OpenMed/multicare-articles huggingface dataset:
Prosthetic Joint Infection due to Actinomyces species: A case series and review of literature
Authors: Ramez Dagher, Talha Riaz, Aaron J Tande, Douglas R Osmon, Anil Jagtiani, James M Steckelberg, Tad Mabry, Elie F Berbari
Journal: J Bone Jt Infect (2019)
Keywords: actinomyces spp, prosthetic joint infection, resection arthroplasty, two stage exchange

Background: Actinomyces prosthetic joint infections (APJIs) are rare and optimal medical and surgical treatment strategies are unknown. The purpose of our study was to characterize the demographics, risk factors, management and outcomes of patients with PJIs due to Actinomyces spp. Methods: Using a retrospective cohort study design, the medical records of all patients with Actinomyces spp. total hip or knee arthroplasty infection (APJI) seen at a single institution between January 1, 1969 and December 31, 2016 were reviewed. We abstracted information including patient demographics, co-morbidities, joint age, surgical history, microbiology, management and outcomes. A simultaneous literature search via PubMed was performed to identify cases of APJI published in literature and a descriptive analysis was performed. Results: Eleven cases were identified over a 47 year study period at our institution. Seven patients (64%) were female. The median age at the time of diagnosis of infection was 71 years (range, 57-89). The knee was involved in six cases (55%) followed by the hip in 5 (45 %) cases. Three cases had dentures, broken teeth, or poor dentition. Actinomyces odonotlyticus was the most commonly found subspecies at our institution. Median ESR and CRP values were 61mm/hr and 64 mg/L respectively. Eight (72%) patients were managed with 2 stage exchange. Most patients received a course of beta-lactam therapy for 6 weeks. Ten cases (91%) were free of failure after a median duration of follow-up of 2 years (range, 0.67 - 5 years). The median duration from joint arthroplasty to the onset of symptoms was 162 days, range (20-3318). Six (54%) had a history of prior PJI with a different microorganism at the same joint site and 4 patients had history of prior 2 stage exchange (36%). In the literature group, we identified 12 cases and the most common subspecies was Actinomyces israelii; most patients underwent two stage exchange and were treated with 6 weeks of beta lactam antibiotics. Conclusions: Based on our observational study, Actinomyces PJI presents as a late complication of TJR, may be associated with prior PJI at the index joint and antecedent dental manipulation may portend as an additional risk factor. Treatment includes two stage exchange and beta- lactam therapy for 6weeks. These results will help clinicians in improved understanding and management of APJIs which although are rare but warrant special attention as population with implanted joint arthroplasties continues to rise.
Thrombocytopenia with absent radii (TAR) syndrome in a female neonate: a case report
Authors: Olayemi Atinuke Alagbe, Adekunle Emmanuel Alagbe, Emmanuel Olugbenga Onifade, Temitope Olugbenga Bello
Journal: Pan Afr Med J (2019)
Keywords: thrombocytopenia, congenital, haemorrhage
MeSH terms: Thrombocytopenia / diagnosis, Upper Extremity Deformities, Congenital / diagnosis, Case Reports, Congenital Bone Marrow Failure Syndromes, Female, Hemorrhage / etiology, Humans, Infant, Newborn, Radius / diagnostic imaging, Radius / physiopathology, Thrombocytopenia / diagnostic imaging, Thrombocytopenia / physiopathology, Upper Extremity Deformities, Congenital / diagnostic imaging, Upper Extremity Deformities, Congenital / physiopathology, Absent radii and thrombocytopenia

Thrombocytopenia absent radius (TAR) syndrome is a rare congenital disorder that is consistently associated with skeletal abnormality and thrombocytopenic haemorrhage. This is a case of a neonate with bilateral absent radius and thrombocytopenia. The rarity of this case prompted this report.
Canthaxanthin retinopathy with visual loss: a case report and review
Authors: Robert A Beaulieu, Ronald E Warwar, Bruce M Buerk
Journal: Case Rep Ophthalmol Med (2013)

