OpenMed/multicare-articles huggingface dataset:
Os odontoideum associated with a retro-odontoid cyst treated with posterior C1-C3 fixation: A case report and literature review
Authors: Bei-Xi Bao, Hui Yan, Jia-Guang Tang
Journal: Front Surg (2023)
Keywords: atlantoaxial stability, gradual resorption of cyst, os odontoideum, posterior c1-c3 fixation, retro-odontoid cyst
MeSH terms: Case Reports

Background:
Os odontoideum is a rare abnormality of the upper cervical spine, and os odontoideum associated with a retro-odontoid cyst has been described as a marker of local instability.
Case description:
This paper reports a case of a 52-year-old female patient who was diagnosed with os odontoideum associated with a retro-odontoid cyst. The patient underwent posterior C1-C3 fixation without surgical removal of the cyst. Magnetic resonance imaging (MRI) two days later revealed that the retro-odontoid cyst was still present and that there were no significant changes to it when compared with the preoperative MRI.
Conclusion:
Retro-odontoid cysts associated with unstable os odontoideum can lead to symptomatic spinal cord compression. Posterior C1-C3 fixation can restore atlantoaxial stability by allowing the gradual resorption of the cyst and ensuring spinal cord decompression. Fixation can also avoid the surgical risk associated with a high-riding vertebral artery.
Epidemiology, treatment, costs, and long-term outcomes of patients with fireworks-related injuries (ROCKET); a multicenter prospective observational case series
Authors: Daan T Van Yperen, Cornelis H Van der Vlies, J Tjeerd H N De Faber, Xander Smit, Suzanne Polinder, Charlotte J M Penders, Esther M M Van Lieshout, Michael H J Verhofstad, ROCKET study group
Journal: PLoS One (2020)
MeSH terms: Wounds and Injuries / epidemiology, Wounds and Injuries / therapy, Multicenter Study, Observational Study, Activities of Daily Living, Adolescent, Adult, Child, Female, Humans, Male, Patient Reported Outcome Measures, Schools, Time Factors, Treatment Outcome, Work, Wounds and Injuries / economics, Young Adult

Objective:
People in the Netherlands are legally allowed to celebrate New Year's Eve with consumer fireworks. The aim of this study was to provide detailed information about the patient and injury characteristics, medical and societal costs, and clinical and functional outcome in patients with injuries resulting from this tradition.
Methods:
A multicenter, prospective, observational case series performed in the Southwest Netherlands trauma region, which reflects 15% of the country and includes a level I trauma center, a specialized burn center, a specialized eye hospital, and 13 general hospitals. All patients with any injury caused by consumer fireworks, treated at a Dutch hospital between December 1, 2017 and January 31, 2018, were eligible for inclusion. Exclusion criteria were unknown contact information or insufficient understanding of Dutch or English language. The primary outcome measure was injury characteristics. Secondary outcome measures included treatment, direct medical and indirect societal costs, and clinical and functional outcome until one year after trauma.
Results:
54 out of 63 eligible patients agreed to participate in this study. The majority were males (N = 50; 93%), 50% were children below 16 years of age, and 46% were bystanders. Injuries were mainly located to the upper extremity or eyes, and were mostly burns (N = 38; 48%) of partial thickness (N = 32; 84%). Fifteen (28%) patients were admitted and 11 (20%) patients needed surgical treatment. The mean total costs per patient were €6,320 (95% CI €3,400 to €9,245). The most important cost category was hospital admission. Only few patients reported complaints in patient-reported quality of life and functional outcome after 12 months follow-up.
Conclusion:
This study found that young males are most vulnerable for fireworks injuries and that most injuries consist of burns, located to the arm and hand, and eye injuries. On the long-term only few patients experienced reduced quality of life and functional limitations.
The Use of Virtual Reality to Facilitate Mindfulness Skills Training in Dialectical Behavioral Therapy for Spinal Cord Injury: A Case Study
Authors: Araceli Flores, Marsha M Linehan, S Rob Todd, Hunter G Hoffman
Journal: Front Psychol (2018)
Keywords: dialectical behavior therapy, emotions, mindfulness training, spinal cord injury (sci), virtual reality therapy
MeSH terms: Case Reports

