High prevalence of early endocrine disorders after childhood brain tumors in a large cohort

Abstract Objective To describe the endocrine follow-up of patients with primary brain tumors. Methods non interventional observational study, data collection from medical records of 221 patients followed at a Pediatric Endocrinology Department. Results 48.9% were female, median age at diagnosis: 6.7 years (range: 0-15.9), median follow-up: 6.7 years (0.3-26.6). Main tumor types: medulloblastoma (37.6%), craniopharyngioma (29.0%) and glioma (20.4%). According to anatomical location, 48% were suprasellar (SS) and 52% non-suprasellar (NSS). GH deficiency (GHD) prevalence was similar in both groups (SS: 83.0%, NSS: 76.5%, p=0.338), appearing in median 1.8 years (-0.8 to 12.4) after diagnosis; post-radiotherapy GHD appeared in median 1.6 years after radiotherapy (0.2 to 10.7). Hypothyroidism was more prevalent in SS (76.4%), than NSS (33.9%), p<0.001, as well as ACTH deficiency (SS: 69.8%, NSS: 6.1%, p<0.001). Early puberty was similar in SS (16%) and NSS (12.2%). Hypogonatropic hypogonadism was predominant in SS (63.1%) vs NSS (1.3%), p<0.001, and post-chemotherapy gonadal toxicity in NSS (29.6%) vs SS (2.8%), p<0.001. Adult height was lower for NSS compared to target height (-1.0 SD, p<0.0001) and to SS patients (p<0.0001). Thyroid nodules were found in 13/45 patients (28.8%), including 4 cancers (4.8-11.5 years after radiotherapy). Last follow-up visit BMI was higher in both groups (p=0.0001), obesity incidence was higher for SS (46.2%) than NSS (17.4%). Conclusions We found a high incidence of early onset endocrine disorders. An endocrine consultation and nutritional evaluation should be mandatory for all patients with a brain tumor, especially when the tumor is suprasellar or after hypothalamus/pituitary irradiation.

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Pseudohypoparathyroidism: application of the Italian common healthcare-pathway for a homogeneous clinical approach and a shared follow up

Italian Journal of Pediatrics ◽

10.1186/s13052-021-01000-z ◽

2021 ◽

Vol 47 (1) ◽

Author(s):

Daniele Tessaris ◽

Elisa Bonino ◽

Giovanna Weber ◽

Malgorzata Wasniewska ◽

Domenico Corica ◽

...

Keyword(s):

Cognitive Impairment ◽

Clinical Practice ◽

Care Pathway ◽

Delayed Puberty ◽

Endocrine Disorders ◽

Clinical Approach ◽

Pediatric Endocrinology ◽

Bone Mineral Status ◽

Few Data

Abstract Background Pseudohypoparathyroidism (PHP) represents a heterogeneous group of rare endocrine disorders caused by (epi) genetic abnormalities affecting the GNAS locus. It is mainly characterized by resistance to PTH and TSH, and by peculiar clinical features such as short stature, obesity, cognitive impairment, subcutaneous ossifications and brachydactyly. Delayed puberty, GHRH and calcitonin resistances have also been described. The healthcare-pathway recently proposed by the Italian Society of Pediatric Endocrinology and Diabetology (ISPED) has provided a standardized clinical approach to these conditions. The purpose of the present study was to evaluate its application in clinical practice, and to collect data for setting future specific studies. Methods Through a semi-structured survey, based on the indications of the care-pathway, data on PHP clinical management were collected. The compilation of each data in the survey was read as an index of the adoption of the healthcare-pathway in clinical practice. Results In addition to the proposing Center, 4 Centers joined the study, thus obtaining a large collection of data on 48 PHP patients. Highest rates in the completion of data were obtained for diagnostic history, auxological measurements and subcutaneous ossifications evaluation. As expected, the availability of data for the other investigated fields was lower, coming from recent research studies. More information has been obtained on hormonal resistance classically involved in PHP (PTH, TSH, GHRH and GnRH) and on cognitive impairment, while a few data has been collected on bone mineral status, calcitonin levels and glucolipid metabolism. Conclusions The presented data show that the ISPED healthcare-pathway could represent a valid tool both to confirm the clinical approach to PHP patients and to allow homogeneous data collection; however, it has not yet been fully adopted. The strengthening of the network among the major Italian Endocrine Centers will contribute to improve its application in clinical practice, optimizing the follow-up of these patients and increasing knowledge on PHP.

