Profound Hypokalemia in Diabetic Ketoacidosis: A Therapeutic Challenge

ObjectiveTo describe profound hypokalemia in a comatose patient with diabetic ketoacidosis.MethodsWe present a case report, review the mechanisms for the occurrence of hypokalemia in diabetic ketoacidosis, and discuss its management in the setting of hyperglycemia and hyperosmolality.ResultsA 22-year-old woman with a history of type 1 diabetes mellitus was admitted in a comatose state. Laboratory tests revealed a blood glucose level of 747 mg/dL, serum potassium of 1.9 mEq/L, pH of 6.8, and calculated effective serum osmolality of 320 mOsm/kg. She was intubated and resuscitated with intravenously administered fluids. Intravenous administration of vasopressors was necessary for stabilization of the blood pressure. Intravenous infusion of insulin was initiated to control the hyperglycemia, and repletion of total body potassium stores was undertaken. A total of 660 mEq of potassium was administered intravenously during the first 12.5 hours. Despite such aggressive initial repletion of potassium, the patient required 40 to 80 mEq of potassium daily for the next 8 days to increase the serum potassium concentration to normal.ConclusionProfound hypokalemia, an uncommon initial manifestation in patients with diabetic ketoacidosis, is indicative of severe total body potassium deficiency. Under such circumstances, aggressive potassium repletion in a comatose patient must be undertaken during correction of other metabolic abnormalities, including hyperglycemia and hyperosmolality. Intravenously administered insulin should be withheld until the serum potassium concentration is ≥ 3.3 mEq/L. (Endocr Pract. 2005;11:331-334)     

Significant Hypercortisolism During Fractionated Radiotherapy in a Patient with a Large Corticotroph Adenoma: A Case Report and Literature Review

ObjectiveWe describe a patient with a large, invasive corticotroph adenoma who developed severe hypercortisolism shortly after starting fractionated radiotherapy.MethodsWe reviewed the patient’s clinical course, along with relevant literature for similar reported cases.ResultsA 29-year-old man was referred for radiotherapy for a residual and recurrent, invasive corticotroph adenoma. Prior to radiotherapy, he had a normal urine free cortisol (UFC) level of 44.7 μg/24 hours, with minimal symptoms. Within 2 weeks of radiotherapy, he developed hypertension, ankle edema, and hypokalemia (potassium level, 2.8 mEq/L), with a markedly elevated UFC level of 9,203 μg/24 hours. His UFC gradually decreased and normalized by the end of radiotherapy. One month later, the patient became adrenal insufficient, with a nondetectable 24-hour UFC. His adrenal function slowly recovered in 3 months. We are aware of only one previous case report of clinically significant hypercortisolism following radiotherapy in Cushing disease.ConclusionRadiotherapy may result in acute severe hypercortisolism in patients with a large corticotroph adenoma. This uncommon, but clinically significant, acute adverse effect of radiotherapy suggests that clinical observation and biochemical monitoring during or soon after radiotherapy may be indicated. (Endocr Pract. 2014;20:e166-e170)     

Diagnostic Challenges in Adrenocortical Carcinoma: Recommendations for Surveillance after Surgical Resection of Selected Adrenal Nodules

ObjectiveTo discuss challenges in the diagnosis of adrenocortical carcinoma and to suggest surveillance measures after removal of selected adrenal nodules.MethodsWe present the case of a 65-year-old man with worsening hypertension and new-onset hypokalemia attributed to primary hyperaldosteronism due to a 3-cm right adrenal nodule.ResultsA laparoscopic right adrenalectomy was performed, and the histologic diagnosis was a benign adenoma. The patient’s hypertension and hypokalemia improved postoperatively but recurred 8 months later, and florid Cushing’s syndrome developed. Magnetic resonance imaging showed an 8-cm mass in the right adrenal bed and multiple hepatic metastatic lesions. Fine-needle biopsy confirmed the presence of adrenocortical carcinoma.ConclusionDespite a comprehensive biochemical, radiologic, and histologic assessment, the diagnosis of adrenocortical carcinoma can be missed. Particularly, we caution against undue reliance on the initial tumor size. We recommend that abdominal imaging be performed every 3 months for the first year and every 6 months for the second year after surgical removal of selected adrenal nodules. (Endocr Pract. 2007;13:636-641)     

