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1.
J Endocrinol Invest ; 45(3): 573-582, 2022 Mar.
Article in English | MEDLINE | ID: mdl-34637114

ABSTRACT

PURPOSE: Conventional therapy (calcium and activated vitamin D) does not restore calcium homeostasis in patients with chronic hypoparathyroidism (HypoPT) and is associated with renal complications and reduced quality of life (QoL). The aim of this study was to evaluate in a case-control, cross-sectional study, the rate of renal complications and QoL in two sex- and age-matched cohort of patients with differentiated thyroid cancer with (n = 89) and without (n = 89) chronic post-operative HypoPT (PoHypoPT) and their relationship with the biochemical control of the disease. METHODS: Serum and urinary parameters, renal ultrasound and QoL were assessed by SF-36 and WHO-5 questionnaires. RESULTS: Forty-three (48.3%) PoHypoPT patients reported symptoms of hypocalcemia. Twenty-six (29.2%) patients were at target for all 6 parameters, 46 (51.6%) for 5. The most frequently unmet targets were gender-specific 24-h urinary calcium (44.9%) and serum calcium (37.1%). Serum phosphate, magnesium and 25(OH)D were in the normal range in > 90% of patients. Renal calcifications were found in 26 (29.2%) patients, with no correlation with 24-h urinary calcium. eGFR did not differ between patients and controls. Conversely, patients had a significant higher rate of renal calcifications and a lower SF-36, but not WHO-5, scores. SF-36 scores did not differ between PoHypoPT patients who were, or not, hypocalcemic. CONCLUSIONS: Our study shows that the rate of renal calcifications was higher in patients with PoHypoPT than in those without. This finding, together with the reduced QoL and the presence of hypocalcemic symptoms in about half patients, underscores that the treatment of chronic HypoPT with conventional therapy is suboptimal.


Subject(s)
Calcium , Hypoparathyroidism , Nephrolithiasis , Postoperative Complications , Quality of Life , Thyroid Neoplasms/surgery , Thyroidectomy , Vitamin D/therapeutic use , Calcium/blood , Calcium/metabolism , Calcium/therapeutic use , Calcium/urine , Calcium-Regulating Hormones and Agents/metabolism , Calcium-Regulating Hormones and Agents/therapeutic use , Female , Humans , Hypocalcemia/blood , Hypocalcemia/etiology , Hypocalcemia/therapy , Hypocalcemia/urine , Hypoparathyroidism/blood , Hypoparathyroidism/complications , Hypoparathyroidism/etiology , Hypoparathyroidism/psychology , Male , Middle Aged , Nephrolithiasis/blood , Nephrolithiasis/etiology , Nephrolithiasis/psychology , Nephrolithiasis/therapy , Outcome Assessment, Health Care , Postoperative Complications/blood , Postoperative Complications/diagnosis , Postoperative Complications/psychology , Postoperative Complications/therapy , Surveys and Questionnaires , Thyroid Neoplasms/pathology , Thyroidectomy/adverse effects , Thyroidectomy/methods
2.
Exp Clin Endocrinol Diabetes ; 129(12): 918-925, 2021 Dec.
Article in English | MEDLINE | ID: mdl-33694151

ABSTRACT

OBJECTIVES: This study aimed to evaluate the current situation of hypoparathyroid patients and to investigate the relationship between treatment adherence and quality of life. STUDY DESIGN: Prospective, multicentre study. METHODS: Adult patients presenting with the diagnosis of hypoparathyroidism to 20 different endocrinology clinics were included. They were receiving conventional therapies for hypoparathyroidism, using calcium, active vitamin D, and magnesium. We collected data on demographic features, disease- and treatment-related information, and results of routine laboratory tests, treatment adherence, and presence of complications. Beck Depression Inventory, Beck Anxiety Inventory, and Short Form-36 quality of life assessments were administered. RESULTS: Among the 300 patients studied, 60.7% were adherent to their treatment, and 34.1% had complications. Anxiety and depression scores were significantly higher in non-adherent versus treatment-adherent patients (p<0.001 and p=0.001, respectively). Most of the domains of quality-of-life scores were also significantly lower in non-adherent patients. Both anxiety and depression scores showed significant, negative correlations with serum calcium and magnesium concentrations (r=-0.336, p<0.001 and r=-0.258, p<0.001, respectively). CONCLUSIONS: Nearly 40% of the patients were non-adherent to conventional treatment for hypoparathyroidism, and such patients had higher anxiety and depression scores and poorer quality of life scores. Conventional treatment might not be sufficient to meet the needs of patients with hypoparathyroidism. In addition to seeking new therapeutic options, factors influencing quality of life should also be investigated and strategies to improve treatment adherence should be developed.


