scholarly journals Hypoparathyroidism and Pseudohypoparathyroidism in Pregnancy: An Italian Retrospective Observational Study

2020 ◽  
Author(s):  
Gemma Marcucci ◽  
Paola Altieri ◽  
Salvatore Benvenga ◽  
Marta Bondanelli ◽  
Valentina Camozzi ◽  
...  
Keyword(s):  
Calcium Carbonate ◽  
Serum Albumin ◽  
Case Reports ◽  
Post Partum ◽  
Case Series ◽  
Urinary Calcium ◽  
Total Calcium ◽  
Maternal Complications ◽  
Pharmacological Management ◽  
The Mean

Abstract Background: Hypoparathyroidism (HypoPT) or pseudo-hypoparathyroidism (pseudo-HypoPT) during pregnancy may cause maternal and fetal/neonatal complications. In this regard, only a few case reports or case series of pregnant or lactating women have been published. The purpose of this study was to describe clinical and biochemical course, pharmacological management, and potential adverse events during pregnancy and post-partum in pregnant women with HypoPT or pseudo-HypoPT. This was a retrospective, observational, multicenter, study involving nine Italian referral centers for endocrine diseases affiliated with the Italian Society of Endocrinology and involved in “Hypoparathyroidism Working Group”.Results: This study identified a cohort of 28 women (between 2005 and 2018) with HypoPT (n=25, 80% postsurgical) and pseudo-HypoPT (n=3). The mean calcium carbonate and calcitriol doses of both groups were increased during pregnancy compared to pre-pregnancy period. During the three trimesters of pregnancy the mean calcium carbonate and calcitriol doses were variable, remaining unchanged throughout gestation respectively in 40% and 60% of women with HypoPT, whilst tended to decrease from the third trimester to the post-partum six months. Most identified women (~70%) did not display maternal complications and (~90%) maintained mean serum albumin-corrected total calcium levels within the low-to-mid normal reference range (8.5±0.8 mg/dl) during pregnancy. The main complications related to pregnancy period included: preterm birth (n=3 HypoPT women), and history of miscarriages (n=6 HypoPT women and n=2 pseudo-HypoPT women). Conclusion: This study shows that mean serum albumin-corrected total calcium levels were carefully monitored during pregnancy and post-pregnancy, with limited evaluation of other biochemical parameters, such as serum phosphate, 24h urinary calcium, 25-OH vitamin D, and creatinine clearance. To avoid complications in mothers and offspring, intense biochemical, clinical and pharmacological monitoring during pregnancy and breastfeeding is highly recommended.

scholarly journals Hypoparathyroidism and pseudohypoparathyroidism in pregnancy: an Italian retrospective observational study

2021 ◽  
Vol 16 (1) ◽  
Author(s):  
Gemma Marcucci ◽  
Paola Altieri ◽  
Salvatore Benvenga ◽  
Marta Bondanelli ◽  
Valentina Camozzi ◽  
...  
Keyword(s):  
Calcium Carbonate ◽  
Serum Albumin ◽  
Post Partum ◽  
Case Series ◽  
Total Calcium ◽  
Third Trimester ◽  
The Third ◽  
The Mean ◽  
The Individual ◽  
In Pregnancy

