bluuebunny/crossref_metadata_2025 huggingface dataset:
The “Ballerina” Sign: Posterior Capsular Rupture during Cataract Surgery
Authors: Ramon Dan , Shemesh Nadav , Achiron Asaf
Published: August 2024

Introduction: In this case study, we present the “Ballerina” sign as a potentially valuable clinical indicator for detecting posterior capsular rupture (PCR) during cataract surgery. The purpose of this study is to highlight the significance of PCR in the context of cataract surgery and introduce this novel sign. Case Presentation: During the cataract operation on a 70-year-old patient, we observed a spiraling fragment of the nucleus attached to a vitreous string. Subsequently, an anterior vitrectomy was successfully performed without further intraoperative complications, followed by the insertion of a 3-piece lens into the sulcus. Positive visual outcomes were observed during postoperative follow-up appointments at 1 day, 2 weeks, and 3 months. Conclusion: The documentation of the “Ballerina” sign in the medical literature is innovative, offering ophthalmic surgeons a valuable tool for early PCR detection during cataract surgeries.
Portable Percussive Massage Device-Related Rectus Sheath Hematoma and Obstructive Uropathy: A Case Report
Authors: Kwon Lawrence
Published: August 2024

Introduction: Rectus sheath hematomas (RSHs) can occur from the rupture of the epigastric arteries, leading to blood accumulation within the rectus abdominis sheath. Herein, we report the unique case of an RSH resulting in acute ureteral obstruction, which was associated with the use of a handheld deep-tissue percussive massage device in attempts to relieve abdominal pain. Case Report: A morbidly obese man in his late 50s was admitted with complications of COVID-19, including acute respiratory syndrome, bilateral peroneal deep vein thromboses, and acute kidney injury. He was treated with anticoagulants (subcutaneous enoxaparin and apixaban), dexamethasone, and remdesivir. He developed severe abdominal pain, and a large (14 × 17 cm) right rectus sheath and an extraperitoneal pelvic hematoma were identified by computed tomography. The hematoma extended across the midline into the left pelvis and the retroperitoneum with associated mild right hydronephrosis. A handheld percussive massage device (Theragun®) was applied repeatedly at the site of enoxaparin injection into the abdominal wall in attempts to alleviate the pain. On day 12, bilateral nephrostomy tubes were inserted. A nephrostogram revealed mild hydroureteronephrosis to the ureterovesicular junction bilaterally and extrinsic compression of the bladder. On day 17, the patient suffered a cardiac arrest and died. Conclusion: This case report offers insights into the pathophysiology of obstructive uropathy and is a reminder of the importance of considering uncommon causes of obstructive uropathy in the diagnosis and management of AKI, particularly in hospitalized patients receiving anticoagulation. We know of no previous reference of obstructive uropathy caused by retroperitoneal hematoma associated with the use of a portable massage device applied to the abdominal wall.
Oesophageal Lichen Planus Successfully Treated with Budesonide Orodispersible Tablets: A Case Report
Authors: Bagger-Jörgensen Harald , Abdulrasak Mohammed , Sandeman Kevin , Binsalman Mohammed , Sjöberg Klas
Published: August 2024

Introduction: Lichen planus is a relatively common inflammatory condition of the nails, skin, and mucosal surfaces. Oesophageal involvement of lichen planus is thought to be very rare, mainly described in case reports, but is associated with a high risk of oesophageal stenosis as well as squamous cell carcinoma. No evidence-based treatment recommendations exist, with the majority of described treatment regimens involving systemic immunosuppression. Case Report: In this case report, we describe a novel approach in treating oesophageal lichen planus in a patient with budesonide orodispersible tablets, a treatment normally reserved for eosinophilic oesophagitis. The patient achieved complete relief of dysphagia, with a follow-up oesophagogastroduodenoscopy 2 months after treatment commencement being macroscopically and microscopically free of inflammatory activity. This case report is to our knowledge the first to report this treatment regimen in oesophageal lichen planus. Conclusion: We consider a trial of budesonide orodispersible tablets a reasonable initial management as it’s a local therapy specific to the oesophagus with a more benign side effect profile than systemic immunosuppression, but further studies need to be undertaken to corroborate our findings. Also, based on the severity and malignant potential of oesophageal lichen planus, we suggest that physicians be liberal in ordering oesophagogastroduodenoscopy with biopsy taking as part of the workup of dysphagia in a patient with known lichen planus.
Characteristic Skin Eruptions with a Distinct Histological Pattern Allow Early Diagnosis of Vitamin C Deficiency
Authors: Mirkovic Goran , Mutke Markus Reinhold , Muehleisen Beda
Published: August 2024

Introduction: Scurvy has become a rare disease in western countries with potentially high morbidity. Early diagnosis is crucial and can be challenging. Case Presentation: We present the case of a 56-year-old male patient who developed hemorrhagic diathesis after trivial impact trauma. Previously, the patient suffered from fatigue and loss of appetite. Characteristic skin eruptions and a distinct skin histology along with a decreased serum vitamin C level led to the diagnosis of scurvy. Following vitamin C supplementation, symptoms improved rapidly. Conclusion: In conclusion, vitamin C deficiency should be considered in cases with unclear hemorrhagic diathesis and a medical history of nutritional irregularities. Especially in cases of scurvy that do not yet show the full clinical spectrum of symptoms or have only moderately decreased serum vitamin C levels, thorough clinical dermatological examination and a skin biopsy are essential for early diagnosis and to prevent complications.
An Unusual Presentation of Myeloperoxidase-Associated Glomerulonephritis and Suspected IgA-Mediated Anti-Glomerular Basement Membrane Disease: A Case Report
Authors: Twomey Brenner Ciaran , Saha Sujit , Bramham Kate , Vinen Katie , Horsfield Catherine , Lioudaki Eirini
Published: August 2024

