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Ekman, Bertil
Alternative names
Publications (10 of 35) Show all publications
Jonsson, A. K., Lövborg, H., Lohr, W., Ekman, B. & Rocklov, J. (2017). Increased Risk of Drug-Induced Hyponatremia during High Temperatures. International Journal of Environmental Research and Public Health, 14(7), Article ID 827.
Open this publication in new window or tab >>Increased Risk of Drug-Induced Hyponatremia during High Temperatures
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2017 (English)In: International Journal of Environmental Research and Public Health, ISSN 1661-7827, E-ISSN 1660-4601, Vol. 14, no 7, article id 827Article in journal (Refereed) Published
Abstract [en]

Purpose: To investigate the relationship between outdoor temperature in Sweden and the reporting of drug-induced hyponatremia to the Medical Products Agency (MPA). Methods: All individual adverse drug reactions (ADR) reported to MPA from 1 January 2010 to 31 October 2013 of suspected drug-induced hyponatremia and random controls were identified. Reports where the ADR had been assessed as having at least a possible relation to the suspected drug were included. Information on administered drugs, onset date, causality assessment, sodium levels, and the geographical origin of the reports was extracted. A case-crossover design was used to ascertain the association between heat exposure and drug-induced hyponatremia at the individual level, while linear regression was used to study its relationship to sodium concentration in blood. Temperature exposure data were obtained from the nearest observation station to the reported cases. Results: During the study period, 280 reports of hyponatremia were identified. More cases of drug-induced hyponatremia were reported in the warmer season, with a peak in June, while other ADRs showed an opposite annual pattern. The distributed lag non-linear model indicated an increasing odds ratio (OR) with increasing temperature in the warm season with a highest odds ratio, with delays of 1-5 days after heat exposure. A cumulative OR for a lag time of 1 to 3 days was estimated at 2.21 at an average daily temperature of 20 degrees C. The change in sodium per 1 degrees C increase in temperature was estimated to be -0.37 mmol/L (95% CI: -0.02, -0.72). Conclusions: Warm weather appears to increase the risk of drug-induced hyponatremia.

Place, publisher, year, edition, pages
MDPI AG, 2017
Keywords
average daily temperature; hyponatremia; adverse drug reaction
National Category
Occupational Health and Environmental Health
Identifiers
urn:nbn:se:liu:diva-140807 (URN)10.3390/ijerph14070827 (DOI)000407370700159 ()28737683 (PubMedID)
Note

Funding Agencies|County Council of Ostergotland [LIO-448321]; Umea Centre for Global Health Research at Umea University; FORTE; Swedish Council for Working Life and Social Research [2006 1512]

Available from: 2017-09-13 Created: 2017-09-13 Last updated: 2018-02-22
Svanberg, C., Norevall, L.-I., Ekman, B., Wahlberg Topp, J. & Bågesund, M. (2016). Cephalometric analysis of adults with Turner syndrome. Swedish Dental Journal, 40(1), 33-41
Open this publication in new window or tab >>Cephalometric analysis of adults with Turner syndrome
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2016 (English)In: Swedish Dental Journal, ISSN 0347-9994, Vol. 40, no 1, p. 33-41Article in journal (Refereed) Published
Abstract [en]

