Bilateral adrenocortical adenomas causing adrenocorticotropic hormone-independent Cushing's syndrome: A case report and review of the literature

BACKGROUND Adrenocorticotropic hormone(ACTH)-independent Cushing's syndrome(CS) is mostly due to unilateral tumors, with bilateral tumors rarely reported. Its common causes include primary pigmented nodular adrenocortical disease,ACTH-independent macronodular adrenal hyperplasia, and bilateral adrenocortical adenomas(BAAs) or carcinomas. BAAs causing ACTHindependent CS are rare; up to now, fewer than 40 BAA cases have been reported. The accurate diagnosis and evaluation of BAAs are critical for determining optimal treatment options. Adrenal vein sampling(AVS) is a good way to diagnose ACTH-independent CS.CASE SUMMARY A 31-year-old woman had a typical appearance of CS. The oral glucose tolerance test showed impaired glucose tolerance and obviously increased insulin and Cpeptide levels. Her baseline serum cortisol and urine free cortisol were elevated and did not show either a circadian rhythm or suppression with dexamethasone administration. The peripheral 1-deamino-8-D-arginine-vasopressin(DDVAP)stimulation test showed a delay of the peak level, which was 1.05 times as high as the baseline level. Bilateral AVS results suggested the possibility of BAAs.Abdominal computed tomography showed bilateral adrenal adenomas with atrophic adrenal glands(right: 3.1 cm × 2.0 cm × 1.9 cm; left: 2.2 cm × 1.9 cm × 2.1 cm). Magnetic resonance imaging of the pituitary gland demonstrated normal findings. A left adenomectomy by retroperitoneoscopy was performed first,followed by resection of the right-side adrenal mass 3 mo later. Biopsy results of both adenomas showed cortical tumors. Evaluations of ACTH and cortisol showed a significant decrease after left adenomectomy but could still not be suppressed, and the circadian rhythm was absent. Following bilateral adenomectomy, this patient has been administered with prednisone until now,all of her symptoms were alleviated, and she had normal blood pressure without edema in either of her lower extremities.CONCLUSION BAAs causing ACTH-independent CS are rare. AVS is of great significance for obtaining information on the functional state of BAAs before surgery.

Paget’s disease of bone: Report of 11 cases

BACKGROUND Paget’s disease of bone(PDB)is a rare metabolic bone disease in China and is characterized by increased bone resorption and disorganized bone formation.The main clinical symptoms of PDB are focal or multiple bone pain and deformity with high disability.The disease has high missed diagnosis and misdiagnosis rates.This report summarizes the clinical manifestations,imaging and pathological features,and treatments of 11 patients with PDB at our hospital from 1993 to 2020 in order to improve the recognition and prognosis of PDB.CASE SUMMARY There were eight male and three female patients whose average age was 48.7±11.0 years with a PDB course of 1-16 years.Nine patients had bone pain and bone deformities in different parts of the body,the majority of which involved the long bones.Laboratory examinations revealed elevated serum alkaline phosphatase(ALP)in all patients with an average of 618±460 IU/L(normal range 0-130 IU/L),and serum calcium and phosphorus levels were in the normal range.Imageology showed that osteolysis was usually combined with osteosclerosis and/or bone deformities in single or multiple bones.^(99m)Tc-methylene diphosphonate bone scintigraphy revealed increased radionuclide uptake in the bone lesions.Six patients underwent bone tissue biopsy,and the typical pathological changes were a mosaic structure of the bone trabeculae with irregularly arranged cement lines and multinuclear osteoclasts.Ten of the 11 patients were effectively treated with bisphosphonates.CONCLUSION Early diagnosis of the rare disease PDB can be made through elevated ALP levels and typical presentations on bone X-ray and from bone tissue biopsy.

Diffuse large B cell lymphoma with bilateral adrenal and hypothalamic involvement: A case report and literature review

BACKGROUND Non-Hodgkin’s lymphoma(NHL)can involve extralymphatic organs,resulting in diverse clinical manifestations,especially if the endocrine organs are affected.This type of involvement can often be difficult to detect accurately.Until now,no patients with NHL and concomitant bilateral adrenal and hypothalamic involvement have been reported.The purpose of this article is to discuss the diagnosis and treatment of lymphoma with bilateral adrenal gland and hypothalamic involvement so as to help physicians avoid misdiagnosis and missed diagnosis.CASE SUMMARY We describe a case of a 52-years-old male patient with bilateral adrenal masses,who presented with a fever of unknown origin on admission.Subsequently,hypopituitarism of the anterior pituitary followed by posterior pituitary developed.18fluorine-fluorodeoxyglucose positron emission tomography/computed tomography(PET/CT)showed lesions with a high metabolism in both adrenal glands,hypothalamus,left supraclavicular lymph nodes,and other organs.The etiological diagnosis was determined based on a left supraclavicular lymph node biopsy.The patient,who eventually present with panhypopituitarism,was finally diagnosed with diffuse large B cell lymphoma with bilateral adrenal gland and hypothalamic involvement.After immunochemotherapy,glucocorticoids administration and desmopressin acetate replacement therapy,the symptoms of fever and panhypopituitarism improved,and all the lesions reduced in size.CONCLUSION This report demonstrates that,although synchronous involvement of two endocrine organs is rare in NHL,extra caution should be taken when dysfunction occurs in multiple endocrine organs.

Association between Self-reported Snoring and Prediabetes among Adults Aged 40 Years and Older without Diabetes

Background：Several previous studies have shown that snoring is associated with glucose metabolism and the development of diabetes,but rare study has shown the association between snoring frequency and prediabetes,particularly in China.We hypothesized that individuals who snore might have a higher risk of prediabetes.This study aimed to investigate the association between self-reported snoring and prediabetes in a Chinese population.Methods：A cross-sectional study was performed in three large communities of Beijing from December 2011 to August 2012 by recruiting individuals aged ≥40 years old.All participants were requested to complete a detailed questionnaire and undergo anthropometric measurements.A 75 g oral glucose tolerance test was performed in individuals without diabetes.Blood samples of all participants were collected;blood glucose and blood fat levels were measured.Multivariate logistic regression models were built to assess the association between snoring frequency and prediabetes.Results：A total of 13,592 participants （female：66.56%;mean age：56.8 ＆#177; 7.9 years;mean body mass index：25.5 ± 3.4 kg/m2） were included in the final analysis.Of these,30.9% were diagnosed with prediabetes,while 41.3% and 25.4% had occasional and habitual snoring,respectively.Habitual snoring was associated with an increased risk of prediabetes （odds ratio [OR]：1.3,95% confidence interval [CI]：1.1-1.4,P 〈 0.001）,after adjusting for diabetes and sleep-related confounders in the multivariable models.Habitual snoring was also associated with isolated impaired fasting glucose （IFG;OR：1.3,95% CI：1.0-1.6;P 〈 0.001） and isolated impaired glucose tolerance （IGT;OR：1.3,95% CI：1.2-1.5;P 〈 0.001）,but not IFG ＋ IGT （OR：1.1,95% CI：0.9-1.4;P =0.281）.When stratified by total cholesterol （TC） levels,this association between habitual snoring and prediabetes was observed only in individuals with TC 〈5.6 mmol/L （OR：1.4,95% CI：1.2-1.6;P〈 0.001）.Conclusions：Habitual snoring is associated with prediabetes,but only in individuals with TC 〈5.6 mmol/L.Further prospective studies are needed to confirm this finding.