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Case report: Dupilumab therapy for alopecia areata in a 4-year-old patient resistant to baricitinib.Frontiers in Medicine 2023Alopecia areata (AA) is a non-scarring hair loss disorder. Alopecia totalis (AT) and alopecia universalis (AU) are the severe subtypes of AA. Age of onset before 6 years...
Alopecia areata (AA) is a non-scarring hair loss disorder. Alopecia totalis (AT) and alopecia universalis (AU) are the severe subtypes of AA. Age of onset before 6 years of age, disease duration of more than 1 year, and extensive alopecia involving more than 50% of the scalp (including AT or AU) suggest a poorer prognosis. Topical corticosteroids are the preferred first-line treatment for pediatric AA. While some treatments, such as intralesional corticosteroids, systemic steroids, contact immunotherapy with squaric acid dibutyl ester, and JAK inhibitors, showed efficacy in adults with AA, their safety profiles limit their use in pediatric AA patients. Dupilumab is a biologic that effectively addresses the patho-physiology of Th2 allergic diseases, and treats atopic diseases by inhibiting the helper Th2 immune axis. AA has been reported to be significantly improved with dupilumab for atopic dermatitis (AD) in children and adults. We report hair regrowth over all of the scalp, eyebrows, and eyelashes after 10 months of dupilumab therapy in a 4-year-old AU patient resistant to baricitinib.
PubMed: 37877023
DOI: 10.3389/fmed.2023.1253795 -
Dermatology and Therapy Aug 2023Alopecia areata (AA) is an autoimmune disease with an underlying immuno-inflammatory pathogenesis. Treatments can include systemic corticosteroids and immunomodulators...
INTRODUCTION
Alopecia areata (AA) is an autoimmune disease with an underlying immuno-inflammatory pathogenesis. Treatments can include systemic corticosteroids and immunomodulators (such as Janus kinase inhibitors); these medications may be associated with a risk of some adverse events. However, large-scale observational studies of baseline incidence rates (IRs) of infection, cardiovascular disease, malignancy, and thromboembolism in US patients with AA, including those with alopecia totalis or alopecia universalis (AT/AU), are limited. This real-world, US claims-based study aimed to estimate the incidence of events in patients with AA compared with matched patients without AA.
METHODS
Patients aged ≥ 12 years enrolled in the Optum Clinformatics Data Mart database from 1 October 2016 to 30 September 2020, with ≥ 2 AA diagnosis codes were included in the AA cohort. Patients without AA were age-, sex-, and race-matched 3:1 to patients with AA. Baseline comorbidities were evaluated during the 12-month period pre-index date. Incident cases of serious/herpes infections, malignancies, major adverse cardiovascular events (MACE), and thromboembolic events were evaluated post-index date. Data are presented using descriptive statistics, proportional percentages, frequencies, and IRs (calculated with 95% CI).
RESULTS
Overall, 8784 patients with AA, 599 of whom had AT/AU, were matched to 26,352 patients without AA. IRs per 1000 person-years among the AA and non-AA cohorts, respectively, were 18.5 and 20.6 for serious infections, 19.5 and 9.7 for herpes simplex infections, 7.8 and 7.6 for herpes zoster infections, 12.5 and 11.6 for primary malignancies, 16.0 and 18.1 for MACE, and 4.9 and 6.1 for venous thromboembolisms. Compared with patients with non-AT/AU AA, patients with AT/AU largely had higher IRs for most baseline comorbidities and outcome events evaluated.
CONCLUSION
Patients with AA had a higher IR of herpes simplex infection than the matched non-AA cohort. Patients with AT/AU generally had higher rates of outcome events than patients without AT/AU.
PubMed: 37296372
DOI: 10.1007/s13555-023-00937-9 -
Journal of Ayurveda and Integrative... 2023Alopecia areata (AA) is a T-cell-mediated autoimmune illness characterized by intermittent, non-scarring hair loss, Alopecia totalis(AT) is a type of AA characterized by...
Alopecia areata (AA) is a T-cell-mediated autoimmune illness characterized by intermittent, non-scarring hair loss, Alopecia totalis(AT) is a type of AA characterized by total hair loss on the face and scalp. Unfortunately, it is projected that 10-15 % of people with AA will advance to total hair loss on the scalp (AT) or hair loss on the scalp and body Alopecia Universalis (AU) only 10 % of patients with AT/AU recover completely. Treatment for severe AA is often unsatisfactory. The most popular AT/AU therapy techniques were topical steroid application and oral steroid administration. We present a case of Alopecia totalis that was treated with cupping therapy and ayurvedic treatments such as Punarnava Mandoor, manjistadi Kashaya, asanadi gana Kashaya, purnachandrodaya rasa, a churna combo, and Malatyadi and Dhurdhurapatradi taila for external application over scalp. The treatment's effectiveness is due to the synergistic action of all the herbs and the immunostimulant activity of cupping.