Turner syndrome (TS) is a genetic disorder of females with a prevalence of 1/2000-3000 live female births. The aim of this study was to compare cephalometric variables from adult women diagnosed with TS to a standardized reference group of 31-year old healthy women, and to evaluate the possible effects of human growth hormone (hGH) therapy in women with TS. Registered TS subjects in the Southeast region of Sweden were invited to take part in the study. Twenty-one women aged 36 +/- 13(18-57) years accepted participation. Lateral radiographs of the head were analyzed using standard cephalometric methods (Hasund analysis) and with the commercially available soft-ware program FACAD. Comparisons were made with roentgen-cephalometric standards from a reference group of nineteen 31-year old Swedish women. Analysis of the cephalometric radiographs from the TS subjects showed a more retrognathic maxilla (SNA 80.3 +/- 5.4) (p=0.0460) and mandible (SNB 77.0 +/- 5.2) (p=0.0014), and a correspondingly backward position of the chin (SN/Pg 78.9 +/- 5.5) (p=0.0046) as compared to the reference values of 31-year old women (SNA 83.2 +/- 3.0, SNB 81.5 +/- 2.3 and SNPg 83.0 +/- 2.3, respectively). In addition there was an increased posterior inclination of the maxilla (SN/NL 8.6 +/- 4.1), as compared to the reference values (SN/NL 5,3 +/- 2.7) (p=0.0048). There were no significant differences regarding sagittal or vertical jaw relations, mandibular inclination or cranial base angle between the TS-group and the 31-year olds with the reference values. No significant difference was seen in jaw relationship, as measured by the ANB value, however the Wits(index) (3.3 +/- 3.5) was higher (p=0.0001) than the reference values (-0.1 +/- 1.8). Subjects with or without previous hGH administration did not show any significant differences in cephalometric values. In conclusion, women with TS had a significantly more retrognathic maxilla (SNA) and mandible (SNB) and a correspondingly significantly posterior position of the chin (SN/Pg), a significantly increased posterior inclination of the maxilla (SN/NL) and a significantly increased Witsindex as compared to the reference group of 31-year old women. No craniofacial variables differed significantly between previously hGH-treated and not hGH-treated women with TS.

Place, publisher, year, edition, pages
SWEDISH DENTAL JOURNAL, 2016
Keywords
Turner syndrome; cephalometric analysis; growth hormone
National Category
Public Health, Global Health, Social Medicine and Epidemiology Dentistry
Identifiers
urn:nbn:se:liu:diva-128984 (URN)000374716300005 ()
Note

Funding Agencies|Folktandvarden Ostergotland; Medical Research Council of Southeast Sweden

Available from: 2016-06-09 Created: 2016-06-07 Last updated: 2018-03-19
Wahlberg Topp, J., Ekman, B., Nystrom, L., Hanson, U., Persson, B. & Arnqvist, H. (2016). Gestational diabetes: Glycaemic predictors for fetal macrosomia and maternal risk of future diabetes. Diabetes Research and Clinical Practice, 114, 99-105
Open this publication in new window or tab >>Gestational diabetes: Glycaemic predictors for fetal macrosomia and maternal risk of future diabetes
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2016 (English)In: Diabetes Research and Clinical Practice, ISSN 0168-8227, E-ISSN 1872-8227, Vol. 114, p. 99-105Article in journal (Refereed) Published
Abstract [en]

Aims: To investigate how glucose levels at diagnosis of gestational diabetes (GDM) are associated with infant birth weight and long-term risk of manifest diabetes mellitus in the mother. Methods: In a case control study GDM pregnancies (n = 2085) were compared with non-GDM pregnancies matched for day of delivery and obstetric unit (n = 3792). GDM was defined as capillary blood glucose (cB-glucose) >9.0 mmol/l (plasma glucose >10.0 mmol/l) after a 75 g oral glucose tolerance test (OGTT). The GDM cohort were followed up 8.5-13.5 yrs after initial diagnosis with a questionnaire, answered by 1324 GDM women (65%). Results: GDM women had higher mean infant birth-weight compared with controls (3682 g vs. 3541 g, P < 0.001). In multiple linear regression analysis, birth weight was positively correlated to fasting cB-glucose at GDM diagnosis (P < 0.001), increased week of gestation (P < 0.001) and BMI before pregnancy (P < 0.003), while 2 h OGTT cB-glucose values >= 9.0 mmol/l were not related. Infants born to mothers with fasting cB-glucose >= 4.5 mmol/l had no increased mean birth-weight or macrosomia (>= 4500 g) compared to controls. In the follow up 334/1324 women (25%) of the GDM women had developed diabetes, 215 type 2 diabetes, 46 type 1 diabetes and 72 unclassified diabetes. In logistic regression fasting cB-glucose and 2 h OGTT cB-glucose at diagnosis of GDM as well as BMI >25 and origin outside Europe were risk factors for manifest diabetes. Conclusions: Fasting blood glucose at diagnosis of GDM gives important information besides 2 h OGTT glucose about pregnancy outcome and future risk for maternal diabetes. (C) 2016 Elsevier Ireland Ltd. All rights reserved.