Subject(s)
Anxiety/psychology , Depression/psychology , Hypoparathyroidism/drug therapy , Hypoparathyroidism/psychology , Medication Adherence/statistics & numerical data , Quality of Life/psychology , Adult , Anxiety/epidemiology , Depression/epidemiology , Female , Humans , Hypoparathyroidism/epidemiology , Male , Middle Aged , Outcome Assessment, Health Care , Prospective Studies , Turkey/epidemiology
3.
Surgery ; 169(1): 70-76, 2021 01.
Article in English | MEDLINE | ID: mdl-32654859

ABSTRACT

BACKGROUND: We sought to better understand the experience of patients with transient hypoparathyroidism using patient interviews and quality of life surveys. METHODS: This is a prospective analysis of 62 patients after total thyroidectomy at a high-volume institution. Semistructured patient interviews and quality of life surveys were conducted preoperatively and postoperatively at 2 weeks, 6 weeks, 6 months, and 1 year and compared based on postoperative parathyroid hormone levels. RESULTS: Postoperative parathyroid hormone levels were <10 pg/mL in 32% of patients (n = 20), 10 to 20 pg/mL in 19% (n = 12), and >20 pg/mL in 48% (n = 30). Hypocalcemic symptoms at 2 weeks were reported in 28 of 55 patients (51%), but patients felt "well prepared" and reported it "wasn't a big deal." If symptoms persisted at 6 weeks, they became more bothersome. At 6 months and 1 year, patients reported calcium supplementation prevented most symptoms and did not interfere with daily activities. Quality of life as measured by the European Organization for Research and Treatment of Cancer and the 12-Item Short Form Survey demonstrated a slight improvement at 1 year postoperatively regardless of parathyroid hormone level. CONCLUSION: Early postoperative transient hypoparathyroidism is common but when appropriately managed did not have a substantial negative impact on the overall quality of life.


Subject(s)
Hypocalcemia/psychology , Hypoparathyroidism/psychology , Postoperative Complications/psychology , Quality of Life , Thyroid Neoplasms/surgery , Thyroidectomy/adverse effects , Adult , Aged , Calcium/blood , Female , Follow-Up Studies , Humans , Hypocalcemia/diagnosis , Hypocalcemia/epidemiology , Hypocalcemia/etiology , Hypoparathyroidism/diagnosis , Hypoparathyroidism/epidemiology , Hypoparathyroidism/etiology , Male , Middle Aged , Parathyroid Glands/injuries , Parathyroid Glands/metabolism , Parathyroid Hormone/blood , Parathyroid Hormone/metabolism , Postoperative Complications/diagnosis , Postoperative Complications/epidemiology , Postoperative Complications/etiology , Preoperative Period , Prospective Studies , Qualitative Research , Risk Factors , Thyroid Neoplasms/blood , Young Adult
4.
J Endocrinol Invest ; 44(5): 905-919, 2021 May.
Article in English | MEDLINE | ID: mdl-32926396

ABSTRACT

CONTEXT AND PURPOSE: Hypocalcemia and low parathyroid hormone levels have been commonly suggested as factors able to induce central nervous system disturbances. However, evidences on the occurrence of cognitive impairment are limited or underestimated. The aim of this review is, therefore, to systematically summarize the available evidence concerning the occurrence of cognitive impairment among subjects suffering from idiopathic or secondary hypoparathyroidism. METHODS: A systematic selection of the available literature was performed by searching the online databases PubMed, Scopus and Web of Knowledge. RESULTS: The present systematic review included sixteen case report articles and one cross-sectional controlled study. Case reports were the most representative literature sources and involved ten women and seven men. The presence of cognitive impairment was mostly discussed in association with idiopathic hypoparathyroidism (HPT); five articles described the occurrence of cognitive impairment following postsurgical HPT. The case-controlled study reported a significant presence of peculiar cognitive deficits (e.g. reduced inhibitory control, impairment in visuo-spatial functioning among, and psychomotor retardation) among HPT subjects compared to healthy controls, with serum total calcium and its product with phosphorus as independent predictors of neuropsychological dysfunctions. CONCLUSION: Even though mostly based on single case reports, the presence of neuropsychological dysfunctions in the context of HPT appears to be a consistent core finding.


Subject(s)
Cognitive Dysfunction , Hypoparathyroidism , Cognitive Dysfunction/blood , Cognitive Dysfunction/diagnosis , Cognitive Dysfunction/physiopathology , Humans , Hypocalcemia/diagnosis , Hypocalcemia/etiology , Hypoparathyroidism/blood , Hypoparathyroidism/etiology , Hypoparathyroidism/psychology , Neuropsychological Tests , Parathyroid Hormone/analysis
5.
J Clin Endocrinol Metab ; 105(12)2020 12 01.
Article in English | MEDLINE | ID: mdl-32918085

ABSTRACT

PURPOSE: Surgical complications such as hypoparathyroidism (HPT) or vocal cord palsy are seldom assessed when the quality of life (QOL) in thyroid cancer patients is investigated. The aim of this study was to measure the QOL difference in thyroid cancer survivors with and without HPT. METHODS: Participants for this analysis were enrolled in 13 countries from a study that pilot-tested a thyroid cancer-specific QOL instrument. They were included if they had been diagnosed with thyroid cancer at least 9 months previously. QOL was measured using the European Organisation for Research and Treatment of Cancer Quality of Life Questionnaire Core (EORTC QLQ-C30) and some items on HPT symptoms (eg, tingling in fingers or toes). HPT status and other clinical data were extracted from the patients' medical charts. Comparisons of QOL domains between patients with and without HPT were performed using Mann-Whitney U test. The occurrence of HPT-related symptoms was compared using chi-square tests. Multiple ordinal regression analysis was performed to evaluate factors that might affect QOL. RESULTS: Eighty-nine patients participated in this study, 17 of whom were considered to have HPT. Patients in the HPT group reported significantly reduced QOL in 9 of the 15 scales of the EORTC QLQ-C30 compared to patients without HPT. Regression analysis showed that HPT was independently negatively associated with various scales of the QLQ-C30. Both groups showed a high prevalence of typical HPT symptoms. CONCLUSION: Thyroid cancer patients with HPT report significantly impaired QOL compared to thyroid cancer survivors without HPT. The assessment of HPT should be considered when measuring QOL in thyroid cancer patients.


