Validation of the Aldosteronoma Resolution Score as a Predictive Resolution Score of Hypertension After Unilateral Adrenalectomy for Primary Aldosteronism in a North-African Population.

BACKGROUND: Unilateral primary aldosteronism (UPA) is the most frequent surgically curable form of endocrine hypertension. Adrenalectomy is the cornerstone of treatment for UPA, but outcomes after surgery are variable. Aldosteronoma Resolution Score (ARS) is a four-item predictive score for the cure of hypertension after adrenalectomy for UPA and has been demonstrated to be valid in different populations. We aimed in this study to validate the accuracy of this score in a North-African population. METHODS: Between 2000 and 2021, the charts of 71 Tunisian patients who underwent laparoscopic adrenalectomy for UPA were retrospectively reviewed. Postoperative outcomes were assessed using the primary aldosteronism surgical outcome (PASO) criterion. The accuracy of the ARS was determined retrospectively by receiver operating characteristic curve and area under the curve. RESULTS: Thirty-four patients (48%) had complete clinical success according to the PASO criteria. Multivariate regression analysis revealed that the main determinants of complete clinical success were the absence of diabetes (OR: 5.205), a BMI <30 (OR: 4.930), a number of antihypertensive medications ≤2 (OR: 8.667), a plasma ARR >332 (OR: 4.554) and an ARS score ≥3 (OR: 2.056). Cure rates were, respectively, 21.1, 51.6, and 66.6% for patients with a score ARS 0-1, 2-3, and 4-5. The AUC of the ARS was 0.837. CONCLUSION: The ARS is a sufficiently predictive score in our North-African population. It may be used preoperatively to predict the outcome after adrenalectomy in these populations.

Pre- and Post-treatment Serum BAFF Levels and BAFF Gene Polymorphisms in Patients with Graves' Disease.

BACKGROUND: B cell activating factor (BAFF), a crucial factor for B cell survival and differentiation, has been linked to several autoimmune conditions. The aim of this study was to evaluate the association of BAFF gene's polymorphisms with its serum levels and to assess their effect on Graves' disease (GD) susceptibility and presentation. METHODS: Sixty-two GD patients and 152 healthy controls have been enrolled to investigate BAFF rs9514827 (-2841 T/C), rs1041569 (-2701 T/A) and rs9514828 (-871 C/T) gene's polymorphism by PCR-RFLP and serum BAFF level's kinetics under medical treatment by ELISA. RESULTS: Median serum BAFF level at baseline was significantly higher in GD patients (841.7 pg/ml [685.23-1058.32]) comparatively to controls (495.75 pg/ml [383.17-595.7]), p = 7.29 E-25. A ROC curve was used to assess BAFF performances in GD diagnosis and revealed an AUC of 94.9% [0.919-0.979], p = 7.29 E-25. At a cutoff value of 654.9 pg/ml of BAFF at baseline, the sensitivity and the specificity were, respectively, 83.9% and 90.8%. BAFF level was significantly increased in smoking patients (1079.55 pg/ml [875.35-1203]) comparatively to nonsmokers (746.95 pg/ml [643.2-915.7]), p = 3.1 E-5. While -2841 T/C and -2701 T/A genotypes and alleles frequencies were similar between patients and controls, the -871*T allele was significantly more prevalent in patients (0.613) than in controls (0.477); p = .01, OR [95% CI] = 1.73 [1.13-2.65]. The three studied polymorphisms were not associated with serum BAFF level at baseline. CONCLUSION: Serum BAFF level is significantly increased in GD especially in smoking patients. rs9514828 - 871*T allele might be a susceptibility variant for GD.

Impact of hydrocortisone replacement on bone mineral density and bone turnover markers in patients with primary adrenal insufficiency.

Objective. The study was aimed to assess the effect of hydrocortisone (HC) replacement therapy on bone mineral density (BMD) and bone turnover markers in patients with primary adrenal insufficiency (PAI). METHODS: A cross-sectional study was conducted in 37 PAI patients treated with HC. BMD and selected bone turnover markers (ß-crosslaps and osteocalcin) were measured. A stepwise binary logistic regression model was applied to determine the independent variables associated with low BMD. RESULTS: Osteoporosis was noted in 14.3% and osteopenia in 34.3% of cases. These patients were older (p=0.01) and received higher daily HC dose compared to patients with normal BMD (p=0.01). BMD values in the lumbar spine and the femoral neck were negatively correlated with daily HC dose (r=-0.36, p=0.03 and r=-0.34, p=0.05, respectively). Plasma osteocalcin was negatively correlated with disease duration (r=-0.38, p=0.02) and cumulative HC dose (r=-0.43, p<0.01). In multivariate analysis, a daily HC dose ≥12 mg/m2/day was independently associated with a higher risk of osteopenia/osteoporosis [OR (95% CI), 9.0 (1.1-74.6); p=0.04]. CONCLUSIONS: Impaired bone mineralization in patients with PAI is correlated with HC dose. A daily HC dose ≥12 mg/m2/day was associated with an increased risk of osteopenia and osteoporosis in these patients.

