scholarly journals SAT-LB49 Persistent vs Recurrent Cushing’s Disease Diagnosed Four Weeks Post-Partum

2020 ◽  
Vol 4 (Supplement_1) ◽  
Author(s):  
Emily V Nosova ◽  
Joshua B Bederson ◽  
Khadeen Christi Cheesman
Keyword(s):  
Weight Gain ◽  
Cushing’S Disease ◽  
Diagnostic Tests ◽  
Post Partum ◽  
Cushing's Disease ◽  
Focal Lesion ◽  
Management Options ◽  
Lower Extremity Weakness ◽  
Urine Cortisol ◽  
Post Partum Period

Abstract Background: Cushing’s disease (CD) recurrence in pregnancy has previously been described and is thought to be associated with predictable estradiol fluctuations during gestation. CD recurrence in the immediate post-partum period has been reported once, but never in a patient with documented dormant disease during pregnancy.Clinical Case: A 30 year old woman with recently diagnosed pre-diabetes presented with weight gain, dorsal hump, depression, oligomenorrhea, and lower extremity weakness. Diagnostic tests were consistent with CD. Results included: three elevated midnight salivary cortisols: 0.33, 1.38 and 1.10 ug/dL (<0.010 - 0.090); 1-mg dexamethasone suppression test (DST) with cortisol 14 ug/dL (<1.8); elevated 24-hr urine cortisol 825 ug/24 hr (6-42); and ACTH 35 pg/mL (7.2-63.3). MRI of the pituitary gland revealed a left 4mm focal lesion. After transsphenoidal resection (TSA), day 1, 2, and 3 morning cortisol values were 18, 5, and 2 ug/dL, respectively. Pathology did not show a definitive pituitary neoplasm. She was rapidly titrated off hydrocortisone (HC) by six weeks post-resection. Her symptoms steadily improved. She resumed normal menses and conceived unexpectedly around 3 months post-TSA. She complained of severe fatigue in her late 2nd trimester. Given low 24-hr urine cortisol of 15 ug/24 hr at 36 weeks gestation, she was started on HC. She was induced at 40 weeks gestation for oligohydramnios and subsequently delivered a healthy baby boy. HC was discontinued immediately after delivery. Around four weeks post-partum she developed symptoms concerning for CD. Diagnostic tests showed elevated midnight salivary cortisol of 0.206 and 0.723 ug/dL and 24-hour urine cortisol of 400 ug/24 hr. MRI pituitary illustrated a 3mm adenoma in the left posterior gland which was thought to represent growth of residual tumor not clearly seen on post-op MRI. During repeat TSA, a discrete lesion was found and resected. Pathology confirmed corticotroph adenoma with MIB-1 < 3%. Post-operative day 1, 2, and 3 cortisol levels were 26, 10 and 2.8 ug/dL, respectively. She was tapered off HC within one month. Her symptoms improved only slightly and she continued to report weight gain, muscle weakness, and fatigue. Three months after repeat TSA, biochemical data showed 1 out of 2 midnight salivary cortisols elevated at 0.124 ug/dL and elevated urine cortisol of 76 ug/24 hr. MRI pituitary demonstrated a 3 x 5 mm left enhancement, concerning for residual or enlarged persistent tumor. Conclusion: We describe the first report of recurrent CD that was quiescent during pregnancy, and subsequently diagnosed in the immediate post-partum period. Treatment options for persistent or recurrent CD include aggressive surgical resection, radiation and/or medical therapy. In the context of additional family planning for this otherwise healthy, reproductive-age woman, ideal management options remain uncertain.

scholarly journals Vaspin in Serum and Urine of Post-Partum Women with Excessive Gestational Weight Gain

Medicina ◽  
2019 ◽  
Vol 55 (3) ◽  
pp. 76
Author(s):  
Marcin Trojnar ◽  
Jolanta Patro-Małysza ◽  
Żaneta Kimber-Trojnar ◽  
Monika Czuba ◽  
Jerzy Mosiewicz ◽  
...  
Keyword(s):  
Weight Gain ◽  
Gestational Weight Gain ◽  
Post Partum ◽  
Enzyme Linked Immunosorbent Assay ◽  
Hydration Status ◽  
Fatty Acid Binding ◽  
Gestational Weight ◽  
Excessive Gestational Weight Gain ◽  
Post Partum Period ◽  
Serum And Urine