Canthaxanthin is a naturally occurring chemical, which is most commonly utilized as a colorant for food and dyes or a skin bronzing agent. Its most prevalent impact on human health is canthaxanthin retinopathy, which appears as birefringent, yellow to red crystals surrounding the macula. This occurs with increasing, dose-dependent exposure. Generally, patients remain asymptomatic and findings may only be evident on funduscopic examination. Cessation of canthaxanthin ingestion appears to reverse the retinopathy, but the time until crystal disappearance is variable. Despite a usually favorable outcome, long-standing visual changes may occur. We report a case of an 84-year-old woman with significant visual loss secondary to canthaxanthin retinopathy that ultimately improved upon cessation of the drug.
Primary omental torsion in children: case report
Authors: Khattala Khalid, Tenorkorang Snr Somuah, Elmadi Aziz, Rami Mohamed, Bouabdallah Youssef
Journal: Pan Afr Med J (2013)
Keywords: childhood, emergency, omental torsion, surgery
MeSH terms: Diagnostic Errors, Omentum / pathology, Peritoneal Diseases / diagnosis, Torsion Abnormality / diagnosis, Case Reports, Abdominal Pain / etiology, Appendicitis / diagnosis, Child, Diagnostic Imaging, False Negative Reactions, Humans, Male, Omentum / surgery, Peritoneal Diseases / surgery, Torsion Abnormality / surgery, Vomiting / etiology

Primary omental torsion is a rare cause of acute abdominal pain, we report a case of 10-year-old boy admitted with crampy abdominal pain, routine laboratory tests and plain abdominal radiography was normal, the patient underwent surgical exploration with the initial diagnosis of appendicitis, primary omental torsion was confirmed and the omentum was untwisted, with good postoperative evolution.
A lurking threat: transfer of peanut allergy through peripheral blood stem cell transplantation
Authors: Birka Brauns, Michael P Schön, Gerald Wulf, Martin Mempel
Journal: World Allergy Organ J (2016)
Keywords: allergy transfer, ige-mediated hypersensitivity, peanut allergy, stem cell transplantation
MeSH terms: Case Reports

Background:
There exist several reports of atopy and allergen-specific IgE-mediated hypersensitivity transferred by bone marrow transplantation, and it has been concluded that the transfer of allergic reactivity results from adoptive transfer of IgE-producing donor-derived B- and/or plasma cells. To the best of our knowledge we report the first case of peanut allergy after PBSCT.
Case presentation:
A 55-year-old anciently non allergic man with secondary acute myeloid leukemia (AML) received an allogeneic peripheral blood stem cell transplantation from a matched unrelated donor following reduced-intensity conditioning. On day 32 after PBSCT, while still on prophylactic systemic immunosuppression, the patient noticed a first episode of angioedema with swelling of the nasal and oral mucosa 30 min after consuming peanut puffs. In a second episode, eight months after PBSCT, he again developed angioedema, generalized pruritus and nausea within minutes after eating biscuits containing hazelnut and peanut. Moreover, after topical application of a peanut oil-containing ointment, the patient experienced facial erythema and angioedema. Nine months after PBSCT an evaluation for peanut allergy revealed a highly increased specific IgE to peanut of 75.9 kU/l. Accordingly, skin prick tests for peanut extract were also positive. In consequence, the patient was counseled to strictly avoid peanut-related products, and provided with an emergency set. No adverse allergic events have occurred since for an observation time of 15 months after PBSCT. The stem cell donor was contacted and confirmed intolerance to peanuts. His specific serum IgE pattern nine month after PBSCT harvest was analysed and showed similar sensitization profiles compared to those of the transplant recipient.
Conclusions:
Because of the close temporal association between the onset of allergic symptoms in the PBSC recipient it is reasonable to assume that the acquired peanut allergy had been transferred from the donor to the recipient by the PBSC graft.
Postoperative intussusception in a neonate with congenital cutis laxa and huge hiatal hernia
Authors: Mohajerzadeh Leily, Sadeghian Naser, Mirshemirani Aliraza, Khaleghnejad Tabari Ahmad, Rouzrokh Mohsen, Jafari Nahid
Journal: APSP J Case Rep (2014)
Keywords: congenital cutis laxa, hiatal hernia, postoperative intussusceptions
MeSH terms: Case Reports

