Unusual case of antiphospholipid syndrome presenting as adrenal insufficiency

2020 ◽  
Vol 13 (3) ◽  
pp. e233631
Author(s):  
Sanval Ahmed Warriach ◽  
Mohamad Mustafa ◽  
Denis O'Keeffe ◽  
Michael Watts

A 64-year-old man presented to the emergency department with generalised fatigue and dizzy spells. His background history includes a previous admission with right leg deep vein thrombosis, provoked by pneumonia. Laboratory results showed evidence of hyponatremia and hyperkalaemia. A synacthen test was performed that indicated hypoadrenalism. CT of his abdomen revealed enlarged adrenal glands bilaterally. Adrenal antibodies and positron emission tomography (PET) scan were performed to assess the cause of enlarged adrenals. PET scan showed no evidence of increased uptake. Adrenal antibodies were found to be negative. Tuberculous (TB) adrenalitis was the principle differential diagnosis. TB QuantiFERON was strongly positive. Following 9 months of TB treatment, surveillance CT scan indicated a significant reduction in adrenal gland size. However, subsequent events culminated in a retrospective review of CT scans questioning the initial clinical diagnosis and suggesting that the observed adrenal gland enlargement was secondary to bilateral adrenal infarction and haemorrhage. Equally, the subsequently observed marked reduction in adrenal gland size was not secondary to an assumed response to TB therapy, but rather the sequela of infracted atrophied adrenal glands, as a manifestation of the underlying antiphospholipid syndrome (APS). The case highlights the importance of recognising adrenal insufficiency in patients with a history of APS. It also illustrates the role of multidisciplinary meetings in the management of such complex cases.

2019 ◽  
Vol 12 (9) ◽  
pp. e231114
Author(s):  
Raghav Bhargava ◽  
Anthony Dixon

Adrenal lymphoma causing primary hypoadrenalism in the elderly is a very rare finding. We describe a case of an 85-year-old man who was admitted to our hospital. He had a short history of hypotension and unresponsiveness. He was referred to the endocrine team, after a CT scan of his thorax abdomen and pelvis demonstrated significant enlargement of both his adrenal glands with associated lymphadenopathy. This scan was originally done to rule out an occult intra-abdominal malignancy. In addition, a tetracosactide (short Synacthen) test demonstrated adrenal insufficiency. This case demonstrates that adrenal lymphoma should be considered as an important differential when there is evidence of adrenal gland hyperplasia, lymphadenopathy and adrenal insufficiency.


2017 ◽  
Vol 7 (3) ◽  
pp. 235-237
Author(s):  
Sultana Marufa Shefin ◽  
Kazi Nazmul Hossain ◽  
Jamal Uddin Ahmed ◽  
Mohammad Mostafizur Rahman

Tuberculosis (TB) is a common infectious disease in developing countries like Bangladesh. Drug-resistant tuberculosis (DR-TB) poses a significant threat to the population. A relatively uncommon site of involvement of TB is adrenal gland, which is an important cause of chronic adrenal insufficiency. Here we present a case of a 40-year-old male with multidrug-resistant tuberculosis (MDR-TB) involving the adrenal glands. He was previously diagnosed as a case of disseminated TB involving lungs, right knee joint and lumbar spine and received anti-TB chemotherapy for several occasions and in different categories in last 12 years before this presentation. He presented with 4-month history of vomiting, diarrhea, generalized weakness, generalized pigmentation and postural hypotension. Investigations revealed hyponatremia with hyperkalemia. Adrenal insufficiency was suspected and serum cortisol was found to be low and serum adreno corticotropic hormone (ACTH) was high. On imaging there was bilateral adrenal enlargement. Fine needle aspiration cytology (FNAC) was done from adrenal gland which revealed granulomatous lesion suggestive of TB. Gene Xpert and culture-sensitivity of aspirated material for Mycobacterium tuberculosis revealed MDR-TB. Patient was finally diagnosed as a case of primary adrenal insufficiency due to MDR-TB of the adrenal glands and was treated with regime for MDR-TB for 20 months.Birdem Med J 2017; 7(3): 235-237


2013 ◽  
Vol 2013 ◽  
pp. 1-5 ◽  
Author(s):  
Adnan Bhatti ◽  
Hindi Al-Hindi ◽  
Ayman Azzam ◽  
Tarek Amin ◽  
Ahmed Abu-Zaid

Primary retroperitoneal teratomas involving adrenal glands are exceedingly uncommon accounting for only 4% of all primary teratomas. They are more common in childhood and rarely occur in adults. Only a very few case reports have been documented in literature so far. Herein, we report a mature (benign) cystic retroperitoneal teratoma in the region of left adrenal gland in a 22-year-old otherwise healthy male patient who presented with a 1-month history of left flank pain. In addition, a literature review on teratomas is included.


