Dermatological Symptoms Research Articles

Chikungunya infection in returned travellers: results from the geosentinel network, 2005-2020.

Chikungunya is an important travel-related disease because of its rapid geographical expansion and potential for prolonged morbidity. Improved understanding of the epidemiology of travel-related chikungunya infections may influence prevention strategies including education and vaccination. We analysed data from travellers with confirmed or probable chikungunya reported to GeoSentinel sites from 2005 to 2020. Confirmed chikungunya was defined as a compatible clinical history plus either virus isolation, positive nucleic acid test or seroconversion/rising titre in paired sera. Probable chikungunya was defined as a compatible clinical history with a single positive serology result. 1202 travellers (896 confirmed and 306 probable) with chikungunya were included. The median age was 43years (range 0-91; interquartile range [IQR]: 31-55); 707 (58.8%) travellers were female. Most infections were acquired in the Caribbean (28.8%), Southeast Asia (22.8%), South Central Asia (14.2%) and South America (14.2%). The highest numbers of chikungunya cases reported to GeoSentinel were in 2014 (28.3%), 2015 (14.3%) and 2019 (11.9%). The most frequent reasons for travel were tourism (n = 592; 49.3%) and visiting friends or relatives (n = 334; 27.7%). The median time to presentation to a GeoSentinel site was 23days (IQR: 7-52) after symptom onset. In travellers with confirmed chikungunya and no other reported illnesses, the most frequently reported symptoms included musculoskeletal symptoms (98.8%), fever/chills/sweats (68.7%) and dermatologic symptoms (35.5%). Among 917 travellers with information available, 296 (32.3%) had a pretravel consultation. Chikungunya was acquired by international travellers in almost 100 destinations globally. Vector precautions and vaccination where recommended should be integrated into pretravel visits for travellers going to areas with chikungunya or areas with the potential for transmission. Continued surveillance of travel-related chikungunya may help public health officials and clinicians limit the transmission of this potentially debilitating disease by defining regions where protective measures (e.g. pretravel vaccination) should be strongly considered.

Orthopedic Dermatopathies: Skin Manifestations in Orthopedic Conditions.

Orthopedic diseases often present with dermatological symptoms that require prompt identification for appropriate treatment. Understanding these dermatologic presentations is crucial for accurate diagnosis and effective management. This article critically reviewed the dermatological manifestations observed in general and regional pathologies, followed by treatment-related manifestations. An extensive literature search was performed and limited to manifestations in orthopedic disease, excluding those pertaining to infection or syndromes. Case reports and case series documenting unusual and rare dermatologic presentations of orthopedic conditions were examined, providing novel perspectives on both prevalent and uncommon illnesses. The identified pathologies are discussed in detail, including their clinical features and diagnosis, while treatment approach varies depending on the severity of the condition, ranging from self-care to surgical intervention. The findings emphasize the need for interdisciplinary collaboration and highlight the importance of careful diagnosis and appropriate management to eliminate unnecessary approaches and ensure optimal outcomes for patients with orthopedic diseases and dermatologic symptoms.

Mpox gastrointestinal manifestations: a systematic review

IntroductionMpox is a viral infection caused by the monkeypox virus, a member of the Poxviridae family and Orthopoxvirus genus. Other well-known viruses of the Orthopoxvirus genus include the variola virus...

Nail Patella Elbow Syndrome Presenting with Depression: A Case Report

Nail-Patella Syndrome (NPS) is a rare autosomal dominant illness. Patients typically exhibit a tetrad of dysplastic finger or toenails, elbow dysplasia, hypoplastic or missing patellae, and iliac horns as their first musculoskeletal and dermatological symptoms. A higher occurrence of psychiatric disorders, such as depression and attention deficit hyperactivity disorder, has also been reported with NPS. This is a rare case of NPS, presenting with depression as the major symptom in an 18-year-old young girl who presented to the psychiatry Outpatient Department (OPD) with depressive symptoms like persistent low mood, loss of pleasure or anhedonia, loss of appetite, disturbed sleep, and thoughts of death. After 10 days of treatment with antidepressant drugs like Benzodiazepines, she was discharged and followed-up on an OPD basis. After a thorough evaluation, hypoplastic patella, swan-necked nails, radial head subluxation, and right eye glaucoma were also noted, and a final diagnosis of Nail-Patella Syndrome was made. A case of NPS can present in any department, such as in Medicine for nail dysplasia, in Orthopaedics for elbow or patella dysplasia, or in Dermatology for iliac horns. Here, the case was primarily presented to the Psychiatry department and later managed by a team of physicians, orthopaedicians, and psychiatrists. Careful illustration of the family history reveals more cases.

