OpenMed/multicare-articles huggingface dataset:
Case Report: Castleman Disease With an Associated Stromal Spindle Cell Proliferation, PDGFRB Mutation and p53 Expression: Clonal Origins of a Rare Disease
Authors: Kunwar I Singh, Sumanth Gollapudi, Jyoti Kumar, Alexandra Butzmann, Corinn Small, Sara Kreimer, Emine Arzu Saglam, Roger Warnke, Oscar Silva, Robert S Ohgami
Journal: Front Oncol (2022)
Keywords: castleman disease, fdc, frc, pdgfrb, tp53, fibroblastic reticular cell, follicular dendritic cell, p53
MeSH terms: Case Reports

Castleman disease (CD) is a rare lymphoproliferative disorder with distinct clinical subtypes. However, our understanding of the underlying pathogenesis of particular subtypes of CD remains unclear. While the characteristic morphologic changes within UCD, including occasional cases of overgrowth of spindled stromal and follicular dendritic cells have been described, the nature and origin of these spindle cells remain elusive. Few reports have suggested that underlying stromal cells in UCD are clonally neoplastic and may be of fibroblastic reticular cell (FRC) or follicular dendritic cell (FDC) origins given their close clonal relationship. Although certain histomorphologic features may aid diagnosis, there are no specific biomarkers that can differentiate a reactive process mimicking UCD from true UCD. Hence, we describe an index case with morphology consistent with the hyaline vascular subtype of UCD with concomitant atypical smooth muscle actin (SMA)-positive stromal spindle cell proliferation containing a recurrent PDGFRB N666S mutation and upregulation of p53 expression. Further analysis of 21 additional cases of UCD identified increased p53 expression by digital image analysis and SMA positive stromal cells predominantly within the paracortical and intrafollicular areas further strengthening the hypothesis of the stromal cellular derivation and origins of UCD.
Systemic air embolism complicating upper gastrointestinal endoscopy: a case report with post-mortem CT scan findings and review of literature
Authors: Zabiullah Ali, Ferdia Bolster, Eric Goldberg, David Fowler, Ling Li
Journal: Forensic Sci Res (2017)
Keywords: forensic science, air embolism, endoscopy, digestive system, forensic pathology, pancreaticoduodenectomy, post-mortem computed tomography scan

Endoscopy of the gastrointestinal and biliary tract is a common procedure and is routinely performed for therapeutic and diagnostic purposes. Perforation, bleeding and infection are some of the more common reported side effects. Air embolism on the other hand, is a rare complication of gastrointestinal endoscopy. We report a 77-year-old African-American female with a history of pancreatic cancer, which was resected with a Whipple procedure. As part of diagnostic and therapeutic procedure, an endoscopic retrograde cholangiopancreatography was planned several months after the surgery. The patient's heart rate suddenly slowed to 40 bpm during the procedure and she became cyanotic and difficult to oxygenate after the endoscope was introduced and CO2 gas was insufflated. A forensic autopsy was performed with post-mortem computed tomography (PMCT) and revealed extensive systemic air embolism. The detailed PMCT and autopsy findings are presented and current literature is reviewed.
Work-related olfactory disorder: a case series and review
Authors: Soon Woo Park, Young Joong Kang, Huisu Eom, Hyun-Jin Cho, Jungho Ahn, Sang-Gil Lee
Journal: Ann Occup Environ Med (2018)
Keywords: anosmia, occupational disease, olfactory disorder, work-relatedness
MeSH terms: Case Reports

