OpenMed/multicare-articles huggingface dataset:
Parathyroid Adenoma With Respiratory-Like Epithelium: Case Report of a Potential Mimic With Unknown Etiology
Authors: C Christofer Juhlin, Jan Zedenius
Journal: Front Endocrinol (Lausanne) (2021)
Keywords: adenoma, case report, epithelium, parathyroid, respiratory
MeSH terms: Adenoma / pathology, Parathyroid Neoplasms / pathology, Respiratory Mucosa / pathology, Case Reports, Research Support, Non-U.S. Gov't, Adenoma / surgery, Humans, Male, Middle Aged, Parathyroid Neoplasms / surgery, Parathyroidectomy, Sweden

Parathyroid adenoma is a tumor composed of increased parenchymal tissue, often built-up by chief cells, transitional cells or oncocytic cells arranged in acinar or solid formations. Occasionally, rare histological patterns are reported, including cystic or trabecular arrangements. We present a 47 year-old male patient with primary hyperparathyroidism who underwent focused parathyroidectomy for a right inferior adenoma. Surgery was uneventful, but histologically, normal parathyroid tissue adjacent to a tumorous structure displaying a cystic growth pattern was detected. The cells lining the cyst walls appeared cylindrical and pseudo-stratified, vaguely reminiscent of a respiratory type of epithelium usually associated to branchial cleft cysts or thyroglossal cyst remnants, albeit with a tumorous appearance. The respiratory-like epithelium stained positive for parathyroid markers PTH and GATA3, thereby confirming them as parathyroid-derived. The patient was cured from surgery as he displayed normal calcium and PTH levels postoperatively, and is currently alive and well without signs of relapse 4 years after surgery. This is to our knowledge the first report of a parathyroid tumor displaying a respiratory-like epithelium. Experimentally, canine parathyroid glands can develop ciliated respiratory epithelium in response to inhalation of ozone. Our patient is a construction worker with a hypothetically increased risk of continuous ozone exposure. Although this association remains purely speculative, future investigations of this tumor phenotype could perhaps yield novel insights regarding the frequency of this histological variant, potential clinical associations, and clues regarding influencing factors.
A case of angiomyolipoma rarely located in the larynx
Authors: Hulya Eyigor, Dinc Suren, Ustun Osma, Cem Sezer, Mustafa Deniz Yilmaz
Journal: Case Rep Otolaryngol (2011)
MeSH terms: Case Reports

Angiomyolipoma is a rare benign mesenchymal tumor, which is mostly renal in origin. A sixty-year-old male patient with the diagnosis of angiomyolipoma located in the larynx has been presented here, and the literature is reviewed.
Coexistence of Rheumatoid Arthritis, Systemic Lupus Erythematosus, Sjogren Syndrome, Antiphospholipid Syndrome, and Ankylosing Spondylitis
Authors: Moshiur Rahman Khasru, Md Abu Bakar Siddiq, Kazi Mohammad Sayeeduzzaman, Tangila Marzen, Abul Khair Mohammad Salek
Journal: Case Rep Rheumatol (2021)
MeSH terms: Case Reports

A 37-year-old Bangladeshi woman presented with low back and several joints pain and swelling for months together; there was significant morning stiffness for more than two hours. Repeated abortions, dry eye, hair fall, photosensitivity, and oral ulcer were the additional complaints. Clinical examination unveiled asymmetrical peripheral and both sacroiliac joint tenderness, positive modified Schober's test, and limited chest expansion. Schirmer's test was positive. The history of rheumatoid arthritis (RA) and ankylosing spondylitis (AS) among 1st-degree relatives was also significant. Biochemical analysis revealed pancytopenia, raised erythrocyte sedimentation rate (ESR) and C-reactive protein (CRP), and mild microscopic proteinuria. The patient was seropositive for rheumatoid factor (RF), antibodies against cyclic citrullinated peptides (anti-CCP), antinuclear antibody (ANA), anti-Sm antibody, anti-Sjögren's-syndrome-related antigen A and B (anti-SSA/SSB), antiphospholipid (aPL-IgG/IgM), and HLA B27; however, serum complement (C3 and C4) levels were normal. Basal cortisol level measured elevated. Besides, X-ray and MRI of lumbosacral spines demonstrated sacroiliitis. There was radiological cardiomegaly, echocardiography unveiled atrial regurgitation, and ascending aorta aneurysm. Based on the abovementioned information, RA, AS, and systemic lupus erythematosus (SLE) have been diagnosed. Moreover, the patient developed Sjogren's syndrome (SS), antiphospholipid lipid syndrome (APS), Cushing syndrome, ascending aorta aneurysm, and atrial regurgitation. Her disease activity score for RA (DAS28), DAS for AS (ASDAS), SLE disease activity index (SLEDAI), and Systemic Lupus International Collaborating Clinics/American College of Rheumatology (SLICC/ACR) scores were 3.46, 2.36, 23, and 5, respectively. The patient received hydroxychloroquine (200 mg daily), pulsed cyclophosphamide, prednisolone (20 mg in the morning), and naproxen 500 mg (twice daily). To our best knowledge, this is the first report documenting RA, AS, and SLE with secondary SS and APS.
Distal humerus as delayed site of metastasis from small cell carcinoma of gallbladder
Authors: Mutahir A Tunio, Mushabbab Alasiri, Asma Mohammed F Ali, Eyad Fawzi Alsaeed, Muhammad Shuja, Hanadi Fatani
Journal: Case Rep Gastrointest Med (2013)