Introduction: Anti-glomerular basement membrane (GBM) disease is a rare cause of glomerulonephritis usually mediated by IgG antibodies and is associated with ANCA-associated glomerulonephritis in up to 50% of cases. IgA-mediated anti-GBM disease is extremely rare and presents diagnostic difficulties as circulating IgA antibodies will not be detected by standard serological tests for anti-GBM disease. Case Presentation: We present the case of a 67-year-old man with rapidly progressive glomerulonephritis requiring haemodialysis at presentation. Serological testing was positive for anti-myeloperoxidase and negative for IgG anti-GBM antibodies. Kidney biopsy revealed necrotizing crescentic glomerulonephritis with linear staining of IgA along the GBM. He was treated with a combination of immunosuppression and plasma exchange and was able to become dialysis-independent. Conclusion: To our knowledge, this is the first documented “double-positive” IgA anti-GBM disease and ANCA-associated glomerulonephritis.
Eruptive Melanocytic Nevi in the Setting of Encorafenib, Cetuximab, and Binimetinib Combination Therapy: A Case Report
Authors: Lam Karen , Gates Gregory A. , Bach Daniel Q. , Cheng Kyle
Published: August 2024

Introduction: The development of new and changing melanocytic lesions has been increasingly reported as an adverse dermatologic toxicity of BRAF inhibitor therapy. Melanocytic lesions and melanomas induced by BRAF inhibitor therapy that lack BRAF V600E expression have been less commonly described. One mechanism that has been proposed for the development of BRAF inhibitor-induced melanocytic lesions, including those lacking BRAF V600E expression, is the paradoxical activation of the MAPK signaling pathway in BRAF wild-type (BRAFWT) cells. Case Presentation: Herein, we report a rare case of a 39-year-old woman who developed numerous BRAF V600E-negative eruptive melanocytic nevi following encorafenib, cetuximab, and binimetinib combination therapy, the current standard of care for the treatment of BRAF-mutant metastatic colorectal cancer. Conclusion: Patients treated with BRAF inhibitors, with or without related combination therapies, who develop BRAFWT melanocytic lesions are at risk for developing both dysplastic nevi and melanoma, thereby warranting baseline dermatoscopic evaluation prior to the initiation of therapy as well as regular follow-up during and after treatment.
Prognostic Value and Clinical Significance of Vesicular Transport-Related Genes in Clear Cell Renal Cell Carcinoma
Authors: Ye Dayong , Luo Jingxian , Wang Yujie , Su Mingqiang , Zhang Dayong , Han Dengjun
Published: August 2024

Introduction: Vesicular transport (VT) has a complex relationship with tumor progression and immunity. But prognostic significance of VT in clear cell renal cell carcinoma (ccRCC) is unclear. Thus, we aimed to establish a prognostic model according to VT to predict overall survival of ccRCC patients. Methods: We used patient data from TCGA database and built a prognostic model with 13 VT-related genes (VTRGs) by differential expression analysis, LASSO regression, and univariate/multivariate Cox analysis. The model was validated internally and externally, and survival analysis and ROC curves depicted excellent predictive ability. Furthermore, higher modeled riskscores corresponded to more advanced tumor progression. To further understand the potential reasons for different prognoses in patients, we did enrichment analysis on differentially expressed genes identified by the model in risk groups. The expression levels and roles of SAA1 and KIF18B in ccRCC were verified by qRT-PCR and cell function experiments. Results: Humoral immune response and cAMP signaling pathway may be the biological processes and pathways leading to poor prognosis. Further analysis of immune microenvironment presented that ccRCC patients with poor prognoses had highly immune-infiltrated characteristics. We compared the drug response data of samples from different prognostic patients in the GDSC database and identified drug sensitivity differences associated with prognosis. Finally, we demonstrated that SAA1 and KIF18B could increase the proliferation, migration, and invasion ability of ccRCC cells using cellular experiments. Conclusion: In summary, we further revealed the importance of VTRGs in ccRCC prognosis.
Reduced CC Chemokine Receptor 4 Expression in Tumor Cells after Lenalidomide Treatment for Adult T-Cell Leukemia/Lymphoma: A Case Report
Authors: Shimizu Masatomo , Yokote Taiji , Hatooka Jun , Kinoshita Yuuki , Imagawa Akihisa , Yasuda Emi
Published: August 2024

Introduction: CC chemokine receptor 4 (CCR4), which is involved in leukocyte migration, is expressed in most tumor cells in patients with adult T-cell leukemia/lymphoma (ATLL). Case Presentation: Here we report the case of a 78-year-old man diagnosed with lymphoma-type ATLL expressing CCR4. The patient was administered two cycles of lenalidomide but died because of sepsis 5 months after the initial diagnosis. Autopsy revealed ATLL cells at several sites. Immunohistochemical analysis revealed that these ATLL cells had reduced CCR4 expression. Conclusion: The present case suggests that treatment should be carefully determined in ATLL with reference to a history of lenalidomide use and CCR4 expression.
The First Pediatric Case of an IFT140 Heterozygous Deletion Causing Autosomal Dominant Polycystic Kidney Disease: Case Report
Authors: Seeman Tomáš , Šuláková Terezie , Bosáková Alice , Indráková Jana , Grečmalová Dagmar
Published: August 2024