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Coexistent Osteoarthritis and Parkinson’s Disease: Data from the Parkinson’s Foundation Outcomes Project

Journal of Parkinson s Disease ◽

10.3233/jpd-202170 ◽

2020 ◽

Vol 10 (4) ◽

pp. 1601-1610

Author(s):

Jaimie A. Roper ◽

Abigail C. Schmitt ◽

Hanzhi Gao ◽

Ying He ◽

Samuel Wu ◽

...

Keyword(s):

Parkinson’S Disease ◽

Parkinson's Disease ◽

Study Data ◽

Functional Mobility ◽

Quality Improvement Initiative ◽

Timed Up And Go ◽

Mobility Performance ◽

Risk Of Mortality ◽

The Impact

Background: The impact of concurrent osteoarthritis on mobility and mortality in individuals with Parkinson’s disease is unknown. Objective: We sought to understand to what extent osteoarthritis severity influenced mobility across time and how osteoarthritis severity could affect mortality in individuals with Parkinson’s disease. Methods: In a retrospective observational longitudinal study, data from the Parkinson’s Foundation Quality Improvement Initiative was analyzed. We included 2,274 persons with Parkinson’s disease. The main outcomes were the effects of osteoarthritis severity on functional mobility and mortality. The Timed Up and Go test measured functional mobility performance. Mortality was measured as the osteoarthritis group effect on survival time in years. Results: More individuals with symptomatic osteoarthritis reported at least monthly falls compared to the other groups (14.5% vs. 7.2% without reported osteoarthritis and 8.4% asymptomatic/minimal osteoarthritis, p = 0.0004). The symptomatic group contained significantly more individuals with low functional mobility (TUG≥12 seconds) at baseline (51.5% vs. 29.0% and 36.1%, p < 0.0001). The odds of having low functional mobility for individuals with symptomatic osteoarthritis was 1.63 times compared to those without reported osteoarthritis (p < 0.0004); and was 1.57 times compared to those with asymptomatic/minimal osteoarthritis (p = 0.0026) after controlling pre-specified covariates. Similar results hold at the time of follow-up while changes in functional mobility were not significant across groups, suggesting that osteoarthritis likely does not accelerate the changes in functional mobility across time. Coexisting symptomatic osteoarthritis and Parkinson’s disease seem to additively increase the risk of mortality (p = 0.007). Conclusion: Our results highlight the impact and potential additive effects of symptomatic osteoarthritis in persons with Parkinson’s disease.

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Social determinants of health affecting treatment of pediatric brain tumors

Journal of Neurosurgery Pediatrics ◽

10.3171/2019.4.peds18594 ◽

2019 ◽

Vol 24 (2) ◽

pp. 159-165

Author(s):

Jillian M. Berkman ◽

Jonathan Dallas ◽

Jaims Lim ◽

Ritwik Bhatia ◽

Amber Gaulden ◽

...

Keyword(s):