Congenital cutis laxa is a genetically heterogeneous condition presenting in the newborn with loose, redundant skin folds, decreased elasticity of the skin, and general connective tissue involvement. A 2-day-old full term neonate with congenital cutis laxa presented with respiratory distress. Investigations revealed huge hiatal hernia. Patient underwent loose Nissen's fundoplication. In postoperative period patient developed intussusception which was manually reduced at re-surgery.
Efficacy of CRISPR-Based Gene Editing in a Sickle Cell Disease Patient as Measured through the Eye
Authors: Alexander Pinhas, Davis B Zhou, Oscar Otero-Marquez, Maria V Castanos Toral, Justin V Migacz, Jeffrey Glassberg, Richard B Rosen, Toco Y P Chui
Journal: Case Rep Hematol (2022)
MeSH terms: Case Reports

Sickle cell disease (SCD) exists on a phenotypic spectrum with variable genetic expressivity, making it difficult to assess an individual patient's risk of complications at any particular point in time. Current and emerging SCD treatments, including CRISPR-based gene editing, result in a variable proportion of affected red blood cells (RBCs) still vulnerable to sickling. Clinical serological indicators of disease such as hemoglobin, indirect bilirubin, and reticulocyte count and clinical metrics including number of emergency department visits and hospitalizations over time often fall short in their ability to objectively quantify ischemic disease activity and efficacy of treatments. Clearly, better clinical biomarkers are needed. The rapidly developing field of oculomics leverages the transparent nature of the ocular tissue to directly study the retinal microvasculature in order to characterize the status of systemic diseases. In this case report, we demonstrate the ability of optical coherence tomography angiography (OCT-A) to detect and measure micro-occlusive events within the retinal capillary bed before and after RBC exchange transfusion and following CRISPR-based gene editing, as an indicator of systemic ischemic disease activity and measure of treatment efficacy. The implications of these findings are discussed.
Kimura disease, a rare cause of inguinal lymphadenopathy: A case report
Authors: Xianwen Hu, Xue Li, Changwei Yang, Dandan Li, Jiong Cai, Pan Wang
Journal: Front Med (Lausanne) (2022)
Keywords: kimura’s disease, pet/ct, flfluoro18-labeled deoxyglucose, inguinal lymphadenopathy, magnetic resonance imaging
MeSH terms: Case Reports

Kimura's disease (KD) is a rare chronic granulomatous disease of unknown etiology that mainly involves damage to lymph nodes, soft tissues, and salivary glands. The clinical symptoms are mainly painless subcutaneous soft tissue masses, often involving head and neck lymph nodes and salivary glands, and are mainly characterized by diffuse eosinophilic infiltration, lymphocyte, and vascular proliferation. There are few reports in the literature that KD affects only inguinal lymph nodes. We report in this study a 41-year-old male patient who presented to the hospital for medical help with soft tissue masses in the groin. Magnetic resonance imaging (MRI) showed multiple abnormal soft tissue nodules around the iliac vessels in the left groin, and a contrast-enhanced scan showed obvious homogeneous enhancement. Diffusion-weighted imaging showed limited movement of water molecules and showed an obvious high signal. Fluoro18-labeled deoxyglucose positron emission tomography/computed tomography (18F-FDG PET/CT) was recommended for further evaluation of the patient's general condition, and the results showed that except for the radioactive uptake in the lesions in the left groin region, no obvious abnormality was found in the rest of the body. Based on these imaging findings, the patient was first suspected to have malignant lesions, and then the patient underwent histopathological examination, which was confirmed to be KD. Our case study suggests that KD affects only the inguinal lymph nodes is rare and should be considered as one of the imaging differential diagnoses for lymphadenopathy such as lymphoma, metastases, and Castleman's disease.
Chemotherapy Reverses Anti-PD-1 Resistance in One Patient With Advanced Non-small Lung Cell Cancer
Authors: Lingdi Zhao, Baozhen Ma, Yonghao Yang, Tiepeng Li, Lu Han, Quanli Gao
Journal: Front Oncol (2020)
Keywords: chemotherapy, immunotherapy, lung cancer, pembrolizumab, resistance
MeSH terms: Case Reports