Background:
The olfactory bulb is anatomically exposed and thus can be directly damaged by external stimulation. This can occur as an occupational injury owing to contact with organic solvents or other causes. We present cases of eight patients who sustained occupation-related exposure to potentially toxic substances and later presented with signs and symptoms of anosmia. We examined the occupational and medical characteristics of the patients and evaluated their work-relatedness.
Case presentation:
Case 1: A 50-year-old man performed high-frequency heat treatments for approximately 11 years. He experienced decreased senses for olfaction and taste during the later years culminating in the diagnosis of anosmia after 3 years (high work-relatedness). Case 2: A 54-year-old man whose work involved exposure to various organic solvents, such as spray painting and application of paint and thinners for approximately 4 years, was subsequently diagnosed with anosmia based on rhinorrhea, headache, and loss of olfaction (high work-relatedness). Case 3: A 44-year-old-man who performed spray painting for approximately 17 years developed anosmia (high work-relatedness). Case 4: A 44-year-old man was involved in ship engine cleaning once a month, for approximately 7 h per cleaning session; he was diagnosed with anosmia based on loss of olfaction (low work-relatedness). Case 5: A 41-year-old man worked in ship building block construction for approximately 13 years; anosmia diagnosis was based on loss of olfaction (low work-relatedness). Case 6: A 47-year-old woman performed product inspection and labeling at a plant manufacturing automobile parts; anosmia diagnosis was based on decreased olfaction and taste (low work-relatedness). Case 7: A 50-year-old woman performed epoxy coating in a plant manufacturing automobile parts; anosmia diagnosis was based on diminishing olfaction (low work-relatedness). Case 8: A 57-year-old woman performed cleaning of the area where mobile phone parts were manufactured; anosmia diagnosis was based on diminishing olfaction (low work-relatedness).
Conclusion:
The study results confirmed work-relatedness when the subject was young, and the duration of exposure was long without any other cause of anosmia. Regarding compensation for occupational diseases, work-relatedness can be recognized as a relative concept.
Case report: Primary pleural giant extraskeletal Ewing sarcoma in a child
Authors: Yang Wu, Chao-Bang Xie, Qiong Huang, Kai-Fei Zhao
Journal: Front Oncol (2023)
Keywords: case report, computed tomography, extraskeletal ewing sarcoma, intrathoracic tumor, malignancy
MeSH terms: Case Reports

Primary extraskeletal Ewing sarcoma (EES) is a rare small round cell malignancy that accounts for less than 1% of all sarcomas. It is found most commonly in the trunk and lower limbs and very rarely in the pleura and can be easily misdiagnosed in clinical practice. This study presents the case of an 11-year-old boy who presented to our hospital with no apparent cause of left shoulder pain for 6 months. On physical examination, tenderness was noted in the left chest wall and shoulder joint, which had a limited range of motion. Computed tomography (CT) and magnetic resonance imaging (MRI) of the chest revealed an irregular soft tissue mass in the upper left thorax, with a wide base attached to the adjacent pleura and bone destruction of the adjacent left first rib. The patient's bone scan showed a dense focus of increased radiotracer accumulation in the left first rib. A subsequent CT-guided aspiration biopsy of the left pleural mass with histomorphology and immunohistochemical phenotyping led to a diagnosis of extraskeletal Ewing sarcoma. To inhibit tumor growth, alternating systemic chemotherapy cycles of vincristine, doxorubicin, and cyclophosphamide (VDC) and isocyclophosphamide and etoposide (IE) were administered at 3-week intervals. After completing three VDC and two IE cycles, the child's condition was well and the pain in the left shoulder joint was relieved. However, a repeat MRI of the chest showed that the mass did not shrink.
An Occult HPV-Driven Oropharyngeal Squamous Cell Carcinoma Discovered Through a Saliva Test
Authors: Kai Dun Tang, Sarju Vasani, Touraj Taheri, Laurence J Walsh, Brett G M Hughes, Lizbeth Kenny, Chamindie Punyadeera
Journal: Front Oncol (2020)
Keywords: biomarker, human papillomavirus, oropharyngeal cancer, saliva, screening tools
MeSH terms: Case Reports