African American ◽

Health Disparities ◽

Brain Tumors ◽

Social Determinants Of Health ◽

Social Determinants ◽

Determinants Of Health ◽

Cns Tumor ◽

Initial Symptoms ◽

Caucasian Patients

OBJECTIVELittle is understood about the role that health disparities play in the treatment and management of brain tumors in children. The purpose of this study was to determine if health disparities impact the timing of initial and follow-up care of patients, as well as overall survival.METHODSThe authors conducted a retrospective study of pediatric patients (< 18 years of age) previously diagnosed with, and initially treated for, a primary CNS tumor between 2005 and 2012 at Monroe Carell Jr. Children’s Hospital at Vanderbilt. Primary outcomes included time from symptom presentation to initial neurosurgery consultation and percentage of missed follow-up visits for ancillary or core services (defined as no-show visits). Core services were defined as healthcare interactions directly involved with CNS tumor management, whereas ancillary services were appointments that might be related to overall care of the patient but not directly focused on treatment of the tumor. Statistical analysis included Pearson’s chi-square test, nonparametric univariable tests, and multivariable linear regression. Statistical significance was set a priori at p < 0.05.RESULTSThe analysis included 198 patients. The median time from symptom onset to initial presentation was 30.0 days. A mean of 7.45% of all core visits were missed. When comparing African American and Caucasian patients, there was no significant difference in age at diagnosis, timing of initial symptoms, or tumor grade. African American patients missed significantly more core visits than Caucasian patients (p = 0.007); this became even more significant when controlling for other factors in the multivariable analysis (p < 0.001). African American patients were more likely to have public insurance, while Caucasian patients were more likely to have private insurance (p = 0.025). When evaluating survival, no health disparities were identified.CONCLUSIONSNo significant health disparities were identified when evaluating the timing of presentation and survival. A racial disparity was noted when evaluating missed follow-up visits. Future work should focus on identifying reasons for differences and whether social determinants of health affect other aspects of treatment.

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Seven-Year Follow-Up of Screening for Hypertension and Diabetes at a Dental Clinic

Journal of Dentistry and Oral Sciences ◽

10.37191/mapsci-2582-3736-1(2)-011 ◽

2019 ◽

Author(s):

Göran Friman

Keyword(s):

Antihypertensive Drugs ◽

Study Data ◽

Initial Screening ◽

Pharmacological Treatments ◽

Medical Screening ◽

Follow Up Study ◽

Undiagnosed Hypertension ◽

Dental Setting ◽

Study Participants

Objective: To describe the distribution of risk, diagnosis and pharmacological treatments for diabetes and hypertension after seven years among patients provided with opportunistic medical screening in a dental setting. Material and Methods: The initial screening’s 170 participants were asked to take part in a seven-year follow-up study. Data were collected through self-reported information in a written health declaration. Outcome measures: • Number of study participants who had passed away • Prescription of antidiabetics or antihypertensives • Changes in weight and height to calculate body mass index (BMI) Results: The follow-up study consisted of 151 participants. Twenty had passed away. The risk needs for medicating with antihypertensive drugs after seven years for those not receiving pharmacological treatment at the initial screening was 3.7 times greater (p=0.025 CI 1.2-11.3) for participants with a diastolic blood pressure (BP) ≥ 90 mm Hg (85 for diabetics) than for the others. The risk was 3.9 times greater (p=0.020 CI 1.2-12.6) for those with a systolic BP of 140-159 mm Hg and 54.2 times greater (p<0.0001 CI 9.8-300.3) for those with a systolic BP ≥ 160 mm Hg than for those with a systolic BP 140 mm Hg. There were no changes in BMI. Conclusion: At least one in ten cases of incorrect medication or undiagnosed hypertension may be identifiable through opportunistic medical screening

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Premature STEMI in Men and Women: Current Clinical Features and Improvements in Management and Prognosis

Journal of Clinical Medicine ◽

10.3390/jcm10061314 ◽

2021 ◽

Vol 10 (6) ◽

pp. 1314

Author(s):

Rebeca Lorca ◽

Isaac Pascual ◽

Andrea Aparicio ◽

Alejandro Junco-Vicente ◽

Rut Alvarez-Velasco ◽

...

Keyword(s):

