ACTA ENDOCRINOLOGICA (BUC)

Background. Mosaic karyotype 45,X/46,XY related mixed gonadal dysgenesis. Aim. To report a case of mosaic karyotype and petroclival meningioma. Methods. Presentation of a clinical case with comments. Results. The case of a 37-year-old woman mosaic karyotype - 45,X/46,XY, infertility, virilisation, Turner syndrome-like phenotype, primary amenorrhea, the absence of labia majora and petroclival meningioma. Concentrations of dehydroepiandrosterone sulphate (DHEAS), testosterone, luteinizing hormone (LH) and follicular stimulating hormone (FSH) were increased indicating hypergonadotropic hypogonadism. Low and high dose dexamethasone suppression tests demonstrated incomplete suppression of DHEAS concentration without connection between pulses of LH/FSH and DHEAS. Response to adrenocorticotropic hormone (ACTH) was normal. The morning/evening concentration ratio of DHEAS was very low in comparison with cortisol, ACTH and testosterone. Head magnetic resonance imaging (MRI) demonstrated petroclival meningioma without any adrenal or ovary abnormality. Menstruation started after treatment with 2 mg of estradiol. At control visit 1.5 years later she had no complaints. MRI did not demonstrate any signs of tumour progression. Conclusions. The main lesson learned from this case is that in searching the DHEAS secreting tumours one can find unusual cases with sustained high DHEAS and lack of confirmations of polycystic ovary syndrome, adrenal or ovary tumours using available ultrasound, CT and MRI.

Introduction. Subacute thyroiditis (SAT) is a self-limiting thyroid disease associated with a triphasic clinical direction of hyperthyroidism, hypothyroidism and back to normal thyroid function. Precise etiology of this clinical condition is unknown. Generally diagnosis is based on clinical-laboratory parameters. Considerable cases of SAT develop after several types of viral infections. We herein describe two cases that developed SAT after dental extraction. Cases. Two-female patients in the forties ages experienced fever and neck pain after dental extraction. The patients presented elevated sedimentation rates and SAT symptoms. After clinical diagnosis and therapy administration, symptoms resolved after one week. Conclusion. We have presented two cases experiencing SAT after dental extraction. The management and possible association with current literature were discussed.

Denosumab,a monoclonal IgG2 antibody, is used as neoadjuvant therapy for giant cell bone tumors, particularly in inoperable or metastatic cases. It targets the receptor activator of nuclear factor kappa-β ligand (RANKL), which is overexpressed in tumor stromal cells. However, denosumab treatment can lead to side effects such as hypocalcemia during treatment and rarely but malignant hypercalcemia after discontinuation. The unpredictable onset time and persistent course of hypercalcemia attacks increase the duration of hospitalization and the risk of complications. Case. A 9-year-old girl with a giant cell bone tumor was treated with denosumab for diffuse tumor recurrence. Severe hypercalcemia occurred four months after completing therapy. Evaluation suggested "rebound-linked" hypercalcemia following denosumab discontinuation. IV bisphosphonate treatment normalized calcium levels initially, but hypercalcemia recurred, requiring repeated IV bisphosphonate administration. Oral alendronate was used weekly to prevent further attacks, resulting in stable calcium levels during follow-up. Results. Rebound hypercalcemia, as an unpredictable recurrent episode at any time, is a potential complication of denosumab cessation, and requires close monitoring post-treatment. Children may be at higher risk due to their rapid bone cycle. In long-term follow-up, IV and oral bisphosphonates can be used effectively in the management of especially life-threatening recurrent attacks.