Introduction: Paralysis from a spinal cord injury (SCI) increases risk of psychological problems including suicide attempts, substance use disorder, negative emotions (e.g., anger), depression, anxiety, ASD/PTSD. Dialectical Behavioral Therapy® (DBT®) has been shown to be effective for treating similar psychological symptoms in non-SCI patient populations. The current study explored for the first time, the feasibility and clinical potential of using Immersive Virtual Reality (VR) enhanced DBT® Mindfulness skills training to help reduce psychological symptoms (negative emotions and anxiety, ASD/PTSD) of two patients with SCI. Patient 1 was a 39-year-old male patient suffering multiple spinal cord injuries, resulting in quadriplegia, after falling out of a four story building. Patient 1 had severe depression, and anxiety symptoms. Patient 2, was a 31 year old male with a C7 vertebral body fracture, leading to paresis, after suffering a blunt force trauma injury during an attempted suicide, jumping from a moving vehicle. Patient 2 had mild depression, and anxiety symptoms. Methods: Each patient looked into VR goggles, and had the illusion of slowly "floating down" a river in virtual reality while listening to DBT® Mindfulness Skills training instructions. Each patient filled out brief psychological ratings before and after each VR session, four VR DBT® sessions for patient 1, and two VR DBT® sessions for patient 2. Results: As predicted, patient 1 reported reductions in negative emotions after each VR DBT® Mindfulness session. Patient 2 had mixed results on some of the measures of negative emotions. And both patients reported feeling less depressed, less anxious, and less emotionally upset, after VR DBT® Mindfulness Skills learning. Patient 2 reported large reductions in short term ASD/PTSD symptoms after his first VR DBT® mindfulness skills training session. Conclusion: This study explored the feasibility of using VR DBT® with quadriplegic or paresis SCI patients. Both SCI patients accepted VR, the patients liked using VR, and, with assistance from the therapist, the patients were able to use the VR equipment, despite being paralyzed. Additional research and development will be needed to determine whether VR DBT® Mindfulness Skills training leads to any long term improvements in outcome.
Complete Response to Nivolumab in Recurrent/Metastatic HPV-Positive Head and Neck Squamous Cell Carcinoma Patient After Progressive Multifocal Leukoencephalopathy: A Case Report
Authors: Laura Deborah Locati, Mara Serena Serafini, Andrea Carenzo, Silvana Canevari, Federica Perrone, Ester Orlandi, Serena Delbue, Stefano Cavalieri, Giulia Berzeri, Anna Pichiecchio, Lisa Francesca Licitra, Enrico Marchioni, Loris De Cecco
Journal: Front Oncol (2022)
Keywords: hnscc, hpv, pml, case report, immunotherapy, oropharynx
MeSH terms: Case Reports

In an immune-competent context nivolumab showed long-term benefit in overall survival in recurrent/metastatic head and neck squamous cell carcinoma (HNSCC); however, in special cancer population such as these patients with immunodeficiency and viral infections, data on checkpoint inhibitors (ICI) activity are scant. Herein, we report a patient with a Human papilloma virus (HPV)-related oropharyngeal cancer (OPC) and CD4 lymphocytopenia. After a first-line treatment complete remission, the patient experienced Human Polyomavirus (JCV) infection in the brain. Consequently, to the recovery from progressive multifocal leukoencephalopathy (PML) the patient metastasized and was enrolled in a single-arm trial with nivolumab (EudraCT number: 2017-000562-30). A complete and durable response (more 3 years) was observed after 10 nivolumab injections Q2wks, interrupted for persistent drug related G2 diarrhea and a syndrome of inappropriate antidiuretic hormone secretion. We describe the circulating immune profile (before-, during-, and after nivolumab), consistent with the clinical history. Moreover, during nivolumab treatment, brain MRI evidenced the presence of small punctuate areas of contrast enhancement, reflecting a mild immune response in perivascular spaces. By cytofluorimetry, we observed that during JCV infection the CD4/CD8 ratio of the patient was under the normal values. After JCV infection recovery and before nivolumab treatment, CD4/CD8 ratio reached the normality threshold, even if the CD4+ T cell count remained largely under the normal values. During ICI, gene expression xCell analyses of circulating immune cells of the patient, showed a progressive normalization of the total immune profile, with significant boost in CD4+ and CD8+ T cells and a reduction in NK T, comparable to the circulating immune profile of reference tumor-free HNSCC patients. The present case supports the activity of ICI in a population of special cancer patients; whether JCV and HPV infections (alone or together) might have a possible role as immune booster(s), require further investigations.
Massive Pulmonary Embolism Complicating Coronavirus Disease 2019 (COVID-19) Pneumonia: A Case Report
Authors: Shruti Hegde, Gemini Yesodharan, John Tedrow, Alena Goldman
Journal: Case Rep Crit Care (2020)
MeSH terms: Case Reports