Introduction: Autosomal dominant polycystic kidney disease (ADPKD) is the most common hereditary kidney disease, which is mainly caused by pathogenic variants in two particular genes: PKD1 and PKD2. ADPKD caused by variants in other genes (GANAB or IFT140) is very rare. Case Report: In a 6-year-old girl examined for abdominal pain, a cystic mass in the upper part of the right kidney was detected during an abdominal ultrasound. She was referred to pediatric oncology and urology for suspicion of a tumorous mass and the condition was assessed as a cystic nephroma. A heminephrectomy was then performed on the upper cystic part of the right kidney. The histological examination was inconclusive; therefore, genetic testing was recommended. Kidney and liver cysts were detected sonographically in the mother, but DNA analysis of the PKD1 and PKD2 genes did not reveal any pathogenic variant; the cause of the pathological formation in the kidneys remained unclear. Nine years later, next-generation sequencing of a panel of genes for kidney disease was performed and a heterozygous deletion was found on chromosome 16; this included exon 13 of the IFT140 gene. The same deletion was found in the patient’s mother. Currently, the patient is 14 years old and has mild sonographic findings, normal glomerular filtration, mild proteinuria, and hypertension. Conclusion: Pathogenic variants of the IFT140 gene very rarely cause ADPKD; however, they should be considered in all children with autosomal dominant forms of PKD and asymmetric/atypical cystic kidney involvement or negative findings of PKD1 and PKD2.
Applications of Synthetic Hybrid-Scale Fiber Matrix in Head and Neck Reconstruction: A Case Series
Authors: Horwich Peter , Sivarajah Shanmugappiriya , Neskey David , Hornig Joshua
Published: August 2024

Introduction: Patients with head and neck cancer often necessitate complex reconstructions, considering both functional and esthetic concerns. Reconstructions are further complicated by previous radiation therapy and patient co-morbidities, which impair wound healing. A recently introduced synthetic hybrid-scale fiber matrix has been shown to provide durable wound coverage and promote tissue healing as an alternative to traditional biologic allogenic and xenogenic skin substitutes. Case Presentation: Thirteen patients were treated at a single academic hospital between December 1, 2021, and May 1, 2023 with the synthetic matrix in head and neck reconstructions. Reconstructions included exposed muscle, scalp wounds, intra-oral defects, and radial forearm free flap donor sites. Wound sizes ranged from 2 × 2 cm to 18 × 10 cm. Serial photographs were taken to evaluate wound healing at 1, 4, 8, 12, and 16 weeks timepoints after application. Outcomes measured at each timepoint included wound size, presence of granulation tissue, and extent of epithelialization. No hematomas or wound complications were encountered. Complete wound healing was noted between 6 and 12 weeks, dependent on wound size. The synthetic matrix significantly promoted wound healing via early granulation tissue formation and epithelialization, or mucosalization, in all head and neck applications. Scar formation and contracture were acceptable in all cases. Conclusion: The use of synthetic hybrid-scale fiber matrix promotes wound healing and avoids patient morbidity associated with traditional allogenic and biogenic treatments, such as split-thickness skin grafts. This synthetic matrix has been demonstrated to be a valuable asset in the head and neck reconstructive armamentarium.
Exploring the Mechanism of Cardiorenal Protection with Finerenone Based on Network Pharmacology
Authors: Si Yachen , Zhu Ying , Liu Jing , Liu Shuyu , Cai Xiaomin , Gu Ying , Li Hanqing , Pan Fang , Wang Wenjuan , Shangguan Jianying , Liu Rui , Xi Chunsheng , Wang Lijun
Published: August 2024

Introduction: Large prospective trials have demonstrated that finerenone could reduce the risk of cardiovascular death and progression of renal failure among patients with chronic kidney disease associated heart failure and/or type 2 diabetes mellitus (T2DM). The aim of this study was to explore the molecular mechanism of finerenone in the treatment of cardiorenal diseases through network pharmacology. Methods: The STITH, SwissTargetPrediction, PharmMapper, DrugBank, and ChEMBL databases were used to screen the targets of finerenone. The disease-related targets were retrieved from the DisGeNET, GeneCards, CTD, OMIM, and MalaCards databases. The protein-protein interaction (PPI) network was conducted with STRING database and Cytoscape software. The clusterProfiler R package was used to perform Gene Ontology (GO) and Kyoto Encyclopedia of Genes and Genomes (KEGG) pathway enrichment analysis. The interactions of key targets and finerenone were analyzed by molecular docking in Autodock software. Diabetes mellitus was induced by intraperitoneal injection of streptozotocin. Histopathology of myocardial and renal tissues was observed by hematoxylin-eosin (HE) staining, and detection of protein expressions was conducted using Western blotting. Results: A total of 111 potential cardiorenal targets of finerenone were identified. The main mechanisms of action may be associated with lipids and atherosclerosis, fluid shear stress and atherosclerosis, AGE-RAGE signaling pathway in diabetic complications, and diabetic cardiomyopathy. The hub targets demonstrated by the PPI network were CASP3, ALB, MMP9, EGFR, ANXA5, IGF1, SRC, TNFRSF1A, IL2, and PPARG, and the docking results suggested that finerenone could bind to these targets with high affinities. HE staining revealed the cardiorenal protection of finerenone on diabetic mice. In addition, the protein expressions of CASP3 and EGFR were increased while ALB was decreased in myocardial and renal tissues in diabetic mice compared with control mice, which were reversed by finerenone. Conclusion: This study suggested that finerenone exerts cardiorenal benefits through multiple targets and pathways.
O80:H2-Associated Hemolytic Uremic Syndrome without Hemorrhagic Colitis: A Case Report
Authors: Yoshida Sawako , Tanaka Eriko , Kiuchi Zentaro , Nunokawa Saaya , Kawahara Ayumi , Iyoda Sunao , Narita Masami
Published: August 2024

