OpenMed/multicare-articles huggingface dataset:
Permanent molar pulpotomy with a new endodontic cement: A case series
Authors: Saeed Asgary, Sara Ehsani
Journal: J Conserv Dent (2009)
Keywords: mature, pulpitis, pulpotomy
MeSH terms: Case Reports

The aim of this case series was to determine the clinical and radiographic success rate of pulpotomy, with new endodontic cement (NEC), in human mature permanent molar teeth. Twelve molars with established irreversible pulpitis were selected from patients 14 - 62 years old. The selection criteria included carious pulp exposure with a positive history of lingering pain. After isolation, caries removal, and pulp exposure, pulpotomy with NEC was performed and a permanent restoration was immediately placed. At the first recall (+1 day) no patients reported postoperative pain. One wisdom tooth had been extracted after two months because of failure in coronal restoration. Eleven patients were available for the second recall, with a mean time of 15.8 months. Clinical and radiographic examination revealed that all teeth were functional and free of signs and symptoms. Histological examination of the extracted teeth revealed complete dentin bridge formation and a normal pulp. Although the results favored the use of NEC, more studies with larger samples and a longer recall period were suggested, to justify the use of this novel material for treatment of irreversible pulpitis in human permanent molar teeth.
Combined adenocarcinoid and mucinous cystadenoma of the appendix: a case report
Authors: A Velusamy, S Saw, J Gossage, Str Bailey, J Schofield
Journal: J Med Case Rep (2009)

Introduction:
Adenocarcinoid of the appendix is a rare malignant tumour with features of both adenocarcinoma and carcinoid, showing both epithelial and endocrine differentiation. Mucinous cystadenoma is the commonest of the benign neoplasms of the appendix, with an incidence of 0.6% in appendicectomy specimens. We report a rare combination of these tumours and discuss the latest treatment options. To the best of our knowledge, only six cases have been reported in the literature to date.
Case presentation:
A 71-year-old Caucasian man presented to our department with a right iliac fossa mass associated with pain. Laparoscopy revealed an adenocarcinoid of the appendix in combination with mucinous cystadenoma. He underwent a radical right hemicolectomy with clear margins and lymph nodes.
Conclusion:
Adenocarcinoids account for 2% of primary appendiceal malignancies. Most tumours are less than 2 cm in diameter and 20% of them metastasize to the ovaries. The mean age for presentation is 59 years and the 5-year survival rate ranges from 60% to 84%. Right hemicolectomy is generally advised if any of the following features are present: tumours greater than 2 cm, involvement of resection margins, greater than 2 mitoses/10 high-power fields on histology, extension of tumour beyond serosa. Chemotherapy mostly with 5-Fluorouracil and Leucovorin is advised for remnant disease after surgery. Cytoreductive surgery with intraperitoneal chemotherapy can offer improved survival for advanced peritoneal dissemination.
Juvenile Systemic Lupus Erythematosus Presenting with Esophagitis and Severe Oral Mucositis
Authors: Emily Schildt, Kristen L Sessions, Deirdre De Ranieri
Journal: Case Rep Rheumatol (2021)
MeSH terms: Case Reports

We present a case of a previously healthy adolescent female who developed severe oral mucositis and acute esophagitis as her presenting symptoms of juvenile systemic lupus erythematosus. Mucositis involving the lips is infrequently reported in systemic lupus erythematosus, and to our knowledge, this is the first reported case of acute, non-infectious esophagitis as a presenting symptom in a pediatric systemic lupus erythematosus patient.
Case Report: Significant Response to Immune Checkpoint Inhibitor Camrelizumab in a Heavily Pretreated Advanced ER+/HER2- Breast Cancer Patient With High Tumor Mutational Burden
Authors: Rong Wang, Yuchen Yang, Wei-Wu Ye, Jianxing Xiang, Songan Chen, Wei-Bin Zou, Xiao-Jia Wang, Tianhui Chen, Wen-Ming Cao
Journal: Front Oncol (2021)
Keywords: ngs, tmb, biomarker, breast cancer, immunotherapy, molecular monitoring
MeSH terms: Case Reports