Background:
Patients with severe COVID-19 pneumonia are hypercoagulable and are at risk for acute pulmonary embolism. Timely diagnosis is imperative for their prognosis and recovery. This case describes an otherwise healthy 55-year-old man with respiratory failure requiring mechanical ventilatory support secondary to COVID-19 pneumonia. Massive acute pulmonary embolism with right heart failure complicated his course.
Case:
A healthy 55-year-old man presented to our emergency department (ED) with a sore throat, cough, and myalgia. A nasopharyngeal swab was obtained, and he was discharged for home quarantine. His swab turned positive for SARS-CoV-2 infection on real-time reverse transcriptase-polymerase chain reaction assay (RT-PCR) on day 2 of his ED visit. A week later, he represented with worsening shortness of breath, requiring intubation for hypoxic respiratory failure due to COVID-19 pneumonia. Initially, he was easy to oxygenate, had no hemodynamic compromise, and was afebrile. On day 3, he became febrile and developed significant hemodynamic instability requiring maximum vasopressor support and oxygenation difficulty. His ECG revealed sinus tachycardia with S1Q3T3 pattern. On bedside TTE, there was evidence of right heart strain and elevated pulmonary artery systolic pressure of 45 mmHg. All data was indicative of a massive APE as the etiology for his hemodynamic collapse. A decision was made to forgo computed tomography pulmonary angiography (CTPA), given his clinical instability, and systemic thrombolytic therapy was administered. Within the next 12-24 hours, his hemodynamic status significantly improved.
Conclusions:
This case highlights the importance of considering massive APE in COVID-19 patients as a cause of the sudden and rapid hemodynamic decline. Furthermore, timely diagnosis can be made to aid in appropriate management with the help of bedside TTE and ECG in cases where CTPA is not feasible secondary to the patient's hemodynamic instability.
Pseudohypertriglyceridemia: A Novel Case with Important Clinical Implications
Authors: Ankur Rughani, Kenneth Blick, Hui Pang, Monica Marin, Jonathan Meyer, Jeanie B Tryggestad
Journal: Case Rep Pediatr (2020)
MeSH terms: Case Reports

Pseudohypertriglyceridemia is an overestimation of serum triglyceride levels that may incorrectly lead to a diagnosis of hypertriglyceridemia. Glycerol kinase deficiency is a condition in which glycerol cannot be phosphorylated to glycerol-3-phosphate, resulting in elevated levels of serum glycerol. Laboratory assays that measure triglycerides indirectly may be affected by elevated glyerol levels and incorrectly report serum tryglyceride levels. We present a case of a novel missense mutation in the GK gene leading to isolated glycerol kinase deficiency and pseudohypertriglyceridemia in a male infant of a mother with gestational diabetes. This paper reviews glycerol kinase deficiency, describes the challenges in diagnosing pseudohypertriglyceridemia, and provides suggestions on improving diagnostic accuracy. Additionally, a potential maternal-fetal interaction between gestational diabetes and glycerol kinase deficiency is discussed.
Spontaneous Bacterial Peritonitis due to Lactobacillus paracasei in Cirrhosis
Authors: Emily Harding-Theobald, Bharat Maraj
Journal: Case Rep Gastrointest Med (2018)
MeSH terms: Case Reports

Lactobacillus species colonize the human gastrointestinal tract and are rarely pathogenic. We present a case involving a cirrhotic patient who presented with sepsis and was found to have peritoneal cultures demonstrating Lactobacillus as the sole pathogen concerning for spontaneous bacterial peritonitis. Treatment was achieved with high-dose penicillin and clindamycin but the patient developed hepatorenal syndrome and died from acute renal failure. Intra-abdominal Lactobacillus infections are typically seen in patients undergoing peritoneal dialysis or who have recently had bowel perforation. There are few case reports of spontaneous Lactobacillus peritonitis in patients with cirrhosis. Our case report addresses the challenges of Lactobacillus treatment and suggests antibiotic coverage of commensal organisms in patients who do not improve with standard management.
Skull Metastasis From Uterine Leiomyosarcoma, a Rare Presentation for a Rare Tumor: A Case Report and Review of the Literature
Authors: Alessandro Rizzo, Maria Concetta Nigro, Vania Ramponi, Carmine Gallo, Anna Myriam Perrone, Pierandrea De Iaco, Giovanni Frezza, Damiano Balestrini, Maika Di Benedetto, Jarno Morbiducci, Maria Abbondanza Pantaleo, Margherita Nannini
Journal: Front Oncol (2020)
Keywords: intracranial recurrence, skull metastasis, uterine cancer, uterine leiomyosarcoma, uterine sarcoma
MeSH terms: Case Reports

