OpenMed/multicare-articles huggingface dataset:
Myroides odoratimimus urinary tract infection in an immunocompromised patient: an emerging multidrug-resistant micro-organism
Authors: Giovanni Lorenzin, Giorgio Piccinelli, Lucrezia Carlassara, Francesco Scolari, Francesca Caccuri, Arnaldo Caruso, Maria Antonia De Francesco
Journal: Antimicrob Resist Infect Control (2018)
MeSH terms: Anti-Bacterial Agents / administration & dosage, Drug Resistance, Multiple, Bacterial, Flavobacteriaceae / drug effects, Flavobacteriaceae / isolation & purification, Flavobacteriaceae Infections / microbiology, Urinary Tract Infections / microbiology, Case Reports, Aged, Flavobacteriaceae / genetics, Flavobacteriaceae / physiology, Flavobacteriaceae Infections / drug therapy, Flavobacteriaceae Infections / immunology, Humans, Immunocompromised Host, Male, Microbial Sensitivity Tests, Sulfamethoxazole / administration & dosage, Trimethoprim / administration & dosage, Urinary Tract Infections / drug therapy, Urinary Tract Infections / immunology, Anti-Bacterial Agents, Trimethoprim, Sulfamethoxazole

Background:

Myroides spp. are common environmental organisms and they can be isolated predominantly in water, soil, food and in sewage treatment plants. In the last two decades, an increasing number of infections such as urinary tract infections and skin and soft tissue infections, caused by these microorganisms has been reported. Selection of appropriate antibiotic therapy to treat the infections caused by Myroides spp. is difficult due to the production of a biofilm and the organism's intrinsic resistance to many antibiotic classes.
Case presentation:
We report the case of a 69-year-old immunocompromised patient who presented with repeated episodes of macroscopic haematuria, from Northern Italy.A midstream urine sample cultured a Gram negative rod in significant amounts (> 105 colony-forming units (cfu)/mL), which was identified as Myroides odoratimimus. The patient was successfully treated with trimethoprim/sulfamethoxazole after antibiotic susceptibility testing confirmed its activity.
Conclusion:
This case underlines the emergence of multidrug resistant Myroides spp. which are ubiquitous in the environment and it demands that clinicians should be more mindful about the role played by atypical pathogens, which may harbour or express multidrug resistant characteristics, in immunocompromised patients or where there is a failure of empiric antimicrobial therapy.
Case report: Thyroid storm in a three-year-old girl presenting with febrile status epilepticus and hypoglycemia
Authors: Yusuke Aoki, Ryo Hanaki, Hidemi Toyoda, Koichi Emori, Masazumi Miyahara, Masahiro Hirayama
Journal: Front Pediatr (2023)
Keywords: graves' disease, beta 1 blocker, febrile convulsion, febrile status epilepticus, hyperthyroid, hypoglycemia, thyroid storm
MeSH terms: Case Reports

Thyroid storm, though extremely rare in toddlers, requires prompt diagnosis and treatment because it can be fatal if left untreated. However, thyroid storm is not often considered in the differential diagnosis of a febrile convulsion due to its rarity in children. Herein, we report the case of a 3-year-old girl with thyroid storm who presented with febrile status epilepticus. Although the seizure was stopped by diazepam administration, her tachycardia and widened pulse pressure persisted, and severe hypoglycemia was observed. Based on the findings of thyromegaly, a history of excessive sweating and hyperactivity, and a family history of Graves' disease, she was eventually diagnosed with a thyroid storm. The patient was successfully treated with thiamazole, landiolol, hydrocortisone, and potassium iodide. Propranolol, a non-selective β-blocker, has been used to manage tachycardia during thyroid storm. However, a cardio-selective β1-blockers, landiolol hydrochloride, was used in our case to avoid worsening hypoglycemia. Febrile status epilepticus is one of the most common medical emergencies in childhood; it is necessary to rule out treatable underlying critical diseases such as septic meningitis and encephalitis. Thyroid storm should be considered in children presenting with prolonged febrile convulsion accompanied by findings that are not usually observed with febrile convulsions.
Cutaneous Squamous Cell Carcinoma with Signet-Ring Cell Component and CDX2 Expression in a Patient Treated with PD-1 Inhibitor: A Case Report of a Common Tumor with Unusual Differentiation
Authors: Sherehan Zada, Jeremiah Tao, Maria Del Valle Estopinal
Journal: Case Rep Pathol (2023)
MeSH terms: Case Reports

