Case Reports in Immunology最新文献

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IgG4-Related Disease Inflamatory Pseudotumor Affecting the Sternoclavicular Joint: A Clinical Challenge. 影响胸锁关节的igg4相关疾病炎性假瘤:一个临床挑战
IF 1.5
Case Reports in Immunology Pub Date : 2025-08-28 eCollection Date: 2025-01-01 DOI: 10.1155/crii/6543528
Orivaldo Alves Barbosa, Talita Guimarães Andrade, Sergio Saldanha Freire Simões, Andre Luis Coutinho de Araújo Macedo, Dower Frota Barroso, João Paulo Uchoa Fontenele, José Walter Correia
{"title":"IgG4-Related Disease Inflamatory Pseudotumor Affecting the Sternoclavicular Joint: A Clinical Challenge.","authors":"Orivaldo Alves Barbosa, Talita Guimarães Andrade, Sergio Saldanha Freire Simões, Andre Luis Coutinho de Araújo Macedo, Dower Frota Barroso, João Paulo Uchoa Fontenele, José Walter Correia","doi":"10.1155/crii/6543528","DOIUrl":"10.1155/crii/6543528","url":null,"abstract":"<p><p>IgG4-related disease (IgG4-RD) is a rare, progressive, and immune-mediated fibroinflammatory disorder that primarily affects middle-aged men and is more prevalent in Asian populations. Although extensively studied, its pathophysiology remains incompletely understood. This case report describes a 44-year-old male presenting with multiple abscesses and progressive inflammatory symptoms, ultimately diagnosed with IgG4-RD with musculoskeletal involvement. Imaging and histopathological evaluation confirmed osteolytic lesions and significant IgG4-positive plasma cell infiltration. Soft tissue tumors in IgG4-RD are exceptionally rare, further emphasizing the uniqueness of this case. The patient showed clinical improvement with corticosteroid therapy. This case highlights the importance of considering IgG4-RD in the differential diagnosis of soft tissue and bone lesions and underscores the need for a multidisciplinary diagnostic approach.</p>","PeriodicalId":42865,"journal":{"name":"Case Reports in Immunology","volume":"2025 ","pages":"6543528"},"PeriodicalIF":1.5,"publicationDate":"2025-08-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12411044/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145016432","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Successfully Overcoming Allergy to Anakinra Through Intravenous Desensitization in a Child "Case Report". 通过静脉脱敏成功克服儿童对阿那白过敏的病例报告。
IF 1.5
Case Reports in Immunology Pub Date : 2025-08-20 eCollection Date: 2025-01-01 DOI: 10.1155/crii/6246850
Tariq Al Farsi
{"title":"Successfully Overcoming Allergy to Anakinra Through Intravenous Desensitization in a Child \"Case Report\".","authors":"Tariq Al Farsi","doi":"10.1155/crii/6246850","DOIUrl":"10.1155/crii/6246850","url":null,"abstract":"<p><p>Anakinra, a recombinant interleukin-1 (IL-1) receptor antagonist, is effective in treating autoinflammatory conditions in children; however, it may elicit a hypersensitive reaction, thus requiring desensitization. We report a case wherein a novel intravenous Anakinra desensitization protocol was applied for a child with hypersensitivity and needle phobia. A 10-year-old girl with tumor necrosis factor receptor-associated periodic syndrome (TRAPS) and Anakinra hypersensitivity underwent a 12-step intravenous desensitization protocol at the Royal Hospital, Oman. Intravenous desensitization was successful without adverse events. Subsequent subcutaneous Anakinra administration was well tolerated without further hypersensitivity or inflammatory episodes. This case demonstrates successful intravenous desensitization against Anakinra in a child with TRAPS and needle phobia, thus enabling continued treatment. This approach may benefit others facing similar challenges and warrants further research.</p>","PeriodicalId":42865,"journal":{"name":"Case Reports in Immunology","volume":"2025 ","pages":"6246850"},"PeriodicalIF":1.5,"publicationDate":"2025-08-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12390699/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144972973","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Diagnostic Dilemma: Adult Onset Still's Disease Mimicking Lymphoma-A Case Report and Literature Review. 诊断困境:成人发病的斯蒂尔氏病模拟淋巴瘤一例报告及文献复习。
IF 1.5
Case Reports in Immunology Pub Date : 2025-08-08 eCollection Date: 2025-01-01 DOI: 10.1155/crii/5533371
