OpenMed/multicare-articles huggingface dataset:
Empedobacter brevis Meningitis in a Neonate: A Very Rare Case of Neonatal Meningitis and Literature Review
Authors: Deepak Sharma, Ankur Patel, Priyanka Soni, Pradeep Sharma, Basudev Gupta
Journal: Case Rep Pediatr (2016)

Empedobacter brevis is gram-negative bacilli that belongs to Flavobacteriaceae family. It was previously known with name of Flavobacterium breve. The reservoir of these bacteria is soil, plants, water, food, hospital water sources, including incubators, sinks, faucets, tap water, hemodialysis systems, saline solutions, and other pharmaceutical solutions. We report a case of term female newborn, admitted with complaint of respiratory distress developing soon after birth and developed clinical features of sepsis at age of 92 hours of postnatal life. The sepsis screen was positive and blood culture and cerebrospinal fluid showed growth of Empedobacter brevis that was resistant to multiple antibiotics. The neonate was treated with appropriate antibiotics and was discharged successfully. The novelty of the case report is that this is the first case report of neonatal sepsis caused by Empedobacter brevis.
Use of Edoxaban for the Treatment of Heparin-Induced Thrombocytopenia
Authors: Ryo Kanamoto, Shinichi Hiromatsu, Tomoyuki Anegawa, Kanako Sakurai, Shohei Yoshida, Yusuke Shintani, Hiroyuki Otsuka, Satoru Tobinaga, Hiroyuki Tanaka
Journal: Case Rep Vasc Med (2020)
MeSH terms: Case Reports

Heparin-induced thrombocytopenia (HIT) is a life-threatening adverse drug reaction of heparin therapy, which increases a patient's risk of developing venous and/or arterial thromboembolism. HIT should be treated through discontinuation of heparin and administration of nonheparin anticoagulants such as argatroban. For long-term anticoagulation, parenteral nonheparin anticoagulants are generally converted to oral treatment with a vitamin K antagonist such as warfarin. Although administration of warfarin is recommended to overlap with a nonheparin anticoagulant for a minimum of 5 days, overlapping with argatroban and warfarin presents high risks of bleeding. We describe a case of HIT treated with edoxaban. A 78-year-old man underwent surgery for esophageal cancer and was administered heparin perioperatively. After surgery, he was diagnosed with HIT and venous thromboembolism. We immediately stopped heparin and initiated parenteral argatroban. The patient was subsequently started on edoxaban without any overlap between the two drugs. The treatment was successful. The treatment of edoxaban following argatroban for HIT could reduce bleeding complications and shorten the length of hospital stay. To the best of our knowledge, this is the first report of the use of edoxaban for HIT treatment.
Deletion of 4q28.3-31.23 in the background of multiple malformations with pulmonary hypertension
Authors: Balazs Duga, Marta Czako, Katalin Komlosi, Kinga Hadzsiev, Katalin Torok, Katalin Sumegi, Peter Kisfali, Gyorgy Kosztolanyi, Bela Melegh
Journal: Mol Cytogenet (2014)
Keywords: 4q283-3123, array cgh, complex malformation syndrome, deletion, developmental delay, face dysmorphia, pulmonary hypertension, vascular malformation of the lung
MeSH terms: Case Reports