Signet-ring cell squamous cell carcinoma (SRCSCC) is an uncommon variant of cutaneous SCC that has been reported in the head and neck region. Herein, we present a case of a 56-year-old female with a cutaneous SCC that recurred after surgical excision, during treatment with cemiplimab (a programmed death receptor-1 (PD-1) inhibitor). Histologically, the recurrent SCC revealed a second component characterized by the presence of signet-ring-like cells (SRLCs). Immunohistochemical studies demonstrated that the tumor cells were positive for P63, CK5/6, CDX2, and P53 while negative for P16, CK7, CK20, and CD68 stains. An abnormal expression of B-catenin was also observed in the tumor. To our knowledge, SRCSCC developing during treatment with an immune checkpoint inhibitor has not been documented in the literature. Our findings suggest a form of acquired SCC cell resistance to immunotherapy that might involve CDX2-related pathways.
Novel Immunotherapy Options for Extranodal NK/T-Cell Lymphoma
Authors: Boyu Hu, Yasuhiro Oki
Journal: Front Oncol (2018)
Keywords: cd30 ligand/cd30, cd38, ebv lymphoma, lmp2, nk t cell lymphoma, latent membrane protein 1, programmed death 1, programmed death ligand 1
MeSH terms: Review

Extranodal NK/T-cell lymphoma (ENKTCL) is a highly aggressive mature NK/T-cell neoplasm marked by NK-cell phenotypic expression of CD3ε and CD56. While the disease is reported worldwide, there is a significant geographic variation with its highest incidence in East Asian countries possibly related to the frequent early childhood exposure of Epstein-Barr virus (EBV) and specific ethnic-genetical background, which contributes to the tumorigenesis. Historically, anthracycline-based chemotherapy such as CHOP (cyclophosphamide, adriamycin, vincristine, and prednisone) was used, but resulted in poor outcomes. This is due in part to intrinsic ENKTCL resistance to anthracycline caused by high expression levels of P-glycoprotein. The recent application of combined modality therapy with concurrent or sequential radiation therapy for early stage disease, along with non-anthracycline-based chemotherapy regimens consisting of drugs independent of P-glycoprotein have significantly improved clinical outcomes. Particularly, this neoplasm shows high sensitivity to l-asparaginase as NK-cells lack asparagine synthase activity. Even still, outcomes of patients with advanced stage disease or those with relapsed/recurrent disease are dismal with overall survival of generally a few months. Thus, novel therapies are needed for this population. Clinical activity of targeted antibodies along with antibody-drug conjugates, such as daratumumab (naked anti-CD38 antibody) and brentuximab vedotin (anti-CD30 antibody conjugated with auristatin E), have been reported. Further promising data have been shown with checkpoint inhibitors as high levels of programmed death-ligand 1 expression are observed in ENKTCL due to EBV-driven overexpression of the latent membrane proteins [latent membrane protein 1 (LMP1) and LMP2] with activation of the NF-κB/MAPK pathways. Initial case series with programmed death 1 inhibitors showed an overall response rate of 100% in seven relapsed patients including five with a complete response (CR). Furthermore, cellular immunotherapy with engineered cytotoxic T lymphocytes targeted against LMP1 and LMP2 have shown encouraging results with durable CRs as either maintenance therapy after initial induction chemotherapy or in the relapsed/refractory setting. In this paper, we review this exciting field of novel immunotherapy options against ENKTCL that hopefully will change the treatment paradigm in this deadly disease.
Gastric Adenocarcinoma Presenting as a Rheumatoid Factor and Anti-cyclic Citrullinated Protein Antibody-Positive Polyarthritis: A Case Report and Review of Literature
Authors: Manuel Silvério-António, Federica Parlato, Patrícia Martins, Nikita Khmelinskii, Sandra Braz, João Eurico Fonseca, Joaquim Polido-Pereira
Journal: Front Med (Lausanne) (2021)
Keywords: gastric cancer, paraneoplastic arthritis, paraneoplastic syndrome, rheumatoid arthritis, seropositive arthritis
MeSH terms: Case Reports