Abstract Background Hypoparathyroidism (HypoPT) or pseudo-hypoparathyroidism (pseudo-HypoPT) during pregnancy may cause maternal and fetal/neonatal complications. In this regard, only a few case reports or case series of pregnant or lactating women have been published. The purpose of this study was to describe clinical and biochemical course, pharmacological management, and potential adverse events during pregnancy and post-partum in pregnant women with HypoPT or pseudo-HypoPT. This was a retrospective, observational, multicenter, study involving nine Italian referral centers for endocrine diseases affiliated with the Italian Society of Endocrinology and involved in “Hypoparathyroidism Working Group”. Results This study identified a cohort of 28 women (followed between 2005 and 2018) with HypoPT (n = 25, 84% postsurgical, 16% idiopathic/autoimmune) and pseudo-HypoPT (n = 3). In HypoPT women, the mean calcium carbonate dose tended to increase gradually from the first to third trimester (+ 12.6%) in pregnancy. This average increase in the third trimester was significantly greater compared to the pre-pregnancy period (p value = 0.03). However, analyzing the individual cases, in 44% the mean calcium dosage remained unchanged throughout gestation. Mean calcitriol doses tended to increase during pregnancy, with a statistically significant increase between the third trimester and the pre-pregnancy period (p value = 0.02). Nevertheless, analyzing the individual cases, in the third trimester most women with HypoPT (64%) maintained the same dosage of calcitriol compared to the first trimester. Both mean calcium carbonate and calcitriol doses tended to decrease from the third trimester to the post-partum six months. Most identified women (~ 70%) did not display maternal complications and (~ 90%) maintained mean serum albumin-corrected total calcium levels within the low-to-mid normal reference range (8.5 ± 0.8 mg/dl) during pregnancy. The main complications related to pregnancy period included: preterm birth (n = 3 HypoPT women), and history of miscarriages (n = 6 HypoPT women and n = 2 pseudo-HypoPT women). Conclusion This study shows that mean serum albumin-corrected total calcium levels were carefully monitored during pregnancy and post-pregnancy, with limited evaluation of other biochemical parameters, such as serum phosphate, 24 h urinary calcium, 25-OH vitamin D, and creatinine clearance. To avoid complications in mothers affected by (HypoPT) or (pseudo-HypoPT) and offspring, intense biochemical, clinical and pharmacological monitoring during pregnancy and breastfeeding is highly recommended.

Analysis of Clinical, Imaging, and Pathologic Features of 36 Patients with Primary Intraspinal Primitive Neuroectodermal Tumors: A Case Series and Literature Review

2021 ◽  
Author(s):  
Xuefeng Wei ◽  
Xu Zhang ◽  
Zimu Song ◽  
Feng Wang
Keyword(s):  
Literature Review ◽  
Single Case ◽  
Case Reports ◽  
Case Series ◽  
Subtotal Resection ◽  
Study Cohort ◽  
Primitive Neuroectodermal Tumors ◽  
Pathologic Features ◽  
Neuroectodermal Tumors ◽  
The Mean

Abstract Background and Study Aims Primary intraspinal primitive neuroectodermal tumors (PNETs) account for ∼0.4% of all intraspinal tumors, but information about these tumors in the medical literature is limited to single case reports. We report four cases of primary intraspinal PNETs and present a systematic literature review of the reported cases. Materials and Methods We retrospectively reviewed and analyzed the clinical data of 4 patients with primary intraspinal PNETs who underwent neurosurgical treatment at our clinic between January 2013 and January 2020, and of 32 cases reported in the literature. Results The female-to-male ratio was 2.6:1. The mean patient age was 21.42 ± 15.76 years (range: 1–60 years), and patients <36 years of age accounted for 83.30% of the study cohort. Progressive limb weakness and numbness were the chief symptoms (accounting for ∼55.6%). The mean complaint duration was 0.89 ± 0.66 months for males and 2.72 ± 3.82 months for females (p = 0.028). Epidural (41.7%) was the most common site, and thoracic (47.3%) was the most frequent location. Most PNETs were peripheral, and magnetic resonance imaging (MRI) appearance was isointense or mildly hypointense on T1-weighted images and hyperintense on T2-weighted images. Homogeneous contrast enhancement was observed. The 1-year survival rate of patients who underwent chemoradiation after total or subtotal lesion resection was better compared with patients who did not undergo chemotherapy, radiotherapy, or total or subtotal resection. The modality of treatment was associated with survival time (p = 0.007). Conclusion Primary intraspinal PNETs mainly occur in young people with a female preponderance. In patients with a rapid loss of lower limb muscle strength and large intraspinal lesions on MRI, PNETs should be considered. Surgical resection and adjuvant radio chemotherapy are key prognostic factors.