Subject(s)
Cancer Survivors , Hypoparathyroidism , Postoperative Complications , Quality of Life , Thyroid Neoplasms/surgery , Adult , Aged , Cancer Survivors/psychology , Cancer Survivors/statistics & numerical data , Cross-Sectional Studies , Female , Health Status , Humans , Hypoparathyroidism/epidemiology , Hypoparathyroidism/etiology , Hypoparathyroidism/psychology , Male , Middle Aged , Pilot Projects , Postoperative Complications/epidemiology , Postoperative Complications/psychology , Surveys and Questionnaires , Thyroid Neoplasms/epidemiology
6.
Patient ; 13(2): 151-162, 2020 04.
Article in English | MEDLINE | ID: mdl-31552607

ABSTRACT

BACKGROUND AND OBJECTIVE: Hypoparathyroidism is a rare endocrine disorder characterized by absent or inappropriately low levels of circulating parathyroid hormone. Patients with hypoparathyroidism receiving standard-of-care therapy report debilitating physical and cognitive symptoms, which may indicate a reduced health-related quality of life. The purpose of this study was to develop a new disease-specific measure of the signs and symptoms of hypoparathyroidism, the Hypoparathyroidism Patient Experience Scale-Symptom (HPES-Symptom), and provide evidence for the content validity of items in the measure based on rigorous qualitative research methodologies for patient-reported outcome development. METHODS: Semi-structured, individual concept elicitation interviews were conducted with five clinical experts and 42 adults with hypoparathyroidism in the USA to identify the signs and symptoms of relevance and importance to those with the condition. Transcripts were coded and analyzed using an adapted grounded theory approach. Following item generation, cognitive debriefing interviews of the draft measure were conducted in an independent sample of 16 adults with hypoparathyroidism. RESULTS: One hundred percent of the concept elicitation patient sample reported experiencing physical symptoms that were attributed to hypoparathyroidism, including tingling/numbness/paresthesia (n = 37, 88%), muscle cramping (n = 36, 86%), and physical fatigue (n = 35, 83%). The majority of patients (n = 36, 86%) further reported experiencing cognitive dysfunction, including impaired memory (n = 24, 57%), impaired ability to have a conversation (n = 21, 50%), and lack of concentration/focus (n = 18, 43%). Seventeen major signs and symptoms were identified during item generation and included in the preliminary measure. After the cognitive debriefing, the 17-item HPES-Symptom was generated. CONCLUSIONS: The findings provided evidence of content validity for the HPES-Symptom in US adults with hypoparathyroidism. Additional research is needed to validate the measure in patients with hypoparathyroidism to assess its psychometric properties.


Subject(s)
Hypoparathyroidism/physiopathology , Hypoparathyroidism/psychology , Patient Reported Outcome Measures , Surveys and Questionnaires/standards , Adult , Aged , Female , Humans , Male , Middle Aged , Psychometrics , Quality of Life/psychology
7.
Clin Endocrinol (Oxf) ; 92(2): 159-168, 2020 02.
Article in English | MEDLINE | ID: mdl-31721256

ABSTRACT

OBJECTIVE: To address knowledge gaps regarding burdens associated with not adequately controlled chronic hypoparathyroidism. DESIGN: Global patient and caregiver survey. STUDY POPULATIONS: Patients with chronic hypoparathyroidism not adequately controlled on conventional therapy and their caregivers. MEASUREMENTS: Health-related quality of life (HRQoL) and health status were evaluated using the 36-item Short Form version 2 (SF-36 v2.0) and Five-Level EuroQoL 5 Dimensions (EQ-5D-5L) instruments, respectively. Hypoparathyroidism-associated symptoms were assessed by a disease-specific Hypoparathyroidism Symptom Diary and caregiver burden via the Modified Caregiver Strain Index (MCSI). RESULTS: Data were obtained from 398 patients and 207 caregivers. Patients' self-rated hypoparathyroidism-related symptom severity was none (3%), mild (32%), moderate (53%) or severe (12%). Per the Hypoparathyroidism Symptom Diary, patients reported moderate, severe or very severe symptoms of physical fatigue (73%), muscle cramps (55%), heaviness in limbs (55%) and tingling (51%) over a 7-day recall period. Impacts (rated 'somewhat' or 'very much') were reported by 84% of patients for ability to exercise, 78% for sleep, 75% for ability to work and 63% for family relationships. Inverse relationships were observed between patient self-rated overall symptom severity and HRQoL and health status assessment scores-the greater the symptom severity, the lower the SF-36 and EQ-5D-5L scores. Caregiver burden increased with patient self-rated symptom severity: none, 1.7 MCSI; mild, 5.4 MCSI; moderate, 9.5 MCSI; and severe, 12.5 MCSI. CONCLUSION: Patients with not adequately controlled hypoparathyroidism reported substantial symptoms and impacts. Greater patient symptom severity was associated with decreased patient HRQoL and health status assessments and increased caregiver burden.