Background and objectives: Data concerning vaspin in obstetric aspects are limited and conflicting. The aim of the study was to evaluate vaspin concentrations in the serum and urine of women with excessive gestational weight gain (EGWG) in the early post-partum period (i.e., 48 h after delivery), when placental function no longer influences the results. Materials and Methods: The study subjects were divided into two groups of 28 healthy controls and 38 mothers with EGWG. Maternal body composition and hydration status were evaluated by the bioelectrical impedance analysis (BIA) method. Concentrations of vaspin, fatty acid-binding protein 4 (FABP4), leptin, and ghrelin were determined via enzyme-linked immunosorbent assay (ELISA). Results: Serum vaspin levels were lower in the EGWG group, whereas no significant differences were noted between the groups, with regard to the urine vaspin concentrations. In both studied groups, the serum vaspin concentrations correlated positively with the urine FABP4 levels and negatively with gestational weight gain, body mass index gain in the period from pre-pregnancy to 48 h after delivery (ΔBMI), and fat tissue index (FTI). In the multiple linear regression models, the serum vaspin concentrations were positively dependent on the serum FABP4 levels, as well as negatively dependent on triglycerides, FTI, and ΔBMI. Conclusions: Our study revealed that the EGWG mothers were characterized by significantly lower serum vaspin concentrations in the early post-partum period compared with the subjects that had appropriate gestational weight gain. Our observation supports previous hypotheses that vaspin might be used as a marker of lipid metabolism in pregnancy and maternal adipose tissue. Considering the fact that FABP4 is widely referred to as a pro-inflammatory adipokine, further research on the protective role of vaspin seems crucial, especially in the context of its relationship to FABP4.

scholarly journals “The Adrenal Gland: Central Relay in Health and Disease - Current Challenges and Perspectives 2018” – Cushing’s Disease

2018 ◽  
Vol 127 (02/03) ◽  
pp. 147-155 ◽  
Author(s):  
Günter Stalla ◽  
Denis Ciato ◽  
Christina Dimopoulou
Keyword(s):  
Cushing’S Disease ◽  
Diagnostic Tests ◽  
Chronically Ill ◽  
Cushing's Disease ◽  
Targeted Drugs ◽  
Recurrence Rates ◽  
Pharmacological Agents ◽  
Late Night ◽  
Individualized Care ◽  
Challenging Situation

Abstract Background Despite advances in diagnostic and therapeutic approach, Cushing’s disease (CD) presents a challenging situation for the treating physician. Aims To elucidate current challenges, present strengths and pitfalls of existing diagnostic tests, enlighten the need for new diagnostic approaches, appraise the effects of surgery and available pharmacological agents and identify future perspectives regarding CD. Materials and methods Systematic search to PubMed and Medline databases for publications mainly over the last five years. Results Mutations in the ubiquitin specific peptidase 8 gene have been recently identified in functional sporadic corticotroph adenomas causing CD. Since the prevalence of obesity and metabolic syndrome is rapidly increasing, new diagnostic tests are necessary to differentiate these conditions. Next to the traditional tests, a cutoff of preoperative ACTH/cortisol ratio, an ultrasensitive late night salivary cortisol assay and the desmopressin test have been suggested as valid tools for the diagnosis and differential diagnosis of CD. Transsphenoidal surgery with variable remission and recurrence rates presents the treatment of choice for CD. Medical therapy consists of adrenal-targeted drugs e. g. ketoconazole, metyrapone, etomidate and mitotane and pituitary-targeted drugs e. g. pasireotide, cabergoline and retinoic acid. Conclusions CD is associated to a significant clinical burden, since numerous comorbidities persist after long-term biochemical control. These chronically ill patients show an increased mortality despite disease remission. Clinicians should treat comorbidities aggressively and seek for appropriate consultations. Structured consultation hours and expert excellence networks are needed in order to allow optimal, individualized care for affected patients, reverse increased morbidity and mortality and identify tumor recurrence early.

scholarly journals SAT-271 Block and Replace Therapy Successfully Improved Symptoms in Recurrent Cyclic Cushing’s Disease