Place, publisher, year, edition, pages
ELSEVIER IRELAND LTD, 2016
Keywords
Birth weight; Pregnancy; GDM; Blood glucose; OGTT
National Category
Clinical Medicine
Identifiers
urn:nbn:se:liu:diva-128751 (URN)10.1016/j.diabres.2015.12.017 (DOI)000375129600015 ()26818892 (PubMedID)
Note

Funding Agencies|Swedish Diabetes Association; Medical Research Council of Southeast Sweden (FORSS); Linkoping University, Sweden

Available from: 2016-05-31 Created: 2016-05-30 Last updated: 2017-04-24
Burman, P., Eden-Engstrom, B., Ekman, B., Anders Karlsson, F., Schwarcz, E. & Wahlberg Topp, J. (2016). Limited value of cabergoline in Cushings disease: a prospective study of a 6-week treatment in 20 patients. European Journal of Endocrinology, 174(1), 17-24
Open this publication in new window or tab >>Limited value of cabergoline in Cushings disease: a prospective study of a 6-week treatment in 20 patients
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2016 (English)In: European Journal of Endocrinology, ISSN 0804-4643, E-ISSN 1479-683X, Vol. 174, no 1, p. 17-24Article in journal (Refereed) Published
Abstract [en]

Context and objective: The role of cabergoline in Cushings disease (CD) remains controversial. The experience is limited to case reports and few open studies that report the effects determined after >= 1 month of treatment. In prolactinomas and dopamine-responsive GH-secreting tumours, effects of cabergoline are seen within days or weeks. Here, we searched for short-term effects of cabergoline in CD. Design: Twenty patients (19 naive and one recurrent) were included in a prospective study. Cabergoline was administered in increasing doses of 0.5-5 mg/week over 6 weeks. Methods: Urinary free cortisol (UFC) 24 h, morning cortisol and ACTH, and salivary cortisol at 0800, 1600 and 2300 h were determined once weekly throughout. Diurnal curves (six samples) of serum cortisol were measured at start and end. Results: At study end, the median cabergoline dose was 5 mg, range 2.5-5 mg/week. The prolactin levels, markers of compliance, were suppressed in all patients. During the treatment, hypercortisolism varied, gradual and dose-dependent reductions were not seen. Five patients had a >50% decrease of UFC, three had a >50% rise of UFC. Salivary cortisol at 2300 h showed a congruent >50% change with UFC in two of the five cases with decreased UFC, and in one of the three cases with increased UFC. One patient with decreases in both UFC and 2300 h salivary cortisol also had a reduction in diurnal serum cortisol during the course of the study. Conclusions: Cabergoline seems to be of little value in the management of CD. Only one patient had a response-like pattern. Given the known variability of disease activity in CD, this might represent a chance finding.

Place, publisher, year, edition, pages
BIOSCIENTIFICA LTD, 2016
National Category
Clinical Medicine
Identifiers
urn:nbn:se:liu:diva-124115 (URN)10.1530/EJE-15-0807 (DOI)000366401700007 ()26582653 (PubMedID)
Note

Funding Agencies|Swedish Association of Local Authorities and Regions to the Swedish Pituitary Study Group; University of Lund; Linkoping; Uppsala

Available from: 2016-01-22 Created: 2016-01-19 Last updated: 2017-04-24
Bergthorsdottir, R., Nilsson, A. G., Gillberg, P., Ekman, B. & Wahlberg, J. (2015). Health-Related Quality of Life In Patients With Adrenal Insufficiency Receiving Plenadren Compared With Immediate-Release Hydrocortisone.. Value in Health, 18(7), A616
Open this publication in new window or tab >>Health-Related Quality of Life In Patients With Adrenal Insufficiency Receiving Plenadren Compared With Immediate-Release Hydrocortisone.
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2015 (English)In: Value in Health, ISSN 1098-3015, E-ISSN 1524-4733, Vol. 18, no 7, p. A616-Article in journal, Meeting abstract (Refereed) Published
Abstract [en]

Background

Previous studies in patients with primary adrenal insufficiency (PAI) on conventional replacement therapy suggest decreased health-related quality of life (HRQoL), and that patients report more frequently fatigue, increased anxiety and inability to work compared to background population.