Uterine leiomyosarcoma (uLMS) is a rare and aggressive malignancy with poor clinical outcomes. Even when localized, uLMS is associated with high rates of local and distant recurrences that are usually fatal. Common sites of recurrence are lung, liver, pelvic lymph nodes, and vertebral and long bones, though atypical patterns of recurrence have been described. Among them, intracranial recurrence appears as a rare finding, almost exceptional in skull and dura. We describe the case of a solitary skull metastasis from uLMS in a 39-year-old woman, which represents the third reported case of skull recurrence in literature. After multidisciplinary discussion, the patient underwent surgery and received adjuvant radiotherapy. After 4 months, she is currently alive, without evidence of extracranial disease. This case highlights the importance of suspecting and recognizing atypical and extremely rare metastasis to this region. We encourage the need for large case series in order to provide further information about cranial recurrences of uLMS taking into account the paucity of data currently available in literature and the frequently unpredictable behavior of this rare and highly lethal disease.
Hypomorphic Mutations in the BCR Signalosome Lead to Selective Immunoglobulin M Deficiency and Impaired B-cell Homeostasis
Authors: Christoph B Geier, Kai M T Sauerwein, Alexander Leiss-Piller, Isabella Zmek, Michael B Fischer, Martha M Eibl, Hermann M Wolf
Journal: Front Immunol (2018)
Keywords: b-cell defects, blnk, btk, marginal-zone b cells, natural antibodies, primary immunodeficiency, selective igm deficiency
MeSH terms: Adaptor Proteins, Signal Transducing / genetics, Agammaglobulinaemia Tyrosine Kinase / genetics, Agammaglobulinemia / genetics, B-Lymphocytes / immunology, Immunoglobulin M / deficiency, Case Reports, Adaptor Proteins, Signal Transducing / metabolism, Adolescent, Adult, Agammaglobulinaemia Tyrosine Kinase / metabolism, Agammaglobulinemia / immunology, B-Lymphocytes / metabolism, Humans, Immunoglobulin M / immunology, Male, Receptors, Antigen, B-Cell / immunology, Receptors, Antigen, B-Cell / metabolism, Signal Transduction / genetics, Signal Transduction / immunology, Adaptor Proteins, Signal Transducing, B cell linker protein, Immunoglobulin M, Receptors, Antigen, B-Cell, Agammaglobulinaemia Tyrosine Kinase, BTK protein, human

B cell activation via the B cell receptor (BCR) signalosome involves participation of signaling molecules such as BTK and BLNK. Genetic defects in these molecules are known to impair B cell differentiation and subsequently lead to agammaglobulinemia. Here we identified novel mutations in BTK and BLNK in two unrelated patients that perturb the intrinsic B-cell receptor signaling pathway and lead to selective IgM deficiency, whereas production of other immunoglobulin isotypes and IgG antibody response remain intact. Currently it is unknown how BCR signaling strength affects mature B cell development in humans. Both patients show reduced levels of BCR signalosome phosphorylation as well as impaired BCR-dependent Ca2+ influx, which was accompanied by a marked decrease in IgD+IgM+CD27+ MZ-like B-cells. We further describe reduced expression of essential B cell differentiation factors such as BAFF-R and T-Bet in the patients' B-cells, which might contribute to the observed deficiency of MZ-like B cells. MZ-like B cells are known to produce natural IgM antibodies that play an essential role in immune homeostasis. By using surface plasmon resonance (SPR) technology and a synthetic blood group A trisaccharide as antigen we were able to show that both patients lack the presence of anti-blood group A IgM considered to be prototypical natural antibodies whereas IgG levels were normal. Antibody binding dynamics and binding affinity of anti-blood group A IgG were comparable between patients and healthy controls. These results indicate that human IgM deficiency can be associated with signaling defects in the BCR signalosome, defective production of natural IgM antibodies in the blood group A/B/0 system and abnormalities in B cell development.
Appendicular endometriosis: A case report and review of literature
Authors: Rahul Gupta, Arvind K Singh, Waad Farhat, Houssem Ammar, Mohamed Azzaza, Abdkader Mizouni, Sami Lagha, Mehdi Ben Latifa, Amal Bouazzi, Ali Ben Ali
Journal: Int J Surg Case Rep (2019)
Keywords: appendicular endometriosis, case report, endometriosis, infertility, pain, right iliac fossa