Hyponatremia: Mechanisms and Newer Treatments

ObjectiveTo review the neural and renal mechanisms of osmotic homeostasis, provide a rationale for the sensitivity of the central nervous system to hyponatremia, and outline modern approaches to therapy of acute and chronic hyponatremia.MethodsReview of relevant literature with focus on physiologic mechanisms.ResultsWith careful monitoring, acute hyponatremia can be managed, while minimizing risks both of continued hyponatremia and the osmotic demyelination that can occur with overly rapid correction of severe hyponatremia. Chronic hyponatremia due to disorders of volume regulation (congestive heart failure or cirrhosis) or to syndrome of inappropriate antidiuretic hormone release can be managed effectively with vasopressin V2 receptor antagonists, but there is no evidence that controlling the hyponatremia enhances survival associated with the underlying diseases.ConclusionsTherapy in the acute setting balances the risk of the osmotic disturbance with the risk of overly rapid correction. The V2 receptor antagonist tolvaptan has enhanced our ability to improve chronic hyponatremia in conditions such as congestive heart failure, cirrhosis, and syndrome of inappropriate antidiuretic hormone hypersecretion. (Endocr Pract. 2010;16:882-887)     

Pheochromocytoma With Histologic Transformation to Composite Type,Complicated By Watery Diarrhea,Hypokalemia, and Achlorhydria Syndrome

ObjectiveTo describe the rare occurrence of histologic transformation of a pheochromocytoma to a composite type of tumor during a long-term follow-up, which was complicated by watery diarrhea, hypokalemia, and achlorhydria syndrome.MethodsWe report the case of a 12-year-old girl who presented with headache, hypertension, and elevated catecholamine levels in the blood and urine. A tumor was found in the right adrenal gland and resected. When she was 15 years of age, multiple metastatic nodules were found in the lung and liver. Intensive chemotherapy was ineffective, and she underwent follow-up with conservative therapy. At 25 years of age, she complained of diarrhea. Laboratory studies revealed hypokalemia and an increase in the level of serum vasoactive intestinal polypeptide (VIP). A year later, she died of extensive metastatic disease. The primary and recurrent tumors at autopsy were histologically examined.ResultsThe primary tumor was pure pheochromocytoma, and the tumors at autopsy were a composite type of pheochromocytoma and ganglioneuroma. Only a few VIP-positive cells were found in the primary tumor, whereas both pheochromocytoma and ganglioneuroma cells of composite tumors were frequently positive for VIP.ConclusionOur case showed histologic transformation from pheochromocytoma to a composite type of tumor during a 14-year clinical course, which was associated with additional hormone production and a change in symptoms. Careful attention should be paid to the alteration of endocrine symptoms and hormone levels during prolonged follow-up of pheochromocytoma in young patients. (Endocr Pract. 2012;18:e91-e96)     

Unusual Cause of Ectopic Secretion of Adrenocorticotropic Hormone: Cushing Syndrome Attributable to Small Cell Prostate Cancer

ObjectiveTo report a rare cause of ectopic adrenocorticotropic hormone (ACTH) secretion leading to severe Cushing syndrome.MethodsWe describe the clinical presentation and management of a case of Cushing syndrome attributable to ectopic ACTH secretion from small cell cancer of the prostate.ResultsIn a 70-year-old man with hypertension and diabetes, congestive heart failure developed. He was found to have severe hypokalemia (serum potassium, 1.7 mEq/L) and a huge pelvic mass on a computed tomographic scan performed because of a complaint of urinary retention. Transurethral biopsy of the prostate showed features of small cell prostate cancer. Hormonal evaluation revealed a high urine free cortisol excretion of 6,214.5 μg/d (reference range, 36 to 137), confirming the diagnosis of Cushing syndrome. A serum ACTH level was elevated at 316 ng/dL (reference range, 10 to 52). An overnight highdose (8 mg orally) dexamethasone suppression test was positive (serum cortisol levels were 43.2 and 41 μg/dL before and after suppression, respectively), and magnetic resonance imaging of the pituitary gland disclosed no abnormalities. A prostate biopsy specimen showed small cell prostate cancer with positive staining for ACTH. The tumor was found to be unresectable, and the poor condition of the patient did not allow for bilateral adrenalectomy. He was treated with ketoconazole and metyrapone, which yielded good temporary control of his Cushing syndrome (24-hour urine free cortisol decreased to 55.2 μg/d). He received 1 cycle of chemotherapy (etoposide and cisplatin), but he died 6 months later as a result of sepsis.ConclusionSmall cell prostate cancer is a rare subtype that can be associated with ectopic secretion of ACTH and severe Cushing syndrome. With this subtype of prostate cancer, Cushing syndrome should be considered and appropriately managed. (Endocr Pract. 2010;16:249-254)     