The 4q deletion syndrome shows a broad spectrum of clinical manifestations consisting of key features comprising growth failure, developmental delay, craniofacial dysmorphism, digital anomalies, and cardiac and skeletal defects. We have identified a de novo interstitial distal deletion in a 9 month-old girl with growth failure, developmental delay, ventricular septum defect in the subaortic region, patent foramen ovale and patent ductus arteriosus, vascular malformation of the lung, dysgenesis of the corpus callosum and craniofacial dysmorphism using array-comparative genomic hybridization. This de novo deletion is located at 4q28.3-31.23 (136,127,048 - 150,690,325), its size is 14.56 Mb, and contains 8 relevant genes (PCDH18, SETD7, ELMOD2, IL15, GAB1, HHIP, SMAD1, NR3C2) with possible contributions to the phenotype. Among other functions, a role in lung morphogenesis and tubulogenesis can be attributed to the deleted genes in our patient, which may explain the unique feature of vascular malformation of the lung leading to pulmonary hypertension. With the detailed molecular characterization of our case with 4q- syndrome we hope to contribute to the elucidation of the genetic spectrum of this disorder.
Chronic gallbladder wall thickening: Is it always malignancy?
Authors: Anjuna Reghunath, Suchana Kushvaha, Rohini G Ghasi, Geetika Khanna, Apurva Surana
Journal: SA J Radiol (2020)
Keywords: computed tomography, gallbladder cancer, magnetic resonance imaging, ultrasound, xanthogranulomatous cholecystitis
MeSH terms: Case Reports

Gallbladder wall thickening, associated with features like perforation, fistula formation and invasion of adjacent organs, is often assumed to be malignant. Xanthogranulomatous cholecystitis (XGC) causes gallbladder wall thickening with similar aggressive features and closely mimics gallbladder carcinoma clinically, radiologically and surgically. Differentiating between these two is crucial for management as misdiagnosis of gallbladder cancer can lead to unnecessary radical surgery. We report a case of chronic gallbladder wall thickening, initially suspected to be malignant, but subsequently diagnosed as XGC.
A giant cell rich osteosarcoma of the proximal ulnar bone treated by elbow arthroplasty: A case report
Authors: S D Cahayadi, A Antoro, B Swandika
Journal: Int J Surg Case Rep (2019)
Keywords: case report, elbow arthroplasty, giant cell rich type, latissimus dorsi flap, osteosarcoma, proximal ulnar bone

Introduction:
Osteosarcoma is a malignant tumor of the bone. The treatment and prognosis have changed dramatically these days. Limb salvage surgery is a widely accepted alternative to amputation in patients with sarcoma. In most cases, the diagnosis of osteosarcoma is not difficult. We reported a rare location and a rare type of osteosarcoma case treated by elbow arthroplasty along with the challenging diagnostic and treatment.
Case presentation:
We reported 46 years old female complained of pain on her left elbow for 5 months. An x-ray showed lytic lesion on the medial side of the elbow while the MRI examination suggested a malignant bone tumor in the epi-meta-diaphyseal of left ulnar bone. Core biopsy showed a Giant Cell containing lesion. The histology of resected tumor and Ki67 staining confirmed the diagnosis of giant cell-rich type osteosarcoma.
Discussion:
Giant cell rich osteosarcoma is an uncommon variant of osteosarcoma which characterized by an abundance of osteoclastic giant cells and lack of tumor osteoid. Involvement of forearm in conventional osteosarcoma is extremely rare. This was the epidemiologic reason why the osteosarcoma was not the first diagnosis of our case. Being a very rare location of the osteosarcoma, the management poses a special challenge. They are the surgical technique, which is not well described, and the implant that need to be custom made.
Conclusion:
Giant cell rich osteosarcoma is difficult to diagnose because its histology and radiologic feature mimic a benign giant cell tumor.
Multiple-system atrophy in long-term professional painter: a case report
Authors: Yusa Nagai, Riko Kitazawa, Miku Nakagawa, Munenori Komoda, Takeshi Kondo, Ryuma Haraguchi, Sohei Kitazawa
Journal: Case Rep Pathol (2012)