Background: Programmed cell death protein 1(PD-1) blockade has become a standard second-line treatment option for patients with advanced non-small cell lung cancer (NSCLC) without a driver gene mutation. Previous clinical studies showed that the objective response rate (ORR) of PD-1 blockade as second-line treatment for patients with NSCLC was ~20%, and the median progression-free survival (PFS) was ~4 months, with most patients eventually developing a resistance to PD-1 blockade. Although the ORR to chemotherapy after PD-1 blockade resistance was relatively high, the survival time of patients could not be significantly prolonged. Clinical oncologists are unclear about which treatment regimen should be selected after PD-1 blockade failure. Here, we report about a patient with advanced NSCLC and initial PD-1 blockade resistance who was observed to have a rapid partial response (PR) following one dose of chemotherapy and subsequent PD-1 blockade treatment. Case presentation: A 70-year-old woman with a history of left lower lobe lung surgery in March 2018 (pathological stage T1N2M0, EGFR wild-type) presented to our hospital. After six cycles of adjuvant chemotherapy, multiple nodules in both the lungs developed, and were suspected to be metastatic lesions. After another 2 months, the nodules in both the lungs enlarged. From November 2018 to March 2019, the patient received six cycles of pembrolizumab, and computed tomography (CT) confirmed a progressive disease status. She was then managed with 260 mg/m2 albumin paclitaxel once every 3 weeks. Subsequently, chemotherapy was discontinued after one cycle owing to grade three neuromuscular toxicity. Follow-up CT revealed a stable disease in May 2019. She then received another six cycles of pembrolizumab, which resulted in a PR. Conclusion: Chemotherapy may play a role in reversing PD-1 blockade resistance. If failure of PD-1 blockade occurs at first, re-administration of PD-1 blockade may be implemented if first followed by several cycles of chemotherapy. Because there are few reports on the use of chemotherapy to reverse PD-1 resistance, it is necessary to conduct clinical studies with larger patient cohorts to investigate whether chemotherapy can reverse PD-1 blockade resistance.
A Remarkable Coexistence of Systemic Capillary Leak Syndrome and Diabetes in an 11-Year-Old Boy: A Case Report and Review of the Literature
Authors: Aysun Ata, Samim Özen, Damla Gökşen, Neslihan Edeer Karaca, Güzide Aksu, Necil Kütükçüler, Hüseyin Onay, Şükran Darcan
Journal: Case Reports Immunol (2020)
MeSH terms: Case Reports