Impact of a Hyperglycemic Crises Protocol

ObjectiveThe purpose of this study was to evaluate the efficacy and safety of an adult hyperglycemic crises protocol based upon the 2009 American Diabetes Association (ADA) consensus statement.MethodsWe performed a retrospective review of patients treated before and after protocol implementation at a university teaching hospital. A total of 256 adult patients met the criteria for diabetic ketoacidosis (DKA) or hyperosmolar hyperglycemic state (HHS) and were treated with an insulin infusion between February 2011 and February 2012 (nonprotocol n = 143, protocol n = 113). Protocol efficacy was evaluated by assessing time to resolution of DKA or HHS, length of stay (LOS) in the intensive care unit (ICU), and LOS in the hospital. Protocol safety was evaluated by assessing the numbers of patients with hypoglycemic and hypokalemic events.ResultsPatients on the hyperglycemic crises protocol experienced a 9.2 hour (95% confidence interval (CI): 4.70-13.70; P<.001) decrease in time to resolution, with nonprotocol patients (n = 143) resolving in 22.78 hours and protocol patients (n = 113) resolving in 13.58 hours. There was no difference in safety outcomes, including the number of patients with moderate hypoglycemia (blood glucose <70 mg/dL), severe hypoglycemia (blood glucose <50 mg/dL), or hypokalemia (K⁺ <3.3 mmol/L).ConclusionImplementation of a hyperglycemic crises protocol decreased times to resolution of DKA and HHS without increasing the rate of hypoglycemia or hypokalemia. (Endocr Pract. 2013;19:953-962)     

A Case Of Severe Neuropat Hy Associated With Hypertriglyceridemia

ObjectiveTo describe a case of severe neuropathy associated with hypertriglyceridemia.MethodsWe describe the clinical and laboratory findings of the study patient and review the relevant literature.ResultsA 45-year-old woman presented to the emergency department with recurrent abdominal pain and severe peripheral neuropathy. Her laboratory data revealed elevated lipase and a very high triglyceride concentration (> 10 000 mg/dL), consistent with a diagnosis of recurrent hypertriglyceridemia-induced pancreatitis. Workup for peripheral neuropathy showed normal concentrations of thyrotropin, fasting blood glucose, vitamin B₁₂, and creatinine, as well as a normal hemoglobin A_1c level, serum protein electrophoresis, and urine protein electrophoresis. Rapid plasma reagin antibodies, antinuclear antibodies, and lyme antibodies were not detected. In the absence of other identifiable causes, hypertriglyceridemia was deemed the likely etiology of severe neuropathy in this patient.ConclusionsPeripheral nerve conduction abnormalities can be identified in patients with mild hypertriglyceridemia in the absence of symptoms. Early recognition and aggressive management of hypertriglyceridemia may prevent the complications of severe peripheral neuropathy.(Endocr Pract. 2008;14:1020-1022)     

Successful Long-Term Treatment of Cushing Disease with Mifepristone (RU486)