Introduction. Multiple system atrophy (MSA) is a rare and severe adult-onset, sporadic, and progressive neurodegenerative disorder. Here, we describe an autopsy case of MSA in a long-term professional painter. Although typical glial cytoplasmic inclusion (GCI) was not observed in a routine histological examination, strong α-synuclein immunostaining in the nucleus confirmed the diagnosis of MSA. Case Presentation. A 48-year-old Japanese man with a long occupational history of professional painter was sent to the emergency room, where he died of multiple organ failure. The patient had suffered tremors and inarticulateness at age 28, developed diabetes at 42 and was diagnosed with spinocerebellar degeneration at 46. A histopathological examination showed severe neuronal loss, gliosis, and tissue rarefaction in the paleostriatum, striate body of the substantia nigra, the pons, and the olivary nucleus of the upper medulla oblongata, intermediolateral of the spinal gray matter (sacral region). α-synuclein-positive GCI in oligodendroglia was occurred in the cerebral cortex, the midbrain, the medulla oblongata, and the spinal cord. These findings confirmed the presence of multiple-system atrophy (OPCA+SDS). Conclusion. Although the pathogenesis of MSA is still unclear, prolonged, and extensive exposure to organic solvents, together with a hyperglycemic morbidity attributed to diabetes, may have contributed to the onset and clinical course of the present case.
Adipsic hypernatremia in a young Sudanese child, challenges in a limited-resource setting: a case report
Authors: Mohammed Abdulrahman Alhassan, Asmahan Tagelsir Abdalla, Samar Sabir Hassan, Mohamed Ahmed Abdullah
Journal: Pan Afr Med J (2021)
Keywords: thirst, case report, child, diabetes insipidus, hypernatremia, hypothalamus
MeSH terms: Dehydration / etiology, Hypernatremia / etiology, Thirst / physiology, Case Reports, Dehydration / diagnosis, Female, Humans, Hypernatremia / diagnosis, Infant, Osmolar Concentration, Recurrence, Sudan

Adipsia is a rare condition characterized by a lack of thirst due to a defect in specific osmoreceptors located in the hypothalamus. The disorder is characterized by failure to maintain the body's normal plasma osmolality (POSM), resulting in chronic or recurrent severe hypernatremia and dehydration. Adipsia is usually accompanied by central diabetes insipidus (DI). Isolated adipsia (without DI) is very rare, with causes ranging from congenital central nervous system malformations to acquired anterior hypothalamic lesions. The diagnosis and management of the condition are considerably challenging for both clinicians and patients/parents, especially in a resource-limited setting. We here in present the first case report of adipsia from Sudan; a young child with isolated adipsia, diagnosed after recurrent severe hypernatemic dehydration episodes. The report portrays the unique challenges in suspecting, diagnosing, and managing the condition in a limited-resource setting.
Photodynamic therapy for the endodontic treatment of a traumatic primary tooth in a diabetic pediatric patient
Authors: Giselle de Sant'Anna
Journal: J Dent Res Dent Clin Dent Prospects (2014)
Keywords: endodontics, dental traumatism, photodynamic therapy
MeSH terms: Case Reports

Conservation of deciduous teeth with pulp alterations caused by caries or trauma is a major therapeutic challenge in pediatric dentistry. It is essential that the sanitizers used in root canal procedures perform well in eliminating bacteria. Antimicrobial photodynamic therapy (PDT) is an emerging and promising adjuvant therapy for endodontic treatment in an attempt to eliminate microorganisms persistent after chemomechanical preparation. This paper reports the case of a five-year-old male with type I diabetes mellitus, presenting the need for pulp therapy in maxillary primary left central incisor due to injury. The proposed treatment included the use of PDT for decontamination of root canals with the application of 50 μg/mL of methylene blue dye for 3-5 minutes and 40 J/cm(2) as energy density, taking into account the need for tissue penetration and effec-tiveness of PDT inside the dentinal tubules.
A Novel Pathogenic Variant in CARMIL2 ( RLTPR) Causing CARMIL2 Deficiency and EBV-Associated Smooth Muscle Tumors
Authors: Jennifer R Yonkof, Ajay Gupta, Cesar M Rueda, Shamlal Mangray, Benjamin T Prince, Hemalatha G Rangarajan, Mohammad Alshahrani, Elizabeth Varga, Timothy P Cripe, Roshini S Abraham
Journal: Front Immunol (2020)
Keywords: carmil2 deficiency, dock8 deficiency, ebv-associated smooth muscle tumor, early-onset inflammatory bowel disease (ibd), rltpr, immune dysregulation, primary immunodeficiencies
MeSH terms: Epstein-Barr Virus Infections / genetics, Herpesvirus 4, Human / physiology, Microfilament Proteins / genetics, Primary Immunodeficiency Diseases / genetics, T-Lymphocytes / physiology, Case Reports, Research Support, Non-U.S. Gov't, CD28 Antigens / metabolism, Cells, Cultured, Cytoskeleton / metabolism, Humans, Lymphocyte Activation, Male, Pedigree, Saudi Arabia, Siblings, Smooth Muscle Tumor, CARMIL1 protein, human, CD28 Antigens, Microfilament Proteins