Paraneoplastic syndromes in patients with melanoma.

Paraneoplastic syndromes are a group of rare clinical conditions characterized by a diverse array of systemic manifestations that arise in association with malignant tumours, often due to the production of bioactive substances by the tumour or an autoimmune response to the tumour. Melanoma, a malignant skin neoplasm originating from melanocytes, has been associated with various paraneoplastic syndromes. This paper provides an overview of the key paraneoplastic syndromes observed in patients with melanoma. Paraneoplastic syndromes in melanoma can manifest with neurological, dermatological, endocrine, haematological, and rheumatological symptoms, among others. Melanoma-associated retinopathy was the most reported paraneoplastic syndrome; this entity is characterized by a spectrum of retinal abnormalities. Paraneoplastic neurological syndromes, such as paraneoplastic encephalitis and paraneoplastic cerebellar degeneration, are among the most frequently reported. The pathophysiology of paraneoplastic syndromes often involves the production of autoantibodies against neuronal or tumour antigens, immune-mediated reactions, or the release of cytokines and growth factors from the tumour. Management strategies for paraneoplastic syndromes associated with melanoma primarily focus on treating the underlying malignancy, which may lead to resolution or improvement of the paraneoplastic manifestations. Immune-modulating therapies, including corticosteroids, intravenous immunoglobulins, and plasmapheresis, may be considered in selected cases to ameliorate symptoms and suppress the autoimmune response. In conclusion, paraneoplastic syndromes in patients with melanoma are a complex and diverse group of clinical entities with a broad range of presentations. Further research is needed to enhance our understanding of the mechanisms and therapeutic options for paraneoplastic syndromes associated with melanoma.

Pseudo-chilblain lesions in children during the COVID-19 pandemic

Introduction and objective: During the COVID-19 pandemic, a range of uncommon cutaneous symptoms were observed, which were linked to the SARS-CoV-2 infection. These dermatological symptoms in the form of reddish or purple-coloured lesions on the fingers and toes, accompanied by blisters and ulcerations, were termed as “COVID fingers/toes” – a pseudo-chilblain cutaneous manifestation of COVID-19. The aetiopathogenesis of pseudo-chilblain lesions has not been conclusively determined. At first, SARS-CoV-2 infection was regarded as an evident cause of chilblains. Various studies conducted over a two-year period have not managed to plausibly confirm its direct implications. Materials and methods: The study was of a retrospective nature. The analysis involved clinical symptoms, results of selected laboratory tests, as well as capillaroscopy of the microcirculation in 15 children diagnosed with skin lesions of chilblain-like morphology. Results: All patients from the study population presented with lesions in distal body parts which were of erythematous or oedematous morphology, and resembled chilblains. None of the hospitalised patients suffered from fever or presented with other systemic symptoms. The reverse transcription polymerase chain reaction test for SARS-CoV-2 was negative in all the patients. SARS-CoV-2 antibodies were found in 5 out of 9 patients tested for their presence. Microcirculation abnormalities were diagnosed in 80% of the patients. In none of the children did capillaroscopy image correspond with microangiopathy characteristic of inflammatory connective tissue diseases. Frequent abnormalities found in more than two fingers/toes were: pericapillary oedema (57%), dilated capillaries (42.8%), venous stasis (35.7%), presence of branched and tortuous capillaries (28.5%). Conclusions: Pseudo-chilblain lesions observed during the SARS-CoV-2 pandemic are probably secondary to multifactorial epiphenomenon.

VEXAS-Syndrome, a newly described autoinflammatory systemic disease with dermatologic manifestations.

