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European Archives of... Jul 2024To review possible risk factors for permanent delayed-onset, progressive sensorineural hearing loss (SNHL) in the paediatric population to recommend follow-up protocols...
PURPOSE
To review possible risk factors for permanent delayed-onset, progressive sensorineural hearing loss (SNHL) in the paediatric population to recommend follow-up protocols for early detection.
METHODS
PRISMA-compliant systematic review was performed, including observational studies on the paediatric population up to 16 years old who have passed the newborn hearing screening programme (NHSP), investigating the development of late-onset, progressive SNHL. Electronic searches were performed through Medline, Embase, Cochrane, and Emcare.
RESULTS
37 studies were included. 21 showed an association between late-onset SNHL and congenital cytomegalovirus (cCMV) infection (age at hearing loss diagnosis 0.75 to 204 months, mean 45.6 ± 43.9), while 16 between late-onset SNHL and other congenital or perinatal factors, namely Neonatal Intensive Care Unit (NICU) stay, prematurity, neonatal respiratory failure, mechanical ventilation, extracorporeal membrane oxygenation (ECMO) support, hypocapnia, hypoxia, alkalosis, seizure activity, congenital diaphragmatic hernia (CDH), inner ear malformation, and gene mutations (age at hearing loss diagnosis 2.5 to 156 months, mean 38.7 ± 40.7).
CONCLUSIONS
cCMV infection may cause late-onset SNHL, which can be missed on standard NHSP. There is, therefore, evidence to support universal screening programmes to enable detection in even asymptomatic neonates. Ongoing audiological follow-up for all children with cCMV is advisable, to enable timely treatment. In the paediatric population presenting conditions such as NICU stay > 5 days, prematurity ≤ 34 weeks gestation, severe neonatal respiratory failure, mechanical ventilation, ECMO support, and CDH surgery, an audiological follow-up from 3 months of age up to at least 3-4 years of age, and at least annually, should be recommended.
Topics: Adolescent; Child; Child, Preschool; Humans; Infant; Infant, Newborn; Age of Onset; Cytomegalovirus Infections; Disease Progression; Hearing Loss, Sensorineural; Neonatal Screening; Risk Factors
PubMed: 38411671
DOI: 10.1007/s00405-024-08527-x -
Journal of Medical Case Reports Feb 2024Hydroxychloroquine and azathioprine have been routinely used to control and treat primary and secondary Sjögren's syndrome, which potentially triggered some overdoses...
INTRODUCTION
Hydroxychloroquine and azathioprine have been routinely used to control and treat primary and secondary Sjögren's syndrome, which potentially triggered some overdoses by these drugs. Toxicity from hydroxychloroquine and azathioprine manifests in the form of cardiac conduction abnormalities, nausea, vomiting, and muscle weakness. Recognizing these unique drug overdoses and management of these toxicities is important. This case report aims to expand our current understanding of these drug overdoses and their management and also underscores the importance of anticipating and identifying fewer common complications, such as hypocalcemia.
CASE REPORT
A 34-year-old Persian woman with a history of Sjögren's syndrome presented to the emergency department 3.5-4 hours after an intentional overdose of hydroxychloroquine and azathioprine and severe hypotension and loss of consciousness. Although the patient was regularly taking other medications, such as fluoxetine, naproxen, and prednisolone, she explicitly clarified that these were not the substances involved in her overdose. Early investigations showed hypokalemia (2.4 mEq/L), hypocalcemia (7.5 mg/dL), and hypoglycemia (65 mg/dL). She was also diagnosed with metabolic acidosis and respiratory alkalosis. The electrocardiogram showed changes in favor of hypokalemia; other lab tests were run on the patient. Supportive treatments were applied, including rapid intravenous fluid dextrose 5%, normal saline, potassium chloride 30 mEq, and calcium gluconate 100 mg. The patient was managed and monitored overnight in the emergency room and recovered without residual side effects.
