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The Kaohsiung Journal of Medical... Dec 2022
Topics: Humans; COVID-19; COVID-19 Vaccines; Lupus Erythematosus, Systemic; Vaccination
PubMed: 36448713
DOI: 10.1002/kjm2.12630 -
Journal of Otolaryngology - Head & Neck... Oct 2019Cold agglutinin disease (CAD) is a rare condition leading to blood agglutination and autoimmune hemolytic anemia. Cutaneous ischemia resulting from CAD in the head and... (Review)
Review
Cold agglutinin disease (CAD) is a rare condition leading to blood agglutination and autoimmune hemolytic anemia. Cutaneous ischemia resulting from CAD in the head and neck is uncommon. Treatment regimens and outcomes vary widely in the literature and no clear protocol exists. This manuscript describes a patient with CAD who developed severe ischemia of the nose that resolved completely without sequellae following a medical regimen of aspirin, low molecular weight heparin, nitroglycerin ointment and hyperbaric oxygen therapy (HBOT). To our knowledge, this is the first reported case where nitroglycerin ointment or HBOT was successfully employed in the treatment of this complication.
Topics: Administration, Topical; Aged, 80 and over; Anemia, Hemolytic, Autoimmune; Anticoagulants; Combined Modality Therapy; Enoxaparin; Female; Humans; Hyperbaric Oxygenation; Ischemia; Nitroglycerin; Nose; Ointments; Vasodilator Agents
PubMed: 31640785
DOI: 10.1186/s40463-019-0369-0 -
Case Reports in Pediatrics 2019Neonatal acute myocardial infarction is an uncommon entity. We describe the case of a 4-day-old term baby who presented with respiratory distress and distal...
Neonatal acute myocardial infarction is an uncommon entity. We describe the case of a 4-day-old term baby who presented with respiratory distress and distal acrocyanosis. The chest radiograph demonstrated cardiomegaly without pleural effusion, and examination revealed hepatomegaly. An electrocardiogram revealed QS pattern in leads I, aVL, and V6, suggestive of ischemia. Cardiac enzymes were elevated, and echocardiogram revealed moderate left ventricular dysfunction with a thrombus at the level of the left atrial appendage. The patient required hemodynamic stabilization, vasodilatation to avoid congestive heart failure, and anticoagulation with heparin and aspirin. In the context of this unusual diagnosis, we reviewed our experience over the last 17 years as well as the existing literature on neonatal myocardial infarction.
PubMed: 31772807
DOI: 10.1155/2019/7203407 -
International Journal of... 2021Hypercoagulability is a risk factor of thromboembolic events in COVID-19. Anti-phospholipid (aPL) antibodies have been hypothesized to be involved. Typical COVID-19...
BACKGROUND
Hypercoagulability is a risk factor of thromboembolic events in COVID-19. Anti-phospholipid (aPL) antibodies have been hypothesized to be involved. Typical COVID-19 dermatological manifestations of livedo reticularis and digital ischemia may resemble cutaneous manifestations of anti-phospholipid syndrome (APS).
OBJECTIVES
To investigate the association between aPL antibodies and thromboembolic events, COVID-19 severity, mortality, and cutaneous manifestations in patients with COVID-19.
METHODS
aPL antibodies [anti-beta2-glycoprotein-1 (B2GP1) and anti-cardiolipin (aCL) antibodies] were titered in frozen serum samples from hospitalized COVID-19 patients and the patients' clinical records were retrospectively analyzed.
RESULTS
173 patients were enrolled. aPL antibodies were detected in 34.7% of patients, anti-B2GP1 antibodies in 30.1%, and aCL antibodies in 10.4%. Double positivity was observed in 5.2% of patients. Thromboembolic events occurred in 9.8% of patients, including 11 pulmonary embolisms, 1 case of celiac tripod thrombosis, and six arterial ischemic events affecting the cerebral, celiac, splenic, or femoral-popliteal arteries or the aorta. aPL antibodies were found in 52.9% of patients with vascular events, but thromboembolic events were not correlated to aPL antibodies (adjusted OR = 1.69, = 0.502). Ten patients (5.8%) had cutaneous signs of vasculopathy: nine livedo reticularis and one acrocyanosis. No significant association was observed between the presence of cutaneous vasculopathy and aPL antibodies ( = 0.692).
CONCLUSIONS
Anti-phospholipid antibodies cannot be considered responsible for hypercoagulability and thrombotic events in COVID-19 patients. In COVID-19 patients, livedo reticularis and acrocyanosis do not appear to be cutaneous manifestations of APS.
Topics: Adult; Aged; Aged, 80 and over; Antibodies, Anticardiolipin; Antibodies, Antiphospholipid; COVID-19; Female; Hospitalization; Humans; Italy; Male; Middle Aged; Retrospective Studies; SARS-CoV-2; Seroepidemiologic Studies; Skin Diseases; Vascular Diseases; beta 2-Glycoprotein I
PubMed: 34541915
DOI: 10.1177/20587384211042115 -
SAGE Open Medical Case Reports 2023A female in her 60s presented to the allergy and immunology clinic for further investigation of ongoing dermatitis. She presented with chronic acrocyanosis, mainly in...