Mohannad N AbuHaweeleh, Al-Jouhara Albaloshi, Mohammed Al-Hor, Majed Al-Theyab, Ahmad Almaslamani, Basant Elsayed, Moaz O Moursi, Abdelrahman Hamad
{"title":"Diagnostic Dilemma: Adult Onset Still's Disease Mimicking Lymphoma-A Case Report and Literature Review.","authors":"Mohannad N AbuHaweeleh, Al-Jouhara Albaloshi, Mohammed Al-Hor, Majed Al-Theyab, Ahmad Almaslamani, Basant Elsayed, Moaz O Moursi, Abdelrahman Hamad","doi":"10.1155/crii/5533371","DOIUrl":"10.1155/crii/5533371","url":null,"abstract":"<p><p><b>Introduction:</b> Adult-onset Still's disease (AOSD) is a rare systemic inflammatory condition with hallmark features of spiking fevers, arthritis, and a salmon-colored maculopapular rash. It typically affects young adults, with a bimodal age distribution of 15-25 and 36-46 years. The prevalence of AOSD ranges from 1 to 34 cases per million people, with an incidence rate of 0.16-0.4 per 100,000 individuals. AOSD's etiology remains unclear but is thought to involve genetic and environmental factors. Diagnosis relies on clinical criteria, such as the Yamaguchi criteria, and exclusion of other conditions. Misdiagnosis is common, particularly in regions where infections and malignancies with overlapping features are prevalent. This case report highlights a diagnostically challenging case of AOSD in a young woman, emphasizing the importance of thorough evaluation and accurate diagnosis. <b>Case Presentation:</b> A 21-year-old female with no prior comorbidities presented with persistent fever, a transient salmon-colored rash, and polyarthritis involving the wrists, shoulders, ankles, and small joints. Three months prior, she had been treated for left submandibular sialadenitis attributed to mumps. Laboratory work revealed elevated C-reactive protein (CRP), ferritin, and LDH levels with a normal erythrocyte sedimentation rate (ESR) and autoimmune profile. Imaging studies, including PET-CT, suggested malignancy, raising suspicion of lymphoma. However, lymph node biopsy showed reactive hyperplasia without evidence of malignancy. Ultimately, a diagnosis of AOSD was established based on clinical and laboratory findings. The patient was initiated on anakinra and prednisolone, leading to significant improvement. At a 2-month follow-up, she had complete resolution of symptoms. <b>Conclusion:</b> AOSD presents a significant diagnostic challenge due to its rarity and symptom overlap with infections, malignancies, and autoimmune diseases. This case highlights the importance of a thorough clinical evaluation and the application of established diagnostic criteria to facilitate early diagnosis and timely management.</p>","PeriodicalId":42865,"journal":{"name":"Case Reports in Immunology","volume":"2025 ","pages":"5533371"},"PeriodicalIF":1.5,"publicationDate":"2025-08-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12356662/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144875809","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Major Histocompatibility Complex Class II Deficiency Beyond Infancy. 主要组织相容性复合体II类缺陷超过婴儿期。
IF 1.5
Case Reports in Immunology Pub Date : 2025-07-27 eCollection Date: 2025-01-01 DOI: 10.1155/crii/8570051
Aziza Bachir Kattra, Ibtihal Benhsaien, Asmaa Drissi Bourhanbour, Zahra Aadam, Abderrahmane Errami, Fatima Ailal, Ahmed Aziz Bousfiha, Jalila El Bakkouri
{"title":"Major Histocompatibility Complex Class II Deficiency Beyond Infancy.","authors":"Aziza Bachir Kattra, Ibtihal Benhsaien, Asmaa Drissi Bourhanbour, Zahra Aadam, Abderrahmane Errami, Fatima Ailal, Ahmed Aziz Bousfiha, Jalila El Bakkouri","doi":"10.1155/crii/8570051","DOIUrl":"10.1155/crii/8570051","url":null,"abstract":"<p><p>Major histocompatibility complex class (MHC)-II deficiency is a rare autosomal recessive combined immunodeficiency, accounting for 4.1% of inborn errors of immunity (IEI) cases in North Africa and the Middle East. Most patients do not survive beyond the age of 10 years. The cases described in this study are rare and unusual for MHC-II deficiency. We report the cases of four unrelated patients of Moroccan origin with MHC-II deficiency. Immunophenotyping of lymphocyte subpopulations and analysis of human