A 64-year-old male presented with a 6-month history of symmetric polyarthritis involving proximal interphalangeal joints and metacarpophalangeal joints of the hands, wrists, and ankles. Associated symptoms included vomiting, progressive fatigue, and weight loss. Laboratory results showed microcytic anemia, leukocytosis, thrombocytosis, elevated C-reactive protein and erythrocyte sedimentation rate, and rheumatoid factor (RF) and anti-cyclic citrullinated protein (ACPA) antibody positivity. Joints radiographs were normal, without erosions. Upper endoscopy and gastric endoscopic ultrasonography showed a gastric adenocarcinoma with lymphatic involvement. Intraoperatively, peritoneal carcinomatosis was documented, and the patient started palliative chemotherapy. A paraneoplastic seropositive arthritis was assumed, and treatment with low-dose prednisolone and hydroxychloroquine was started. Arthritis remission was achieved and sustained up to 18 months of follow-up, although gastric cancer progression was documented. We describe a unique phenotype of paraneoplastic arthritis (PA) presenting as a seropositive (RF and ACPA positivity) rheumatoid arthritis (RA) with a good response to both low dose corticosteroids and hydroxychloroquine therapy. We also review the literature of PA, mostly the RA-like pattern, and the association between PA and ACPA positivity. This case highlights the importance of considering underlying cancer in elderly male patients, presenting with polyarthritis and systemic symptoms, even in those with ACPA-positive RA-like arthritis.
Rare features associated with Mobius syndrome: Report of two cases
Authors: Rumela Ghosh, Vikram Shetty, Shruthi Hegde, G Subhas Babu, Vidya Ajila, Nanda Kishore P, Mithula Nair
Journal: J Dent Res Dent Clin Dent Prospects (2017)
Keywords: congenital, facial nerve, mobius syndrome, palsy, strabismus
MeSH terms: Case Reports

Mobius syndrome is a rare congenital disorder with the preliminary diagnostic criteria of congenital facial and abducent nerve palsy. Involvement of other cranial nerves, too, is common. Prevalence rate of this syndrome is approximately 1 in 100,000 neonates. It is of unknown etiology with sporadic occurrence. However, data regarding the occurrence rate in India is limited. Features such as orofacial malformations, limb defects, and musculoskeletal, behavioral, and cognitive abnormalities might be associated. A thorough evaluation to identify the condition and establishing an adequate treatment plan is of utmost important in this condition. We are reporting clinical and radiographic features of Mobius syndrome in two cases along with unusual findings of limb and neck deformity.
Diaphragmatic Rupture and Hernia after Cardiopulmonary Resuscitation
Authors: Feridoun Sabzi, Reza Faraji
Journal: Tanaffos (2017)
Keywords: cardiopulmonary resuscitation, diaphragmatic hernia, double valve replacement
MeSH terms: Case Reports

A 55-year-old man underwent surgical replacement of a mitral valve 10 years earlier. In a retrospective evaluation of a chest radiograph, the diaphragm was intact at the time of initial surgery. He was then admitted to our emergency room with a complaint of vertigo. During evaluation, he developed decreased consciousness. Ventricular fibrillation was diagnosed, and external massage and full cardiopulmonary resuscitation were performed. After 20 minutes, his sinus rhythm returned and hemodynamic status stabilized with inotropic drugs. Transthoracic echocardiography showed normal valvular function and no evidence of left cardiac malfunction or clot. Electrocardiography showed ST elevation in inferior leads, and levels of cardiac enzymes were elevated. Angiography showed an embolic lesion in the mid right coronary artery that was treated with percutaneous coronary intervention (PCI) and insertion of a stent. After 24 hours, the patient was extubated in good condition, but had mild dyspnea that progressed to CO2 narcosis and subsequent reintubation. Post-extubation chest radiography showed herniation of abdominal organs into the right hemithorax. The diaphragmatic defect was closed with a polytetrafluoroethylene patch by a thoracic surgeon, and the postoperative course was uncomplicated.
Case Report: A Delayed Diagnosis of Congenital Syphilis-Too Many Missed Opportunities
Authors: Sarah Khafaja, Yolla Youssef, Nidale Darjani, Nour Youssef, Captain Mohammad Fattah, Rima Hanna-Wakim
Journal: Front Pediatr (2021)
Keywords: awareness, congenital syphilis, delayed diagnosis, maternal screening, prevention
MeSH terms: Case Reports

Congenital syphilis remains a significant public health problem nowadays. We describe the presentation of an infant with a delayed diagnosis of congenital syphilis, with a negative initial non-treponemal test. Our aim is to shed light on the incidence of missed prevention, the importance of awareness, maternal screening, and early diagnosis.
Case Report: Cerebral Venous Sinus Thrombosis and COVID-19 Infection
Authors: Manasa Anipindi, Amanda Scott, Li Joyce, Salman Wali, Mark Morginstin
Journal: Front Med (Lausanne) (2021)
Keywords: covid-19, anticoagulant, case report, cerebral venous sinus thrombosis (cvst), headache
MeSH terms: Case Reports