VEXAS syndrome is a recently identified autoinflammatory systemic disease caused by an acquired somatic mutation of the X-linked UBA1 gene, the key enzyme of the first step of ubiquitylation. The acronym VEXAS stands for the characteristics Vacuoles, E1 enzyme, X-linked, autoinflammatory and somatic. The disease occurs in advanced adulthood preferentially in men and is characterized by hematological, rheumatological and dermatological symptoms. The latter include neutrophil-rich lesions reminiscent of Sweet's syndrome, erythema nodosum- and panniculitis-like skin manifestations and recurrent polychondritis of the nose and auricles. The presence of cytoplasmic vacuoles in myeloid and erythroid precursors in the bone marrow is characteristic. In up to half of the cases, VEXAS syndrome is associated with myelodysplastic syndrome. Dermatologists should be familiar with the clinical picture, as skin symptoms are often the first indicator of the disease. Molecular diagnostics are essential for confirming the diagnosis and risk stratification of affected patients. In this minireview we provide an overview of the pathophysiology, diagnosis and therapy of VEXAS syndrome and illustrate its clinical picture with two own cases.

Long COVID and Physical Therapy: A Systematic Review.

Prolonged COVID is a persistent condition following the initial COVID-19 infection, which is characterized by a variety of symptoms that may include fatigue, muscle pain, sleep disturbances, "brain fog", respiratory, cardiovascular, digestive, neurological and dermatological symptoms. Physical therapy has been identified as a crucial aspect of the management of patients with long COVID, as it can help improve symptoms and overall physical function. The investigation of long COVID poses significant challenges due to the diversity and variability of symptoms, lack of clear diagnostic criteria, and limited understanding of the underlying mechanisms. The aim of this study is to conduct a systematic review of studies conducted in patients with long COVID in conjunction with interventions targeting respiratory function, particularly involving physical activity. To this end, we conducted a systematic review to analyze studies conducted on treatment programs for long COVID based on some form of physical activity. The protocol of the review was registered in the PROSPERO website, and the databases PubMed, Scopus, CINAHL and WOS were searched. Of the 62 initial articles, six were included in the review. The results obtained have positive implications for the advancement of physical activity as a therapeutic intervention for individuals with long COVID-19 and the conceptualization of evidence-based treatment protocols. Statistically significant results have been observed in studies of at least 6 weeks duration, in which inspiratory muscle training exercises are proposed. Further research is needed to better understand long COVID and develop effective treatment strategies.

A statewide population-based approach to examining Long COVID symptom prevalence and predictors in Michigan

ObjectiveThe current broad definition of Long COVID, and an overreliance on clinical and convenience samples, is leading to a wide array of Long COVID estimates with limited generalizability. Our objective was to examine Long COVID symptoms using a statewide population-based probability sample. MethodsAmong 8000 sampled adults with polymerase-chain-reaction-confirmed SARS-CoV-2 between June 2020 and July 2021 in the Michigan Disease Surveillance System, 2533 completed our survey (response rate 32.2%). Using modified Poisson regression, we examined sociodemographic, behavioral, and clinical predictors of eight Long COVID symptom clusters, defined as at least one applicable symptom lasting 90 or more days post COVID-19 onset. ResultsNeuropsychiatric Long COVID symptoms, including brain fog, were most prevalent (23.7%), followed by systemic symptoms (17.1%), including fatigue, musculoskeletal (11.4%), pulmonary (10.4%), dermatologic (6.7%), cardiovascular (6.1%), gastrointestinal (5.4%), and ear, nose, and throat symptoms (5.3%). In adjusted analyses, female sex, a pre-existing psychological condition, and intensive care unit admission were strong predictors of most Long COVID symptom clusters. Older age was not associated with a higher prevalence of all symptoms – cardiovascular and dermatologic symptoms were most prevalent among middle-aged adults and age was not associated with neuropsychiatric or gastrointestinal symptoms. Additionally, there were fewer associations between pre-existing conditions and cardiovascular, neuropsychiatric, and dermatologic symptoms compared to other symptom clusters. ConclusionsWhile many predictors of Long COVID symptom clusters were similar, the relationship with age and pre-existing conditions varied across clusters. Cardiovascular, neuropsychiatric, and dermatologic symptoms require further study as potentially distinct from other Long COVID symptoms.

