scholarly journals Pregnancies after Childhood Craniopharyngioma: Results of KRANIOPHARYNGEOM 2000/2007 and Review of the Literature

2020 ◽  
Vol 111 (1-2) ◽  
pp. 16-26
Author(s):  
Panjarat Sowithayasakul ◽  
Svenja Boekhoff ◽  
Brigitte Bison ◽  
Hermann L. Müller

<b><i>Background:</i></b> Data on female fertility, pregnancy, and outcome of offspring after childhood-onset craniopharyngioma (CP) are rare. <b><i>Study Design:</i></b> Observational study on pregnancy rate and offspring outcome in female CP patients recruited in KRANIOPHARYNGEOM 2000/2007 since 2000. <b><i>Results:</i></b> A total of 451 CP patients (223 female) have been recruited, and 269 (133 female) were postpubertal at study. Six of 133 female CP patients (4.5%) with a median age of 14.9 years at CP diagnosis had 9 pregnancies, giving birth to 10 newborns. Three patients achieved complete surgical resections. No patient underwent postoperative irradiation. Five natural pregnancies occurred in 3 CP patients without pituitary deficiencies. Four pregnancies in 3 CP patients with hypopituitarism were achieved under assisted reproductive techniques (ART) (median 4.5 cycles, range: 3–6 cycles). Median maternal age at pregnancy was 30 years (range: 22–41 years). Six babies (60%) were delivered by caesarean section. Median gestational age at delivery was 38 weeks (range: 34–43 weeks); median birth weight was 2,920 g (range: 2,270–3,520 g), the rate of preterm delivery was 33%. Enlargements of CP cysts occurred in 2 women during pregnancy. Other complications during pregnancy, delivery, and postnatal period were not observed. <b><i>Conclusions:</i></b> Pregnancies after CP are rare and were only achieved after ART in patients with hypopituitarism. Close monitoring by an experienced reproductive physician is necessary. Due to a potentially increased risk for cystic enlargement, clinical, ophthalmological, and MRI monitoring are recommended in patients at risk. Severe perinatal complications, birth defects, and postnatal morbidity of mothers and offspring were not observed.

2020 ◽  
Vol 22 (Supplement_3) ◽  
pp. iii455-iii455
Author(s):  
Panjarat Sowithayasakul ◽  
Svenja Boekhoff ◽  
Brigitte Bison ◽  
Hermann L Müller

Abstract BACKGROUND Data on female fertility, pregnancy, and outcome of offspring after childhood-onset craniopharyngioma (CP) are rare. STUDY DESIGN Observational study on pregnancy rate and offspring outcome in female CP patients recruited in KRANIOPHARYNGEOM 2000/2007. RESULTS A total of 451 CP patients (223 female) have been recruited, and 269 (133 female) were postpubertal at study. Six of 133 female CP patients (4.5%) with median age of 14.9 years at CP diagnosis had 9 pregnancies, giving birth to 10 newborns. Three patients achieved complete surgical resections. No patient underwent postoperative irradiation. Five natural pregnancies occurred in 3 CP patients without pituitary deficiencies. Four pregnancies in 3 CP patients with hypopituitarism were achieved under assisted reproductive techniques (ART) (median 4.5 cycles, range: 3–6 cycles). Median maternal age at pregnancy was 30 years (range: 22–41 years). Six babies (60%) were delivered by caesarean section. Median gestational age at delivery was 38 weeks (range: 34–43 weeks); median birth weight was 2,920 g (range: 2,270– 3,520 g), the rate of preterm delivery was 33%. Enlargements of CP cysts occurred in 2 women during pregnancy. Other complications during pregnancy, delivery, and postnatal period were not observed. CONCLUSIONS Pregnancies after CP are rare and were only achieved after ART in patients with hypopituitarism. Close monitoring by an experienced reproductive physician is necessary. Due to a potentially increased risk for cystic enlargement, clinical, ophthalmological, and MRI monitoring are recommended in patients at risk. Perinatal complications, birth defects, and morbidity of mothers and offspring were not observed.