Objectives

To study self-reported health status with EQ-5D in patients with PAI. Patients treated with Plenadren (modified-release hydrocortisone) were compared with patients treated with immediate release hydrocortisone (IRHC) replacement therapy.

Methods

This was a cross-sectional, multi-centre, non-interventional survey of patients with PAI receiving Plenadren or immediate release hydrocortisone (IRHC) replacement.

Subjects

One hundred thirty-four adult patients with PAI of whom 36 (19 females [53%]) were treated with Plenadren and 98 (77 females [79%]) were treated with IRHC, were included.

MAIN OUTCOME MEASURE

HRQoL described by the EQ-5D, a generic preference-based measure of health.

RESULTS

Patients on Plenadren and on IRHC had a mean ± SD age of 53.1 ± 12.7 years and 48.0 ± 13.1 years, respectively (P=0.043). The majority of the patients were diagnosed more than 5 years ago (69%). The mean ± SD daily Plenadren and IRHC doses were 27.0 ± 6.8 mg and 26.6 ± 10.9 mg, respectively (P=0.807). 47% of the Plenadren patients had been receiving Plenadren and 82% of the IRHC patients had been receiving IRHC for more than 3 years. Patients receiving Plenadren had better HRQoL measured by the EQ-5D questionnaire compared to patients replaced with IRHC (0.76 ± 0.18 vs 0.68 ± 0.18, respectively [P=0.040]).

CONCLUSIONS

Replacement therapy with Plenadren in patients with PAI confers measurable benefit on HRQoL relative to IRHC as estimated by the EQ-5D questionnaire, and may therefore be advantageous when compared to IRHC substitution.

National Category
Health Care Service and Management, Health Policy and Services and Health Economy Surgery
Identifiers
urn:nbn:se:liu:diva-124656 (URN)10.1016/j.jval.2015.09.2145 (DOI)26533455 (PubMedID)
Available from: 2016-02-09 Created: 2016-02-08 Last updated: 2017-11-30
Ekman, B., Wahlberg Topp, J. & Landberg, E. (2015). Urine oligosaccharide pattern in patients with hyperprolactinaemia. Glycoconjugate Journal, 32(8), 635-641
Open this publication in new window or tab >>Urine oligosaccharide pattern in patients with hyperprolactinaemia
2015 (English)In: Glycoconjugate Journal, ISSN 0282-0080, E-ISSN 1573-4986, Vol. 32, no 8, p. 635-641Article in journal (Refereed) Published
Abstract [en]

Free milk-type oligosaccharides are produced during pregnancy and lactation and may have an impact on several cells in the immune system. Our aim was to investigate if patients with isolated hyperprolactinaemia, not related to pregnancy, also have increased synthesis and urinary excretion of milk-type oligosaccharides and to compare the excretion pattern with that found during pregnancy. Urine samples were collected as morning sample from 18 patients with hyperprolactinaemia, 13 healthy controls with normal prolactin levels and four pregnant women. After purification, lactose and free oligosaccharides were analysed and quantified by high-performance anion-exchange chromatography with pulsed amperometric detection. The identity of peaks was confirmed by exoglycosidase treatment and comparison with oligosaccharide standards. Prolactin was measured in serum collected between 09 and 11 a.m. by a standardized immunochemical method. Patients with hyperprolactinaemia had higher urinary excretion of lactose than normoprolactinemic controls and urinary lactose correlated positively to prolactin levels (r = 0.51, p less than 0.05). Increased levels of the fucosylated oligosaccharides 2-fucosyl lactose and lacto-di-fucotetraose were found in urine from three and two patients, respectively. The acidic oligosaccharide 3-sialyl lactose was found in high amount in urine from two patients with prolactin of greater than 10,000 mU/l. However, pregnant women in their third trimester had the highest concentration of all these oligosaccharides and excretion increased during pregnancy. This study is first to show that both lactose and certain fucosylated and sialylated milk-type oligosaccharides are increased in some patients with hyperprolactinaemia. It remains to elucidate the functional importance of these findings.