PubMed: 38043433
DOI: 10.1016/j.jaim.2023.100805 -
The Pan African Medical Journal 2023
Topics: Humans; Alopecia
PubMed: 38188881
DOI: 10.11604/pamj.2023.46.44.41181 -
Indian Dermatology Online Journal 2024Alopecia areata (AA) presents with noncicatricial alopecia and has multifactorial etiology. Janus Kinase inhibitors (JAKibs) with potential efficacy and favorable...
A Real-World Study of Steroid-Free Monotherapy with Tofacitinib in Severe and Therapy-Recalcitrant Alopecia Areata, Alopecia Totalis, and Alopecia Universalis Cases: A Retrospective Analysis.
BACKGROUND
Alopecia areata (AA) presents with noncicatricial alopecia and has multifactorial etiology. Janus Kinase inhibitors (JAKibs) with potential efficacy and favorable side-effect profile are the first class of drugs to receive FDA approval in AA.
OBJECTIVES
Our primary objective was to assess the complete response rates to tofacitinib monotherapy in severe and recalcitrant AA, alopecia totalis (AT), and alopecia universalis (AU) patients using the latest percentage change in Severity of alopecia tool (SALT) score. We also aimed to analyze the various systemic agents used by these patients prior to the use of tofacitinib.
MATERIALS AND METHODS
Institutional records of 17 patients with severe or refractory AA, AT, and AU treated with tofacitinib monotherapy were analyzed, retrospectively. The response to tofacitinib therapy was determined after calculating percentage change in SALT score. End of treatment was defined as the dose which resulted in a significant response (complete/near complete response was ≥75% hair regrowth from baseline as determined by SALT score).
RESULTS
Majority of patients had severe AA (SALT ≥ 50) ( = 9/17, 52.94%), while five patients had AT and three had AU. All patients had received either systemic glucocorticoids (GCS), which included oral mini pulse (OMP) ( = 8), intravenous pulse steroids ( = 4), and daily oral GCS ( = 6) or immunosuppressive agents (ISAs) which included cyclosporine ( = 14) followed by methotrexate ( = 6) and azathioprine ( = 6). Mean SALT score prior to starting tofacitinib was 74.23. Mean dose of tofacitinib used was 13.23 mg (10-15 mg) and mean duration of treatment was 9.23 months. Latest percentage change of SALT score ranged from 70.58% to 100%, with an average of 91.47%. Most patients showed complete/near complete response (13/17, 76.47%).
CONCLUSION
Tofacitinib was found to be safe and effective in severe/refractory AA, AU, and AT patients recalcitrant to other treatment modalities in our study. Further studies are needed to assess the effect of these targeted drugs on JAK-STAT expression or tissue cytokines involved in the pathogenesis of AA using immunohistochemistry.
PubMed: 38282998
DOI: 10.4103/idoj.idoj_131_23 -
Anais Brasileiros de Dermatologia 2024Alopecia Areata (AA) is an acquired autoimmune form of non-scarring hair loss. Adiponectin and its gene polymorphism were related to many autoimmune disorders.
BACKGROUND
Alopecia Areata (AA) is an acquired autoimmune form of non-scarring hair loss. Adiponectin and its gene polymorphism were related to many autoimmune disorders.
OBJECTIVE
Assessment of adiponectin serum levels and adiponectin gene (ADIPOQ) (rs2241766) Single Nucleoid Polymorphism (SNP) in AA patients and correlating the results with the disease severity in those patients.
METHODS
This study included 75 AA patients and 75 age and gender-matched healthy subjects (controls). The severity of Alopecia Tool (SALT) score assessment to evaluate AA severity was done. Adiponectin serum levels by ELISA and ADIPOQ (rs2241766) SNP using PCR were performed.
RESULTS
Adiponectin serum levels were significantly lower in AA patients than controls (p = 0.001). ADIPOQ (rs2241766) TG genotype and G allele were significantly predominant in AA patients increasing its risk by 5 and 4 folds (OR = 5.17, p = 0.001), (OR = 3.82, p = 0.001) respectively. Serum adiponectin levels were negatively correlated with SALT score (r = -0.435, p = 0.001) and associated with alopecia totalis (p = 0.016). ADIPOQ (rs2241766) TG genotype was significantly associated with low serum adiponectin levels and higher SALT score (p = 0.001).