Endocrine treatment plus CDK4/6 inhibitors have become standard of care for estrogen receptor positive (ER+) breast cancer. Although immune checkpoint inhibitors (ICIs) have shown promising antitumor activity in a variety of cancer types, only limited success has been achieved for metastatic breast cancer (mBC) patients, especially the ER+ subtype, which usually exhibit lower tumor mutation burden (TMB) compared with other subtypes and therefore perceived as immunologically quiescent. Here we present a case of an ER+/HER2- but TMB-high mBC patient who had significant response to combination therapy with anti-PD-1 antibody camrelizumab and vinorelbine and obtained partial response (PR) with a progression-free survival (PFS) of 5 months after failure of multiple lines of therapy. Our case indicates that TMB may serve as a potential biomarker in immunotherapy selection for normally immunologically "cold" tumors such as ER+ mBC, also molecular monitoring during the whole treatment course plays an important role in patient management.
Bilateral true giant aneurysm of the profunda femoral artery: case report and review of the literature
Authors: Alessandro Robaldo, Mauro Maselli, Daniele Maggio
Journal: Case Rep Surg (2012)

We report a rare case of a bilateral true giant aneurysm of the profunda femoral artery aneurysms (PFAAs) in a 80-year-old man with a previous history of "open" abdominal aortic surgery and small bilateral popliteal artery aneurysm. In the English Literature only seven previously cases of true bilateral PFAAs are reported. Due to its location, this lesion may require surgical intervention and removal. The presentation, the diagnostic evaluation, and the surgical management of the aneurysm are discussed.
Double Intussusceptions in a 20-Year-Old Lady Harboring a Tubulovillous Adenoma with High-Grade Dysplasia: A Case Report and Literature Review
Authors: Fawaz Alkeraithe, Ahmad Alghamdi, Abdullah Algharbi, Anas Aluwishiq, Zeyad Alsolami
Journal: Cureus (2023)
Keywords: colonic adenoma, double, dysplasia, intussusception, tubulovillous
MeSH terms: Case Reports

Underlying malignancy is a concern when intussusception is diagnosed in adults and the elderly. Management includes oncological resection of the intussusception. We report a case of a 20-year-old female patient who presented with signs of bowel obstruction. Computed tomography demonstrated double intussusceptions (ileo-cecal and transverse colo-colonic). During laparotomy, the mid-transverse intussusception reduced spontaneously while the other did not. Both intussusceptions were managed with oncological resection. The final pathology showed a tubulovillous adenoma with high-grade dysplasia. As a result, intussusception in adults should be investigated thoroughly to exclude malignant potential.
Renal Tubular Acidosis in the Postpartum Period: A Case Report and Literature Review
Authors: Carlos E Duran, Mayra Estacio, Fredy Lozano, Esteban Echeverri, Maria Juliana Riascos, Juan Guillermo Posada, Johanna Schweineberg
Journal: Case Rep Nephrol (2021)
MeSH terms: Case Reports

Case Presentation. Distal renal tubular acidosis (dRTA) is characterized by impaired hydrogen ion secretion in the distal nephron resulting either from decreased net activity of the proton pump or from increased luminal membrane hydrogen ion permeability. Typical complications of dRTA include severe hypokalemia, normal anion gap metabolic acidosis, nephrolithiasis, and nephrocalcinosis. The patient is a 25-year-old woman in immediate puerperium with hypokalemia leading to paralysis, and the laboratory findings in this patients were concerning for dRTA. It is rare to encounter this entity during pregnancy, and the impact of this pathology is unknown.
Extensively Drug-Resistant Myroides odoratus in Critically Ill Patients: A Case Series and Literature Review
Authors: Melissa O'Neal, Caitlin E Labay, Jesse E Harris, William L Musick, Patricia L Cernoch, Kevin A Grimes, Jiejian Lin, Shivan Shah, Elizabeth Ramos-Salazar
Journal: Case Rep Infect Dis (2022)
MeSH terms: Case Reports

The bacterial genus Myroides, like other members of the Flavobacteriaceae family, consists of aerobic, non-motile, Gram-negative bacilli. Myroides spp. is considered predominantly opportunistic pathogens as, historically, most documented infections have been in immunocompromised individuals. Along with advancements in molecular assay testing, there are growing reports of clinically relevant Myroides spp. infections in immunocompetent individuals. These organisms display broad antimicrobial resistance, and while research into their mechanisms of resistance is progressing, genetic testing has revealed metallo-β-lactamases present in their genome. The sporadic identification of Myroides spp. and ongoing clarification of resistance patterns make empiric treatment difficult. This report documents two cases of extensively drug-resistant Myroides odoratus isolated from critically ill but otherwise immunocompetent patients followed by a review of available literature on Myroides spp. antibiotic sensitivities. Our findings indicate that minocycline and moxifloxacin have the highest documented in vitro activity against Myroides spp.
Three-Dimensional Printed Model and Virtual Reconstruction: An Extra Tool for Pediatric Solid Tumors Surgery
Authors: Ángela Sánchez-Sánchez, Óscar Girón-Vallejo, Ramón Ruiz-Pruneda, Maria Fernandez-Ibieta, Darío García-Calderon, Vanesa Villamil, María Cristina Giménez-Aleixandre, Carlos Andrés Montoya-Rangel, Juan Pedro Hernández Bermejo
Journal: European J Pediatr Surg Rep (2018)
Keywords: 3d printing, oncology, pediatrics, stereolithography, virtual reconstruction
MeSH terms: Case Reports

