63 results found
Boharoon H, Tomlinson J, Limback-Stanic C, et al., 2021, A case series of patients with isolated IgG4-related hypophysitis treated with rituximab, Journal of the Endocrine Society, Vol: 4, Pages: 1-9, ISSN: 2472-1972
ContextThe acute presentation of Immunoglobulin G4 (IgG4)-related hypophysitis can be indistinguishable from other forms of acute hypophysitis and histology remains the diagnostic gold standard. The high recurrence rate necessitates long term immunosuppressive therapy. Rituximab (RTX) has been shown to be effective in systemic IgG4-related disease (IgG4-RD), but experience with isolated pituitary involvement remains limited.Case descriptionWe report three female patients with MRI findings suggestive of hypophysitis. All patients underwent transsphenoidal biopsy and fulfilled diagnostic criteria for IgG4-related hypophysitis. Treatment with GCs (GC) resulted in good therapeutic response in patients 1 and 2, but the disease recurred on tapering doses of GCs. GC treatment led to emotional lability in Patient 3 necessitating dose reduction. All three patients received RTX and Patients 2 and 3 received further courses when symptoms returned and B-cells repopulated. Patient 3 did not receive RTX until 12 months from onset of symptoms. Patient 1 was not able to have further RTX treatments due to an allergic reaction when receiving the second dose. RTX treatment resulted in sustained remission and full recovery of anterior pituitary function in Patients 1 and 2 with complete resolution of pituitary enlargement. By contrast, Patient 3 only showed symptomatic response following RTX treatment, but pituitary enlargement and hypofunction persisted.ConclusionRTX treatment for IgG4-related hypophysitis resulted in sustained remission in two patients treated early in the disease process, but only achieved partial response in a patient with chronic disease suggesting that early therapeutic intervention may be crucial to avoid irreversible changes.
Hussein Z, Glynn N, Martin N, et al., 2021, Temporal trends in craniopharyngioma management and long-term endocrine outcomes: a multicentre cross-sectional study., Clinical Endocrinology, Vol: 94, Pages: 242-249, ISSN: 0300-0664
BACKGROUND: The optimal management of craniopharyngiomas remains controversial. OBJECTIVES: To examine temporal trends in the management of craniopharyngioma with a focus on endocrine outcomes. METHODS: This was a cross-sectional, multicentre study. Patients treated between 1951 and 2015 were identified and divided into four quartiles. Demographics, presentation, treatment and outcomes were collected. RESULTS: In total, 142 patients with childhood onset craniopharyngioma (48/142; 34%) and adult onset disease (94/142; 66%) were included. The median follow-up was 15 years (IQR 5 - 23 years). Across quartiles, there was a significant trend towards using transsphenoidal surgery (p <0.0001). The overall use of radiotherapy was not different among the four quartiles (p = 0.33). At the latest clinical review, the incidence of GH, ACTH, gonadotrophins deficiencies as well as anterior panhypopituitarism fell significantly across the duration of the study. Anterior panhypopituitarism was not affected by treatment modality (surgery vs surgery and radiotherapy) (p = 0.23). There was no difference in the incidence of high BMI (≥ 25 kg/m2 ) among the four quartiles (p = 0.14). BMI was higher in patients who treated with surgery and radiotherapy than those treated with surgery only (p = 0.006). Tumour regrowth occurred in 51 patients (51/142; 36%) with no difference in regrowth among quartiles over the time course of the study (p = 0.15). CONCLUSION: We demonstrate a significant reduction in panhypopituitarism in craniopharyngioma patients over time, most likely because of a trend towards more transsphenoidal surgery. However, long term endocrine sequelae remain common and lifelong follow-up is required.