Background. Small cell carcinoma (SCC) of the gallbladder is a rare entity and is often seen in elderly women. SCC of gallbladder is typically a nonsecretory carcinoid tumor without overt clinical symptoms and is often discovered at advanced stages. SCC of gallbladder carries a dismal prognosis as compared to SCC of lung and adenocarcinoma of gallbladder. To date, only 73 case reports have been published in the world literature. Case Presentation. Herein, we report a case of a 73-year-old Saudi woman who presented with one week history of right upper quadrant abdominal pain and obstructive jaundice and was found to be a case of locally advanced, metastatic SCC of gallbladder cT4N1M1 (liver, para-aortic lymph nodes, and bone). The patient was treated with neoadjuvant etoposide and cisplatin (EP) chemotherapy three cycles after biliary stenting followed by radical cholecystectomy, lymphadenectomy, and adjuvant EP chemotherapy and then one year later developed distal humerus osseous metastasis. Conclusion. SCC of the gallbladder is very rare entity and is often seen at advanced stages. Osseous metastases of peripheral skeleton from SCC gallbladder are rarely reported. Surgery is curative option but only for early stage tumors. Incorporation of chemotherapy along with radical resection increases the survival.
Pneumatosis Coli Associated with Pseudomembranous Colitis in a Patient following Colonic Surgery
Authors: Jennifer Bailey, Eldon A Shaffer
Journal: Case Rep Med (2010)
MeSH terms: Case Reports

Pneumatosis intestinalis is a rare disorder characterized by gas-filled cysts within the subserosal and/or submucosal regions of the intestinal wall. The source of this gas and its translocation across the mucosa is incompletely understood. Most (85%) cases are associated with medical conditions, ranging from psychiatric through respiratory disorders to gastrointestinal-related diseases; the remaining 15% lack any recognizable cause or association. In this case report, pneumatosis coli (affecting the colon) occurred in a patient following abdominal surgery and was associated with pseudomembranous colitis, which was Clostridium difficile toxin negative-presumably a false negative. Supportive care and appropriate antibacterial agents sufficed to alleviate symptoms and resolve the pneumatosis. Recognizing this uncommon but important association can avoid high financial and personal costs from unnecessary testing and invasive surgical explorations. Consideration should be given to pseudomembranous colitis as the basis for pneumatosis coli developing in patients who have received antibiotics, once gut ischemia has been ruled out.
Urethral Triplication Without Bladder Duplication: Endourologic Diagnosis and Management
Authors: Alberto Parente, Ruben Ortiz, Laura Burgos, Jose Maria Angulo
Journal: J Endourol Case Rep (2018)
Keywords: children, triplication, urethral, urethral duplication
MeSH terms: Case Reports

Urethral triplication is a rare congenital anomaly of the lower urinary system, with <15 cases reported so far. We present a 24-month-old boy with accessory hypoplastic urethra ending in glans. At the beginning of toilet training, urine output was observed along the rectum. Rigid cystoscopy shows a perineal urethra starting in the posterior urethra. Subsequently, flexible cystoscopy showed entry of epispadic urethra in the bladder immediately superior to the bladder neck. It was running parallel to primary urethra. Then, we observed two most frequent types of urethral duplication in the sagittal plane in a single patient.
Aggressive CD5-Positive Primary Bone Marrow Diffuse Large B-Cell Lymphoma with Leukemic Presentation
Authors: Mark G Evans, Sherif A Rezk, Lauren C Pinter-Brown, Xiaohui Zhao
Journal: Case Rep Hematol (2021)
MeSH terms: Case Reports