Systemic capillary leak syndrome (ISCLS) is a rare disease characterized by unexplained reversible capillary hyperpermeability followed by hypoperfusion, hemoconcentration, and either hypoalbuminemia or total hypoproteinemia. An 11-year-old boy was admitted with vomiting, generalized edema, and hyperglycemia, which was preceded by 5 days of coryzal symptoms, lethargy, and oral aft, without fever. On physical examination, he had tachycardia and hypotension, with severe generalized systemic nonitchy edema, and the laboratory tests supported the conclusion that he had severe hemoconcentration with hemoglobin: 184 g/L, hematocrit: 51.3 %, urea: 20 mmol/L, blood glucose: 11.1 mmol/L, and albumin: 19 gr/L, with normal urine analysis. On the fourth day, the patient was diagnosed with ISCLS, by ruling out other causes of shock and hypoalbuminemia. Intravenous immunoglobulin (IVIG) treatment regimen was administered on two consecutive days (day five and day six). His edema decreased on the fifth day, and the patient was deemed clinically well. There was no compartment syndrome, rhabdomyolysis, or pulmonary edema in the recovery period. However, respiratory virus panel PCR was positive for respiratory syncytial virus (RSV) and enterovirus, which were thought to be the triggering cause of ISCLS. For the differential diagnosis of diabetes, his fasting serum glucose was 13.4 mmol/L, simultaneous C-peptide was 0.44 nmol/L, and HbA1c was 64 mmol/mol, and urine ketone was positive. However, antiglutamic acid decarboxylase, anti-insulin antibody, and islet cell antibody were negative. At the last outpatient visit, 22 months after the diagnosis, his insulin dose was still 0.4 IU/kg/day and HbA1c was 40 mmol/mol, and without prophylaxis, there was no ISCLS attack. Conclusion. Early recognition of ISCLS is important for therapeutic awareness, since it is very rare in childhood and occurs usually without any prior provoking factors in healthy children. With the increase in awareness of the disease, knowledge and experiences about pediatric patients may also increase. We think that our case will contribute to the literature since there have been no pediatric diabetic patients with ISCLS reported.
Diagnosis of paracardiac castleman disease by dynamic gadolinium-enhanced first pass perfusion magnetic resonance imaging
Authors: Andrew Crean, Narinder Paul, Naeem Merchant, Lianne Singer, Yves Provost
Journal: Clin Med Case Rep (2008)
Keywords: castleman disease, first pass perfusion, gadolinium
MeSH terms: Case Reports

Summary Castleman disease is an uncommon disorder affecting the lymphatic system and is characterised by atypical lymphocyte proliferation. The usual clinical presentation is of a solitary mass lesion, frequently within the thorax. A number of different imaging findings have been reported on CT and MRI. We present a case of paracardiac Castleman disease where the diagnosis was suggested by dramatic enhancement of the tumour mass during a dynamic MR perfusion sequence. To our knowledge this is the first report of the use of a first pass bolus tracking technique in the diagnosis of Castleman disease.
Case Report: Four Cases of Cortical/Brainstem Encephalitis Positive for Myelin Oligodendrocyte Glycoprotein Immunoglobulin G
Authors: Wan Wang, Juntao Yin, Zhiliang Fan, Juxian Kang, Jia Wei, Xiaoqian Yin, Shaohua Yin
Journal: Front Neurol (2022)
Keywords: brainstem encephalitis, cortical encephalitis, epileptic seizures, myelin oligodendrocyte glycoprotein, neuromyelitis optica spectrum disorder
MeSH terms: Case Reports

