OpenMed/multicare-articles huggingface dataset:
Case Report: Disseminated Edwardsiella tarda infection in an immunocompromised patient
Authors: Lucia An, June L Chan, Margaret Nguyen, Shangxin Yang, Jaime G Deville
Journal: Front Cell Infect Microbiol (2023)
Keywords: edwardsiella tarda, case report, disseminated infection, immunocompromised, spontaneous bacterial peritonitis
MeSH terms: Bacteremia / diagnosis, Bacteremia / drug therapy, Enterobacteriaceae Infections / diagnosis, Enterobacteriaceae Infections / drug therapy, Case Reports, Anti-Bacterial Agents / therapeutic use, Edwardsiella tarda, Humans, Immunocompromised Host, Anti-Bacterial Agents

Human infection caused by bacteria of the Edwardsiella genus is rare and most often presents with gastroenteritis that rarely requires antibiotics. Our case report describes a medically complex patient with chronic steroid use contributing to an immunocompromised state, who presented with fever and abdominal pain. The patient was later found to have Edwardsiella tarda (E. tarda) bacteremia and underwent paracentesis confirming E. tarda bacterial peritonitis requiring a prolonged antibiotic course. This case report aims to illustrate the presentation, diagnosis, and management of an uncommon infection that can have severe complications especially among immunocompromised patients.
Huge intermediate-grade myofibroblastic sarcoma in the retroperitoneum revealed by 18F-FDG PET/CT: a case report
Authors: Xiaotian Li, Tengyue Mei, Pan Wang, Jiong Cai, Xianwen Hu
Journal: Front Med (Lausanne) (2024)
Keywords: 18f-fdg, pet/ct, computed tomography, myofibroblastic sarcoma, retroperitoneal
MeSH terms: Case Reports

Myofibroblastic sarcoma (MS) is a relatively rare malignant bone and soft tissue tumor, which originates from myofibroblasts, with some characteristics of both smooth muscle cells and fibroblasts. It can develop in individuals at any age and can affect various regions, especially the head and neck; however, it is rarely reported retroperitoneally. Generally, this type of sarcoma is considered a low-grade malignancy, and cases classified as moderate and high-grade malignancy are rare. In this study, we describe a case of intermediate-grade myofibroblastic sarcoma (IGMS) originating from the retroperitoneum, which was confirmed through pathological diagnosis. The 18F-fluorodeoxyglucose (18F-FDG) positron emission tomography (PET)/computed tomography (CT) scan revealed a large, borderless mass located retroperitoneally with a significantly increased 18F-FDG uptake, accompanied by adjacent visceral and soft tissue infiltration and peripheral lymph node metastasis. The patient received chemotherapy for 3 weeks; however, the tumor did not shrink significantly. Therefore, the patient discontinued the treatment. After 5 months, his condition gradually deteriorated, which eventually led to death. Through this case report, the diagnosis and treatment of moderate malignant retroperitoneal myofibroblastoma were discussed, aiming to increase clinicians' understanding of this disease.
IgG4-related disease complicated by PLA2R-associated membranous nephropathy: A case report
Authors: Meichun Huang, Jun Liu, Xiuxiu Li
Journal: Open Life Sci (2024)
Keywords: igg4-related disease, igg4-related kidney disease, tripterygium wilfordii, membranous nephropathy, phospholipase a2 receptor
MeSH terms: Case Reports

IgG4-related tubulointerstitial nephritis (IgG4-related TIN) is the prevalent clinical manifestation of IgG4-related diseases (IgG4-RD). However, there are limited cases of IgG4-RD occurring with membranous nephropathy (MN) in the absence of phospholipase A2 receptor (PLA2R). There have been no indications of treatment using Tripterygium wilfordii. This study reported a rare case of IgG4-RD with PLA2R-associated MN without any of the distinct IgG4-related TIN. The patient was treated effectively with T. wilfordii. A 71-year-old patient was admitted to the medical facility after presenting with a 1 month history of edema and 8 months of albuminuria. The renal biopsy tissue examination confirmed the presence of MN (phase II) in the absence of pathological manifestations of IgG4-related TIN. Immunohistochemistry identified PLA2R++ (granular capillaries). The serum PLA2R antibody titer was 1:180 (1:20). The patient met the diagnosis with IgG4-RD. Over 8 years of follow-up, the patient was effectively treated with low-dose hormones and T. wilfordii, without any adverse effects. This MN is considered a unique form of IgG4-RD, regardless of whether PLA2R antibodies are present or not. Research suggests that T. wilfordii could be a promising option for elderly people with IgG4-related MN, as it has been found to have fewer adverse effects.
Case Report: A case suffered acute cerebral infarction after removal of temporary cardiac pacing lead which led to the perforation of interventricular septum
Authors: Lu Zhang, Jiechu Chen, Kailun Zhu, Jianghai Liu, Guiyang Li, Dong Chang, Qiang Li
Journal: Front Cardiovasc Med (2024)
Keywords: anticoagulants, artificial, brain, cardiac pacing, embolism and thrombosis, septal perforation
MeSH terms: Case Reports