Introduction: Hemolytic uremic syndrome (HUS) is characterized by progressive kidney injury accompanied by thrombotic microangiopathy, which is clinically defined as microangiopathic hemolytic anemia with thrombocytopenia and organ injury. Shiga toxin-producing Escherichia coli (STEC)-HUS is caused by infection with pathogenic E. coli strains, typically O157, O26, and O111. However, the prevalence of other types of pathogenic E. coli has been increasing, and these pathogens sometimes cause atypical clinical manifestations of STEC-HUS. Case Presentation: We report the case of a 3-year-old girl diagnosed with STEC-HUS associated with a rare O80:H2 stx2 serotype, characterized by an atypical clinical course. She presented with severe hemolytic anemia and mild renal dysfunction but did not have enterohemorrhagic diarrhea. The first culture test of her stool sample collected using a swab upon admission yielded no signs of STEC, leading to an initial diagnosis of atypical HUS; thus, eculizumab was administered adding to red blood cell transfusion and recombinant thrombomodulin alfa and haptoglobin. However, a subsequent culture test of her second stool sample revealed the presence of O80:H2 stx2, confirming the diagnosis of STEC-HUS. Subsequently, the patient’s condition improved, and her serum creatinine level gradually normalized over the course of 3 months. Conclusion: Diligently diagnosis is crucial in cases lacking typical STEC-HUS symptoms. We advocate for repeated stool culture testing to ensure accurate identification and timely management of such cases.
Unilateral Anterior Spinal Artery Syndrome following Spinal Anesthesia for Cesarian Section: A Case Report
Authors: Kraus Matthew J. , Nguyen Joseph
Published: August 2024

Introduction: Spinal cord infarction is a rare but serious neurologic complication of spinal anesthesia. Direct vessel injury, intra-arterial anesthetic injection, and anesthetic-induced local hypotension are potential mechanisms of infarction during this procedure. The proximity of the artery of Adamkiewicz to the spinal levels used for spinal anesthesia may also play a role. This case of unilateral anterior spinal artery syndrome highlights the potential for an atypical pattern of injury and deficits due to the complexity of the spinal cord’s anterior circulation. Case Presentation: We present a 38-year-old female patient who presented with left lower extremity weakness, loss of temperature sensation, and urinary retention following spinal anesthesia for cesarian section. Magnetic resonance imaging of the spine demonstrated T2 hyperintensities in the left central spinal cord from T8 to the conus medullaris. A diagnosis of spinal cord infarction was made after lumbar puncture testing showed no evidence of inflammatory myelitis. The patient was treated with steroids empirically until lumbar puncture testing showed no inflammation. The patient was discharged on daily aspirin with persistent left lower extremity weakness and loss of temperature sensation. A plan for outpatient physical therapy was made for rehabilitation. Conclusion: Awareness of the potential for spinal cord infarction secondary to spinal anesthesia must increase among anesthesiologists, obstetricians, and neurologists. The risk of systemic hypotension during and after spinal anesthesia is important to recognize for both primary and secondary prevention of this complication. The hyperacute onset of myelopathic symptoms should point neurologists to investigate an ischemic etiology in the proper clinical context.
Motivational Interviewing to Address Vaccine Hesitancy: Insights from an Intervention in Portugal
Authors: Mendonça Joana , Gouveia Luís , Hilário Ana Patrícia
Published: August 2024

Research, to date, suggests the need for healthcare professionals to adapt their communication style to the needs and characteristics of parents when discussing childhood vaccination. Motivational interviewing (MI) has been recognized as an important communication style, based on a patient-centred model of care, leading to the responsibility and motivation to change in cases of vaccine hesitancy. The current study intended to assess the attainment of MI competencies by healthcare professionals in Portugal who may interact with vaccine-hesitant parents. The pre- and post-training survey results showed a significant increase in the participants’ scores in all three pre-defined dimensions: acquisition of knowledge, perception of skills application, and sense of confidence. Our results suggest that integrating MI training into healthcare professionals’ curriculum appears to be a promising avenue to pursue to foster their immunization-related communication skills in Portugal and elsewhere.
Apocrine Adenoma of the Breast Showing Unique Image Findings
Authors: Kondo Senri , Oura Shoji
Published: August 2024

Introduction: Apocrine adenoma of the breast is extremely rare and its typical images remain uncertain. Case Presentation: A 66-year-old woman was incidentally found of her left breast tumor with computed tomography for ascending colon cancer staging work. Mammography showed a well-demarcated oval mass. Ultrasonography showed an oval mass with indistinct borders, internal iso-echoes with multiple high echo spots, and unchanged posterior echoes. Magnetic resonance imaging of the mass showed a hypo-intense pattern both on T1- and T2-weighted images and a long-lasting rim enhancement pattern up to the late phase on time-signal intensity images. Probably due to the inappropriate tissue sampling, no definitive pathological diagnosis was obtained with core needle biopsy, forcing us to do lumpectomy of the breast mass at the time of colon cancer surgery. The resected breast mass was hard, clearly demarcated, and 8 mm in size. Cut surface of the mass was flesh-colored and had multiple white spots. Pathological study showed dense predominantly oval glandular ducts with luminal apocrine cell layers, outer myoepithelium cell layers, and slight fibrosis. Glandular cavities on pathology well matched to the white spots on macroscopic view, seemed vacant in a cyst-like fashion, and had aggregated foamy histiocytes in some of them. These pathological findings led us to the diagnosis of apocrine adenoma of the breast. Conclusion: Diagnostic physicians should note this extremely rare breast disorder and its image findings.
Clearance of Hepatitis C Virus following Immune Checkpoint Inhibitor Therapy for Hepatocellular Carcinoma: Case Report
Authors: Wilson Harry , Macdonald Douglas , Bryce Kathleen
Published: August 2024