Aim:
Despite a significant improvement in the number of studies on myelin oligodendrocyte glycoprotein (MOG)-immunoglobulin G (IgG)-associated disorder (MOGAD) over the past few years, MOG-IgG-associated cortical/brainstem encephalitis remains a relatively uncommon and less-reported presentation among the MOGAD spectrum. This study aimed to report the clinical course, imaging features, and therapeutic response of MOG-IgG-associated cortical/brainstem encephalitis.
Methods:
Data of four patients who suffered from cortical encephalitis with epileptic seizures and/or brainstem encephalitis during the course of the disease were retrospectively collected and analyzed.
Results:
In this study, three male patients and one female patient, with a median age of onset of 21 years (ranging 20-51 years) were enrolled. An epileptic seizure was the main symptom of cortical encephalitis in these patients, while the manifestations of brainstem encephalitis were diverse. Cranial MRI demonstrated abnormal signals in unilateral or bilateral cortical or brainstem. Cerebrospinal fluid studies showed normal or mildly elevated leukocyte counts and protein levels, and a cell-based assay detected positive MOG-IgG in the serum of all patients. Two patients were misdiagnosed at the first attack, and both experienced a relapse. All of them accepted the first-line immunotherapy after a confirmed diagnosis and had a good outcome.
Conclusion:
Early suspicion of MOG-IgG-associated encephalitis is necessary for any patient with sudden onset of seizures or symptoms of brainstem damage, especially with lesions on unilateral/bilateral cortical or brainstem on brain MRI.
Incidental benign metastasizing leiomyoma in a patient with bone sarcoma: a case report
Authors: Zanndor Jacob Del Real-Romo, Carlos Montero-Cantú, Oscar Villegas-Cabello, José Antonio Díaz-Elizondo, Danae Reyes-Salas, Rene Palomo-Hoil, Guillermo Peralta-Castillo, David Martínez-Sánchez, Eduardo Flores-Villalba
Journal: Case Rep Surg (2014)

Background. The benign metastasizing leiomyoma is an exceptionally rare entity; it presents with ectopic leiomyoma nodules with a benign pattern. Symptoms vary according to the anatomic location. The diagnosis is histopathological, usually in patients with history of hysterectomy. Case Presentation. A 36-year-old female with 2-month history of left knee pain was diagnosed with bone fibrosarcoma. A CT scan showed pulmonary nodules. The patient started neoadjuvant chemotherapy. Conservative surgery of pelvic limb was achieved. A new CT scan reported pulmonary nodules that remained in relation to the previous CT. A nodule resection by thoracotomy and TOB (transoperative biopsy) was performed. The final pathology report described benign proliferative lesions consistent with benign metastatic leiomyoma. Conclusions. Benign metastatic leiomyoma is a rare condition presenting with uterine and extrauterine nodules most commonly in the lung. The diagnosis is histopathological. The surgical procedure must be reserved for selected patients.
Differential Diagnosis of Fulminant Myocarditis and Acute Coronary Syndromes in the Case of Failure of Coronary Angiography: A Case Report
Authors: Xiangfei Huang, Yi Gao, Fuzhou Hua, Jun Ying
Journal: Front Cardiovasc Med (2021)
Keywords: acute coronary syndromes, coronary angiography, coronary artery anomaly, extracorporeal membrane oxygenation, fulminant myocarditis, intra-aortic balloon pump
MeSH terms: Case Reports

Fulminant myocarditis (FM) is a severe disease with a rapidly progressive and life-threatening course caused mainly by viral infection. The symptoms, laboratory findings, and presence of ECG changes resemble acute coronary syndrome. Therefore, coronary angiography is usually helpful in making the appropriate diagnosis. However, failure to obtain complete coronary artery images due to coronary artery anatomic variations poses a challenge for the diagnosis of FM. Here, we report a case of FM preliminarily diagnosed as acute coronary syndrome (ACS) due to the presence of coronary artery anomaly.
Solitary plasmacytoma of mandible: an unusual bilateral presentation
Authors: Sandip Kulkarni, Jashika Adil Shroff, S M Meghana, Pournima Godge, Monica Yadav, Charudatta Shridhar Naik
Journal: Autops Case Rep (2021)
Keywords: bilateral, case report, mandible, multiple myeloma, plasma cells, plasmacytoma
MeSH terms: Case Reports

Plasmacytoma is a neoplastic proliferation of monoclonal plasma cells, which can present clinically as solitary bone neoplasm, extramedullary plasmacytoma, and multiple myeloma. The biological behavior of these tumors is variable from periods of clinical latency to rapid growth and progression from localized forms to more disseminated multiple myeloma. We present the case of solitary plasmacytoma of the mandible with rare bilateral involvement in a 65-year-old female patient. This paper highlights the importance of understanding the maxillofacial manifestations of the disease by the dentist for early diagnosis and thus better prognosis.