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OBJECTIVE: The objective of this study was to evaluate the role of adrenal venous sampling (AVS) in guiding the management of patients with corticotropin (ACTH)-independent glucocorticoid secretory autonomy and bilateral adrenal masses. DESIGN AND PATIENTS: A cohort with 25 patients underwent AVS and surgical management. MEASUREMENTS: Cortisol was measured from the adrenal veins (AVs) and inferior vena cava (IVC). AV/IVC cortisol ratio and cortisol lateralization ratio (CLR) (dominant AV cortisol concentration divided by the nondominant AV cortisol concentration) were calculated. Posthoc receiver-operating characteristic curves were generated to determine the specificity of revised AV/IVC cortisol ratio and CLR in differentiating unilateral from bilateral disease. RESULTS: Patients underwent unilateral (n = 21) or bilateral (n = 4) adrenalectomy. The mean AV/IVC cortisol ratio for unilateral adrenalectomy was 12.1 ± 9.6 (dominant) and 4.7 ± 3.8 (contralateral) with a mean CLR of 3.6 ± 3.5. The mean AV/IVC cortisol ratio for bilateral adrenalectomy was 7.5 ± 2.1, with a mean CLR of 1.1 ± 0.6. At a mean follow-up of 22 months, one patient who underwent unilateral adrenalectomy for the predicted bilateral disease developed recurrent mild autonomous cortisol secretion. Posthoc analyses demonstrated a specificity of 95%-100% for unilateral disease with AV/IVC cortisol ratio >9 for one side, <2.0 for the opposite side and a CLR > 2.3. The specificity was 80%-90% for bilateral disease with AV/IVC cortisol ratio >5.1 bilaterally and a CLR < 1.1. CONCLUSIONS: Among patients with bilateral adrenal masses and ACTH-independent autonomous cortisol secretion, AVS can distinguish between unilateral and bilateral disease with high specificity and may guide surgical management.
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Doenças das Glândulas Suprarrenais , Hiperaldosteronismo , Humanos , Hidrocortisona , Glândulas Suprarrenais/irrigação sanguínea , Adrenalectomia , Hormônio Adrenocorticotrópico , Hiperaldosteronismo/cirurgia , Estudos Retrospectivos , AldosteronaRESUMO
BACKGROUND: Primary aldosteronism (PA) is a common cause of secondary hypertension, whereas pheochromocytoma is a rare cause of it. Thus, concomitant PA and pheochromocytoma is a very rare condition. CASE PRESENTATION: A 52-year-old woman was admitted to our hospital with suspected PA based on the presence of hypertension, spontaneous hypokalemia, and a high aldosterone-to-renin ratio. She had no catecholamine excess symptoms other than hypertension. Abdominal computed tomography (CT) showed a right lipid-rich adrenal mass and a left lipid-poor adrenal mass. PA was diagnosed by the captopril challenge test. The 24-h urinary fractionated metanephrines were slightly elevated. Adrenal vein sampling (AVS) confirmed that the right adrenal gland was responsible for aldosterone hypersecretion. Medical therapy with eplerenone was started because the patient refused surgery. Five years later, she requested surgery for PA. The second AVS confirmed right unilateral hyperaldosteronism, as expected. Repeated abdominal CT showed the enlargement of the left adrenal mass. The 24-h urinary fractionated metanephrines had risen to the diagnostic level. 123I- metaiodobenzylguanidine (MIBG) scintigraphy showed a marked tracer uptake in the left adrenal mass with no metastatic lesion. After preoperative management with α-blockade, laparoscopic left partial adrenalectomy was performed. Immunohistochemical examination of the tumor showed chromogranin A positivity leading to the diagnosis of left pheochromocytoma. CONCLUSIONS: We report an extremely rare case of concomitant unilateral PA and contralateral pheochromocytoma. When diagnosing unilateral PA by AVS, especially in cases with a lipid-poor adrenal mass, clinicians should rule out the possibility of the presence of pheochromocytoma before proceeding to undergo unilateral adrenalectomy. Although there is no standard treatment for this rare condition, it is essential to select personalized treatment from the perspective of conserving the adrenal gland.