Subject(s)
Caregivers , Cost of Illness , Hypoparathyroidism/epidemiology , Hypoparathyroidism/therapy , Quality of Life , Adult , Aged , Caregiver Burden/epidemiology , Caregiver Burden/etiology , Caregivers/psychology , Caregivers/statistics & numerical data , Chronic Disease , Female , Health Status , Humans , Hypoparathyroidism/blood , Hypoparathyroidism/psychology , Male , Middle Aged , Parathyroid Hormone/blood , Stress, Psychological/epidemiology , Stress, Psychological/etiology , Surveys and Questionnaires
8.
J Clin Endocrinol Metab ; 104(7): 2748-2756, 2019 07 01.
Article in English | MEDLINE | ID: mdl-30776291

ABSTRACT

CONTEXT: Calcium and vitamin D treatment does not improve reduced quality of life (QOL) in hypoparathyroidism. Recombinant human (rh) PTH(1-84) therapy improves QOL metrics for up to 5 years. Data on QOL beyond this time point are not available. OBJECTIVES: To evaluate the effects of 8 years of rhPTH(1-84) therapy on QOL and factors associated with long-term benefit. DESIGN: Prospective, open-label trial. SETTING: Referral center. PATIENTS: Twenty patients with hypoparathyoidism. MAIN OUTCOME MEASURES: RAND 36-Item Short Form Health Survey (SF-36). RESULTS: rhPTH therapy led to substantial improvement in five of the eight SF-36 domains [vitality, social functioning (SF), mental health (MH), bodily pain (BP) and general health] and three of these domains (SF, MH, BP) were no longer lower than the reference population. The improvement in the mental component summary (MCS) score was sustained through 8 years, while the physical component summary (PCS) score improved through 6 years. A lower baseline QOL score was associated with greater improvement. A threshold value <238 (MCS) and <245 (PCS) predicted long-term improvement in 90% and 100% of the cohort, respectively. In patients whose calcium supplementation was reduced, MCS and PCS scores improved more than those whose supplementation did not decline to the same extent. Improvement in PCS was greater in patients whose calcitriol dosage was reduced and duration of disease was shorter. CONCLUSIONS: rhPTH(1-84) improves long-term well-being in hypoparathyroidism. The improvements are most prominent in those with impaired SF-36 at baseline and those whose requirements for conventional therapy decreased substantially.


Subject(s)
Calcium-Regulating Hormones and Agents/therapeutic use , Hypoparathyroidism/drug therapy , Parathyroid Hormone/therapeutic use , Quality of Life , Adult , Aged , Calcitriol/therapeutic use , Calcium/blood , Calcium/therapeutic use , Cholecalciferol/therapeutic use , Ergocalciferols/therapeutic use , Female , Humans , Hypoparathyroidism/blood , Hypoparathyroidism/physiopathology , Hypoparathyroidism/psychology , Longitudinal Studies , Male , Middle Aged , Prospective Studies , Recombinant Proteins , Treatment Outcome , Vitamin D/analogs & derivatives , Vitamin D/blood
9.
Endocrinol Metab Clin North Am ; 47(4): 855-864, 2018 12.
Article in English | MEDLINE | ID: mdl-30390818

ABSTRACT

Patients with hypoparathyroidism have a multitude of physical, emotional, and cognitive complaints consistent with reduced quality of life (QOL). Impaired QOL in patients treated with conventional therapy with calcium and active vitamin D has been documented in epidemiologic (registry) studies, case-controlled studies, and surveys, and at baseline in clinical trials of parathyroid hormone (PTH). Treatment with PTH has been shown to improve QOL in some but not all studies.


Subject(s)
Hormone Replacement Therapy/psychology , Hypoparathyroidism/psychology , Quality of Life , Humans , Hypoparathyroidism/drug therapy , Parathyroid Hormone/therapeutic use , Vitamin D/therapeutic use
11.
Arch Endocrinol Metab ; 62(3): 319-324, 2018 Jun.
Article in English | MEDLINE | ID: mdl-29791658

ABSTRACT

OBJECTIVE: Hypoparathyroidism is characterized by parathyroid hormone deficiency and hypocalcemia. It has been demonstrated that these patients may also present psychiatric symptoms and decrease of quality of life. The aims of this study were to evaluate the presence of psychopathological symptoms in a cohort of patients with hypoparathyroidism and compare to a control group. SUBJECTS AND METHODS: Patients were submitted to a cross-sectional Symptom Checklist-90-R (SCL-90-R) questionnaire that evaluates psychopathological symptoms by means of the Global Severity Index (GSI), Positive Symptoms Total (PST) and Positive Symptom Distress Index (PSDI). A score based in the positive symptoms was calculated (T-score). The test group was composed of patients with hypoparathyroidism, and control by thyroidectomized patients without hypoparathyroidism. A correlation between the presence of psychological symptoms and clinical features was analyzed. RESULTS: The study included 57 patients with a mean age of 51.1 ± 16.4 years; 20 as a control and 37, test group. There were no differences between groups regarding gender, mean age and age at diagnose. Hypoparathyroidism patients presented higher GSI index than the control group (p = 0.038). Mean T-score of the test group was as elevated as 58.2 ± 5.3 (reference range < 55). No correlation of the number of psychological symptoms to clinical and laboratorial parameters was observed. CONCLUSION: Patients with hypoparathyroidism attending our outpatient clinics presented an increase in the number of self-report of psychological symptoms when compared with a control group. However, no correlation with hypocalcemia and clinical parameters was observed. Future studies are necessary to evaluated if the absence of PTH play a role on it.