Martin N, Sovetkina A, Nadir R, et al., 2020, Development of autoimmune thyroid disease in multiple sclerosis patients post-alemtuzumab improves treatment response, The Journal of Clinical Endocrinology & Metabolism, Vol: 105, Pages: e3392-e3399, ISSN: 0021-972X
ContextAlemtuzumab is an anti-CD52 monoclonal antibody used in the treatment of relapsing-remitting multiple sclerosis (MS). Between 20-40% of alemtuzumab-treated MS patients develop autoimmune thyroid disease (AITD) as a side effect.ObjectiveTo determine whether MS disease progression following alemtuzumab treatment differs in patients that develop AITD compared to those who do not.Design, setting and patientsA retrospective analysis of 126 patients with relapsing-remitting MS receiving alemtuzumab from 2012 to 2017 at a tertiary referral centre.Main outcome measuresThyroid status, new relapses, Expanded Disability Status Score (EDSS) change and disability progression following alemtuzumab were evaluated.ResultsTwenty-six percent (33 out of 126, 25 female, 8 male) of alemtuzumab-treated patients developed AITD, 55% of which was Graves’ disease. EDSS score following alemtuzumab was reduced in patients who developed AITD compared to those who did not (median [IQR]; AITD: -0.25 [-1 - 0.5] vs non-AITD: 0 [1 - 0]. P=0.007]. Multivariate regression analysis confirmed that the development of AITD was independently associated with EDSS score improvement (p=0.011). Moreover, AITD patients had higher relapse-free survival following alemtuzumab (p=0.023). There was no difference in the number of new focal T2-lesions and contrast-enhancing MRI lesions developed following alemtuzumab between the two groups.ConclusionGraves’ disease was the most common form of AITD developed by MS patients following alemtuzumab. This study suggests that MS patients who develop AITD may have an improved response to alemtuzumab, as measured by reduced disability and lower relapse rate.
Fountas A, Lim ES, Drake WM, et al., 2020, Outcomes of patients with Nelson's Syndrome after primary treatment: a multicenter study from 13 UK pituitary centers, Journal of Clinical Endocrinology and Metabolism, Vol: 105, Pages: 1-11, ISSN: 0021-972X
ContextLong-term outcomes of patients with Nelson’s syndrome (NS) have been poorly explored, especially in the modern era.ObjectiveTo elucidate tumor control rates, effectiveness of various treatments, and markers of prognostic relevance in patients with NS.Patients, design, and settingRetrospective cohort study of 68 patients from 13 UK pituitary centers with median imaging follow-up of 13 years (range 1–45) since NS diagnosis.ResultsManagement of Cushing’s disease (CD) prior to NS diagnosis included surgery+adrenalectomy (n = 30; eight patients had 2 and one had 3 pituitary operations), surgery+radiotherapy+adrenalectomy (n = 17; two received >1 courses of irradiation, two had ≥2 pituitary surgeries), radiotherapy+adrenalectomy (n = 2), and adrenalectomy (n = 19). Primary management of NS mainly included surgery, radiotherapy, surgery+radiotherapy, and observation; 10-year tumor progression-free survival was 62% (surgery 80%, radiotherapy 52%, surgery+radiotherapy 81%, observation 51%). Sex, age at CD or NS diagnosis, size of adenoma (micro-/macroadenoma) at CD diagnosis, presence of pituitary tumor on imaging prior adrenalectomy, and mode of NS primary management were not predictors of tumor progression. Mode of management of CD before NS diagnosis was a significant factor predicting progression, with the group treated by surgery+radiotherapy+adrenalectomy for their CD showing the highest risk (hazard ratio 4.6; 95% confidence interval, 1.6–13.5). During follow-up, 3% of patients had malignant transformation with spinal metastases and 4% died of aggressively enlarging tumor.ConclusionsAt 10 years follow-up, 38% of the patients diagnosed with NS showed progression of their corticotroph tumor. Complexity of treatments for the CD prior to NS diagnosis, possibly reflecting corticotroph adenoma aggressiveness, predicts long-term tumor prognosis.
