OpenMed/multicare-articles huggingface dataset:
BK virus-associated hemorrhagic cystitis in pediatric stem cell transplantation: a case report and scoping review
Authors: Julia E Moss, William J Muller
Journal: Front Pediatr (2024)
Keywords: bk polyomavirus, cystitis, hematuria, stem cell transplantation, viral infection

Introduction:
BK virus-associated hemorrhagic cystitis (BK-HC) is a debilitating and poorly understood complication of hematopoietic stem cell transplantation (SCT). Hematuria, dysuria, and other symptoms associated with BK-HC are common in the immediate post-SCT period, making BK-HC difficult to distinguish from other conditions presenting with these symptoms. Despite published criteria for diagnosis, the degree to which these criteria are consistently applied to either clinical diagnosis or to studies informing BK-HC management is unclear. We present a case of BK-HC in a pediatric SCT recipient, and discuss the challenges associated with treatment in the absence of rigorous data to inform clinical management.
Methods:
We reviewed all cases of BK viruria at our center in patients undergoing SCT between January 2015 and December 2019. We then performed a scoping review of publications in PubMed addressing BK-HC, specifically focusing on how BK-HC was defined. Publications using the keywords "BK polyomavirus" and "hemorrhagic cystitis" were included if they involved a clinical study of SCT recipients and a full-text article was available in English. Case reports were excluded. Analysis focused on whether BK-HC was explicitly defined and whether the definition incorporated elements of diagnostic criteria published by European Conference on Infections in Leukemia (ECIL).
Results:
A total of 30 studies published between January 2018 and 30 June 2021 met criteria for review, including 4 clinical trials, 7 prospective observational studies, and 19 retrospective observational studies. Fifteen of these studies included pediatric patients (7 pediatric only, 8 combined adult and pediatric). Of the 30 publications, 19 included a definition of either BK-HC or BK cystitis, with only five using ECIL criteria, all of which were observational studies. Multiple interventions are described for treatment of BK-HC, including cidofovir, leflunomide, quinolones, hyperbaric oxygen, keratinocyte growth factor, and BK-specific cytotoxic T lymphocytes. However, evidence to support efficacy for any of these interventions is lacking.
Discussion:
Although BK-HC is a well-known complication of SCT, evidence to support available treatment options is limited. Well-controlled studies that incorporate clear diagnostic criteria are needed to better define the risk factors, natural history, and ideal interventions.
The Importance of Serial POCUS Exams - Dual Pathologies in Play
Authors: Rahul Nair, Jonathan Zuo, Ariel L Shiloh
Journal: POCUS J (2023)
Keywords: butterfly iq, cardiac pocus, pocus, pericardial effusion, takotsubo, tamponade, assessment case
MeSH terms: Case Reports

Serial point of care ultrasound (POCUS) exams are essential to assess acute pericardial effusions which can rapidly evolve into cardiac tamponade. A typical presentation includes dyspnea, tachycardia, and chest pain. Importantly, serial cardiac exams in such high-risk patients can detect other concurrent pathologies. We present an unusual case of a patient who initially presented with an acute circumferential pericardial effusion and upon serial POCUS exams developed an unexpected Takotsubo cardiomyopathy in the setting of cardiac tamponade.
Supraglottic myxoedema successfully treated orally
Authors: Yu Arai, Satoru Okada, Taiju Miyagami, Narumi Sue, Chisato Kainaga
Journal: Endocrinol Diabetes Metab Case Rep (2024)

Summary:
Myxoedema coma is a severe form of hypothyroidism with multiple organ dysfunction, characterised by an altered state of consciousness and hypothermia. Intravenous thyroid hormone replacement therapy is the preferred treatment for myxoedema. The mortality rate associated with this disease is high, and early detection and intervention are essential. Supraglottal myxoedema is a rare form of periglottic oedema and can be fatal. A previously healthy 66-year-old man presented with impaired consciousness, hypothermia, and nonpitting oedema. Blood tests revealed the presence of hypothyroidism and respiratory acidosis. He was intubated for type 2 respiratory failure; however, severe laryngeal oedema made the procedure difficult to perform. Oral thyroid hormone therapy was initiated under the diagnosis of myxoedema coma. Tracheostomy was performed because of prolonged type 2 respiratory failure and laryngeal oedema. Three weeks after admission, the patient was weaned off the ventilator. Approximately a week later, laryngeal oedema improved, and the tracheostomy tube was removed. The patient was discharged and remained stable for 3 months. This case report describes a patient with comatose myxoedema and supraglottic oedema who was successfully treated with oral medication alone. This case shows that supraglottic oedema should be considered even in the absence of wheezing or other signs of upper airway obstruction.
Learning points:
Myxoedema coma is a differential diagnosis of respiratory acidosis. In myxoedematous coma, the possibility of difficult intubation due to supraglottic oedema should be considered. Tracheostomy should be considered for supraglottic myxoedema, which often results in prolonged ventilator use. Supraglottic myxoedema can be treated with oral medications.
Malignant transformation of desmoplastic infantile tumors in a non-infant: a case report
Authors: Yan Yang, Bin Tian, Xuzhu Chen, Xin Liu, Shiguang Li
Journal: Front Oncol (2023)
Keywords: case report, computed tomography, desmoplastic infantile tumors, magnetic resonance imaging, malignant transformation
MeSH terms: Case Reports