Sporadic Schwannomatosis: A Systematic Review Following the 2005 Consensus Statement

2018 ◽  
Vol 79 (05) ◽  
pp. 408-415 ◽  
Author(s):  
Jan Victor ◽  
Thibault Poujade ◽  
Nadine Hollevoet ◽  
Gregoire Chick
Keyword(s):  
Systematic Review ◽  
Consensus Statement ◽  
Case Reports ◽  
Case Series ◽  
Spinal Tumors ◽  
The Mean ◽  
Tumor Distribution ◽  
Key Aspects ◽  
Number Of Individuals

Aims To identify the frequency of reports of sporadic schwannomatosis, the types of patients affected, and the nerves affected. Patients and Methods We identified all case reports and case series that reported on patients with sporadic schwannomatosis according to established criteria. Results The initial search yielded 1,597 studies, of which 15 were included. A total of 38 of 55 individuals met the inclusion criteria. The mean age of the patients was 48 years; 41% were male. Thirty-three patients had peripheral nerve tumors, and 17 had spinal tumors. Twelve had tumors in both locations. Tumor distribution was unilateral in 25 of 30 cases (83.3%) and segmental (limited to one limb or five or fewer contiguous segments of the spine) in 28 of 38 cases (73.7%). Conclusion This systematic review quantified the number of individuals who meet the criteria for sporadic schwannomatosis and better described this population to facilitate the classification of neurofibromatosis in regard to the 2005 consensus statement. Unilateral or segmental distribution of nerve tumors are key aspects when dealing with multiple noncutaneous schwannomas without involvement of the vestibular nerve.

scholarly journals COVID-19 in HIV: a Review of Published Case Reports

2020 ◽  
Vol 2 (12) ◽  
pp. 2647-2657
Author(s):  
Zoya Morani ◽  
Saumil Patel ◽  
Sudeshna Ghosh ◽  
Falah Abu Hassan ◽  
Shriya Doreswamy ◽  
...  
Keyword(s):  
Morbid Obesity ◽  
Web Of Science ◽  
Case Reports ◽  
Case Series ◽  
Hiv Patients ◽  
High Burden ◽  
Cardiovascular Comorbidities ◽  
Life Threatening ◽  
The Mean ◽  
Immunocompromised State

AbstractPatients with COVID-19 present with a myriad of comorbidities. An immunocompromised state like HIV in patients with COVID-19 can be life-threatening. We searched PubMed/Medline, Scopus, and Web of Science for case reports and case series about COVID-19 in HIV patients. We finally reviewed 20 case reports including cases of 43 patients with HIV and COVID-19. The mean age of 43 adult patients was 51.56 ± 27.56 years (range 24–76 years). Of these, 30 were male (69.77%), 11 were female (25.58%), and 2 were transgender (4.65%). A total of 25 patients (58.14%) were above 50 years of age. The most common cardiovascular comorbidities were hypertension and hyperlipidemia (48.8%), diabetes (20.93%), and morbid obesity (11.63%). Out of 43 HIV patients with COVID-19, 6 resulted in death (13.95%). All the patients who died were elderly above 50 years and required mechanical ventilation. HIV patients infected with COVID-19 had a high mortality rate. A high burden of pre-existing comorbidities and an advanced age in these patients make them prone to disease progression and worse outcomes.

scholarly journals Mad Honey Intoxication: A Case Series of 21 Patients

2011 ◽  
Vol 2011 ◽  
pp. 1-3 ◽  
Author(s):  
Hasan Demir ◽  
Arzu Denizbasi ◽  
Ozge Onur
Keyword(s):  
Blood Pressure ◽  
Case Reports ◽  
Sinus Bradycardia ◽  
Case Series ◽  
Length Of Hospital Stay ◽  
Clinical State ◽  
Emergency Setting ◽  
Mad Honey ◽  
The Mean ◽  
Excessive Perspiration