Amin A, Neophytou C, Thein S, et al., 2018, L-Arginine increases post-prandial circulating GLP-1 and PYY levels in humans, Obesity, Vol: 26, Pages: 1721-1726, ISSN: 1930-7381
ObjectiveThe satiating effect of protein compared with other nutrients has been well described and is thought to be mediated, in part, by gut hormone release. Previously, it has been shown that oral L‐arginine acts as a GLP‐1 secretagogue both in vitro and in vivo in rodents. Here, the effect of L‐arginine on gut hormone release in humans was investigated.MethodsThe hypothesis was tested in two separate studies. The first study assessed the tolerability of oral L‐arginine in healthy human subjects. The second study assessed the effect of oral L‐arginine on gut hormone release following an ad libitum meal. Subjects were given L‐arginine, glycine (control amino acid), or vehicle control in a randomized double‐blind fashion.ResultsAt a dose of 17.1 mmol, L‐arginine was well tolerated and stimulated the release of plasma GLP‐1 (P < 0.05) and PYY (P < 0.001) following an ad libitum meal. Food diaries showed a trend toward lower energy intake and particularly fat intake following L‐arginine treatment.ConclusionsL‐arginine can significantly elevate GLP‐1 and PYY in healthy human volunteers in combination with a meal. Further work is required to investigate whether L‐arginine may have utility in the suppression of appetite and food intake.
Abbara A, Clarke S, Eng PC, et al., 2018, Clinical and biochemical characteristics of patients presenting with pituitary apoplexy, Endocrine Connections, Vol: 7, Pages: 1058-1066, ISSN: 2049-3614
PurposeTo review the clinical and biochemical characteristics and clinical outcome of patients presenting with pituitary apoplexy to a tertiary centre.MethodsWe retrospectively reviewed the clinical features, predisposing factors, biochemistry and clinical outcome of patients presenting with pituitary apoplexy to Imperial College Healthcare NHS Trust between 1991 and 2015.ResultsWe identified 64 patients with pituitary apoplexy (more complete clinical records were available in 52 patients). The median age at presentation was 46.7 years (IQR 31.5–57.0 years). Pituitary apoplexy was the first presentation of pituitary disease in 38/52 of patients and predisposing factors were identified in 28/52. Pituitary apoplexy predominantly occurred in patients with non-functioning pituitary adenomas (47/52). Headache was most commonly described as sudden onset, severe, lateralising to the frontal or temporal regions. Symptoms of meningeal irritation were reported in 7/18 and visual abnormalities in 22/35. A pre-treatment serum cortisol <100 nmol/L was recorded in 12/31 of patients. All patients with visual disturbance had some resolution of their visual symptoms whether managed surgically (14/14) or conservatively (5/5), although pituitary endocrine function did not fully recover in any patient.ConclusionsIn conclusion, these data describe the clinical features of pituitary apoplexy to aid the clinician in diagnosing this rare emergency presentation of pituitary disease. Prospective multicentre studies of the presentation of pituitary apoplexy are required to further characterise presentation and outcomes.
Abbara A, Clarke S, Nesbitt A, et al., 2018, Interpretation of serum gonadotropin levels in hyperprolactinemia, Neuroendocrinology, Vol: 107, Pages: 105-113, ISSN: 0028-3835
Background/Aims: Hyperprolactinemia is a common cause of amenorrhea due to hypogonadotropic hypogonadism. Prolactin is hypothesized to impede the reproductive axis through an inhibitory action at the hypothalamus. However, limited data exists to aid the interpretation of serum gonadotropins in the context of hyperprolactinemia. Methods: Serum gonadotropin values were reviewed in 243 patients with elevated serum monomeric prolactin due to discrete etiologies at a tertiary reproductive endocrine centre between 2012 and 2015. The cause of hyperprolactinemia was categorized by an experienced endocrinologist / pituitary multidisciplinary team, unless superseded by histology. The most frequently encountered diagnoses were Microprolactinoma (n=88), Macroprolactinoma (n=46), Non-Functioning Pituitary Adenoma (NFPA) (n=72), Drug-Induced Hyperprolactinemia (DIH) (n=22) and Polycystic Ovarian Syndrome (PCOS) (n=15). Results: In patients with prolactinoma and modestly raised serum prolactin levels (<4000 mU/L), increasingly FSH-predominant gonadotropin values were observed with rising prolactin level, consistent with a progressive reduction in hypothalamic GnRH pulsatility. Patients with prolactinoma and higher prolactin values (>4000 mU/L) were more likely to have a reduction in serum levels of both FSH and LH, consistent with direct pituitary gonadotrope dysfunction. Patients with macroadenoma and extremes of serum gonadotropin values (either serum FSH or LH >8 IU/L) were more likely to have NFPA than prolactinoma. Patients with polycystic ovarian syndrome (PCOS) and hyperprolactinemia had LH-predominant secretion in keeping with increased GnRH pulsatility despite a raised prolactin level. Conclusion: The pattern of gonadotropin secretion in patients may reflect the etiology of hyperprolactinemia.