Oropharyngeal cancer (OPC) caused by human papillomavirus (HPV) is a rising global concern. Early lesions are small and are often located in difficult to access areas (such as the crypts of the tonsils or base of tongue). Unlike cervical cancer, there is no standard or routine screening program for HPV-driven OPC. HPV DNA from OPC tumors may shed directly into saliva, and this can be used as a biomarker for early diagnosis. In this study, we report the first-ever clinically occult OPC in an asymptomatic patient discovered through a saliva test. This case relied upon serial measurements of HPV-16 DNA in saliva, which fell to undetectable levels following low morbidity, curative treatment.
Case Report: Non-episodic Angioedema With Eosinophilia in a Young Lactating Woman
Authors: Mizuho Hirmatsu-Ito, Nobuhisa Nakamura, Megumi Miyabe, Tatsuaki Matsubara, Keiko Naruse
Journal: Front Immunol (2021)
Keywords: angioedema, case report, chemokine, cytokine, edema, eosinophil
MeSH terms: Angioedema / immunology, Eosinophilia / immunology, Case Reports, Adult, Chemokines / analysis, Chemokines / physiology, Cytokines / analysis, Cytokines / physiology, Electric Impedance, Female, Humans, Lactation, Chemokines, Cytokines

Angioedema with eosinophilia is classified into two types: episodic angioedema with eosinophilia (EAE), known as Gleich's syndrome, and non-episodic angioedema with eosinophilia (NEAE). We present the case of a young lactating woman with non-episodic angioedema. She had no history of parasitic or nonparasitic infections. Physical examination showed striking, non-pitting edema in both lower extremities. Her weight had not changed significantly throughout the course of the illness. She exhibited no other symptoms, and her vital signs were normal. There was no evidence of anemia, hypoalbuminemia, thyroid dysfunction, heart failure, renal failure, or postpartum cardiomyopathy. Based on these findings, we diagnosed her with angioedema with eosinophilia. Given the scarcity of information about this condition, we explored the dynamics between cytokines/chemokines and edema in this patient. We successfully quantified the edema by bioimpedance analysis. In addition, we revealed the involvement of interleukin-5 (IL-5), thymus- and activation-regulated chemokine/C-C motif chemokine ligand-17 (TARC/CCL-17), eotaxin-3/CCL-26, tumor necrosis factor-α (TNF-α), vascular endothelial growth factor (VEGF), monocyte chemotactic protein-4/CCL-13 (MCP-4/CCL-13), eotaxin-1/CCL-11, and regulated on activation, normal T expressed and secreted/CCL-5 (RANTES/CCL-5) in NEAE. Lastly, we elucidated the strong association between these parameters. To the best of our knowledge, this is the first such study of its kind.
Moxifloxacin Monotherapy in Left-Sided Staphylococcus aureus Endocarditis
Authors: Yucel Colkesen
Journal: Case Rep Infect Dis (2021)
MeSH terms: Case Reports

Staphylococcus aureus is the major cause of endocarditis, and its mortality has remained high despite therapeutic procedures over time. A case of left-sided native valve endocarditis caused by methicillin-sensitive Staphylococcus aureus which responded well to moxifloxacin monotherapy is described. An 83-year-old woman with a history of current hospitalization presented with fatigue and fever. Transthoracic echocardiography depicted vegetation, and blood cultures were positive for Staphylococcus aureus. After a 14-day intravenous administration of moxifloxacin, a good clinical response was achieved, and antibiotic regimen transitioned to oral moxifloxacin for an additional four-week therapy.
Hyperleukocytosis during clozapine treatment: A rare presentation of B-cell Acute lymphoblastic leukemia
Authors: Neslyne B Augustin, Michael Maroules
Journal: Leuk Res Rep (2021)
Keywords: b-cell acute lymphoblastic leukemia, clozapine toxicity, hyperleukocytosis, schizophrenia
MeSH terms: Case Reports