leukocyte antigen-DR (HLA-DR) expression were performed using flow cytometry. Genetic analysis was conducted through direct sequencing. The mean age of our patients was 18.75 years (range 16-26 years); the mean age at diagnosis was 14.07 years, and the mean age of onset of symptoms was 5.25 months. The clinical presentation is characterized by recurrent pulmonary infections with predominant bronchial dilatation and hemorrhagic rectocolitis. The diagnosis was confirmed in all patients by absence of HLA-DR expression and detection of the c.338-25_338del mutation in <i>RFXANK</i>. Three (75%) of our patients are still alive and are on monthly intravenous immunoglobulin (IVIG) therapy. It is important to consider MHC-II deficiency in the differential diagnosis of combined immunodeficiencies across all age groups. Further studies are needed to elucidate the various phenotypes associated with this condition.</p>","PeriodicalId":42865,"journal":{"name":"Case Reports in Immunology","volume":"2025 ","pages":"8570051"},"PeriodicalIF":1.5,"publicationDate":"2025-07-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12318625/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144776408","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Coexistence of a Leaky SCID Phenotype With Hyperphenylalaninemia in an Adult Case. 漏型SCID与高苯丙氨酸血症在成人病例中的共存
IF 0.7
Case Reports in Immunology Pub Date : 2025-03-19 eCollection Date: 2025-01-01 DOI: 10.1155/crii/9988821
Ugur Musabak, Tuba Erdogan, Muserref Sule Akcay, Serdar Ceylaner
{"title":"Coexistence of a Leaky SCID Phenotype With Hyperphenylalaninemia in an Adult Case.","authors":"Ugur Musabak, Tuba Erdogan, Muserref Sule Akcay, Serdar Ceylaner","doi":"10.1155/crii/9988821","DOIUrl":"10.1155/crii/9988821","url":null,"abstract":"<p><p>In recent years, due to the widespread use of advanced molecular diagnostic methods, it has become clear that individuals in particular born from consanguineous marriages may be carriers of different genetic diseases. For this reason, cases where diseases related to inborn errors of immunity (IEI) and metabolism errors are detected in the same patient are encountered more frequently. In patients affected by different genetic defects, the pathophysiology is more complex, and disease management is more difficult. In this article, we aimed to draw attention to this complex genetic carrier state in a male with primary immunodeficiency (PID). In the patient who presented with recurrent lower respiratory tract infections, bronchiectasis, asthma and nasal polyps, and antibody deficiencies as well as cellular immunodeficiency findings were detected in the immunological analyses. In the whole exome sequencing (WES) study, three different variants were detected, two in genes related to PIDs (DCLRE1C and TNFRSF13B) and one in the gene related to phenylalanine metabolism (phenylalanine hydroxylase (PAH)). In the light of the current findings, the patient was evaluated as having leaky severe combined immunodeficiency (SCID) with immune phenotype T-B-natural killer (NK)+ and hyperphenylalaninemia (HPA). This case showed us that metabolic diseases may accompany a delay in the diagnosis of SCID and patients should be evaluated with a multidisciplinary approach.</p>","PeriodicalId":42865,"journal":{"name":"Case Reports in Immunology","volume":"2025 ","pages":"9988821"},"PeriodicalIF":0.7,"publicationDate":"2025-03-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11944794/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143732214","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Hemizygous Moesin (MSN) Gene Deletion in an Adult With Chronic Neutropenia. 成人慢性中性粒细胞减少症的半合子Moesin (MSN)基因缺失。
IF 0.7
Case Reports in Immunology Pub Date : 2024-12-31 eCollection Date: 2024-01-01 DOI: 10.1155/crii/3860726
Yandy Marx Castillo-Aleman, Francisco Sotomayor-Lugo, Sherjeel Sana, Nameer Abdul Raheem Kadhum Al-Saadawi, Yendry Ventura-Carmenate, David Dennison, Gianina Statache, Julieta Osorio-Zuluaga, Ahmad Raza, David Grossman