Wernig F, Hatfield E, Smith C, et al., 2018, Early Treatment With Rituximab Can Improve Clinical Outcomes In Isolated IgG4- related Hypophysitis, The Endocrine Society Annual Meeting, Publisher: Oxford University Press (OUP), ISSN: 0163-769X
Drake WM, Stiles CE, Bevan JS, et al., 2016, A follow-up study of the prevalence of valvular heart abnormalities in hyperprolactinemic patients treated with cabergoline, Journal of Clinical Endocrinology & Metabolism, Vol: 101, Pages: 4189-4194, ISSN: 1945-7197
CONTEXT Uncertainty exists whether the long-term use of ergot-derived dopamine agonist (DA) drugs for the treatment of hyperprolactinemia may be associated with clinically significant valvular heart disease; and whether current regulatory authority guidelines for echocardiographic screening are clinically appropriate. OBJECTIVE: To provide follow-up echocardiographic data on a previously described cohort of patients treated with DA for lactotrope pituitary tumors; and to explore possible associations between structural and functional valve abnormalities with the cumulative dose of drug used. DESIGN: Follow-up echocardiographic data were collected from a proportion of our previously reported cohort of patients; all had received continuous DA therapy for at least 2 years in the intervening period. Studies were performed according to British Society of Echocardiography minimum standards for adult transthoracic echocardiography. Generalised estimating equations with backward selection were used to determine odds ratios of valvular heart abnormalities according to tertiles of cumulative cabergoline dose, using the lowest tertile as the reference group. SETTING: Thirteen centers of secondary/tertiary endocrine care across the United Kingdom. RESULTS: There were 192 patients (81 males; median age, 51 years; interquartile range [IQR], 42-62). Median (IQR) cumulative cabergoline doses at the first and second echocardiograms were 97mg (20-377) and 232mg (91-551) respectively. Median (IQR) duration of uninterrupted cabergoline therapy between echocardiograms was 34 months (24-42). No associations were observed between cumulative doses of dopamine agonist used and the age-corrected prevalence of any valvular abnormality. CONCLUSION: This large UK follow-up study does not support a clinically significant association between the use of DA for the treatment of hyperprolactinemia and cardiac valvulopathy.
