American Journal of Case Reports最新文献

{"title":"A 43-Year-Old Man with a 30-Year History of a Retained Glass Intraocular Foreign Body Presenting with Retinal Detachment.","authors":"Jilin Zhou, Sahil Thakur, Chaoxu Qian","doi":"10.12659/AJCR.947369","DOIUrl":"10.12659/AJCR.947369","url":null,"abstract":"<p><p>BACKGROUND This report describes the case of a 43-year-old man with a 30-year history of a retained glass intraocular foreign body (IOFB) presenting with retinal detachment. CASE REPORT We describe this case using patient history, ocular examination, and imaging, including anterior segment photographs, fundus photographs, and B-scan ultrasonography. A 43-year-old man presented with retinal detachment in the right eye. Medical history revealed that the patient had suffered an explosive injury to the right eye 30 years ago, for which he underwent surgical repair at the time of the incident. However, a glass IOFB was left in the eye, either due to an oversight during the initial treatment or an intentional decision by the treating physician. On examination, indirect ophthalmoscopy revealed a half-disc-sized retinal hole at the 9 o'clock position in the peripheral retina, along with long-standing atrophic lesions extending from 12 to 2 o'clock. Temporal macular-involving retinal detachment extending from 6 to 11 o'clock was observed. B-scan ultrasonography confirmed the presence of the glass IOFB. The patient subsequently underwent surgical treatment, including pars plana vitrectomy, removal of the IOFB, retinal reattachment, photocoagulation, and silicone oil tamponade. CONCLUSIONS Patients with open globe injuries should be carefully evaluated with the presumption of an IOFB. IOFBs may remain asymptomatic for decades, underscoring the importance of regular follow-up for patients with retained IOFBs. If complications such as retinal detachment occur, they can be managed routinely.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e947369"},"PeriodicalIF":1.0,"publicationDate":"2025-05-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12077246/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144057160","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}

{"title":"Rhabdomyolysis Induced by Parainfluenza 2 Virus in a Healthy 18-Year-Old Male Patient: A Case Study.","authors":"Mustafa K Al Nuaimi, Sara Ubosy, Hakeem A Mohammed, Kwabena Ayesu, Mario Madruga, Stephen J Carlan","doi":"10.12659/AJCR.947909","DOIUrl":"10.12659/AJCR.947909","url":null,"abstract":"<p><p>BACKGROUND Rhabdomyolysis is a medical condition characterized by the cellular breakdown of skeletal muscle tissue, leading to the release of basic muscle components into the bloodstream. Multiple causes have been reported, including infections that involve skeletal muscle cells. One of the most common classes of infections causing rhabdomyolysis is viral in origin. Clinically, the patient usually reports a short latency period consisting of malaise followed by muscle soreness, myalgia, and even an antalgic gait. Higher degrees of skeletal muscle damage result in higher levels of serum myoglobin. When the levels of myoglobin exceed the protein binding capacity, the molecule can precipitate during glomerular filtration and damage the kidneys. CASE REPORT An 18-year-old healthy, active male patient was brought to the Emergency Department (ED) after experiencing 5 days of generalized malaise and 2 days of progressively debilitating skeletal muscle pain and malfunction, leading to the inability to walk. He had dismissed the symptoms until he was non-ambulatory. On arrival at the ED, his creatine kinase (CK) level was elevated at 6859 U/L (reference, 10-70 U/L), and parainfluenza type 2 virus was identified by a nasopharyngeal respiratory polymerase chain reaction panel. He received symptomatic treatment including acetaminophen, methocarbamol, and diuresis. He was discharged on day 5, and his CK level returned to normal by day 12 of his disease. CONCLUSIONS Parainfluenza virus is a rare cause of rhabdomyolysis. If the myalgia of a common \"cold or flu\" progresses to muscle swelling and tenderness, especially in the lower extremities, and impairs ambulation, urgent medical evaluation should be considered.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e947909"},"PeriodicalIF":1.0,"publicationDate":"2025-05-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12070817/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144050347","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}