2020 ◽  
Vol 4 (Supplement_1) ◽  
Author(s):  
Daniela Maria DiCenso ◽  
Carlos A Penaherrera ◽  
Alejandro Ayala

Abstract Introduction: Primary Adrenal insufficiency is an uncommon complication of antiphospholipid syndrome, with an incidence of 0.4% [2]. It is often secondary to bilateral adrenal hemorrhage. We present a case of bilateral adrenal hemorrhage in a patient with APLS on anticoagulation with rivaroxaban. Case: 46-year-old m with a history of antiphospholipid syndrome, recently transitioned from warfarin to rivaroxaban for anticoagulation, who presented to the ED after a syncopal episode following a prior episode of abdominal pain with an unremarkable work-up. He subsequently developed severe fatigue, dizziness, headaches, nausea and 15 lbs weight loss. On presentation, the patient was hypotensive(72/45 mmHg) and tachycardic. Intravenous hydration was started with minimal response. Initial laboratory testing showed serum sodium of 121mmol/L, potassium of 5.5mmol/L and random cortisol of 0.8mcg/dL. The patient was admitted to the intensive care unit where he was started on vasopressors and hydrocortisone 50 mg IV every 8 hours. A non-contrast CT of the abdomen and pelvis showed thickening of the adrenal glands with decreased attenuation. MRI of the abdomen showed hyper-intensity of the adrenal glands bilaterally (T1 images), without post-contrast enhancement suggestive of bilateral adrenal hemorrhage. His electrolytes normalized, and he was successfully discharged home on hydrocortisone and fludrocortisone replacement with outpatient follow-up. Discussion: Atraumatic bilateral adrenal hemorrhage is rare, but remains one of the most common endocrine-related complications of antiphospholipid syndrome (APLS). The venous anatomical configuration of the adrenal gland increases risk of thrombotic hemorrhagic infarction[1]. Patients with APLS are commonly anticoagulated to prevent thrombosis. The ideal anticoagulation regimen remains controversial. Only three other cases of spontaneous bilateral adrenal hemorrhage on patients with APLS using new oral anticoagulants (NOACs) were reported. The use of NOACs seem to increase the already-elevated risk of adrenal hemorrhage seen in patients with APLS. References: 1. Aldaajani, H. et al. Bilateral adrenal hemorrhage in antiphospholipid syndrome: Anticoagulation for the treatment of hemorrhage. Saudi Med J. 2018; 39(8): 829-833. 2. Cervera R, Piette JC, Font J, Khamashta MA, Shoenfeld Y, Camps MT, et al. Antiphospholipid syndrome: Clinical and immunologic manifestations and patterns of disease expression in a cohort of 1,000 patients. Arthritis Rheum. 2002;46:1019-27.


Author(s):  
Neeta Thakur

Antiphospholipid syndrome (APLS) is an acquired is auto immune disorder that is defined by the presence of antibodies known as antiphospholipid antibodies in addition to clinical thrombosis and/ or poor obstetric outcome. Although the incidence remains unknown, 10% to 15% of women with a history of recurrent pregnancy loss will meet the criteria for APLS. In these Patients, the annual incidence of deep vein thrombosis and stroke is 1.46% and 0.32% respectively. Compared with the annual incidence in the general population of 0.1% for deep vein thrombosis and 0.1% for stroke.1


2008 ◽  
Vol 159 (6) ◽  
pp. 819-824 ◽  
Author(s):  
S A Whitley ◽  
V J Moyes ◽  
K M Park ◽  
A M Brooke ◽  
A B Grossman ◽  
...  