An Unaccustomed Presentation of Besnier-Boeck-Schaumann Disease

Eliminating other potential causes of granulomatous inflammation helps to diagnose the mysterious sarcoidosis. All clinicians face a diagnostic dilemma because of the clinical polymorphism and the multisystemic sarcoidosis symptoms. After the lungs, the skin is the organ most frequently impacted by sarcoidosis. Before, during, or after systemic involvement, dermatological symptoms can occur, and the kind of skin lesion can have a bearing on the prognosis. Additionally, compared to a biopsy of visceral organs, a skin lesion biopsy is easier to perform and less intrusive. Therefore, there are some instances in which cutaneous symptoms can help with the diagnosis and outlook of a systemic illness. A high index of clinical suspicion is needed for early diagnosis and timely management, which can improve the quality of life for those patients and reduce morbidy and mortality due to this disease. This article has emphasised the prevalence, clinical characteristics, and treatment of the common skin lesions of sarcoidosis.

Effect of Eriobotrya japonica Leaf Supplements on Allergic Rhinitis Symptoms and Skin Conditions in Healthy Adults: A Randomized, Double-Blind, Placebo-Controlled Study

Eriobotrya japonica (E. japonica) leaves have been used as an herbal traditional medicine in China and Japan owing to their anti-inflammatory and protective effects against skin conditions and allergy symptoms. These beneficial effects are likely mediated by the various triterpenoids present in E. japonica leaves. However, the efficacy of E. japonica leaves in the treatment of allergic rhinitis has not been evaluated in humans. Therefore, in the present study, a randomized, controlled, double-blind trial was performed on healthy adults of age >20 (n = 27) who were randomly assigned to receive either 2.5 g of placebo or E. japonica leaf supplements once daily for 4 weeks. The Japanese Allergic Rhinitis Quality of Life Standard Questionnaire (JRQLQ), dermatological allergy symptoms, Dermatology Life Quality Index, and skin condition parameters were assessed at baseline and after 4 weeks. Significant differences were observed in the variability of the itchy nose, itchy eyes, and eye symptoms between the E. japonica supplementation and placebo groups after 4 weeks. Arm skin transepidermal water loss was improved only in the E. japonica supplementation group. This study suggests that E. japonica leaves can be used as a functional food ingredient to relieve allergic symptoms.

Fatal eosinophilic myocarditis and submassive hepatic necrosis in lamotrigine induced DRESS syndrome

Drug Reaction with Eosinophilia and Systemic Symptoms (DRESS) syndrome is a rare but severe and sometimes fatal adverse drug reaction that is known to occur with a number of antiepileptic drugs. It often follows a prolonged clinical course, which can worsen even after discontinuing the causative drug and administering steroid treatment. Failure to promptly identify the delayed involvement of vital organs, such as the heart and liver, may result in irreversible organ failure and death. We report a case of a presumed sudden death of a young woman who had a documented history of a protracted intermittent hypersensitivity reaction to lamotrigine. Postmortem examination revealed the presence of eosinophilic myocarditis and submassive hepatic necrosis diagnostic of fatal DRESS syndrome that progressed despite early discontinuation of the medication and improvement of dermatologic and hematologic symptoms following steroid therapy.

A Prospective Open-Label Study of the Antifungal Activity of External Forms of Activated Zinc Pyrithione in the Treatment of Malassezia- Associated Skin Diseases

Background: There are insufficient data on the antifungal activity of active zinc pyrithione, which is widely used in practice. Considering the reported role of Malassezia spp. in the pathogenesis of several dermatologic diseases, it is of scientific and practical importance to investigate this issue. Aim: To evaluate the antifungal activity of external forms of activated zinc pyrithione in the treatment of psoriasis, seborrheic dermatitis, and pityriasis versicolor. Method: An open-label prospective study was conducted between March and July 2022. Patients with psoriasis, seborrheic dermatitis, and pityriasis versicolor were treated with external forms of activated zinc pyrithione for 21 days. Skin scales and circular prints from lesion foci, as well as from skin areas without clinical manifestations before and after therapy were studied. A quantitative assessment of skin colonization by micromycetes of Malassezia was performed using microscopic and cultural methods of examination. Clinical efficacy and drug safety of the therapy was assessed using the Dermatological Symptom Scale Index, by recording adverse events at weeks 0, 1, 2, and 3. Results: 64 patients aged 18 to 65 years with diagnoses of psoriasis, seborrheic dermatitis, and pityriasis versicolor were included. 60 patients completed the study, 4 were excluded due to failure to adhere to the schedule. In patients with seborrheic dermatitis and pityriasis versicolor in the lesion foci after therapy, a significant decrease was observed in the colonization level according to the results of microscopic and cultural studies. In psoriasis patients, a significant decrease in the colonization level was obtained only based on the results of microscopic examination. In all groups, significant differences in comparison to the initial level were observed at the 1st week of treatment. There was no adverse events observed. Conclusion: Activated zinc pyrithione in the form of cream and aerosol showed moderate antifungal activity against micromycetes of the genus Malassezia.