Koulouri O, Kandasamy N, Hoole AC, et al., 2016, Successful treatment of residual pituitary adenoma in persistent acromegaly following localisation by 11C-methionine PET co-registered with MRI, European Journal of Endocrinology, Vol: 175, Pages: 485-498, ISSN: 1479-683X
OBJECTIVE: To determine if functional imaging using (11)C-methionine positron emission tomography computed tomography, co-registered with 3D gradient echo MRI (Met-PET/MRI), can identify sites of residual active tumour in treated acromegaly, and discriminate these from post-treatment change, to allow further targeted treatment. DESIGN/METHODS: Twenty-six patients with persistent acromegaly following previous treatment, in whom MRI appearances were considered indeterminate, were referred to our centre for further evaluation over a 4.5-year period. Met-PET/MRI was performed in each case and findings were used to inform decision-making regarding adjunctive therapy. Four patients with clinical and biochemical remission post-transsphenoidal surgery (TSS), but in whom residual tumour was suspected on postoperative MRI, were also studied. RESULTS: Met-PET/MRI demonstrated tracer uptake only within the normal gland in the four patients who had achieved complete remission following primary surgery. In contrast, in 26 patients with active acromegaly, Met-PET/MRI localised sites of abnormal tracer uptake in all but one case. Based on these findings, fourteen subjects underwent endoscopic TSS, leading to a marked improvement in (n=7), or complete resolution of (n=7), residual acromegaly. One patient received stereotactic radiosurgery and two patients with cavernous sinus invasion were treated with image-guided fractionated radiotherapy, with good disease control. Three subjects await further intervention. Five patients chose to receive adjunctive medical therapy. Only one patient developed additional pituitary deficits following Met-PET/MRI-guided TSS. CONCLUSIONS: In patients with persistent acromegaly following primary therapy, Met-PET/MRI can help identify the site(s) of residual pituitary adenoma when MRI appearances are inconclusive, and direct further targeted intervention (surgery or radiotherapy).
Sam AH, Shah S, Saleh K, et al., 2015, Clinical outcomes in patients with nonfunctioning pituitary adenomas managed conservatively, CLINICAL ENDOCRINOLOGY, Vol: 83, Pages: 861-865, ISSN: 0300-0664
McGavigan AK, O'Hara HC, Amin A, et al., 2015, L-cysteine suppresses ghrelin and reduces appetite in rodents and humans, INTERNATIONAL JOURNAL OF OBESITY, Vol: 39, Pages: 447-455, ISSN: 0307-0565
Lee M, Lupp A, Mendoza N, et al., 2015, SSTR3 is a putative target for the medical treatment of gonadotroph adenomas of the pituitary, ENDOCRINE-RELATED CANCER, Vol: 22, Pages: 111-119, ISSN: 1351-0088
Ramachandran R, Bech P, Murphy KG, et al., 2015, Comparison of the Utility of Cocaine- and Amphetamine-Regulated Transcript (CART), Chromogranin A, and Chromogranin B in Neuroendocrine Tumor Diagnosis and Assessment of Disease Progression, Journal of Clinical Endocrinology & Metabolism, Vol: 100, Pages: 1520-1528, ISSN: 1945-7197
Bech PR, Martin NM, Ramachandran R, et al., 2014, The biochemical utility of chromogranin A, chromogranin B and cocaine- and amphetamine-regulated transcript for neuroendocrine neoplasia, ANNALS OF CLINICAL BIOCHEMISTRY, Vol: 51, Pages: 8-21, ISSN: 0004-5632
Pinato DJ, Tan TM, Toloue S, et al., 2013, An expression signature of the angiogenic response in gastrointestinal neuroendocrine tumours: correlation with tumour phenotype and survival outcomes., British Journal of Cancer, Vol: 110, ISSN: 1532-1827
Parker JA, McCullough KA, Field BCT, et al., 2013, Glucagon and GLP-1 inhibit food intake and increase c-fos expression in similar appetite regulating centres in the brainstem and amygdala, INTERNATIONAL JOURNAL OF OBESITY, Vol: 37, Pages: 1391-1398, ISSN: 0307-0565