Coronavirus disease-2019 is caused by the severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2 virus). Coronavirus disease-2019 (COVID-19) was declared a pandemic in March 2020 and has changed our lives in many ways. This infection induces a hypercoagulable state leading to arterial and venous thrombosis, but the exact pathophysiology of thrombosis is unknown. However, various theories have been postulated including excessive cytokine release, endothelial activation, and disseminated intravascular coagulation (DIC). We present a patient diagnosed with cerebral venous sinus thrombosis (CVST) with COVID-19 infection. A 66-year-old man presented to a hospital for evaluation of persistent headaches. He tested positive for COVID-19, and MRI of the brain and CT venogram revealed CVST. He was started on heparin drip in the hospital and transitioned to oral anticoagulants at the time of discharge. His headaches improved with treatment. Even though headache is the most frequent and initial symptom of cerebral venous thrombosis, it is rarely the only symptom. A high index of suspicion is therefore required to diagnose CVST especially if the patient presents with a simple complaint like a headache. Common complaints can delay the diagnosis leading to disease progression. Considering the high mortality rates in patients diagnosed with CVST, we suggest the importance of knowing the association between COVID-19 infection and CVST, especially in susceptible patients.
11C-UCB-J synaptic PET and multimodal imaging in dementia with Lewy bodies
Authors: Nicolas Nicastro, Negin Holland, George Savulich, Stephen F Carter, Elijah Mak, Young T Hong, Selena Milicevic Sephton, Tim D Fryer, Franklin I Aigbirhio, James B Rowe, John T O'Brien
Journal: Eur J Hybrid Imaging (2020)
Keywords: amyloid, brain atrophy, dementia, lewy bodies, synaptic imaging, tau
MeSH terms: Case Reports

Objective:
Dementia with Lewy bodies (DLB) is a common cause of dementia, but atrophy is mild compared to Alzheimer's disease. We propose that DLB is associated instead with severe synaptic loss, and we test this hypothesis in vivo using positron emission tomography (PET) imaging of 11C-UCB-J, a ligand for presynaptic vesicle protein 2A (SV2A), a vesicle membrane protein ubiquitously expressed in synapses.
Methods:
We performed 11C-UCB-J PET in two DLB patients (an amyloid-negative male and an amyloid-positive female in their 70s) and 10 similarly aged healthy controls. The DLB subjects also underwent PET imaging of amyloid (11C-PiB) and tau (18F-AV-1451). 11C-UCB-J binding was quantified using non-displaceable binding potential (BPND) determined from dynamic imaging. Changes in 11C-UCB-J binding were correlated with MRI regional brain volume, 11C-PiB uptake and 18F-AV-1451 binding.
Results:
Compared to controls, both patients had decreased 11C-UCB-J binding, especially in parietal and occipital regions (FDR-corrected p < 0.05). There were no significant correlations across regions between 11C-UCB-J binding and grey matter, tau (18F-AV1451) or amyloid (11C-PiB) in either patient.
Conclusions:
Quantitative imaging of in vivo synaptic density in DLB is a promising approach to understanding the mechanisms of DLB, over and above changes in grey matter volume and concurrent amyloid/tau deposition.
Supplementary information:
The online version contains supplementary material available at 10.1186/s41824-020-00093-9.
Mutations of MACF1, Encoding Microtubule-Actin Crosslinking-Factor 1, Cause Spectraplakinopathy
Authors: Lulu Kang, Yi Liu, Ying Jin, Mengqiu Li, Jinqing Song, Yi Zhang, Yao Zhang, Yanling Yang
Journal: Front Neurol (2020)
Keywords: macf1, myopathy, neuromuscular disorder, spectraplakinopathy, whole exome sequencing
MeSH terms: Case Reports

As a member of spectraplakin family of cytoskeletal crosslinking proteins, microtubule-actin crosslinking factor 1 (MACF1) controls cytoskeleton network dynamics. Knockout of Macf1 in mice resulted in the developmental retardation and embryonic lethality. Spectraplakinopathy type I, a novel neuromuscular condition characterized by periodic hypotonia, lax muscles, joint contracture, and diminished motor skill, was reported to be associated with heterozygous genomic duplication involving the MACF1 loci, with incomplete penetrance and highly variable clinical presentation in a single pedigree. In this study, parental-derived compound heterozygous novel missense mutations of MACF1, c.1517C>T (p.Thr506Ile) and c.11654T>C (p.Ile3885Thr), were found to co-segregate with disease status in two affected brothers presenting with progressive spastic tetraplegia, dystonia, joint contracture, feeding difficulty and developmental delay. We speculated that MACF1 mutations cause spectraplakinopathy inherited in an autosomal recessive manner. Our clinical findings expanded the phenotype of this neuromuscular disorder and provided new insights into the function of MACF1.
Bortezomib Use for a Critically Ill Patient with Angioimmunoblastic T-Cell Lymphoma
Authors: Motoharu Shibusawa
Journal: Case Rep Hematol (2022)
MeSH terms: Case Reports

