-
Infection and Drug Resistance 2024Nocardiosis is primarily an opportunistic infection affecting immunocompromised individuals, with a predilection for the lungs, brain, or skin in those with compromised...
BACKGROUND
Nocardiosis is primarily an opportunistic infection affecting immunocompromised individuals, with a predilection for the lungs, brain, or skin in those with compromised immune function. Granulomatous hepatitis caused by Nocardia is a rare clinical manifestation. This study aims to provide a systematic overview of the clinical features of Nocardiosis caused by Nocardia farcinica, enhancing our understanding of this disease.
METHODS
We report a case of a 75-year-old male with no underlying diseases presenting with a history of "recurrent fever for more than 4 months", along with fatigue, poor appetite, and pleural and abdominal effusion. Despite treatment at multiple hospitals, the patient showed little improvement. Chest CT revealed chronic inflammation, small nodules, bilateral pleural effusion, and pleural thickening. Abdominal CT indicated multiple low-density lesions in the liver, multiple small calcifications, and abdominal effusion.
RESULTS
Liver biopsy suggested inflammatory changes, with focal granuloma formation. Metagenomic next-generation sequencing (mNGS) of liver tissue indicated Nocardia farcinica, leading to the final diagnosis of disseminated Nocardia farcinica granulomatous hepatitis.
CONCLUSION
Nocardia infection is a rare disease primarily observed in immunocompromised patients but can also occur in those with normal immune function. The clinical and radiological features lack specificity; however, the utilization of mNGS technology enables rapid identification of the pathogenic microorganism. Nocardia farcinica is generally susceptible to sulfonamide drugs and amikacin, offering viable treatment options.
PubMed: 38645888
DOI: 10.2147/IDR.S461494 -
The Journal of Dermatological Treatment Dec 2024Granuloma formation is an uncommon and persistent skin inflammatory condition caused by the injection of dermal fillers. The exact cause of this reaction is not well... (Review)
Review
BACKGROUND
Granuloma formation is an uncommon and persistent skin inflammatory condition caused by the injection of dermal fillers. The exact cause of this reaction is not well understood, but it may be associated with irritating components or abnormal immune function. Treating granulomas can be difficult. However, recent research has shown that Janus kinase (JAK) inhibitors hold promise as a potential therapy for refractory granulomatous diseases.
OBJECTIVES
The aim was to evaluate the efficacy and safety of tofacitinib as a treatment for granulomas secondary to filler injection and the possible mechanisms were discussed and summarized.
METHODS
This study focuses on three cases of patients who experienced granuloma formation after receiving filler injections and were subsequently treated with tofacitinib. The efficacy and safety of the treatment were evaluated using parameters such as photographs and monitoring for any adverse reactions. In addition, a literature review was conducted to explore the underlying mechanisms and potential effects of tofacitinib.
RESULTS
All three cases recovered from swelling and nodules without side effects through the off-label use of oral tofacitinib. Existing data review reveals some approaches for cutaneous granulomatous disorders like inhibiting macrophage activation and downregulation of the JAK-STAT pathway.
CONCLUSION
This report emphasizes the effectiveness of JAK inhibitors in treating granulomas caused by filler injections. Recent advancements in understanding the underlying mechanisms of granulomatous reactions have paved the way for JAK inhibitors to be regarded as a promising treatment choice. However, further research is necessary to fully assess the safety and long-term effectiveness of using tofacitinib for granuloma treatment.
Topics: Humans; Dermal Fillers; Janus Kinase Inhibitors; Janus Kinases; Signal Transduction; STAT Transcription Factors; Granuloma; Skin Diseases; Piperidines; Pyrimidines
PubMed: 38632962
DOI: 10.1080/09546634.2024.2338281 -
EBioMedicine May 2024Prurigo nodularis (PN) presents with intensely itchy hard nodules. Despite being limited to the skin, PN was noted to be associated with systemic diseases including...
BACKGROUND
Prurigo nodularis (PN) presents with intensely itchy hard nodules. Despite being limited to the skin, PN was noted to be associated with systemic diseases including diabetes and chronic renal failure. In previous smaller retrospective studies, several cardiac and vascular diseases were found more frequently in patients with PN. However, small cohort sizes, partially discrepant outcomes, missing data, and incomplete risk assessment limit these findings.
METHODS
Electronic health records (EHR)s of 64,801 patients (59.44% females) with PN and an equal sized propensity-matched control group were retrieved. In these cohorts, the risks to develop cardiac and vascular diseases and mortality following the diagnosis of PN were determined. Sub-analyses included stratification for sex, ethnicity, and treatments.
