OpenMed/multicare-articles huggingface dataset:
Chimaeribacter arupi a new member of the Yersineacea family has the characteristics of a human pathogen
Authors: Matthias Riediger, Katharina Hoffmann, Riekje Isberner, Annika Dreyer, Aljoscha Tersteegen, Pauline Marquardt, Achim J Kaasch, Andreas E Zautner
Journal: Front Cell Infect Microbiol (2023)
Keywords: chimaeribacter arupi, blood stream infection, case report, comparative genomics, genome, virulence factors, “nissabacter archeti”
MeSH terms: Fimbriae, Bacterial / metabolism, Sepsis / metabolism, Case Reports, Research Support, Non-U.S. Gov't, Base Sequence, Female, Gram-Negative Bacteria / genetics, Humans, Infant, Virulence Factors / genetics, Virulence Factors / metabolism, Virulence Factors

Chimaeribacter arupi (heterotypic synonym: "Nissabacter archeti") is a facultative anaerobic, newly described Gram-negative rod and belongs to the Yersineacea family. Here, we report the case of a 19-month-old female infant patient who presented to the emergency unit with somnolence and fever. C. arupi was isolated from a positive blood culture, taken via an implanted Broviac catheter, proving a bloodstream infection by the pathogen. The objective of this study was to utilize whole genome sequencing to assess the genes encoding potential virulence associated factors, which may play a role in host tropism, tissue invasion and the subsequent stages in the pathogenesis of a bloodstream infection with C. arupi. The genome of the isolate was completely sequenced employing Illumina MiSeq and Nanopore MinION sequencing and the presumptive virulence associated factors and antimicrobial resistance genes were investigated in more detail. Additionally, we performed metabolic profiling and susceptibility testing by microdilution. The presence of predicted TcfC-like α-Pili suggests that C. arupi is highly adapted to humans as a host. It utilizes flagellar and type IV pili-mediated motility, as well as a number of γ1-pili and a σ-pilus, which may be used to facilitate biofilm formation and adherence to host epithelia. Additionally, long polar fimbriae may aid in tissue invasion. The bacterium possesses antioxidant factors, which may enable temporary survival in phagolysosomes, and a capsule that potentially provides protection from phagocytosis. It may acquire iron ions from erythrocytes through the type 6 secretion system and hemolysins. Furthermore, the isolate exhibits beta-lactamase-mediated penicillin and aminopenicillin resistance. Based on the analysis of the whole genome, we conclude that C. arupi possesses virulence factors associated with tissue invasion and may thus be a potential opportunistic pathogen of bloodstream infections.
IgA-dominant postinfectious glomerulonephritis: a case report
Authors: Rodolfo Moreno-Alvarado, Guillermo Navarro-Blackaller, Werner De Leon-Pérez, David Armas-Eguizabal, Jonathan Chávez-Iñiguez
Journal: Front Nephrol (2023)
Keywords: acute kidney injury, case report, immunoglobulin a, kidney biopsy, postinfectious glomerulonephritis
MeSH terms: Case Reports

Introduction:
Acute postinfectious glomerulonephritis (APIGN) is an immunological glomerular disease that is an important health issue in developing countries. The incidence remains high in developing countries with a male-to-female ratio of 2:1 and age predominantly above 50 years. In this case study, we present a patient with a history of Staphylococcus epidermidis infection, a past medical history of diabetes mellitus, and histopathological findings of APIGN with Immunoglobulin A (IgA) deposition.
Methods:
A 58-year-old male presented to the emergency room with a 6-day history of severe low back pain. Three days later, the patient developed fever, chills, abdominal pain in the upper quadrant and a subsequent lower limb cellulitis. Various immunological tests, imaging studies, and kidney biopsy were performed to arrive at a diagnosis.
Results:
Following the diagnosis and treatment of Cholangitis and Staphylococcus epidermidis, further investigation led to a diagnosis of IgA-dominant APIGN. IgA-dominant APIGN was treated with antibiotics, renin-angiotensin-aldosterone system inhibitors and steroids, and the patient was discharged from the hospital.
Conclusion:
In developing countries, APIGN is a relatively common presentation of kidney damage due to acute kidney injury and nephritic syndrome. IgA-dominant APIGN is a rare but increasingly recognized morphological variant in which IgA is the sole or dominant immunoglobulin. This unique presentation and multidisciplinary approach for diagnosing and treating IgA-dominant APIGN need to be considered and understood by healthcare professionals to better help these patients. Further investigation is needed to understand the best treatment of this IgA-dominant APIGN presentation and its prognosis.
Fallopian canal arachnoid cyst with acute facial nerve paralysis in children: a report of two cases and literature review
Authors: Jianbin Sun, Weiju Han
Journal: Front Neurol (2023)
Keywords: arachnoid cyst, cerebrospinal fluid otorrhea, facial nerve paralysis, fallopian canal, meningitis
MeSH terms: Case Reports