AimsTo review the morphology of the adrenal glands in multiple endocrine neoplasia type 1 (MEN1) on computed tomography (CT) to compare the results with established normal values for adrenal size and nodularity and to correlate adrenal size with serum cortisol secretory dynamics.Materials and methodsTwo observers independently reviewed the adrenal CT in 28 patients with MEN1, measuring the maximum width of the body of the gland and the medial and lateral limbs. Incidence and location of nodules >5 mm within the gland were recorded. Following exclusion of known cases of Cushing's syndrome, adrenal gland size was compared with previously documented normative data. Adrenal gland size was compared between patients with normal and abnormal cortisol dynamics.ResultsComparison of mean adrenal size in MEN1 patients with normative data showed that the adrenal limbs were significantly larger in MEN1 than normal (P<0.0001 in all four limbs). Adrenal body was also significantly larger (P<0.05). Nodules were demonstrated in 17 (60%) of patients (versus 0.4–2% in the normal population). No statistically significant correlation was demonstrated between adrenal limb hyperplasia and abnormal cortisol dynamics.ConclusionsIn patients with MEN1, adrenal limb hyperplasia and adrenal nodules are significantly more common than in the normal population, a phenomenon not previously documented in a quantitative manner. There was no significant correlation between adrenal limb hyperplasia and abnormal cortisol dynamics.


2018 ◽  
Vol 90 (4) ◽  
pp. 23-28 ◽  
Author(s):  
Agnieszka Dworzyńska ◽  
Katarzyna Paduszyńska ◽  
Lech Pomorski

Introduction Adrenal insufficiency is a typical complication after surgical treatment of adrenal tumors, especially after the removal of both adrenal glands. Human beings are not able to survive without adrenal glands and without proper hormonal substitution. Autotransplantation of a fragment of the adrenal gland may prevent this complication. This can be done by transplanting the entire adrenal glands or its fragment, such as the adrenal cortex cells. In the case of adrenal tumors, the entire adrenal gland can not be transplanted. However, it is possible to transplant cells from the tumor-free part. Succesful adrenal autografts may result in a new treatment of adrenal insufficiency. Materials and Methods: Autograft transplantation was performed on 3 groups of Sprague Dawley rats. In the first group, physiological corticosterone concentrations were determined. These animals were not operated. In the second group, both adrenal glands were removed. Corticosterone concentrations were determined after bilateral adrenalectomy. The third group was divided into two parts. In the first subgroup, bilateral adrenalectomy was performed simultaneosly with adrenal transplant into the omentum. In the second subgroup, right adrenalectomy was performed simultaneosly with and adrenal transplant into the omentum followed a month later by left adrenalectomy. During the experiment, corticosterone concentrations were measured at 4 time points. Results. The statistical difference between corticosterone concentrations in rats after two timed adrenalectomies and rats after bilateral adrenalectomy was statistically different, but these results were far from physiological concentrations.


2020 ◽  
pp. 1098612X2097496
Author(s):  
Laura Pérez-López ◽  
Ana María Wägner ◽  
Pedro Saavedra ◽  
Jose Raduan Jaber ◽  
Carlos Melián

Objectives Adrenal gland size and its association with body weight have been rarely evaluated in cats. This study was undertaken to assess the association between feline body weight and adrenal gland thickness, and to propose reference intervals (RIs) for adrenal gland thickness in healthy cats. Methods This was a cross-sectional study in which 39 healthy cats were included. The cats were divided into two weight categories, classified as ⩽4.0 kg and >4–8 kg of ideal body weight (with 13 and 26 cats in each group, respectively), which took into consideration the body condition score of the cats. All cats underwent an ultrasound examination that was taken from a subcostal position. Maximum dorsoventral thicknesses of the left (MTL) and right (MTR) adrenal glands were measured in a sagittal plane. RIs were obtained for the maximum thickness (MT), which included the MTLs and MTRs of each cat. RIs with the 90% confidence intervals were calculated according to American Society for Veterinary Clinical Pathology guidelines on RIs. Results No statistical differences for adrenal gland thickness were observed between the left and right ( P = 0.543) adrenal glands or between male and female cats ( P = 0.943). Mean MT was significantly greater in the group of cats weighing >4–8 kg compared with the group of cats weighing ⩽4 kg (3.7 ± 0.6 vs 3.2 ± 0.4 mm; P <0.005). The lower limit of the RI for MT was 2.4 mm (range 2.2–2.6 mm) in the group weighing ⩽4 kg and 2.6 mm (range 2.4–2.8 mm) in the group weighing >4–8 kg. The upper limit of the RI for MT was 3.9 mm (range 3.7–4.1 mm) in the group of cats weighing ⩽4 kg and 4.8 mm (range 4.6–5.1 mm) in the group of cats weighing >4–8 kg. Conclusions and relevance The use of RIs based on two group sizes allows for a more accurate ultrasonographic evaluation of adrenal gland thickness in cats. The maximum normal adrenal gland thickness is lower in smaller cats (3.9 mm for those weighing ⩽4 kg and 4.8 mm for those weighing >4–8 kg).