FINDINGS
PN was associated with a higher risk for a broad range of acute cardiac events including heart failure and myocardial infarction. For example, the hazard ratio of myocardial infarction was 1.11 (95%-CI: 1.041-1.184, p = 0.0015) following PN diagnosis. Also, all-cause mortality was higher in patients with PN. Further, chronic vascular as well as structural heart diseases, e.g., peripheral arterial disease, chronic ischaemic heart disease and valval disorders were found more frequently following a PN diagnosis. Risks were more pronounced in white and female patients. Having established an increased risk for death and cardiovascular disease, we next addressed if dupilumab that has been recently licenced for use in this indication can modulate these risks. The risk of death but not of any cardiovascular disease was slightly reduced in patients with PN treated with dupilumab as opposed to those treated with systemic therapies other than dupilumab. The study is limited by retrospective data collection and reliance on ICD10-disease classification.
INTERPRETATION
PN is associated with higher mortality and an increased risk for the development of a wide range of cardiac and vascular diseases. Health care professionals should take this into account when managing patients with PN.
FUNDING
This work was supported by the University of Lübeck, the Deutsche Forschungsgemeinschaft and the State of Schleswig-Holstein.
Topics: Humans; Female; Male; Prurigo; Cardiovascular Diseases; Middle Aged; Aged; Adult; Cohort Studies; Risk Factors; Retrospective Studies
PubMed: 38631094
DOI: 10.1016/j.ebiom.2024.105123 -
Cureus Mar 2024Musculoskeletal coccidioidomycosis is a rare disseminated fungal infection caused by either or endemic to the southwestern United States and northwestern Mexico, as...
Musculoskeletal coccidioidomycosis is a rare disseminated fungal infection caused by either or endemic to the southwestern United States and northwestern Mexico, as well as Guatemala, Brazil, and other locations in Central and South America. Symptomatic primary infection of coccidioidomycosis can present as pneumonia with influenza-like symptoms, but the majority of cases remain asymptomatic. When dissemination occurs, the most common extrapulmonary sites include the skin, lymph nodes, musculoskeletal system, and meninges. We present a case of a 53-year-old female with a history of breast cancer and ankylosing spondylitis treated with adalimumab who presented with disseminated coccidioidomycosis. On presentation, she reported subcutaneous nodules on the right forearm and elbow. Radiologic evaluation utilizing magnetic resonance imaging (MRI) and positron emission tomography (PET) scan revealed multiple subcutaneous and bony enhancing lesions in her right forearm, lumbar spine, iliac wing, and axillary lymphadenopathy. Given the patient's history of breast cancer, there was concern for metastatic disease. Axillary lymph node biopsies were negative for malignancy, but immunoreactive for with a positive Grocott methenamine silver (GMS) stain and a antibody panel confirmed the diagnosis of disseminated coccidioidomycosis. Treatment with fluconazole was initiated along with discontinuation of adalimumab. Fluconazole was transitioned to itraconazole due to adverse effects. Treatment was successful as evidenced by improved PET imaging and downtrending antibody titers. This case highlights the concerning potential for dissemination of endemic mycoses with anti-tumor necrosis factor-α (TNF-α) therapies and the unique ways in which they can present. Further investigation is needed to determine the long-term implications of the disease and the role that immunosuppressive medications play in disease susceptibility.
PubMed: 38629004
DOI: 10.7759/cureus.56321 -
Frontiers in Veterinary Science 2024Lumpy skin disease (LSD) is a transboundary viral infection, affecting cattle with characteristic manifestations involving multiple body systems. A distinctive...
Lumpy skin disease (LSD) is a transboundary viral infection, affecting cattle with characteristic manifestations involving multiple body systems. A distinctive characteristic of lumpy skin disease is the subclinical disease manifestation wherein animals have viremia and shed the virus through nasal and ocular discharges, while exhibiting no nodules but enlarged lymph nodes that are easily oversighted by inexperienced vets. Further research on the role of subclinically ill animals in the transmission of LSD virus (LSDV) can contribute to the development of more effective tools to control the disease worldwide. Thus, this study aims to determine the potential role of subclinical infection in virus transmission in a non-vector-borne manner. To achieve this, we inoculated animals with the recombinant vaccine-like strain (RVLS) Udmurtiya/2019 to cause clinical and subclinical LSDV infection. After the disease manifestation, we relocated the subclinically ill animals to a new clean facility followed by the introduction of another five animals to determine the role of RVLS-induced subclinical infection in the virus transmission via direct/indirect contact. After the introduction of the naïve animals to the relocated subclinically ill ones in a shared airspace, two introduced animals contracted the virus (clinically and subclinically), showing symptoms of fever, viremia, and seroconversion in one animal, while three other introduced animals remained healthy and PCR-negative until the end of the study. In general, the findings of this study suggest the importance of considering LSDV subclinical infection as a high-risk condition in disease management and outbreak investigations.