Revealing the therapeutic potential of dupilumab: a case report on managing post-COVID-19 chronic pruritus in a geriatric patient

This case report describes a 73-year-old Caucasian male who contracted COVID-19 and struggled with chronic, severe pruritus after that. The patient's pruritus resisted numerous conventional and unconventional therapy modalities, highlighting the difficulty of treating post-COVID-19 dermatological symptoms. Pruritus in the patient was significantly alleviated due to therapy with Dupixent, a monoclonal antibody that inhibits interleukin-4 and interleukin-13 signaling. Treatment of the condition improved significantly with the use of Dupixent. The report describes the story of the patient's journey, from the first-time symptoms showed up after COVID-19 infection, through several treatments that was not effective, to when the patient's symptoms disappeared after starting Dupixent. The patient's recent SARS-CoV-2 infection may be connected to the immune-mediated process shown by the patient's histopathological results. Even though these results are based on a single instance, they highlight COVID-19's ability to cause chronic pruritus and recommend Dupixent as a possible treatment approach. More study is required to confirm these findings and investigate possible immunological connections between COVID-19 and chronic pruritus.

Efficacy of an extensively hydrolyzed formula with the addition of synbiotics in infants with cow's milk protein allergy: a real-world evidence study.

Cow's milk protein allergy (CMPA) is the most frequent food allergy in early childhood. For those infants requiring breastmilk substitutes, formulas with extensively hydrolyzed proteins (EHF), should be the treatment of choice. As there are limited data showing the progression of initial symptoms in infants newly diagnosed with CMPA who are treated with EHF with added synbiotics, the main objective of this study was to evaluate the resolution of symptoms in said infants after 4 weeks of treatment. As a secondary objective this study aimed to assess the impact of the treatment on the family's quality of life. observational, longitudinal, prospective, and multicentric real-world evidence study. The intervention phase (EHF with synbiotics) lasted 28 days and was completed by 65 patients. Treating physicians registered child´s anthropometry, Infant Gastrointestinal Symptoms Questionnaire (IGSQ-13) and CoMiSS (Cow´s Milk Allergy Symptoms Score) both at baseline and after 28 days of treatment. During treatment, caregivers reported child´s regurgitation and stools, PO-SCORAD (Patient Oriented Scoring of Atopic Dermatitis) and FAQL-PB (Family Quality of Life-Parental Burden). Data were collected using Google Forms and analyzed through the STATA program. 95.4% of the patients showed an improvement or disappearance of the overall initial symptoms after 4 weeks of treatment. Gastrointestinal symptoms improved or disappeared in 92% of patients (p < 0.05) while dermatological symptoms improved or disappeared in 87.5% of patients (p < 0.05). The median CoMiSS at baseline was 9, with 21 patients exceeding the cut-off point of 12. After 4 weeks of treatment, the median dropped to 3, and no patient exceeded the 12-cut-off point (p = 0.000). At baseline, patients had a PO-SCORAD of 11.5 (interquartile range 1-23) that went to 1.0 (interquartile range 1-6) at day 28 (p = 0.000). The treatment diminished stool frequency (p < 0.05), improved stool consistency (p = 0.004) and decreased the frequency of regurgitation in infants with CMPA (p = 0.01). The percentage of patients who no longer had any episode of regurgitation increased from 11% to 31% on day 28 (p = 0.003). At baseline, 13% of patients cried more than 3 h per day, while at day 28 that percentage dropped to 3% (p = 0.03). An improvement in the infants' sleep pattern was also appreciated with the treatment. At study onset, 56% of the families reported feeling very overwhelmed, a percentage that dropped to 17% after 28 days of treatment (p < 0.05). The small percentage of families who did not feel overwhelmed at study onset (17%), grew to 43% on day 28 (p < 0.05). The use of an EHF with synbiotics for the management of infants diagnosed with or suspected to have CMPA suggested a good safety profile, an adequate infant growth, and improvement of overall, gastrointestinal, and dermatological symptoms. It also suggested a lower daily frequency of regurgitations and stools, and an improvement in stool consistency, sleeping pattern, and quality of life of the infant and his family.