STUDY LIMITATIONS
The small sample size.
CONCLUSIONS
ADIPOQ (rs2241766) gene polymorphism (TG genotype and G allele) may modulate AA risk and contribute to the development of AA in Egyptian populations. Decreased circulating adiponectin levels may have a dynamic role in AA etiopathogenesis. Adiponectin serum concentration can be considered a severity marker of hair loss in AA.
Topics: Humans; Adiponectin; Polymorphism, Single Nucleotide; Alopecia Areata; Egypt; Case-Control Studies; Genetic Predisposition to Disease
PubMed: 37985302
DOI: 10.1016/j.abd.2023.05.003 -
Skin Research and Technology : Official... Mar 2024Alopecia areata (AA) is an autoimmune condition characterized by sudden and unpredictable hair loss, with a lifetime incidence of 2%. AA can be divided into three...
BACKGROUND
Alopecia areata (AA) is an autoimmune condition characterized by sudden and unpredictable hair loss, with a lifetime incidence of 2%. AA can be divided into three categories: patchy alopecia, alopecia totalis, and alopecia universalis. It can affect a person's psychological health and overall quality of life. Elevated C-reactive protein (CRP) levels in the liver may indicate an inflammatory response in autoimmune diseases. Vitamin D, essential for immune system control and skin health, may be related to AA. Hair follicles contain vitamin D receptors, which control immunological responses in the skin. However, no study has found a relationship between CRP and vitamin D in AA patients in our region.
SUBJECTS AND METHODS
An analytical cross-sectional study with a case-control design research investigation of 82 AA patients and 81 healthy controls was carried out. Both groups' medical histories were taken. Biochemical analysis was done for both groups as well as the serum vitamin D levels, and CRP. Genetic analysis for CDX2 rs11568820 variant detected by PCR (T-ARMS-PCR) method and vitamin D receptor (VDR) gene expression measured by real-time PCR analysis for both patients and healthy subjects.
RESULTS
CRP levels are higher in AA patients, AA patients with G/G genotypes exhibited higher concentrations of CRP when compared to those with A/A and A/G genotypes while patients with A/A genotypes have higher levels of Serum vitamin D as compared to the A/G and G/G genotypes. G allele was more abundant in AA patients. VDR gene expression was lower in AA compared to control and lower in ophiasis compared to localized and multiple patchy AA. An important inverse linear correlation was observed between vitamin D and CRP levels in ophiasis AA.
CONCLUSION
CRP concentrations were found to be elevated in AA patients. The considerable accuracy of CRP in the diagnosis of AA is substantiated by a statistically significant al. A noteworthy inverse linear association was observed between serum vitamin D and CRP concentrations in ophiasis AA.
Topics: Humans; Alopecia Areata; C-Reactive Protein; Cross-Sectional Studies; Quality of Life; Vitamin D Deficiency; Vitamin D; Biomarkers
PubMed: 38528743
DOI: 10.1111/srt.13657 -
Dermatology and Therapy Apr 2024Alopecia areata (AA) is an autoimmune skin disease presenting as nonscarring hair loss. Information on the epidemiology of AA, especially the occurrence of AA and its...
INTRODUCTION
Alopecia areata (AA) is an autoimmune skin disease presenting as nonscarring hair loss. Information on the epidemiology of AA, especially the occurrence of AA and its subtypes within the general population, is scarce. The study aimed to estimate the incidence rates and prevalence of hospital-treated AA and its subtypes in Denmark and to examine the demographic and clinical characteristics of patients with AA, including comorbidities and use of prescription medications.
METHODS
This was a cohort study based on data from administrative and health registers in Denmark in 1995-2016. The study included individuals who were (1) registered with a hospital inpatient or hospital-based outpatient clinic diagnosis of AA between 1995 and 2016 in the Danish National Patient Registry covering encounters at all Danish hospitals, (2) alive and resided in Denmark anytime between 1995 and 2016, (3) aged ≥ 12 years, and (4) resided uninterrupted in Denmark during the 12 months before the first AA diagnosis during the study period.