Introduction:
Symptoms induced by arachnoid cysts in the fallopian canal are uncommon, and facial nerve paralysis without cerebrospinal fluid otorrhea is comparatively rarer.
Methods:
Herein, we present two cases of arachnoid cysts in the fallopian canal with acute severe facial nerve paralysis and review the relevant literature.
Results:
The symptoms and imaging findings of these two cases resembled those of facial nerve schwannomas. Cerebrospinal fluid otorrhea occurred upon removal of the arachnoid cyst, and the facial nerve was observed to be separated into multiple filaments or compressed and atrophied. Facial-hypoglossal nerve anastomosis and decompression were conducted after packing the dehiscence of cerebrospinal fluid otorrhea for the two cases.
Conclusion:
Arachnoid cysts of the fallopian canal rarely cause facial nerve paralysis. Enhanced magnetic resonance imaging is vital for differentiating schwannomas. Different treatment strategies should be adopted for patients with different degrees of facial nerve paralysis; however, concurrent repair of cerebrospinal fluid otorrhea and facial nerves during surgery can occasionally be challenging.
Lenacapavir with Fostemsavir in a Multidrug-Resistant HIV-Infected Hemodialysis Patient
Authors: Ferdinand Bigirimana, Sigi Van den Wijngaert, Christelle Fosso, Karolien Stoffels, Charlotte Martin, Evelyne Maillart, Philippe Clevenbergh
Journal: Case Rep Infect Dis (2023)
MeSH terms: Case Reports

We report a hemodialysis MDR HIV-infected patient switched to fostemsavir with lenacapavir plus lamivudine for more than a year. She maintained a suppressed viral replication and did not present any clinical or biological drug-related side effects. The combination of lenacapavir plus fostemsavir looks promising in terms of safety and efficacy even in patients with end-stage renal disease awaiting renal transplant. Both drugs are first in class ARVs so that there is no cross resistance with previous drugs, maintaining their efficacy against MDR HIV.
Case report: Area of focus of management of severe pityriasis rubra pilaris by dose optimization of adalimumab biosimilar in combination with acitretin and montelukast
Authors: Kristine Heidemeyer, S Morteza Seyed Jafari, Lena Farnina, Simon Bossart, Laurence Feldmeyer, Nikhil Yawalkar
Journal: Front Med (Lausanne) (2023)
Keywords: adalimumab, biosimilar, dose-optimization, montelukast, phospholipase a2, pityriasis rubra pilaris
MeSH terms: Case Reports

Pityriasis rubra pilaris (PRP) is a rare inflammatory skin disorder characterized by hyperkeratotic follicular papules, orange-red scaling plaques with islands of sparing and palmoplantar keratoderma. While spontaneous resolution occurs in some cases, treatment can be challenging for others. The use of biologics in PRP management has gained attention in recent studies, although their high costs and potential side effects present limitations. We present a case of a 71-year-old patient with treatment-resistant PRP who showed significant improvement through optimized adalimumab treatment. Considering the emerging role of phospholipase A2 in PRP pathogenesis, montelukast was added, further enhancing the therapeutic response. By maintaining montelukast and prolonging the adalimumab interval to 3 and 4 weeks, effective dose optimization was achieved without PRP relapse. This case report highlights the potential for adalimumab dose optimization by shortening the initial treatment interval for increased effectiveness and lengthening the interval during the maintenance phase to conserve medication doses. Montelukast appears to assist in sustaining clinical outcomes during interval prolongation, necessitating further investigation through additional studies.
Case report: Development of clonal hematologic disorders from inherited bone marrow failure
Authors: Jaroslav Cermak
Journal: Front Oncol (2024)
Keywords: cytopenia, diagnosis, inherited bone marrow failure, myelodysplastic syndromes, transplantation, treatment
MeSH terms: Case Reports