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Neoplasias das Glândulas Suprarrenais , Hiperaldosteronismo , Hipertensão , Feocromocitoma , Feminino , Humanos , Pessoa de Meia-Idade , Neoplasias das Glândulas Suprarrenais/complicações , Neoplasias das Glândulas Suprarrenais/diagnóstico , Neoplasias das Glândulas Suprarrenais/cirurgia , Glândulas Suprarrenais/irrigação sanguínea , Adrenalectomia , Aldosterona , Hiperaldosteronismo/complicações , Hiperaldosteronismo/diagnóstico , Hipertensão/complicações , Hipertensão/cirurgia , Lipídeos , Feocromocitoma/complicações , Feocromocitoma/diagnóstico , Feocromocitoma/cirurgiaRESUMO
Background: Laparoscopic adrenalectomy is currently considered the gold standard method for adrenal surgery. Open surgery is the most frequent technique preferred amongst surgeons who are faced with tumours of larger sizes or challenging lesions. Despite the increasing interest in laparoscopy, most centres still utilise open surgery for challenging adrenal cases. and Methods: We retrospectively evaluated our successive 30 robotic adrenalectomies performed in the past 10 years and assigned the patients into 'difficult' and 'easy' groups. Patients with malignant tumours or tumour size of over 8 cm were assigned to the 'difficult group' and others to the 'easy group'. Groups were evaluated according to the demographic features of the patients, side of the operation, the body mass index (BMI) and laparotomy history. The duration of anaesthesia, amount of bleeding during surgery and the hospitalisation periods were also evaluated. Results: There is no statistically significant difference between the two groups considering age, gender, BMI value, operation side, presence or absence of a laparotomy history, the amount of bleeding during the operation and hospitalisation duration (P > 0.05). The anaesthesia duration was found to be higher in the 'difficult' patient group (P < 0.05). Conclusion: Our results present robust evidence to support the idea that robotic adrenalectomy is not only a doable but also a safe option for malignant and large adrenal masses.
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Adrenal incidentalomas are incidentally discovered adrenal masses greater than one centimeter in diameter. An association between insulin resistance and adrenal incidentalomas has been established. However, the pathophysiological link between these two conditions remains incompletely characterized. This review examines the literature on the interrelationship between insulin resistance and adrenal masses, their subtypes, and related pathophysiology. Some studies show that functional and non-functional adrenal masses elicit systemic insulin resistance, whereas others conclude the inverse. Insulin resistance, hyperinsulinemia, and the anabolic effects on adrenal gland tissue, which have insulin and insulin-like growth factor-1 receptors, offer possible pathophysiological links. Conversely, autonomous adrenal cortisol secretion generates visceral fat accumulation and insulin resistance. Further investigation into the mechanisms and timing of these two pathologies as they relate to one another is needed and could be valuable in the prevention, detection, and treatment of both conditions.
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Neoplasias das Glândulas Suprarrenais , Resistência à Insulina , Neoplasias das Glândulas Suprarrenais/patologia , Humanos , Hidrocortisona , Gordura Intra-Abdominal/patologiaRESUMO
Introduction: [11C]Metomidate ([11C]MTO), the methyl ester analogue of etomidate, was developed as a positron emission tomography (PET) radiotracer for adrenocortical tumours and has also been suggested for imaging in primary aldosteronism (PA). A disadvantage of [11C]MTO is the rather high non-specific binding in the liver, which impacts both visualization and quantification of the uptake in the right adrenal gland. Furthermore, the short 20-minute half-life of carbon-11 is a logistic challenge in the clinical setting. Objectives: The aim of this study was to further evaluate the previously published fluorine-18 (T1/2=109.5 min) etomidate analogue, para-chloro-2-[18F]fluoroethyl etomidate; [18F]CETO, as an adrenal PET tracer. Methods: In vitro experiments included autoradiography on human and cynomolgus monkey (non-human primate, NHP) tissues and binding studies on adrenal tissue from NHPs. In vivo studies with [18F]CETO in mice, rats and NHP, using PET and CT/MRI, assessed biodistribution and binding specificity in comparison to [11C]MTO. Results: The binding of [18F]CETO in the normal adrenal cortex, as well as in human adrenocortical adenomas and adrenocortical carcinomas, was shown to be specific, both in vitro (in humans) and in vivo (in rats and NHP) with an in vitro Kd of 0.66 nM. Non-specific uptake of [18F]CETO in NHP liver was found to be low compared to that of [11C]MTO. Conclusions: High specificity of [18F]CETO to the adrenal cortex was demonstrated, with in vivo binding properties qualitatively surpassing those of [11C]MTO. Non-specific binding to the liver was significantly lower than that of [11C]MTO. [18F]CETO is a promising new PET tracer for imaging of adrenocortical disease and should be evaluated further in humans.