CARMIL2 deficiency is a rare combined immunodeficiency (CID) characterized by defective CD28-mediated T cell co-stimulation, altered cytoskeletal dynamics, and susceptibility to Epstein Barr Virus smooth muscle tumors (EBV-SMTs). Case reports associated with EBV-SMTs are limited. We describe herein a novel homozygous CARMIL2 variant (c.1364_1393del) in two Saudi Arabian male siblings born to consanguineous parents who developed EBV-SMTs. CARMIL2 protein expression was significantly reduced in CD4+ T cells and CD8+ T cells. T cell proliferation on stimulation with soluble (s) anti-CD3 or (s) anti-CD3 plus anti-CD28 antibodies was close to absent in the proband, confirming altered CD28-mediated co-signaling. CD28 expression was substantially reduced in the proband's T cells, and was diminished to a lesser degree in the T cells of the younger sibling, who has a milder clinical phenotype. Defects in both T and B cell compartments were observed, including absent central memory CD8+ T cells, and decreased frequencies of total and class-switched memory B cells. FOXP3+ regulatory T cells (Treg) were also quantitatively decreased, and furthermore CD25 expression within the Treg subset was substantially reduced. These data confirm the pathogenicity of this novel loss-of-function (LOF) variant in CARMIL2 and expand the genotypic and phenotypic spectrum of CIDs associated with EBV-SMTs.
Pure red cell aplasia associated with autoimmune hepatitis successfully treated with cyclosporine A
Authors: Akira Sato, Fumiaki Sano, Toshiya Ishii, Kayo Adachi, Ryujirou Negishi, Nobuyuki Matsumoto, Chiaki Okuse
Journal: Clin J Gastroenterol (2014)
Keywords: autoimmune hepatitis, cyclosporine, pure red cell aplasia
MeSH terms: Cyclosporine / therapeutic use, Hepatitis, Autoimmune / drug therapy, Hepatitis, Autoimmune / etiology, Immunosuppressive Agents / therapeutic use, Red-Cell Aplasia, Pure / complications, Red-Cell Aplasia, Pure / drug therapy, Case Reports, Adult, Female, Humans, Remission Induction, Immunosuppressive Agents, Cyclosporine

