OpenMed/multicare-articles huggingface dataset:
Autoinflammatory Reaction in Dogs Treated for Cancer via G6PD Inhibition
Authors: Jonathan W Nyce
Journal: Case Rep Vet Med (2017)
MeSH terms: Case Reports

Glucose-6-phosphate dehydrogenase (G6PD) is an oncoprotein that is overexpressed in cancer cells to provide the NADPH required for their increased anabolism. NADPH, sourced from G6PD fuels nucleotide biosynthesis, maintains redox potential of thioredoxin and glutathione and drives the mevalonate pathway that powers many of the basic mechanisms by which cancer cells escape host control. G6PD is thus a target for cancer treatment being addressed by many groups around the world. We have discovered that systemic inhibition of G6PD by high dose dehydroepiandrosterone (DHEA) causes a severe autoinflammatory response in dogs, which does not occur in mice or rats. Since dogs more closely model the human adrenal androgen system than do common laboratory animals, this finding is relevant to the design of G6PD-inhibiting drugs for humans. The autoinflammatory reaction observed closely resembles mevalonate kinase deficiency (MKD), a rare autosomal recessive disease in humans characterized by recurrent febrile attacks, arthralgia, skin rash, and aphthous ulcers of mucocutaneous tissues. In a manner comparable to animal models of MKD, the reconstitution of protein geranylgeranylation blocked the autoinflammatory reaction caused by systemic G6PD inhibition. This autoinflammatory response to systemic G6PD inhibition represents an unexpected result that must be taken into consideration when targeting this oncoprotein.
Direct Midline Diastema Closure with Composite Layering Technique: A One-Year Follow-Up
Authors: Bora Korkut, Funda Yanikoglu, Dilek Tagtekin
Journal: Case Rep Dent (2016)

Objective. Maxillary anterior spacing is a common aesthetic complaint of patients. Midline diastema has a multifactorial etiology such as labial frenulum, microdontia, mesiodens, peg-shaped lateral incisors, agenesis, cysts, habits such as finger sucking, tongue thrusting, or lip sucking, dental malformations, genetics, proclinations, dental-skeletal discrepancies, and imperfect coalescence of interdental septum. Appropriate technique and material for effective treatment are based on time, physical, psychological, and economical limitations. Direct composite resins in diastema cases allow dentist and patient complete control of these limitations and formation of natural smile. Clinical Considerations. In this case report a maxillary midline diastema was closed with direct composite resin restorations in one appointment without any preparation. One bottle total etch adhesive was used and translucent/opaque composite resin shades were layered on mesial surfaces of the teeth that were isolated with rubber dam and Teflon bands. Finishing and polishing procedures were achieved by using polishing discs. Patient was informed for recalls for every 6 months. Conclusions. At one-year recall no sensitivities, discolorations, or fractures were detected on teeth and restorations. Direct composite resins seemed to be highly aesthetic and durable restorations that can satisfy patients as under the conditions of case presented.
Multimodal postpartum imaging of a severe case of Couvelaire uterus
Authors: Josef Jackson, Verghese George, Jennifer McKinney, Karin A Fox
Journal: Case Rep Perinat Med (2022)
Keywords: couvelaire uterus, magnetic resonance imaging, retained placenta, ultrasound
MeSH terms: Case Reports