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Córtex Suprarrenal/diagnóstico por imagem , Etomidato/análogos & derivados , Tomografia por Emissão de Pósitrons/métodos , Compostos Radiofarmacêuticos/administração & dosagem , Neoplasias do Córtex Suprarrenal/diagnóstico , Animais , Avaliação Pré-Clínica de Medicamentos , Etomidato/administração & dosagem , Etomidato/farmacocinética , Radioisótopos de Flúor/administração & dosagem , Radioisótopos de Flúor/farmacocinética , Humanos , Hiperaldosteronismo/diagnóstico , Macaca fascicularis , Camundongos , Compostos Radiofarmacêuticos/química , Compostos Radiofarmacêuticos/farmacocinética , Ratos , Distribuição TecidualRESUMO
OBJECTIVES: To evaluate the features of adrenal masses on ultrasonography and correlate the findings with the pathologic diagnoses to help distinguish benign from malignant adrenal lesions. METHODS: Ultrasonography was performed in 1363 patients with adrenal lesions. The following ultrasonographic parameters were recorded: size, shape, margin, echogenicity, echo texture, cystic necrosis, calcifications, and blood supply. The sensitivity and specificity of aggressive features for predicting malignancy were calculated. RESULTS: Of 1385 adrenal lesions, 66 (4.8%) were malignant: 33 malignant pheochromocytomas, 20 adrenal cortical carcinomas, 10 metastases, 2 leiomyosarcomas, and 1 primitive neuroectodermal tumor. The remaining 1319 (95.2%) lesions were benign: 832 adenomas, 182 pheochromocytomas, 153 nodular adrenal hyperplasia, 54 myelolipomas, 42 cysts, 23 ganglioneuromas, 10 schwannomas, 8 cortical tumor eosinophils, 4 teratomas, 4 tuberculosis, 4 primary pigmented nodular adrenocortical disease, and 3 Castleman disease. Most of the tumors were regular and hypoechoic. The malignant lesions were significantly larger than the benign lesions (mean ± SD, 6.3 ± 2.4 versus 4.6 ± 1.7 cm; P < .01). Significant differences between malignant and benign tumors were observed when comparing the shape, margins, echo texture and vascularization of the analyzed lesions (P < .01). An irregular shape, poor definition of margins, heterogeneous echo texture, and vascularization indicated malignancy, with sensitivity and specificity of 45.5% and 97.0%, 34.8% and 99.2%, 92.4% and 97.3%, and 15.2% and 97.5%, respectively. CONCLUSIONS: Ultrasonography could be an effective diagnostic supplementary tool for adrenal tumors. A large size, irregular shape, poorly defined margins, heterogeneous echo texture, and vascularization of adrenal lesions could be effective indicators of malignancy.
