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Background: Lymphoproliferation is the persistent proliferation of lymphoid cells and it's incidence in inborn errors of immunity varies from 0.7 to 18%. Material(s) and Method(s): This is a retrospective analysis of patients referred to the department of Immunology, B. J. Wadia Hospital for Children, Mumbai between March 2017 to December 2022. Inclusion criteria consisted of 3 months duration of significant lymphadenopathy and/or splenomegaly or history of lymphoma. The clinical characteristics, laboratory and molecular findings of the included patients were analyzed. Result(s): A total of 66 patients were included. There was a male preponderance with male:female ratio of 25:8. Median age of onset of lymphoproliferation was 4.75 years(Range 1 year to 60 years). Splenomegaly was seen in 75%. Infections included recurrent pneumonia (14/66), recurrent ear infections(5/66), COVID(4/66), one episode of pneumonia(6/66), herpes zoster(3/66), recurrent subcutaneous abscess (3/66), abdominal koch(3/66), chronic sinusitis(2/66), dermatophytosis(2/66), esophageal candidiasis(2/66), recurrent malaria(1/66), recurrent varicella(1/66), cryptococcal meningitis(1/66), gram negative sepsis(1/66), BCG adenitis(1/66), pseudomonas osteomyelitis(1/66), impetigo (1/66), pseudomonas urinary tract infection (1/66), chicken pox(1/66), herpes keratitis(1/66), dengue(1/66), Other manifestations included Evans plus phenotype(10/66), Evans phenotype(8/66), Autoimmune hemolytic anemia(5/66), bronchiectasis(5/66), Type 1 diabetes(3/66), hyper reactive airway disease(2/66), inflammatory bowel disease(4/66), autoimmune thrombocytopenia(2/66), stroke(3/66), hemophagocytic lymphohistiocytosis(2/66), hypertriglyceridemia(2/66), hypothyroidism(2/66), celiac disease(1/66), Type 2 diabetes(1/66), autoimmune encephalitis(1/66), autoimmune hepatitis(2/66), anti-parietal cell antibody(1/66), arthritis(1/66), autoimmune enteropathy(1/66), systemic lupus erythromatosus(1/66), primary biliary cirrhosis requiring liver transplant(1/66), nephrotic syndrome(1/66), lymphoedema(1/66), hypersplenism(1/66), recurrent oral ulcers(1/66), gout(1/66), dermatitis(1/66), ovarian teratoma(1/66), alopecia areata(1/66). Hodgkin's lymphoma(HL) was the most common malignancy(9/66), followed by non Hodgkin lymphoma(NHL)(6/66), transformation from NHL to HL(1/66), Burkitt to T-cell lymphoma(1/66), HL to DLBCL(1/66), HL to anaplastic T-cell lymphoma(1/66). EBV driven lymphoproliferation was seen in biopsy of21/66. Genetic testing showed mutations in LRBA(11/66), PIK3CD(5/66), CTLA4(3/66), TET2(2/66), IL2RA (1/66), IL12RB1(1/66), BACH2(1/66), PRKCD(1/66), TNFSFR13B(1/66), TNFAIP3(1/66), FAS(2/66), FASL(1/66), Caspase8(1/66), CARD11(1/66), RTEL1(1/66), AICD(1/66), PIK3R1(1/66), IKBKB(1/66). Treatment included IVIG, chemotherapy, rituximab, sirolimus, abatacept, HSCT. Conclusion(s): All children with persistent lymphoproliferation, with or without autoimmunity and/or infections should be worked up for an underlying monogenic disorder of immune dysregulation. Lymphomas presenting at abnormal site and/or age, relapse and EBV driven lymphomas require further evaluation. Presence of monogenic cause helps in providing targeted therapy.Copyright © 2023 Elsevier Inc.