PubMed: 38628945
DOI: 10.3389/fvets.2024.1330657 -
Frontiers in Neurology 2024Capillary hemangiomas, usually found in skin and mucosal tissues, are rarely encountered within the spinal cord, presenting a significant diagnostic challenge. We report...
Capillary hemangiomas, usually found in skin and mucosal tissues, are rarely encountered within the spinal cord, presenting a significant diagnostic challenge. We report a rare case of intradural extramedullary capillary hemangioma at the conus medullaris in a 66-year-old female patient. Our initial diagnosis leaned towards a cystic hemangioblastoma based on MRI findings due to the presence of cystic formation with an enhanced mural nodule. However, surgical exploration and subsequent pathological examination revealed the lesion as a capillary hemangioma. To the authors' knowledge, this case may represent the first documented instance of a spinal capillary hemangioma that mimics a cystic hemangioblastoma.
PubMed: 38606279
DOI: 10.3389/fneur.2024.1350780 -
Cureus Mar 2024Here, we report the case of recurrent swelling and pain in the proximal interphalangeal joint of the left ring finger, which was later diagnosed as a localized...
Here, we report the case of recurrent swelling and pain in the proximal interphalangeal joint of the left ring finger, which was later diagnosed as a localized tenosynovial giant cell tumor in a young adult female. The first presentation was at the same anatomical site four years prior. Examination at presentation showed a firm skin-colored nodule in the volar aspect of the left ring finger. The swelling was seen to be partly attached to underlying structures and was non-tender. After a careful physical examination and plain radiograph imaging of the hand, the two differential diagnoses considered were tenosynovial giant cell tumor and ganglion cyst. A surgical excision was performed, and histopathologic evaluation showed features consistent with a tenosynovial giant cell tumor, localized type. The resection margins were clear of tumor. The patient had no intraoperative or postoperative complications. Postoperative physiotherapy was recommended. No recurrence was seen after postoperative surgical follow-up for one year. This report highlights the importance of histopathologic evaluation and confirmation of clear surgical margins in the management of tenosynovial giant cell tumors. In recurrent cases, surgical re-excision with clear margins provides good clinical outcomes. Before surgical excision, patients should be informed about the biologic nature of the lesion and the high risk of recurrence. The management modalities to prevent recurrence and the need for long-term follow-up should also be discussed with the patient.
PubMed: 38601375
DOI: 10.7759/cureus.55962 -
Neurobiology of Pain (Cambridge, Mass.) 2024Spinal cord injury (SCI) affects roughly 300,000 Americans with 17,000 new cases added annually. In addition to paralysis, 60% of people with SCI develop neurogenic...
BACKGROUND AND AIMS
Spinal cord injury (SCI) affects roughly 300,000 Americans with 17,000 new cases added annually. In addition to paralysis, 60% of people with SCI develop neurogenic bowel (NB), a syndrome characterized by slow colonic transit, constipation, and chronic abdominal pain. The knowledge gap surrounding NB mechanisms after SCI means that interventions are primarily symptom-focused and largely ineffective. The goal of the present studies was to identify mechanism(s) that initiate and maintain NB after SCI as a critical first step in the development of evidence-based, novel therapeutic treatment options.
METHODS
Following spinal contusion injury at T9, we observed alterations in bowel structure and function reflecting key clinical features of NB. We then leveraged tissue-specific whole transcriptome analyses (RNAseq) and fecal 16S rRNA amplicon sequencing in combination with histological, molecular, and functional (Ca imaging) approaches to identify potential mechanism(s) underlying the generation of the NB phenotype.
RESULTS
In agreement with prior reports focused on SCI-induced changes in the skin, we observed a rapid and persistent increase in expression of calcitonin gene-related peptide (CGRP) expression in the colon. This is suggestive of a neurogenic inflammation-like process engaged by antidromic activity of below-level primary afferents following SCI. CGRP has been shown to disrupt colon homeostasis and negatively affect peristalsis and colon function. As predicted, contusion SCI resulted in increased colonic transit time, expansion of lymphatic nodules, colonic structural and genomic damage, and disruption of the inner, sterile intestinal mucus layer corresponding to increased CGRP expression in the colon. Gut microbiome colonization significantly shifted over 28 days leading to the increase in a pathogenic, gram-negative microbe. Moreover, colon specific vagal afferents and enteric neurons were hyperresponsive after SCI to different agonists including fecal supernatants.