Introduction: Patients with advanced hepatocellular carcinoma (HCC) have limited treatment options in the context of decompensated cirrhosis. HCC occurs in patients with hepatitis C virus (HCV) infection and cirrhosis at 1–4% per year. Direct-acting antiviral (DAA) efficacy is decreased in the presence of HCC. We present a case where immunotherapy may have resulted in HCV clearance, when DAA therapy had been ineffective. We hypothesise that immune checkpoint inhibitors targeting the PD-1/PD-L1 pathway can reverse T-cell exhaustion and aid in the clearance of chronic HCV. Case Presentation: This case study describes a male in his 40 s identified by a re-engagement initiative for HCV, who had been unaware of his diagnosis. On further investigation he was found to have compensated for liver cirrhosis and HCC. He was treated with HCV DAA therapy (sofosbuvir/velpatasvir) and then systemic immunotherapy for HCC with atezolizumab and bevacizumab, in an attempt to downstage the disease. Hepatitis C therapy did not achieve sustained virological response, with viral relapse after the end of treatment. This, combined with ongoing alcohol use, resulted in hepatic decompensation and cessation of immunotherapy after the fifth cycle. The HCV RNA subsequently became undetectable without further DAA re-treatment. Conclusion: To our knowledge, this is the first case of HCV clearance after DAA relapse and the timing of this event after immunotherapy suggests a causal link. We hypothesise that this may be due to the reversal of antiviral T-cell exhaustion. This would therefore support further investigation into other chronic viral infections that create tumour associated with immunosuppressive microenvironments.
Intramuscular Hemangioma of Lateral Rectus Muscle with Rare Presentation as an Epibulbar Mass: A Case Report and Review of Literature
Authors: Nag Adwaita , Krema Hatem , Kamil Zaid Saeed , Al-Mbaideen Suzan
Published: August 2024

Introduction: Intramuscular hemangiomas of extraocular muscles are extremely rare tumors that usually present as retro-orbital masses causing proptosis. We describe a previously unreported presentation, in the form of an epibulbar mass; this easily accessible location allows direct imaging, complete surgical resection, and histopathological confirmation, providing a unique perspective. Case Presentation: A 69-year-old woman presented with a painless dark red mass in the lateral part of the right eye, which had been slowly enlarging over the last 18 months. Clinical features and imaging were suggestive of a benign vascular tumor of the conjunctiva. During surgical resection, the mass was observed to be enmeshed within the fibers of the lateral rectus muscle. Careful dissection from muscle fibers was needed for complete excision. Histopathology revealed the diagnosis of an intramuscular hemangioma of extraocular muscle. Conclusion: In this report, we describe the atypical anterior epibulbar presentation of intramuscular hemangioma of the lateral rectus muscle. We discuss the differential diagnoses and management of this rare tumor along with a review of existing literature. Careful surgical resection achieved complete resolution in this case without recurrence.
Severe Cutaneous Manifestation of Malignant Syphilis in a Patient with Behçet’s Uveitis: A Case Report
Authors: Charoenphol Pulthip , Choopong Pitipol , Sitthinamsuwan Panitta , Leeyaphan Charussri , Rujitharanawong Chuda , Boonsopon Sutasinee , Tesavibul Nattaporn , Tungsattayathitthan Usanee
Published: August 2024

Introduction: Syphilis exhibits a wide range of clinical presentations, mimicking various systemic and ocular diseases. Ocular syphilis, in particular, manifests with multiple presentations, ranging from anterior uveitis to panuveitis, making it a potential differential diagnosis for Behçet’s uveitis. Here, we present a unique case of Behçet’s panuveitis that was undergoing immunomodulatory therapy and was complicated by ocular syphilis. Notably, this case also featured rare cutaneous manifestations associated with secondary syphilis, commonly known as malignant syphilis. Case Presentation: A 38-year-old Thai man with refractory end-stage Behçet’s panuveitis reported a maculopapular rash accompanied by increased intraocular inflammation. The escalation of immunomodulatory therapy, intended to manage the provisional diagnosis of active ocular and cutaneous Behçet’s disease, resulted in clinical deterioration, with the rash transforming into multiple noduloulcerative lesions. Despite negative serologic tests for syphilis at baseline before initiating immunomodulatory therapy, syphilis infection was eventually diagnosed following reevaluation and the observation of spirochetes in a skin biopsy specimen. The patient was treated with intravenous penicillin G, resulting in an improvement in intraocular inflammation and resolution of noduloulcerative rashes. Conclusion: Intraocular inflammation and mucocutaneous lesions in syphilis can mimic the presentation of Behçet’s disease. The introduction of immunomodulatory therapy may alter the clinical picture, leading to a severe and atypical presentation. A high index of suspicion for reevaluating serologic tests or performing tissue biopsies is warranted for an accurate diagnosis.
The Pleural Origin of Retiform Hemangioendothelioma: An Unusual Origin of a Rare Diagnosis
Authors: Ullah Hidayat , Khan Imran , Khan Aria , Tangri Apoorva , Lamichhane Saral , Maqbool Shahzaib , Nashwan Abdulqadir J.
Published: August 2024