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Introduction: The pandemic of COVID-19 has several implications for patients with chronic stress-sensitive diseases such as alopecia areata (AA). On the other hand, the vulnerability of AA patients using immunosuppressives to a more severe infection is in the shadow of ambiguity. This teledermatology study aimed to evaluate the course and outcome of AA in patients during this challenging period. Methods: Patients with AA who had previously received systemic therapy included in this study. Information about demographic data, AA history, characteristics, and treatments, hair loss progression, Corona Disease Anxiety Scale (CDAS), adherence to protective measures against the COVID-19, possible infection, and its features obtained via a telephone call. Results: A total of 57 patients participated. The majority (84.2%) of the participants had mild anxiety assessed by CDAS. Two patients (3.5%) had got infected with COVID-19. Twenty-one (36.8%) participants experienced hair loss progression. Hair loss progression correlated with drug dose reduction (OR: 46.09, 95% CI 5.48-387.14, p < 0.001) although it did not influence by the level of anxiety evaluated by the CDAS (p > 0.05). Conclusion: The anxiety perceived by severe AA patients about COVID-19 is mild; however, many experience hair loss progressions owing to their drug dose reduction.
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Key Clinical Message: Alopecia areata may develop in patients after COVID-19 vaccination. Platelet-rich plasma (PRP) has an outstanding anti-inflammatory effect and could be an alternative treatment for alopecia patients who are refractory or intolerant to corticosteroids. Abstract: A 34-year-old female with no systemic illness presented with non-scarring hair loss after the second COVID-19 vaccination shot 4 weeks ago. The hair loss worsened and progressed to severe alopecia areata. We started double-spin PRP therapy. Her hair recovered completely after six courses of PRP treatment.
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Alopecia areata is a common autoimmune condition that causes a non-scarring form of hair loss. It is associated with several viruses and diseases. One of the viruses that have been linked to alopecia areata is the coronavirus disease of 2019 (COVID-19). It was found to cause the onset, aggravation, or recurrence of alopecia areata in previously infected patients. We report the case of a 20-year-old woman who was medically free and presented with the severe and progressive onset of alopecia areata after one month of contracting COVID-19. This study aimed to explore the literature on COVID-19-associated severe onset alopecia areata in terms of timeline and clinical presentation.
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Hair-loss diseases comprise heterogenous conditions with respective pathophysiology and clinicopathological characteristics. Major breakthroughs in hair follicle biology and immunology have led to the elucidation of etiopathogenesis of non-scarring alopecia (e.g., alopecia areata, AA) and cicatricial alopecia (e.g., lichen planopilaris, LPP). High-throughput genetic analyses revealed molecular mechanism underlying the disease susceptibility of hair loss conditions, such as androgenetic alopecia (AGA) and female pattern hair loss (FPHL). Hair loss attracted public interest during the COVID-19 pandemic. The knowledge of hair loss diseases is robustly expanding and thus requires timely updates. In this review, the diagnostic and measurement methodologies applied to hair loss diseases are updated. Of note, novel criteria and classification/scoring systems published in the last decade are reviewed, highlighting their advantages over conventional ones. Emerging diagnostic techniques are itemized with clinical pearls enabling efficient utilization. Recent advances in understanding the etiopathogenesis and management for representative hair diseases, namely AGA, FPHL, AA, and major primary cicatricial alopecia, including LPP, are comprehensively summarized, focusing on causative factors, genetic predisposition, new disease entity, and novel therapeutic options. Lastly, the association between COVID-19 and hair loss is discussed to delineate telogen effluvium as the predominating pathomechanism accounting for this sequela.
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Alopecia areata (AA) is an autoimmune disorder that results in patchy hair loss. There have been increasing reports, though limited, in the medical literature of relapse of AA in affected patients and sudden onset of AA in previously unaffected patients after COVID-19 infection or vaccination. We describe a unique case of a middle-aged woman in the United States with sudden onset of severe AA immediately after receiving the Pfizer-BioNTech vaccine. We explain our case's trichoscopic findings, review the literature surrounding AA and COVID-19, and discuss the pathogenesis of how COVID-19 vaccination and infection may induce AA in patients without previous history of alopecia.
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Alopecia Areata (AA) is an autoimmune disorder characterized by hair loss, due to a T-lymphocyte-mediated inflammatory response targeting anagen-phase hair follicles. Former studies have suggested that vaccination and viral infections may play a role in the onset of AA through a variety of different mechanisms. We report here two cases of AA following COVID-19 vaccination.
