration can indicate an intact HPA axis in patients with an incomplete serum F response, avoiding the need to start glucocorticoid replacement treatment.


2012 ◽  
Vol 2 (2) ◽  
pp. 5
Author(s):  
Anne Corbould ◽  
Matthew Jarvis ◽  
Joanne Campbell ◽  
Deborah Kunde ◽  
Wade Clarkson ◽  
...  

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