Primary bone marrow diffuse large B-cell lymphoma is an exceedingly rare form of non-Hodgkin lymphoma. It may demonstrate a leukemic presentation, and a proportion of cases have CD5 expression. The prognostic implications of this CD5-positivity remain unknown. Here, we present a 78-year-old man who presented with circulating peripheral blood lymphoma cells and a hypercellular marrow involved by diffuse large B-cell lymphoma, germinal center B-cell subtype. The patient responded favorably to six cycles of etoposide, prednisone, vincristine, cyclophosphamide, doxorubicin, and rituximab (EPOCH-R) and intrathecal methotrexate. He unfortunately relapsed in several enlarged inguinal lymph nodes and succumbed to the lymphoma approximately one year after diagnosis, demonstrating the particularly aggressive clinical course of his disease.
Augmentation of Antipsychotic Medications with Low-Dose Clozapine in Treatment-Resistant Schizophrenia-Case Reports and Discussion
Authors: Zoe Harrison, Owen Haeney, William Brereton
Journal: Case Rep Psychiatry (2021)
MeSH terms: Case Reports

Treatment resistance in schizophrenia is often encountered in clinical practice, with clozapine usually recommended as the appropriate therapy. However, where clozapine proves ineffective or cannot be tolerated due to side effects, treatment options are limited. In patients within forensic mental health services, residual symptomatology often presents a barrier to discharge and can have lasting effects on prospects for rehabilitation as well as risk to self and others. This paper presents a review of the relevant literature and three cases of a novel approach, utilising clozapine in doses usually considered subtherapeutic, in combination with the primary antipsychotic treatment. In all three patients, it improved clinical efficacy as well as tolerability, resulting in improvement that allowed discharge from the forensic hospital.
Prosthetic valve endocarditis caused by Bartonella quintana
Authors: John L Klein, Sukumaran K Nair, Tim G Harrison, Ian Hunt, Norman K Fry, Jon S Friedland
Journal: Emerg Infect Dis (2002)
MeSH terms: Bartonella quintana / isolation & purification, Heart Valve Prosthesis / adverse effects, Prosthesis-Related Infections / diagnosis, Prosthesis-Related Infections / microbiology, Trench Fever / diagnosis, Trench Fever / microbiology, Case Reports, Antibodies, Bacterial / isolation & purification, Aortic Valve / surgery, Bartonella quintana / genetics, Bartonella quintana / immunology, DNA, Bacterial / isolation & purification, Diagnosis, Differential, Female, Humans, Middle Aged, Risk Factors, Antibodies, Bacterial, DNA, Bacterial

We describe the first case of Bartonella quintana endocarditis affecting a prosthetic valve in a person with no known risk factors for this infection. Bartonella should be considered as a cause of endocarditis in any clinical setting.
Spinal cord stimulation for refractory pericarditis: a case report and a review of the mechanism of action
Authors: Simon Elsliger, Jacob Saucier, Andre Schneider, Antonios El Helou
Journal: Front Pain Res (Lausanne) (2023)
Keywords: heart failure, mechanism of action, pericarditis, refractory chest pain, spinal cord stimulation
MeSH terms: Case Reports

Background and objectives:
In recent years, spinal cord stimulation (SCS) has emerged as a promising management option for chronic pain of multiple etiologies. While its effectiveness has been strongly suggested in many patients, the exact mechanism of action of SCS is incompletely understood. This article reviews the leading mechanisms underlying the analgesic and cardiovascular effects of SCS and reports its novel benefits in a case of recurrent pericarditis.
Literature review:
Throughout history, the analgesic properties of SCS were thought to arise via stimulation of the spinothalamic tract. Although this mechanism has been thoroughly reported, new research and patient outcomes from SCS have revealed various additional properties that cannot be fully explained by this mechanism alone. Evidence suggests that SCS enhances calcitonin gene-related peptide release and modulates inflammatory cytokine secretion, sympathetic tone, and inhibitory neurotransmitter secretion. These distinct mechanisms likely collectively contribute to the therapeutic effects of SCS on the cardiovascular system and pain management.
Case report:
We report the case of a 48-year-old male patient with recurrent pericarditis, characterized by refractory angina-like pain and reduced left ventricular ejection fraction (LVEF). After 1 year of having a spinal cord stimulator implanted, the patient is free from pain and narcotics, with a reduction of 428 mg equivalent dose of morphine. The patient's LVEF increased from 40% to 45% without changes to his previous medical treatment. This is the first reported case of refractory pericarditis managed with spinal cord stimulation.
Conclusion:
Recognizing the improved pain management, reduced narcotic usage, and improved LVEF in our patient following SCS is critical to paving the way toward a complete understanding of the mechanism of action of SCS. This case reveals the therapeutic potential of SCS for cardiovascular pathologies other than refractory angina pectoris.
Acute Pain in Right Iliac Fossa during Pregnancy
Authors: Ophelye Chiabai, Cristina Anca Dragean
Journal: J Belg Soc Radiol (2022)
Keywords: epiploic appendagitis, pregnancy, right iliac fossa
MeSH terms: Case Reports