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OBJECTIVE: We sought to determine the risk of contracting coronavirus disease (COVID-19) in individuals with alopecia areata (AA) compared to individuals without AA. METHOD(S): We queried the Symphony Health-derived data from the COVID-19 Research Database, and individuals with a diagnosis of AA from 2019 to 2020 were included in the AA cohort. Subjects with no record of AA diagnosis from 2019 to 2020 were randomly placed in the control group in a 4:1 size ratio compared with the AA group. Laboratory-confirmed cases of COVID-19 between January 1, 2020, and September 1, 2021, were identified. RESULT(S): The AA and non-AA cohorts included 73,784 and 280,991 subjects, respectively. The COVID-19 incidence rate ratio (IRR) for adults with AA was 0.72 (95% CI 0.68, 0.76) compared with adults without AA (p<0.001). Within the AA cohort, moderate-severe AA showed a similar decreased risk in COVID-19 infection compared to mild AA. LIMITATIONS: This study is limited by its retrospective nature and the use of ICD-10 codes for the identification of individuals with AA and COVID-19, which may underestimate the true burden of disease. CONCLUSION(S): Individuals with AA have a slightly decreased risk of contracting COVID-19. Notably, it has been demonstrated that interferon-gamma (IFN- gamma) leads to the downregulation of the angiotensin-converting enzyme 2 (ACE2), the SARS-CoV receptor.1 Thus, it is possible that increased levels of IFN- gamma seen in individuals with AA confer some protection against this viral infection.Copyright © 2022 Matrix Medical Communications. All rights reserved.
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BACKGROUND: Alopecia areata (AA) is a CD8+ T cell mediated autoimmune disease characterized by non-scarring hair loss. Ivarmacitinib, a selective oral Janus kinase 1 (JAK1) inhibitor, may interrupt certain cytokine signaling implicated in the pathogenesis of AA. OBJECTIVE: To evaluate the efficacy and safety of ivarmacitinib in adult AA patients who have ≥25% scalp hair loss. METHODS: Eligible patients were randomized 1:1:1:1 to receive ivarmacitinib 2 mg, 4 mg, or 8 mg QD or placebo for 24 weeks. The primary endpoint was percentage change from baseline in Severity of Alopecia Tool (SALT) score at week 24. RESULTS: A total of 94 patients were randomized. At week 24, the least squares mean (LSM) difference in percentage change from baseline in SALT score for ivarmacitinib 2 mg,4 mg, 8 mg, and placebo groups were -30.51% (90% confidence interval [CI]: -45.25, -15.76), -56.11% (90% CI: -70.28, -41.95), -51.01% (90% CI: -65.20, -36.82) and -19.87% (90% CI: -33.99, -5.75), respectively. Two SAEs, follicular lymphoma, and COVID-19 pneumonia were reported. LIMITATIONS: Small sample size limits the generalizability of the results. CONCLUSION: Treatment with ivarmacitinib 4 mg and 8 mg doses in moderate and severe AA patients for 24 weeks was efficacious and generally tolerated.
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Numerous cutaneous manifestations related to the COVID-19 (severe acute respiratory syndrome coronavirus 2) viral infection have been reported in literature. In this case report, we describe two acute hair-associated manifestations-the first being alopecia areata and the second is a case of Marie Antoinette syndrome or Canities subita where all the scalp hair has become white almost overnight. Both entities of hair changes were seen in the same patient.
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COVID-19 vaccines have positively changed the course of the pandemic. They entered the market after only one year of the initial trials, which that yielded positive results in terms of safety and efficacy. However, after inoculating billions of people in the most extensive vaccination campaign worldwide, mild but common and some rare but potentially fatal adverse events have been reported. Among several self-reported adverse events, hair loss and alopecia have been linked to COVID-19 mRNA or viral vector vaccines. We tracked and followed a series of five cases with post-vaccine telogen effluvium and alopecia development in Ecuador. Here, we reported the clinical presentation of two women and three men with the diagnosis of post-vaccine hair loss. All patients received a heterologous vaccination scheme (mRNA and attenuated virus vaccine) with an additional viral vector booster associated with the apparition of telogen effluvium and alopecia universalis between 3 and 17 days after the vaccine was administered.