ObjectiveWe describe a girl with Cushing disease for whom surgery and radiation treatments failed and the sub- sequent clinical course with mifepristone therapy.MethodsWe present the patient’s clinical, biochemi- cal, and imaging findings.ResultsA 16-year-old girl presented with classic Cushing disease. After transsphenoidal surgery, Cyberknife radiosurgery, ketoconazole, and metyrapone did not control her disease, and she was prescribed mifepristone, which was titrated to a maximal dosage of 1200 mg daily with subsequent symptom improvement. Mifepristone (RU486) is a high-affinity, nonselective antagonist of the glucocor- ticoid receptor. There is limited literature on its use as an off-label medication to treat refractory Cushing disease. Over her 8-year treatment with mifepristone, her therapy was complicated by hypertension and hypokalemia requir- ing spironolactone and potassium chloride. She received a 2-month drug holiday every 4 to 6 months to allow for withdrawal menstrual bleeding with medroxyprogester- one acetate. Urinary cortisol, serum cortisol, and cortico- tropin levels remained elevated during mifepristone drug holidays. While on mifepristone, her signs and symptoms of Cushing disease resolved. Repeated magnetic resonance imaging demonstrated stable appearance of the residual pituitary mass. Bilateral adrenalectomy was performed, and mifepristone was discontinued after 95 months of medical therapy.ConclusionsWe describe the longest duration of mifepristone therapy thus reported for the treatment of refractory Cushing disease. Mifepristone effectively controlled all signs and symptoms of hypercortisolism. Menstruating women who take the drug on a long-term basis should receive periodic drug holidays to allow for menses. The lack of reliable serum biomarkers to monitor the success of mifepristone therapy requires careful clini- cal judgment and may make its use difficult in Cushing disease. (Endocr Pract. 2012;18:e114-e120)     

Endocrine Disturbances in Empty Sella Syndrome: Case Reports and Review of Literature

ObjectiveTo report 5 cases of empty sella syndrome (ESS) manifesting with various degrees of pituitary dysfunction.MethodsWe describe the initial manifestations in 5 patients with primary ESS and in previous cases of ESS reported in the English language literature.ResultsReview of our recent medical records identified 5 patients referred for evaluation of pituitary deficiencies in whom ESS was diagnosed. Glucocorticoid replacement was required in 3 patients, 2 of whom presented initially with symptoms of severe glucocorticoid deficiency. In each case, magnetic resonance imaging of the brain demonstrated an empty sella.ConclusionOur cases suggest that endocrine abnormalities are not rare as the initial manifestation of ESS and that, contrary to many studies in the literature, the endocrine abnormalities may be quite severe. (Endocr Pract. 2005;11:120-124)     

The Endocrinopathies of Anorexia Nervosa

ObjectiveTo describe the hormonal adaptations and alterations in anorexia nervosa.MethodsWe performed a PubMed search of the English-language literature related to the pathophysiology of the endocrine disorders observed in anorexia nervosa, and we describe a case to illustrate these findings.ResultsAnorexia nervosa is a devastating disease with a variety of endocrine manifestations. The effects of starvation are extensive and negatively affect the pituitary gland, thyroid gland, adrenal glands, gonads, and bones. Appetite is modulated by the neuroendocrine system, and characteristic patterns of leptin and ghrelin concentrations have been observed in anorexia nervosa. A thorough understanding of refeeding syndrome is imperative to nutrition rehabilitation in these patients to avoid devastating consequences. Although most endocrinopathies associated with anorexia nervosa reverse with recovery, short stature, osteoporosis, and infertility may be long-lasting complications. We describe a 20-year-old woman who presented with end-stage anorexia nervosa whose clinical course reflects the numerous complications caused by this disease.ConclusionsThe effects of severe malnutrition and subsequent refeeding are extensive in anorexia nervosa. Nutrition rehabilitation is the most appropriate treatment for these patients; however, it must be done cautiously. (Endocr Pract. 2008;14:1055-1063)     