Teaching Point: The diagnosis of epiploic appendagitis in case of pain in the right iliac fossa in pregnant woman shouldn't be forgotten.
A practical guide to botulinum neurotoxin treatment of shoulder spasticity 2: Injection techniques, outcome measurement scales, and case studies
Authors: Jörg Wissel, Alexandre Camões-Barbosa, Stefano Carda, Damon Hoad, Jorge Jacinto
Journal: Front Neurol (2022)
Keywords: assessment tools, botulinum neurotoxin, injection, muscle spasticity, pain, shoulder

Introduction:
Botulinum neurotoxin type A (BoNT-A) is a first-line treatment option for post-stroke spasticity, reducing pain and involuntary movements and helping to restore function. BoNT-A is frequently injected into the arm, the wrist, the hand, and/or the finger muscles but less often into the shoulder muscles, despite clinical trials demonstrating improvements in pain and function after shoulder BoNT-A injection.
Methods:
In part 2 of this two-part practical guide, we present an experts' consensus on the choice of outcome measurement scales and goal-setting recommendations for BoNT-A in the treatment of shoulder spasticity to increase awareness of shoulder muscle injection with BoNT-A, alongside the more commonly injected upper limb muscles. Expert consensus was obtained from five European experts with a cumulative experience of more than 100 years of BoNT-A use in post-stroke spasticity. Case studies are included as examples of approaches taken in the treatment of shoulder spasticity.
Results:
Although the velocity-dependent increase in muscle tone is often a focus of patient assessment, it is only one component of spasticity and should be assessed as part of a wider range of measurements. For outcome measurement following BoNT-A injection in shoulder muscles, shoulder-specific scales are recommended. Other scales to be considered include Pain Numerical Rating and/or global functioning, as well as the quality of life and global perception of benefit scores.Goal setting is an essential part of the multidisciplinary management process for spasticity; goals should be patient-centric, realistic, and achievable; functional-focused goal statements and a mixture of short- (3-6 month) and long-term (9-18 month) goals are recommended. These can be grouped into symptomatic, passive function, active function, involuntary movement, and global mobility.Clinical evaluation tools, goal setting, and outcome expectations for the multipattern treatment of shoulder spasticity with BoNT-A should be defined by the whole multidisciplinary team, ensuring patient and caregiver involvement.
Discussion:
These recommendations will be of benefit to clinicians who may not be experienced in evaluating and treating spastic shoulders.
Full-Term Delivery and Complete Lung Recovery following VV ECMO Support Midpregnancy in a Patient with COVID-19 ARDS
Authors: Shelley Leong, Guillermo Moreno, Mohamed Fayed, Crystal Ives Tallman
Journal: Case Rep Crit Care (2023)
MeSH terms: Case Reports

Pregnant women are especially vulnerable to coronavirus disease 2019 (COVID-19). We present a twin pregnancy case with acute respiratory distress syndrome following COVID-19 infection at 19 weeks. The patient's ARDS was successfully managed with veno-venous extracorporeal membrane oxygenation (VV ECMO). She recovered completely and delivered healthy twins.
Transport and Golgi organization 2 deficiency with a prominent elevation of C14:1 during a metabolic crisis: A case report
Authors: Katsuyuki Yokoi, Yoko Nakajima, Yoshihisa Takahashi, Takashi Hamajima, Go Tajima, Kazuyoshi Saito, Shunsuke Miyai, Hidehito Inagaki, Tetsushi Yoshikawa, Hiroki Kurahashi, Tetsuya Ito
Journal: JIMD Rep (2022)
Keywords: c14
MeSH terms: Case Reports