Desmoplastic infantile tumors (DITs) are rare benign intracranial tumors in infants with benign biological behavior and rare malignant transformation characteristics. We present a DIT case that underwent malignant transformation and metastasis. A 6-year-old girl presented with DITs and underwent surgical resection. 12 years later, the tumor recurred and underwent surgical resection again. The pathology report confirmed the lesion to be a glioblastoma multiforme. She received adjuvant treatment. A year after the surgical operation of the lesions, she had intraspinal metastasis and underwent surgical resection again. Multiple spinal cord metastases were subsequently identified in the patient. The patient's condition exhibited severe deterioration during the follow-up period. This case report focuses on the occurrence of DITs and their potential malignant transformation, as assessed through computed tomography and magnetic resonance imaging.
Inflammatory endobronchial polyps unleashing recurrent pneumothorax: A case report
Authors: Ummi Nadira Daut, Muhammad Halmii Faisal Thena, Tan Hui-Xin, Mona Zaria Nasaruddin, Jamalul Azizi Abdul Rahaman
Journal: Respirol Case Rep (2024)
Keywords: debulking, endobronchial inflammatory polyp, pneumothorax, rigid bronchoscopy
MeSH terms: Case Reports

Inflammatory endobronchial polyps (IEPs) are rare benign lesions that originate from the bronchial mucosa. While pneumothorax is a well-known complication of various pulmonary conditions, its association with IEPs is exceedingly uncommon and poorly understood. This case report presents a unique and explosive encounter of a patient with an inflammatory endobronchial polyp who experienced a pneumothorax, shedding light on the clinical presentation, diagnostic challenges, and management strategies for this rare entity.
Edward O. Bixler, PhD: from the Apollo project and chimpanzees to sleep epidemiology
Authors: Julio Fernandez-Mendoza, Susan L Calhoun, Edward O Bixler
Journal: Sleep Adv (2024)
Keywords: cardiovascular risk, cohort studies, epidemiology, insomnia, pharmacology, psychiatry, sedative-hypnotics, sleep apnea

What an honor to write about Dr. Edward O. Bixler's contributions to the sleep field. In 1967, Dr. Bixler published a case report on a chimpanzee with implanted brain electrodes while working at an Air Force base in New Mexico. A few years later, in 1971, he published on the sleep effects of flurazepam in individuals with insomnia together with Dr. Anthony Kales, data that he had collected when the Sleep Research & Treatment Center (SRTC) was housed at the University of California Los Angeles. Dr. Bixler, a meticulous scientist, learned from Dr. Kales, a devoted clinician, to study "the whole patient, and all aspects of sleep," a legacy that continued when the SRTC moved to Penn State in Hershey. Indeed, Dr. Bixler's tenure at Penn State from 1971 until 2019 kept the science of the SRTC focused on that premise and helped translate scientific evidence into clinical care. He not only contributed early to the pharmacology of sleep and the effects of hypnotics, but he was also a pioneer in "sleep epidemiology." His "Prevalence of sleep disorders in the Los Angeles metropolitan area" study of 1979 was the first rigorous epidemiological study on sleep disturbances. Starting in 1990, he established the Penn State Adult Cohort to estimate the prevalence and natural history of sleep-disordered breathing and other sleep disorders in adults. Inspired by life-course epidemiology, he established in 2001 the Penn State Child Cohort to estimate the same phenomena in children. This Living Legend paper captures and highlights Dr. Bixler's enduring legacy to sleep science.
Pituitary stalk interruption syndrome and liver cirrhosis associated with diabetes and an inactivating KCNJ11 gene mutation: a case report and literature review
Authors: Zhaoxiang Liu, Wenhui Zhao, Chenxiang Cao, Yanlei Wang, Luqi Xiao, Xiaojing Wang, Chenxi Jin, Jianzhong Xiao
Journal: Front Endocrinol (Lausanne) (2023)
Keywords: kcnj11, case report, diabetes mellitus, liver cirrhosis, pituitary stalk interruption syndrome
MeSH terms: Diabetes Mellitus / genetics, Diabetes Mellitus / pathology, Non-alcoholic Fatty Liver Disease / pathology, Pituitary Diseases / complications, Review, Case Reports, Research Support, Non-U.S. Gov't, Adult, Female, Humans, Liver Cirrhosis / complications, Liver Cirrhosis / genetics, Mutation, Pituitary Gland / pathology, Combined Pituitary Hormone Deficiency

