scholarly journals The use of cinacalcet in pregnancy to treat a complex case of parathyroid carcinoma

2014 ◽  
Vol 2014 ◽  
Author(s):  
K Nadarasa ◽  
M Bailey ◽  
H Chahal ◽  
O Raja ◽  
R Bhat ◽  
...  
Keyword(s):  
Parathyroid Carcinoma ◽  
Thyroid Tissue ◽  
Complex Case ◽  
High Dose ◽  
List Type ◽  
Radiological Findings ◽  
Chronic Knee Pain ◽  
Healthy Infants ◽  
Learning Points ◽  
In Pregnancy

Summary We present the case of a patient with metastatic parathyroid carcinoma whose hypercalcaemia was medically managed through two pregnancies. The diagnosis was made when the patient presented with chronic knee pain and radiological findings consistent with a brown tumour, at the age of 30. Her corrected calcium and parathyroid hormone (PTH) levels were significantly elevated. Following localisation studies, a right parathyroidectomy was performed with histology revealing parathyroid carcinoma, adherent to thyroid tissue. Aged 33, following biochemical recurrence of disease, the patient underwent a second operation. A subsequent CT and FDG–PET revealed bibasal pulmonary metastases. Aged 35, the patient was referred to our unit for treatment of persistent hypercalcaemia. The focus of treatment at this time was debulking metastatic disease using radiofrequency ablation. Despite advice to the contrary, the patient conceived twice while taking cinacalcet. Even though there are limited available data regarding the use of cinacalcet in pregnancy, both pregnancies continued to term with the delivery of healthy infants, using intensive medical management for persistent hypercalcaemia. Learning points Parathyroid carcinoma is a rare cause of primary hyperparathyroidism. Hypercalcaemia during pregnancy can result in significant complications for both the mother and the foetus. The use of high-dose cinacalcet in pregnancy has been shown, in this case, to aid in the management of resistant hypercalcaemia without teratogenicity.

scholarly journals A case of pituitary apoplexy in pregnancy

2014 ◽  
Vol 2014 ◽  
Author(s):  
Aimee R Hayes ◽  
Anthony J O'Sullivan ◽  
Mark A Davies
Keyword(s):  
Pituitary Apoplexy ◽  
Rare Event ◽  
Visual Disturbance ◽  
List Type ◽  
The Subject ◽  
Magnetic Resonance Imaging Mri ◽  
Evidence Of Impact ◽  
Learning Points ◽  
Visual Abnormalities ◽  
In Pregnancy

Summary Pituitary apoplexy is a rare event in pregnancy. A 41-year-old woman with a known pituitary microadenoma presented with visual disturbance and headache during the second trimester of pregnancy. Magnetic resonance imaging (MRI) demonstrated pituitary apoplexy with chiasmal compression. After treatment with corticosteroid therapy, she underwent transsphenoidal excision of the pituitary adenoma. Visual abnormalities were completely restored and pituitary function preserved. There was no evidence of impact on the foetus. The literature on the subject is reviewed with emphasis on the management of the apoplectic patient with mild and stable neuro-ophthalmological signs. Learning points There are no clear guidelines on the management of pituitary apoplexy in pregnancy. A multidisciplinary approach can minimise morbidity and mortality. Pituitary apoplexy has an unpredictable clinical course and determining which clinical situations warrant early surgery needs to take into consideration the presence and severity of neurological signs and their stability. The management of conscious apoplectic patients with absent or mild and stable neuro-ophthalmological signs is controversial.

scholarly journals Diagnosis and treatment challenges of parathyroid carcinoma in a 27-year-old woman with multiple lung metastases

2017 ◽  
Vol 2017 ◽  
Author(s):  
Liudmila Rozhinskaya ◽  
Ekaterina Pigarova ◽  
Ekaterina Sabanova ◽  
Elizaveta Mamedova ◽  
Iya Voronkova ◽  
...  
Keyword(s):  
Parathyroid Carcinoma ◽  
Kinase Inhibitor ◽  
Lung Metastases ◽  
Treatment Options ◽  
Distant Metastases ◽  
List Type ◽  
Medical Centers ◽  
Resected Tissue ◽  
Promising Treatment ◽  
Learning Points