CONCLUSION
Hydroxychloroquine and azathioprine toxicity are considered rare, but it is likely to increase in frequency given the prevalence and increase in autoimmune diseases and the increasing usage of these drugs in treating such diseases. We found hypocalcemia as the presentation to this patient, which needs further investigation into the probable mechanism. Clinicians need to consider the unique effects of hydroxychloroquine and azathioprine poisoning and initiate appropriate emergency interventions to improve the outcomes in similar patients.
Topics: Female; Humans; Adult; Hydroxychloroquine; Azathioprine; Hypocalcemia; Sjogren's Syndrome; Hypokalemia; Drug Overdose; Drug-Related Side Effects and Adverse Reactions
PubMed: 38409169
DOI: 10.1186/s13256-024-04390-w -
Case Reports in Endocrinology 2024The triad of hypercalcemia, metabolic alkalosis, and acute kidney injury associated with ingesting high doses of calcium and absorbable bases characterizes the...
INTRODUCTION
The triad of hypercalcemia, metabolic alkalosis, and acute kidney injury associated with ingesting high doses of calcium and absorbable bases characterizes the calcium-alkali syndrome. . We report the case of a patient with postthyroidectomy hypoparathyroidism 15 years ago due to differentiated thyroid cancer who presented with severe hypercalcemia. He had adequate control of calcemia for many years on treatment with calcitriol and calcium carbonate and hypertension treated with amlodipine, losartan, and hydrochlorothiazide. After a period of loss to follow-up, he suddenly presents with severe hypercalcemia, metabolic alkalosis, and loss of renal function. Upon hydration and withdrawal of calcitriol and calcium replacements, hypercalcemia resolved. The etiological investigation identified no granulomatous or neoplastic diseases, but an aldosterone-producing adrenal incidentaloma was found. The cause of hypercalcemia in this patient was calcium-alkali syndrome due to calcium carbonate replacement potentiated by hydrochlorothiazide and primary aldosteronism. Six months after the hospitalization and suspension of calcium and vitamin D, the patient returned to hypocalcemia, reinforcing the diagnosis.
CONCLUSION
Although seldom described, the calcium-alkali syndrome is an expected complication for individuals with postoperative hypoparathyroidism, as they require lifelong calcium and vitamin D supplementation. This case also shows the importance of hydrochlorothiazide use and primary aldosteronism as possible triggers of life-threatening hypercalcemia.
PubMed: 38404648
DOI: 10.1155/2024/3067354 -
Advances in Medical Sciences Mar 2024Hypocalcaemia predicts coronavirus disease 2019 (COVID-19) severity and mortality. We hypothesized an association between respiratory alkalosis secondary to hypoxia and...
BACKGROUND
Hypocalcaemia predicts coronavirus disease 2019 (COVID-19) severity and mortality. We hypothesized an association between respiratory alkalosis secondary to hypoxia and low ionised calcium (iCa) concentration in patients with COVID-19.
METHODS
Arterial blood gas samples taken from January 2019 to March 2021 were retrospectively matched with infection status. Principal components regression was undertaken to determine the correlation between pH, partial pressure arterial oxygen (PaO), partial pressure arterial carbon dioxide (PaCO), and iCa.
RESULTS
We included 4056 patients (300 COVID-19 detected, 19 influenza detected), corresponding to 5960 arterial blood samples. The COVID-19 detected group had a statistically significantly lower iCa, PaO and PaCO, and more alkalotic pH than infection-free groups. The influenza group had a lower iCa and PaCO, higher PaO, and a more alkalotic pH than infection-free groups, but these differences were non-significant. Principal components regression revealed that pH, PaCO, and PaO explain just 2.72 % of the variance in iCa. An increase in pH by 1 unit was associated with an iCa reduction of 0.141 mmol/L (p < 0.0001).
CONCLUSION
Reduction in iCa concentration in patients with COVID-19 is not associated with pH derangement. Influenza infection was associated with a minor reduction in iCa in our small sample, a hitherto unreported finding, although statistical significance was not demonstrated.