Mutations in transport and Golgi organization 2 homolog (TANGO2) have recently been described as a cause of an autosomal recessive syndrome characterized by episodes of metabolic crisis associated with rhabdomyolysis, cardiac arrhythmias, and neurodegeneration. Herein, we report a case of a one-and-a-half-year-old Japanese girl, born to nonconsanguineous parents, who presented with metabolic crisis characterized by hypoglycemia with hypoketonemia, rhabdomyolysis, lactic acidosis, and prolonged corrected QT interval (QTc) at the age of 6 months. Acylcarnitine analysis during the episode of crisis showed prominent elevation of C14:1, suggesting very-long-chain acyl-CoA dehydrogenase (VLCAD) deficiency. In addition, worsening rhabdomyolysis was observed after intravenous administration of L-carnitine. VLCAD deficiency was initially suspected; however, the enzyme activity in lymphocytes was only mildly decreased at the gene carrier level, and no mutation in the VLCAD gene (ADADVL) was detected. Subsequently, acylcarnitine analysis was nonspecific at 17-h fasting and almost normal during the stable phase. Eventually, a trio whole-exome sequencing revealed a compound heterozygous variant of two novel variants in the TANGO2 gene, a missense variant, and a deletion of exon 7. This is the first case of TANGO2 deficiency in Asians. Our case suggests that elevated C14:1 may be seen in severe metabolic crises and that the use of L-carnitine should be avoided during metabolic crises.
Case Report: A Novel Homozygous Variant Identified in a Chinese Patient With Benign Recurrent Intrahepatic Cholestasis-Type 1
Authors: Huayu Chen, Dongbo Wu, Wei Jiang, Ting Lei, Changli Lu, Taoyou Zhou
Journal: Front Med (Lausanne) (2021)
Keywords: atp8b1 gene, bric, case report, diagnosis, intrahepatic cholestasis, treatment
MeSH terms: Case Reports

Benign recurrent intrahepatic cholestasis (BRIC) is a rare hereditary cholestatic liver disorder. Accurate diagnosis and timely interventions are important in determining outcomes. Besides clinical and pathologic diagnosis, genetic study of BRIC remains limited. Here, we report a young man enduring recurrent jaundice and severe pruritus for 15 years. The increased level of direct bilirubin was the main biochemical abnormality, and the work-up for common causes of jaundice were unremarkable. Liver biopsy showed extensive cholestasis of hepatocytes in zone 3. The novel homozygous variant including c.1817T > C and p.I606T was detected on his ATP8B1gene. The patient was finally diagnosed with BRIC-1. His symptoms were relieved, and liver function tests returned to normal after taking ursodeoxycholic acid. This case provides a different perspective to the methodology employed when dealing with cases of jaundice and helping diagnose rare diseases.
Communicating Food Risk-Benefit Assessments: Edible Insects as Red Meat Replacers
Authors: Emilia Boehm, Dan Borzekowski, Ermolaos Ververis, Mark Lohmann, Gaby-Fleur Böl
Journal: Front Nutr (2021)
Keywords: edible insects, food communication, food perception, health communication, novel foods, red meat substitute, risk communication, risk-benefit assessment (rba)

Risk-benefit Assessment (RBA) is an emerging methodology in the area of Food and Nutrition that offers a simultaneous evaluation of both risks and benefits linked to dietary choices. Communication of such research to consumers may present a challenge due to the dual nature of RBA. We present a case study of a communication strategy developed for the NovRBA-project. The NovRBA-project (Novel foods as red meat replacers-an insight using Risk Benefit Assessment methods) performed a risk-benefit assessment to evaluate the overall health impact of substituting red meat (beef) by a novel food (house cricket), considering the microbial, toxicological and nutritional characteristics of the respective dietary choices. A literature review of risk perceptions and acceptance of beef and insects as food formed the basis of the communication strategy for the study's results, drawing on environmental and emotional as well as health-related motivations to consume or avoid either food and considering the sociodemographic characteristics of likely consumers. Challenges and future directions for consumer protection organizations communicating findings of risk-benefit analyses on food safety are discussed.
Positive Retrospective SARS-CoV-2 Testing in a Case of Acute Respiratory Distress Syndrome of Unknown Etiology
Authors: A Burkett, S McElwee, C Margaroli, P Bajpai, A Elkholy, U Manne, K Wille, P Benson
Journal: Case Rep Pulmonol (2021)
MeSH terms: Case Reports

In order to elucidate the cause of acute respiratory distress syndrome of unknown etiology in a pre-pandemic patient, molecular techniques were used for detection of SARS-CoV-2. We used a SARS-CoV-2 nucleocapsid protein immunofluorescence stain to retrospectively identify an individual with diffuse alveolar damage on autopsy histology who had negative respiratory virus panel results in February, 2020, in Birmingham, Alabama. In situ hybridization for SARS-CoV-2 RNA revealed evidence of widespread multiorgan SARS-CoV-2 infection. This death antecedes the first reported death of a State of Alabama resident diagnosed with SARS-CoV-2 by 26 days.