Place, publisher, year, edition, pages
SPRINGER, 2015
Keywords
Prolactin; Prolactinoma; Urine; Oligosaccharides; Lactose
National Category
Clinical Medicine
Identifiers
urn:nbn:se:liu:diva-122650 (URN)10.1007/s10719-015-9610-x (DOI)000363488300006 ()26275984 (PubMedID)
Note

Funding Agencies|FORSS (Medical Research Council of Southeast Sweden) [4065]; Faculty of Health and Sciences, Linkoping University, Sweden

Available from: 2015-11-16 Created: 2015-11-13 Last updated: 2017-12-01
Ekman, B., Alstrand, N., Bachrach-Lindström, M., Jenmalm, M. C. & Wahlberg, J. (2014). Altered Chemokine Th1/Th2 Balance in Addison's Disease: Relationship with Hydrocortisone Dosing and Quality of Life. Hormone and Metabolic Research, 46(1), 48-53
Open this publication in new window or tab >>Altered Chemokine Th1/Th2 Balance in Addison's Disease: Relationship with Hydrocortisone Dosing and Quality of Life
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2014 (English)In: Hormone and Metabolic Research, ISSN 0018-5043, E-ISSN 1439-4286, Vol. 46, no 1, p. 48-53Article in journal (Refereed) Published
Abstract [en]

The adrenalitis found in autoimmune Addison’s disease (AAD) is considered having a Th1-driven pathogenesis. Circulating Th1- and Th2-associated chemokines responsible for the trafficking of leukocytes to inflammatory sites are markers for the Th1/Th2 balance. The aim of the study was to assess if the same daily hydrocortisone dose of 30 mg given in either 2 or 4 doses to patients with AAD could affect the Th1/Th2 balance of circulating chemokines.

Fifteen patients (6 women) with AAD were included in this randomised, placebo controlled, double blind cross-over study. Samples for chemokines, Th1-associated (CXCL10, CXCL11) and Th2-associated (CCL17, CCL22), were drawn 5 times during a 24-h period at the end of each treatment period and analysed with Luminex. Seven control subjects did the same diurnal blood sampling once. Subjects with AAD had higher median diurnal levels of the Th1-associated chemokines than controls, CXCL10 [43 (33–56) pg/ml vs. 22 (19–34) pg/ml, p<0.01] and CXCL11 [37 (29–48) pg/ml vs. 16 (9–24) pg/ml, p<0.001], whereas no significant difference was found regarding the Th2-related chemokines. Similar chemokine levels were found when the same hydrocortisone dose of 30 mg was divided in 2 or 4 doses. Levels of CXCL11 correlated negatively with scores of SF-36 domains (high score indicate better health) of General Health (GH) and total score for Physical Component Summary (PCS), and these negative correlations were most pronounced at 04:00 h on the 2-dose regimen. Patients with AAD have a dominant Th1 chemokine profile that partially correlates to reduced quality of life.

National Category
Endocrinology and Diabetes
Identifiers
urn:nbn:se:liu:diva-104003 (URN)10.1055/s-0033-1351291 (DOI)000329563500004 ()
Available from: 2014-02-06 Created: 2014-02-06 Last updated: 2019-01-09
Ekman, B., Fitts, D., Marelli, C., Murray, R. D., Quinkler, M. & Zelissen, P. M. J. (2014). European Adrenal Insufficiency Registry (EU-AIR): a comparative observational study of glucocorticoid replacement therapy. BMC Endocrine Disorders, 14(40)
Open this publication in new window or tab >>European Adrenal Insufficiency Registry (EU-AIR): a comparative observational study of glucocorticoid replacement therapy
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2014 (English)In: BMC Endocrine Disorders, ISSN 1472-6823, E-ISSN 1472-6823, Vol. 14, no 40Article in journal (Refereed) Published
Abstract [en]

BACKGROUND:

Increased morbidity and mortality associated with conventional glucocorticoid replacement therapy for primary adrenal insufficiency (primary AI; estimated prevalence 93-140/million), secondary AI (estimated prevalence, 150-280/million, respectively) or congenital adrenal hyperplasia (estimated prevalence, approximately 65/million) may be due to the inability of typical glucocorticoid treatment regimens to reproduce the normal circadian profile of plasma cortisol. A once-daily modified-release formulation of hydrocortisone has been developed to provide a plasma cortisol profile that better mimics the daytime endogenous profile of cortisol. Here, we describe the protocol for the European Adrenal Insufficiency Registry (EU-AIR), an observational study to assess the long-term safety of modified-release hydrocortisone compared with conventional glucocorticoid replacement therapies in routine clinical practice (ClinicalTrials.gov identifier: NCT01661387).