Summary Parathyroid carcinoma is an extremely rare disorder with little treatment options. It could be misdiagnosed in medical centers with little experience in management of such cases. Our clinical case shows that the initial misdiagnosis of a parathyroid carcinoma in a young woman has led to the development of multiple lung metastases, thus making its treatment hardly possible. Initiation of treatment with sorafenib – a multi-kinase inhibitor approved for treatment of different types of cancer but not parathyroid carcinoma – has led to a significant decrease in the size of lung metastases and has prevented the progression of hyperparathyroidism, which is usually severe in cases of parathyroid carcinoma. The detection of a germline CDC73 mutation in this patient has raised additional concerns about the necessity of periodic screening for early detection of renal, jaw and uterine lesions. Learning points: Diagnosis of parathyroid carcinoma may be challenging due to the absence of reliable diagnostic criteria. Thus, thorough histological examination is needed using immunohistochemical staining of resected tissue in suspicious cases. CDC73 genetic testing should be considered in patients with parathyroid carcinoma. Sorafenib may be a promising treatment of patients with parathyroid carcinoma with distant metastases.

scholarly journals Primary hyperparathyroidism and Zollinger Ellison syndrome during pregnancy: a case report

2021 ◽  
Vol 2021 ◽  
Author(s):  
Dalal Ali ◽  
Patrick Divilly ◽  
Ruth Prichard ◽  
Dermot O’Toole ◽  
Donal O’Shea ◽  
...  
Keyword(s):  
Primary Hyperparathyroidism ◽  
Unplanned Pregnancy ◽  
Case Series ◽  
Medical Attention ◽  
High Dose ◽  
List Type ◽  
Childbearing Age ◽  
Zollinger Ellison Syndrome ◽  
In Pregnancy

Summary Multiple endocrine neoplasia type 1 (MEN1) is a rare inherited endocrine disorder with a high rate of penetrance. The incidence of MEN1 is 1/30,000 in the general population; however, it is quite rare for a patient to present for medical attention with MEN1 for the first time in pregnancy. Primary hyperparathyroidism (PHPT) is one of the most common features of MEN1. The incidence of PHPT occurring in pregnancy is 1%. Despite advances in the medical, surgical and obstetric care over the years, management of this condition during pregnancy may be challenging. It can be difficult to identify pregnant women with PHPT requiring intervention and to monitor safely. Hypercalcemia can result in significant maternal and fetal adverse outcomes including: miscarriage, intrauterine growth restriction, preterm delivery, neonatal hypocalcaemia, pre-eclampsia and maternal nephrolithiasis. Herein, we present a case study of a lady with a strong family history of MEN1, who was biochemically proven to have PHPT and evidence of Zollinger Ellison Syndrome (ZE) on endoscopy. This patient delayed her assisted pregnancy plans for in vitro fertilization (IVF) until completion of the MEN1 workup; nevertheless, she spontaneously achieved an unplanned pregnancy. As a result, she required intervention with parathyroidectomy in the second trimester of her pregnancy as her calcium level continued to rise. This case study highlights the workup, follow up and management of MEN1 presenting with PHPT and ZE in pregnancy. Learning points Women of childbearing age who are suspected to have a diagnosis of primary hyperparathyroidism ideally should have genetic testing and avoid pregnancy until definitive plans are in place. Zollinger Ellison syndrome in pregnancy means off-label use of high dose of proton pump inhibitors (PPI). Use of PPI in pregnancy is considered to be safe based on retrospective studies. Omeprazole, however, is FDA class C drug because of lack of large prospective studies or large case series during pregnancy. Calcium supplements in the form of calcium carbonate must be converted to calcium chloride by gastric acid in order to be absorbed, however, patients rendered achlorhydric as a result of PPI use will have impaired absorption of calcium. Therefore, use of calcium citrate might be considered a better option in this case.

scholarly journals Too little or too much corticosteroid? Coexisting adrenal insufficiency and Cushing's syndrome from chronic, intermittent use of intranasal betamethasone

2013 ◽  
Vol 2013 ◽  
Author(s):  
Adrienne Dow ◽  
Run Yu ◽  
John Carmichael
Keyword(s):  
Adrenal Insufficiency ◽  
Cushing’S Syndrome ◽  
Cushing's Syndrome ◽  
High Dose ◽  
List Type ◽  
Secondary Adrenal Insufficiency ◽  
Severe Fatigue ◽  
Learning Points ◽  
Endocrinology Clinic ◽  
Iatrogenic Cushing’S Syndrome