2020 ◽  
Vol 4 (Supplement_1) ◽  
Author(s):  
Panjarat Sowithayasakul ◽  
Svenja Boekhoff ◽  
Hermann Lothar Muller

Abstract Background: Hypopituitarism is associated with an increased risk of pregnancy complications, such as abortion, anemia, pregnancy-induced hypertension, placental abruption, premature birth, and postpartum hemorrhage. The advance of assisted reproductive techniques makes it possible to improve the pregnancy rate in hypopituitary patients. Data on female fertility, pregnancy, and outcome of offspring after childhood-onset, adamantinomatous craniopharyngioma (CP) are rare. Study design: Observational study on pregnancy rate and outcome of offspring after childhood-onset CP in adult, female patients recruited in KRANIOPHARYNGEOM 2000/2007. Patient cohorts: Since 2000, 451 CP patients (223 f / 228 m) have been recruited with high grade of completeness. 263 CP patients (128 f / 135 m) have reached adult age. 6 of 128 adult, female CP patients (5%) reported on 9 pregnancies giving birth to 10 healthy newborns. Results: The median age at time of CP diagnosis was 14.9 years. Complete surgical CP resections were achieved in 3 patients. No patient underwent postoperative irradiation. 5 natural pregnancies occurred in 3 CP patients presenting with postoperative normal pituitary function. 4 pregnancies were achieved in 3 CP with hypopituitarism under assisted reproductive techniques (after in median 4.5 cycles, range: 3-6 cycles). Median maternal age at pregnancy was 30 years, ranging from 22 to 41 years. 6 of 10 babies were delivered by caesarean section. Gestational age at delivery was in median 38 weeks, ranging from 34 to 43 weeks; median birth weight was 2,920 gram (range: 2,270-3,520 gram), the rate of preterm delivery (&lt;38 weeks of gestation) was 33%. The rate of breastfeeding was 56%. Enlargements of CP cysts occurred in 2 women during pregnancy. Other severe complications during pregnancy, delivery and postnatal period were not observed. Conclusions: Pregnancies after CP are rare (5%) and almost half of the patients (45%) achieved pregnancies after assisted reproductive techniques, which are effective and safe in CP patients. With regard to existing deficiencies of hypothalamic-pituitary axes, close monitoring and care by an experienced reproductive physician is necessary. Furthermore, MRI monitoring especially of CP cysts is recommended during pregnancy. Severe perinatal complications, birth defects, and postnatal morbidity of the mothers and their offspring were not observed. Most CP patients complained about their initial lack of information on potential fertility under assisted reproductive techniques.


2021 ◽  
Vol 0 (0) ◽  
Author(s):  
Julita Szarpak ◽  
Karol Ciejka ◽  
Weronika Perczyńska ◽  
Michał Flis ◽  
Paulina Wróbel-Knybel

Abstract Introduction: According to the neurodevelopmental theory, schizophrenia is a cognitive-behavioral manifestation of the non-physiological development of the CNS, which was caused by the interaction of genetic and environmental factors during its formation. Aim, material and methodology: The aim of this article is to present a clinical case in which exposure to numerous adverse environmental factors in the pre and postnatal period could cause the development of schizophrenia in adulthood. The analysis of the clinical case and the medical records of a patient diagnosed with paranoid schizophrenia, suffering from numerous perinatal complications, was performed. Case report: A woman, 23 years old, unmarried, studying at post-secondary school - archivistics. Currently, psychiatrically hospitalized for the second time. A patient from the first, twin, fraternal pregnancy. The delivery took place by caesarean section, complicated. In the APGAR scale, she scored 4 points in the 1st minute, and 6 points in the 3rd minute. By day 30, the patient had experienced preterm retinopathy, viral infection, salmonella, jaundice and anemia. In the second month of her life, she was treated with surgery due to intestinal obstruction. Psychomotor development was disturbed. At 9 years of age, there was a retinal detachment. Until the end of junior high school, she was educated in a special school for the visually impaired. Conclusions: The neurodevelopmental theory assumes that the increased risk of developing schizophrenia is associated with unfavorable factors in the developmental period. Identifying the causes underlying the development of the disease is a key step towards prevention, more efficient diagnosis, and improvement of the effectiveness of treatment of patients suffering from this disease.


Biomedicines ◽  
2021 ◽  
Vol 9 (7) ◽  
pp. 764
Author(s):  
Shih-Lung Cheng ◽  
Kuo-Chin Chiu ◽  
Hsin-Kuo Ko ◽  
Diahn-Warng Perng ◽  
Hao-Chien Wang ◽  
...  