A female in her 60s presented to the allergy and immunology clinic for further investigation of ongoing dermatitis. She presented with chronic acrocyanosis, mainly in her left lower extremity, extending distally from her mid thigh with concurrent ulcerations in her foot resulting in immobility secondary to pain. She experienced these symptoms for years without a definitive diagnosis. The lack of diagnosis was due, in part, to her atypical symptoms and laboratory findings that required a high level of clinical suspicion to diagnose. Extensive autoimmune workup was largely unrevealing with the exception of a cold agglutinin titer of 1:250 and a positive anticomplement C3b direct antiglobulin test. A diagnosis of cold agglutinin disease was made and treatment with rituximab monotherapy was initiated.
PubMed: 37654547
DOI: 10.1177/2050313X231191899 -
Pediatric Rheumatology Online Journal Oct 2019Infrared Thermography (IRT) has been used for over 30 years in the assessment of Raynaud Phenomenon (RP) and other peripheral microvascular dysfunctions in adults but,...
BACKGROUND
Infrared Thermography (IRT) has been used for over 30 years in the assessment of Raynaud Phenomenon (RP) and other peripheral microvascular dysfunctions in adults but, to date, very little experience is available on its use in children for this purpose. The first aim of the study was to assess reproducibility of thermographic examination after cold exposure by comparing inter-observer agreement in thermal imaging interpretation. The secondary aim was to evaluate whether IRT is reliable to diagnose and differentiate peripheral circulation disturbances in children.
METHODS
Children with clinical diagnosis of primary Raynaud's phenomenon (PRP), secondary RP (SRP), acrocyanosis (AC) and age-matched controls underwent sequential measurements of skin temperature at distal interphalangeal (DIP) and metacarpophalangeal (MCP) joints with IRT at baseline and for 10 min after cold challenge test. Intraclass correlation coefficient (ICC) was calculated for inter-rater reliability in IRT interpretation, then temperature variations at MCP and DIP joints and the distal-dorsal difference (DDD) were analysed.
RESULTS
Fourteen PRP, 16 SRP, 14 AC and 15 controls entered the study. ICC showed excellent agreement (> 0.93) for DIPs and MCPs in 192 measures for each subject. Patients with PRP, SRP and acrocyanosis showed significantly slower recovery at MCPs (p < 0.05) and at DIPs (p < 0.001) than controls. At baseline, higher temperature at DIPs and lower at MCPs was observed in PRP compared with SRP with significantly lower DDD (p < 0.001). Differently from AC, both PRP and SRP showed gain of temperature at DIPs and less at MCPs after cold challenge. PRP but not SRP patients returned to DIPs basal temperature by the end of re-warming time. Analysis of DDD confirmed that controls and PRP, SRP and AC patients significantly differed in fingers recovery pattern (p < 0.05).
CONCLUSION
IRT appears reliable and reproducible in identifying children with peripheral microvascular disturbances. Our results show that IRT examination pointed out that PRP, SRP and AC patients present significant differences in basal extremities temperature and in re-warming pattern after cold challenge therefore IRT can be suggested as an objective tool for diagnosis and monitoring of disease.
Topics: Adolescent; Case-Control Studies; Child; Child, Preschool; Diagnosis, Differential; Female; Humans; Infrared Rays; Male; Microvessels; Peripheral Vascular Diseases; Raynaud Disease; Reproducibility of Results; Skin Temperature; Thermography
PubMed: 31619252
DOI: 10.1186/s12969-019-0371-0 -
The American Journal of Tropical... Apr 2017AbstractChikungunya is a mosquito-borne infectious disease that has emerged as a global pathogen. The virus can pass vertically from mother to child especially during...
AbstractChikungunya is a mosquito-borne infectious disease that has emerged as a global pathogen. The virus can pass vertically from mother to child especially during the perinatal period, with an intrapartum vertical transmission rate of 50%. Approximately half of the neonates infected with chikungunya present with severe symptoms and infrequently death. This report summarizes two severe cases of vertically transmitted neonatal chikungunya infection. One case was confirmed by real-time reverse transcription polymerase chain reaction and the other fulfilled clinical and epidemiological criteria. Both infants presented on day 3 with abdominal distension, reduced perfusion pressure, and hypotension; acrocyanosis progressing to ischemic digits; and respiratory distress. Both died within 24-48 hours of presentation. The severity of symptoms observed is likely due to a combination of contamination of the fetal blood from highly viremic mothers during delivery and a low innate antiviral type-1 interferon response. Further examination of the neonate's innate immune response to chikungunya may provide clues for the development of potential treatment or vaccine interventions.
Topics: Chikungunya Fever; Fatal Outcome; Female; Humans; Infant, Newborn; Infectious Disease Transmission, Vertical; Jamaica; Male; Pregnancy; Pregnancy Complications, Infectious
PubMed: 28167590
DOI: 10.4269/ajtmh.16-0491 -
Infection and Drug Resistance 2023Chlamydia is a zoonotic pathogen that mainly infects poultry and pet birds. This Gram-negative obligate intracellular parasite also causes human psittacosis, the...