RESULTS
During the study period, 2778 individuals with an incident hospital-based diagnosis of AA were identified; 63.1% were female and 28.7% of the patients were aged ≥ 50 years. Over the study period, the overall incidence rate for any hospital-treated AA per 100,000 person-years was 2.62 (95% confidence interval [CI], 2.53-2.72), and the overall prevalence in 2016 was 71.7 (95% CI 69.4-74.1) per 100,000 persons. Both incidence rate and prevalence increased over time. Prevalence of most hospital-treated comorbidities or history of medication use was below 10% and was similar in the alopecia totalis (AT)/alopecia universalis (AU) and non-AT/AU subtypes of AA.
CONCLUSION
This cohort study reported incidence rates and prevalence over time and characteristics of individuals with hospital-treated AA in Denmark, which are in agreement with those previously reported in this population.
PubMed: 38625633
DOI: 10.1007/s13555-024-01145-9 -
Skin Appendage Disorders Oct 2023Alopecia areata (AA) is an autoimmune condition that results in nonscarring hair loss. AA is comorbid with mental health disorders including anxiety and depression. This...
INTRODUCTION
Alopecia areata (AA) is an autoimmune condition that results in nonscarring hair loss. AA is comorbid with mental health disorders including anxiety and depression. This study aimed to evaluate the presence of post-traumatic stress disorder (PTSD) in relation to hair loss in patients with AA.
METHODS
A cross-sectional national survey was distributed using the National Alopecia Areata Foundation's (NAAF) email list. This study was approved by the Mass General Brigham Institutional Review Board. Participants were asked to complete the PTSD Checklist for the DSM-5 (PCL-5), a validated screening tool for PTSD in the context of their AA.
RESULTS
Of the 1,449 completed surveys (completion rate 79.6%), most respondents were female (83.8%) and white (76.6%) with an average age of 50.6 ± 15.6 years. Respondents had AA for an average of 17.7 ± 15.8 years, with 91.4% experiencing current active hair loss. A total of 33.9% of respondents screened positively for PTSD, with an average score of 48.8 ± 12.3 on the PCL-5 in participants who screened positively. Participants with alopecia totalis have the highest average PCL-5 score of 30.1 ± 19.2, followed by participants with alopecia universalis with an average score of 26.0 ± 19.9, and lastly patchy AA with an average score of 24.5 ± 18.3 ( = 0.003). Feelings of intrusion and avoidance were the predominant reported symptoms. Total PTSD scores were significantly higher in respondents who were younger and identified as Black or African American and Hispanic when compared to white and non-Hispanic respondents, respectively.
CONCLUSION
These findings identify that one in 3 patients with AA in this cohort meet the screening criteria for PTSD specifically relating to their hair loss experience. These results further highlight the mental health comorbidities associated with AA and emphasize that these symptoms may persist even after hair regrowth. Limitations include the nonrandomized NAAF population with most participants being white females. Future studies should confirm these findings in other patient populations. Finally, respondent's baseline mental health was not assessed; therefore, a causal relationship between AA and PTSD cannot be deduced.
PubMed: 37900780
DOI: 10.1159/000530356 -
Pediatric Rheumatology Online Journal Jan 2024STING-associated vasculopathy with onset in infancy (SAVI) is a rare type I interferonopathy caused by heterozygous variants in the STING gene. In SAVI, STING variants...
BACKGROUND
STING-associated vasculopathy with onset in infancy (SAVI) is a rare type I interferonopathy caused by heterozygous variants in the STING gene. In SAVI, STING variants confer a gain-of-function which causes overactivation of type I interferon (IFN) signaling leading to autoinflammation and various degrees of immunodeficiency and autoimmunity.
CASE PRESENTATION
We report the case of a 5 year old child and his mother, both of whom presented with systemic inflammatory symptoms yet widely varying organ involvement, disease course and therapeutic response. Genetic testing revealed a heterozygous STING variant, R281Q, in the child and his mother that had previously been associated with SAVI. However, in contrast to previously reported SAVI cases due to the R281Q variant, our patients showed an atypical course of disease with alopecia totalis in the child and a complete lack of lung involvement in the mother.
CONCLUSIONS
Our findings demonstrate the phenotypic breadth of clinical SAVI manifestations. Given the therapeutic benefit of treatment with JAK inhibitors, early genetic testing for SAVI should be considered in patients with unclear systemic inflammation involving cutaneous, pulmonary, or musculoskeletal symptoms, and signs of immunodeficiency and autoimmunity.
Topics: Child, Preschool; Humans; Immunologic Deficiency Syndromes; Inflammation; Interferon Type I; Lung; Mutation; Vascular Diseases; Male; Female
PubMed: 38178067
DOI: 10.1186/s12969-023-00934-4