Objective. We aimed to reach a diagnosis for patients with pituitary failure (PF) with more detailed multifaceted examination, to obtain new gains in this way and to apply this experience to our future patients. Patients and Method. The study included 72 patients with pituitary failure diagnosis. Patients were divided into two groups of 32 female patients with Sheehan syndrome (SS) and 40 non-Sheehan syndrome patients (20 female and 20 male). Results. The mean age of the 32 patients with Sheehan syndrome was 56.1±11.7 years with mean disease duration 20.3±8.3 years and mean age at disease onset 34.3±7.6 years. For the 40 non-SS pituitary failure patients mean age was 46.5±16.6 years, mean disease duration was 4.7±5.1 years and mean age at disease onset was 41.8±18.2 years. The prolactin response (PRL) on the TRH test was found to be significantly lower in SS patients compared to non-SS patients(p<0.05). Both patient groups had low basal pituitary hormones, with mean PRL level in SS patients 4.9±4.0 ng/mL which was significantly low compared to non-SS patients with 36.8±84.7 ng/mL(p=0.022). Conclusion. Pituitary failure patients may present with a broad range of clinical symptoms and findings. Early diagnosis and timely replacement treatment have vital importance for SS patients.

Context. Sodium-glucose co-transporter 2 inhibitors (SGLT2-I) are a new class of oral antidiabetic drugs. Objective. Side effects of drugs are frequently encountered with increasing use in clinical practice. Design/Subjects and Methods. We present a case with an unidentified body odor that has not been described yet in the literature as an adverse effect of SGLT2-I. Results. A 49-year-old female patient with Type 2 DM was applied to our clinic for routine control. She had been receiving sitagliptin/metformin 50/1000 mg b.i.d. Her fasting plasma glucose was 160 mg/dL and HbA1c was 8%. Empagliflozin 10 mg/day was added to therapy. The patient noticed that two weeks after starting empagliflozin treatment, she began to smell herself and felt a bad/foul odor was released from her body. The patient associated these complaints with the new drug(empagliflozin) stopped two weeks ago. She also stated that all these complaints had disappeared one week after stopping the medication and there were no complaints at the moment. Conclusions. Here we report an unidentified body odor as a potential empagliflozin-associated adverse event. There was a close temporal relationship between empagliflozin intake and the development and the occurrence of the described odor.

Thyroid carcinomas associated with Graves? ophthalmopathy had been described in the literature, before. The rate of this coincidence is 1.1% - 7.1%. Papillary carcinoma is more common than thyroid cancer in Graves? disease. Herein, we present a 37-year-old male Graves? disease patient with thyroid papillary carcinoma.

Background. Fine needle aspiration biopsy (FNAB) is an important tool in the diagnosis of thyroid nodules. Aim. Our aim was to investigate the malignancy criteria in thyroid nodules by gray-scale ultrasonography (US) and duplex Doppler ultrasonography (DDUS), and their usefulness in reducing the number of unnecessary FNAB’s. Study design. This was a prospective observational study. Subjects and methods. 181 benign and 18 malignant thyroid nodules were evaluated by US and DDUS before FNAB or thyroidectomy. US was used to note size, shape, internal structure, nodule echogenicity, marginal properties, peripheral hypoechogenic halo, and microcalcifications. DDUS studies were used to evaluate the maximum and minimum flow velocity (Vmax and Vmin), systolic/diastolic flow velocity ratio (S/D), pulsatility index (PI), resistive index (RI), acceleration time (AT) and acceleration value. Results. Contour irregularity, size and presence of microcalcifications (p<0.001, p=0.02 and p=0.002, respectively) and S/D, Vmin, PI, RI and AT were significantly different (p=0.004, p=0.007, p=0.032, p=0.003 and p=0.003, respectively) were significant for malignant nodules. Benign and malignant nodules with or without suspicious US findings had similar DDUS findings. Vmax, Vmin, PI, RI, and AT were significantly different in the presence of microcalcification (p=0.043, p=0.001, p=0.031, p=0.04, and p=0.019 respectively). AT was significantly different in the case of absence of microcalcification (p=0.019). Comparing the irregular margins, Vmin, PI and RI were significantly different (p=0.014, p=0.003, and p=0.014 respectively). Conclusion. Benign and malignant thyroid nodules can be differentiated using gray-scale US findings and DDUS based indices together to reduce the number of unnecessary FNAB’s.