2020 ◽  
Vol 4 (Supplement_1) ◽  
Author(s):  
Risa Kamigaki ◽  
Hiraku Kameda ◽  
Hiroshi Iesaka ◽  
Rimi Izumihara ◽  
Yuki Ohe ◽  
...  
Keyword(s):  
Weight Gain ◽  
Pituitary Adenoma ◽  
Cushing’S Disease ◽  
Brain Mri ◽  
Urinary Free Cortisol ◽  
Cushing's Disease ◽  
Subjective Symptoms ◽  
Free Cortisol ◽  
Level Elevation ◽  
Cortisol Levels

Abstract BACKGROUND: Cyclic Cushing’s disease is rare and treatments have not been established for post-surgical recurrent cases. Here, we report a patient with recurrent cyclic Cushing’s disease, whose subjective symptoms improved by administration of metyrapone and hydrocortisone. Clinical Case: A 45-year-old woman had exhibited face and peripheral edema, hyperphagia, weight gain, hair loss and limb numbness since September X-10. In May X-9, her ACTH and cortisol levels were high (87.8 pg/mL and 28.8 µg/dL, respectively), and she was referred to our department. A brain MRI revealed a pituitary adenoma of 7mm in diameter. Because blood ACTH and cortisol levels turned normal and typical Cushingoid features were absent at the admission to our department, cyclic Cushing’s disease was suspected. Later in September, because subjective symptoms recurred accompanied with blood cortisol level elevation, she was diagnosed as cyclic Cushing’s disease with the examinations including inferior petrosal sinus sampling. Transsphenoidal surgery was performed in November, and immunohistology confirmed ACTH-producing pituitary adenoma based on ACTH positivity. After the surgery, endocrine test results were normalized and subjective symptoms were ameliorated. In March X-3, the blood ACTH level increased again; however, no subjective symptoms were observed. From May X, she had experienced limb numbness, hyperphagia and weight gain again. MRI showed no apparent recurrence, but endocrine tests showed the activity of Cushing’s disease. Urinary free cortisol (UFC) increased to 300–400 µg/day in a 1-week cycle, indicating the recurrence of cyclic Cushing’s disease. Metyrapone treatment was initiated, and the patient was finally discharged after block and replace therapy with metyrapone 2,000 mg/day and hydrocortisone 15 mg/day. After metyrapone treatment, subjective symptoms improved and UFC was normalized. Conclusion: Block and replace therapy with metyrapone and hydrocortisone may be effective for recurrent cyclic Cushing’s disease, especially in cases with a very short cycle.

scholarly journals SAT-262 A Case of Pseudotumor Cerebri After Removal and Recurrence of ACTH-Producing Tumor

2020 ◽  
Vol 4 (Supplement_1) ◽  
Author(s):  
Karla Bermudez Saint Andre ◽  
Steven Petak ◽  
Laila Tabatabai
Keyword(s):  
Weight Loss ◽  
Cerebrospinal Fluid ◽  
Weight Gain ◽  
Cushing’S Syndrome ◽  
Cushing’S Disease ◽  
Pseudotumor Cerebri ◽  
Cushing's Syndrome ◽  
Cushing's Disease ◽  
Pituitary Macroadenoma ◽  
Second Surgery

Abstract Introduction: Pseudotumor cerebri also known as idiopathic intracranial hypertension (IIH) is a condition of elevated cerebrospinal fluid pressure which as a result causes headaches and vision problems. Several case reports and studies have reported a relation of IIH with Cushing’s disease (CD) in adults and children, particularly after surgical or medical therapy. We describe a very uncommon presentation of persistent intracranial hypertension (ICH) in a patient with recurrence of cushing’s disease after her initial surgical resection. Case presentation:38-year-old African-American female with a BMI of 45, type 2 diabetes, HTN and history of Cushing’s syndrome from an ACTH-producing pituitary macroadenoma. She initially presented with increasing weight gain and features of Cushing’s syndrome including hypertension, hyperglycemia, truncal obesity and moon facies. Imaging studies showed a 2 cm intrasellar mass with suprasellar extension without compression of the optic chiasm. Hormonal evaluation confirmed Cushing’s syndrome from an ACTH-producing pituitary macroadenoma. Patient underwent initial trans-sphenoidal hypophysectomy (TSR) and was tapered down to physiologic doses of glucocorticoids. Post-operatively she started complaining of significant headaches and transient vision loss. She followed up with neuro-ophthalmology and was diagnosed with papilledema that was not present in the pre-operative examination. An LP (lumbar puncture) was recommended to assess for ICH, however patient declined the procedure. A year after, a second surgery had to be performed for recurrent pituitary adenoma. Unfortunately, a repeat MRI pituitary shortly after her second surgery revealed recurrent pituitary macroadenoma of 2.2 cm. Patient continued with headaches and underwent an LP with an opening pressure of 28 cm H20 (ICH > 26 cm H2O). She underwent a third TSR with follow up MRI showing gross total resection of the previously seen pituitary mass. For her ICH she was started on acetazolamide but was not able to tolerate due to paresthesias and metallic taste. Her symptoms have improved after her last resection and last MRI brain shows no residual tumor. She is currently on furosemide and focusing on weight loss. Conclusion: Our patient’s presentation is an interesting and unusual case because we believe she had both pseudotumor cerebri (IIH) and real tumor cerebri from the complications of her ACTH-secreting macroadenoma. The cause of IIH after treatment of cushing’s disease is believed to be mostly due to steroid withdrawal after surgical resection or medical treatment comprising hormonal control of cerebrospinal fluid production and absorption. In our patient we suppose that the persistent weight gain caused by the recurrence of her CD could also contribute to her IIH. The treatment in general is the same with physiologic doses of corticosteroids, diuretics and weight loss.