Introduction: Vascular malignancies are categorized into benign hemangiomas, malignant angiosarcomas, and tumors of intermediate malignancy. Retiform hemangioendothelioma (RH) is one of the rare diagnoses belonging to the class of intermediate malignancies that was initially diagnosed. Case Presentation: In this case report, we have reported a rare case of RH, a locally aggressive, vascular tumor of malignant potential presenting at an unusual site. The most observed locations of this soft tissue tumor are skin and subcutaneous tissues of extremities; however, this case of RH was observed in an unusual site called pleura, which is a rare occurrence related to this tumor. The presence of cell surface markers like CD31 and ERG of vascular and endothelial origin and the presence of hobnail appearance of endothelial channels on the biopsy profile supported the vascular origin of the tumor; the clinical features and imaging studies further confirmed the diagnosis of RH. Conclusion: In this case report, a rare case of RH with an unusual site of origin was highlighted. RH is a vascular neoplasm; commonly observed sites of this vascular tumor were skin and subcutaneous tissue of the extremities; however, in this case, the site was unusually different, which was the pleura of the left lung.
Individualized Decision-Making and Outcomes for the 87-Year-Old Living Kidney Donor: A Case Report
Authors: Kigitovica Dana , Kuzema Viktorija , Jusinskis Janis , Mesecko Veronika , Suhorukovs Vadims , Petersons Aivars , Ziedina Ieva
Published: August 2024

Introduction: Latvia faces a challenging shortage of available kidney donors, leading to a significant mismatch between demand for kidney transplantation and supply. Although older adult donors require a thorough pre-donation workup to rule out significant medical comorbidities, it offers hope for potential kidney transplantation candidates. Case Presentation: This case study presents the unique scenario of an 87-year-old living kidney donor, where individualized decision-making resulted in outstanding outcomes for both the donor and recipient. Conclusions: The initial assessment for donation, which involves renal scintigraphy, serves as a preventive measure. In cases where one of the kidneys exhibits insufficient function, this approach avoids the necessity for further costly tests, thus preserving resources in the healthcare budget. The decision concerning an older donor should undergo thorough discussion by a multidisciplinary team to minimize perioperative and long-term risks. Nonetheless, a thoughtful approach to elderly donors offers a valuable opportunity to expand the living donor pool in the context of the organ shortage problem.
A Challenging Case of Reactive Angioendotheliomatosis
Authors: Kravvas Georgios , Meghoma Lola , Vilenchik Victoria , Oxley Jon , Keith Daniel J.
Published: August 2024

Introduction: Reactive angioendotheliomatosis (RAE) is a rare, benign, angioproliferative disorder with poorly understood aetiopathogenesis. It is characterised by vascular occlusion that occurs in patients with coexistent systemic or autoimmune disease. Case Presentation: A 60-year-old female presented with an 8-week history of a painful, non-healing, and non-traumatic ulcer on the left thigh. Her past medical history included smoking, peripheral vascular disease (PVD) and previously treated rectal squamous cell carcinoma. The diagnosis of pyoderma gangrenosum with superimposed cellulitis was considered and treatment with oral antibiotics was initiated. Following failure to improve, a biopsy was undertaken leading to the diagnosis of RAE. The patient was referred for urgent consideration of surgical correction of PVD, but was deemed unsuitable for surgical treatment due to a poor performance status. The patient was treated with conservative measures, but her condition rapidly deteriorated and she passed away a few weeks later. Conclusion: RAE is notorious for mimicking a wide spectrum of diseases. It is an important differential diagnosis to consider in patients with non-healing ulceration and underlying systemic or autoimmune disorders. Our case raises awareness of this rare condition and the mortality that it carries if left untreated. In an attempt to reverse disease progression and mortality, we urge clinicians to attempt surgical correction of PVD even when faced with multiple comorbidities and poor performance status.
Chemical Leukoderma or Vitiligo – Where Do We Draw the Line? A Case Report
Authors: AlAfeefi Mariam , Abdelhadi Shaden
Published: August 2024

Introduction: Vitiligo is a skin depigmenting disorder influenced by both genetic and environmental factors. In contrast, chemical leukoderma (CL) is an acquired form of depigmentation caused by repeated exposure to specific chemical compounds. There is growing evidence of a similar pathogenesis in both entities; however in some cases, it remains unclear. Case Presentation: We present a case of a 26-year-old female who developed widespread depigmentation after using “lightning creams” containing Monobenzyl Ether Hydroquinone purchased online. The lesions displayed various tones of skin color, including discrete macules and confluent patches with defined borders. Her history of chemical exposure and subsequent depigmentation strongly supports the diagnosis of CL. Conclusion: We aim to share this interesting case of widespread CL, review of pathogenesis and highlight the importance of awareness about commercially distributed products, particularly those sold online.
A Case Report of Immune Checkpoint-Related Hemophagocytic Lymphohistiocytosis and Review of the Literature
Authors: Herman Michael , Lee Andrea , Fawcett Sandra , Deodhare Sanjeev
Published: August 2024