Edinburgh questionnaire in the screening for peripheral artery disease in type 2 diabetic patients.

AIM: to analyze Edinburgh questionnaire (EQ) screening performance for peripheral artery disease (PAD) in type 2 diabetic patients. METHODS: Cross sectional study including 150 type 2 diabetic patients without PAD history. All patients responded to EQ and had peripheral pulse checkup and measurement of the ankle/brachial index (ABI). PAD was considered to be present when the ABI was ≤ 0.9 Results: Participants mean age was 57.46 ± 8.04 years and sex-ratio (men/women) was 1.3. EQ has revealed intermittent claudication in 18 patients (12%). On examination, 42 patients (28%) had at least one weakened or abolished pulse in upper limbs. ABI has revealed the presence of PAD in 16% of patients. The EQ sensibility specificity, positive and negative predictivevalueswere 29, 91,39 and 87%, respectively. Among patients with false negative results (n=17), nine had peripheral neuropathy. CONCLUSION: In diabetic patients, EQ had a very low sensibility for the PAD screening. In fact, the important false negative rate, due to the coexisting of peripheral neuropathy, had limited the use of this questionnaire.

Benzylthiouracil induced ANCA associated glomerulonephritis in patients with graves' disease.

BACKGROUND: Renal complications in Graves' disease are rare and may be related either to the disease itself or secondary to antithyroid drugs. AIM: We report 6 cases of renal damage in patients with Graves' disease treated with Benzylthiouracil collected over a period of 14 years. METHODS: There were 6 women with a mean age of 37.86 ± 14.25 years. All patients developed renal vasculitis associated with ANCA. The signs were dominated by renal proteinuria and renal failure associated with hematuria in all cases. The lung involvement was the most common extrarenal manifestation occurred in 4 patients (alveolar hemorrhage in 2 cases, lymphocytic alveolitis in 1 case and pleurisy in 1 case). The benzylthiouracil was discontinued in 3 patients still under treatment. Corticosteroid therapy was used alone or in combination with cyclophosphamide in all cases. Plasmapheresis sessions were made during the alveolar hemorrhage. A complete remission was obtained in one case and incomplete remission in 2 cases. The other 3 patients required chronic hemodialysis. One patient died of sepsis. CONCLUSION: The possibility of renal impairment in antithyroid drugs treated Graves' disease requires monitoring to detect urinary abnormalities in order to early initiate therapy and improve patient's outcome.

Vitamin B12 levels in type 2 diabetic patients on Metformin compared to those never on Metformin: a cross sectional study in Tunisia.

INTRODUCTION: Recent studies suggest that long-term use of metformin may decrease the plasma level of Vitamin B12. AIM: To assess the Vitamin B12 status in Tunisian patients treated with metformin and to study its association with the dose, the duration of metformin use, and the clinical and biological parameters. METHODS: It was a cross-sectional, comparative study on 200 type 2 diabetic (T2D) patients. A vitamin B12 assay was performed with a neurological examination and a Complete blood count. RESULTS: The mean level of Vitamin B12 assayed in our population was 398.5±188.3 pg/ml. The serum Vitamin B12 levels were 356.9±153.5 pg/ml in the metformin group and 460.9±218.6 pg/ml in the no metformin group (p <0.01). Metformin intake was associated with an increased prevalence of Vitamin B12 deficiency and borderline level. The level of Vitamin B12 was correlated with the duration and the dose of metformin. Vitamin B12 deficiency was significantly associated with anemia, macrocytosis, and diabetic neuropathy. Multivariate analysis concluded that Vitamin B12 deficiency was significantly associated with the duration, cumulative metformin dose, clinical neuropathy, anemia, and macrocytosis. CONCLUSION: Our study showed an association of Vitamin B12 deficiency with the dose and duration of metformin intake in Tunisian T2D patients, with hematological and neurological repercussions.

Predictors of successful outcome after adrenalectomy for unilateral primary aldosteronism.

Introduction: Unilateral primary aldosteronism (UPA) is the most frequent surgically curable form of endocrine hypertension. Adrenalectomy is the cornerstone of treatment for UPA, but outcomes after surgery are variable. The cause of resistant hypertension after surgery is still a matter of debate. Our aim was to investigate cure rates after surgery and to evaluate preoperative factors that might influence the surgical outcome. Methods: Between 2000 and 2021, the charts of 71 Tunisian patients who underwent laparoscopic adrenalectomy for UPA were retrospectively reviewed. Preoperative medical records were collected and follow-up data (1-158 months) were registered. Antihypertensive medication doses were calculated using defined daily doses (DDD) and postoperative outcomes were assessed using the Primary Aldosteronism Surgical Outcome (PASO) criterion. Results: Of 91 enrolled patients, 71 (59% women, mean age 46 years, median length of follow-up 21 months) were suitable for evaluation. Thirty-four patients (48%) had complete clinical success according to the PASO criteria. The most relevant factors associated with complete clinical success on univariate analysis were: absence of diabetes (p= 0.007), low body mass index (BMI) (p= 0.001), lower preoperative DDD (p= 0.01), preoperatively controlled blood pressure (p= 0.024), higher plasma aldosterone to renin ratio (ARR) (p= 0.001), adenoma subtyping (p <0.001) and aldosteronoma resolution score (ARS) (p= 0.002). Multivariate regression analysis showed that the major predictors of complete clinical success were absence of diabetes (OR: 5.205), a BMI < 30 (OR: 4.930), a plasma ARR > 332 (OR: 4.554) and an ARS ≥ 3 (OR: 2.056). Conclusion: Complete and partial clinical response rates were achieved in respectively 48 and 43% of cases. The main predictors of complete resolution of hypertension were absence of diabetes, low BMI, high plasma ARR and high ARS. Taking these factors into account may help identify patients at risk of persistent postoperative hypertension who may require long-term surveillance and medication.