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Neoplasias das Glândulas Suprarrenais/diagnóstico por imagem , Ultrassonografia/métodos , Adolescente , Glândulas Suprarrenais/diagnóstico por imagem , Adulto , Idoso , Diagnóstico Diferencial , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Estudos Retrospectivos , Sensibilidade e Especificidade , Adulto JovemRESUMO
INTRODUCTION: Congenital adrenal hyperplasia (CAH) autosomal recessive disorders characterized by impaired adrenal steroid hormone synthesis. The most common form is 21-hydroxylase deficiency (21OHD). Testicular adrenal rest tumors (TARTs) are benign intratesticular masses that occur in male patients with CAH. TARTs are quite common in patients with 21OHD who were diagnosed late. CASE REPORT: A 41-year-old male patient with CAH secondary to 21OHD. The patient was referred to our endocrinology department from the andrology clinic for bilateral adrenal masses. Bilateral orchiectomy had been performed due to bilateral testicular masses and azoospermia two years ago. The pathology was reported as Leydig cell tumor. In hormonal assessment, baseline cortisol levels were low, 17-hydroxyprogesterone levels with baseline and after cosyntropin stimulation test were high. As a result of clinic and laboratory assessment, the patient was diagnosed with simple virilising CAH due to 21OHD and adrenal insufficiency. Then, prednisolone replacement was initiated. Bilateral orchiectomy tissue blocks of the patient were re-assessed and were considered TART. Magnetic resonance imaging revealed bilateral adrenal masses with 88x55 mm on the right and 41x22 mm on the left. Laparoscopic right adrenalectomy was applied and pathology was reported as myelolipoma. Follow-up of the mass on the left adrenal gland is ongoing. The patient is monitored under prednisolone and testosterone replacement therapy. Early diagnosis of CAH is very important because of the complications it causes. It should be considered especially for bilateral testicular and/or adrenal masses. Both fertility and adrenal glands can be protected with an early diagnosis and an early glucocorticoid replacement.
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Generalized glucocorticoid resistance is associated with glucocorticoid receptor (GR; NR3C1) mutations. Three novel heterozygous missense NR3C1 mutations (R477S, Y478C, and L672P) were identified in patients presenting with adrenal incidentalomas, glucocorticoid excess without Cushing syndrome. Dexamethasone (DXM) binding studies demonstrated that the affinity of GRR477S and GRY478C mutants, located in the DNA-binding domain (DBD) of GR, was similar to wild-type GR (Kd = 2-3 nM). In contrast, GRL672P mutant, located in the ligand-binding domain (LBD) of GR, was unable to bind glucocorticoids and was more sensitive to protein degradation. GR subcellular distribution revealed a marked decrease in DXM-induced nuclear translocation of GRR477S and GRY478C mutants, whereas GRL672P remained exclusively cytoplasmic. Chromatin immunoprecipitation demonstrated impaired recruitment of DBD mutants onto the regulatory sequence of FKBP5. Transactivation assays disclosed the lack of transcriptional activity of GRR477S and GRL672P , whereas GRY478C had a reduced transactivation capacity. Three-dimensional modeling indicated that R477S lost two essential hydrogen bonds with DNA, Y478C resulted in altered interaction with surrounding amino-acids, destabilizing DBD, whereas L672P altered the H8 helix folding, leading to unstructured LBD. This study identifies novel NR3C1 mutations with their molecular consequences on altered GR signaling and suggests that genetic screening of NR3C1 should be conducted in patients with subclinical hypercorticism.
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Neoplasias das Glândulas Suprarrenais/genética , Resistencia a Medicamentos Antineoplásicos , Glucocorticoides/farmacologia , Mutação Puntual , Receptores de Glucocorticoides/química , Receptores de Glucocorticoides/genética , Neoplasias das Glândulas Suprarrenais/tratamento farmacológico , Adulto , Animais , Sítios de Ligação , Células COS , Núcleo Celular/metabolismo , Chlorocebus aethiops , Síndrome de Cushing/genética , Citoplasma/metabolismo , DNA de Neoplasias/metabolismo , Dexametasona/farmacologia , Feminino , Células HEK293 , Humanos , Masculino , Pessoa de Meia-Idade , Modelos Moleculares , Mutação de Sentido Incorreto , Estrutura Secundária de Proteína , Transporte Proteico , Receptores de Glucocorticoides/metabolismoRESUMO