Summary To report the puzzling, rare occurrence of coexisting adrenal insufficiency and Cushing's syndrome from chronic, intermittent use of intranasal betamethasone spray. A 62-year-old male was referred to our endocrinology clinic for management of adrenal insufficiency. This previously healthy individual began to experience chronic sinus symptoms in 2007, was treated with multiple ensuing sinus surgeries, and received oral glucocorticoid for 6 months. In the following 5 years, he suffered severe fatigue and was diagnosed with secondary adrenal insufficiency. He could not be weaned from corticosteroid and developed clear cushingoid features. In our clinic, careful inquiry on medications revealed chronic, intermittent use of high-dose intranasal betamethasone since 2008, which was not apparent to his other treating physicians. His cushingoid features significantly improved after holding intranasal betamethasone. Learning points Chronic, intermittent intranasal betamethasone can cause secondary adrenal insufficiency and iatrogenic Cushing's syndrome when used in excess. Topical corticosteroid use should be considered in the differential diagnosis of adrenal insufficiency or Cushing's syndrome.

THIOURACIL TREATMENT IN PREGNANCY AND LACTATION IN THE DOG

1962 ◽  
Vol 40 (3) ◽  
pp. 430-440 ◽  
Author(s):  
K. E. Arosenius ◽  
H. Derblom ◽  
G. Nylander
Keyword(s):  
Thyroid Gland ◽  
List Type ◽  
Histological Changes ◽  
Morphological Effect ◽  
Iodine Metabolism ◽  
Pregnancy And Lactation ◽  
In Pregnancy

ABSTRACT The offspring of bitches treated with thiouracil during pregnancy and lactation were studied with regard to certain aspects of iodine metabolism and also to the morphological effect on the thyroid gland, as compared with controls of the same age and weight. At the age of six months, when the dogs were virtually fully grown, the thyroid gland still exhibited the effects of the thiouracil treatment of the mothers as manifested by an increased uptake of 131I, enlargement, and histological changes of a hyperplastic type. The significance of these deviations from normal iodine metabolism and morphology is discussed with particular reference to the dangers of thiouracil medication during pregnancy.

A METHOD FOR EVALUATING AN EXPERIMENTAL ATHYREOSIS

1972 ◽  
Vol 70 (2) ◽  
pp. 289-294 ◽  
Author(s):  
K. Schimmelpfennig ◽  
A. Kaul ◽  
U. Haberland
Keyword(s):  
Weight Loss ◽  
Thyroid Tissue ◽  
Cervical Region ◽  
List Type ◽  
Experimental Animals

ABSTRACT A micro-131I-test is presented which allows an easy separation of completely surgically thyroidectomised experimental animals from animals with aberrant or residual thyroid tissue. The application of the method is easy and not time-consuming: the total time spent on one animal is approximately 3 min. The method is based on the principle that the 131I storage is measured over the cervical region. The application of this method gives the following advantages: When performing studies with proven athyroid rats it is not necessary subsequently to demonstrate athyreosis (histologically, BMR, or PBI). Time-consuming experiments with animals which are not definitely athyroid can be avoided. The additional fractionated radio-iodine resection after surgical thyroidectomy, used by many authors to destroy residual thyroid tissue, becomes superfluous. Such a procedure takes 4 to 8 weeks. The animals may be used after a 5-day-period. This excludes secondary changes like weight loss and disturbed development which have to be taken into consideration when using a radio-iodine resection.

scholarly journals 312 Female sex independently predicts adjuvant immunotherapeutic benefit from CTLA4 immune checkpoint inhibition

2020 ◽  
Vol 8 (Suppl 3) ◽  
pp. A339-A339
Author(s):  
Ahmad Tarhini ◽  
Ni Kang ◽  
Sandra Lee ◽  
F Stephen Hodi ◽  
Gary Cohen ◽  
...  
Keyword(s):  
Overall Survival ◽  
Treatment Efficacy ◽  
Significant Interaction ◽  
Cox Regression ◽  
Performance Status ◽  
Phase Iii ◽  
High Dose ◽  
List Type ◽  
Female Sex ◽  
Interaction Term