We report an elderly male patient with frequent episodes of dizziness due to a complete atrioventricular block who underwent temporary pacemaker insertion in a local hospital. After the implantation of a permanent pacemaker and removal of the temporary pacemaker lead, the patient developed sudden neurological symptoms, upon which an acute cerebellar infarction was diagnosed via head CT. We will discuss the adequacy of the periprocedural administration.
Digital subtraction angiography-guided peripheral nerve stimulation via the foramen rotundum for refractory trigeminal postherpetic neuralgia: a case report and literature review
Authors: Qingbang Xu, Fangyuan Zhou, Dong Yang
Journal: Front Neurol (2024)
Keywords: case report, foramen rotundum, maxillary division, peripheral nerve stimulation, trigeminal postherpetic neuralgia
MeSH terms: Case Reports

Postherpetic neuralgia (PHN) is a debilitating complication of varicella-zoster virus infection. This case report presents a novel approach to treating refractory trigeminal maxillary postherpetic neuralgia using digital subtraction angiography (DSA)-guided peripheral nerve stimulation via the foramen rotundum. A 72-year-old female with severe, treatment-resistant pain underwent this intervention. The results demonstrated the disappearance of tactile allodynia, a significant reduction in oral analgesic requirements, and no observed complications or side effects during a 3-year follow-up period. This case highlights the potential effectiveness of DSA-guided peripheral nerve stimulation using a new dorsal root ganglion (DRG) stimulator as an alternative therapy for refractory trigeminal postherpetic neuralgia (TPHN).
Dermal cylindroma of the external auditory canal
Authors: Vivek Dokania, Indranil Mukherjee
Journal: Autops Case Rep (2023)
Keywords: ear, ear canal, ear neoplasms
MeSH terms: Case Reports

External auditory canal (EAC) cylindroma is a rare tumor that mainly presents as a painless mass over the lateral aspect of the ear canal. They have been designated under different nomenclatures in the literature, and controversies persist about their etiology and histogenesis. Moreover, a clinical diagnosis of EAC cylindroma is often challenging because of their rarity and a close resemblance with other adnexal benign and malignant tumors. None of the previous authors have extensively reviewed the dermal cylindroma of the EAC. We provide an extensive review involving PubMed and Google Scholar and report by Preferred Reporting Items for Systematic Reviews and Meta-Analyses standards. A total of 8 cases are included in the current study. The mean age is 55.13 years. There are six females and two males. The left and right ear are involved in 62.50% and 37.50% of cases, respectively. The most common sign/symptom is painless mass (50%). Five authors reported a primary lesion (62.50%), while the remaining 3 reported a recurrent tumor (37.50%). Benign versus malignant cylindroma is reported in 87.50% and 12.50% of cases, respectively. All, except one case, reported a solitary swelling. Surgical excision was employed in all the cases. Primary defect closure versus defect closure with local/distant skin graft /flap is utilized in 37.50% and 62.50% of cases, respectively.
Uncommon Presentation of a Perforated Appendicitis Leading to Duodenal Fistula: Case Report and Literature Review
Authors: Jad El Bitar, Hani Maalouf, Souad Ghattas, Ribal Aby Hadeer, Ahmad Younes, Hind Rahban, Ziad El Rassi
Journal: Case Rep Surg (2024)
MeSH terms: Case Reports

Multiple types of fistulas associated with the appendix have been reported; however, duodenal fistula resulting from perforated acute appendicitis has only been documented in one previous case. In this report, we present the case of an 18-year-old male patient who was diagnosed to have a complicated appendicitis in its normal position with abscess formation. He was started on IV antibiotics and underwent a CT-guided drainage of the abscess with drain placement. Two days later due to biliary output from the drain, CT fistulography and diagnostic laparoscopy were performed that revealed the presence of a duodenal fistula. The potential for duodenal fistula formation in patients with complicated appendicitis must always be taken into consideration. Consequently, it is crucial to establish an appropriate management plan aimed at preventing additional serious complications arising from duodenal perforation.
Generalized lymphatic anomaly involving the pleura and bone in an older male: A case report
Authors: Atsushi Yanagisawa, Akihiro Tamiya, Takayuki Takimoto, Hiromitsu Sumikawa
Journal: Respir Med Case Rep (2023)
Keywords: adult, generalized lymphatic anomaly (gla), multiple bone lesion, pleural lesion
MeSH terms: Case Reports