Background. The “grayanotoxin (mad honey)” poisoning is not known commonly, there are some case series and case reports in the medical literature about it, especially in Turkey. The aim of this study was to describe the presentation of 21 natural honey intoxication cases and to review the literature. Material and Method. This study is retrospective analysis of twenty one patients who were admitted to the emergency department due to honey poisoning. Results. Median age of 21 patients was 55. The mean length of delay after consumption is 3.4 hrs. Dizziness, weakness, excessive perspiration, nausea-vomiting, and low blood pressure were the most observed symptoms. Mean pulse rate was 56/min. Mean systolic blood pressure was 102 mmHg. The mean length of hospital stay is 14.7 hrs. Patient rhytms on arrival were as follows: 10 patients were in normal sinus rhytm, 7 sinus bradycardia, 3 nodal rhytm, 1 atrial fibrillation. Atropine was given to 18 patients. None of our patients died and all were discharged home without any complication. Discussion. In the emergency setting, poisoning is a clinical state which is very hard to identify. We have to keep in mind that drugs and toxins may cause lethal dysrhythmias.

scholarly journals Hereditary leiomyomatosis and renal cell carcinoma: a case series and literature review

2021 ◽  
Vol 16 (1) ◽  
Author(s):  
Zahraa Chayed ◽  
Lone Krøldrup Kristensen ◽  
Lilian Bomme Ousager ◽  
Karina Rønlund ◽  
Anette Bygum
Keyword(s):  
Renal Cell Carcinoma ◽  
Cell Carcinoma ◽  
Renal Cell ◽  
Case Reports ◽  
Case Series ◽  
Surveillance Program ◽  
Uterine Leiomyosarcoma ◽  
Dermatological Examination ◽  
The Mean ◽  
Hereditary Leiomyomatosis

Abstract Background Hereditary leiomyomatosis and renal cell carcinoma (HLRCC) is a rare genodermatosis characterized by cutaneous leiomyoma (CLM), uterine leiomyoma (ULM) and renal cell carcinoma (RCC). Five HLRCC patients are presented with a compiled database of published HLRCC cases to increase understanding of HLRCC. Furthermore, a surveillance program is suggested. Our review is based on a PubMed search which retrieved case reports and cohort studies published before November 2019. The search yielded 97 original papers with a total of 672 HLRCC patients. Results CLMs were present in 474 patients (71.5%), developed at the mean age of 28 years. Five patients had cutaneous leiomyosarcomas. ULMs were present in 356 women (83%), while two had uterine leiomyosarcoma. ULMs were diagnosed at a mean age of 32 years, with the youngest diagnosed at age 17 years. The most common surgical treatment for ULMs was hysterectomy, performed at a mean age of 35 years, with the youngest patient being 19 years old. RCCs were present in 189 patients (34.9%), of which half had metastatic disease. The mean age of diagnosis was 36 years with the youngest patient diagnosed with RCC at the age of 11 years. Conclusion We suggest a surveillance program for HLRCC including a dermatological examination once every 2 years, annual magnetic resonance imaging starting at the age of 10 years to monitor for early RCCs, annual gynecological examinations from the age of 15 years and counseling regarding risk of hysterectomy and family planning at the age of 18 years. CLMs are often the earliest manifestation of HLRCC, which is why recognizing these lesions, performing a biopsy, and making a prompt referral to genetic counseling is important in order to diagnose HLRCC early.

scholarly journals A Retrospective Review of 25 cases of Lethal Fetal Anomalies

2020 ◽  
Vol 15 (1) ◽  
Author(s):  
Dalia FA ◽  
Hamizah I ◽  
Zalina N ◽  
Yong SL ◽  
Mokhtar A
Keyword(s):  
Gestational Age ◽  
Retrospective Review ◽  
Post Partum ◽  
Case Series ◽  
Tertiary Hospital ◽  
Prenatal Ultrasound ◽  
Age At Diagnosis ◽  
Maternal Complications ◽  
Breech Delivery ◽  
Failed Induction