A 47-year-old female with a 17-year history of autoimmune hepatitis had been treated with prednisolone, azathioprine, and ursodeoxycholic acid. Although her alanine aminotransferase level occasionally showed mild abnormality, the prednisolone dose could not be increased because she had developed cataract during the course of her illness. In May 2012, she developed severe normochromic normocytic anemia without hemorrhage, and azathioprine was discontinued because it was suspected of being the cause. However, anemia recurred frequently even after discontinuation, necessitating repeated blood transfusions. Bone marrow analysis revealed selective erythroblastopenia, thus leading to a diagnosis of pure red cell aplasia. Cyclosporine A was administered, which led to a dramatic recovery from anemia, and stabilized her alanine aminotransferase levels. Furthermore, the prednisolone dose could be gradually tapered. Pure red cell aplasia associated with autoimmune hepatitis is extremely rare. The present case shows that patients with autoimmune hepatitis refractory to the standard treatment regimen and those with concomitant pure red cell aplasia may be treated with cyclosporine A.
Ghost cell odontogenic carcinoma of the maxilla: a case report with a literature review
Authors: Elneel Ahmed Mohamed Ali, Musadak Ali karrar, Abeer Abdalla El-Siddig, Nahla Gafer, Ali Abdel satir
Journal: Pan Afr Med J (2015)
Keywords: odontogenic carcinoma, sudan, ghost cell, mandible
MeSH terms: Maxillary Neoplasms / diagnosis, Odontogenic Tumors / diagnosis, Case Reports, Review, Humans, Male, Maxillary Neoplasms / pathology, Maxillary Neoplasms / therapy, Odontogenic Tumors / pathology, Odontogenic Tumors / therapy, Radiotherapy, Adjuvant / methods, Young Adult

The aim of this report is to present a rare case of a Ghost cell odontogenic carcinoma (GCOC) of a 21-year-old man with review of the literature. The patient was treated surgically, and one of the surgical margins was involved, the patient received adjuvant radiotherapy for local control. Five months later, patient presented with infraorbital lesion which was proven histological to be GCOC. Radiological images, histological sections and clinical photographs are also presented. One year after the second surgery, the patient was tumor free. The only effective treatment modality was surgical removal.
Minimal Change Disease After First Dose of Pfizer-BioNTech COVID-19 Vaccine: A Case Report and Review of Minimal Change Disease Related to COVID-19 Vaccine
Authors: Jessica Hanna, Alistair Ingram, Tiffany Shao
Journal: Can J Kidney Health Dis (2021)
Keywords: covid-19 vaccine, sars-cov-2, acute kidney injury, minimal change disease
MeSH terms: Case Reports

Rationale:
While severe complications are generally uncommon with novel coronavirus disease 2019 (COVID-19) vaccine, there has been a steady increase in the number of patients presenting with nephrotic syndrome and acute kidney injury after the administration of COVID-19 vaccine. Physicians should be made aware of minimal change disease as a potential complication associated with COVID-19 vaccine.
Presenting concerns:
A 60-year-old male without significant past medical history presented with new onset of nephrotic syndrome approximately 10 days after his first dose of Pfizer-BioNTech COVID-19 vaccine. Laboratory findings showed hypoalbuminemia (20 g/L), elevated urine albumin/creatinine ratio (668 mg/mmol), and elevated creatinine of 116 µmol/L from a baseline of 79 µmol/L.
Diagnosis:
A diagnostic kidney biopsy was performed 6 weeks after the onset of the edema and approximately 8 weeks after his first dose of Pfizer-BioNTech COVID-19 vaccine. The kidney biopsy findings were consistent with minimal change disease with focal acute tubular injury.
Interventions:
The patient was treated conservatively with ramipril 10 mg and furosemide 80 mg daily 5 weeks after the onset of swelling. Prednisone 1 mg/kg was initiated immediately when the kidney biopsy result became available (approximately 6 weeks after the onset of edema).
Outcomes:
The patient remitted with rapid weight loss starting 2 weeks post prednisone initiation.
Novel findings:
De novo minimal change disease with acute tubular injury is a kidney manifestation following the administration of Pfizer-BioNTech COVID-19 vaccine. Minimal change disease is potentially a rare complication of Pfizer-BioNTech COVID-19 vaccine.
Unilateral macular neovascularization formation during the follow-up of a 15-year-old boy with Bietti crystalline dystrophy and the successful treatment outcome with a single intravitreal ranibizumab injection
Authors: Mustafa Kayabaşi, Ferdane Ataş, Ali Osman Saatci
Journal: GMS Ophthalmol Cases (2023)
Keywords: bietti crystalline dystrophy, macula, macular neovascularization, optical coherence tomography, optical coherence tomography angiography
MeSH terms: Case Reports