Subject(s)
Hypoparathyroidism/psychology , Quality of Life , Case-Control Studies , Cohort Studies , Cross-Sectional Studies , Female , Humans , Hypoparathyroidism/surgery , Male , Middle Aged , Self Report , Surveys and Questionnaires
12.
Arch. endocrinol. metab. (Online) ; 62(3): 319-324, May-June 2018. tab
Article in English | LILACS | ID: biblio-950073

ABSTRACT

ABSTRACT Objective: Hypoparathyroidism is characterized by parathyroid hormone deficiency and hypocalcemia. It has been demonstrated that these patients may also present psychiatric symptoms and decrease of quality of life. The aims of this study were to evaluate the presence of psychopathological symptoms in a cohort of patients with hypoparathyroidism and compare to a control group. Subjects and methods: Patients were submitted to a cross-sectional Symptom Checklist-90-R (SCL-90-R) questionnaire that evaluates psychopathological symptoms by means of the Global Severity Index (GSI), Positive Symptoms Total (PST) and Positive Symptom Distress Index (PSDI). A score based in the positive symptoms was calculated (T-score). The test group was composed of patients with hypoparathyroidism, and control by thyroidectomized patients without hypoparathyroidism. A correlation between the presence of psychological symptoms and clinical features was analyzed. Results: The study included 57 patients with a mean age of 51.1 ± 16.4 years; 20 as a control and 37, test group. There were no differences between groups regarding gender, mean age and age at diagnose. Hypoparathyroidism patients presented higher GSI index than the control group (p = 0.038). Mean T-score of the test group was as elevated as 58.2 ± 5.3 (reference range < 55). No correlation of the number of psychological symptoms to clinical and laboratorial parameters was observed. Conclusion: Patients with hypoparathyroidism attending our outpatient clinics presented an increase in the number of self-report of psychological symptoms when compared with a control group. However, no correlation with hypocalcemia and clinical parameters was observed. Future studies are necessary to evaluated if the absence of PTH play a role on it.


Subject(s)
Humans , Male , Female , Middle Aged , Quality of Life , Hypoparathyroidism/psychology , Case-Control Studies , Cross-Sectional Studies , Surveys and Questionnaires , Cohort Studies , Self Report , Hypoparathyroidism/surgery
13.
Endocrine ; 58(1): 14-20, 2017 Oct.
Article in English | MEDLINE | ID: mdl-28822059

ABSTRACT

PURPOSE: Hypoparathyroidism is a rare endocrine disorder for which replacement therapy of the missing parathyroid hormone is not the standard therapeutic option. Current standard treatment consists of calcium and vitamin D supplementation. The intake of calcium and vitamin D supplementation can lead to complications and therefore might negatively influence patients' quality of life. METHODS: A systematic literature review was performed to assess the current knowledge on the influence of hypoparathyroidism on patients' quality of life. The literature search was conducted in PubMed and Web of Science; all relevant literature published by August 24, 2016, was included. RESULTS: In total 372 records were found. After title and abstract screening, 14 studies remained for a full-text screening. The full-text screening resulted in five studies which were included into the systematic review. Comparing the results with a norm-based reference population, three studies reported lower SF-36 scores for hypoparathyroidism patients. Two studies showed a reduced quality of life in hypoparathyroidism patients when their results were compared to control populations. CONCLUSION: Most hypoparathyroidism patients receiving standard treatment show stable calcium and vitamin D levels. However, hypoparathyroidism patients still report reduced quality of life and experience physical, mental, and emotional symptoms. Therefore, it is assumed that the lack of parathyroid hormone directly influences the patients' quality of life. This review indicates that patients with hypoparathyroidism have a reduced quality of life in comparison to norm-based populations or matched controls. Further studies are required to quantify the effect of hypoparathyroidism on patients' quality of life using disease-specific questionnaires and controlling for the co-morbidities and etiologies of the patients.


Subject(s)
Hypoparathyroidism/psychology , Hypoparathyroidism/therapy , Quality of Life , Hormone Replacement Therapy , Humans , Hypoparathyroidism/drug therapy
14.
Intern Med ; 54(17): 2245-50, 2015.
Article in English | MEDLINE | ID: mdl-26328655

ABSTRACT

Idiopathic hypoparathyroidism (IHP) is accompanied by cognitive impairment. We report the case of a 70-year-old IHP patient with cognitive disturbance. Brain computed tomography showed bilateral calcification in basal ganglia, thalamus, and cerebellum. Neuropsychological assessment revealed low scores for intelligence, memory, and perseverative errors. Brain positron emission tomography showed a significant reduction in [(18)F]-Fludeoxyglucose (FDG) uptake in bilateral frontal, left temporal and parietal cortices, along with a marked reduction in [(11)C]-flumazenil binding in left frontal, temporal, parietal, and bilateral cerebellum. These findings suggest cognitive impairment in IHP may be ascribed to GABAergic dysfunction, thus leading to, or coexisting with, cerebral hypometabolism.