Introduction: To review the gestational age at diagnosis, method of diagnosis, pregnancy outcome and maternal complications of prenatally diagnosed lethal foetal anomalies. Methods: Retrospective review of 25 women who had aborted or delivered foetuses with lethal anomalies in a tertiary hospital in 2011 based on patient medical records. Results: There were a total of 10,088 deliveries, in which 25 (0.24%) women were found to have conceived foetuses with lethal anomalies. All of them were diagnosed by prenatal ultrasound and only 7 (28.0%) had both prenatal ultrasound and genetic study done. The women’s mean age was 29.9 years old. The mean gestational age at diagnosis of lethal foetal anomalies was 25.5 weeks (SD=12.5) and mean gestational age at termination of pregnancy (TOP) or delivery was 28.5 weeks (SD=12.5). Seven (28%) women had early counseling and TOP at the gestation of < 22 weeks. Beyond 22 weeks of gestation, eight (32%) women had TOP and ten (40%) women had spontaneous delivery. Twenty (80%) women delivered or aborted vaginally, three (12%) women with assisted breech delivery and two (8%) women with abdominal delivery which were performed due to transverse foetal lie in labour and a failed induction, leading to emergency hysterotomy complicated by hysterectomy due to intraoperative finding of ruptured uterus. Overall, the associated post-partum adverse events included post-partum haemorrhage (12%), retained placenta (12%), blood transfusion (8%), uterine rupture (4%) and endometritis (4%). Mean duration of hospital stay was 6.6 days (SD 3.7 days). Conclusion: Late diagnosis of lethal foetal anomalies leads to various maternal morbidities, in this case series , which could have been prevented if they were diagnosed and terminated at early trimester. A new direction is needed in our local practice.

Abstract 52: Case Reports of Short-Term Outcome Following Mechanical Thrombectomy for Childhood Stroke May Be Overly Optimistic

Stroke ◽  
2020 ◽  
Vol 51 (Suppl_1) ◽  
Author(s):  
Catherine Amlie-Lefond ◽  
Dwight Barry ◽  
Keyword(s):  
Mechanical Thrombectomy ◽  
Care Delivery ◽  
Case Reports ◽  
Case Series ◽  
Arterial Ischemic Stroke ◽  
Short Term ◽  
Term Outcome ◽  
The Mean ◽  
Meta Analyses ◽  
Iv Tpa

Background: Meta-analyses of case reports and case series of mechanical thrombectomy in childhood arterial ischemic stroke (AIS) suggest that mechanical thrombectomy is associated with good short-term outcomes in approximately three-quarters of children. Methods: Retrospective data on 38 children treated between 2010-2019 with mechanical thrombectomy for stroke were collected from former Thrombolysis in Pediatric Stroke (TIPS) sites. To minimize reporting bias, sites were required to report all patients ≤ 17 years treated with mechanical thrombectomy either at the study site or at an outside hospital prior to transfer. Short-term outcomes, defined as change in NIHSS prior to and following mechanical thrombectomy (MT), were compared with 85 children from case reports/case series published between 2010-2019. Results: The mean age of children from former TIPS sites was 11.9 years, 50% male, 34% treated with IV tPA prior to thrombectomy. The mean age of published cases was 10.4 years, 65% males, 19% treated with IV tPA prior to thrombectomy. Case reports had a wider distribution of before-MT NIHSS scores, and a steeper decrease in distributions of after-MT NIHSS scores than those from study sites within 24 hours of MT (Figure 1A; medians: 15 to 4 in published cases, 12 to 8 in cases from sites) and at discharge (Figure 1B; medians: 15 to 3 in published cases, 12 to 5 in cases from sites). Conclusion: Patients reported to former TIPS sites had discernably lower rates of good short-term outcomes compared with published case reports and case series, suggesting publication bias. Capturing key metrics of safety and effectiveness of MT in children with AIS, including variations in care delivery in actual practice, will be critical in the creation of evidence-based guidelines for acute management of pediatric AIS.

Valproic Acid in the Management of Delirium

2021 ◽  
pp. 104990912110383
Author(s):  
Carlos Fernandez Cuartas ◽  
Mellar Davis
Keyword(s):  
Valproic Acid ◽  
English Language ◽  
Retrospective Studies ◽  
Case Reports ◽  
Controlled Trial ◽  
Case Series ◽  
Open Label ◽  
Mesh Terms ◽  
Starting Dose ◽  
The Mean