Clozapine has been widely used as an antipsychotic drug for the treatment of refractory schizophrenia. Unfortunately, a wide range of blood dyscrasias have been reported as adverse effects of this drug. Agranulocytosis has gotten the most clinical vigilance; however, there are substantial reports of other blood dyscrasias associated with Clozapine some more serious than others. Of relevance, there have been previous claims of Clozapine-associated leukocytosis and acute myeloid leukemia. We report the case of a 31-year-old patient who developed Acute lymphoblastic leukemia shortly after starting treatment with Clozapine for refractory schizophrenia. We suggest Clozapine may play a causal role in the development of leukemias in patients taking this medication and we encourage vigilance for such correlation.
Keratoconus with Central Serous Chorioretinopathy: A Rare Combination
Authors: Anastasia Tsiogka, Apostolos Gkartzonikas, Konstantinos Markopoulos, Iordanis Georgiou, George L Spaeth
Journal: Case Rep Ophthalmol Med (2020)
MeSH terms: Case Reports

Keratoconus and central serous chorioretinopathy are two rare diseases. They can occur together in some individuals. We report a case of a 48-year-old man, who presented to our clinic with decreased visual acuity on his left eye. Physical examination, biomicroscopy, corneal topography, and optical coherence tomography revealed keratoconus and central serous chorioretinopathy. We discuss the possible connection between these two conditions.
Methimazole-induced cholestatic hepatitis: two cases report and literature review
Authors: Hai Zou, Lie Jin, Li-Ren Wang, Martin Braddock, Wen-Wei Cai, Ming-Hua Zheng
Journal: Oncotarget (2016)
Keywords: adverse effect, cholestatic hepatitis, methimazole
MeSH terms: Antithyroid Agents / adverse effects, Hepatitis / etiology, Hyperthyroidism / drug therapy, Jaundice, Obstructive / chemically induced, Methimazole / adverse effects, Case Reports, Research Support, Non-U.S. Gov't, Adult, Anti-Inflammatory Agents / therapeutic use, Drug-Related Side Effects and Adverse Reactions, Female, Hepatitis / drug therapy, Humans, Jaundice, Obstructive / drug therapy, Methylprednisolone / therapeutic use, Middle Aged, Prognosis, Anti-Inflammatory Agents, Antithyroid Agents, Methimazole, Methylprednisolone

Methimazole is commonly prescribed for patients who are thyrotoxic. Cholestatic hepatitis is a rare but serious adverse event which may be associated with interventional therapy. In this case report, we present two Chinese women with cholestatic jaundice due to methimazole treatment. Both patients had a history of hyperthyroidism; initial laboratory studies of liver function were normal and cholestatic hepatitis occurred after treatment with methimazole. Concomitant liver disease, such as viral hepatitis (A, B, C, D, E), autoimmune hepatitis, primary biliary cirrhosis and calculus of bile duct, were excluded. Liver enzyme levels in both patients returned to normal after stopping methimazole therapy and taking hepatoprotective drugs. It is essential that patients are informed about the earliest symptoms of serious adverse effects of antithyroid drugs, such as hepatic toxicity, and that they are advised to stop taking the drug immediately and contact their physician if such symptoms occur.
Parathyroid adenoma with rare severe pathological osteolytic lesion: a case report and literature review
Authors: Jia Chen, Gusheng Tang, Ye Peng, Hui Cheng
Journal: Front Oncol (2023)
Keywords: parathyroid adenoma, parathyroid hormone, parathyroidectomy, pathological osteolytic lesions, serum calcium
MeSH terms: Case Reports

Parathyroid adenomas are benign proliferative disorders of parathyroid glands. Patients typically exhibit hyperparathyroidism and elevated serum calcium levels due to elevated levels of parathyroid hormone (PTH). We report a newly diagnosed case of a rare pathological osteolytic lesion. Radiological evaluation revealed multiple bony lesions in multiple parts of the pelvis, vertebral body, and spinous process, suggesting hematological neoplasms or bone marrow metastatic carcinoma. The morphology revealed many abnormal cells in the bone marrow smear. Furthermore, serum calcium and PTH levels were significantly increased compared to normal levels. Doppler color ultrasound showed a thyroid mass (left), suspected parathyroid adenoma, thyroid, and isthmus nodular goiter (right). The patient underwent bilateral neck exploration with parathyroidectomy, and serum calcium and PTH levels significantly decreased on the second day after surgery and had a surgical cure.
Presentation of Ocular Syphilis in a HIV-Positive Patient with False-Negative Serologic Screening
Authors: Mahsaw N Motlagh, Cameron G Javid
Journal: Case Rep Infect Dis (2019)
MeSH terms: Case Reports

