ACTA ENDOCRINOLOGICA (BUC)

Sustained exposure to high serum insulin-like growth factor (IGF-1) levels is likely to play a role in the development of the thyroid tumor in acromegaly; however, there is no report that indicate a promoting effect on the secretion of hormones by the thyroid tumor. We report a case of acromegaly in a seventy-one-year-old female with primary subclinical hyperthyroidism. Autoantibodies including\r\nanti-thyroglobulin antibody, anti-thyroid peroxidase antibody and thyroid stimulating hormone (TSH) receptor antibody were all negative. Ultrasonography of the thyroid\r\ndemonstrated a solid adenoma in the left thyroid lobe and Technetium-99m thyroid scintigraphy showed a high level of\r\naccumulation into the same lesion, indicating that toxic nodular goiter (TNG) was the cause of hyperthyroidism. Despite serum thyroid hormones remaining at normal levels,\r\nserum TSH levels went on decreasing gradually until transsphenoidal surgery to treat a growth hormone (GH) secreting pituitary adenoma. Interestingly, as IGF-I\r\nand GH levels normalized after operation, the serum TSH level increased and finally reached the normal level without\r\nintervention to the thyroid. Additionally, accumulation of Technetium-99m seen with thyroid scintigraphy slightly decreased. The evidence obtained from this case may\r\nsuggest a novel mechanism whereby excessive secretion of thyroid hormones from TNG in an acromegalic patient is\r\nregulated by IGF-I.

Cervical lymph nodes could be a starting sign for a complex diagnosis work-up. Depending on co-morbidities, medical unit and physician’s previous experience, the differential diagnosis includes thyroid malignancy, lymphoma, chronic infectious disorders, etc.

Thyroid isthmus agenesia (TIA) which is characterized by the absence of isthmus is one of these anomalies and its etiology is unclear. Marine Lenhart Syndrome (MLS), on the other hand, is defined as the coexistence of Graves disease and hyperactive nodules,although the diagnostic criteria are not clearly defined. Fifty five years old male patient with no concomitant disease had been diagnosed with MLS and accompanying subclinical hyperthyroidy for two years. He had dysphagia for a year. Neck ultrasound revealead retrosternal goiter with multiple hypoechoic nodules with the largest size of 33x30 mm. He had normal FT3 and FT4 levels with a decreased level of TSH. Scintigraphy revealed diffuse thyroid uptake with a hyperactive nodule of 16x11 mm on right upper lobe. Due to compressive sypmtoms and MLS he underwent surgery and diagnosed with thyroid isthmus agenesia peroperatively. MLS and TIA are rare thyroid pathologies and their etiopathogenesis has not been clarified yet. This case is thought to be the first case in which MLS and TIA coexistence was reported. Even though it is a rare disorder,having a basic knowledge about TIA can be an important step in establishing a treatment plan and avoiding possible complications. Especially in management of MLS, when surgery is considered the physician should be careful for presence of a TIA.

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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 reassessed 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.

Introduction. Pituitary apoplexy is a rare, acute, and life threatening condition due to haemorrhage or necrosis within a pituitary adenoma. Its prognosis may be poor leading to a fatal issue, or be good inducing a decrease or cure of the pituitary adenoma as in the case presented.\r\nCase report. A man, aged 28 years, having a history of well proved acromegaly [Growth hormone =GH=205ng/mL (n<5),\r\nand pituitary adenoma measuring 19x16mm] with diabetes mellitus (Glycaemia 4g/L, glycosuria++++, with ketoacidosis) treated with insulin for one year, was referred for\r\nnumerous hypoglycaemias which obliged him to stop insulin and diet. Just before, he had an acute episode of headaches, nauseas and vomiting. Clinical exam showed typical\r\nacromegaly, but pituitary assessment demonstrated low GH=0.05ng/mL, normal IGF1, without pituitary deficits. Routine analyses were normal. Fasting glycaemias, and glycaemias after glucose loading were normal too. Cerebral MRI showed a significant decrease in pituitary tumour (10 x\r\n16mm). Ten years later, glycaemias, GH, IGF1, and other pituitary functions remained normal, the necrotic pituitary process decreased up to 4.5 mm in height.\r\nConclusion. This clinical history and outcome argued for a pituitary apoplexy secondary to apparently spontaneous\r\nnecrosis of a somatotrop adenoma that induced a total cure of acromegaly and diabetes mellitus.

Pituitary apoplexy (PA) is a life-threatening clinical syndrome. Dopamine receptor agonists are the drugs of choice in the treatment of prolactinomas. The use of cabergoline is reported to cause an increased risk of PA, particularly in macroprolactinomas of cystic nature. In this report, we present a patient with a cystic macroprolactinoma who developed PA on the 16th week of cabergoline treatment.

Objective. This study aims to determine the frequency and prognostic significance of lactic acidosis in children with diabetic ketoacidosis (DKA) admitted to the pediatric intensive care unit. Methods. The study was carried out retrospectively by examining the patients admitted to the pediatric intensive care unit for the treatment of DKA. The ages of the patients ranged from 2 to 18 years. The patients with the following parameters were enrolled in the study: serum blood glucose>200 mg/dL, ketonuria presence, venous blood gas pH ≤7.1, bicarbonate <15. Results. A total of 56 patients were included in the study with a mean age of 111.07 ± 51.13 months. The recovery time from DKA was 16.05 ± 6.25 h in the group with low lactate level and it was 13.57 ± 8.34 h in the group with high lactate level with no statistically significant difference. There was a negative correlation between lactate levels and the recovery time from DKA. Conclusion. Lactic acidosis is common in DKA, and unlike other conditions, such as sepsis, it is not always a finding of poor prognosis that predicts the severity of the disease or mortality. We think that high lactate may even protect against possible brain edema-cerebral damage in DKA.

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Context. Phosphoinositide-3-kinase (PI3K) pathway inhibitors are increasingly used as targeted therapy in malignancies. We discuss here three cases of PI3K inhibitor induced hyperglycemia and discuss the mechanism of action of these medications and treatment of this class side effect. Objectives. Alpelisib (Piqray) is the newest PI3K inhibitor used in conjunction with Fulvestrant to treat specific types of breast cancer. Since PI3K is a critical mediator of insulin signaling, hyperglycemia is an on-target, unfortunate side effect of this treatment. We present a case series of severe hyperglycemia induced by the alpelisib in three women without a history of diabetes. Design. All three women in this study had hormone receptor (HR) positive, human epidermal growth factor receptor 2 (Her2) negative, PI3K mutated breast cancer. They were referred to our clinic by Oncology for alpelisibinduced hyperglycemia. Subjects and Methods. Review of laboratory values and glucometer values were conducted during each visit allowing treatment decisions. Two of these women are actively managed by us for their diabetes. One woman recently died due to progression of malignancy. Results. All three women presented with new onset of severe hyperglycemia after the initiation of PI3K inhibitor, alpelisib. At least one case noted maximal glucose elevation in the hours following drug ingestion. In another, cessation of Alpelisib reversed the hyperglycemia within the span of one week. Conclusion. Hyperglycemia induced by PI3K inhibitors can be recalcitrant and might necessitate interruption of chemotherapy. Optimal glucose-lowering therapy remains unclear as exogenous insulin has the theoretical potential to overcome PI3K inhibition.