CONCLUSIONS
Our data suggest that SCI results in overexpression of colonic CGRP which could alter colon structure and function. Neurogenic inflammatory-like processes and gut microbiome dysbiosis can also sensitize vagal afferents, providing a mechanism for visceral pain despite the loss of normal sensation post-SCI. These data may shed light on novel therapeutic interventions targeting this process to prevent NB development in patients.
PubMed: 38601267
DOI: 10.1016/j.ynpai.2024.100156 -
Frontiers in Genetics 2024Neurofibromatosis type 1 (NF1) is an autosomal dominant genetic condition with complete age-dependent penetrance, variable expressivity and a global prevalence of...
Neurofibromatosis type 1 (NF1) is an autosomal dominant genetic condition with complete age-dependent penetrance, variable expressivity and a global prevalence of ∼1/3,000. It is characteriszed by numerous café-au-lait macules, skin freckling in the inguinal or axillary regions, Lisch nodules of the iris, optic gliomas, neurofibromas, and tumour predisposition. The diagnostic testing strategy for NF1 includes testing for DNA single nucleotide variants (SNVs), copy number variants (CNVs) as well as RNA analysis for deep intronic and splice variants, which can cumulatively identify the causative variant in 95% of patients. In the present study, NF1 patients were screened using a next-generation sequencing (NGS) assay targeting exons and intron/exon boundaries for SNV and multiple ligation-dependent probe amplification (MLPA) analysis for CNV detection. Twenty-six unrelated Southern African patients clinically suspected of having NF1, based on the clinical diagnostic criteria developed by the National Institute of Health (NIH), were included in the current study. A detection rate of 58% (15/26) was obtained, with SNVs identified in 80% (12/15) using a targeted gene panel and gene deletion in 20% (3/15) identified using MLPA. Ten patients (38%) had no variants identified, although they met NF1 diagnostic criteria. One VUS was identified in this study in a patient that met NF1 diagnostic criteria, however there was no sufficient information to classify variant as pathogenic. The clinical features of Southern African patients with NF1 are similar to that of the known NF1 phenotype, with the exception of a lower frequency of plexiform neurofibromas and a higher frequency of developmental/intellectual disability compared to other cohorts. This is the first clinical and molecular characterisation of a Southern African ancestry NF1 cohort using both next-generation sequencing and MLPA analysis. A significant number of patients remained without a diagnosis following DNA-level testing. The current study offers a potential molecular testing strategy for our low resource environment that could benefit a significant proportion of patients who previously only received a clinical diagnosis without molecular confirmation.
PubMed: 38596211
DOI: 10.3389/fgene.2024.1331278 -
Cureus Mar 2024Squamous cell carcinoma (SCC) is the second leading form of skin cancer. In the elderly population, surgery may carry more risk and significant morbidity in comparison...
Squamous cell carcinoma (SCC) is the second leading form of skin cancer. In the elderly population, surgery may carry more risk and significant morbidity in comparison to less invasive forms of treatment. This case report describes the successful use of intralesional 5-fluorouracil (IL5-FU) to treat cutaneous squamous cell carcinoma (cSCC). A 98-year-old white woman presented in early May 2017 with a 3.5-cm rapidly growing crusted nodule on her left proximal-lateral arm. She had a past medical history of chronic obstructive pulmonary disease, atrial fibrillation, and heart failure. The patient also had a frail body habitus and weighed 80 pounds. Physical examination revealed a large, ulcerated, crateriform mass on the left proximal-lateral arm. A shave biopsy was performed, which revealed a well-differentiated SCC, composed of nodular masses of neoplastic squamous cells with atypical nuclei, keratin pearl formation, and scattered mitotic figures with surrounding fibrosis and inflammation. The patient was wheelchair-bound and oxygen-dependent and, thus, not considered a good surgical or radiation candidate. Treatment was decided with 5-fluorouracil. At a four-week follow-up appointment, there was no visible or palpable evidence of the tumor. There was no sign of recurrence at three months, indicating treatment success. The patient later died due to cardiac arrest in September 2017. The elderly population with cSCC can benefit from intervention and treatment with IL5-FU when surgery is not an option due to patient comorbidities. IL5-FU can potentially be used in areas where access to a dermatologist, surgeon, or surgical services is limited.
PubMed: 38590475
DOI: 10.7759/cureus.55855