Risk Factors ◽

Cardiovascular Risk ◽

Coronary Artery ◽

General Population ◽

Cardiovascular Risk Factors ◽

Young Women ◽

Artery Dissection ◽

Men And Women ◽

High Prevalence

Background: Coronary artery disease (CAD) is the most frequent cause of ST-segment elevation myocardial infarction (STEMI). Etiopathogenic and prognostic characteristics in young patients may differ from older patients and young women may present worse outcomes than men. We aimed to evaluate the clinical characteristics and prognosis of men and women with premature STEMI. Methods: A total 1404 consecutive patients were referred to our institution for emergency cardiac catheterization due to STEMI suspicion (1 January 2014–31 December 2018). Patients with confirmed premature (<55 years old in men and <60 in women) STEMI (366 patients, 83% men and 17% women) were included (359 atherothrombotic and 7 spontaneous coronary artery dissection (SCAD)). Results: Premature STEMI patients had a high prevalence of classical cardiovascular risk factors. Mean follow-up was 4.1 years (±1.75 SD). Mortality rates, re-hospitalization, and hospital stay showed no significant differences between sexes. More than 10% of women with premature STEMI suffered SCAD. There were no significant differences between sexes, neither among cholesterol levels nor in hypolipemiant therapy. The global survival rates were similar to that expected in the general population of the same sex and age in our region with a significantly higher excess of mortality at 6 years among men compared with the general population. Conclusion: Our results showed a high incidence of cardiovascular risk factors, a high prevalence of SCAD among young women, and a generally good prognosis after standardized treatment. During follow-up, 23% suffered a major cardiovascular event (MACE), without significant differences between sexes and observed survival at 1, 3, and 6 years of follow-up was 96.57% (95% CI 94.04–98.04), 95.64% (95% CI 92.87–97.35), and 94.5% (95% CI 91.12–97.66). An extra effort to prevent/delay STEMI should be invested focusing on smoking avoidance and optimal hypolipemiant treatment both in primary and secondary prevention.

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Prognostic Value of Oxygenated Hemoglobin Assessed during Acute Exacerbations of Chronic Pulmonary Disease

Respiration ◽

10.1159/000513440 ◽

2021 ◽

pp. 1-8

Author(s):

Clemens F. Hinke ◽

Rudolf A. Jörres ◽

Peter Alter ◽

Robert Bals ◽

Florian Bornitz ◽

...

Keyword(s):

Pulmonary Disease ◽

Predictive Value ◽

Hemoglobin Concentration ◽

Blood Gas Analysis ◽

Study Data ◽

Gas Analysis ◽

Chronic Obstructive ◽

Acute Exacerbations ◽

All Cause Mortality

Background: Oxygenated hemoglobin(OxyHem) is a simple-to-measure marker of oxygen content capable of predicting all-cause mortality in stable chronic obstructive pulmonary disease (COPD). Objectives: We aimed to analyze its predictive value during acute exacerbations of COPD (AECOPD). Methods: In this retrospective study, data from 227 patients discharged after severe AECOPD at RoMed Clinical Center Rosenheim, Germany, between January 2012 and March 2018, was analyzed. OxyHem (hemoglobin concentration [Hb] × fractional SpO2, g/dL) was calculated from oxygen saturation measured by pulse oximetry and hemoglobin assessed within 24 h after admission. The follow-up (1.7 ± 1.5 years) covered all-cause mortality, including readmissions for severe AECOPD. Results: During the follow-up period, 127 patients died, 56 due to AECOPD and 71 due to other reasons. Survivors and non-survivors showed differences in age, FVC % predicted, C-reactive protein, hemoglobin, Cr, Charlson Comorbidity Index (CCI), and OxyHem (p < 0.05 each). Significant independent predictors of survival were BMI, Cr or CCI, FEV1 % predicted or FVC % predicted, Hb, or OxyHem. The predictive value of OxyHem (p = 0.006) was superior to that of Hb or SpO2 and independent of oxygen supply during blood gas analysis. OxyHem was also predictive when using a cutoff value of 12.1 g/dL identified via receiver operating characteristic curves in analyses including either the CCI (hazard ratio 1.85; 95% CI 1.20, 2.84; p = 0.005) or Cr (2.04; 95% CI 1.35, 3.10; p = 0.001) as covariates. Conclusion: The concentration of OxyHem provides independent, easy-to-assess information on long-term mortality risk in COPD, even if measured during acute exacerbations. It therefore seems worth to be considered for broader clinical use.

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Long-term remission of acromegaly after somatostatin analogues withdrawal: a single-centre experience

Journal of Endocrinological Investigation ◽

10.1007/s40618-021-01562-z ◽

2021 ◽

Author(s):

E. Sala ◽

G. Carosi ◽

G. Del Sindaco ◽

R. Mungari ◽

A. Cremaschi ◽

...