Topics: Humans; COVID-19; Hydrogen-Ion Concentration; Female; Male; Middle Aged; Retrospective Studies; Calcium; Blood Gas Analysis; SARS-CoV-2; Aged; Oxygen; Hypocalcemia; Carbon Dioxide; Adult
PubMed: 38364757
DOI: 10.1016/j.advms.2024.02.001 -
BMJ Case Reports Feb 2024Bartter syndrome (BS) is a rare genetic tubulopathy affecting the loop of Henle leading to salt wasting. It is commonly seen in utero or in the early neonatal period....
Bartter syndrome (BS) is a rare genetic tubulopathy affecting the loop of Henle leading to salt wasting. It is commonly seen in utero or in the early neonatal period. Rare cases of acquired BS are reported in association with infections like tuberculosis, granulomatous conditions like sarcoidosis, autoimmune diseases and drugs. The mainstay of management includes potassium, calcium and magnesium supplementation. We report the case of a woman in her 50s with a history of type 2 diabetes mellitus for the last 10 years, who presented with diabetic foot ulcers and generalised weakness with ECG changes suggestive of hypokalaemia. She had severe hypokalaemia with high urine potassium excretion and hypochloraemic metabolic alkalosis. She poorly responded to intravenously administered potassium supplements and had persistent hypokalaemia. On further evaluation of the persistent hypokalaemia, a diagnosis of idiopathic Bartter-like phenotype was made. She responded well to tablet indomethacin and is presently asymptomatic and is being maintained on tablet indomethacin after 6 months of follow-up.
Topics: Infant, Newborn; Female; Humans; Bartter Syndrome; Hypokalemia; Diabetes Mellitus, Type 2; Phenotype; Potassium; Indomethacin; Tablets
PubMed: 38350705
DOI: 10.1136/bcr-2023-257953 -
Gastric outlet obstruction due to an intragastric balloon in a patient returning from the Caribbean.Clinical Case Reports Feb 2024Gastric outlet obstruction can be a dangerous complication of intragastric balloons, as it can result in severe metabolic alkalosis. As weight loss procedures and...
KEY CLINICAL MESSAGE
Gastric outlet obstruction can be a dangerous complication of intragastric balloons, as it can result in severe metabolic alkalosis. As weight loss procedures and medical tourism become more popular, physicians should have a high index of suspicion for complications of invasive procedures, particularly in returning travelers.
ABSTRACT
Intragastric balloons for weight loss have decreased in frequency in the United States. However, they are still frequent in low- and middle-income countries. Severe complications occur in less than 3% of patients who undergo this procedure. Herein, we present a case of gastric outlet obstruction, severe metabolic alkalosis, and refeeding syndrome in a patient returning from the Dominican Republic. She presented with 2 weeks of emesis and obstipation, followed by a pre-syncope and altered mental status. An intragastric mass was observed on computerized tomography, which was characterized as an intragastric balloon and retrieved endoscopically. All metabolic derangements were corrected, and the patient improved without sequelae. As weight loss procedures and medical tourism become more popular, physicians should have a high index of suspicion for complications of invasive procedures, particularly in returning travelers.
PubMed: 38333653
DOI: 10.1002/ccr3.8509 -
Pediatric Critical Care Medicine : a... May 2024Mechanical ventilation (MV) is pervasive among critically ill children. We sought to validate a computerized physiologic equation to predict minute ventilation...
OBJECTIVES
Mechanical ventilation (MV) is pervasive among critically ill children. We sought to validate a computerized physiologic equation to predict minute ventilation requirements in children and test its performance against clinician actions in an in silico trial.
DESIGN
Retrospective, electronic medical record linkage, cohort study.
SETTING
Quaternary PICU.
PATIENTS
Patients undergoing invasive MV, serial arterial blood gas (ABG) analysis within 1-6 hours, and pharmacologic neuromuscular blockade (NMB).