Purpose: To understand the association between biomarkers and exacerbations of severe asthma in adult patients in Taiwan. Materials and Methods: Demographic, clinical characteristics and biomarkers were retrospectively collected from the medical charts of severe asthma patients in six hospitals in Taiwan. Exacerbations were defined as those requiring asthma-specific emergency department visits/hospitalizations, or systemic steroids. Enrolled patients were divided into: (1) those with no exacerbations (non-exacerbators) and (2) those with one or more exacerbations (exacerbators). Receiver operating characteristic curves were used to determine the optimal cut-off value for biomarkers. Generalized linear models evaluated the association between exacerbation and biomarkers. Results: 132 patients were enrolled in the study with 80 non-exacerbators and 52 exacerbators. There was no significant difference in demographic and clinical characteristics between the two groups. Exacerbators had significantly higher eosinophils (EOS) counts (367.8 ± 357.18 vs. 210.05 ± 175.24, p = 0.0043) compared to non-exacerbators. The optimal cut-off values were 292 for EOS counts and 19 for the Fractional exhaled Nitric Oxide (FeNO) measure. Patients with an EOS count ≥ 300 (RR = 1.88; 95% CI, 1.26–2.81; p = 0.002) or FeNO measure ≥ 20 (RR = 2.10; 95% CI, 1.05–4.18; p = 0.0356) had a significantly higher risk of exacerbation. Moreover, patients with both an EOS count ≥ 300 and FeNO measure ≥ 20 had a significantly higher risk of exacerbation than those with lower EOS count or lower FeNO measure (RR = 2.16; 95% CI, 1.47–3.18; p = < 0.0001). Conclusions: Higher EOS counts and FeNO measures were associated with increased risk of exacerbation. These biomarkers may help physicians identify patients at risk of exacerbations and personalize treatment for asthma patients.


2020 ◽  
Vol 30 (5) ◽  
pp. 686-691
Author(s):  
Christina J. Ge ◽  
Amanda C. Mahle ◽  
Irina Burd ◽  
Eric B. Jelin ◽  
Priya Sekar ◽  
...  

AbstractObjective:To evaluate delivery management and outcomes in fetuses prenatally diagnosed with CHD.Study design:A retrospective cohort study was conducted on 6194 fetuses (born between 2013 and 2016), comparing prenatally diagnosed with CHD (170) to those with non-cardiac (234) and no anomalies (5790). Primary outcomes included the incidence of preterm delivery and mode of delivery.Results:Gestational age at delivery was significantly lower between the CHD and non-anomalous cohorts (38.6 and 39.1 weeks, respectively). Neonates with CHD had a significantly lower birth weights (p < 0.001). There was an approximately 1.5-fold increase in the rate of primary cesarean sections associated with prenatally diagnosed CHD with an odds ratio of 1.49 (95% CI 1.06–2.10).Conclusions:Our study provides additional evidence that the prenatal diagnosis of CHD is associated with a lower birth weight, preterm delivery, and with an increased risk of delivery by primary cesarean section.


2021 ◽  
Vol 20 (1) ◽  
Author(s):  
Emma V. Preston ◽  
Victoria Fruh ◽  
Marlee R. Quinn ◽  
Michele R. Hacker ◽  
Blair J. Wylie ◽  
...  

Abstract Background Prenatal endocrine disrupting chemical (EDC) exposure has been associated with increased risk of preterm birth. Non-Hispanic Black women have higher incidence of preterm birth compared to other racial/ethnic groups and may be disproportionately exposed to EDCs through EDC-containing hair products. However, research on the use of EDC-associated hair products during pregnancy and risk of preterm birth is lacking. Therefore, the objective of this pilot study was to estimate associations of prenatal hair product use with gestational age at delivery in a Boston, Massachusetts area pregnancy cohort. Methods The study population consisted of a subset of participants enrolled in the Environmental Reproductive and Glucose Outcomes (ERGO) Study between 2018 and 2020. We collected self-reported data on demographics and hair product use using a previously validated questionnaire at four prenatal visits (median: 12, 19, 26, 36 weeks’ gestation) and abstracted gestational age at delivery from medical records. We compared gestational age and hair product use by race/ethnicity and used linear regression to estimate covariate-adjusted associations of product use and frequency of use at each study visit with gestational age at delivery. Primary models were adjusted for maternal age at enrollment and delivery method. Results Of the 154 study participants, 7% delivered preterm. Non-Hispanic Black participants had lower mean gestational age at delivery compared to non-Hispanic White participants (38.2 vs. 39.2 weeks) and were more likely to report ever and more frequent use of hair products. In regression models, participants reporting daily use of hair oils at visit 4 had lower mean gestational age at delivery compared to non-users (β: -8.3 days; 95% confidence interval: -14.9, -1.6). We did not find evidence of associations at earlier visits or with other products. Conclusions Frequent use of hair oils during late pregnancy may be associated with shorter gestational duration. As hair oils are more commonly used by non-Hispanic Black women and represent potentially modifiable EDC exposure sources, this may have important implications for the known racial disparity in preterm birth.