Introduction Three-dimensional (3D) technology is increasingly applied for planning challenging surgical interventions. We report our experience using 3D printing and virtual reconstruction for surgical planning of complex tumor resections in children. Methods Data were obtained from preoperative magnetic resonance. imaging analysis and 3D virtual recreations were performed using specialized computer software. 3D real-scale geometry models, including tumor, adjacent organs, and relevant vascularization, were printed in colorimetric scale and different materials for optimal structures discrimination. Results Four complex cases were selected. The first case was a bilateral Wilms tumor. The volumetric reconstruction proved the presence of enough healthy renal tissue, allowing bilateral nephron-sparing surgery. In the second case, reconstruction contributed to the location of pulmonary metastases. The third case was an abdominal neuroblastoma stage L2. The 3D model was of high value for planning and as a reference during the intervention. The last case is a cervico-thoracic neuroblastoma with an anatomopathological diagnosis of ganglioneuroma, located at the cervico-mediastinal juncture, in close relationship with the cervical vessels. Conclusions 3D reconstruction and the full-scale printing models are a useful tool in cases of complex tumor resections as they contribute to a better understanding of the relationships between the tumor and adjacent organs, helping to anticipate certain surgical complications. They also provide additional information to conventional imaging tests, being able to influence therapeutic decisions and facilitate the understanding by the family, improving doctor-patient communication.
Open-Label Fosmetpantotenate, a Phosphopantothenate Replacement Therapy in a Single Patient with Atypical PKAN
Authors: Yiolanda-Panayiota Christou, George A Tanteles, Elena Kkolou, Annita Ormiston, Kostas Konstantopoulos, Maria Beconi, Randall D Marshall, Horacio Plotkin, Kleopas A Kleopa
Journal: Case Rep Neurol Med (2017)
MeSH terms: Case Reports

Objective. Pantothenate kinase-associated neurodegeneration (PKAN) is an autosomal recessive disorder with variable onset, rate of progression, and phenotypic expression. Later-onset, more slowly progressive PKAN often presents with neuropsychiatric as well as motor manifestations that include speech difficulties, progressive dystonia, rigidity, and parkinsonism. PKAN is caused by biallelic PANK2 mutations, a gene that encodes pantothenate kinase 2, a regulatory enzyme in coenzyme A biosynthesis. Current therapeutic strategies rely on symptomatic relief. We describe the treatment of the first, later-onset PKAN patient with oral fosmetpantotenate (previously known as RE-024), a novel replacement therapy developed to bypass the enzymatic defect. Methods. This was an open-label, uncontrolled, 12-month treatment with fosmetpantotenate of a single patient with a later-onset, moderately severe, and slowly progressive form of PKAN. Results. The patient showed improvement in all clinical parameters including the Unified Parkinson's Disease Rating Scale (UPDRS), Barry-Albright Dystonia Scale, the EuroQol five-dimensional three-level (EQ-5D-3L) scale, timed 25-foot walk test, and electroglottographic speech analysis. Fosmetpantotenate was well-tolerated with only transient liver enzyme elevation which normalized after dose reduction and did not recur after subsequent dose increases. Conclusions. Fosmetpantotenate showed promising results in a single PKAN patient and should be further studied in controlled trials.
Ehlers-Danlos Syndrome Type VIII: A Rare Cause of Leg Ulcers in Young Patients
Authors: Sophie Ronceray, Juliette Miquel, Antoine Lucas, Gérald E Piérard, Trinh Hermanns-Lê, Anne De Paepe, Alain Dupuy
Journal: Case Rep Dermatol Med (2013)