METHODS:

Patients enrolled in EU-AIR have primary or secondary AI and are receiving either modified-release or conventional glucocorticoid replacement therapy. The primary endpoints of EU-AIR are the incidence of intercurrent illness, adrenal crisis and serious adverse events (SAEs), as well as the duration of SAEs and dose changes related to SAEs. Data relating to morbidity, mortality, adverse drug reactions, dosing and concomitant therapies will be collected. Patient diaries will record illness-related dose changes between visits. All decisions concerning medical care are made by the registry physician and patient. Enrolment is targeted at achieving 3600 patient-years of treatment (1800 patient-years per group) for the primary analysis, which is focused on determining the non-inferiority of once-daily modified-release replacement therapy compared with conventional glucocorticoid therapy.

RESULTS:

Recruitment began in August 2012 and, as of March 2014, 801 patients have been enrolled. Fifteen centres are participating in Germany, the UK and Sweden, with recruitment soon to be initiated in the Netherlands.

CONCLUSIONS:

EU-AIR will provide a unique opportunity not only to collect long-term safety data on a modified-release preparation of glucocorticoid but also to evaluate baseline data on conventional glucocorticoid replacement. Such data should help to improve the treatment of AI.

Place, publisher, year, edition, pages
BioMed Central, 2014
National Category
Clinical Medicine
Identifiers
urn:nbn:se:liu:diva-109287 (URN)10.1186/1472-6823-14-40 (DOI)000338366200001 ()24884782 (PubMedID)
Available from: 2014-08-11 Created: 2014-08-11 Last updated: 2017-12-05Bibliographically approved
Nilsson, A. G., Marelli, C., Fitts, D., Bergthorsdottir, R., Burman, P., Dahlqvist, P., . . . Johannsson, G. (2014). Prospective evaluation of long-term safety of dual-release hydrocortisone replacement administered once daily in patients with adrenal insufficiency. European Journal of Endocrinology, 171(3), 369-377
Open this publication in new window or tab >>Prospective evaluation of long-term safety of dual-release hydrocortisone replacement administered once daily in patients with adrenal insufficiency
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2014 (English)In: European Journal of Endocrinology, ISSN 0804-4643, E-ISSN 1479-683X, Vol. 171, no 3, p. 369-377Article in journal (Refereed) Published
Abstract [en]

Objective: The objective was to assess the long-term safety profile of dual-release hydrocortisone (DR-HC) in patients with adrenal insufficiency (AI). Design: Randomised, open-label, crossover trial of DR-HC or thrice-daily hydrocortisone for 3 months each (stage 1) followed by two consecutive, prospective, open-label studies of DR-HC for 6 months (stage 2) and 18 months (stage 3) at five university clinics in Sweden. Methods: Sixty-four adults with primary AI started stage 1, and an additional 16 entered stage 3. Patients received DR-HC 20-40 mg once daily and hydrocortisone 20-40 mg divided into three daily doses (stage 1 only). Main outcome measures were adverse events (AEs) and intercurrent illness (self-reported hydrocortisone use during illness). Results: In stage 1, patients had a median 1.5 (range, 1-9) intercurrent illness events with DR-HC and 1.0 (1-8) with thrice-daily hydrocortisone. AEs during stage 1 were not related to the cortisol exposure-time profile. The percentage of patients with one or more AEs during stage 1 (73.4% with DR-HC; 65.6% with thrice-daily hydrocortisone) decreased during stage 2, when all patients received DR-HC (51% in the first 3 months; 54% in the second 3 months). In stages 1-3 combined, 19 patients experienced 27 serious AEs, equating to 18.6 serious AEs/100 patient-years of DR-HC exposure. Conclusions: This long-term prospective trial is the first to document the safety of DR-HC in patients with primary AI and demonstrates that such treatment is well tolerated during 24 consecutive months of therapy.