Atypical parathyroid adenoma: A case report.

The atypical parathyroid adenoma is a histological diagnosis. It is a parathyroid tumor with atypical histological features different from an adenoma and not similar enough to be considered as a carcinoma. It has an uncertain malignant potential. We report the case of a 55-year-old woman, diagnosed with severe hypercalcemia during her follow-up with the rheumatologist for osteoporosis. The laboratory testing led to the detection of a very important level of parathormone PTH 1325.62 pg/ml (20 fold the normal level), confirming the diagnosis of primary hyperparathyroidism. Investigations of the potential causes led to the presence of a large left inferior parathyroid adenoma. The patient was operated and on the examination of the removed tissue, the histopathological examination concluded to an atypical parathyroid adenoma, and the post-operatory PTH level dropped significantly to 152 pg/ml. The atypical parathyroid adenoma is a very rare tumor, and the diagnosis is still a challenge, the outcome of patients is not well known yet, there for the surveillance is important and must be regularly.

Synchronous parathyroid carcinoma and papillary thyroid carcinoma.

The simultaneous occurrence of parathyroid carcinoma and nonmedullary thyroid carcinoma is unusual. We report the case of 60-year-old woman who was found to have concurrent parathyroid carcinoma with severe clinical manifestations of primary hyperparthyroidism in addition to an incidental papillary thyroid carcinoma. Parathyroid hormone level was 569 pg/ml (normal range 10-65), and the serum calcium concentration was 13.83 mg/dl (normal range, 8.8-10.4). Preoperative investigation found a large 3 cm anterior cervical nodule suggestive of parathyroid adenoma. Total thyroidectomy and left parathyroidectomy were performed, and the final anatomopathological examination of the operative specimen concluded the coexistence of papillary microcarcinoma and parathyroid carcinoma. Although parathyroid carcinoma is an uncommon cause of hypercalcemia, it should be considered when severe hypercalcemia is observed, and in case of coexistence of thyroid nodules. The possibility of both malignancies must also be considered since parathyroid and nonmedullary thyroid carcinoma rare cases have previously been reported.

Non-islet-cell tumor hypoglycemia as first manifestation of an advanced hepatocellular carcinoma.

In a nondiabetic patient, fasting hypoglycemia is uncommon and warrants careful assessment. Although rare, NICTH should be considered in patients with recurrent hypoglycemia especially in those with risk factors for HCC.

Pheochromocytoma presenting as fulminant myocarditis mimicking COVID-19 pneumonia.

Adrenergic cardiomyopathy is uncommon but can be fulminant and life-threatening. Nowadays, the need to exclude the possibility of COVID-19 pneumonia in patients with acute dyspnea in a previously healthy adult may cause a delay in the diagnosis.

Langerhans Cell Histiocytosis of the Thyroid Leading to the Diagnosis of a Disseminated Form.

Langerhans cell histiocytosis (LCH) is a rare sporadic proliferative disorder of Langerhans cells. LCH rarely involves the thyroid gland. We report herein a case of a disseminated chronic form of LCH with a diagnosis established by histological examination of the thyroid gland. It is about a 37-year-old female who underwent total thyroidectomy for a thyroid nodule of the right lobe. Histological study showed a granulomatous thyroiditis, and the immunohistochemistry study revealed a strong positivity of histiocytes for the CD1 antigen and for the S100 protein. The incidence of LCH involving the thyroid gland, either as an isolated lesion or as a part of multisystemic disease, is extremely rare.

[Anicteric cholestasis: a rare hepatic dysfunction caused by hyperthyroidism]. / Cholestase anictérique: une manifestation hépatique rare d'hyperthyroïdie.

Clinical manifestations and visceral involvements described in patients with thyroid disease are many and varied. However, cases of patients with hepatic manifestations have been rarely reported in the literature. We here report the case of a 50 year old patient diagnosed with Graves' disease without a goiter or ocular manifestations. Follow-up laboratory testing led to the detection of anicteric cholestasis. Investigations of the potential causes were negative and given the normalization of the liver parameters after appropriate treatment of hyperthyroidism, the diagnosis of hepatic involvement secondary to Graves' disease was retained. The purpose of this study was to report a new case of rare hepatic manifestation (cholestasis anictérique) in a patient with hyperthyroidism: Graves' disease.