Background:
Pituitary stalk interruption syndrome (PSIS) is a congenital disease commonly found in patients with combined pituitary hormone deficiency (CPHD). Most PSIS patients manifest growth retardation and delayed puberty. We report a rare case of PSIS with tall stature, liver cirrhosis and diabetes, possibly caused by an inactivating KCNJ11 gene mutation.
Case presentation:
A 37-year-old female patient initially presented with liver cirrhosis and diabetes, without any secondary sexual characteristics. Endocrine investigation indicated CPHD. Small anterior pituitary, invisible pituitary stalk and no eutopic posterior lobe hypersignal in the sella turcica viewed in magnetic resonance imaging (MRI) confirmed the diagnosis of PSIS. Despite receiving no growth hormone or sex hormone therapy, she reached a final height of 186 cm. Liver histopathology revealed nonalcoholic fatty cirrhosis. Genetic testing identified a heterozygous p.Arg301Cys mutation in the KCNJ11 gene.
Conclusion:
This is a rare case of PSIS with liver cirrhosis and diabetes associated with an inactivating KCNJ11 gene mutation. It's supposed that early hyperinsulinism caused by the KCNJ11 gene mutation, as well as delayed epiphyseal closure due to estrogen deficiency, contributed to the patient's exceptionally tall stature. Untreated growth hormone deficiency (GHD) resulted in increased visceral fat, leading to nonalcoholic fatty liver disease (NAFLD) and cirrhosis. The decline in β cell function with age, combined with NAFLD, may have played a role in the development of diabetes.
Progressive Neuropsychiatric Symptoms Following Recurrent COVID-19 Infections in a Previously Healthy Adolescent
Authors: Christopher Shawl, R Hunter Clark, Matthew T Edwards, Casey Berson, Megan Zappitelli
Journal: Case Rep Psychiatry (2023)
MeSH terms: Case Reports

This is the almost 2-year-long course of a 16-year-old male without significant psychiatry history who abruptly developed symptoms of obsessive-compulsive disorder (OCD) and psychosis following a confirmed coronavirus disease 2019 (COVID-19) infection. His symptoms worsened following a confirmed reinfection with COVID-19. He responded poorly to treatment with selective serotonin reuptake inhibitors, antipsychotics, and benzodiazepines. This case highlights an emerging phenomenon of post-COVID-19 neuropsychiatric sequelae and presents a complicated diagnostic and treatment challenge. The differential for this patient was explored and outlined in detail, and the medical workup recommendations for new-onset mental status changes were reviewed as they pertain to the patient's assessment and treatment course. While there are several case reports of adolescents with abrupt-onset OCD and psychosis symptoms following COVID-19 infections, none of these reports include worsening of symptoms following reinfection, and few reports follow patients beyond initial hospitalization and treatment.
Exploring the Potential of Lateral Wedge Insoles in Alleviating Bone Marrow Lesions in End-Stage Knee Osteoarthritis: A Preliminary Case Report
Authors: So Tanaka, Takanori Taniguchi, Masami Tokunaga, Takaaki Yoshimoto, Tomohiko Nishigami
Journal: Cureus (2024)
Keywords: bone marrow lesion, co-contraction ratio, gait, knee osteoarthritis/ koa, lateral wedge insole, varus thrust
MeSH terms: Case Reports