Introduction:
Patients with appendicular endometriosis (AE) constitute <1% cases of all pelvic endometriosis cases. AE presents with pain in right iliac fossa and symptomatically mimics appendicitis and definitive diagnosis is possible only after histopathological examination of excised appendix.
Presentation of case:
In this case report, we present a rare case of appendicular endometriosis in a young woman suffering from pain in right iliac fossa and periumbilical region (on/off) for the past one year. She had a past history of infertility. Blood investigations were normal and CT scan of abdomen demonstrated findings suggestive of appendicitis. Laparoscopic appendicectomy was performed, which revealed 2 cm tumor-like mass (1.5 cm diameter) present at the tip of appendix involving the mesoappendix. There was no ascites, peritoneal or omental deposits or any signs of inﬂammation. Post-operative recovery was good. Dissected appendix specimen was sent for histopathological examination which confirmed endometriosis of appendix.
Discussion:
Gastrointestinal endometriosis (GE) accounts for 3-37% of all endometriosis cases, whereas AE is present in only ∼3% of all GE cases and constitutes <1% of all the endometriosis cases. AE frequently involves tip and body of the appendix. The layers of appendix most commonly affected are muscular and seromuscular (∼2/3rd cases), followed by the serosa (∼1/3rd cases).
Conclusion:
Thus, appendicular endometriosis is a rare entity and should be included in differential diagnosis in young women presenting with pain in right iliac fossa and having history of infertility.
Acute human self-poisoning with imidacloprid compound: a neonicotinoid insecticide
Authors: Fahim Mohamed, Indika Gawarammana, Thomas A Robertson, Michael S Roberts, Chathura Palangasinghe, Shukry Zawahir, Shaluka Jayamanne, Jaganathan Kandasamy, Michael Eddleston, Nick A Buckley, Andrew H Dawson, Darren M Roberts
Journal: PLoS One (2009)
MeSH terms: Imidazoles / poisoning, Insecticides / poisoning, Nitro Compounds / poisoning, Self-Injurious Behavior, Case Reports, Research Support, Non-U.S. Gov't, Adult, Cohort Studies, Female, Humans, Male, Neonicotinoids, Prospective Studies, Sri Lanka, Imidazoles, Insecticides, Neonicotinoids, Nitro Compounds, imidacloprid

Background:
Deliberate self-poisoning with older pesticides such as organophosphorus compounds are commonly fatal and a serious public health problem in the developing world. The clinical consequences of self-poisoning with newer pesticides are not well described. Such information may help to improve clinical management and inform pesticide regulators of their relative toxicity. This study reports the clinical outcomes and toxicokinetics of the neonicotinoid insecticide imidacloprid following acute self-poisoning in humans.
Methodology/principal findings:
Demographic and clinical data were prospectively recorded in patients with imidacloprid exposure in three hospitals in Sri Lanka. Blood samples were collected when possible for quantification of imidacloprid concentration. There were 68 patients (61 self-ingestions and 7 dermal exposures) with exposure to imidacloprid. Of the self-poisoning patients, the median time to presentation was 4 hours (IQR 2.3-6.0) and median amount ingested was 15 mL (IQR 10-50 mL). Most patients only developed mild symptoms such as nausea, vomiting, headache and diarrhoea. One patient developed respiratory failure needing mechanical ventilation while another was admitted to intensive care due to prolonged sedation. There were no deaths. Median admission imidacloprid concentration was 10.58 ng/L; IQR: 3.84-15.58 ng/L, Range: 0.02-51.25 ng/L. Changes in the concentration of imidacloprid in serial blood samples were consistent with prolonged absorption and/or saturable elimination.
Conclusions:
Imidacloprid generally demonstrates low human lethality even in large ingestions. Respiratory failure and reduced level of consciousness were the most serious complications, but these were uncommon. Substitution of imidacloprid for organophosphorus compounds in areas where the incidence of self-poisoning is high may help reduce deaths from self-poisoning.
Case report: Sclerosed hemangioma of the liver: A diagnostic challenge
Authors: M Poras, G Katsanos, A C Agrafiotis, P Demetter, M Pezzullo, V Lucidi
Journal: Front Surg (2022)
Keywords: cholangiocarcinoma, hemangioma, liver, sclerosed, sclerosing
MeSH terms: Case Reports

Hemangiomas are the most common noncystic benign hepatic tumors and are usually incidentally discovered during routine radiological examinations. The diagnosis of hepatic hemangiomas with a typical presentation is generally easy with plain and cross-sectional imaging; however, it can be complicated when hemangiomas undergo histological changes such as fibrosis. Sclerosed hepatic hemangioma (SHH) is the extreme presentation of this fibrotic process. These atypical lesions can be misdiagnosed as primary hepatic malignancies or metastasis. Their diagnosis is established by histological examination. We report the case of a patient with an SHH, which was misdiagnosed as an intrahepatic cholangiocarcinoma. This article's aim is to draw attention to this infrequent pathology and underline the features of this benign tumor that could suggest its diagnosis prior to surgery to avoid unnecessary hepatic resections.