Subject(s)
Brain/metabolism , Brain/pathology , Cognition Disorders/etiology , Cognition , Hypoparathyroidism/metabolism , Hypoparathyroidism/pathology , Receptors, GABA-B/metabolism , gamma-Aminobutyric Acid/metabolism , Aged , Basal Ganglia/pathology , Brain/diagnostic imaging , Brain/physiopathology , Calcinosis/diagnostic imaging , Cerebellum/metabolism , Cerebellum/pathology , Cerebral Cortex/metabolism , Cognition Disorders/metabolism , Fluorodeoxyglucose F18/administration & dosage , Humans , Hypoparathyroidism/complications , Hypoparathyroidism/psychology , Male , Neuropsychological Tests , Positron-Emission Tomography/methods , Radiopharmaceuticals/administration & dosage , Thalamus/pathology , Tomography, X-Ray Computed
15.
Dtsch Med Wochenschr ; 140(16): 1195-7, 2015 Aug.
Article in German | MEDLINE | ID: mdl-26261924

ABSTRACT

Hypoparathyroidism and quality of life: Even in adequately substituted patients, hypoparathyroidism (HPT) is associated with impaired quality of life. It has been hypothesized that there is a correlation between reduced quality of life and lack of parathyroid hormone (PTH) in the central nervous system. Positive effects on quality of life have been reported when treating HPT with PTH. Hypoparathyroidism and comorbidities: Comorbidities associated with HPT include an increased risk of nephrolithiasis/nephrocalcinosis and neuro-psychiatric disease. Additionally, cardiovascular risk profile and an increased susceptibility for infections have been reported. Mortality rates seem not to be increased in HPT. Hypoparathyroidism and new therapeutic strategies: Recombinant PTH has not been approved for the treatment of HPT in Europe yet. However, in early 2015, the FDA approved PTH (1-84). Daily subcutaneous delivery of PTH (1-84) and PTH (1-34) (Teriparatide) has emerged as a promising therapeutic tool. However, its use should be restricted to patients insufficiently controlled with the standard treatment consisting of active vitamin d and calcium.


Subject(s)
Hypoparathyroidism/psychology , Quality of Life/psychology , Comorbidity , Drug Administration Schedule , Drug Approval , Germany , Humans , Hypoparathyroidism/complications , Hypoparathyroidism/drug therapy , Injections, Subcutaneous , Parathyroid Hormone-Related Protein/therapeutic use , Peptide Fragments/therapeutic use , Recombinant Proteins/therapeutic use , Risk Factors , Teriparatide/therapeutic use , United States , United States Food and Drug Administration
16.
J Clin Endocrinol Metab ; 99(10): 3694-9, 2014 Oct.
Article in English | MEDLINE | ID: mdl-24978675

ABSTRACT

CONTEXT: In hypoparathyroidism, quality of life (QOL) is compromised as compared to normal subjects. We previously reported our results showing an association with recombinant human PTH(1-84) therapy in hypoparathyroidism and improvement in QOL measures for 1 year. OBJECTIVE: We tested the hypothesis that PTH(1-84) therapy in hypoparathyroidism through 5 years would be associated with continued improvement in QOL measures. DESIGN: Sixty-nine hypoparathyroid subjects received open-label PTH(1-84). Before and during therapy, subjects completed the RAND 36-Item Short Form (SF-36) Health Survey, a measure of health-related QOL covering eight domains of physical and mental health. RESULTS: At baseline, subjects scored significantly lower than the normative reference range in all 8 domains (T-scores -1.4 to -0.9; P < .001 for all). With PTH therapy, intention-to-treat analysis showed significant improvement in the overall score at 2 months that persisted through 5 years (386 ± 19 to 482 ± 25; P < .0001). The mental component summary score improved at 2 months and was sustained through 5 years (199 ± 11 to 246 ± 14; P = .001), as did all four individual mental health domains and T-scores (vitality, social functioning, role emotional, mental health). The physical component summary score improved at 2 months and was sustained through 5 years (187 ± 10 to 237 ± 13; P < .0001), as did 3 physical health domains and T-scores (physical functioning, role physical, general health). CONCLUSIONS: PTH(1-84) therapy is not only associated with improvement in biochemical and skeletal indices, previously well-documented, but also in mental and physical health as determined by the SF-36 metric.