{"title":"Hemizygous Moesin (MSN) Gene Deletion in an Adult With Chronic Neutropenia.","authors":"Yandy Marx Castillo-Aleman, Francisco Sotomayor-Lugo, Sherjeel Sana, Nameer Abdul Raheem Kadhum Al-Saadawi, Yendry Ventura-Carmenate, David Dennison, Gianina Statache, Julieta Osorio-Zuluaga, Ahmad Raza, David Grossman","doi":"10.1155/crii/3860726","DOIUrl":"https://doi.org/10.1155/crii/3860726","url":null,"abstract":"<p><p>X-linked moesin-associated immunodeficiency (X-MAID) is a recently identified combined immunodeficiency caused by a mutation in the moesin (<i>MSN</i>) gene. It is characterized by cytopenias, hypogammaglobulinemia, poor immune response to vaccine antigens, and increased susceptibility to early-life infections. We report a patient with adult-onset neutropenia, lymphopenia, inadequate response to the pneumococcal polysaccharide vaccine (PPSV23), and recurrent bacterial infections associated with a hemizygous <i>MSN</i> deletion. Notably, the patient has no history of significant childhood infections, cytopenias, or hypogammaglobulinemia. Although only a few cases have been documented worldwide, we underscore the importance of whole-genome sequencing (WES) in diagnosing this atypical immunodeficiency disease in adulthood. Moreover, this report may shed light on our understanding of further variants of X-MAID and enrich the known spectrum of the disease.</p>","PeriodicalId":42865,"journal":{"name":"Case Reports in Immunology","volume":"2024 ","pages":"3860726"},"PeriodicalIF":0.7,"publicationDate":"2024-12-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11707056/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142956495","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Allergic Contact Dermatitis Reaction to Permanent Tattoo Containing Paraphenylenediamine: A Case Report. 含有对苯二胺的永久性纹身引起的过敏性接触性皮炎反应:病例报告。
IF 0.7
Case Reports in Immunology Pub Date : 2024-09-25 eCollection Date: 2024-01-01 DOI: 10.1155/2024/2118096
Eliot Parascandolo, Samuel Puglisi, Miguel Marenco, Gregory Puglisi
{"title":"Allergic Contact Dermatitis Reaction to Permanent Tattoo Containing Paraphenylenediamine: A Case Report.","authors":"Eliot Parascandolo, Samuel Puglisi, Miguel Marenco, Gregory Puglisi","doi":"10.1155/2024/2118096","DOIUrl":"10.1155/2024/2118096","url":null,"abstract":"<p><p>Paraphenylenediamine (PPD) is a well-known culprit allergen in the literature and clinical practice. Although this has been described in temporary tattoos, the definite implication of PPD in permanent tattoos has not been described. We report a patient who developed severe allergic contact dermatitis (ACD) requiring skin grafting after receiving a permanent tattoo with ink containing PPD. A 30-year-old female with a past history of atopic dermatitis and psoriasis presented with a 2-week history of cutaneous reaction to a recent tattoo. The patient noticed inflammation and irritation of the tattoo site the day after administration. The patient was previously identified on patch testing to have a PPD allergy after evaluation for dermatitis after hair dye application. Following the tattoo placement, she applied soap and bacitracin cream which she had used several years prior on a similar tattoo. On presentation 2 weeks later, she was found to have a deep ulcerated plaque with an indurated border encompassing the area of the tattoo. She was referred to the emergency department and admitted for treatment, ultimately requiring debridement and skin grafting. The patient obtained the safety data sheets for the tattoo inks which revealed PPD as an ingredient in every color. We believe this is the first confirmed case of PPD being implicated as the causative agent for ACD to a permanent tattoo. Tattoo ink is unregulated, and formulas are proprietary which makes safe practice difficult for patients with sensitivities. We advocate for consistent ingredient labeling, regulation, and transparency within the tattoo ink industry.</p>","PeriodicalId":42865,"journal":{"name":"Case Reports in Immunology","volume":"2024 ","pages":"2118096"},"PeriodicalIF":0.7,"publicationDate":"2024-09-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11446613/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142366871","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Two Unrelated Iranian Patients with Adenosine Deaminase 2 Deficiency: A Case Report and Review of Treatment. 两名无亲属关系的伊朗腺苷脱氨酶 2 缺乏症患者:病例报告和治疗回顾