MEASUREMENTS AND MAIN RESULTS
ABG values were filtered to those occurring during periods of NMB. Simultaneous ABG and minute ventilation data were linked to predict serial Pa co2 and pH values using previously published physiologic equations. There were 15,121 included ABGs across 500 encounters among 484 patients, with a median (interquartile range [IQR]) of 20 (10-43) ABGs per encounter at a duration of 3.6 (2.1-4.2) hours. The median (IQR) Pa co2 prediction error was 0.00 (-3.07 to 3.00) mm Hg. In Bland-Altman analysis, the mean error was -0.10 mm Hg (95% CI, -0.21 to 0.01 mm Hg). A nested, in silico trial of ABGs meeting criteria for weaning (respiratory alkalosis) or escalation (respiratory acidosis), compared the performance of recommended ventilator changes versus clinician decisions. There were 1,499 of 15,121 ABGs (9.9%) among 278 of 644 (43.2%) encounters included in the trial. Calculated predictions were favorable to clinician actions in 1124 of 1499 ABGs (75.0%), equivalent to clinician choices in 26 of 1499 ABGs (1.7%), and worse than clinician decisions in 349 of 1499 ABGs (23.3%). Calculated recommendations were favorable to clinician decisions in sensitivity analyses limiting respiratory rate, analyzing only when clinicians made changes, excluding asthma, and excluding acute respiratory distress syndrome.
CONCLUSIONS
A computerized equation to predict minute ventilation requirements outperformed clinicians' ventilator adjustments in 75% of ABGs from critically ill children in this retrospective analysis. Prospective validation studies are needed.
Topics: Humans; Retrospective Studies; Critical Illness; Respiration, Artificial; Female; Male; Child, Preschool; Child; Infant; Intensive Care Units, Pediatric; Blood Gas Analysis; Adolescent; Neuromuscular Blockade; Carbon Dioxide
PubMed: 38329377
DOI: 10.1097/PCC.0000000000003462 -
Radiology Case Reports Apr 2024Typhoid fever, caused by Salmonella Typhi, is a severe bacterial infection prevalent in developing countries, and can result in life-threatening complications if...
Typhoid fever, caused by Salmonella Typhi, is a severe bacterial infection prevalent in developing countries, and can result in life-threatening complications if untreated. Nutcracker Syndrome is a rare vascular disorder involving compression of the left renal vein between the aorta and the superior mesenteric artery. It can lead to various symptoms and poses diagnostic and management challenges. We present a case study of a patient diagnosed with typhoid fever in a Unit of Critical Emergency Care. Coincidentally, the evaluation through CT-scan revealed the presence of Nutcracker Syndrome. This report underscores the incidental discovery of Nutcracker Syndrome during the assessment of a patient with typhoid fever in a critical emergency care setting.
PubMed: 38327558
DOI: 10.1016/j.radcr.2024.01.034 -
Zhonghua Xin Xue Guan Bing Za Zhi Feb 2024To explore the clinical manifestations and genotype of an infant with hyperuricemia, pulmonary hypertension, renal failure in infancy, and alkalosis syndrome (HUPRAS).... (Review)
Review
To explore the clinical manifestations and genotype of an infant with hyperuricemia, pulmonary hypertension, renal failure in infancy, and alkalosis syndrome (HUPRAS). Clinical data of the patient were collected. Peripheral blood samples from the patient and his parents were acquainted for whole exome sequencing. The filtrated variants were verified by Sanger sequencing. The pathogenicity of the variants was predicted by bioinformatic tools. The patient is a male infant of 6 months old, carrying two missense variants in the SARS2 allele: a paternal inherited c.1205G>A (p. Arg402His) and a maternal inherited c.680G>A (p. Arg227Gln). The two variants were in extremely low population frequencies. The pathogenetic prediction tools categorized them as deleterious. Arg402 and Arg227 were highly conserved in evolution. The variants led to changes in the hydrogen bonds and hydrophobicity of seryl-tRNA synthetase encoded by SARS2. c.1205G>A (p. Arg402His) and c.680G>A (p. Arg227Gln) are the possible causative variants of the HUPRA syndrome.
Topics: Humans; Infant; Male; Mutation; Hypertension, Pulmonary; COVID-19; Mutation, Missense; Genotype; Kearns-Sayre Syndrome; Mitochondrial Myopathies
PubMed: 38326069
DOI: 10.3760/cma.j.cn112148-20231009-00268