OpenMed/multicare-articles huggingface dataset:
The pacemaker-twiddler's syndrome: an infrequent cause of pacemaker failure
Authors: Mohammad Salahuddin, Fathima Aaysha Cader, Sahela Nasrin, Mashhud Zia Chowdhury
Journal: BMC Res Notes (2016)
MeSH terms: Equipment Failure, Pacemaker, Artificial, Postoperative Complications / diagnosis, Case Reports, Aged, 80 and over, Brugada Syndrome / physiopathology, Brugada Syndrome / therapy, Cardiac Conduction System Disease, Electrocardiography, Female, Humans, Patient Compliance, Postoperative Complications / diagnostic imaging, Postoperative Complications / physiopathology, Radiography, Thoracic, Syndrome

Background:
The pacemaker-twiddler's syndrome is an uncommon cause of pacemaker malfunction. It occurs due to unintentional or deliberate manipulation of the pacemaker pulse generator within its skin pocket by the patient. This causes coiling of the lead and its dislodgement, resulting in failure of ventricular pacing. More commonly reported among elderly females with impaired cognition, the phenomenon usually occurs in the first year following pacemaker implantation. Treatment involves repositioning of the dislodged leads and suture fixation of the lead and pulse generator within its pocket.
Case presentation:
An 87 year old Bangladeshi lady who underwent a single chamber ventricular pacemaker (VVI mode: i.e. ventricle paced, ventricle sensed, inhibitory mode) implantation with the indication of complete heart block, and presented to us again 7 weeks later, with syncopal attacks. She admitted to repeatedly manipulating the pacemaker generator in her left pectoral region. Physical examination revealed a heart rate of 42 beats/minute, blood pressure 140/80 mmHg and bilateral crackles on lung auscultation. She had no cognitive deficit. An immediate electrocardiogram showed complete heart block with pacemaker spikes and failure to capture. Chest X-ray showed coiled and retracted right ventricular lead and rotated pulse generator. An emergent temporary pace maker was set at a rate of 60 beats per minute. Subsequently, she underwent successful lead repositioning with strong counselling to avoid further twiddling.
Conclusion:
Twiddler's syndrome should be considered as a cause of pacemaker failure in elderly patients presenting with bradyarrythmias following pacemaker implantation. Chest X-ray and electrocardiograms are simple and easily-available first line investigations for its diagnosis. Lead repositioning is required, however proper patient education and counselling against further manipulation is paramount to long-term management.
Immediate latissimus dorsi pedicle flap reconstruction following the removal of an eight kilogram giant phyllodes tumour of the breast: a case report
Authors: Romesh Sarvanandan, Ramesh Thangaratnam, Ai Chen Leong
Journal: J Med Case Rep (2011)

Introduction:
Phyllodes tumors account for less than 1% of breast tumors in women, and giant phyllodes tumors are those that are larger than 10 cm in diameter. Removal of such large tumors places a huge burden on the surgeon to reconstruct a breast that is aesthetically acceptable by the patient. We report what may be the largest giant phyllodes tumor and, most likely, the first latissimus dorsi flap used to cover such a large defect caused by the resection.
Case presentation:
We report the case of a 36-year-old Malaysian woman who presented with a three-year history of gradually increasing swelling of the left breast, with skin changes. Examination revealed a huge, globular, lobulated mass measuring 400 mm by 350 mm. The patient had a mastectomy with an immediate latissimus dorsi pedicled myocutaneous flap reconstruction. The breast weighed 8.27 kg, and ex vivo, the tumor measured 280 mm by 250 mm by 180 mm. Histopathologic analysis confirmed the diagnosis as a giant phyllodes tumor. At 12-month follow-up, the patient reports no complications and is satisfied with the aesthetic outcome.
Conclusion:
Giant phyllodes tumors are very rare tumors that can reach up to 40 cm in diameter. Reconstruction of such a defect is a great challenge, and we report what we believe is the first latissimus dorsi flap to cover successfully a defect of approximately 400 mm by 350 mm.
Stress Cardiomyopathy Managed with Extracorporeal Support after Self-Injection of Epinephrine
Authors: Jeremy Bourenne, Bourenne Jeremy, Raphaëlle Fresco, Fresco Raphaëlle, François Kerbaul, Kerbaul François, Pierre Michelet, Michelet Pierre, Marc Gainnier, Gainnier Marc
Journal: Case Rep Crit Care (2017)
MeSH terms: Case Reports