Ehlers-Danlos syndrome type VIII (EDS-VIII) is a very rare autosomal dominant disease characterized by early-onset periodontitis associated with features of Ehlers-Danlos syndrome. We report a 32-year-old man whose chronic leg ulcer led to the diagnosis of EDS-VIII. He had severe periodontitis with complete loss of permanent teeth and skin fragility with thin skin, atrophic scars, and brownish atrophic pretibial plaques. Leg ulcer is not a prominent feature of EDS-VIII. We suggest adding EDS-VIII to the list of rare diseases accounting for chronic leg ulcers, if this case report prompts others to report leg ulcers associated with EDS-VIII.
An uncommon cause of seizures in children living in developed countries: neurocysticercosis--a case report
Authors: Irene Raffaldi, Carlo Scolfaro, Federica Mignone, Sonia Aguzzi, Federica Denegri, Pier-Angelo Tovo
Journal: Ital J Pediatr (2011)
MeSH terms: Frontal Lobe, Neurocysticercosis / complications, Seizures / etiology, Case Reports, Child, Developed Countries, Diagnosis, Differential, Humans, Italy, Magnetic Resonance Imaging, Male, Neurocysticercosis / diagnosis, Seizures / diagnosis

Neurocysticercosis represents an important cause of seizures in children in endemic countries, such as Latin America, Asia and sub-Saharan Africa, while in Europe, especially in Italy, the cases of neurocysticercosis are anectodal. We report the case of a 6 year old boy, born and lived for four years in Cameroon, who presented a right emiconvulsion. The diagnosis was neurocysticercosis. This case accentuates the need to consider neurocysticercosis in a child presenting with non febrile seizures, mainly if he emigrated from an area of high prevalence or if he had long-term stay in endemic regions.
Cervical neurofibromatosis with quadriparesis: Management by fibular strut graft
Authors: Wichien Laohacharoensombat, Wiwat Wajanavisit, Patarawan Woratanarat
Journal: Indian J Orthop (2010)
Keywords: cervical kyphosis, cervical neurofibromatosis, cervical spondyloptosis, fibular strut graft

This is a case report of an eight-year old boy with neurofibromatosis and a 120 degrees dystrophic kyphosis of the cervical spine. He presented with progressive quadriparesis caused by spondyloptosis of the C2/C3, and was successfully treated by skull traction and one-stage anterior fibular strut graft lying between the tubercle of the atlas through the C2 body slot and lower vertebrae. At seven years follow-up there was, loosening of lower vertebral screws which allowed growth and residual mobility of lower vertebral joints while the fusion of upper cervical spines was still solid.
TMEM126A mutation in a Moroccan family with autosomal recessive optic atrophy
Authors: Julie Désir, Frauke Coppieters, Nicole Van Regemorter, Elfride De Baere, Marc Abramowicz, Monique Cordonnier
Journal: Mol Vis (2012)
MeSH terms: Membrane Proteins / genetics, Optic Atrophy / genetics, Case Reports, Research Support, Non-U.S. Gov't, Adolescent, Adult, Chromosomes, Human, Pair 11, Consanguinity, Genes, Recessive, Genetic Loci, Heterozygote, Homozygote, Humans, Optic Atrophy / ethnology, Pedigree, Sequence Analysis, DNA, Siblings, Membrane Proteins, TMEM126A protein, human

Purpose:
Nonsyndromic autosomal recessive optic atrophy (arOA) is extremely rare and its existence was disputed until a locus, optic atrophy 6 (OPA6), was mapped to 8q. Recently, a second locus, OPA7, was found on 11q in several families from North Africa, with one presumably ancestral mutation of transmembrane protein 126A (TMEM126A). Here we report an independently ascertained large consanguineous family of Moroccan descent with three siblings affected with nonsyndromic arOA.
Methods:
Assuming autosomal recessive inheritance, we identified a locus on 11q with homozygosity mapping, with a multipoint logarithm of the odds score of 3.84, and sequenced two candidate genes. Direct sequencing of the complete coding sequence of TMEM126A revealed mutation p.Arg55X, homozygous in all affected siblings and heterozygous in both unaffected parents.
Results:
This mutation was identical to that recently reported in families from North Africa, consistent with a single ancestral origin. In contrast to the recently reported patients, however, the siblings reported in this study had a relatively mild clinical course, with sudden onset in adolescence in the proband. Interestingly, the proband, but not the other affected siblings, had sensory-motor axonal neuropathy with electrophysiological data strongly suggestive of focal demyelinating abnormalities. An unaffected sibling had transient loss of vision after exercise, i.e., Uhthoff's sign of optic neuropathy, and was found to be a heterozygous carrier of the mutation.
Conclusions:
Our results confirm genetic heterogeneity in arOA, illustrate clinical variability between families with the p.Arg55X mutation including the description of a mild phenotype in a heterozygote, and underscore the implication of mitochondrial proteins in optic and peripheral neuropathy.
Recrudescence of Focal Stroke Symptoms during Pain Management with Hydromorphone
Authors: Joshua D Bernstock, Craig S Budinich, Leonardo G Cohen, Oluwole O Awosika
Journal: Front Neurol (2016)
Keywords: opioids, pain management, plasticity, recrudescence of symptoms, stroke
MeSH terms: Case Reports