Keyword(s):

Median Time ◽

Pituitary Surgery ◽

Somatostatin Analogues ◽

Exclusion Criterion ◽

Single Centre ◽

Disease Evolution ◽

Single Centre Experience ◽

Pituitary Irradiation

Abstract Purpose A long-lasting remission of acromegaly after somatostatin analogues (SAs) withdrawal has been described in some series. Our aim was to update the disease evolution after SAs withdrawal in a cohort of acromegalic patients. Methods We retrospectively evaluated 21 acromegalic patients previously included in a multicentre study (Ronchi et al. 2008), updating data at the last follow-up. We added further 8 patients selected for SAs withdrawal between 2008–2018. Pituitary irradiation represented an exclusion criterion. The withdrawal was suggested after at least 9 months of clinical and hormonal disease control. Clinical and biochemical data prior and after SAs withdrawal were analysed. Results In the whole cohort (29 patients) mean age was 50 ± 14.9 years and 72.4% were females. In 69% pituitary surgery was previously performed. Overall, the median time of treatment before SAs withdrawal was 53 months (IQR = 24–84). At the last follow up in 2019, 23/29 patients (79.3%) had a disease relapse after a median time of 6 months (interquartile range or IQR = 3–12) from the drug suspension, while 6/29 (20.7%) were still on remission after 120 months (IQR = 66–150). IGF-1 levels were significantly lower before withdrawal in patients with persistent remission compared to relapsing ones (IGF-1 SDS: -1.5 ± 0.6 vs -0.11 ± 1, p = 0.01). We did not observe any other difference between patients with and without relapse, including SAs formulation, dosage and treatment duration. Conclusion A successful withdrawal of SAs is possible in a subset of well-controlled acromegalic patients and it challenges the concept that medical therapy is a lifelong requirement.

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RONC-19. TWO CASES OF RE-IRRADIATION FOR LATE RECURRENT OR RADIATION-INDUCED TUMOR AFTER RADIATION THERAPY FOR PEDIATRIC BRAIN TUMORS

Neuro-Oncology ◽

10.1093/neuonc/noaa222.788 ◽

2020 ◽

Vol 22 (Supplement_3) ◽

pp. iii459-iii459

Author(s):

Takashi Mori ◽

Shigeru Yamaguchi ◽

Rikiya Onimaru ◽

Takayuki Hashimoto ◽

Hidefumi Aoyama

Keyword(s):

Radiation Therapy ◽

Brain Tumors ◽

Tumor Resection ◽

Intensity Modulated Radiation Therapy ◽

Late Recurrence ◽

Pediatric Brain Tumors ◽

Suprasellar Region ◽

Radiation Induced ◽

Pediatric Brain

Abstract BACKGROUND As the outcome of pediatric brain tumors improves, late recurrence and radiation-induced tumor cases are more likely to occur, and the number of cases requiring re-irradiation is expected to increase. Here we report two cases performed intracranial re-irradiation after radiotherapy for pediatric brain tumors. CASE 1: 21-year-old male. He was diagnosed with craniopharyngioma at eight years old and underwent a tumor resection. At 10 years old, the local recurrence of suprasellar region was treated with 50.4 Gy/28 fr of stereotactic radiotherapy (SRT). After that, other recurrent lesions appeared in the left cerebellopontine angle, and he received surgery three times. The tumor was gross totally resected and re-irradiation with 40 Gy/20 fr of SRT was performed. We have found no recurrence or late effects during the one year follow-up. CASE 2: 15-year-old female. At three years old, she received 18 Gy/10 fr of craniospinal irradiation and 36 Gy/20 fr of boost to the posterior fossa as postoperative irradiation for anaplastic ependymoma and cured. However, a anaplastic meningioma appeared on the left side of the skull base at the age of 15, and 50 Gy/25 fr of postoperative intensity-modulated radiation therapy was performed. Two years later, another meningioma developed in the right cerebellar tent, and 54 Gy/27 fr of SRT was performed. Thirty-three months after re-irradiation, MRI showed a slight increase of the lesion, but no late toxicities are observed. CONCLUSION The follow-up periods are short, however intracranial re-irradiation after radiotherapy for pediatric brain tumors were feasible and effective.

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NCOG-42. INITIAL PRESENTATION AND OUTPATIENT VISIT COMPLIANCE OF INMATES WITH BRAIN TUMORS

Neuro-Oncology ◽

10.1093/neuonc/noaa215.580 ◽

2020 ◽

Vol 22 (Supplement_2) ◽

pp. ii138-ii138

Author(s):

Iyad Alnahhas ◽

Appaji Rayi ◽

Yasmeen Rauf ◽

Shirley Ong ◽

Pierre Giglio ◽

...