Introduction:
Inherited bone marrow failure (IBMF) syndromes are caused by mutations forming pathologic germline variants resulting in the production of defective hematopoietic stem cells (HSC) and in congenital failure in the production of one or more blood lineages. An acquisition of subsequent somatic mutations is determining further course of the disease. Nevertheless, a certain number of patients with IBMF may escape correct diagnosis in childhood, especially those with mild cytopenia and minimal clinical features without non-hematologic symptoms. These patients usually present in the third decade of life with unexplained cytopenia or myelodysplastic syndrome (MDS).
Methods and results:
We report 2 patients with IBMF who were correctly diagnosed between 20 and 40 years of age when they were referred with progressive MDS with adverse prognostic factors that affected their outcome.
Discussion:
IBMF syndromes should be excluded in all patients below 40 years of age with unexplained cytopenia. Early hematopoietic stem cell transplantation (HSCT) is the treatment of choice in these patients.
A Neglected Disease: Hidradenitis Suppurativa a Rare Cause of Amyloidosis Complicated With Sepsis and Renal Failure: A Case Report
Authors: Orhun Demir, Omer Zuhtu Yondem, Mehmet Doganay
Journal: Case Rep Crit Care (2024)
MeSH terms: Case Reports

Background: Hidradenitis suppurativa (HS) is a painful relapsing inflammatory disease characterized with subcutaneous nodules, abscesses, tunnels, and deforming scars. We present a case of HS complicated with amyloidosis who was admitted with sepsis and acute renal failure. Case Report: A 53-year-old male patient with a history of HS and amyloidosis was accepted to the intensive care unit suffering from acute kidney failure and sepsis symptoms. He was receiving adalimumab during admission. He received hemodialysis and piperacillin-tazobactam antibiotherapy. After 15 days of hospitalization, his sepsis was treated, but he was taken to a routine hemodialysis programme. Conclusion: In the presence of amyloidosis with intervenient infections, the risk of chronic renal failure in HS cases can be kept in mind even if the patient is receiving TNF inhibitors.
HIV False-Positive Test in the Setting of CD4 Lymphocytopenia
Authors: Hussien Mohamed, Hanna D Hedriana, Emily A Holbrook, Heather Henderson, Jason W Wilson
Journal: Cureus (2024)
Keywords: cd4 lymphocytopenia, cdc hiv algorithm, false hiv test, false positive, false-positive hiv, hiv testing
MeSH terms: Case Reports

In 2016, we implemented a non-targeted Emergency Department (ED)-based HIV screening program at our academic medical center following revised CDC guidelines utilizing the Abbott Alinity 4th generation HIV-1/2 antigen (Ag)/antibody (Ab) immunoassay (Abbott Laboratories, Abbott Park, IL). Following the CDC algorithm, after reactive fourth-generation testing, HIV-1/2 Ab testing is conducted. Patients undergoing acute seroconversion (acutes) may express p24 Ag but have a negative confirmatory Ab test. Acutes have the same laboratory signature during the ED encounter as those that are false positive (False +), and the two patient groups are denoted as "equivocals" until viral load testing specifies a definitive HIV status. Among False + patients (Ab/Ag positive, Ab negative, viral load undetectable), there have been limited studies on those also demonstrating a reduction in CD4+ count, an uncommon phenomenon known as "idiopathic CD4 lymphocytopenia." We review a patient with a reactive fourth-generation HIV Ab/p24 Ag test on two separate occasions. Despite lymphopenia with a reduced CD4 count, his symptoms resolved, and an RNA PCR test did not detect any presence of HIV (False +). This patient was unique as False + patient with p24 Ag reactive, as well as a coincidental low CD4 count in the absence of HIV infection. A low CD4 count is often a sign of significant HIV infection.
Hypoparathyroidism, Sensorineural Deafness, and Renal Disease Syndrome Presenting With Febrile Seizures and Hypocalcemia
Authors: Anne Gandolfi, Kevin Ratnasamy, Carla Minutti
Journal: JCEM Case Rep (2023)
Keywords: barakat syndrome, gata3, hdr syndrome, hypocalcemia, hypoparathyroidism
MeSH terms: Case Reports

HDR syndrome is a rare genetic disorder caused by mutations in the GATA3 gene and characterized by hypoparathyroidism, sensorineural deafness, and renal disease. Here, we report case of a 9-month-old male with history of hydronephrosis and sensorineural deafness who presented with febrile seizures. He was found to have hypocalcemia and inappropriately normal parathyroid hormone. His neurologic and infectious workup were negative. Genetic testing revealed a nonsense mutation in the GATA3 gene, consistent with HDR syndrome. Hypocalcemia was responsive to calcium carbonate and calcitriol treatment. This case highlights hypocalcemia caused by hypoparathyroidism as a potential etiology of seizures. When hypoparathyroidism is detected with either hearing loss or renal disease, HDR syndrome should be considered, and other features of the syndrome should be investigated.
Case report: Waldenstrom macroglobulinemia with systemic amyloidosis as the main manifestation
Authors: Junjing Yin, Xia Zhou, Shuyuan Yu, Hongying Wu, Yuping Zhong
Journal: Front Med (Lausanne) (2024)
Keywords: waldenström macroglobulinemia, case report, diarrhea, lymphoplasmacytic cells, systemic amyloidosis
MeSH terms: Case Reports