BackgroundSex differences in tumor immunity and response to immunotherapy were shown in murine models and descriptive analyses from recent clinical trials. Female sex hormones have been implicated in melanoma development and response to systemic therapy. We hypothesized a gender difference in response to adjuvant immunotherapy with ipilimumab (3 or 10 mg/kg; ipi3 or ipi10) versus high dose IFNα (HDI) as tested in the E1609 trial.MethodsE1609 demonstrated significant overall survival (OS) benefit with ipi3 versus HDI.1 We investigated treatment efficacy between ipi and HDI in the subgroups by sex (female, male), age (< 55 or ≥55), stage at study entry (IIIB, IIIC, M1a/1b), ECOG performance status (PS 0, 1), ulceration (yes, no), primary tumor (known, unknown), number of lymph nodes involved (0, 1, 2–3, 4+). Forest plots were created to compare OS and RFS with ipi3 vs. HDI and ipi10 vs. HDI using the concurrently randomized ITT populations. For the estimated HRs, 95% confidence intervals were created for all subgroups.ResultsThe subgroups of female, stage IIIC, PS=1, ulcerated, in-transit without lymph node involvement demonstrated significant improvement in overall survival (OS) and/or relapse free survival (RFS) with ipi3 versus HDI as summarized in table 1. Female sex was significant for both OS and RFS and was further explored. In investigating RFS with ipi3 versus HDI, a multivariate Cox regression model including sex, treatment and interaction term of sex*treatment, indicated a significant interaction between sex and treatment (P = 0.026). Including sex, PS (0 vs. 1), age (<55 vs. 55+), ulceration (yes vs. no), stage (IIIB, IIIC, M1a, M1b), treatment and interaction term of sex*treatment, indicated a significant interaction between sex and treatment (P = 0.024). While similar trends were seen, no significant interactions between sex and treatment effect were found in the OS multivariate analysis or in the comparison of ipi10 versus HDI. When exploring age, in the univariate analyses in the ipi3 versus HDI comparison older women appeared to drive most of the difference (age ≥55: OS, P=0.02 and RFS, P=0.08; differences non-significant for women <55). Table 1.Abstract 312 Table 1Treatment efficacy between ipi3 and HDI by subgroupConclusionsFemale sex was independently associated with RFS adjuvant immunotherapeutic benefit from ipi3, supporting a potentially important role for female related factors in the immune response against melanoma, and these warrant further investigation.Trial RegistrationNCT01274338Ethics ApprovalThe study protocol was approved by the institutional review board (IRB) of each participating institution and conducted in accordance with Good Clinical Practice guidelines as defined by the International Conference on Harmonisation. This study was monitored by the ECOG-ACRIN DataSafety Monitoring Committee and the NCI.ConsentAll patients provided IRB-approved written informed consent.ReferenceTarhini AA, Lee SJ, Hodi FS, Rao UNM, Cohen GI, Hamid O, Hutchins LF, Sosman JA, Kluger HM, Eroglu Z, Koon HB, Lawrence DP, Kendra KL, Minor DR, Lee CB, Albertini MR, Flaherty LE, Petrella TM, Streicher H, Sondak VK, Kirkwood JM. Phase III Study of Adjuvant Ipilimumab (3 or 10 mg/kg) Versus High-Dose Interferon Alfa-2b for Resected High-Risk Melanoma: North American Intergroup E1609. J Clin Oncol. 2020 Feb 20;38(6):567–575. PMID: 31880964.

Directional Persistence in the Rewarded Alternation Model of Obsessive-compulsive Disorder is Responsive to both Dopaminergic and Serotonergic Manipulations

2009 ◽  
Vol 24 (S1) ◽  
pp. 1-1
Author(s):  
D. Kontis ◽  
V. Boulougouris ◽  
S. Papadopoulos ◽  
V.-M. Papakosta ◽  
S. Kalogerakou ◽  
...  
Keyword(s):  
Obsessive Compulsive Disorder ◽  
Low Dose ◽  
Receptor Activation ◽  
Common Mechanism ◽  
High Dose ◽  
List Type ◽  
Type Number ◽  
Obsessive Compulsive ◽  
Compulsive Disorder ◽  
Serotonin Agonist

Rationale:In the rewarded alternation model of obsessive compulsive disorder (OCD), the serotonin agonist m-chlorophenylpiperazine (mCPP) increases persistent behaviour, while chronic pretreatment with selective serotonin reuptake inhibitor (SSRI-fluoxetine) but not benzodiazepine or desipramine abolishes mCPP effects. However, we noted that acute SSRI administration also causes transient persistence increases, counteracted by mCPP pretreatment.Objectives:This studya.further explores the apparent cross-tolerance between fluoxetine and mCPP andb.extends the model by investigating its sensitivity to dopaminergic manipulations (D2,3 agonism - quinpirole).Methods:In both experiments, baseline and drug testing was carried out under daily T-maze alternation training.Exp.1:Matched group (n=8) pairs of rats received one of the following 20-day pretreatments (daily intraperitoneal administration):1.saline,2.low-dose fluoxetine (2.5mg/kg),3.low-dose mCPP (0.5mg/kg) or4.combined fluoxetine+mCPP.One group per pretreatment then received a 4-day challenge with high-dose fluoxetine (10mg/kg), the other with high-dose mCPP (2.5mg/kg).Exp.2:One group (n=12) of rats received 20-day treatment with saline, another with quinpirole (0.5 mg/kg).Results:Exp.1:Saline and low-dose mCPP- or fluoxetine-pretreated animals showed significant persistence increases under both challenges, while combined low-dose fluoxetine+mCPP pretreatment afforded full protection from either challenge.Exp.2:Quinpirole significantly increased directional persistence after 13 administration days.Conclusions:These results establish the sensitivity of the rewarded alternation OCD model to D2,3receptor activation, thereby extending its profile of pharmacological isomorphism with OCD. Furthermore, they suggest a common mechanism of action of an SSRI and a serotonin agonist in the control of directional persistence.