Equine Cushing's Disease: Plasma Immunoreactive Proopiolipomelanocortin Peptide and Cortisol Levels Basally and in Response to Diagnostic Tests*

Endocrinology ◽  
1982 ◽  
Vol 110 (4) ◽  
pp. 1430-1441 ◽  
Author(s):  
DAVID N. ORTH ◽  
MYRON A. HOLSCHER ◽  
MARGARET G. WILSON ◽  
WENDELL E. NICHOLSON ◽  
RAYMOND E. PLUE ◽  
...  
Keyword(s):  
Cushing’S Disease ◽  
Diagnostic Tests ◽  
Cushing's Disease ◽  
Cortisol Levels

scholarly journals The Utility of Preoperative ACTH/Cortisol Ratio for the Diagnosis and Prognosis of Cushing's Disease

2018 ◽  
Vol 09 (01) ◽  
pp. 106-111 ◽  
Author(s):  
Alev Selek ◽  
Berrin Cetinarslan ◽  
Zeynep Canturk ◽  
Ilhan Tarkun ◽  
Ozlem Zeynep Akyay ◽  
...  
Keyword(s):  
Cushing’S Disease ◽  
Diagnostic Tests ◽  
Clinical Laboratory ◽  
University Hospital ◽  
Cushing's Disease ◽  
Control Group ◽  
Sustained Remission ◽  
Diagnosis And Prognosis ◽  
Diagnostic Difficulties ◽  
Cortisol Ratio

ABSTRACT Purpose: Cushing's syndrome (CS) is a rare disease having diagnostic difficulties. Many diagnostic tests have been defined but none of these are diagnostic alone. Determination of the cause is another problem which sometimes requires more sophisticated and invasive procedures. Therefore, we aimed to evaluate the utility of pretreatment plasma adrenocorticotropic hormone (ACTH)/cortisol ratios in patients with confirmed endogenous CS for the diagnosis and differential diagnosis of CS. Materials and Methods: This retrospective evaluation included 145 patients with the diagnosis of CS, 119 patients with Cushing's disease (CD), and 26 patients with ACTH-independent CS (AICS), in a university hospital. Furthermore, 114 individuals in whom CS diagnosis was excluded with at least one negative screening test were enrolled to the study as control group. The clinical, laboratory, imaging, postsurgical pathologic records and also clinical follow-up data of all patients were evaluated. Results: The median basal ACTH/cortisol ratio of the patients with CD was significantly higher than AICS and controls. A cutoff ACTH/cortisol ratio >2.5 was found to be diagnostic for CD with 82% specificity and 63% sensitivity. Among CD group, patients with recurrent disease had higher preoperative ACTH levels and ACTH/cortisol ratio than patients with sustained remission. Furthermore, these patients had more invasive, atypical, and larger tumors. Conclusion: An ACTH/cortisol ratio >2.5 would be beneficial to diagnose CD together with other diagnostic tests. It is a simple test with no additional cost. Higher ratios might be related with larger, invasive, and atypical adenoma and also might be helpful to predict recurrence.