A 28-year-old man was admitted to the ICU for self-injection of Epinephrine. This injection resulted in the rapid development of a catecholamine-induced cardiomyopathy (inverted Takotsubo) with a severe cardiogenic shock. The importance of ventricular dysfunction required the implementation of a temporary arteriovenous circulatory support until the recovery of myocardial stunning. This case allows redefining the role of circulatory assistance during cardiotropic agents intoxication.
Correct Identification of Ochrobactrum anthropi From Blood Culture Using 16rRNA Sequencing: A First Case Report in an Immunocompromised Patient in Mexico
Authors: Ma G Aguilera-Arreola, Martha L Ostria-Hernández, Enrique Albarrán-Fernández, Sara R Juárez-Enriquez, Cristina Majalca-Martínez, Beatríz Rico-Verdín, Enrico A Ruiz, María Del Socorro Ruiz-Palma, María R Morales-García, Araceli Contreras-Rodríguez
Journal: Front Med (Lausanne) (2018)
Keywords: ochrobactrum anthropi, ochrobactrum bacteraemia, ochrobactrum identification, ochrobactrum in humans, misidentifying brucella
MeSH terms: Case Reports

The present report describes the misidentification of Brucella spp. from a positive blood culture using traditional microbiology tests. A molecular test identified the bacterium as Ochrobactrum anthropi. According to the information available, this report is the first to include this type of case in Mexico.
High-Grade Leiomyosarcoma Arising in a Previously Replanted Limb
Authors: Tiffany J Pan, Liron Pantanowitz, Kurt R Weiss
Journal: Case Rep Oncol Med (2015)

Sarcoma development has been associated with genetics, irradiation, viral infections, and immunodeficiency. Reports of sarcomas arising in the setting of prior trauma, as in burn scars or fracture sites, are rare. We report a case of a leiomyosarcoma arising in an arm that had previously been replanted at the level of the elbow joint following traumatic amputation when the patient was eight years old. He presented twenty-four years later with a 10.8 cm mass in the replanted arm located on the volar forearm. The tumor was completely resected and pathology examination showed a high-grade, subfascial spindle cell sarcoma diagnosed as a grade 3 leiomyosarcoma with stage pT2bNxMx. The patient underwent treatment with brachytherapy, reconstruction with a free flap, and subsequently chemotherapy. To the best of our knowledge, this is the first case report of leiomyosarcoma developing in a replanted extremity. Development of leiomyosarcoma in this case could be related to revascularization, scar formation, or chronic injury after replantation. The patient remains healthy without signs of recurrence at three-year follow-up.
Intestinal Tuberculosis Masquerading as Crohn's Disease? A Case of Disseminated Tuberculosis after Anti-TNF Therapy for Suspected Crohn's Disease
Authors: Alexander P Abadir, James Y Han, Fady A Youssef
Journal: Case Rep Gastrointest Med (2019)
MeSH terms: Case Reports

Intestinal tuberculosis (ITB) and Crohn's disease (CD) very closely resemble each other in symptomatology, imaging, appearance, and pathology. While ITB is rare in the United States, its prevalence is significantly higher in endemic areas, thus presenting a diagnostic dilemma in immigrant populations from high-risk countries. This patient was diagnosed with CD and treated with anti-TNF agents after indeterminate screening for latent tuberculosis. He was then admitted with septic shock and intestinal perforation due to disseminated tuberculosis. This case demonstrates the importance the consideration of ITB when a patient with risk factors for TB fails to respond to treatment for CD.
Case report: Significant benefits of tislelizumab combined with anlotinib in first-line treatment of metastatic renal pelvic urothelial carcinoma with sarcomatoid carcinoma differentiation
Authors: Shibin Zhu, Chenhao Yu, Chongwei Wang, Guoqing Ding, Sheng Cheng
Journal: Front Oncol (2022)
Keywords: immune checkpoint inhibitor, pseudoprogression, renal pelvic urothelial carcinoma, sarcomatoid carcinoma differentiation, tislelizumab, tyrosine kinase inhibitor, upper tract urothelial carcinoma
MeSH terms: Case Reports