Efficacy of intravenous immunoglobulins (IVIg) in improving skin symptoms in patients with dermatomyositis: a post-hoc analysis of the ProDERM study

Dermatomyositis (DM) is a rare autoimmune disease characterized by skin involvement, with or without proximal muscle weakness. Recently, following the ProDERM study, intravenous immunoglobulin (IVIg) was approved for treatment of DM. Until ProDERM evidence from large, placebo-controlled studies supporting its use for dermatological symptoms, was lacking. Here we present efficacy data from ProDERM of IVIg versus placebo for treatment of the cutaneous aspect of DM. ProDERM was a double-blind, randomized, multicenter, Phase 3 study. In the First Period (Weeks 0-16), adults with active DM received 2.0g/kg IVIg (Octagam 10%; Octapharma AG) or placebo every 4 weeks. In the open-label Extension Period (Weeks 16-40), all patients received IVIg for 6 additional cycles. Cutaneous disease was assessed using measures including modified cutaneous DM disease area and severity index activity (CDASI-A) and damage (CDASI-D) scores, and myositis disease activity assessment tool (MDAAT) including visual analogue scale (VAS). This trial is registered with ClinicalTrials.gov, NCT02728752. The study took place from February 2017 to November 2019. 95 patients received IVIg (N=47) or placebo (N=48) in the First Period. Together, 664 IVIg infusion cycles were administered (median dose, 2.0g/kg). At Week 16, mean CDASI-A change from baseline was-9.36 (95% CI:-12.52,-6.19) in the IVIg group versus-1.16 (-3.32, 0.99) in placebo group (p<0.0001). At the end of the Extension Period, mean changes from baseline were-10.44 (95% CI:-13.94,-6.94) and-10.03 (-13.12,-6.94), respectively. Similar changes were seen for CDASI-D and VAS of MDAAT. These observations were seen regardless of baseline disease severity. ProDERM is the first large prospective, randomized trial to demonstrate the efficacy of IVIg to improve the cutaneous manifestations of DM. IVIg treatment significantly improved dermatological symptoms in patients with DM, regardless of disease severity before treatment, suggesting that IVIg is effective for even the most severe cutaneous DM. This study was sponsored by Octapharma Pharmazeutika Produktionsges m.b.H.

Diagnosis, Treatment and Prevention of Pediatric Lyme disease

Lyme disease remains a significant public health challenge in the United States, particularly in the Northeast and Midwest regions, and is likely to increase in the coming years with population growth to suburban areas and warmer weather. Lyme disease is a bacterial infection caused by the spirochete, Borrelia burgdorferi, that is transmitted to humans through deer ticks. The presentation of Lyme disease can involve multiple systems with dermatologic, neurologic, rheumatologic, and cardiac symptoms commonly described. However, there is substantial variation in the presentation of Lyme disease, creating a challenge to diagnosis and treatment. The standard treatment for Lyme disease is administration of antibacterial agents. Although the majority of pediatric patients achieve symptom resolution with treatment, a subset continues to experience fatigue, pain, and psychological disturbances known as post-treatment Lyme disease syndrome. Prevention of Lyme disease largely relies on behavioral measures such as wearing long sleeves and pants and checking for ticks given the absence of an effective vaccine. Prophylactic antibiotic treatment is also highly effective for children who have experienced a Ixodes tick bite, occurring in an endemic region with tick attachment for at least 36 hours. Although Lyme disease disproportionately impacts children, the existing literature primarily focuses on its presentation in adults. In this review, we summarize the latest literature surrounding the diagnosis, treatment, and prevention of Lyme disease with a specific emphasis on pediatric populations and the recent advancements in vaccine development.

Safety of Longer-Term Doxycycline Use: A Systematic Review and Meta-Analysis With Implications for Bacterial Sexually Transmitted Infection Chemoprophylaxis.