IF 0.7
Case Reports in Immunology Pub Date : 2024-08-12 eCollection Date: 2024-01-01 DOI: 10.1155/2024/4380689
Parvaneh Karimzade, Aziz Eghbali, Mohammad Keramatipour, Reza Shiari, Zahra Golchehre, Mahdieh Taghizadeh, Mazdak Fallahi, Shahrzad Fallah, Nasrin Khakbazan Fard, Narges Eslami, Narges Bazgir, Mahnaz Jamee, Zahra Chavoshzadeh
{"title":"Two Unrelated Iranian Patients with Adenosine Deaminase 2 Deficiency: A Case Report and Review of Treatment.","authors":"Parvaneh Karimzade, Aziz Eghbali, Mohammad Keramatipour, Reza Shiari, Zahra Golchehre, Mahdieh Taghizadeh, Mazdak Fallahi, Shahrzad Fallah, Nasrin Khakbazan Fard, Narges Eslami, Narges Bazgir, Mahnaz Jamee, Zahra Chavoshzadeh","doi":"10.1155/2024/4380689","DOIUrl":"10.1155/2024/4380689","url":null,"abstract":"<p><strong>Background: </strong>Adenosine deaminase deficiency 2 (DADA2) is an autoinflammatory disorder, caused by the <i>CECR1</i> gene mutation. The major clinical manifestations include recurrent vasculitis, neurological disorders such as stroke, hematologic abnormalities, and immunodeficiency. As reported in previous studies, DADA2 may be manifested by ischemic or hemorrhagic strokes. This disorder also includes various hematological manifestations (pure red cell aplasia, pancytopenia, hemolytic anemia, and pancytopenia with bone marrow involvement). <i>Case Presentation</i>. In this case report, we present the clinical and immunological findings of two unrelated patients with DADA2. The first patient was a 7-year-old female who experienced recurrent neurological symptoms such as vertigo, tinnitus, hearing loss, and right-sided hemiparesis. Her brain magnetic resonance imaging (MRI) revealed a left-sided stroke, and she responded well to antitumor necrosis factor alpha agents and plasmapheresis. The second patient was a 6-year-old female who had recurrent fever and bicytopenia, aphthous lesions, cervical lymphadenopathy, and elevated liver enzymes. We also discussed the strategies used to manage the clinical manifestations in these two DADA2 patients.</p><p><strong>Conclusion: </strong>In this case report, we discussed two cases with DADA2 deficiency and their respective manifestations. The first case showed neurological symptoms while the second case had hematological symptoms. Although there is no established treatment for DADA2 due to its rarity, steroids are commonly used to treat this disorder. Antitumor necrosis factor is also effective in controlling the symptoms, especially the neurological ones. In cases where there is no appropriate response to these treatments, hematopoietic stem cell transplantation can be beneficial.</p>","PeriodicalId":42865,"journal":{"name":"Case Reports in Immunology","volume":"2024 ","pages":"4380689"},"PeriodicalIF":0.7,"publicationDate":"2024-08-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11333127/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142005433","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Risankizumab Efficacy in Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis (SAPHO) Remission: A Case Report on Rheumatologic and Dermatologic Disease Manifestations with Literature Review. 利桑珠单抗对滑膜炎、痤疮、脓疱病、骨质增生和骨膜炎(SAPHO)缓解的疗效:关于风湿病和皮肤病表现的病例报告及文献综述。
IF 1
Case Reports in Immunology Pub Date : 2024-03-19 eCollection Date: 2024-01-01 DOI: 10.1155/2024/9076852
Mario Ferraioli, Luigi Fiannacca, Elisabetta Greco, Eneida Cela, Mauro Fatica, Alberto Bergamini, Maria Sole Chimenti
{"title":"Risankizumab Efficacy in Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis (SAPHO) Remission: A Case Report on Rheumatologic and Dermatologic Disease Manifestations with Literature Review.","authors":"Mario Ferraioli, Luigi Fiannacca, Elisabetta Greco, Eneida Cela, Mauro Fatica, Alberto Bergamini, Maria Sole Chimenti","doi":"10.1155/2024/9076852","DOIUrl":"10.1155/2024/9076852","url":null,"abstract":"<p><p>SAPHO syndrome is a complex disease that encompasses both inflammatory arthritis and/or osteitis and dermatologic manifestations. It is considered a rare disease, in fact, no clinical trials have been conducted on its therapy and management. Therefore, therapeutic approach is based on small case studies. Here, we described