Background:
Renal pelvic urothelial carcinoma with sarcomatoid carcinoma differentiation is a very dangerous malignant tumor and extremely rare in clinical practice. In general, these tumors with a dismal prognosis, and there is no standard treatment.
Case presentation:
In this case, an 81-year-old male patient was diagnosed with right renal pelvic carcinoma. After an open right radical nephroureterectomy, postoperative pathological examination showed infiltrating urothelial carcinoma with sarcomatoid differentiation. Overexpression of programmed death ligand-1 by immunohistochemistry. The carcinoma recurred 4.5 months after surgery. After informed, tislelizumab combined with anlotinib were used as first-line treatment. The patients showed a clinical partial response that lasted for 20 months.
Conclusion:
This case demonstrates the efficacy of tislelizumab combined with anlotinib in patients diagnosed with metastatic renal pelvic urothelial carcinoma with sarcomatoid carcinoma differentiation. Moreover, to our knowledge, this is the first application of this treatment.
Suspected Transfusion Related Acute Lung Injury Improving following Administration of Tranexamic Acid: A Case Report
Authors: Stan Ryniak, Piotr Harbut, Anders Ostlund, Andrzej Mysiak, Jan G Jakobsson
Journal: Case Rep Anesthesiol (2014)

A 16-year-old woman with craniofacial injury developed severe acute respiratory failure under the primary reconstructive surgical procedure requiring several units of blood and plasma. A transfusion related acute lung injury (TRALI) was suspected and supportive treatment was initiated. Because of the severity of symptoms, acute extracorporeal membrane oxygenation (ECMO) was planned. During preparation for ECMO, a single intravenous dose, 1 g of tranexamic acid, was administered and a remarkable improvement was observed shortly thereafter. The patient was placed on ECMO for 16 hours. The further course was uncomplicated and the patient was discharged from ICU on the 6th day after admission fully and she recovered. A clinical improvement was observed in a timely fashion following the administration of tranexamic acid. The handling of a suspected TRALI and potential benefit from administration of tranexamic acid are discussed in this case report.
Endodontic Management of a Severely Dilacerated Mandibular Third Molar: Case Report and Clinical Considerations
Authors: Suraj Arora, Gurdeep Singh Gill, Priyanka Setia, Anshad Mohamed Abdulla, Ganapathy Sivadas, Vaishnavi Vedam
Journal: Case Rep Dent (2018)
MeSH terms: Case Reports

This article aims at providing an insight to the clinical modifications required for the endodontic management of severely dilacerated mandibular third molar. A 35-year-old patient was referred for the root canal treatment of the mandibular left third molar. An intraoral periapical radiograph revealed a severe curvature in both the canals. A wide trapezoidal access was prepared following the use of intermediate-sized files for apical preparation. Owing to increased flexibility, Hero Shaper NITI files were used for the biomechanical preparation and single cone obturation was carried out. Third molars owing to their most posterior location-limited access coupled with a severe curvature pose utmost clinical challenges require meticulous skill, advanced technology, and patience to achieve success.
Traumatic Fracture: Dislocation of Cervicothoracic Junction-Grand Round Presentation of C7-T1 Instabilities and Different Instrumentation Techniques
Authors: Mohammad A Alsofyani, Soufiane Ghailane, Sultan Alsalmi, Sreenath Jakinapally, Louis Boissière, Ibrahim Obeid, Jean-Marc Vital
Journal: Case Rep Orthop (2020)
MeSH terms: Case Reports

Introduction:
Acute traumatic cervicothoracic junction spinal lesions are rare disorders and poorly documented. We report a case of a traumatic cervicothoracic fracture-dislocation. We present our experience in the operative treatment of an unstable fracture-dislocation at the cervicothoracic junction. Materials and Method. A seventy-year-old man was transferred to our hospital. We found paresthesia in the corresponding dermatome of C7 and C8 bilaterally. Initial CT scan shows vertebral body fracture of T1 with retropulsion into the spinal canal and anteroposterior dislocation of cervicothoracic junction type C according to AOSpine subaxial injury. Traumatic disc material at C7-T1 was removed by anterior cervical discectomy and fusion of C6-T2. Fixation was done from C6 to T2 in the prone position.
Results:
At one-year postoperative follow-up, radiographs revealed bony fusion at the level of C7-T1, and the patient had no major functional disability.
Conclusion:
We opted for the ventral-dorsal approach in our case for maximum stabilization and to prevent mechanical complications.
Combined prosthesis with extracoronal castable precision attachments
Authors: Naveen Gupta, Abhilasha Bhasin, Parul Gupta, Pankaj Malhotra
Journal: Case Rep Dent (2013)

Satisfactory restoration in a patient with a partially edentulous situation can be challenging especially when unilateral or bilateral posterior segment of teeth is missing. Successful restoration can be done with various conventional and contemporary treatment options. One such treatment modality is attachment-retained cast partial dentures. This paper describes a case report of a patient with maxillary bilateral distal extension edentulous span restored with a cast partial denture having an extracoronal castable precision attachment (RHEIN 83 OT CAP attachments system).
Postpneumonectomy Compression of the Mitral Annulus: Rare Vascular Complication in Sportive Patient
Authors: David Debeaumont, Susana Bota, Jean-Marc Baste, Marie Bellefleur, Dimitri Stepowski, Florence Vincent, Tristan Bonnevie, Francis-Edouard Gravier, Marie Netchitailo, Catherine Tardif, Alain Boutry, Jean-François Muir, Jérémy Coquart
Journal: Case Rep Pulmonol (2016)