OBJECTIVES: To determine the frequency of adrenal enlargement of participants in a CT-screening program for lung cancer and demonstrate the progression during follow-up, separately for baseline and annual repeat rounds. MATERIALS AND METHODS: HIPAA-compliant informed consent was obtained in 4,776 participants. The adrenal gland was defined as enlarged if it measured ≥6 mm at its largest diameter. Logistic regression analyses were performed. RESULTS: At baseline, 202 (4 %) of 4,776 participants had adrenal enlargement. Significant factors were age (OR = 1.4, 95 % CI: 1.2-1.7) and current smoker (OR = 1.8, 95 % CI: 1.3-2.4). Follow-up 7-18 months after baseline for 133 cases with adrenal enlargement <40 mm showed it decreased or was stable in 85 (64 %), and increased by <10 mm in 48 (36 %). Five (0.04 %) cases of adrenal enlargement were newly identified, none increased beyond 40 mm on follow-up. Adrenal enlargement was a significant predictor of a subsequent diagnosis of lung cancer (OR = 2.0, 95 % CI: 1.2-3.4). CONCLUSION: Participants with adrenal enlargement <40 mm identified at baseline and on repeat screening could be reasonably assessed on subsequent annual screening. Adrenal enlargement increased with increasing pack-years of smoking. Adrenal enlargement was an independent predictor of a subsequent diagnosis of lung cancer. KEY POINTS: ⢠Adrenal enlargement was seen in 4 % of participants at baseline screening. ⢠Age and currently smoking were significantly associated with adrenal enlargement. ⢠0.04 % of participants were newly identified with adrenal enlargement. ⢠Annual follow-up for adrenal enlargement <40 mm was appropriate. ⢠Adrenal enlargement was an independent predictor of a diagnosis of lung cancer.
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Doenças das Glândulas Suprarrenais/diagnóstico por imagem , Glândulas Suprarrenais/diagnóstico por imagem , Neoplasias Pulmonares/diagnóstico por imagem , Tomografia Computadorizada por Raios X , Doenças das Glândulas Suprarrenais/epidemiologia , Glândulas Suprarrenais/patologia , Assistência ao Convalescente , Fatores Etários , Idoso , Progressão da Doença , Detecção Precoce de Câncer , Feminino , Humanos , Incidência , Achados Incidentais , Modelos Logísticos , Neoplasias Pulmonares/epidemiologia , Masculino , Programas de Rastreamento , Pessoa de Meia-Idade , Tamanho do Órgão , Estudos Retrospectivos , Fatores de Risco , Fumar/epidemiologiaRESUMO
PURPOSE: To implement in-phase and out-of-phase (IP/OP) techniques with Magnetization-Prepared Gradient Recalled Echo (MP-GRE) and to evaluate the feasibility and diagnostic image quality among pre and post-optimized MP-GRE sequences, including patients unable to cooperate with breath-hold requirements. MATERIALS AND METHODS: Institutional review board approval with waiver of informed consent was obtained for this HIPAA-compliant retrospective study. Two groups of patients were included in the study, before and after optimization of MP-GRE parameters, with seventy-three (24 noncooperative/49 cooperative) and sixty-four (22 noncooperative/42 cooperative) consecutive patients, respectively. The motion-insensitive sequence used in this study was a single-shot 2D MP-GRE. Two radiologists qualitatively evaluated the sequences to identify the presence of phase cancellation artifact in OP images and to determine image quality, extent of artifacts (respiratory ghosting, bounce-point artifact, spatial misregistration and pixel graininess) and lesion conspicuity on the various sequences. The ability to visually detect liver steatosis and fatty adrenal adenomas was evaluated. Qualitative analyses were compared using the Wilcoxon and Mann-Whitney tests. RESULTS: There were statistically significant differences between all MP-GRE sequences concerning phase cancellation artifact (P<.0001) which was present in MP-GRE OP sequences and negligible to absent in the pre (IP1) and post-optimized (IP2) MP-GRE IP sequences, respectively, in all patients. Bounce point artifacts were significantly more pronounced in MP-GRE IP1 (P<.0001). Spatial misregistration was slightly more prominent in noncooperative patients with MP-GRE IP2 (P=.0027). MP-GRE OP and MP-GRE IP2 showed significantly higher overall image quality (P<.0001). MP-GRE sequences subjectively identified hepatic steatosis (n=20) and adrenal adenomas (n=5) based on signal loss from IP to OP sequence. CONCLUSION: Single shot IP/OP MP-GRE is feasible and allows motion resistant imaging with adequate diagnostic image quality. This technique is able to provide IP and OP information in patients unable to suspend respiration.