The efficacy of lateral wedge insoles (LWIs) in patients with end-stage knee osteoarthritis (OA) is unclear. A 43-year-old male underwent two anterior cruciate ligament reconstructions in his right knee and was later diagnosed with end-stage knee OA. An LWI combining arch support with a lateral heel wedge was fabricated for this patient and used over 12 months. As a result, after 12 months, the bone marrow lesion (BML), as measured by the magnetic resonance imaging Osteoarthritis Knee Score (MOAKS), was downgraded from grade 2 to grade 1. The use of LWI in a patient with end-stage knee OA showed lower co-contraction ratios in knee muscles even after 12 months. The results provide preliminary evidence suggesting the use of LWI in patients with end-stage knee OA has potential benefits for reducing BML.
Gastroblastoma in a 5-year-old child: a case report and literature review
Authors: Jizhen Feng, Chunxiang Ling, Yingjie Xue, Jiamei Li
Journal: Front Oncol (2023)
Keywords: biphasic tumor, case report, gastroblastoma, imaging, stomach
MeSH terms: Case Reports

Background:
Gastroblastoma is an extremely rare stomach tumor with a biphasic cell morphology of epithelioid and spindle cells. Due to the low incidence rate and the lack of specific clinical characteristics, it is easy to misdiagnose. Detailed imaging analysis is also unavailable. At present, we reported a case of gastroblastoma to analyze its clinical and imaging characteristics. In addition, we reviewed the imaging findings, current diagnosis, treatment, and outcome of gastroblastoma.
Case presentation:
A 5-year-old girl was admitted to our hospital with upper abdominal pain and melena. Endoscopic examination showed a protuberant submucosal mass on the greater curvature of the gastric body. Abdominal ultrasonography and an abdominal enhanced computed tomography further confirmed the mass. The patient was pathologically diagnosed with gastroblastoma after radical surgery in February 2021.
Conclusion:
We described a rare case of gastroblastoma and may provide a new perspective on imaging diagnosis, treatment, and outcome of this tumor. Gastroblastoma tends to occur in male patients, typically affects young people, and has low malignant potential and a low rate of recurrence and metastasis. Gastroblastoma usually arises in the gastric muscularis propria with hypoecogenic and submucosal characteristics in ultrasound examination and significant enhancement in computed tomography (CT) scan. Surgical resection and regular follow-up after surgery are the main management of the disease. Clinicians should strengthen the understanding of this rare tumor for early detection and treatment.
Case report: Isolated dissection of the left gastric artery: an unusual cause of acute abdominal pain
Authors: V Frey, R P Engelberger, E Psathas
Journal: Front Cardiovasc Med (2023)
Keywords: conservative treatment, contrast-enhanced computed tomography, left gastric artery dissection, sudden epigastric pain, visceral artery dissection
MeSH terms: Case Reports

Spontaneous and isolated dissection of the left gastric artery is a rare occurrence, with only a handful of cases reported in the medical literature. Clinical presentation may mimic more common intra-abdominal pathologies; however, it is imperative to identify this condition promptly due to its potential serious consequences. This underscores the importance of maintaining a high level of clinical suspicion and including this pathology in the differential diagnosis of patients presenting with acute abdominal symptoms. Hence, this case report aims to increase awareness among clinicians about the importance of identifying and treating this rare condition promptly. A 69-year-old female experienced severe epigastric pain while attending a yoga class, prompting her admission to the emergency department 24 h later due to the persistence of her symptoms. Following imaging work-up utilizing computed tomography angiography (CTA), she was diagnosed with a dissection of the left gastric artery. Notably, there was no associated aneurysm or any evidence of ischemia in the esophageal or gastric wall. Conservative management, including low-dose aspirin and blood pressure control, was implemented. After 6 months of follow-up, CTA demonstrated expansion of the true lumen and the absence of secondary aneurysm formation, leading to discontinuation of aspirin. The management of spontaneous dissection of visceral arteries is primarily determined by the presence of complications and organ ischemia. In the case of uncomplicated visceral artery dissections, first-line treatment comprises surveillance and antiaggregation. Nevertheless, the optimal duration of antiplatelet therapy and the necessity for long-term follow-up remain unclear. Endovascular or surgical interventions should be reserved for patients exhibiting deteriorating symptoms or complications, and the decision to pursue these interventions should be made on a case-by-case basis.
Point-of-Care Ultrasound for Mimicker Lesions of Incarcerated Inguinal Hernia
Authors: Takahiro Hosokawa, Shinsuke Yoshizawa, Kyoichi Deie, Kensuke Ohashi, Hiroshi Kawashima
Journal: Case Rep Pediatr (2023)
MeSH terms: Case Reports