Patients who have recovered from a prior stroke may experience a reemergence of their original stroke syndrome secondary to metabolic derangements, sedation, infection, and/or fatigue. Critically, the molecular/cellular mechanisms mediating symptom recurrence after exposure to analgesic agents remain unknown. Accordingly, herein, we report a unique case of a patient with hydromorphone-induced recrudescence 30 years after her initial stroke event(s) and in so doing propose a putative mechanism related to post-infarction functional neuroplasticity.
Full Digital Workflow in the Esthetic Dental Restoration
Authors: Suchada Kongkiatkamon, Dinesh Rokaya
Journal: Case Rep Dent (2022)
MeSH terms: Case Reports

This case report presented a fully digital workflow in esthetic dental restoration. A 51-year-old female patient was referred to BDMS Wellness Clinic due to a fracture of the maxillary left central incisor. An immediate dental implant was planned to restore tooth 21 with esthetic crown lengthening of upper front teeth and new zirconia crowns for teeth 11, 12, 13, 21, 22, and 23. Digital impression was made using a digital scanner (PRIMESCAN®, Dentsply Sirona, Bensheim, Germany); surgical guide (Cerec Guide 3, Dentsply Sirona, Bensheim, Germany) was designed by using a designing software (Galileos Software, Dentsply Sirona, Bensheim, Germany) and was milled by using a milling machine (MCXL milling machine, Dentsply Sirona, Bensheim, Germany) to create a precise surgical guide. 3D smile design was done by using the Digital Smile Design (DSD) program, the crown lengthening guide was designed according to DSD designed by using a designing software (Inlab 19 Software, Dentsply Sirona, Bensheim, Germany), and the guide was fabricated by a 3D printer (FormLabs Form 2, Formlab, MA, USA). Provisional crowns (splinted) for teeth 11, 12, 13, 21, 22, and 23 were milled by using polymer (VIPI BLOCK TRILUX®, VIPI Industria, Pirassununga, SP, Brazil) using a milling machine (MCX5, Dentsply Sirona, Bensheim, Germany). The zirconia crowns were designed by using software (Inlab19 Software, Dentsply Sirona, Bensheim, Germany) and milled using the same milling machine. At the implant position, Ti-base is cemented by using an abutment (Multilink Hybrid Abutment, Ivoclar Vivadent, Schaan, Lichtenstein, Germany) with zirconia coping (Cercon base white shade, Dentsply Sirona, Bensheim, Germany) utilized as the abutment. The zirconia crowns (Cercon Xt, Dentsply Sirona, Bensheim, Germany) were sintered and characterized and then cemented. The patient was satisfied with the esthetic outcome of the treatment.
Delayed bleeding and pelvic haematoma after low-energy osteoporotic pubic rami fracture in a warfarin patient: an unusual cause of abdominal pain
Authors: Andrea Sandri, Dario Regis, Nicola Bizzotto
Journal: Case Rep Emerg Med (2014)

Introduction. Acute abdominal pain may be the presenting symptom in a wide range of diseases in the elderly. Acute abdominal pain related to a delayed bleeding and pelvic haematoma after a low-energy pubic rami fracture is rare and can have important consequences; to the best of our knowledge, only one case has been previously described. Case Report. We present an unusual case of an 83-year-old woman taking warfarin for atrial fibrillation, admitted to the Emergency Department (ED) with acute abdominal pain and progressive anemia related to a delayed bleeding and pelvic haematoma 72 hours after a low-energy osteoporotic pubic rami fracture. Warfarin was withheld, anticoagulation was reversed by using fresh frozen plasma and vitamin K, and concentrated red blood cells were given. Haemoglobin level gradually returned to normal with a progressive resorption of the haematoma. Conclusion. Delayed bleeding and pelvic haematoma after osteoporotic pubic rami fracture should be considered in the differential diagnosis of acute abdominal pain in the elderly. This case indicates the need for hospital admission, careful haemodynamic monitoring, and early identification of bleeding in patients with "benign" osteoporotic pubic rami fracture, especially those receiving anticoagulants, to provide an adequate management and prevent severe complications.