Two Cases of Thyroid Storm-Associated Cholestatic Jaundice

ObjectiveTo describe the association of the rare and serious complication of jaundice with severe thyrotoxicosis, a potentially lethal endocrine disorder.MethodsWe report the clinical, laboratory, and pathologic findings of 2 cases of severe jaundice (total bilirubin levels: 35.2 mg/dL in case 1 and 42 mg/dL in case 2) associated with thyroid storm in the absence of a history of liver disease, thionamide exposure, or congestive heart failure. We also present other relevant reports available in the literature.ResultsCase 1 was a 38-year-old woman who presented with nausea, vomiting, fatigue, pruritus, and frequent nonbloody diarrhea. She was transferred to our institution because of worsening hyperbilirubinemia. Case 2 was a 35-year-old woman admitted to a community hospital with thyroid storm and jaundice. Upon transfer to our institution, the patient was unconscious, mechanically ventilated, and in atrial fibrillation. In case 2, liver biopsy results revealed diffuse hepatocellular ballooning with intrahepatic cholestasis with mild portal lymphocytic infiltration. Both patients presented with severe cholestatic jaundice in the absence of congestive heart failure; underlying liver disease (infectious or autoimmune); or previous exposure to thionamides, other hepatotoxic agents, or complementary and alternative medications. In both cases, jaundice responded to therapy with antithyroid medications. Both patients eventually underwent thyroidectomy with complete resolution of the jaundice.ConclusionThe data strongly suggest that in these patients, the hepatic dysfunction was primarily due to hyperthyroidism. These cases indicate that the mere presence of hyperbilirubinemia during severe thyrotoxicosis should not per se delay the use of potentially life-saving thionamides once a thorough evaluation for other causes of liver disease has been completed. (Endocr Pract. 2007;13:476-480)     

Apparent Mineralocorticoid Excess Caused by a Novel Mutation in 11-β Hydroxysteroid Dehydrogenase type 2 Enzyme: its Genetics and Response to Therapy

ObjectiveTo present a case of apparent mineralocorticoid excess (AME) due to a novel mutation in the HSD11B2 gene and describe the patient’s response to therapy.MethodsThe clinical, biochemical, and genetic features of the proband and his family are presented. For the genetic study, DNA was extracted from peripheral leucocytes. The exons and exon-intron boundaries were polymerase chain reaction (PCR)-amplified and directly sequenced.ResultsA 10-year-old male presented with hypertension (HTN) and weakness and was found to have hypokalemia of 2.6 mmol/L. Plasma renin was undetectable, and plasma and urinary aldosterone were low. Serum cortisol and deoxycorticosterone were normal. Daily urinary excretion of cortisol was normal, but urinary and serum cortisone levels were undetectable. The patient was treated with spiranolactone with inadequate response. A small dose of dexamethasone was added and led to excellent control of HTN and hypokalemia. Genetic studies showed a novel missense biallelic mutation changing guanine to adenine in exon 3 (c.G526A) of the HSD11B2. This mutation changes the amino acid aspartic acid to asparagine at codon 176 (p.D176N). A monoallelic form of the same mutation was found in the parents and 3 of his 4 healthy siblings but not in a healthy sister or 100 normal subjects.ConclusionsA case of AME due to a novel mutation in HSD11B2 showed the usual features of AME but exhibited an inadequate response to spironolactone. A small dose of dexamethasone resulted in an excellent response. (Endocr Pract. 2014;20:e151-e156)     

Danger of Hypoglycemia Due to Acute Tramadol Poisoning

ObjectiveTo report a case of prolonged hypoglycemia after acute tramadol poisoning.MethodsWe describe a patient’s clinical presentation and outcome with prolonged hypoglycemia attributable to acute tramadol poisoning. In addition, the possible mechanism for the hypoglycemia is discussed, and a brief review of the pertinent literature is presented.ResultsA 54-year-old woman had previously under- gone a partial hepatectomy because of involvement of her liver by a gastrointestinal stromal tumor. After ingestion of 3,000 mg of tramadol with suicidal intent, she developed prolonged hypoglycemia that necessitated treatment with continuous intravenous glucose infusion for 24 hours. Reports in the literature have described central nervous system depression, nausea, vomiting, tachycardia, seizures, and even death from tramadol overdoses.ConclusionThis report alerts clinicians to the potential danger of severe hypoglycemia in tramadol poisoning. (Endocr Pract. 2012;18:e151-e152)     

Association of Dengue Hemorrhagic Fever With Multiple Risk Factors for Pituitary Apoplexy