Objectives:
Placental abruption occurs when a normally implanted placenta prematurely separates, causing rupture of decidual spiral arteries and retroplacental hemorrhage. Estimates place the incidence of placental abruption somewhere between 0.22% and 1% of all deliveries. Clinical abruption represents a spectrum from mild to the most severe form, in which blood can extravasate into or through the myometrium, the broad ligament, or the peritoneum, causing the uterus and surrounding structures to take on a blue discoloration. This phenomenon is a clinical entity known as Couvelaire uterus, so named because it was first described by French physician Alexandre Couvelaire in the early 20th century as "uteroplacental apoplexy." Its incidence is difficult to estimate because it has classically been diagnosed only by direct intraoperative visualization. Imaging is not usually indicated in this clinical setting, so radiologic correlation with operative findings has not been previously described.
Case presentation:
In this report, we discuss the case of a multipara who presented with abdominal pain and vaginal discharge several days after a classical cesarean delivery. Her prolonged and complex clinical course led to evaluation via several radiologic modalities. At first, a focal placenta accreta or retained products of conception were suspected, however these diagnoses did not correlate with the patient's reported intraoperative findings of a clean endometrial cavity or with histopathology that was consistent with massive abruption.
Conclusions:
The clinical presentation and features identified on multimodal imaging were ultimately most consistent with the patient's intraoperative diagnosis of Couvelaire uterus.
Percutaneous Valve-in-Valve Treatment of a (Very Old and Fluoroscopy Invisible) Degenerated Tricuspid Prosthesis Through the Right Jugular Vein Approach
Authors: Cristina Aurigemma, Francesco Burzotta, Michele Corrado, Christian Colizzi, Carlo Trani
Journal: Front Cardiovasc Med (2019)
Keywords: degenerated prosthesis, percutaneous valve in valve, tricuspid regurgitation, tricuspid stenosis, tricuspid valve
MeSH terms: Case Reports

Tricuspid valve dysfunction adversely affects prognosis and may cause severe symptoms. Among the different opportunity offered by transcatheter techniques, the valve in valve represents an emerging strategy to treat patients with degenerated surgical biological prosthesis. We describe a case report of a percutaneous valve in valve treatment of a very old and fluoroscopy invisible tricuspid degenerated bioprosthesis. In the reported case, pivotal issue for percutaneous valve in valve procedure success was the achievement of perfect alignment between transcatheter valve and degenerated bioprosthesis despite the horizontal right chamber axis and the poor valve visibility. Of note, the combination of jugular vein approach, transapical delivery system rotation, right ventricle guidewire placement, and right atrium angiography made the valve in valve procedure safely.
GNQ-209P Mutation in Metastatic Uveal Melanoma and Treatment Outcome
Authors: Nagla Abdel Karim, Ihab Eldessouki, Ahmad Taftaf, Deeb Ayham, Ola Gaber, Abouelmagd Makramalla, Zelia M Correa
Journal: Case Rep Oncol Med (2018)
MeSH terms: Case Reports

Metastatic prognosis in uveal melanoma is assessed by gene expression profiling (GEP) testing of the tumor cells, usually obtained by fine needle aspiration (FNA). GEP has demonstrated high accuracy in distinguishing class I and II tumors, both having different metastatic potential. Transcriptomic studies identified distinct mutations including somatic mutations in GNAQ and GNA11, detected in more than 80%, and contribute to the upregulation of the mitogen-activated protein kinase (MAPK) pathway and the development of uveal melanoma (UM). The role of these mutations in treatment selection and possible benefit from targeted therapy are somewhat unclear. However, until the discovery of novel agents, local versus systemic therapies remain options for treatment that can still be considered for disease control in certain cases. We report a series of patients with metastatic UM with distinct mutational profiles. One had significant liver metastases with proven GNQ-209P mutation on tissue biopsy while peripheral blood molecular profiling did not show these mutations. The other three cases had no GNQ-209P mutation. All cases received nab-paclitaxel (Abraxane) as a treatment drug, and we record their responses to treatment and their molecular-profiling results.
Management of root fracture: a novel, noninvasive treatment approach
Authors: M S Rangareddy, Archana Daga, Y Vishnu Vardhan, M Daneswari
Journal: Case Rep Dent (2013)

Traumatic injuries to teeth account for approximately 25% of dental conditions where a patient seeks dentist for emergency treatment. Radicular fractures are one such entity which is very challenging to address due to various complications like periodontal communication, increased mobility, and continued pulpal infection leading to necrosis. Radicular fractures in the middle third have long been considered teeth of salvage due to their unfavourable fracture pattern. During the recent years introduction of biomimetic materials has opened the horizon for saving these teeth. In the present case report a novel approach to the management of radicular fractures in the middle third has been presented.
Pediatric chronic myeloid leukemia in myeloid blast crisis
Authors: Biswajit Dey, Anirban Dutta
Journal: Autops Case Rep (2023)
Keywords: blast crisis, leukemia, myelogenous, chronic, bcr-abl positive, leukemia, myeloid, leukemia, myeloid, acute, pediatrics
MeSH terms: Case Reports