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Imageamento por Ressonância Magnética/métodos , Artefatos , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Estudos RetrospectivosRESUMO
Introduction: Adrenal masses are a rare diagnosis in children, with bilateral masses even less common. At present, appearance of the mass on imaging and histology can give important clues to the diagnosis; however, there is significant overlap in 10-30% of cases and it can be difficult to distinguish benign from malignant adrenal masses. As a result, the clinical presentation remains a large part of the diagnostic process, as well as thorough endocrinology evaluation to determine if the tumor is functional versus nonfunctional. Case Presentation: We present a recent case of bilateral adrenal masses in a pediatric patient at our institution, with an unusual diagnosis of histiocytosis. Conclusion: In this case, the diagnosis was unclear, until genetic testing and pathology allowed for expedient diagnosis and targeted therapy for this patient. We hope that presenting this case will increase physician awareness of this condition and expedite diagnosis and treatment in other patients with this rare presentation.
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Background: Laparoscopic adrenalectomy (LA) has emerged as the primary treatment for adrenal masses. This systematic review and meta-analysis assessed LA's feasibility, safety, effectiveness, and complications for adrenal masses exceeding 5 cm. Methods: The study was conducted using PRISMA guidelines with PROSPERO registration No. CRD42023462901. Adults with unilateral adrenal masses >5 cm who underwent unilateral LA were included. Intraoperative and postoperative measurements and complications were assessed. A systematic literature review employed a comprehensive search strategy which was last searched on September 8, 2023, through PubMed, Google Scholar, Web of Science, and ProQuest databases. Meta-analysis was utilized to analyze the outcomes. Risk of bias was assessed using the Newcastle-Ottawa scale. Results: This systematic review encompassed 25 studies involving 963 patients who underwent LA. Tumor size varied 7.05 cm [95% confidence interval (CI): 6.24-7.70], with 50% on the right and 45% on the left. The subgroup meta-analysis comparing the transperitoneal and retroperitoneal approaches revealed the transperitoneal approach h was utilized for the largest tumor size with a mean of 12.10 cm (95% CI: 11.30-12.96), compared to the retroperitoneal approach 5.83 cm (95% CI: 5.52-6.14). Notably, the mean operative time across studies was 137.4 minutes (95% CI: 113.36-150.94), bleeding prevalence was 0.02% (95% CI: 0.01-0.03%), and average blood loss was 110.6 mL (95% CI: 78.2-156.3). Postoperative complications such as pulmonary edema, pulmonary embolism, gastric dysfunction, and wound infection were very low, ranging from 0.03% to 0.4%. Out of 963 patients, only 49 were converted to open surgery. Patient hospital stay averaged 3.72 days (95% CI: 2.97-4.66); blood transfusion was required in 1.3% (95% CI: 0.30-8.88%). Conclusions: The feasibility and safety of LA for tumors exceeding 5 cm in size have notable implications for intraoperative and postoperative outcomes. Underreporting in the included studies may impact the generalizability of findings.
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Nasal-type natural killer/T-cell lymphoma is a rare non-Hodgkin's malignant lymphoma. A distinct clinicopathological entity associated with the Epstein-Barr virus, it typically presents with otorhinolaryngeal symptomatology. We report a rare case of a 24-year-old patient with nasal-type lymphoma with an atypical inaugural testicular presentation associated with the discovery of bilateral adrenal involvement.