Keyword(s):

Brain Tumors ◽

Civil Liberties ◽

Delayed Diagnosis ◽

Low Grade ◽

Small Series ◽

Initial Symptoms ◽

The Ohio State University ◽

American Civil Liberties Union ◽

Lost To Follow Up

Abstract INTRODUCTION While advocacy for inmates with cancer has recently gained momentum, little is known about management of brain tumors in inmates. Delays in acknowledging or recognizing nonspecific initial symptoms can lead to delayed diagnosis and treatment. Inmates with cancer are reported to either be ignored or receive substandard care due in part to cost or logistics (American Civil Liberties Union; ASCO Post 2018). METHODS In this retrospective study, we identified inmates with gliomas seen in the Ohio State University Neuro-oncology Center between 1/1/2010-4/20/2019. RESULTS Twelve patients were identified. Median age at presentation was 39.5 years (range 28-62). Eleven patients were Caucasian and one was African American. Diagnoses included glioblastoma (GBM) (n=6), anaplastic astrocytoma (n=1), anaplastic oligodendroglioma (n=1), low-grade astrocytoma (n=3) and anaplastic pleomorphic xanthroastrocytoma (n=1). Patients were more likely to present early after seizures or focal neurologic deficits (9/12) than after headaches alone. Patients with GBM started RT 12-71 days after surgery (median 34.5). One patient’s post-RT MRI was delayed by a month and another with GBM had treatment held after 4 cycles of adjuvant temozolomide (TMZ) due to “incarceration issues”. For one patient who received adjuvant TMZ, the facility failed to communicate with the primary team throughout treatment. Two patients suffered significant nausea while on chemotherapy due to inability to obtain ondansetron in prison, or due to wrong timing. 7/12 (58%) patients were lost to follow-up for periods of 3-15 months during treatment. Three patients refused adjuvant treatment. CONCLUSIONS Although this is a small series, our results highlight the inequities and challenges faced by inmates with gliomas who are more likely to forego treatments or whose incarceration prevents them from keeping appropriate treatment and follow-up schedules. Additional studies are needed to define and address these deficiencies in the care of inmates with brain tumors and other cancers.

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Symptom Burden of Patients with Advanced Pancreas Cancer (APC): A Provincial Cancer Institute Observational Study

Current Oncology ◽

10.3390/curroncol28040244 ◽

2021 ◽

Vol 28 (4) ◽

pp. 2789-2800

Author(s):

Stephanie Lelond ◽

Julie Ward ◽

Pascal J. Lambert ◽

Christina A. Kim

Keyword(s):

Cox Regression ◽

Advanced Pancreatic Cancer ◽

Symptom Burden ◽

Distress Score ◽

Ambulatory Oncology ◽

High Prevalence ◽

Associated Symptoms ◽

Report Symptom

Patients with advanced pancreatic cancer (APC) experience many disease-related symptoms. ESAS-r measures the severity of 9 symptom domains and has been validated for use in the ambulatory oncology setting. We aimed to describe symptom burden at baseline for patients with APC treated with modern chemotherapy (CT), and to determine whether symptom burden at baseline is prognostic. Patients diagnosed with APC between 2012–2016, treated with ≥1 cycle of CT, who completed ≥1 ESAS-r were identified. Descriptive statistics were used to report symptom burden and common moderate-to-severe symptoms. A joint model was used to describe the trajectory of ESAS-r during follow-up while controlling for death. Multivariable Cox regression was used to identify independent predictors of death. Of 123 patients identified, the median age was 65 and 61% had metastatic disease. The median baseline ESAS-r total symptom distress score (TSDS) was 24. A total of 86% of patients had at least one symptom score of ≥4 at baseline, with the most common being: fatigue, nausea, anxiety, and shortness of breath. Median overall survival was 10.2 months. Baseline TSDS was not predictive for worse survival in the era of modern CT. Patients with APC have a high burden of cancer-associated symptoms and a high prevalence of moderate-to-severe symptoms. Early intervention has the potential to improve quality of life in this group of patients and should be investigated.

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