Subject(s)
Hypoparathyroidism/drug therapy , Hypoparathyroidism/psychology , Parathyroid Hormone/administration & dosage , Quality of Life/psychology , Adolescent , Adult , Aged , Emotions , Female , Health Surveys , Humans , Longitudinal Studies , Male , Mental Health , Middle Aged , Recombinant Proteins/administration & dosage , Treatment Outcome , Young Adult
17.
Osteoporos Int ; 25(6): 1717-26, 2014 Jun.
Article in English | MEDLINE | ID: mdl-24687385

ABSTRACT

UNLABELLED: The effects of treatment with 100 µg parathyroid hormone (PTH) (1-84) or an identical placebo on muscle function and quality of life (QoL) was studied in hypoparathyroid patients. At baseline, we found reduced QoL but no myopathy in the patients. Six months of treatment did not improve QoL, and muscle strength decreased slightly. INTRODUCTION: A reduced quality of life (QoL) and myopathy that may be due to the absence of PTH have been reported in patients with hypoparathyroidism (hypoPT). METHODS: Sixty-two patients with chronic hypoPT were randomized to 6 months of treatment with either PTH(1-84) 100 µg/d s.c. or placebo, given as add-on therapy to conventional treatment. Muscle function and postural stability were investigated using a dynamometer chair, a stadiometer platform, the repeated chair stands test, the timed up and go test, and electromyography. QoL was assessed using the 36-item Short Form Health Survey and the WHO-5 Well-Being Index. RESULTS: The mean age of the patients was 52 ± 11 years, and 85 % were females. At baseline, QoL was significantly reduced in comparison with norm-based scores. Compared with placebo, PTH did not improve QoL or muscle function. Rather, max force production decreased significantly by 30 % at elbow flexion in the PTH group compared with the placebo group. Moreover, there was a nonsignificant trend for muscle strength to decrease in the upper extremities and on knee extension in response to PTH. Treatment did not affect postural stability. Electromyography showed a slight decrease in the duration of motor unit potentials in the PTH group, indicating a tendency toward myopathy, which, however was not symptomatic. CONCLUSIONS: Overall, our data do not support an immediate beneficial effect of PTH replacement therapy on muscle function or QoL. A high frequency of hypercalcemia among our patients may have compromised the potential beneficial effects of reversing the state of PTH insufficiency.


Subject(s)
Hormone Replacement Therapy/methods , Hypoparathyroidism/drug therapy , Muscle Strength/drug effects , Parathyroid Hormone/therapeutic use , Quality of Life , Adult , Aged , Body Composition/drug effects , Double-Blind Method , Electromyography/methods , Female , Humans , Hypoparathyroidism/physiopathology , Hypoparathyroidism/psychology , Male , Middle Aged , Muscle, Skeletal/physiopathology , Parathyroid Hormone/pharmacology , Postural Balance/drug effects , Postural Balance/physiology , Psychometrics , Treatment Outcome
18.
Endocr Pract ; 20(7): 671-9, 2014 Jul.
Article in English | MEDLINE | ID: mdl-24449664

ABSTRACT

OBJECTIVE: Hypoparathyroidism is a rare disease caused by lack of parathyroid hormone (PTH) leading to hypocalcemia, hyperphosphatemia, and a variety of symptoms. This study aimed to quantify the clinical and social burden of illness from the perspective of affected patients. METHODS: A web-based instrument was developed with input from patients, clinical experts, and the Hypoparathyroidism Association. Qualifying participants were ≥18 years old, diagnosed with hypoparathyroidism for ≥6 months, and U.S. residents. Questions focused on demographics, diagnosis perceptions, current attitudes, medical management, current symptoms, acute episodes, comorbidities, personal life, and employment. RESULTS: A total of 374 adults (mean age, 49 ± 12 years; female, 85%) with hypoparathyroidism (mean duration, 13 ± 12 years; severe condition, 30.5%) completed the survey. Patients reported visiting a mean of 6 ± 8 physicians before and after their diagnosis. The majority strongly agreed with feeling unprepared to manage the condition at diagnosis (56%), that controlling their hypoparathyroidism is harder than expected (60%), and that they were concerned about long-term complications of their current medications (75%). More than 10 symptoms were experienced by 72% of patients in the preceding 12 months, despite current management regimens. Symptoms were experienced for a mean of 13 ± 9 hours/day. Hospital stays or emergency department visits were required by 79% of patients. 45% reported significant interference with their lives, 85% reported an inability to perform household activities, and 20% experienced a disease-associated change in employment status. CONCLUSION: Patients with hypoparathyroidism have a high burden of illness and experience a broad spectrum of symptoms, with a multidimensional impact on their lives.


Subject(s)
Cost of Illness , Hypoparathyroidism/economics , Adult , Aged , Employment , Female , Humans , Hypoparathyroidism/psychology , Male , Middle Aged , Quality of Life
19.
Endocr Pract ; 20(5): 427-46, 2014 May.
Article in English | MEDLINE | ID: mdl-24325999