the case of a 63-year-old woman affected by SAPHO syndrome, treated with the selective IL-23p19 antagonist, Risankizumab, after unsuccessful therapies with Methotrexate, Infliximab, Adalimumab, and an allergic reaction to Secukinumab. At the beginning of therapy, in November 2022, the patient presented with arthritis in both knees associated with palmar pustulosis and guttate psoriasis on the trunk. DAPSA score was 24, PtGA 80 mm, PASI score 11.1, and BSA 40%. Thereafter, Risankizumab was started at the standard dosage of 150 mg. At week 24 patient achieved clinical remission, DAPSA score was 8, PtGA was 30 mm, PASI was 1, and BSA 2.5. Patient maintained clinical remission state at the subsequent week 52 evaluation. At the same time, the patient did not report any adverse effects. Health-related quality of life was also assessed at the same time points aforementioned, showing significant improvement. In conclusion, this case report wants to point out the efficacy and safety of Risankizumab in SAPHO syndrome, reporting a sustained disease remission through a 12 months long follow-up period. We can consider IL-23p19 targeted therapy as a novel treatment option for SAPHO-with a high efficacy potential-especially on patients that have already been treated with other biologics.</p>","PeriodicalId":42865,"journal":{"name":"Case Reports in Immunology","volume":"2024 ","pages":"9076852"},"PeriodicalIF":1.0,"publicationDate":"2024-03-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10965285/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140294901","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Polymyalgia Rheumatica Post-SARS-CoV-2 Infection. SARS-CoV-2 感染后的多发性风湿痛
IF 1
Case Reports in Immunology Pub Date : 2024-03-14 eCollection Date: 2024-01-01 DOI: 10.1155/2024/6662652
Carolina Duarte-Salazar, José Eugenio Vazquez-Meraz, Lucio Ventura-Ríos, Cristina Hernández-Díaz, José Arellano-Galindo
{"title":"Polymyalgia Rheumatica Post-SARS-CoV-2 Infection.","authors":"Carolina Duarte-Salazar, José Eugenio Vazquez-Meraz, Lucio Ventura-Ríos, Cristina Hernández-Díaz, José Arellano-Galindo","doi":"10.1155/2024/6662652","DOIUrl":"10.1155/2024/6662652","url":null,"abstract":"<p><p>There is growing evidence that infection with severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) can lead to dysregulation of the immune system and, consequently, the development of autoimmune phenomena. Here, we describe the case of a 75-year-old woman with rheumatic manifestations characterized by intense musculoskeletal pain and stiffness in the neck and shoulders, with sudden onset and with the inability to raise her arms. The patient was admitted with severe pain located in the neck and shoulders. Previously, she had oropharyngeal pain, severe fatigue, and fever; a real-time polymerase chain reaction test for COVID-19 was positive. Two weeks later, the patient presented localized musculoskeletal pain in the neck and shoulders. Relevant laboratory results included an erythrocyte sedimentation rate of 46 mm/hr and a negative rheumatoid factor test; ultrasound findings with bilateral subacromial-subdeltoid bursitis were observed. A diagnosis of polymyalgia rheumatica (PMR) was initially made according to the EULAR/ACR provisional classification criteria for PMR; however, due to C-reactive protein negativity, the diagnosis was established based on symptoms. Management was with prednisone at the dose of 25 mg/day for 4 weeks and progressive reduction until prednisone suspension. The patient showed complete recovery at 6 months of follow-up. In this case, COVID-19 was implicated in the development of autoimmune and inflammatory rheumatic manifestations. PMR is a rare rheumatic condition that should be included in the wide range of rheumatologic manifestations expressed post-SARS-CoV-2 infection.</p>","PeriodicalId":42865,"journal":{"name":"Case Reports in Immunology","volume":"2024 ","pages":"6662652"},"PeriodicalIF":1.0,"publicationDate":"2024-03-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10957256/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140185867","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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