2021 ◽  
Vol 12 ◽  
pp. 204209862098569
Author(s):  
Phyo K. Myint ◽  
Ben Carter ◽  
Fenella Barlow-Pay ◽  
Roxanna Short ◽  
Alice G. Einarsson ◽  
...  

Background: Whilst there is literature on the impact of SARS viruses in the severely immunosuppressed, less is known about the link between routine immunosuppressant use and outcome in COVID-19. Consequently, guidelines on their use vary depending on specific patient populations. Methods: The study population was drawn from the COPE Study (COVID-19 in Older People), a multicentre observational cohort study, across the UK and Italy. Data were collected between 27 February and 28 April 2020 by trained data-collectors and included all unselected consecutive admissions with COVID-19. Load (name/number of medications) and dosage of immunosuppressant were collected along with other covariate data. Primary outcome was time-to-mortality from the date of admission (or) date of diagnosis, if diagnosis was five or more days after admission. Secondary outcomes were Day-14 mortality and time-to-discharge. Data were analysed with mixed-effects, Cox proportional hazards and logistic regression models using non-users of immunosuppressants as the reference group. Results: In total 1184 patients were eligible for inclusion. The median (IQR) age was 74 (62–83), 676 (57%) were male, and 299 (25.3%) died in hospital (total person follow-up 15,540 days). Most patients exhibited at least one comorbidity, and 113 (~10%) were on immunosuppressants. Any immunosuppressant use was associated with increased mortality: aHR 1.87, 95% CI: 1.30, 2.69 (time to mortality) and aOR 1.71, 95% CI: 1.01–2.88 (14-day mortality). There also appeared to be a dose–response relationship. Conclusion: Despite possible indication bias, until further evidence emerges we recommend adhering to public health measures, a low threshold to seek medical advice and close monitoring of symptoms in those who take immunosuppressants routinely regardless of their indication. However, it should be noted that the inability to control for the underlying condition requiring immunosuppressants is a major limitation, and hence caution should be exercised in interpretation of the results. Plain Language Summary Regular Use of Immune Suppressing Drugs is Associated with Increased Risk of Death in Hospitalised Patients with COVID-19 Background: We do not have much information on how the COVID-19 virus affects patients who use immunosuppressants, drugs which inhibit or reduce the activity of the immune system. There are various conflicting views on whether immune-suppressing drugs are beneficial or detrimental in patients with the disease. Methods: This study collected data from 10 hospitals in the UK and one in Italy between February and April 2020 in order to identify any association between the regular use of immunosuppressant medicines and survival in patients who were admitted to hospital with COVID-19. Results: 1184 patients were included in the study, and 10% of them were using immunosuppressants. Any immunosuppressant use was associated with increased risk of death, and the risk appeared to increase if the dose of the medicine was higher. Conclusion: We therefore recommend that patients who take immunosuppressant medicines routinely should carefully adhere to social distancing measures, and seek medical attention early during the COVID-19 pandemic.


Rheumatology ◽  
2020 ◽  
Vol 59 (Supplement_2) ◽  
Author(s):  
Mariam Malik ◽  
Himashi Anver ◽  
Ernest Wong