Generalized lymphatic anomaly (GLA) is a congenital malformation of the lymphatic vessels that is often diagnosed in early childhood. Owing to the rarity and heterogeneity of its clinical course, GLA is frequently misdiagnosed, especially in adults. A 67-year-old man was incidentally found to have bone and pleural lesions. Multiple bone lesions detected on magnetic resonance images were mistaken for malignancy, and pathological evaluation led to the diagnosis of GLA. GLA should be considered in the differential diagnosis of multiple bone lesions, and a proactive biopsy to confirm the diagnosis may help avoid unnecessary invasive procedures.
Diffuse subarachnoid hemorrhage following ventriculo-peritoneal shunt insertion for acute obstructive hydrocephalus from large glomus jugulare tumor: case report
Authors: Mestet Yibeltal Shiferaw, Tsegazeab Laeke Teklemariam, Eyob Zenebe Wondimagegnewu, Dejen Tekiea Gebrewahd, Ermias Fikru Yesuf, Bereket Hailu Mekuria, Sebboona Baisa Abelti
Journal: Front Surg (2024)
Keywords: endovascular embolization, glomus jugulare tumors, hydrocephalus, subarachnoid hemorrhage, vascular malformation, ventriculoperitoneal shunt insertion
MeSH terms: Case Reports

Background:
Glomus jugulare tumors (GJTs) are rare intra-cranial tumors. Commonly, these lesions present with cranial nerve palsies, headaches, and hydrocephalus. Rarely, GJTs present with spontaneous subarachnoid hemorrhage. However, there has never been a report of diffuse subarachnoid hemorrhage following ventriculoperitoneal shunt insertion in a patient who developed hydrocephalus secondary to any brain tumor in general or glomus jugulare tumors in particular.
Observation:
The authors presented an extremely rare complication of diffuse subarachnoid hemorrhage following the insertion of a ventriculoperitoneal shunt (VPS) in a 61-year-old female patient who was diagnosed to have both clinical and radiologic features of acute obstructive hydrocephalus secondary to a highly vascular huge glomus jugulare tumor.
Conclusion:
Subarachnoid hemorrhage following ventriculoperitoneal shunt insertion for hydrocephalus caused by a mass lesion is an extremely rare complication. Preoperative CT angiography should be strongly considered to look for the associated vascular malformations in extremely vascularized mass lesions. Given the not ubiquitous availability of all therapeutic options for GJTs, especially in low and middle income settings contributes for the poor outcome of GJTs and it fosters a global neurosurgery agenda.
Case Report: Adrenal schwannoma associated with ganglioneuroma
Authors: Dong-Lin He, Run-Lin Feng, Jia-Gui Chai, Xin Guo, Chang-Xing Ke
Journal: Front Surg (2024)
Keywords: adrenal gland, adrenal tumors, collision tumors, ganglioneuroma, schwannoma
MeSH terms: Case Reports

Background:
An adrenal collision tumor (ACT) denotes the presence of distinct tumors with diverse behavioral, genetic, and histological features independently co-existing within the adrenal tissue without intermingling, and occurrences of such cases are infrequent. The concurrent occurrence of adrenal schwannoma and adrenal ganglioneuroma is exceedingly rare, and the diagnosis of these ACTs has been notably challenging due to their atypical clinical manifestations and imaging characteristics.
Case summary:
A 37-year-old man presented to the hospital 3 weeks after a computed tomography (CT) examination that revealed a left adrenal mass. Physical examination findings were unremarkable. Both CT and magnetic resonance imaging scans indicated the presence of a left adrenal mass. Plasma cortisol, adrenocorticotropic hormone, and renin-angiotensin-aldosterone system tests yielded normal results. Preoperative imaging confirmed the diagnosis of left adrenal pheochromocytoma. After thorough surgical preparation, a laparoscopic partial left adrenalectomy was performed. Subsequent postoperative pathological analysis identified adrenal schwannoma in conjunction with adrenal ganglioneuroma. The patient recovered well and was discharged on postoperative day 4. A routine urology clinic visit was included in his postoperative care plan. During follow-up assessments, CT scans of the left adrenal gland revealed no abnormalities.
Conclusion:
Adrenal schwannoma combined with ganglioneuroma represents an exceptionally rare collision tumor characterized by the absence of typical clinical or imaging features, leading to potential misdiagnosis. Adrenal incidentalomas present as multifaceted conditions, and this case serves to heighten awareness of their intricate nature. Due to the challenges in preoperative differentiation of various adrenal mass types, postoperative pathological analysis is imperative for guiding the subsequent treatment course for the patient.
Non-financial access to healthcare services in rural areas: A case study of people with disabilities living in Northern Iran
Authors: Lida Shams, Tahere Darvish, Mohamad Meskarpour Amiri, Sayyed-Morteza Hosseini-Shokouh, Taha Nasiri
Journal: PLoS One (2023)
MeSH terms: Disabled Persons, Delivery of Health Care, Health Services, Health Services Accessibility, Humans, Income, Iran