Introduction: Hemophagocytic lymphohistiocytosis (HLH) secondary to immune checkpoint inhibitors (irHLH) is rare, and consequently optimal management strategies remain to be defined. There are sparse reports of the successful treatment of irHLH with steroids and tocilizumab. This case adds to the body of literature supporting this management strategy. Case Description: We present a case of a patient with thoracic malignancy who received dual checkpoint inhibitors and developed weakness, fever, confusion, and cytopenias. Further testing revealed an extremely elevated ferritin level. Cytokine levels as well as HLH-2004 criteria and HScore results were consistent with irHLH. Clinical parameters and symptoms promptly improved after the administration of corticosteroids and tocilizumab. Conclusion: This case highlights an important treatment strategy for an immune checkpoint inhibitor toxicity associated with a high mortality rate.
Pembrolizumab plus Pharmacologic Ascorbate in the Treatment of Leiomyosarcoma
Authors: Rieth John M. , Belzer Alex C. , Walhof Mackenzie L. , Milhem Mohammed M.
Published: August 2024

Introduction: Leiomyosarcoma (LMS) is a malignancy with smooth muscle differentiation. Metastatic LMS is associated with poor prognosis and limited efficacy of systemic treatment. Novel treatment modalities are desperately needed for this entity. Case Presentation: We report the first use of pembrolizumab plus pharmacologic ascorbate in 3 patients with metastatic LMS. All cases resulted in persistent objective responses and disease control significantly better than has been reported with chemotherapy or other immunotherapeutic approaches. Three patients with metastatic LMS, one each of uterine, vascular, and soft tissue origin, were treated with pembrolizumab plus pharmacologic ascorbate. The patient with uterine LMS received combination therapy at presentation and had persistent response for 12 months, which is ongoing. The patient with metastatic LMS of the inferior vena cava received combination therapy at presentation and had persistent response for 12 months, at which time new metastases were found. The patient with soft tissue LMS had disease progression on pembrolizumab monotherapy prior to the addition of ascorbate, after which she had a 17-month response, which is ongoing. No side effects attributed to treatment were reported. Conclusion: Pembrolizumab plus pharmacologic ascorbate is a novel immunotherapeutic approach and warrants further study in LMS.
Primary Vitreoretinal Lymphoma Presenting Solely with Asymptomatic Peripheral Drusenoid Lesions
Authors: Lu Brianna , Wong Jovi C.Y. , Kritzinger Justin , Wong David T.
Published: August 2024

Introduction: Primary vitreoretinal lymphoma (PVRL) is a rare malignant tumor that typically involves the retina, vitreous, or optic nerve head. PVRL often occurs concurrently with central nervous system lymphoma. Here, we present the first report of a patient with biopsy-confirmed PVRL presenting solely with asymptomatic peripheral drusenoid lesions. Case Presentation: A woman in her 70s presented with new elevated amelanotic yellow lesions with overlying pigment in both of her eyes not previously seen prior to cataract surgery. Over the next 4 months, there was waxing and waning of lesions which resolved and first appeared in the right eye and then the left. A diagnostic vitrectomy of the left eye revealed B-cell lymphoma. The patient elected for initial treatment with radiation therapy of both orbits. A new lesion was identified in her right eye nearly 18 months after starting maintenance therapy with ibrutinib, following which systemic chemotherapy with methotrexate was initiated. Conclusion: Elevated clinical suspicion for a malignant process is needed for patients with progressive new retinal lesions in older age. Local radiation therapy to the orbits alone may not be sufficient to prevent progression despite initial presentation showing confinement of disease to the intraocular space.
Health Benefits of Intermittent Fasting
Authors: Reddy B. Lakshmi , Reddy Vamsee S. , Saier Jr. Milton H.
Published: August 2024

Summary: We propose that intermittent fasting (time-restricted eating), in agreement with the conclusions of other biologists, as revealed in recent publications, promotes the achievement of numerous health benefits including the extension of human and animal lifespans. Background: There is evidence, obtained both with animal model systems and with humans, that intermittent fasting has health benefits. These benefits include extended longevity, weight loss, and counteracting various disease conditions. Such procedures positively influence the benefits of human tissue-specific microbiomes and minimize the consequences of organellar apoptosis. Key Messages: In this review, we attempt to summarize the predominant evidence, published in the scientific literature, relevant to the conclusions that in general, and in many specific instances, intermittent fasting has long-term benefits to animals, including humans, with respect to overall and specific organismal health and longevity.
Lung Adenocarcinoma Presenting as Early Cardiac Tamponade: A Case Report
Authors: Chapiolkina Volha , Saadati Homa , Guevara-Rodriguez Nehemias Antonio , Francis-Morel Garry
Published: August 2024

Introduction: Lung cancer remains the most common cause of cancer death in the USA and worldwide despite continued advances in lung cancer screening and treatment. Pericardial effusion (PerF) has been found in up to 50% of postmortem patients with cancer; lung and breast cancers are the most frequent malignancies. Furthermore, it is a sign of poor outcomes with fewer than 5 months of survival. Nevertheless, PErF with or without tamponade as a presentation of lung cancer is uncommon. Case Presentation: We present a 72-year-old male without medical history who presented with 1 month of cough with white sputum and shortness of breath, progressively worsening, associated with weight loss (20 pounds). Further studies demonstrated early cardiac tamponade secondary to malignancy. Conclusion: Cardiac tamponade can arise secondarily from various etiologies and have different presentations depending on the cause. In general, it is a slowly developing and clinically silent disease process. Therefore, malignant PerFs can rarely present with hemodynamic instability and be the initial manifestation of an underlying malignancy. Our case review presents a rare case of metastatic lung adenocarcinoma manifesting as early symptomatic cardiac tamponade and as an emergency. The results might be life-threatening if this presentation is not recognized and managed appropriately. Clinicians must be aware of such atypical presentations of thoracic malignancies to take action adequately.
Enhancing Visual Acuity through Combined Intraocular Lens Fixation, Penetrating Keratoplasty, and Vitrectomy: The Role of Temporary Landers Wide-Field Keratoprosthesis
Authors: Zusho Takahiro , Ono Takashi , Taketani Yukako , Kimakura Mikiko , Toyono Tetsuya , Sugimoto Koichiro , Toyama Taku , Ueta Takashi , Aihara Makoto , Miyai Takashi
Published: August 2024