A Woman with Weight Gain and Fatigue

2017 ◽  
Author(s):  
Alexandra Lovett ◽  
Whitney W. Woodmansee
Keyword(s):  
Pituitary Adenoma ◽  
Cushing’S Disease ◽  
Dexamethasone Suppression Test ◽  
Cushing's Disease ◽  
Distal Muscle ◽  
Suppression Test ◽  
Urine Cortisol ◽  
Acth Secreting ◽  
Dexamethasone Suppression ◽  
Transsphenoidal Resection

Cushing’s syndrome is a descriptive term of a syndrome secondary to elevated levels of steroids or cortisol, while Cushing’s disease is hypercortisolemia that results from an adrenocorticotropic (ACTH)-secreting pituitary adenoma. Patients will present with cushingoid features on physical examination and can be myopathic with proximal rather than distal muscle weakness. Diagnosis can be obtained by multiple avenues including but not limited to checking 24 hour urine cortisol, a dexamethasone suppression test, checking ACTH levels, a CRH (corticotrophin-releasing hormone) stimulation test, and inferior petrosal sinus sampling (IPSS). Once Cushing’s disease is confirmed, treatment is via transsphenoidal resection of the pituitary adenoma.

scholarly journals Cushing’s Disease: Not Always Black and White

2021 ◽  
Vol 5 (Supplement_1) ◽  
pp. A580-A581
Author(s):  
Kajal Shah ◽  
Alan Chang ◽  
Natalie Jarahzadeh
Keyword(s):  
Weight Gain ◽  
Pituitary Adenoma ◽  
Cushing’S Disease ◽  
Pituitary Surgery ◽  
Cushing's Disease ◽  
Nuclear Staining ◽  
Corticotroph Adenoma ◽  
Post Surgery ◽  
Free Cortisol ◽  
Initial Symptoms

Abstract Cushing’s Disease is a well known entity but the difficulty of diagnosis is often underappreciated. Given the wide spectrum of clinical presentation and pitfalls with each diagnostic evaluation, diagnosis of Cushing’s Disease (CD) is often difficult in clinical practice, even with pathological analysis. A 37 year old female with a history of Cushing’s Disease presented for her third transphenoidal resection to our institution. She was diagnosed with a pituitary microadenoma 16 years ago based on MRI, as she had persistent headaches and vision abnormalities. The patient also reported unexplained weight gain, “buffalo hump”, and moon facies at the time. She was diagnosed with CD due to an abnormal 1 mg overnight dexamethasone suppression test (DST) with AM cortisol of 3.03 ug/dl (normal <1.8 microgram/dl). She was on an oral contraceptive pill (OCP) at the time. The patient underwent her first pituitary transsphenoidal resection and was symptom free for about 10 years, after which she had a recurrence of her initial symptoms. She had another abnormal DST while on an OCP and pituitary MRI revealed growth of the pituitary adenoma. Patient underwent a second pituitary surgery with benign postoperative course with a recurrence about 5 years later. The workup prior to her third surgery revealed an abnormal DST while on OC pills with the 8am cortisol being 3.36 ug/dl (<1.8ug/dl), urinary free cortisol 35.4 mcg/24 hour (4-50 mcg/24 hour). 8am ACTH done on a separate day was 48 pg/ml (6-50 pg/ml) with a cortisol of 14.5 ug/dl. Midnight salivary cortisol was not performed. Interval history was still positive for weight gain and headaches, hence she was referred for her third pituitary surgery. Post surgery, the patient was on a short taper of hydrocortisone and 8am cortisol was 32 ug/dl the next day. After a discussion with the pathologist, it was determined that the pathology was suggestive of a corticotroph adenoma with moderate ACTH staining and patchy nuclear staining for TPIT, although the pathologist stated that it was difficult to be certain due to the small tissue size. From the current literature, this patient had an incomplete and equivocal biochemical work up while on OCPs but still had tissue diagnosis supporting CD. Whether this was a recurrence of Cushing’s disease, silent corticotroph adenoma, or non-functioning pituitary adenoma is unclear. This case illustrates the multiple challenges in the diagnosis of Cushing’s disease that may be encountered. 1.Braun LT et al. Recurrence after pituitary surgery in adult Cushing’s disease: a systematic review on diagnosis and treatment. Endocrine. 2020;70(2):218-231.

Sign in / Sign up

Export Citation Format

Share Document