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Congenital adrenal hyperplasia (CAH) is caused by genetic defects in the enzymes involved in cortisol biosynthesis in the adrenal gland and, in more than 90% of cases, due to a deficiency in the 21-hydroxylase enzyme. Classical CAH due to 21-hydroxylase deficiency is a severe form of the disease that presents with cortisol deficiency and is further categorized into salt-wasting or simple-virilizing types. Appropriate steroid replacement has been shown to effectively treat patients with classical CAH and prevent complications. Individuals who receive inadequate treatment or fail to comply with their prescribed steroid hormone regimen are susceptible to the development of adrenal myelolipomas. Myelolipomas are benign tumors composed of both adipose and hematopoietic tissues. While documented cases of adrenal myelolipomas exist in medical literature, instances of large bilateral myelolipomas remain exceedingly rare. This case report highlights a 40-year-old female patient with a known history of classical congenital adrenal hyperplasia who presented with unusually large bilateral adrenal myelolipomas. A diagnostic CT scan of the abdomen and pelvis revealed a 13.4 x 10.8 cm myelolipoma on the left adrenal gland and a 10 x 8.6 cm myelolipoma on the right adrenal gland. Prior to her presentation, the patient experienced recurrent nausea and vomiting, along with left upper quadrant pain, over five months. Hormonal assessments indicated significantly elevated serum androgen levels, suggesting inadequate management of her CAH. In this report, we present a rare case of symptomatic bilateral large adrenal myelolipomas, underscoring the significance of adhering to treatment regimens, diagnostic assessments, and management for adrenal myelolipomas in individuals diagnosed with CAH.
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Bilateral adrenal hyperplasia associated with primary lung cancer may arise from either an adrenocorticotropic hormone (ACTH)-dependent paraneoplastic syndrome or metastatic hyperplasia. Ectopic Cushing's syndrome manifests as a paraneoplastic syndrome characterized by the secretion of ACTH from malignant cells. This secretion can in turn overstimulate the adrenal cortex, resulting in adrenal cortical hyperplasia followed by hypercortisolism. Though rare, lung cancer can metastasize to the adrenal glands bilaterally, occasionally resulting in hemorrhage causing a rapid increase in size. This cadaveric report aims to delineate an unusual case of metastatic lung cancer, presenting with bilateral adrenal hyperplasia and two abdominal aortic aneurysms (AAA). An 84-year-old white male cadaver was dissected during routine coursework. Dissection of the right lung revealed a pale-yellow mass measuring 10.0 x 7.4 x 7.0cm. An irregularly defined, necrotic, ulcerated lesion measuring 5.5 x 4.5 x 3.3cm was evident on the lateral surface of the left shoulder, consistent with a history of metastatic lung carcinoma. Upon abdominal dissection, bilaterally enlarged adrenal glands and two AAAs were observed. These AAAs measured 6.0cm and 11.0cm at their respective widest transverse diameters. The right and left adrenal glands measured 10.0 x 6.5 x 4.5cm and 7.3 x 4.7 x 3.5cm, respectively. We aim to discuss the possible pathophysiological correlation of these unusual findings in this cadaver.
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We report a case of a 78-year-old immunocompetent man who presented with worsening fatigue and lethargy for one month. He had also been complaining of cough and SOB for two months which had been attributed to his underlying COPD and possible pneumonia. CT showed bilateral pleural effusions, ground-glass opacities, cirrhosis, splenomegaly and bilateral adrenal masses which was highly suspicious for malignancy. After pheochromocytoma was ruled out, EUS-FNA guided biopsy was performed on the left adrenal gland. Histology was positive for yeast cells, with fungal staining (PAS) revealing narrow-based budding compatible with Histoplasma. The patient was treated with amphotericin and itraconazole. Our case is unique as he presented with hepatosplenomegaly, which is reported in less than a quarter of cases. Although typically a diagnosis in immunocompromised patients, a high index of clinical suspicion is required to diagnose disseminated histoplasmosis in an immunocompetent patient. The gold standard for diagnosis is fungal tissue culture. However results may take up to weeks. EUS-FNA guided biopsy of adrenal glands can aid in early definitive diagnosis and management.