Introduction: Complex corneal conditions present surgical challenges and necessitate innovation. Here, we present two cases where we performed intraocular lens trans-scleral fixation using the double-needle Yamane technique, followed by penetrating keratoplasty and vitrectomy using a temporary Landers wide-field keratoprosthesis. Case Presentation: Case 1 involved a 70-year-old man with an aphakic eye of bullous keratopathy and corneal opacity owing to multiple penetrating and endothelial keratoplasty, endophthalmitis, and herpetic keratitis. His visual acuity was counting fingers at 20 cm before surgery. Penetrating keratoplasty with vitrectomy and intraocular lens scleral fixation was performed using the double-needle Yamane technique, and 10 months postoperatively, his best-corrected visual acuity improved to 0.6, presenting a clear cornea. Case 2 involved a 62-year-old man who underwent penetrating keratoplasty twice for corneal perforation and therapeutic penetrating keratoplasty with vitrectomy for traumatic globe rupture, resulting in the loss of the intraocular lens. The patient exhibited graft failure, and his best-corrected visual acuity was 0.03. Utilizing a temporary Landers wide-field keratoprosthesis, we performed penetrating keratoplasty and intraocular lens trans-scleral fixation without complications. His final best-corrected visual acuity improved to 0.15 with a clear cornea. Conclusions: Trans-scleral fixation of intraocular lens with penetrating keratoplasty, using temporary Landers wide-field keratoprosthesis, yielded positive clinical outcomes without serious complications.
Unusual Synchronous Multiple Primary Early-Stage Lung Adenocarcinoma with Concomitant MET Exon 14 Skipping, PIK3CA and KRAS Mutations: A Case Report
Authors: Zhang Yongjing , Gao Qiqi
Published: August 2024

Instruction: Synchronous multiple primary lung cancer (sMPLC) constitutes a distinct subtype of NSCLC, where accurate diagnosis and prognostic evaluation remain challenging. Case Presentation: The case involves a 70-year-old male patient admitted to the hospital due to bilateral pulmonary nodules. The patient underwent staged resection. Molecular pathological examination revealed that tumor A harbored concurrent mutations in MET exon 14 skipping and PIK3CA (p.E545K), while tumor B exhibited a KRAS exon 2 (p.G12S/D) mutation. Postoperatively, the patient demonstrated a favorable recovery, with no evidence of recurrence for 1 year. Conclusion: This study presents a case of sMPLC in early-stage lung cancer, illustrating the genetic heterogeneity in early-stage lung adenocarcinoma and underscoring the significance of precise evaluation of sMPLC and intrapulmonary metastases.
Large Spontaneous Subcutaneous Abscess Formation due to Finegoldia magna in a Diabetic Patient: A Case Report
Authors: Sato Toshiyuki , Tomita Mayuka , Kohno Atsuhiro , Chujo Satomi , Waki Yuma , Nobeyama Yoshimasa , Kawase Masaaki , Asahina Akihiko
Published: August 2024

Introduction: Finegoldia magna is a member of the Gram-positive anaerobic cocci group and constitutes the flora of the skin and other parts of the body. It sometimes colonizes diabetic foot and rarely infects skin or soft tissue of non-immunocompromised patients. Case Presentation: Here, we report the case of a severe subcutaneous abscess on the back caused by F. magna involving an immunocompromised patient with poorly controlled diabetes. A 48-year-old woman with diabetes mellitus and anemia associated with uterine fibroids was referred to us with a 1-month history of a skin manifestation on her back, with a body temperature of 35.9°C and blood pressure of 115/73 mm Hg. The manifestation involved a subcutaneous mass of 36 × 45 cm with a foul odor, partly covered with necrotic tissue, which had the appearance of a tortoiseshell-like pattern. Blood examination revealed C-reactive protein of 21.4 mg/dL and hemoglobin A1c of 9.1%. Contrast-enhanced computed tomography showed a subcutaneous abscess with internal emphysema. Emergency debridement was performed, resulting in drainage of foul-smelling gray-green pus. F. magna was detected in the pus and skin tissue. Conclusion: Skin and soft tissue infectious disease caused by F. magna is extremely rare, but the disease tends to become severe once developing in an immunocompromised patient, such as a patient with poorly controlled diabetes. Therefore, physicians should consider F. magna as a causative agent when poorly controlled diabetic patients suffer from severe infectious cutaneous manifestations.
Neural Process Models Can Help Keep Theory at the Forefront of Developmental Neuroscience: Commentary on Marshall, Arsalidou, Rueda, Crone and van Drunen
Authors: Spencer John P. , Johns Eleanor
Published: August 2024

We discuss the four chapters in this special issue, focusing on three key questions: (1) what developmental theories are being used across chapters; (2) what neuroscience methods are being used; and (3) how are these integrated? We organize our discussion by chapter moving from the broadest conceptual chapter by Peter Marshall through to the integration of theory and methods by Crone and van Drunen. We end by discussing work that is missing from the special issue that uses neural process models to integrate developmental theory with cognitive neuroscience methods. We highlight how this approach can yield a tighter integration between theory and methods, enabling researchers to test specific hypotheses about the mechanisms that underlie change in development. We suggest that this approach might prove to be a particularly effective way to keep theory at the forefront of developmental neuroscience.