Context: Antipsychotics and benzodiazepines do not improve delirium. Valproic acid (VPA) has been used recently to treat agitation in delirium. Objectives: To review the evidence for VPA in the management of Delirium. Methods: Systematic review. English language, age 19 and above, from 1946 to January 12, 2021. Mesh Terms: “Valproic acid”, “valproate”, “sodium valproate”, “delirium”, “acute mania with delirium” in PubMed and Ovid. Exclusion: Studies of VPA used for diagnoses other than delirium. Results: 21 abstracts were identified and 10 studies were included in the review (252 patients): One prospective open label study (n: 7), 2 case series (n: 22), 4 retrospective studies (n: 219) and 3 case reports (n: 4). No randomized controlled trial (RCT) evaluates the effect of VPA in delirium. 237/250 (94.8%) patients were in the ICU. Mean age was 59.7 (27-87). 153/204 (74%) were male. The mean starting dose was 733 mg/day in 148 patients and the mean dose at follow up was 1061 mg/day in 205 patients. CAM ICU was used to diagnose delirium in 6 reviews. Delirium improved in case series in 19/22 patients. Delirium improved in retrospective studies at day 3 compared to day 1. VPA levels were not consistently reported. Hyperammonemia (12-19%) and thrombocytopenia (9-13%) were the most common side effects. No deaths were attributed to VPA. Conclusion: VPA is being used more frequently for delirium. The evidence is limited to retrospective studies and case series. There is a need for RCT to evaluate the effect of VPA in delirium compared to other alternatives and placebo.

625 EFFICACY OF MYOTOMY IN PATIENTS WITH SIGMOID MEGAESOPHAGUS: A SYSTEMATIC REVIEW AND META-ANALYSIS

2021 ◽  
Vol 34 (Supplement_1) ◽  
Author(s):  
Marina Orlandini ◽  
Maria Carolina Serafim ◽  
Letícia Datrino ◽  
Clara Santos ◽  
Luca Tristão ◽  
...  
Keyword(s):  
Systematic Review ◽  
Heller Myotomy ◽  
Selection Process ◽  
Case Reports ◽  
Meta Analysis ◽  
Case Series ◽  
Cochrane Library ◽  
Cross Sectional ◽  
Manual Search ◽  
The Mean

Abstract   Megaesophagus progress to sigmoid megaesophagus (SM) in 10–15% of patients, presenting tortuosity and sigmoid colon aspect. Esophagectomy is the choice treatment but is associated with high complications and mortality rates. To avoid the esophagectomy inherent morbidity, several authors recommend Heller myotomy (HM) with pull-down technique for SM, mainly for patients with comorbidities and the elderly. This systematic review and meta-analysis is the first to analyze the effectiveness of HM for treating SM. Methods A systematic review was conducted in PubMed, Embase, Cochrane Library Central, Lilacs (BVS), and manual search of references. Inclusion criteria were: a) clinical trials, cohort studies, case series; b) patients with SM and esophageal diameter ≥ 6 cm; and c) patients undergoing primary myotomy. The exclusion criteria were: a) reviews, case reports, cross-sectional studies, editorials, letters, congress abstracts, full-text unavailability; b) animal studies, c) previous surgical treatment for achalasia; and d) pediatric studies. There were no restrictions on language or date of publication, and no filters were applied for the selection process. Random model and a 95% confidence interval (CI) were used. Results Sixteen articles were selected, encompassing 231 patients. The mean age ranged from 36 to 61 years old, and the mean follow-up ranged from 16 to 109 months. The analyzed outcomes include mortality, complications (pneumonia, pneumothorax, gastroesophageal reflux), need for reintervention (remyotomy, dilation and esophagectomy), and results classified as ‘good’ and ‘excellent’. Mortality rate was 0.035 (CI: 0.017–0.07; p &lt; 0.01). Complications rate was 0.08 (CI: 0.04–0.153; p = 0.01). Need for retreatment rate was 0.161 (CI: 0.053–0.399; p &lt; 0.01). Probability of good or excellent outcomes after myotomy was 0.762 (CI: 0.693–0.819; p &lt; 0,01). Conclusion Heller myotomy is an option for avoiding esophagectomy in achalasia, with a low morbimortality rate and good results. It is effective for most patients but will fail in a minority of patients and demand retreatment, be it a remyotomy, endoscopic treatment or esophagectomy.

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