Numerous postpneumonectomy complications exist. We present a rare clinical case of postpneumonectomy exertional dyspnea revealing compression of the mitral annulus by the descending aorta. The patient was 42-year-old former smoker with pulmonary emphysema. He has been operated on, in 2012 (i.e., right pneumonectomy). Before the surgery, the patient was a recreational runner. However, after some months, it was difficult for the patient to resume running. Cardiopulmonary exercise testing indicated moderate exercise intolerance with important oxygen desaturation. More interestingly, a decrease of low oxygen pulse was noticed from the first ventilatory threshold with no electrical modification on the electrocardiogram. This decrease was indicative of a decline in stroke volume. The thoracic scan revealed a right pneumonectomy pocket with a liquid abnormal content. Moreover, the mediastinum had shifted toward the pneumonectomy space and the left lung was distended and emphysematous. Echocardiography revealed a major change in the mediastinal anatomy. The mitral annulus was observed to be compressed by the rear wall of the descending aorta. The diagnosis of postpneumonectomy syndrome or platypnea-orthodeoxia syndrome was ruled out in this patient. Mitral annular compression by the descending aorta is rare complication, which must be researched in patients with postpneumonectomy exertional dyspnea.
A Rare Case Report of Giant Urinary Bladder Stone Causing Recurrent Dysuria in a Woman
Authors: Rojan Adhikari, Hari Prasad Baral, Utsav Bhattarai, Ravi Kiran Gautam, Kiran Jung Kunwar, Dipesh Shrestha, Bijay Mansingh Katwal
Journal: Case Rep Urol (2022)
MeSH terms: Case Reports

Background:
Large urinary bladder stones are not common and even less common in females. We report a case of large bladder stone presented with acute retention of urine in a female patient. Case Report. A 62-year-old female presented in emergency department with retention of urine for 12 hours with history of recurrent UTIs for last 1 year. She was also complaining of mild dull lower abdominal pain for last 6 months. She had no history of incontinence of urine and fever. On physical examination, hard mass was palpable on suprapubic region on palpation of abdomen. Urine culture shows Escherichia coli for which antibiotics was given. An X-ray kidney ureter bladder showed a radio-opacity in the pelvic region measuring 9 × 8 cm in size. Ultrasonography revealed bilateral mild hydronephrosis with a large bladder stone. Open cystolithotomy was performed, and the stone was taken out. Stone biochemical analysis showed predominantly urate crystals. Patient had uneventful postoperative course, and she was discharged on 4th postoperative day and was followed up for 1 months after operation.
Conclusions:
Large urinary bladder stones are not common and even less common in females. Clinician should have think regarding large bladder stone as a cause of recurrent lower urinary tract symptoms like dysuria and should assess renal function for proper treatment. Open cystolithotomy is choice of operation in large bladder stone.
Surgical Treatment of Intrapelvic Pseudotumour after Hip Resurfacing Arthroplasty: Case Report and Literature Review
Authors: Cristian Barrientos, Julian Brañes, José-Luis Llanos, Alvaro Martinez, Maximiliano Barahona
Journal: Case Rep Orthop (2018)
MeSH terms: Case Reports

Hip replacement is the surgery of the last century due to its impact on the quality of life. A pseudotumour is a rare complication of hip arthroplasty, and it is related to a metal-bearing surface. Pseudotumour is a challenging scenario for hip surgeons due to poor clinical outcomes. The patient consulted for hip pain and paresthesia in the left lower extremity, and analyses showed that the cause was a sizeable intrapelvic pseudotumour. A multidisciplinary team surgery was planned. At first, an infraumbilical approach was made to resect the intrapelvic-retroperitoneum portion of the pseudotumour. Then, a posterolateral hip approach was performed, to resect the remaining portion of the pseudotumour and revision arthroplasty. At five years of follow-up, there are no clinical or imaging signs of recurrence of the pseudotumour. Treatment evidence is limited to a series of cases and expert opinions; we encourage complete resection and revision arthroplasty.
Rhabdomyolysis and Acute Kidney Injury due to Severe Heat Stroke
Authors: Máximo H Trujillo, Carlos Fragachán G
Journal: Case Rep Crit Care (2011)
MeSH terms: Case Reports