Place, publisher, year, edition, pages
BioScientifica, 2014
National Category
Clinical Medicine
Identifiers
urn:nbn:se:liu:diva-112489 (URN)10.1530/EJE-14-0327 (DOI)000343670900015 ()24944332 (PubMedID)
Note

Funding Agencies|ViroPharma SPRL, Maidenhead, UK

Available from: 2014-11-28 Created: 2014-11-28 Last updated: 2017-12-05
Wahlberg, J. & Ekman, B. (2013). Atypical or typical adrenocorticotropic hormone-producing pulmonary carcinoids and the usefulness of 11C-5-hydroxytryptophan positron emission tomography: two case reports. Journal of Medical Case Reports, 7, 80
Open this publication in new window or tab >>Atypical or typical adrenocorticotropic hormone-producing pulmonary carcinoids and the usefulness of 11C-5-hydroxytryptophan positron emission tomography: two case reports
2013 (English)In: Journal of Medical Case Reports, ISSN 1752-1947, E-ISSN 1752-1947, Vol. 7, p. 80-Article in journal (Refereed) Published
Abstract [en]

Introduction

Pulmonary carcinoids associated with ectopic adrenocorticotropic hormone secretion have a good prognosis if histological examination shows typical pulmonary carcinoid and low proliferation, whereas a poor outcome is linked to atypical pulmonary carcinoid and high proliferation. Here we describe the diagnostic challenges to find the tumor in Cushing’s syndrome secondary to ectopic adrenocorticotropic hormone secretion in two cases with an atypical and a typical pulmonary carcinoid, respectively.     

Case presentation

A 63-year-old Caucasian woman presented with aggressive clinical features related to Cushing’s syndrome, having very high levels of urinary cortisol and circulating adrenocorticotropic hormone and cortisol. Magnetic resonance imaging showed no pituitary tumor, and bilateral inferior petrosal sinus sampling revealed no central peripheral ratio of adrenocorticotropic hormone. Computed tomography and 111Indium-pentetreoide somatostatin receptor scintigraphy could not visualize any ectopic tumor. The patient was referred for an 11C-5-hydroxytryptophan positron emission tomography, and a small 8mm nodule in her left lung was found. The tumor was removed via a lateral thoracic incision and wedge excision. The histological examination showed an atypical carcinoid with Ki-67 index of 9 to 10%, and an additional lobectomy was performed.     

The second patient, a 22-year-old Caucasian man, also presented with aggressive Cushing’s syndrome, with very high urinary cortisol levels and increased circulating cortisol as well as adrenocorticotropic hormone levels. A magnetic resonance imaging scan of the pituitary showed no tumor, whereas a 12×9×14mm tumor was detected in the right lung on the primary computed tomography scan and no further investigation was performed. The tumor was removed via a lateral thoracic incision and wedge excision. A typical carcinoid with Ki-67 index of 1 to 2% was found and no further surgery was performed.     

After surgical removal, the biochemical disturbances resolved and significant clinical improvement were achieved in both patients after 24 months of follow up.     

Conclusions

Diagnostic evaluation time is limited due to the aggressive course in ectopic adrenocorticotropic hormone-dependent Cushing’s syndrome. We suggest that 11C-5-hydroxytryptophan positron emission tomography could be considered early as a secondary diagnostic tool when primary computed tomography and/or magnetic resonance imaging scans fail to show any tumor.

Place, publisher, year, edition, pages
BioMed Central, 2013
Keywords
ACTH; ACTH syndrome; Cortisol; Cushing’s syndrome; Ectopic; Pulmonary carcinoid
National Category
Medical and Health Sciences
Identifiers
urn:nbn:se:liu:diva-102327 (URN)10.1186/1752-1947-7-80 (DOI)
Available from: 2013-12-05 Created: 2013-12-05 Last updated: 2017-12-06Bibliographically approved
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