Inguinal hernia is the most common surgical disease in pediatric patients, and urgent intervention such as manual reduction is needed for incarcerated inguinal hernia. Torsion of undescended testes, inguinal herniated ovarian torsion, and purulent lymphadenitis are mimickers of this condition. If these mimicker lesions are misdiagnosed as incarcerated inguinal hernia, manual reduction usually fails, and edematous and erythematous changes may occur in these mimicker lesions due to manual reduction. For physicians in the emergency department, prompt decisions and familiarity with the sonographic appearance of different contents within an inguinal hernia are important to accurately diagnose these mimickers. In this case series, we present sonographic images of a typical case of incarcerated inguinal hernia (an 11-month-old male with right incarcerated inguinal hernia) and three cases of mimicker lesions (a 7-month-old female with herniated ovarian torsion, a 7-year-old boy with undescended testicular torsion, and a 2-month-old male with purulent lymphadenitis). The incidence of incarcerated inguinal hernia is reported to be higher in males (80%), on the right side (60%), and in infants and toddlers. This information is important for diagnosing mimicker lesions. In addition, to prevent manual reduction in mimicker diseases, point-of-care ultrasound before manual reduction in suspected cases of incarcerated inguinal hernia is important.
Rapid progression of condyloma acuminatum caused by IL-17A antibody treatment: a case report
Authors: Fang Sun, Zhenze Yu
Journal: Front Med (Lausanne) (2024)
Keywords: hpv, il-17a antagonist, condyloma acuminatum, psoriasis, secukinumab
MeSH terms: Case Reports

Anti interleukin (IL)-17A therapy is a common and effective treatment for psoriasis, but there are also risks of infection. In this case, we presented a patient who experienced a swift progression of condyloma acuminatum on the genitals during psoriasis treatment with secukinumab, a human IL-17A antagonist. Through this case, we strongly suspect that anti IL-17A treatment may promote the onset and rapid progression of low-risk HPV-associated condyloma acuminatum.
Severe Acquired Hypothyroidism and Van Wyk-Grumbach Syndrome in Two Children
Authors: Corina Ramona Nicolescu, Lucie Bazus, Jean-Louis Stephan
Journal: Case Rep Pediatr (2024)
MeSH terms: Case Reports

The primary manifestations of chronic hypothyroidism in children include growth arrest, delayed skeletal maturity, and delayed puberty. In 1960, Van Wyk and Grumbach reported three girls with hypothyroidism and a combination of incomplete isosexual precocious puberty (early breast development, menstruation, and absence of pubic hair), galactorrhea, delayed bone age, and pituitary enlargement. All abnormalities regressed after appropriate thyroid hormone replacement therapy. Over the years, an increasing number of reported cases has allowed for a more precise understanding of the clinical, biochemical, and radiological phenotypes of the Van Wyk-Grumbach syndrome (VWGS). These varying clinical manifestations are thought to result from a unique pathophysiological process where the thyroid-stimulating hormone (TSH) is a key element. We describe the cases of two patients (a boy and a girl) with severe autoimmune thyroiditis and VWGS. The clinical, biochemical, and radiological imaging characteristics were similar in both patients and included growth failure, absence of clinical goiter, markedly elevated TSH concentrations >100 mIU/L, undetectable free thyroxine levels, "normal" thyroglobulin levels, high follicle-stimulating hormone (FSH) and prolactin levels, prepubertal levels of luteinizing hormone (LH), delayed bone age, and hyperplasia of the pituitary gland. The two patients displayed differences, especially in the absence of clinical pubertal development, moderate anemia, abnormal renal function, and moderate goiter detected via ultrasonography (in the female patient). Thyroxine replacement therapy reversed the VWGS phenotype and hypothyroidism, with satisfactory growth velocity, strictly normal thyroid function, and normal pituitary size detected via magnetic resonance imaging at the 6-month follow-up visit.
Case Report: Chronic pancreatitis in children as the cumulative effect of bilio-pancreatic abnormalities and genetic mutations
Authors: Francesca Destro, Eleonora Durante, Raffaele Salerno, Alessandro Campari, Milena Meroni, Veronica Diotto, Marco Brunero, Gloria Pelizzo
Journal: Front Pediatr (2024)
Keywords: prss1, anatomic variants, bilio-pancreatic malformation, children, chronic pancreatitis, endoscopy, genetic malformations, pancreatic duct duplication
MeSH terms: Case Reports

Pancreatitis, in general, is a high-morbidity condition. Genetic conditions and anatomic variants are sometimes seen, especially in children, where biliary etiologies and alcohol are less common than in adults. The decision to intervene, the combined operative-endoscopic strategy, and the timing pose unique challenges. We report the case of a 10-year-old boy with PRSS1 mutation and pancreatic duct duplication, discussing the management and reviewing the recent reports in the Literature.