Purpose:
The ocular sequelae of syphilis are devastating and may cause blindness. The ambiguous nature of its ocular manifestations makes syphilis difficult to detect. Though uncommon, the rise of syphilis in the United States requires a renewed understanding of its ophthalmic presentation to prevent devastating outcomes. We present this case to raise awareness for the increasing prevalence of ocular syphilis and appropriate serologic testing.
Observations:
We describe a 65-year-old HIV-positive male with worsening retinitis, uveitis, and rapid visual loss. Initial lab results showed a nonreactive rapid plasma reagin (RPR) for syphilis. However, subsequent Treponema pallidum antibody testing was positive 48 hours after initial false-negative serologic screening. The patient had a rapid and successful recovery following treatment with penicillin.
Conclusions and importance:
The incidence of syphilis is on the rise once again, and patients living with HIV are at increased risk. Ocular syphilis should be considered in susceptible populations in the clinical setting of retinitis, uveitis, and worsening visual loss with unknown cause. In addition, retesting for syphilis will decrease the prevalence of false-negative results, especially in patients with high clinical suspicion.
Lack of Conventional Acinar Cells in Parotid Salivary Gland of Patient Taking an Anti-PD-L1 Immune Checkpoint Inhibitor
Authors: Sarah Pringle, Bert van der Vegt, Xiaoyan Wang, Nico van Bakelen, T Jeroen N Hiltermann, Fred K L Spijkervet, Arjan Vissink, Frans G M Kroese, Hendrika Bootsma
Journal: Front Oncol (2020)
Keywords: anti pd-l1 therapy, cancer treatment, checkpoint inhibitors, hyposalivation, immune related adverse event, salivary gland dysfunction, sicca syndrome
MeSH terms: Case Reports