Objective. The aim of this study was to compare the clinical effects of a triple oral antidiabetic combination versus basal insulin and metformin combination treatment in patients with poorly controlled type 2 diabetes.\r\nMethods. Eighty patients with type 2 diabetes, who were treated by metformin and sulphonylurea combination, and had\r\nHbA1c values between 7.5 and 10 % (58 and 86 mmol/L), were randomized into two groups. The first group was given triple oral antidiabetic therapy (pioglitazone, metformin, and sulphonylurea) and the second group was given metformin and a bedtime basal insulin (insulin detemir) combination for 12 weeks. Metabolic parameters were evaluated.\r\nResults. The mean fasting plasma glucose and HbA1c levels decreased in both groups. The decrease in HbA1c was slightly\r\nhigher in triple oral antidiabetic group (p=0.046). The patients in triple oral combination group gained 0.2 kg (p=0.881) and those in the metformin-insulin detemir combination group lost 1.7 kg (p=0.001) in 12 weeks (p=0.29 between groups). The frequency of hypoglycemia was higher in\r\ntriple oral antidiabetic group (11 vs. 2 episodes, respectively).\r\nConclusion. Both sulphonyureametformin-pioglitazone and insulin detemir-metformin therapies provided significant improvements in glycemic control. However, sulphonylurea,\r\npioglitazone and metformin combination led to more frequent hypoglycemic events, and weight management seemed in favor of insulin detemir-metformin combination.

Background. Paraganglioma develop from embryological neural crest cells from the base of the skull, mediastinum, retroperitoneum, urinary bladder and scrotum. The most common location of the retroperitoneal form is situated between the aorta and vena cava, at the level of left renal vein, or more distally, to the aortic bifurcation. The care of patients with paraganglioma is a challenge for many physicians and surgeons because of its clinical features and therapeutic implications. Case presentation. We report the case of a 22-years old female patient with symptomatic retroperitoneal paraganglioma who was successfully treated with complete surgical excision. The pathological report confirmed the presence of chromaffin tumours. Postoperative care was uneventful, the patient being discharged after 10 days. Follow-up evaluation showed no recurrence after 4 years. Conclusion. Although a very rare condition, retroperitoneal paraganglioma has to be considered in the differential diagnosis of retroperitoneal tumours. Complete surgical resection is crucial for treatment and histological assessment as these tumours are potentially curable if they are diagnosed correctly. In our experience, the best surgical approach is open surgical excision for large masses located in the interaortocaval region, with a clear operatory field and good control over the major blood vessels. Follow-up is strongly recommended.

Background. Iodine deficiency and/or thyroid autoimmunity are the most common causes of hypothyroidism development among pregnant women. In this study, we aimed to investigate the effect of iodine consumption and thyroid autoimmunity on TSH levels and abortus. The study sample consisted of 104 patients, 79 in abortus and 25 in control groups. TSH, free T4, free T3, anti-TPO, anti-Tg, spot urinary iodine concentrations, and thyroid volumes of the cases were measured by ultrasonography. The spot urine concentration was below 100 μg/L in 93% of the cases included in the study. The TSH levels of the abortus group cases were significantly higher than those of the controls (p=0.025). The percentage of subclinical hypothyroid cases were significantly higher among the cases evaluated due to abortus compared to the control group (p<0.001). Abortus and control groups did not differ statistically with respect to the presence of autoimmune thyroid diseases (p=0.424). Spot urine iodine concentrations of abortus cases with subclinical hypothyroid were significantly lower than those with TSH levels below the defined range (p=0.001). Spot urine iodine concentrations of the cases with subclinical abortus with negative thyroid autoantibodies were also significantly lower than those with TSH levels below the defined range (p=0.017). TSH levels above 1 μIU/mL for the first trimester and 2 μIU/mL for the second trimester may be indicators of iodine nutrition in pregnancy losses.