ObjectiveTo describe pituitary apoplexy that developed during the course of dengue hemorrhagic fever.MethodsWe describe the clinical findings, laboratory test results, imaging findings, and clinical course of the study patients.ResultsPatient 1 was a 40-year-old man who developed clinical signs and symptoms of dengue, which was confirmed by serologic testing. He presented with thrombocytopenia and developed severe headache and vomiting. During hospitalization, acromegaly was suspected because of the characteristic disease phenotype. Magnetic resonance imaging confirmed the diagnosis of pituitary apoplexy. Subsequently, the biochemical diagnosis of acromegaly was confirmed, and the patient underwent transsphenoidal surgery. Histopathologic examination showed signs of recent bleeding. Patient 2 was a 38-year-old man with a macroprolactinoma, who had been treated with cabergoline for 10 weeks and had shown improvement on laboratory testing and imaging. The patient then presented with clinical symptoms of dengue (confirmed serologically) and thrombocytopenia. He developed bilateral hemianopsia, and magnetic resonance imaging showed enlargement of the pituitary adenoma with signs of intratumoral bleeding. The patient underwent transsphenoidal surgery, and histopathologic examination documented a pituitary adenoma diffusely infiltrated by blood cells.ConclusionsWe describe dengue as a probable novel condition for pituitary apoplexy because it may be associated with multiple risk factors for pituitary infarction or bleeding. Physicians should suspect pituitary apoplexy in patients with dengue hemorrhagic fever who develop a rapid onset of severe headache and vision defects, even in those without known pituitary adenomas. (Endocr Pract. 2012;18:e97-e101)     

严重高甘油三酯血症小鼠血液流变学的异常分析

目的:本研究旨在检测两种严重高甘油三酯血症(HTG)小鼠的血液流变学,建立具有异常血液流变学特点的HTG小鼠模型,以应用于进一步的相关机制研究。方法:采用ApoC3转基因与GPIHBP1基因敲除的HTG小鼠及野生型小鼠,所用小鼠均为C57BL/6J背景下8周龄雄性小鼠,通过生物物理法检测和比较其血浆粘度、红细胞渗透脆性、变形性与电泳率。结果:与野生小鼠相比,ApoC3转基因与GPIHBP1基因敲除的HTG小鼠血浆甘油三酯含量明显升高(P0.05),红细胞渗透脆性显著增加(P0.05),变形指数与电泳率明显降低(P0.05),血浆粘度显著升高(P0.05),但血细胞计数等血常规指标均未见显著差异(P0.05)。结论:Apo C3tg与GPIHBP1KO小鼠血液流变学发生异常改变,可能作为探讨HTG血液流变学异常和动脉粥样硬化心血管疾病病理机制的动物模型。     

Severe Hyponatre mia Due to Rosiglita zone Use in an Elderly Woman With Diabetes Mellitus: A Rare Cause of Syndrome of Inappropriate Antidiuretic Hormone Secretion

ObjectiveTo describe the first case of syndrome of inappropriate antidiuretic hormone secretion with lifethreatening hyponatremia due to rosiglitazone therapy.MethodsWe describe the clinical, laboratory, and imaging findings of the study patient.ResultsAn 89-year-old woman with a 5-year history of type 2 diabetes mellitus was admitted to the emergency department because of unconsciousness. She had reported generalized weakness for 15 days and nausea and vomiting for 3 days. Findings from laboratory analysis showed severe hyponatremia (sodium, 110 mEq/L). She had normal renal, cardiac, and adrenal function, and she did not have edema or volume depletion. The cause of hyponatremia was syndrome of inappropriate antidiuretic hormone secretion. We did not find any cause for her condition other than rosiglitazone, an antihyperglycemic drug that is increasingly being used in patients with type 2 diabetes mellitus. According to her medical history, rosiglitazone was prescribed 1 month previously after withdrawal of gliclazide. We stopped the rosiglitazone and administered hypertonic saline infusion to treat the hyponatremia. Saline infusion was stopped and blood sodium levels were stabilized in the normal range after 2 days. The patient’s plasma sodium concentration has remained in the reference range at follow-up visits.ConclusionsThis is the first reported case of syndrome of inappropriate antidiuretic hormone secretion as an adverse effect of rosiglitazone, and this drug should possibly be considered for addition to the list of drugs that cause this condition. (Endocr Pract. 2008;14:1017-1019)     