We present a case of heat stroke (HS) and acute kidney injury (AKI) due to severe rhabdomyolysis in a 14-year-old previously healthy female patient. When she was practicing strenuous exercise she suffered acute seizures and high fever. These symptoms were followed by coma and multiple organ failure (MOF), which included AKI, encephalopathy, fulminant hepatic failure (FHF), and disseminated intravascular coagulation (DIC). The patient was managed in the ICU with renal replacement therapy, ventilatory support, and other vital supporting measures. After three weeks of ICU treatment she made a full recovery.
High Levels of IL-18 and IFN-γ in Chronically Inflamed Tissue in Chronic Granulomatous Disease
Authors: Virginia Meda Spaccamela, Rocio G Valencia, Oleksandr Pastukhov, Andrea Duppenthaler, Matthias S Dettmer, Juliane Erb, Urs C Steiner, Sven Hillinger, Carsten Speckmann, Stephan Ehl, Janine Reichenbach, Ulrich Siler
Journal: Front Immunol (2019)
Keywords: il-18/ifn-γ loop, chronic granulomatous disease, hyperinflammation, macrophage priming, macrophage re-priming
MeSH terms: Granulomatous Disease, Chronic / immunology, Interferon-gamma / immunology, Interleukin-18 / immunology, Macrophages / immunology, Paracrine Communication / immunology, Signal Transduction / immunology, Case Reports, Clinical Trial, Research Support, Non-U.S. Gov't, Adolescent, Adult, Child, Female, Granulomatous Disease, Chronic / genetics, Granulomatous Disease, Chronic / pathology, Humans, Infant, Inflammation / genetics, Inflammation / immunology, Inflammation / pathology, Interferon-gamma / genetics, Interleukin-18 / genetics, Macrophages / pathology, Male, Paracrine Communication / genetics, Signal Transduction / genetics, IFNG protein, human, IL18 protein, human, Interleukin-18, Interferon-gamma

Background: Chronic granulomatous disease (CGD) is caused by a malfunctioning nicotinamide adenine dinucleotide phosphate (NADPH) oxidase complex in phagocytes, leading to impaired bacterial and fungal killing and hyperinflammation. Objective: To characterize macrophage subsets and cytokine/chemokine signaling loops involved in CGD tissue hyperinflammation. Methods: Cytokine/chemokine production and surface marker expression were analyzed in inflamed tissue of four CGD patients and compared to cytokine/chemokine released by CGD macrophages upon priming to different macrophage subpopulations. Furthermore, the re-priming capacity of CGD pro-inflammatory M1 to M2a anti-inflammatory macrophages was evaluated. Results: In human CGD inflammatory tissue, IL-18 and IFN-γ were detected in significant quantity. Immunofluorescence analysis identified macrophages as one source of IL-18 in inflamed tissue. In vitro, CGD macrophages could be primed and re-primed into all inflammatory/anti-inflammatory macrophage subpopulations. IL-18 was also released by M1 CGD and control macrophages. Conclusion: CGD pro-inflammatory M1 macrophages remain M1 primed in vivo. As CGD M1 macrophages can be re-primed to anti-inflammatory M2a phenotype in vitro, macrophages are kept in M1 state in vivo by a persistent pro-inflammatory environment. Our results suggest a paracrine signaling loop between M1 macrophage derived IL-18 and non-macrophage derived IFN-γ maintaining macrophage pro-inflammatory activity in CGD tissue.
Spinal cord compression due to extramedullary hematopoiesis in a patient with E-beta-thalassemia managed without radiation or surgery
Authors: Van T La, Michael Diatte, Johnathan Gaston, Dallas Dick, Raed Sweiss, Zahra Pakbaz
Journal: J Community Hosp Intern Med Perspect (2018)
Keywords: e-beta-thalassemia, blood transfusion, extramedullary hematopoiesis, hydroxyurea, spinal cord compression
MeSH terms: Case Reports

Extramedullary hematopoiesis (EMH) in individuals with thalassemia is often the result of undertreated severe anemia. Radiation or surgery is often the chosen approach to handle spinal cord compression due to these paraspinal EMH elements. Our patient is a 28-year-old male with E-beta-thalassemia who presented with both upper thoracic and lower extremity symptoms of spinal cord compression and was successfully managed with the combination of transfusion and hydroxyurea. Given the variation in symptoms as a result of the sporadic location as well as the extent of these EMH elements along the spinal canal, the hematological communities will continue to benefit from case reports that offer treatment therapy.