Background: Salivary glands (SGs) can be damaged by immune checkpoint inhibitor (ICI) therapy. In patients with ICI-induced SG dysfunction, 60% progress to fulfill classification criteria for primary Sjögren's syndrome (pSS), owing to immune foci in SGs and/or anti-SSA autoantibody positivity. We report the SG tissue analysis of a patient with SG dysfunction after treatment with a programmed death ligand-1 (PD-L1) inhibitor, compared to that of a dry mouth ("sicca") control and pSS patient. Case presentation: The patient received the PD-L1 inhibitor durvalumab (10 mg/kg, every 2 weeks by intravenous infusion) as adjuvant treatment for stage 3 non-small cell lung carcinoma, following concurrent chemo radiotherapy. At 43 weeks after 21 cycles of Durvalumab, the patient was not capable of producing unstimulated or stimulated parotid gland saliva, and a biopsy was taken. Immunohistochemical analysis showed no classical AQP5+ CK7- acinar cell clusters (CK7 marks intercalated ducts, IDs). In contrast, the parenchyma was dominated by hybrid epithelial "structures" with ID-like morphology, containing a mixture of AQP5+CK7-, AQP5-CK7+, and AQP5+CK7+ cells (30 structures/mm2). These structures were present at lower frequencies in sicca control (2/mm2) and pSS (10/mm2) tissue. Hybrid structures contained proliferating (Ki67+) cells and senescent (p16+) cells. Striated ducts showed no abnormal morphology post PD-L1 treatment, in contrast to pSS tissue. PD-L1 expression was detected in the SG parenchyma following anti-PD-L1 therapy. The SG post-PD-L1 therapy further demonstrated focal lymphocytic sialadentitis, harboring disperse, and focal CD4+ T cell-rich infiltrates. CD8+ T cells were also present. In this patient, these CD4+ and CD8+ T cells were observed in-between and inside hybrid structures. CD20+ B-cells were infrequently detected following PD-L1 blockade, in contrast to their preponderance in pSS SG tissue. Conclusion: This patient lacked conventional SG acinar cells following anti-PD-L1 therapy and demonstrated presence of hybrid intercalated duct-like structures. Understanding which mechanisms and dynamics underpinning this aberrant parenchyma may be crucial to understand how SG dysfunction post ICI therapy, and potentially other affected organs. Furthermore, although the patient treated with anti-PD-L1 antibody examined here fulfills the criteria for pSS and demonstrated focal lymphocytic sialadentitis, the further histopathological characteristics do not resemble pSS.
Emergency deep hypothermic circulatory arrest in a 1-year-old undergoing cardiac surgery in a Nigerian hospital - anaesthesia and critical care interventions
Authors: Chidiebele Samuel Ikenga, Balswaroop Sahu, Vincent Okwulehie, Friday Umeh
Journal: Pan Afr Med J (2020)
Keywords: deep hypothermic circulatory arrest, aberrant right subclavian artery, cerebral perfusion, interrupted aortic arch, patent ductus arteriosus, ventricular septal defect
MeSH terms: Cardiovascular Abnormalities / surgery, Circulatory Arrest, Deep Hypothermia Induced / methods, Ductus Arteriosus, Patent / surgery, Heart Septal Defects, Ventricular / surgery, Subclavian Artery / abnormalities, Case Reports, Anesthesia / methods, Cardiovascular Abnormalities / diagnosis, Critical Care / methods, Female, Humans, Infant, Nigeria, Subclavian Artery / surgery, Treatment Outcome, Aberrant subclavian artery

Deep Hypothermic Circulatory Arrest (DHCA) is a technique used to obtain optimal operating conditions while providing cerebral protection. The case report presented a DHCA on an infant that was basically done as an emergency in an attempt to correct a previously unrecognized anomaly. We report a case of a 1-year-old that had surgery for Ventricular Septal Defect (VSD) and Patent Ductus Arteriosus (PDA) ligation but following closure was noted to have no palpable peripheral pulses, only carotid pulsation. This necessitated an emergency reopening of the chest. Close inspection revealed an interrupted aortic arch with aberrant right subclavian artery and pre-surgery PDA supplying both upper and lower limb. DHCA was immediately commenced and the patient cooled to 16°C. The surgeon promptly set out to attach the subclavian artery to the ascending aorta and descending aorta. At the completion of the surgery, the patient was taken to Intensive Care Unit (ICU) for critical care support. She was subsequently discharged after spending a little more than a week in ICU. This procedure is rarely done as an emergency but was instituted in our case in effort to immediately achieve perfusion to the limbs. As the expertise to carry out the procedure is limited, it might be better to develop ways to efficiently ensure the skill set is continually updated.
Odontogenic Cutaneous Fistula: A Cause of Persistent Cervical Discharge
Authors: Nicholas Figaro, Solaiman Juman
Journal: Case Rep Med (2018)
MeSH terms: Case Reports

Odontogenic cutaneous fistulas often lead to intense levels of patient discomfort and suffering. Due to its rarity and the absence of dental symptoms, a considerable number of patients are usually misdiagnosed which results in inappropriate management. This case report presents a 16-year-old patient with a 2-year history of a nonhealing, persistently discharging lesion in the left submandibular region of the neck. The patient underwent exploration of the left submandibular region, and a fistulous tract directed superomedially to the ipsilateral lower molar teeth was excised. A subsequent panoramic orthopantomogram performed one week postoperatively demonstrated radiolucency is the distal root of tooth 37. A final diagnosis of odontogenic cutaneous fistula was made, and the patient was referred to the Maxillofacial Department for treatment of the offending tooth.