Systemic amyloidosis is a rare protein misfolding and deposition disorder leading to progressive organ failure. Waldenström macroglobulinemia (WM) with systemic amyloidosis as the main manifestation is even rarer. The patient in this study presented with recurrent diarrhea and had not been diagnosed in other hospitals on multiple occasions. Later, his diarrhea worsened and was accompanied by sunken edema of both lower limbs and dizziness. Renal biopsy showed deposits of PAS light-staining material in the glomeruli, interstitium, and small arteries, which stained positively with Congo red. Cardiac ultrasound showed interventricular septum thickening of 17 mm, right ventricular wall myocardial thickening of approximately 0.6 cm, and septal thickening of approximately 0.5 cm, considering myocardial amyloidosis. Electromyography showed abnormal peripheral nerve conduction. Lymphoplasmacytic cells were found in the bone marrow. Taken together, he was diagnosed with WM. He was treated with a BR (Bendamustine + Rituximab) regimen. After 6 courses, the patient's discomfort was relieved, his weight gained 5 kg, the level of serum IgM and dFLC decreased, and cardiac and renal assessments were more relieved. The patient has been followed up for more than 1 month.
Directional Stimulus-Evoked Pallidal Electrophysiology in Primary Dystonia
Authors: Aditya V Boddu, Sarah Brinkerhoff, Adam E Bashir, Camerron M Crowder, Mohammed Awad, Christopher L Gonzalez, Harrison C Walker
Journal: Tremor Other Hyperkinet Mov (N Y) (2024)
Keywords: ano3, dbs, erna, deep brain stimulation, dystonia, globus pallidus
MeSH terms: Deep Brain Stimulation / methods, Dystonic Disorders / physiopathology, Dystonic Disorders / therapy, Globus Pallidus / physiopathology, Case Reports, Adult, Evoked Potentials / physiology, Female, Humans, Male, Middle Aged

Background:
Deep brain stimulation for dystonia improves motor symptoms but variable and delayed responses challenge patient selection, targeting, and device programming.
Case report:
Here we studied intracranial electrophysiology in a patient with primary dystonia and observed evoked resonant neural activity (ERNA) in the globus pallidus interna. These local stimulus-evoked potentials displayed refractory periods and paired-pulse facilitation at clinically relevant interstimulus intervals. Sensing from directional DBS contacts localized ERNA to an effective stimulation site in the ventral posterolateral portion of the pallidum.
Discussion:
To the best of our knowledge, this is the first observation of ERNA in the globus pallidus interna in a patient with primary dystonia. Stimulus-evoked activity could eventually guide both directional and adaptive stimulation for dystonia and other complex neuropsychiatric disorders.
Locating and Treating Three Calcified Canals in a Mandibular First Molar With Radix Entomolaris and Five Canals: A Case Report
Authors: He Liu, Ya Shen
Journal: Cureus (2024)
Keywords: endodontic treatment, middle mesial canal, permanent mandibular first molar, radix entomolaris, root canal calcification, root canal treatment
MeSH terms: Case Reports

The success of root canal treatment is highly contingent on the comprehensive shaping, cleaning, and filling of the entire root canal system. Failure to address one or more canals often results in an increased likelihood of post-treatment apical periodontitis. Typically, mandibular first molars feature two roots and three canals, but they may also exhibit anatomical variations, such as a mesial middle canal or radix entomolaris (RE). This article presents a case where three calcified canals in a mandibular first molar with RE and five canals were successfully located and treated.
Case report: a case report of excision of giant lipoma in the posterior neck
Authors: Aichao Du, Hongyu Wang, Junqiang Dai, Qiang Dong, Guoqiang Yuan, Yawen Pan
Journal: Front Oncol (2024)
Keywords: case report, giant lipoma, lipoma, neck, surgery
MeSH terms: Case Reports