Angioimmunoblastic T-cell lymphoma (AITL) accounts for 18.5% of all peripheral T-cell lymphomas. There is still no gold standard chemotherapy for treating newly diagnosed AITL. This case describes the use of bortezomib in newly diagnosed AITL. A 53-year-old man with no previous illness presented with erythema and swelling in the left neck. A diagnosis of AITL was made based on the results of lymph node biopsies. AITL progression led the patient to a severely deteriorated general condition. Bortezomib was thus administered, which resulted in a reduction in lymphadenopathies, the disappearance of tumor fever, and a decrease in serum lactate dehydrogenase levels. Subsequently, the patient's general condition gradually improved. Despite the patient's poor condition, bortezomib was well tolerated. After bortezomib administration, the patient did not require chemotherapy for approximately 10 months. The present case indicates that bortezomib is a possible treatment option for patients with AITL.
A case of wolfram syndrome
Authors: Gholamali Naderian, Fereshteh Ashtari, Kia Nouri-Mahdavi, Valleh Sajjadi
Journal: J Ophthalmic Vis Res (2010)
Keywords: deafness, diabetes insipidus, diabetes mellitus, optic atrophy
MeSH terms: Case Reports

Purpose:
To report a case of Wolfram syndrome characterized by early onset diabetes mellitus and progressive optic atrophy.
Case report:
A 20-year-old male patient with diabetes mellitus type I presented with best corrected visual acuity of 1/10 in both eyes with correction of -0.25+1.50@55 and -0.25+1.50@131 in his right and left eyes, respectively. Bilateral optic atrophy was evident on fundus examination. The patient also had diabetes insipidus, neurosensory deafness, neurogenic bladder, polyuria and extra-residual voiding indicating atony of the urinary tract, combined with delayed sexual maturity.
Conclusion:
One should consider Wolfram syndrome in patients with juvenile onset diabetes mellitus and hearing loss. Ophthalmological examination may disclose optic atrophy; urologic examinations are vital in such patients.
Anasarca, Fever, Thrombocytopenia, Organomegaly, and Multiorgan Failure in a 24-Year-Old Pregnant Woman
Authors: Guillaume Morel, Joy Mootien, Philippe Guiot, Khaldoun Kuteifan
Journal: Case Rep Crit Care (2017)
MeSH terms: Case Reports

TAFRO syndrome is a distinct idiopathic multicentric Castleman disease characterized by the association of thrombocytopenia, anasarca, fever, reticulin fibrosis, and organomegaly. We report the first case occurring in a Caucasian pregnant woman. At 34 weeks of gestation, our patient presented with all clinical and biological symptoms compatible with a TAFRO syndrome. Tough quick cesarean section was performed as symptoms got worse with onset of multiorgan failure requiring mechanical ventilation for acute respiratory distress, continuous renal replacement, and vasopressors. Nine days after ICU admission, steroid boluses were started and allowed spectacular clinical and biological improvement. As systemic inflammatory manifestations are important, TAFRO syndrome can be mistaken with severe autoimmune diseases, systemic infections, hematological malignancies, or hemophagocytic lymphohistiocytosis.
Severe Vaginal Bleeding in a Case of Renal Cell Carcinoma
Authors: Ryan Machiele, Taylor Renbarger, Bret Guidry
Journal: Case Rep Obstet Gynecol (2019)
MeSH terms: Case Reports

Renal Cell Carcinoma (RCC) accounts for approximately 2-3% of all adult cancers and carries the highest mortality of the genitourinary cancers. Metastatic disease is seen in approximately 16% of cases and when present represents an advanced status. Metastasis of RCC to the vagina has rarely been cited in literature and when present can mimic primary vaginal cancer in clinical presentation and symptoms. Biopsy is performed to delineate the etiology and, in the presence of clear cells and certain immunohistochemistry markers, RCC needs to be included in the differential diagnosis. Treatment protocols are limited due to the rarity of the condition, with retrospective and comparative studies alongside cervical cancer treatment protocols serving as the basis. Herein, we describe a unique case of profuse vaginal bleeding secondary to vaginal metastases of RCC and discuss the relevant aspects of diagnosis and treatment.