Sexually transmitted infections (STIs) such as syphilis, gonorrhea, and chlamydia have significantly increased over the past decade in the United States. Doxycycline as chemoprophylaxis (i.e., postexposure prophylaxis) offers promise for addressing bacterial STIs. The goal of the current study was to evaluate the safety of longer-term doxycycline use (defined as 8 or more weeks) in the context of potential use as STI chemoprophylaxis through a systematic literature review and meta-analysis. This review used the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines to search MEDLINE/PubMed for clinical studies published from August 2003 to January 2023 that reported on adverse events with doxycycline use with a focus on side effects and metabolic effects of long-term use. A total of 67 studies were included in the systematic review. Overall, studies on longer-term doxycycline use reported 0% to greater than 50% adverse events ranging from mild to severe. Most common adverse events included gastrointestinal symptoms (i.e., nausea, vomiting, and abdominal pain), dermatologic (i.e., rash), and neurological (i.e., headache and dizziness) symptoms. Discontinuation of doxycycline due to adverse events was relatively uncommon in most studies. A meta-analysis of placebo controlled clinical trials (N = 18) revealed that gastrointestinal and dermatological adverse events were more likely to occur in the doxycycline group. Longer-term (8+ weeks) doxycycline use is generally safe and may be associated with minor side effects. Further research is needed on the potential metabolic impact of longer-term doxycycline use.

Cutaneous microvascular occlusion syndrome as the first manifestation of catastrophic lupus-associated antiphospholipid antibody syndrome: a case report

BackgroundAntiphospholipid syndrome (APS), defined by thrombotic events or obstetric complications in the presence of persistently high antiphospholipid antibodies, is characterized by a wide variety of clinical presentations and the effects of vascular occlusion can impact almost any organ system or tissue. Since adult-onset APS classification criteria are not well verified in pediatrics (where pregnancy-related problems are rare), estimating childhood prevalence is challenging. Stroke and pulmonary embolism are thromboembolic events occurring in children that can cause considerable long-term morbidity. Children with APS are more prone to recurrent thromboembolism than adults. Cutaneous symptoms are prominent and typically represent the first clue of APS. Although dermatologic findings are exceedingly heterogeneous, it is essential to consider which dermatological symptoms justify the investigation of antiphospholipid syndrome and the required further management.Case presentationWe describe a seven-year-old Iranian boy with retiform purpura and acral cutaneous ischemic lesions as the first clinical presentation of antiphospholipid syndrome in the setting of systemic lupus erythematous.ConclusionAPS in pediatrics, is associated with a variety of neurologic, dermatologic, and hematologic symptoms. Therefore, it is essential for pediatricians to be aware of the rare appearance of Catastrophic APS as an initial indication of APS.

Sporadic late onset nemaline myopathy with concurrent dermatological symptoms responding to immunosuppressive treatment

BackgroundSporadic late onset nemaline myopathy is a rare, progressive muscle disease, presenting in adulthood, mainly affecting proximal limb and bulbar muscles. Muscle biopsies show characteristic nemaline rods. The putative mechanism is considered immune-related. Other manifestations aside from neuromuscular symptoms have not been described previously.Case presentationWe present a case with atypical sporadic late onset nemaline myopathy (SLONM) of a non-HIV, non-MGUS subtype, where skin manifestations preceded neuromuscular symptoms, and a residual thymus with the histology of thymic follicular hyperplasia was detected during the diagnostic workup. Thorough dermatological investigations could not explain the skin presentations. Muscle biopsy revealed variation in fiber diameter, ragged-red and COX-negative fibers associated with discrete fibrosis. Electron microscopy detected atrophic muscle fibres with disorganization of the myofibrils, nemaline rods and abnormal mitochondria. Single-fiber EMG suggested signs of a neuromuscular transmission defect, EMG showed signs of myopathy. Analyses of antibodies associated with myasthenia gravis were negative. The patient showed improvement after intravenous immunoglobulin treatment regarding both the skin and the muscle symptoms.ConclusionsOur case highlights the heterogeneity of SLONM with its varied spectrum of presentation. A unique combination of dermatological symptoms and SLONM could be seen with skin lesions as primary presenting symptoms. An association can be considered between the different manifestations, presumably based on immune etiology, where immunosuppressive therapy has been beneficial.