scholarly journals Managing asthma in pregnancy

Breathe ◽  
2015 ◽  
Vol 11 (4) ◽  
pp. 258-267 ◽  
Author(s):  
Vanessa E. Murphy
Keyword(s):  
Pregnant Women ◽  
Inhaled Corticosteroids ◽  
Action Plan ◽  
Asthma Management ◽  
Perinatal Outcomes ◽  
List Type ◽  
Night Time ◽  
Exacerbation Rate ◽  
Common Chronic Disease ◽  
In Pregnancy

Asthma is a common comorbidity during pregnancy and its prevalence is increasing in the community. Exacerbations are a major clinical problem during pregnancy with up to 45% of women needing to seek medical help, resulting in poor outcomes for mothers and their babies, including low birth weight and preterm delivery. The goals of effective asthma management in pregnancy are to maintain the best possible asthma control and prevent exacerbations. This is achieved by aiming to prevent day- and night-time symptoms, and maintain lung function and normal activity. In addition, maintaining fetal oxygenation is an important consideration in pregnancy. Guidelines recommend providing asthma advice and review prior to conception, and managing asthma actively during pregnancy, with regular 4-weekly review, provision of a written action plan, use of preventer medications as indicated for other adults with asthma, and management of comorbid conditions such as rhinitis.Improvements have been made in recent years in emergency department management of asthma in pregnancy, and multidisciplinary approaches are being proposed to optimise both asthma outcomes and perinatal outcomes. One strategy that has demonstrated success in reducing exacerbations in pregnancy is treatment adjustment using a marker of eosinophilic lung inflammation, the exhaled nitric oxide fraction (FeNO). The use of an algorithm that adjusted inhaled corticosteroids (ICS) according toFeNOand added long-acting β-agonists when symptoms remained uncontrolled resulted in fewer exacerbations, more women on ICS but at lower mean doses, and improved infant respiratory health at 12 months of age. Further evidence is needed to determine whether this strategy can also improve perinatal outcomes and be successfully translated into clinical practice.Key pointsAsthma is the most common chronic disease to affect pregnant women.Exacerbations occur in up to 45% of pregnant women with asthma.Asthma should be managed during pregnancy as for other adults.Treatment adjustment using a marker of airway inflammation reduces the exacerbation rate in pregnancy.Educational aimsTo identify the goals of and steps associated with effective asthma management in pregnancy.To understand the maternal and perinatal risks associated with asthma during pregnancy.To describe a management strategy that has been shown to reduce exacerbations in pregnant women with asthma.

scholarly journals Anecdotal Response to Amphotericin in a Patient with Probable Susac Syndrome: Implications in the Pathogenesis

2019 ◽  
Vol 32 (11) ◽  
pp. 727
Author(s):  
Raquel Gil-Gouveia ◽  
Natália Marto ◽  
Pedro Vilela ◽  
Ana Catarino
Keyword(s):  
Hearing Loss ◽  
Fungal Infection ◽  
Infectious Agent ◽  
Brain Biopsy ◽  
High Dose ◽  
Radiological Findings ◽  
Susac Syndrome ◽  
Infection Treatment ◽  
Immune Mediated

Susac syndrome is a rare, probably immune-mediated endotheliopathy presenting with encephalopathy, sensorineural hearing loss and retinal arterial occlusions. A 33-year-old female with Susac syndrome was worsening despite high-dose steroids so a brain biopsy was performed which suggested a possible fungal infection. Treatment with amphotericin B resulted in prompt reversal of symptoms and radiological findings, and no further symptoms occurred during 8 years of follow-up. A diagnosis of fungal infection was not confirmed. The etiology of Susac syndrome is unknown and this anecdotal observation suggests that an infectious agent susceptible to amphotericin might have caused or triggered Susac syndrome in this patient.

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