Chronic myeloid leukemia (CML) accounts for 2-3% of childhood leukemias. About 5% of cases present in a blastic phase of CML which clinically and morphologically mimics more common acute leukemias of childhood. We report a case of a 3-year-old male who presented with gradual onset swelling of the abdomen and extremities along with generalized weakness. Examination revealed massive splenomegaly, pallor, and pedal edema. Initial workup showed anemia, thrombocytopenia, and leukocytosis (120,000/uL) with a blast percentage of 35%. Blasts were positive for CD13, CD33, CD117, CD34 and HLA-DR, and stained negative for Myeloperoxidase and Periodic Acid Schiff. Fluorescence in situ hybridization was positive for b3a2/e14a2 junction BCR-ABL1 transcript and negative for RUNX1-RUNX1T1/t(8;21), clinching the diagnosis of CML in myeloid blast crisis. The patient expired within 17 days of diagnosis and initiation of therapy.
Extraosseous Accumulation of Technetium-99m-Methyl Diphosphonate (99mTc-MDP) in a Child with ALL: A Case Report
Authors: Farnaz Banezhad, Narjess Ayati, Farrokh Seilanian Toosi, Samine Boloursaz, S Rasoul Zakavi
Journal: Asia Ocean J Nucl Med Biol (2018)
Keywords: acute lymphoblastic leukemia, bone scan, hypercalcemia, lung uptake, technetium-99m-methyl diphosphonate (99mtc-mdp)
MeSH terms: Case Reports

Extraosseous accumulation of technetium-99m-methyl diphosphonate (99mTc-MDP) on bone scan is not common. This phenomenon is often attributed to abnormality of calcium metabolism and has been reported in a variety of conditions including metabolic diseases and malignancies. A five years old boy is presented here, who was admitted to the pediatric emergency suffering from fatigue, respiratory symptoms, weight loss, intermittent fevers, anorexia, nausea and vomiting, edema of legs and abdominal distension for one month. The initial laboratory analysis revealed hypercalcemia. The patient was referred for whole body bone scan with suspicion of malignancy and bone metastasis. The bone scan revealed highly increased radiotracer uptake in both lungs in the perfusion and blood pool phases. Delayed images also showed increased activity in lungs and gastric wall. The skeleton was not seen clearly. Bone marrow aspiration was done and established the diagnosis of ALL. The patient deceased due to respiratory failure 20 days later. Diffuse lung uptake in this patient was consistent with respiratory failure and poor prognosis. It is reported that bone scan may be useful for assessment of the extent of metastatic calcification and may establish suitable management to prevent organ failure.
Lactobacillus paracasei endocarditis of bioprosthetic aortic valve presenting with recurrent embolic strokes
Authors: Abdulfatah Osman, Michael Taipale, Mazen Najjar, Baraa Osman
Journal: Access Microbiol (2019)
Keywords: lactobacillus paracasei, endocarditis
MeSH terms: Case Reports

Introduction:

Lactobacillus prosthetic valve endocarditis is a rare infection caused by Lactobacillus bacteria. This bacterium is found in the normal flora of the human mouth, gastrointestinal tract and female genital tract. While there have been isolated cases of Lactobacillus bacteraemia and endocarditis, the infections are associated with comorbidities, immune deficiency, dental manipulation procedures and other medical history. This case of bioprosthetic valve endocarditis caused by Lactobacillus paracasei is unusual, as the patient was immune-competent and treated with pre-procedural antibiotics.
Case:
We present a case of a 65-year-old male who underwent a dental extraction. He presented after 3 months of fever, chills and fatigue. On initial presentation, blood cultures were positive for alpha-haemolytic streptococcus bacteraemia. He was treated with IV penicillin and underwent aortic valve replacement with a bioprosthetic valve and excision of the mitral vegetation with repair of the mitral valve. Two years later, he had a tooth extraction after being treated properly with antibiotics. Three months later he presented with difficulty speaking, left leg weakness and increased drooling. All testing was normal. Three months later he presented with left side lower extremity weakness and expressive aphasia. He was diagnosed with bioprosthetic aortic valve endocarditis and was treated with IV penicillin and gentamicin for 6 weeks and then switched to oral penicillin. He remained stable.
Conclusions:

L. paracasei can potentially be a cause of complicated endocarditis in patients with prosthetic heart valves undergoing dental procedures. Timely culture-guided antibiotic therapy is critical and may obviate the need for valve surgery.
Parathyroid Carcinoma All-In-One, a Rare Life-Threatening Case With Multiple Systemic Manifestations: Case Report and Review of the Literature
Authors: Lorenzo Zelano, Pietro Locantore, Carlo Antonio Rota, Caterina Policola, Andrea Corsello, Esther Diana Rossi, Vittoria Rufini, Luca Zagaria, Marco Raffaelli, Alfredo Pontecorvi
Journal: Front Endocrinol (Lausanne) (2022)
Keywords: neuropathy, brown tumors, hungry bone, hypercalcemia, pancreatitis, parathyroid carcinoma, synovitis, venous thrombosis
MeSH terms: Hypercalcemia / complications, Hypercalcemia / diagnosis, Osteoporosis / complications, Parathyroid Neoplasms / complications, Parathyroid Neoplasms / pathology, Parathyroid Neoplasms / surgery, Case Reports, Review, Humans

Parathyroid carcinoma (PC) is an extremely rare disease. Although it may occasionally occur in genetic syndromes, it is more often sporadic. It is usually associated with a consistent secretion of PTH, causing severe hypercalcemia and potentially all clinical conditions due to primary hyperparathyroidism. Management of PC can be challenging: some clinical, biochemical, and radiological features may be useful, but the final diagnosis of malignancy strictly relies on histological criteria. To date, radical surgery is the first-choice treatment and is the only effective therapy to control hypercalcemia and other clinical manifestations. On the other hand, chemo- or radiotherapy, local treatments, or novel drugs should be reserved for selected cases. We report an exceptionally unusual case of life-threatening PC, associated with several systemic manifestations: moderate pancreatitis, portal thrombosis, kidney stones, brown tumors, osteoporosis, hungry bone syndrome (HBS), chondrocalcinosis, neuropathy, and depression. The clinical case also represents an opportunity to provide a review of the recent literature, associated with a complete evaluation of the main diagnostic and therapeutic approaches.
Recurrent Adult Sacrococcygeal Teratoma Developing Adenocarcinoma: A Case Report and Review of Literatures
Authors: Shengjie Cui, Jing Han, Binny Khandakar, Barak Friedman, Domingo Nunez, Gabriel A Sara, Gabriel S Levi
Journal: Case Rep Pathol (2021)
MeSH terms: Case Reports

Sacrococcygeal teratomas (SCT) are most commonly seen in infants and children but are rare in adults. Most adult SCT are benign and mature with a minority of tumors having immature components or overt malignancy. Here, we report a 65-year-old female with a SCT developing adenocarcinoma. The patient was diagnosed with benign sacrococcygeal cystic teratoma on her initial hospital visit and was treated with surgical resection. She was followed up postoperatively and was noted to have a markedly elevated CA 19-9 level 13 months after the surgery. Radiological and clinical examination revealed thickening of the perirectal soft tissues, located near the inferior portion of her previous incision site. Histological evaluation of the lesion showed invasive, moderately differentiated adenocarcinoma. Immunohistochemical staining results were suggestive, but not diagnostic, of anal gland adenocarcinoma. This case report expands the knowledge regarding an adenocarcinoma arising from a previously benign, adult SCT.
Case Report: Unusual Heterotopic Ossification of the Hindfoot
Authors: Falcioni Danya, Baldini Marco, Coppa Valentino, Marinelli Mario, Gigante Antonio Pompilio
Journal: Front Surg (2022)
Keywords: bone, case report, children, foot, heterotopic ossification
MeSH terms: Case Reports