Lipomas, benign tumors originating from the anomalous proliferation of adipocytes, predominantly emerge in regions rich in adipose tissue. However, their presence in the head and neck areas remains rare, constituting approximately 13% of all diagnosed lipoma cases. This study presents a case involving a substantial subcutaneous lipoma located at the posterior neck, measuring about 20 cm × 19 cm × 10 cm. The patient presented with swelling and pain in the back of the neck. And the considerable dimensions of this lipoma significantly impacted the patient's quality of life and aesthetic appearance. Concurrently, the patient exhibited symptoms indicative of degenerative cervical spine disease and cervical disc herniation. After admission, a comprehensive examination, including ultrasound, CT scan, and MRI, was conducted. Given the clinical complexity, the decision for surgical intervention was deemed essential. The surgical strategy entailed a meticulous total excision of the tumor through an incision made in the posterior cross-neck, coupled with the strategic removal of excess skin. To facilitate wound healing, postoperative management included the use of negative pressure drainage. Pathological examination conclusively identified the mass as a lipoma. Postoperative follow-ups indicated successful recovery, as evidenced by the restoration of the neck's aesthetic contour and the complete resolution of the previously observed restrictions in sagittal neck movement.
A good response to anti-PD-1 monoclonal antibody plus SBRT in a patient with PD-L1-negative recurrent advanced esophageal cancer: a long-term follow-up case report of a possible abscopal effect
Authors: Tao Hai, Jia Liu, Jialu Lai, Lin Zhou
Journal: Front Oncol (2024)
Keywords: abscopal effect, anti-pd-1 monoclonal antibody, case report, esophageal cancer, stereotactic body radiotherapy
MeSH terms: Case Reports

There are limited treatment options for recurrent advanced esophageal squamous cell carcinoma. A good response with a possible abscopal effect was observed in a patient with programmed death-ligand 1 (PD-L1)-negative recurrent advanced esophageal squamous cell carcinoma treated with an anti-PD-1 monoclonal antibody plus stereotactic body radiotherapy (SBRT). A 66-year-old male patient was diagnosed with recurrent advanced esophageal squamous cell carcinoma with multiple lung metastases (13 metastatic nodules in total) four months after completing radical radiotherapy plus concurrent and consolidated chemotherapy, and PD-L1 expression in the primary esophageal tumor was negative. This patient received 25 cycles of camrelizumab (an anti-PD-1 monoclonal antibody) in total plus upfront SBRT for two metastatic nodules, which was administered after the first cycle of camrelizumab. After this combined treatment, for most nontarget nodules, an obvious volume decrease and fuzzy change were observed, including two nodules that completely vanished. At the end of follow-up, the progression-free survival and duration of response of this patient were 34 months and 32 months, respectively. This case report indicated that an anti-PD-1 monoclonal antibody combined with SBRT was a promising therapeutic strategy for recurrent esophageal squamous cell carcinoma even in patients with negative PD-L1 expression.
Case Report: Mechanical hemolysis resulting from left ventricular outflow tract obstruction after aortic valve replacement relieved by transapical beating-heart septal myectomy
Authors: Qingwen Kang, Jie Tian, Ying Zhu, Wei Zhou, Xiang Wei, Yani Liu
Journal: Front Cardiovasc Med (2024)
Keywords: case report, echocardiographic imaging, hematuria, left ventricular outflow tract obstruction, transapical beating-heart septal myectomy
MeSH terms: Case Reports

Background:
Aortic stenosis (AS) in combination with left ventricular outflow tract obstruction (LVOTO) has occasionally been reported. However, making a precise diagnosis and successfully treating this combination is challenging due to the hemodynamic interaction between the two conditions.
Case summary:
A 56-year-old male patient who had been diagnosed with severe AS and asymmetric left ventricular hypertrophy underwent aortic valve replacement (AVR) and a conventional septal myectomy. Immediately after the procedure, significant systolic anterior motion and mitral regurgitation developed, necessitating a surgical mitral edge-to-edge repair. Ten days after the procedure, the patient developed hematuria and LVOTO, which was confirmed by echocardiography. Because the LVOTO might have been the cause of the hematuria, the patient underwent alcohol septal ablation, but this had little effect. Three months later, a transapical beating-heart septal myectomy (TA-BSM) was performed in our hospital. Postoperatively, the LVOTO had been significantly ameliorated and the hematuria had resolved.
Conclusion:
For patients with AS and LVOTO due to a hypertrophic interventricular septum, inadequate amelioration of the LVOTO after AVR may lead to severe hemolytic hematuria. TA-BSM is a minimally invasive, safe, and effective surgical procedure for ameliorating LVOTO in patients with aortic valve prostheses.