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Introduction: Alopecia areata (AA) is a common autoimmune disease characterized by non-scarring hair loss. New onsets of AA have been associated with coronavirus disease 2019 (COVID-19). Various skin diseases have already been reported because of the vaccines (the Pfizer-BioNTech COVID-19 vaccine, the Moderna COVID-19 vaccine, the AstraZeneca vaccine) against severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2). Case Presentation: We report 5 cases of AA after COVID-19 vaccination. The trend shown by patients in this study is an initial worsening after the first dose of the vaccine with the stability of the disease even with subsequent doses. However, it is worth highlighting the case reported by one of our patients who suffered a "booster effect" of the disease with progressive and worsening alopecia with each vaccine booster. Discussion: The possible mechanism of action lies in the ability of COVID-19 vaccines to induce spike protein, which can lead to molecular mimicry phenomena. In an organism predisposed to autoimmunity, the mRNA vaccine acts as a trigger. Furthermore, we would like to point out how even cytokine storm and simple oxidative stress from SARS-CoV-2 infection can induce not only AA but also other types of hair loss such as telogen effluvium. Thus, this highlights how complex and multifaceted the phenomenon is.
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Background: Among many coronavirus disease 2019 (COVID-19) integumentary system involvements, alopecia is one of the least investigated. This disorder has been reported in many individuals who have suffered from varying severities of COVID-19. We aimed to systematically review studies evaluating the onset of different types of alopecia following COVID-19. Method(s): The PubMed, Scopus, Web of Science, and Embase databases were searched with a specified string of relevant keywords. After quality assessments, the data of eligible studies were qualitatively and quantitatively synthesized. Result(s): Sixteen studies were included in our review, six of which underwent meta-analysis for the differences in the risk of alopecia between males and females. Though the disorder was not differentiated in all the cases, we found that COVID-19 was associated with several different types of alopecia, including telogen effluvium, alopecia areata, alopecia parvimaculata, and lichen planopilaris. Moreover, although statistically insignificant, the odds of developing alopecia favored the female population. Conclusion(s): Although many hypotheses have been suggested as to why an individual might be more predisposed to developing alopecia following COVID-19, the data obtained from the studies yielded results that could not lead to definite conclusions. Therefore, we recommend that further studies be conducted to evaluate the association between the two phenomena more confidently. Copyright © 2022, Iranian Society of Dermatology. All rights reserved.
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Olumiant® (Baricitinib) is a newly approved treatment for alopecia areata. Baricitinib is a selective and reversible inhibitor of Janus kinase that has shown promising results in two randomized, placebo-controlled phase-3 trials. A significantly higher number of patients achieved at least 80% scalp coverage following 36 weeks of treatment, compared to the placebo group. Adverse effects reported include acne and urinary tract infections, in addition to the warnings and precautions as highlighted in the product monograph. The current recommended regimen is 2 mg or 4 mg taken once daily, depending on the extent of hair loss. Other approved indications for baricitinib treatment include management of rheumatoid arthritis and the COVID-19 infections. (SKINmed. 2022;20:452-455).
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With the onset of the global pandemic due to the new virus SARS-CoV-2 and the development of vaccines against it, various adverse events have been reported to them. We present a clinical case of a 53-year-old Caucasian woman who presented one week after Pfizer vaccination against SARS-CoV-2 with severe muscle weakness, myalgia in upper and lower extremities, alopecia 2 cm in diameter, and two mucosal ulcerations on lower lip. No deviations from the hematological, biochemical and immunological indicators were found from the laboratory tests carried out. Two weeks later, the patient developed total alopecia on the scalp. Due to the causal relationship with the administered vaccine, the patient was not given a second dose and a diagnosis of Systemic Connective Tissue Disease was not accepted. Six months later, recovery and partial hair growth was observed in the affected areas of the capillitium. Alopecia areata is autoimmune in nature, with literature reporting initial or recurrent alopecia after vaccination. It is important that adverse events after vaccination are properly evaluated in order to choose a proper therapeutic approach. © 2022 Medical Information Center. All rights reserved.