Heterotopic ossification (HO) is a pathologic condition in which aberrant lamellar bone deposits in soft tissues, outside of the normal skeleton. Pathogenesis is still unclear, but different risk factors are known. Here we report a case of a 14 year-old girl presenting with pain in the medial calcaneal region and evidence of a rapidly growing, firm and solid neoformation. The lesion was diagnosed 6 years earlier, but it was consistently smaller and asymptomatic so that the patient did not undergo any follow up. The patient had no previous trauma or surgery, no other risk factors for HO and did not show any clinically evident HO in other districts. Xray and CT showed a heterogeneous bony lesion in the context of soft tissues, isolated from the calcaneus. After complete excision, histological analysis confirmed the diagnosis of HO. In conclusion, lone non congenital HO can occur regardless of known risk factors. Small HO lesion may also enter a proliferative phase without evidence of triggering events. More studies are required to better understand etiopathogenesis of HO in these clinical settings.
Restoration of a malformed primary incisor using digital technology in a pediatric patient with congenital Zika virus syndrome: A case report
Authors: Isabella Fernandes Carvalho, Louise Cristina Pereira Freitas, Phillipe Nogueira Barbosa Alencar, Maria Cláudia de Freitas Lima, Daniel de Sá Cavalcante, José Luciano Pimenta Couto, Paulo Tarcio Aded Silva, Dhaniel Anderson Olímpio Barbosa, Ellaine Dóris Fernandes Carvalho, Fabrício Bitu Sousa
Journal: J Dent Res Dent Clin Dent Prospects (2022)
Keywords: dental anomalies, indirect restorations, oral manifestations, zika virus
MeSH terms: Case Reports

Zika virus congenital syndrome (ZVCS) is a congenital viral infection resulting from the transmission of the Zika virus (ZV) to the fetus during pregnancy. This report describes a clinical case involving a 20-month-old female child with ZVCS, who presented with systemic changes related to the syndrome, such as microcephaly, arthrogryposis, ocular and auditory changes, and oral changes such as delayed dental eruption, ogival (high-arched) palate, short lip frenum, and altered morphology of a superior primary incisor. For esthetic and functional rehabilitation of the oral health of this child, an indirect composite resin restoration was performed using intraoral digital scanning technology. This case presents an accurate, rapid, and comfortable restorative treatment option that might result in excellent outcomes in children with ZVCS or similar syndromes with neurological impairment.
Spontaneous Renal Allograft Rupture Caused by Acute Tubular Necrosis: A Case Report and Review of the Literature
Authors: Deepak Shankar Ray, Sharmila Thukral
Journal: Case Rep Transplant (2017)
MeSH terms: Case Reports

Renal allograft rupture (RAR) is a rare but lethal complication of renal transplantation. It potentially threatens graft and patient survival. RAR is frequently associated with acute rejection, but other causes like renal vein thrombosis and acute tubular necrosis have also been observed. Most commonly a graft nephrectomy is required, but graft repair can also be attempted in selected cases to salvage the graft. Herein, we describe a rare case of spontaneous renal allograft rupture in the early posttransplant period due to acute tubular necrosis. A 42-year-old male, living donor renal allograft recipient, experienced RAR on the sixth posttransplant day. Surgical exploration showed two lacerations of 10 cm and 5 cm length at the upper and mid pole of the kidney. Histologically, the graft demonstrated acute tubular injury; no features of humoral or cellular rejection were identified. The successful management of this complication resulted in the salvage of the patient and the graft. This case demonstrates that early diagnosis and prompt treatment of a life-threatening RAR can salvage the graft.
Case Report: Rare Dynein Axonemal Heavy Chain 9 Mutations in a Han-Chinese Patient With Kartagener Syndrome
Authors: Jingjing Feng, Junqing Li, Yong Du, Tianyun Shi, Lokesh Sharma, Zhijun Jie
Journal: Front Med (Lausanne) (2022)
Keywords: dnah9, pcd, gene mutation, kartagener syndrome, situs inversus
MeSH terms: Case Reports

A 52-year-old woman presented with respiratory symptoms of productive cough and shortness of breath. She had suffered from repeated pneumonia. The CT scans revealed chronic sinusitis, tree bud signs in pulmonary imaging, and situs inversus. She received a primary diagnosis of Kartagener syndrome of primary ciliary dyskinesia (PCD) and a genetic examination was performed. Compound heterozygous mutations in dynein axonemal heavy chain 9 (DNAH9) were identified, which encoded outer dynein arms (ODAs) components. DNAH9 mutations are relatively rare events in PCD, and this is the first report of PCD patients with DNAH9 mutations in the Chinese population. Further, a literature review of mutations in PCD was conducted.