Introduction:
Access to healthcare for persons with disabilities (PWDs) is an important but often ignored issue for achieving universal health coverage. The current study aimed to investigate PWDs' access to healthcare in the rural areas in north of Iran.
Methods:
Following a descriptive-analytical design, 471 persons with disabilities (PWDs) living in the Nor city, Mazandaran province, were selected using quota sampling. Data were collected by a valid and reliable questionnaire that contained dimensions of time, geography, physical, and acceptability using face-to-face interviews. The findings are provided by central and dispersion indicators and analyses are performed with linear Regression using SPSS version 17.
Results:
PWDs had moderate access to healthcare services in all dimensions. The regression models for access to health services in all four dimensions were significant (p<0.05). The results showed that in the geographical dimension, the variables of marital status, income, receipt of financial aid, supplementary insurance, and type of disability; in the physical dimension, the variables of income, responsibility for taking care of the family, supplementary insurance, and type of disability; in the time dimension, supplementary insurance, home area, and type of disability; and in the aspect of service acceptability, only the variables of type of disability and internet access had a significant effect (p<0.05).
Conclusion:
A small percentage of PWDs had high access to health services. Hence, improving their access to healthcare services, particularly in rural and less developed areas, and developing appropriate policies should be the focus of Iranian policy-makers.
Case Report: In situ pulmonary artery thrombosis in a 12-year-old girl classified as systemic lupus erythematosus
Authors: Yong Feng, Ning Chen, Bing Dai, Yunxiao Shang
Journal: Front Pediatr (2024)
Keywords: antiphospholipid syndrome, child, in situ pulmonary artery thrombosis, pulmonary embolism, systemic lupus erythematosus
MeSH terms: Case Reports

In situ pulmonary artery thrombosis (ISPAT) is a relatively rare but potentially life-threatening complication of systemic lupus erythematosus (SLE) in children. We report the case of a 12-year-old girl who presented with fever, chest pain, and dyspnea. Immune thrombocytopenia was identified due to purpura and menorrhagia 3 months before presentation with a lowest platelet count of 12 × 109/L. The sudden onset of fever, chest pain, and dyspnea were misdiagnosed as hyperinflammatory responses caused by pneumonia; these symptoms ameliorated with glucocorticoid and antibiotic treatment. The reappearance of symptoms after dose reduction of glucocorticoids and the observation of bloody bronchoalveolar lavage fluid necessitated further evaluation. Pulmonary artery thrombosis/embolism was identified using computed tomography pulmonary angiography and high D-dimer quantitative level of 4,118 μg/L (normal <252 μg/L). Ultrasonography of the deep and superficial veins of both lower limbs and renal veins revealed no thrombosis, suggesting the diagnosis of ISPAT. Further etiological evaluation revealed positive antinuclear antibodies, lupus anticoagulant, and anti-SSA antibodies, confirming SLE. Repeated normal urine analysis indicated that lupus nephritis was unlikely. Further, the negative anticardiolipin and anti-β2 glycoprotein antibodies and temporary positive lupus anticoagulant suggested that antiphospholipid syndrome was unlikely. The patient received anticoagulants, glucocorticoids, hydroxychloroquine, and mycophenolate therapy. Her symptoms gradually improved, and she was discharged. At the 1-month follow-up, the thrombosis had resolved. During the 1-year follow-up, her condition remained well without SLE relapse. Our experience with this case emphasizes searching for SLE in the case of ISPAT and pulmonary hemorrhages. ISPAT can occur in children with SLE and may be caused by hyperinflammatory response during SLE flare.
Bilateral adrenal giant medullary lipoma combined with disorders of sex development: a rare case report and literature review
Authors: Chenghao Zhanghuang, Na Long, Zhen Yang, Yucheng Xie
Journal: Front Oncol (2023)
Keywords: adrenal gland, adrenal myelolipoma, diagnosis, disorders of sex development (dsd), pathology
MeSH terms: Case Reports