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PURPOSE: Incidental adrenal masses (IAMs) are common but rarely evaluated. To improve this, we developed a standardized radiology report recommendation template and investigated its implementation and effectiveness. METHODS: We prospectively studied implementation of a standardized IAM reporting template as part of an ongoing quality improvement initiative, which also included primary care provider (PCP) notifications and a straightforward clinical algorithm. Data were obtained via medical record review and a survey of radiologists. Outcomes included template adoption rates and acceptability (implementation measures), as well as the proportion of patients evaluated and time to follow-up (effectiveness outcomes). RESULTS: Of 4,995 imaging studies, 200 (4.0%) detected a new IAM. The standardized template was used in 54 reports (27.0%). All radiologists surveyed were aware of the template, and 91% affirmed that standardized recommendations are useful. Patients whose reports included the template were more likely to have PCP follow-up after IAM discovery compared with those with no template (53.7% versus 36.3%, P = .03). After adjusting for sex, current or prior malignancy, and provider ordering the initial imaging (PCP, other outpatient provider, or emergency department or inpatient provider), odds of PCP follow-up remained 2.0 times higher (95% confidence interval 1.02-3.9). Patients whose reports included the template had a shorter time to PCP follow-up (log-rank P = .018). PCPs ultimately placed orders for biochemical testing (35.2% versus 18.5%, P = .01), follow-up imaging (40.7% versus 23.3%, P = .02), and specialist referral (22.2% versus 4.8%, P < .01) for a higher proportion of patients who received the template compared with those who did not. CONCLUSIONS: Use of a standardized template to communicate IAM recommendations was associated with improved IAM evaluation. Our template demonstrated high acceptability, but additional strategies are necessary to optimize adoption.
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Achados Incidentais , Radiologia , Humanos , Estudos Prospectivos , Radiografia , Diagnóstico por ImagemRESUMO
Histoplasma capsulatum is a geographically specific dimorphic fungus that can cause a spectrum of diseases. While most cases are asymptomatic pulmonary infections, in severe cases, particularly in immunocompromised patients, disseminated disease can occur. Histoplasmosis in California is limited to only a few case reports. In this article, we describe a rare case of disseminated histoplasmosis in a non-endemic region presenting with diagnostically challenging symptomatology, including altered mental status, status epilepticus, septic shock, and bilateral adrenal masses.
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Histoplasmose , Linfo-Histiocitose Hemofagocítica , Humanos , Histoplasmose/complicações , Histoplasmose/diagnóstico , Histoplasmose/microbiologia , Linfo-Histiocitose Hemofagocítica/complicações , Linfo-Histiocitose Hemofagocítica/diagnóstico , Histoplasma , CaliforniaRESUMO
A 59-year-old woman underwent an abdominal and pelvic computed tomography (CT) scan to rule out non-obstructive urolithiasis. The patient was asymptomatic, with the exception of occasional bilateral low back pain. A physical examination did not reveal any notable findings. The CT scan revealed the presence of an incidental solid left adrenal lesion, which displaced the body of the pancreas and the left kidney. The lesion measured 7 cm × 6.5 cm and exhibited a rounded morphology with well-defined margins. It showed progressive and heterogeneous contrast uptake. Additionally, magnetic resonance imaging (MRI) was performed, confirming the presence of an adrenal lesion with intense and heterogeneous hypersignal on T2. The lesion also demonstrated heterogeneous and persistent enhancement in a dynamic study. Furthermore, there were some indistinct and non-specific hypointense areas identified on both T1 and T2 sequences. The lesion exhibited moderately restricted diffusion. Although the imaging features were non-specific, there were no indications of invasion or distant metastasis, which made a benign large adrenal mass the most likely diagnosis. Non-functioning pheochromocytoma, lipid-poor adrenal adenoma, as well as metastasis or primary adrenal carcinoma, were considered differential diagnoses. The patient underwent an elective adrenalectomy, during which the identified lesion was completely resected. The patient's postoperative recovery was uneventful, and she was discharged three days after the procedure. Subsequent histopathological evaluation revealed an oncocytic neoplasm of the adrenal cortex - specifically, an oncocytoma.
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Bilateral adrenal hemorrhage (BAH) is a rare condition that can lead to acute adrenal insufficiency and death if not recognized and treated promptly. We report the case of a 30-year-old male who presented to the emergency department with acute abdominal pain, nausea, and vomiting. On emergency room admission, the first abdominal CT revealed normal adrenal glands without enlargement, but with the development of hypotension and hypoglycemia, a second CT performed four days later showed enlargement due to hemorrhage in both adrenals. The diagnosis of BAH associated with acute adrenal insufficiency was retained. Prompt treatment with intravenous and oral corticosteroids resulted in successful conservative management. We describe the clinical, biological, radiological and etiological features of this condition based on a review of the literature.