Abstract Background Toxoplasma gondii is thought to infect up to a third of world’s population. Incidence rate of 0.4/100,000 has been calculated in Britain, culminating in a life-time risk of 18/100,000. Cats are primary hosts, but humans and warm-blooded animals can be infected by consumption of contaminated food/water. Although in most patients, it’s self-limiting, it can be devastating in immunosuppressed patients and may cause eye manifestations, cerebral abscesses or disseminated infection. Immunosuppressive therapies including treatment with biologics increases the risk and may also cause toxoplasmosis reactivation. Methods This is case of 57 year old lady with psoriatic arthritis. She has past history of congenital vision impairment in the left eye and is HLA B27 negative. She enjoyed horse-riding and had a pet dog. Initially she was started on methotrexate. Sulfasalazine was added later. Due to ongoing active disease, etanercept was used for 6 months, before being switched to cetrolizumab due to ineffectiveness. She had this for 5 months and then switched to infliximab, 3mg/kg, 8 weekly. In May 2019, she was seen by Ophthalmology for 2 weeks history of blurred vision and floaters in right eye. She was diagnosed to have panuveitis and had positive IgM for toxoplasma. Bloods revealed negative TB screen, HIV, Hep B&C, syphilis, lyme and anti-streptolysin antibody tests were negative. Infliximab levels were sub-therapeutic. She was commenced on 30mg prednisolone for possible inflammatory process secondary to seronegative arthropathy, but acute toxoplasmosis could not be excluded. Hence, she was started on azithromycin and had vitreous biopsy. Toxoplasma was detected in the sample, confirming acute infection. Methotrexate and infliximab were stopped. MRI head ruled out intracranial involvement. Following treatment of acute toxoplasmosis, adalimumab is now being considered for management of her inflammatory disease, with close monitoring by local infectious-disease team and specialist ophthalmology unit. Results This lady developed ocular toxoplasmosis and panuveitis, whilst on immunosuppression for psoriatic arthritis. She was a horse-rider and had exposure to dogs. Diagnosing toxoplasma in immunocompromised can be difficult. Isolation of T. gondii in tissue usually confirms diagnosis. Some forms of immunosuppressive treatment may be associated with increased risk of reactivation of toxoplasmosis but there is not much evidence to assess the relative risk of various therapies. Conclusion Ocular toxoplasmosis needs to be considered in patients receiving immunosupression and presenting with inflammatory eye symptoms. Management requires specialist input and close monitoring. Further research into diagnostic techniques, possibility of using prophylaxis in high-risk patients and management guidelines would be helpful. Disclosures M. Malik None. H. Anver None. E. Wong None.


2010 ◽  
Vol 28 (1) ◽  
pp. 160-165 ◽  
Author(s):  
Joseph Ciccolini ◽  
Laetitia Dahan ◽  
Nicolas André ◽  
Alexandre Evrard ◽  
Muriel Duluc ◽  
...  

Purpose Anticipating toxicities with gemcitabine is an ongoing story, and deregulation in cytidine deaminase (CDA) could be associated with increased risk of developing early severe toxicities on drug exposure. Patients and Methods A simple test to evaluate CDA phenotypic status was first validated in an animal model investigating relationships between CDA activity and gemcitabine-related toxicities. Next, relevance of this test as a marker for toxicities was retrospectively tested in a first subset of 64 adult patients treated with gemcitabine alone, then it was tested in a larger group of 130 patients who received gemcitabine either alone or combined with other drugs and in 20 children. Additionally, search for the 435 T>C, 208 G>A and 79 A>C mutations on the CDA gene was performed. Results In mice, CDA deficiency impacted on gemcitabine pharmacokinetics and had subsequent lethal toxicities. In human, 12% of adult patients experienced early severe toxicities after gemcitabine administration. A significant difference in CDA activities was observed between patients with and without toxicities (1.2 ± 0.8 U/mg v 4 ± 2.6 U/mg; P < .01). Conversely, no genotype-to-phenotype relationships were found. Of note, the patients who displayed particularly reduced CDA activity all experienced strong toxicities. Gemcitabine was well tolerated in children, and no CDA deficiency was evidenced. Conclusion Our data suggest that CDA functional testing could be a simple and easy marker to discriminate adult patients at risk of developing severe toxicities with gemcitabine. Particularly, this study demonstrates that CDA deficiency, found in 7% of adult patients, is associated with a maximum risk of developing early severe toxicities with gemcitabine.


2012 ◽  
Vol 141 (6) ◽  
pp. 1223-1231 ◽  
Author(s):  
E. R. C. MILLETT ◽  
D. NOEL ◽  
P. MANGTANI ◽  
I. ABUBAKAR ◽  
M. E. KRUIJSHAAR

SUMMARYCompletion of treatment is key to tuberculosis control. Using national surveillance data we assessed factors associated with tuberculosis patients being lost to follow-up before completing treatment (‘lost’). Patients reported in England, Wales and Northern Ireland between 2001 and 2007 who were lost 12 months after beginning treatment were compared to those who completed, or were still on treatment, using univariable and multivariable logistic regression. Of 41 120 patients, men [adjusted odds ratio (aOR) 1·29; 95% confidence interval (CI) 1·23–1·35], 15- to 44-year-olds (P<0·001), and patients with pulmonary sputum smear-positive disease (aOR 1·25, 95% CI 1·12–1·45) were at higher risk of being lost. Those recently arrived in the UK were also at increased risk, particularly those of the White ethnic group (aOR 6·39, 95% CI 4·46–9·14). Finally, lost patients had a higher risk of drug resistance (aOR 1·41, 95% CI 1·17–1·69). Patients at risk of being lost require enhanced case management and novel case retention methods are needed to prevent this group contributing towards onward transmission.


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