Bilateral adrenal myelolipoma is rare in clinics and patients with disorders of sex development (DSDs). One case was reported in our center. A 45-year-old patient was admitted to the hospital after discovering a left abdominal mass for more than a year and worsening abdominal pain for 18 days. An imaging examination showed bilateral adrenal masses. Physical examination showed clitoris hypertrophy with patelliform changes, thick and dense pubic hair, normal development of bilateral labia majora without labia minora, and urethral opening. After the relevant preoperative examinations, bilateral adrenal mass resection was performed under general anesthesia. The postoperative pathology confirmed adrenal myelolipoma. The incision healed well without recurrence over 10 years after the operation. Her enlarged clitoris decreased in size. This case report has a detailed diagnosis and treatment process and sufficient examination results. It can provide a reference for diagnosing and treating patients with bilateral adrenal myelolipoma and DSD and reduce the risk of misdiagnosis and mistreatment.
Case Report: Culture-negative Prosthetic Valve Endocarditis
Authors: Ignacio D Velazquez, Kenneth K Woo, Mohammed Siddiqui, Sion K Roy
Journal: Curr Cardiol Rev (2023)
Keywords: fungal endocarditis, aspergillus endocarditis, culture-negative infective endocarditis, prosthetic valve endocarditis, transesophageal echocardiograms, treatment
MeSH terms: Anti-Infective Agents, Endocarditis, Bacterial / complications, Endocarditis, Bacterial / diagnostic imaging, Heart Valve Prosthesis / adverse effects, Case Reports, Aortic Valve / diagnostic imaging, Aortic Valve / surgery, Echocardiography / methods, Humans, Anti-Infective Agents

Background:
Prosthetic valve endocarditis can be difficult to diagnose and cause significant morbidity and mortality, especially when no culture data are available to guide therapy. Transthoracic and transesophageal echocardiograms, the studies of choice for initial endocarditis evaluation, can be less reliable due to artifact and post-surgical changes. Some less common forms of endocarditis may be difficult to culture and, due to their fastidious nature, may delay the identification of causative organisms. Given the lack of directed antimicrobial treatment, culturenegative prosthetic valve endocarditis is specifically difficult. A wide differential diagnosis is critical to make a timely diagnosis and initiate treatment.
Case presentation:
We present a case of a patient presenting with dyspnea which was found to have culture-negative endocarditis requiring mitral and aortic valve replacement that ultimately was complicated with culture-negative prosthetic valve endocarditis. Identifying a culprit organism made appropriate and timely antimicrobial treatment difficult, ultimately resulting in the patient dying from endocarditis complications.
Conclusion:
A high index of suspicion is needed when managing infective endocarditis, especially when prosthetic valves are involved. Diagnostic accuracy of cultures and echocardiography may be reduced when dealing with prosthetic valve endocarditis; thus, alternative methods of diagnosis may be required to make a timely diagnosis of causative organisms.
Case Report: Transthyretin Glu54Leu-a rare mutation with predominant cardiac phenotype
Authors: Mariana Gospodinova, Sashka Zhelyazkova, Teodora Chamova, Ognyan Asenov, Zornitsa Pavlova, Tihomir Todorov, Dilyana Mikova, Yordan Palashev, Ivan Gruev, Atanas Kundurdjiev, Albena Todorova, Ivailo Tournev
Journal: Front Cardiovasc Med (2023)
Keywords: amyloidosis, cardiomyopathy, pglu74leu (glu54leu) mutation, polyneuropathy, transthyretin
MeSH terms: Case Reports

We report two unrelated Bulgarian families with hereditary transthyretin (ATTR) amyloidosis due to a rare p.Glu74Leu (Glu54Leu) pathogenic variant found in seven individuals-three of them symptomatic. Only one family with the same variant and with a Swedish origin has been clinically described so far. Our patients are characterized by predominant cardiac involvement, very much similar to the Swedish patients. Although the initial complaint was bilateral carpal tunnel syndrome, advanced amyloid cardiomyopathy was found in two symptomatic carriers at diagnosis with heart failure manifestations. The neurological involvement was considered as mild, with mainly sensory signs and symptoms being present. We followed a non-biopsy algorithm to confirm the diagnosis. Tafamidis 61 mg has been initiated as the only approved disease modifying treatment for ATTR cardiomyopathy. Clinical stability in the absence of adverse events has been observed at follow up.