ABSTRACT

OBJECTIVE: To test the hypothesis that surgeons and their patients underestimate the potential negative impact that permanent hypoparathyroidism has on quality of life (QOL). METHODS: We used a modified SF-36 assessment tool to compare the perceptions of patients with permanent hypoparathyroidism to the perceptions of control subjects who were given a standardized preoperative statement about the complications of hypoparathyroidism. We also elicited the perceptions of endocrine surgeons regarding the QOL impacts of hypoparathyroidism using a subset of questions from the modified SF-36. RESULTS: A total of 340 postsurgical patients with permanent hypoparathyroidism, 200 controls, and 102 surgeons participated in the study. Both surgeons and controls underestimated the negative impact of hypoparathyroidism on QOL when compared to patients living with permanent hypoparathyroidism. Forty-seven percent of hypoparathyroid patients believed that their health was "much worse" than before surgery, compared with 16% of surgeons (P<.001) and 7% of controls (P<.001). Postoperative hypoparathyroid patients also reported far more negative effects on QOL, from interference with social activities, paresthesias, muscle cramping, and medications than were anticipated by surgeons or controls (P<.05 for all comparisons). In each of the 8 dimensions of QOL, including physical functioning, role limitations due to physical health, role limitations due to emotional problems, energy/fatigue, emotional well-being, social functioning, pain, and general health, hypoparathyroid patients reported a significantly lower mean score compared to the control group (P<.001 for all comparisons). CONCLUSION: The impact of postoperative hypoparathyroidism on patient QOL is consistently and significantly underestimated by surgeons and subjects receiving surgical consultation.


Subject(s)
Hypoparathyroidism/psychology , Postoperative Complications/psychology , Thyroidectomy/adverse effects , Adolescent , Adult , Aged , Aged, 80 and over , Humans , Middle Aged , Prospective Studies , Quality of Life , Surgeons
20.
Encephale ; 40(3): 271-5, 2014 Jun.
Article in French | MEDLINE | ID: mdl-23816059

ABSTRACT

UNLABELLED: Fahr's syndrome is a rare disorder characterized by abnormal deposits of calcium in areas of the brain that control movement, including the basal ganglia and the cerebral cortex associated with many neurological and psychiatric abnormalities such as a rigid hypokinetic syndrome, mood disorders and cognitive impairment. Fahr's syndrome is secondary to some disorders, such as hypoparathyroidism. CASE REPORT: We report the case of a 56 year-old man, with a history of cataract, who was admitted to our psychiatric hospital for the first time in his life because of psychotic symptoms associated with irritability and aggressiveness. Since the age of 38 the patient had become nervous, 10 years later he developed tonic-clonic seizures. Two months ago, he began expressing delusions of persecution against his wife and sons and making fugues. According to his family during this period, he was agitated, aggressive, and suffered from insomnia and anorexia. The general and psychiatric examination showed an upright and bronzed patient with neglected hygiene. He was indifferent to his environment and expressed poor mimics and gestures. He was anxious, suspicious and not very talkative. He was conscious but his attention was slightly decreased. Moreover, he was not aware of his problems. The neurological examination showed extrapyramidal syndrome with postural tremor and cerebellar ataxia. A cranial computed tomography brain scan found bilateral, symmetric basal ganglia calcifications, in favour of Fahr's syndrome. Phosphocalcic investigations revealed low concentration of serum calcium at 1.01mmol/L (normal 2.15 to 2.57mmol/L) and hyperphosphoremia at 2.69mmol/L (normal 0.81 to 1.55mmol/L). He also had low concentrations of 25-OH vitamin as well as decreased urinary levels of phosphate and calcium. The blood level of parathyroid hormone was 0ng/L. The diagnosis of Fahr's syndrome, revealing a hypoparathyroidism was posed. He was supplemented with calcium and alpha cholecalciferol and treated with clozapine (100mg per day). After four weeks, psychotic symptoms responded well to this treatment without expressing any side effects, notably seizures. DISCUSSION: Psychotic symptoms seen in Fahr's disease include auditory and visual hallucinations, complex perceptual distortions, delusions, and fugue state. Some of them were manifest in this patient. It is likely that the psychosis in both Fahr's disease and schizophrenia share a similar pathology. Positive psychotic symptoms, hallucinations, and paranoia are not necessarily generated by the classical hypothesis of dopamine-mediated attachment of salience to internally generated stimuli. Still, there is some evidence that disruption of the cortex involved in the pathophysiology of schizophrenia is also seen in Fahr's disease, particularly in areas of the limbic system. CONCLUSION: Psychiatrists should consider Fahr's syndrome as a differential diagnosis in the evaluation of psychosis associated with seizures. This case, along with others in the literature, further emphasizes the importance of the role of neuro-imaging and the search for disrupted phosphocalcic metabolism in patients with atypical psychotic symptoms. Moreover, further research should focus on pharmacologic interventions. The efficacy and risks of neuropharmacologic and psychopharmacologic interventions in Fahr's syndrome, and correlates of good and poor outcome with these interventions remain to be defined.


Subject(s)
Basal Ganglia Diseases/diagnosis , Brain Diseases/diagnosis , Calcinosis/diagnosis , Neurocognitive Disorders/diagnosis , Neurodegenerative Diseases/diagnosis , Basal Ganglia/pathology , Basal Ganglia Diseases/drug therapy , Basal Ganglia Diseases/psychology , Brain Diseases/drug therapy , Brain Diseases/psychology , Calcinosis/drug therapy , Calcinosis/psychology , Calcium/therapeutic use , Cholecalciferol/therapeutic use , Clozapine/therapeutic use , Humans , Hypoparathyroidism/diagnosis , Hypoparathyroidism/drug therapy , Hypoparathyroidism/psychology , Male , Middle Aged , Neurocognitive Disorders/drug therapy , Neurocognitive Disorders/psychology , Neurodegenerative Diseases/drug therapy , Neurodegenerative Diseases/psychology , Tomography, X-Ray Computed
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