Chlamydia is a zoonotic pathogen that mainly infects poultry and pet birds. This Gram-negative obligate intracellular parasite also causes human psittacosis, the severity of which varies from mild flu-like symptoms to life-threatening severe pneumonia, including sepsis, acute respiratory distress syndrome, and multiple organ failure. Inhalation of aerosols from contaminated bird excreta through the respiratory tract is the main route of transmission to humans. Here, we present a case of pneumonia accompanied by lower extremity atherosclerotic occlusive disease. A 48-year-old man was admitted to the emergency department with a four-day history of cough and dyspnea. A detailed history revealed his contact with domestic pigeons. The results of metagenomic next-generation sequencing of bronchoalveolar lavage fluid suggested infection. Antibacterial agents were switched to targeted doxycycline, but in the next week, skin examination revealed acrocyanosis of both lower extremities, and the remarkable palpable purpura progressively worsened. Re-examination of the lower extremity vascular ultrasound suggested left dorsalis pedis artery occlusion and right peroneal vein thrombosis, which resulted in the amputation of both legs. This case is the first report of pneumonia combined with arterioocclusive sclerosis of both lower extremities.
PubMed: 37077253
DOI: 10.2147/IDR.S393256 -
Case Reports in Emergency Medicine 2015Prilocaine-induced methemoglobinemia is a rarely seen condition. In this paper, a case is presented with methemoglobinemia developed secondary to prilocaine use in a...
Prilocaine-induced methemoglobinemia is a rarely seen condition. In this paper, a case is presented with methemoglobinemia developed secondary to prilocaine use in a liposuction procedure, and the importance of this rarely seen condition is emphasized. A 20-year-old female patient presented with complaints of prostration, lassitude, shivering, shortness of breath, and cyanosis. It was learned that the patient underwent nearly 1000 mg prilocaine infiltration 8 hours priorly during a liposuction procedure. At admission, her blood pressure (130/80 mmHg), pulse rate (140 bpm), body temperature (36°C), and respiratory rate (40/min) were recorded. The patient had marked acrocyanosis. The arterial blood gas methemoglobin level was measured as 40%. The patient received oxygen therapy with a mask and was administered vitamin C in normal saline (500 mg tid), N-acetylcysteine (300 mg tid), and 50 mg 10% methylene blue in the intensive care unit of the internal medicine department. Methemoglobin level dropped down to 2% after her treatment with methylene blue and she was clinically cured and discharged 2 days later. Emergency service physicians should remember to consider methemoglobinemia when making a differential diagnosis between dyspnea and cyanosis developing after prilocaine infiltration performed for liposuctions in the adult age group.
PubMed: 26199764
DOI: 10.1155/2015/282347 -
Frontiers in Pediatrics 2022Bier anemic spots, cyanosis, and urticaria-like eruption (BASCULE) syndrome is an underreported pediatric vascular disorder from the group of acrosyndromes. In children,...
INTRODUCTION
Bier anemic spots, cyanosis, and urticaria-like eruption (BASCULE) syndrome is an underreported pediatric vascular disorder from the group of acrosyndromes. In children, these include paroxysmal acrosyndromes (Raynaud's phenomenon and chilblain-like lesions), permanent acrosyndromes (acrocyanosis), and transient acrosyndromes, in which their pathogeneses are associated with virus infections, Epstein-Barr virus, and, more recently, SARS-CoV-2, respectively.
METHODS
We reported a case of BASCULE syndrome associated with postural orthostatic tachycardia syndrome (POTS) and provided a narrative review of case reports describing the BASCULE syndrome in children. Moreover, we presented the results of a prospective practice survey that we performed in the French medical community.
RESULTS
A 14-years-old boy reported pruritic erythrocyanic lesions on the lower limbs, which occurred whenever he was in a standing position and fully resolved when he laid down. He reported asthenia and cramps. He presented a typical BASCULE syndrome associated with POTS confirmed by a tilt-test. Physical and vascular examinations were within the normal range. We identified 12 case reports, describing 21 pediatric cases since 2016. Most patients were adolescents between 12 and 19 years of age or were newborns. Furthermore, 20% of cases in the literature have presented POTS or orthostatic intolerance. Our survey among 95 French physicians confirmed that BASCULE syndrome is an underdiagnosed and under recognized disease in the general pediatric practice, at least in France. Among these physicians, 65% had already encountered patients with similar symptoms, but only 30% declared that they had knowledge of the BASCULE syndrome.
CONCLUSION
The under-recognition of the clinical manifestations leads the patients to consult emergency rooms, with multiple unnecessary investigations performed. Therefore, we suggest that the diagnosis of BASCULE syndrome is based on clinical observations, without the need for laboratory tests, to avoid unnecessary health costs. We suggest physicians to perform a tilt-test when POTS is suspected.
PubMed: 35463901
DOI: 10.3389/fped.2022.849914