Objective:
To report the successful outcome with a single intravitreal ranibizumab injection in a 15-year-old boy with Bietti crystalline dystrophy (BCD) who developed a unilateral macular neovascularization (MNV).
Methods:
A retrospective case report.
Results:
A 15-year-old Caucasian boy with Bietti crystalline dystrophy was diagnosed to have a unilateral MNV a year after the initial examination with the help of multimodal imaging and he was treated with a single intravitreal ranibizumab injection. Five months later, visual acuity improved and anatomically a dry-looking macula was noted.
Conclusion:
MNV is among the rare macular complications of BCD. The present case is the third reported pediatric case with MNV in association with BCD and the first pediatric BCD patient who received a vascular endothelial growth factor inhibitor (anti-VEGF) agent.
Isotretinoin in Acne Vulgaris Complicated by Underlying Major Depression: A Case Report and Review of Literature
Authors: Matthew DeLuca, Maxsaya Baez Nuñez, Ezequiel Rodriguez, Krishan Chirimunj
Journal: Case Rep Psychiatry (2021)
MeSH terms: Case Reports

We relay the case of a teenage female with severe facial acne vulgaris and a past psychiatric history of major depressive disorder who presented to the emergency department with a primary complaint of ongoing suicidal ideation. Defining features of this case stem from the patient endorsing that her suicidal ideation was a result of her severe acne and the coinciding social perturbation it caused. Additionally, the patient reported that just four months prior to the current presentation, her dermatologist started her on isotretinoin therapy for the management of acne vulgaris. To the best of the authors' knowledge, there have been no reported cases which describe a teenage female presenting with active suicidal ideation secondary to severe acne vulgaris while concurrently undergoing treatment with isotretinoin. Given the controversial but reported association between isotretinoin and increased suicidality, we considered the appropriateness of continuing this medication for our patient. We then conducted a literature search evaluating the evidence concerning this association. In what follows, we present a unique case report and provide a thorough review of the evidence-or lack thereof-surrounding the relationship between isotretinoin and suicidality. Additionally, the authors aim to provide recommendations for the management of future patients who may present under similar circumstances.
Eribulin Provides a Remarkable Effect in Trabectedin-Resistant Myxoid Liposarcoma
Authors: Hironari Tamiya, Hideaki Sabe, Katsunari Yamashita, Yoshinori Imura, Toru Wakamatsu, Satoshi Takenaka
Journal: Case Rep Orthop (2020)
MeSH terms: Case Reports

Adriamycin-based chemotherapy is commonly used for malignant soft tissue sarcoma including myxoid liposarcoma. However, in the case of unavailability or failure of the adriamycin-based regimen, trabectedin or eribulin can produce a good antitumor effect for myxoid liposarcoma. We relate the experience of a 64-year-old female with myxoid liposarcoma, who noticed a nodule on her left thigh and visited our institute. At initial presentation, the tumor was 18.7 cm in diameter, and the magnetic resonance imaging (MRI) showed a malignant lipomatous tumor with a myxoid component. We recommended that she undergo treatment; however, she refused. Three years later, the tumor had grown larger, so she finally decided to undergo treatment. A needle biopsy revealed a myxoid liposarcoma. The tumor massively involved the neurovascular structures; we thus determined that hip disarticulation was inevitable. Two years later, metastases in the right thigh, left lung, right ileum, and abdominal space were pointed out and chemotherapy was initiated. Adriamycin was unusable due to cardiac dysfunction, so trabectedin was administered; however, the tumors progressed. Eribulin was subsequently started and has been considerably effective for more than 2 years without severe adverse effects. In conclusion, we experienced a case showing the remarkable and long-lasting effect of eribulin against trabectedin-resistant myxoid liposarcoma.