Sam A, Busbridge M, Amin A, et al., 2013, Hepcidin levels in diabetes mellitus and polycystic ovary syndrome, Diabetic Medicine, Vol: 30, Pages: 1495-1499, ISSN: 0742-3071
Cegla J, Jones B, Seyani L, et al., 2013, Comparison of the overnight metyrapone and glucagon stimulation tests in the assessment of secondary hypoadrenalism, CLINICAL ENDOCRINOLOGY, Vol: 78, Pages: 738-742, ISSN: 0300-0664
Pinato DJ, Ramachandran R, Toussi STK, et al., 2013, Immunohistochemical markers of the hypoxic response can identify malignancy in phaeochromocytomas and paragangliomas and optimize the detection of tumours with VHL germline mutations, BRITISH JOURNAL OF CANCER, Vol: 108, Pages: 429-437, ISSN: 0007-0920
Martin NM, Patel M, Nijher GMK, et al., 2012, Adjuvant lithium improves the efficacy of radioactive iodine treatment in Graves' and toxic nodular disease, CLINICAL ENDOCRINOLOGY, Vol: 77, Pages: 621-627, ISSN: 0300-0664
Maurice JB, Troke R, Win Z, et al., 2012, A comparison of the performance of Ga-68-DOTATATE PET/CT and I-123-MIBG SPECT in the diagnosis and follow-up of phaeochromocytoma and paraganglioma, EUROPEAN JOURNAL OF NUCLEAR MEDICINE AND MOLECULAR IMAGING, Vol: 39, Pages: 1266-1270, ISSN: 1619-7070
Higham CE, Atkinson AB, Aylwin S, et al., 2012, Effective Combination Treatment with Cabergoline and Low-Dose Pegvisomant in Active Acromegaly: A Prospective Clinical Trial, OBSTETRICAL & GYNECOLOGICAL SURVEY, Vol: 67, Pages: 475-476, ISSN: 0029-7828
Ramachandran R, Bech P, Murphy KG, et al., 2012, Improved diagnostic accuracy for neuroendocrine neoplasms using two chromogranin A assays., Clin Endocrinol (Oxf), Vol: 76, Pages: 831-836
Chromogranin A (Cg A) is the best available diagnostic marker for neuroendocrine neoplasms (NENs). However, clinical interpretation of Cg A results may be limited by considerable heterogeneity between commonly available Cg A assays. Variation in diagnostic accuracy of these assays largely reflects differences in antibody specificities. We compared the diagnostic utility of four Cg A assays [Imperial Supra-regional Assay Service radioimmunoassay (SAS) and three commercial assays, Cisbio, DAKO and Eurodiagnostica].
Bech PR, Ramachandran R, Dhillo WS, et al., 2012, Quantifying the Effects of Renal Impairment on Plasma Concentrations of the Neuroendocrine Neoplasia Biomarkers Chromogranin A, Chromogranin B, and Cocaine- and Amphetamine-Regulated Transcript, CLINICAL CHEMISTRY, Vol: 58, Pages: 941-943, ISSN: 0009-9147
Salem V, Hopkins TG, El-Gayar H, et al., 2012, Adrenal venous sampling as a diagnostic procedure for primary hyperaldosteronism: experience from a tertiary referral centre, HORMONES-INTERNATIONAL JOURNAL OF ENDOCRINOLOGY AND METABOLISM, Vol: 11, Pages: 151-159, ISSN: 1109-3099
Higham CE, Atkinson AB, Aylwin S, et al., 2012, Effective Combination Treatment with Cabergoline and Low-Dose Pegvisomant in Active Acromegaly: A Prospective Clinical Trial, JOURNAL OF CLINICAL ENDOCRINOLOGY & METABOLISM, Vol: 97, Pages: 1187-1193, ISSN: 0021-972X
Ghourab S, Beale KE, Semjonous NM, et al., 2011, Intracerebroventricular administration of vasoactive intestinal peptide inhibits food intake, REGULATORY PEPTIDES, Vol: 172, Pages: 8-15, ISSN: 0167-0115
Ford HE, Peters V, Martin NM, et al., 2011, Effects of oral ingestion of sucralose on gut hormone response and appetite in healthy normal-weight subjects, EUROPEAN JOURNAL OF CLINICAL NUTRITION, Vol: 65, Pages: 508-513, ISSN: 0954-3007
Ramachandran R, Bech P, Murphy KG, et al., 2011, PLASMA GASTRIN: SOMATOSTATIN RATIO IS DECREASED IN GASTRINOMAS, Annual Meeting on British-Society-of-Gasenterology, Publisher: B M J PUBLISHING GROUP, Pages: A117-A117, ISSN: 0017-5749
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