2021 ◽  
Vol 5 (Supplement_1) ◽  
pp. A160-A160
Author(s):  
Moin U Sattar ◽  
Priyanka L Mahali ◽  
Natalia Chamorro-Pareja ◽  
Nisha Suda ◽  
Noah Bloomgarden

Abstract Background: Testosterone supplementation has been associated with a variety of side effects, such as polycythemia, and can potentially increase the risk of cardiovascular disease. Testosterone use has also been associated with increased thrombotic events, especially in patients with underlying hypercoagulable state. Clinical Case: A 57-year-old man presented with abdominal pain and distention. He had history of previous intramuscular (IM) and oral testosterone use for ten years. For 8 weeks prior to initial presentation, he reported using weekly IM 500mg Testosterone injections. Computed Tomography (CT) of the abdomen and pelvis revealed multiple thrombi of the portal, splenic, superior mesenteric and inferior mesenteric veins. He was started on Warfarin and discharged home. A few weeks later, he presented with similar symptoms with labs now showing an International Normalized Ratio (INR) of 10.2. Repeat CT was significant for presence of bilateral adrenal hemorrhage, measuring 2.9cm on the right and 2.4cm on the left, which were not seen on previous imaging done one week prior. During the hospital course, he was found to be hypotensive with low platelet count so Intravenous Immunoglobulin therapy was initiated for suspected catastrophic antiphospholipid syndrome (CAPS). Morning cortisol was 5.82 ug/dL (6.2–29.0) so this critically-ill patient was started on stress dose hydrocortisone, which was subsequently tapered to physiological dose after clinical improvement. Cosyntropin stimulation test was performed after withholding the prior dose of hydrocortisone. The baseline cortisol was 0.88 ug/dL (6.20–29.00ug/dL), after administration of 250mcg of Cosyntropin 30- and 60-minute cortisol levels were 1.5 ug/dL (4.3–22.4ug/dL) and 1.6 ug/dL (4.3–22.4ug/dL) respectively. Baseline ACTH of 121.0 pg/mL (7.2–63.3pg/mL), consistent with primary adrenal insufficiency. Dehydroepiandrosterone Sulfate (DHEA-S) level was 15.7 ug/dL (80.0–560.0ug/dL). Hypercoagulability workup was significant for the presence of lupus anticoagulant and antibodies positive for heparin induced thrombocytopenia, so patient was diagnosed with Antiphospholipid syndrome. Conclusion: This is a case of hypercoagulability in a patient with history of anabolic steroid misuse who developed extensive intraabdominal venous thrombosis, adrenal hemorrhage, and primary adrenal insufficiency. Adrenal vein thrombosis and hemorrhage can be life threatening sequalae of testosterone misuse and should be considered in the differential for patients with history of testosterone misuse and adrenal insufficiency.


2021 ◽  
Vol 26 (4) ◽  
pp. 124-131
Author(s):  
О.B. Kutovyi ◽  
E.V. Zhmurenko

Objective – to improve the results of treatment of adrenal tumors by optimizing a differentiated approach based on the analysis of the results of various surgical invasions. The paper analyzed the results of treatment of 145 patients with various adrenal tumors, which were divided into 2 groups: І (n=71) - patients after adrenalectomies, II (n=74) – after adrenal resections. Organ-preserving invasions showed better results during the early and long-term postoperative period in patients with corticosteromas, pheochromocytomas and incidentalomas by reducing the severity of fluctuations in hemodynamic parameters and the frequency of adrenal insufficiency. Due to the introduction of the treatment algorithm, the number of adrenal resections increased by 26.6% in patients with neoplasms more than 4,0 cm in diameter (p=0.0001). The choice of surgical invasion should be individualized, taking into account the clinical features of the disease, hormone indicator parameters, the size of tumors, the comprehensiveness of the adrenal gland lesion and the ratio of the latter to nearby organs and structures. Adrenal insufficiency was detected 14.2% more often after adrenalectomies (р=0.044) compared to adrenal resections. Treatment according to the developed algorithm reduced the incidence of adrenal insufficiency by 5.8% (p=0.028). In the long-term postoperative period, contralateral gland tumors occurred with the same frequency (4.8%) among patients after adrenalectomies and resections. Relapse of the neoplasm after resections was detected in 3 (7.1%), as a result of which 2.4% underwent repeated operations with the final removal of the gland. An increase in the size of a single adrenal gland was more common by 21.4% after adrenalectomies.


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