Apathetic Thyrotoxicosis Secondary to Atypical Subacute Thyroiditis

ObjectiveTo report a case of apathetic thyrotoxicosis with an etiology of subacute thyroiditis.MethodsWe describe the patient’s clinical findings, laboratory findings, and clinical course.ResultsAn 85-year-old woman with no history of thyroid disease presented with severe obtundation and altered mental status. Laboratory testing documented elevated free thyroxine and free triiodothyronine concentrations and a suppressed thyrotropin concentration. Thyroid antibodies were absent. A radioactive iodine study revealed severely diminished uptake, suggestive of thyroiditis. After a short course of steroids, the patient’s mental status returned to baseline. Follow-up laboratory testing showed normalizing thyroid function.ConclusionEven in the absence of a history of thyroid disease, we recommend considering thyroid dysfunction in the differential diagnosis of patients who present with altered mental status, particularly in the elderly population. (Endocr Pract. 2012;18:e127-e129)     

Gonadotropin-Releasing Hormone Agonist-Induced Pituitary Apoplexy in Treatment of Prostate Cancer: Case Report and Review of Literature

ObjectiveTo describe a case and review the literature on the rare complication of pituitary apoplexy after administration of a gonadotropin-releasing hormone agonist (GnRHa) for treatment of patients with prostate cancer.MethodsWe present a detailed case report of a patient with immediate signs of pituitary apoplexy after receiving a GnRHa and review the 6 previously reported cases in the literature.ResultsA 60-year-old man presented to a local hospital with severe headache, nausea, vomiting, and diplopia. Prostate cancer had recently been diagnosed, and he had received his first dose of a GnRHa 4 hours before this presentation. On physical examination, he was confused and had ptosis of the left eye. A head computed tomographic scan without contrast enhancement showed soft tissue filling the sella, without intracranial hemorrhage or mass effect. He was discharged with the diagnosis of viral meningitis. Three weeks later, he presented again with severe headache and diplopia. He had confusion, lethargy, disorientation, a blood pressure of 88/64 mm Hg, and left cranial nerve III, IV, and VI paralysis. Magnetic resonance imaging of the brain revealed an enhancing pituitary mass with hemorrhage extending to the optic chiasm, consistent with pituitary apoplexy. Laboratory results were consistent with panhypopituitarism. Surgical excision revealed a necrotic pituitary macroadenoma with hemorrhage. Tumor immunohistochemical staining was positive only for luteinizing hormone.ConclusionWe describe a rare adverse effect of GnRHa therapy, which unmasked a gonadotropin-secreting pituitary macroadenoma. This case adds to the 6 previously reported cases of GnRHa administration inducing pituitary apoplexy in men with prostate cancer. (Endocr Pract. 2007;13:642-646)     

Residual dysphasia after severe hypoglycemia in a patient with immune-mediated primary Adrenal insufficiency and type 1 diabetes mellitus: Case report and systematic review of the literature

ObjectiveTo describe a 24-year-old patient with immune-mediated primary adrenal insufficiency and type 1 diabetes mellitus (T1DM) receiving intensive diabetes management who was comatose at presentation attributable to severe hypoglycemia and had residual dysphasia after recovery and to summarize the related literature.MethodsWe present a case report and the findings on systematic review of the pertinent literature to identify the cumulative incidence of severe hypoglycemia with use of intensive insulin therapy in patients with primary adrenal insufficiency and T1DM and to determine the incidence of dysphasia after severe hypoglycemia.ResultsAfter 5 days of mechanical ventilation, our patient was revived. He had severe dysphasia after recovery of consciousness. Magnetic resonance imaging of the brain revealed encephalomalacia in the left temporal, frontal, and parietal lobes. After 6 years of follow-up, he continues to have residual deficits of expressive dysphasia and difficult-to-control seizures but no other neurologic disorders. Systematic review of the literature revealed that studies from the 1950s reported mortality due to hypoglycemia in such a cohort, but no recent studies have described the cumulative incidence of severe hypoglycemia in a cohort of patients with primary adrenal insufficiency and T1DM. To the best of our knowledge, we report the first findings on magnetic resonance imaging of the head in such a patient.ConclusionFortunately, residual dysphasia is an infrequent outcome after severe hypoglycemia. (Endocr Pract. 2007;13:384-388)