{"title":"A Case of Undifferentiated Connective Tissue Disease with Bilateral Auricular Polychondritis Manifestations: A Rare Clinical Association.","authors":"Angelo Nigro, Pasquale Santarcangelo, Antonio Bonelli, Serena Digregorio, Giuseppe Nicoletti","doi":"10.12659/AJCR.946827","DOIUrl":"10.12659/AJCR.946827","url":null,"abstract":"<p><p>BACKGROUND Undifferentiated connective tissue disease (UCTD) is a heterogeneous autoimmune condition characterized by clinical features of connective tissue involvement without meeting the full classification criteria for a defined connective tissue disease (CTD). It often includes arthralgias, Raynaud's phenomenon, and serologic abnormalities. In rare cases, UCTD presents with features resembling relapsing polychondritis, raising questions about a possible overlap syndrome or a shared immunopathogenic mechanism. This case report describes an atypical presentation of UCTD with bilateral auricular inflammation and its therapeutic management. CASE REPORT A 45-year-old man diagnosed with UCTD initially presented with acral cyanosis and inflammatory arthralgias, which responded well to hydroxychloroquine and low-dose prednisone. Over time, he developed progressive bilateral auricular pain, erythema, and nodular swelling, mimicking polychondritis. In the absence of systemic features of relapsing polychondritis, this was considered an unusual manifestation within the UCTD spectrum rather than a distinct overlap syndrome. His symptoms improved significantly following an increase in corticosteroid therapy. CONCLUSIONS This case highlights the importance of recognizing atypical inflammatory manifestations in UCTD and adjusting treatment accordingly. The findings suggest auricular inflammation is part of the broader UCTD spectrum rather than a separate disease entity. These observations underscore the role of shared autoimmune pathways in connective tissue disorders and the need for individualized treatment approaches.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e946827"},"PeriodicalIF":1.0,"publicationDate":"2025-05-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12070814/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143989771","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}

{"title":"Rare Coexisting Hepatocellular and Gallbladder Carcinomas: A Case Report.","authors":"Kristína Cmarková, Lenka Nosakova, Miroslav Pindura, Roman Kyčina, Juraj Marcinek, Juraj Miklušica","doi":"10.12659/AJCR.946739","DOIUrl":"https://doi.org/10.12659/AJCR.946739","url":null,"abstract":"<p><p>BACKGROUND Multiple primary malignancies in a single patient are considered to be quite rare. However, due to the wider availability of advanced imaging methods and, more regular check-ups, they are becoming more common in clinical practice. To determine the diagnosis of multiple primary malignancies, each tumor must be completely separate and cannot be from metastasis of one or the other. Coexisting hepatocellular carcinoma and gallbladder carcinoma are extremely rare, with only a few case reports published. We believe that to determine the most effective therapeutic management, the results of as many cases as possible should be documented. Regardless of limited data, curative resection may be the most beneficial treatment option in terms of overall survival. CASE REPORT We present the case of a 64-year-old man first diagnosed with hepatocellular carcinoma with possible infiltration of the gallbladder wall as appeared on a CT scan. However, the definitive histopathologic examination revealed the coexistence of hepatocellular carcinoma and gallbladder carcinoma. In this case report, we offer an insight into the entire diagnostic process, as well as the chosen surgical approach and adjuvant therapy. Moreover, we present our approach to preoperative biopsy, the decision-making process throughout the whole diagnostic and therapeutic course, and the achieved results. CONCLUSIONS In cases of double primary malignancies, the choice of therapeutic strategy depends on the type and stage of both malignancies, but procedures with curative intent are superior. We present the outcome and overall survival of the patient after the surgery and adjuvant chemotherapy for synchronous hepatocellular carcinoma and gallbladder carcinoma. Since the liver is the organ most commonly affected by metastatic dissemination of primary tumors, exclusion of metastatic disease is particularly important.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e946739"},"PeriodicalIF":1.0,"publicationDate":"2025-05-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12067998/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144054890","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}

{"title":"A Rare Case of Hypoxia and Cyanosis Secondary to Multifactorial Medication-Induced Sulfhemoglobinemia.","authors":"Megan M Mansour, Robert M Seby, Aya Elalfy, Mario El Hayek, Tuhama S Rihani, Edson J Mwakyanjala, Libardo Rueda Prada","doi":"10.12659/AJCR.947587","DOIUrl":"https://doi.org/10.12659/AJCR.947587","url":null,"abstract":"<p><p>BACKGROUND Fatigue and dyspnea, especially with hypoxia, are common in acute care and can indicate various conditions. Sulfhemoglobinemia is a rare but important consideration when a large saturation gap is present, as most blood gas analyzers do not detect it. Medication exposure, particularly to phenazopyridine and other oxidant drugs, is a leading cause. Early recognition of this diagnosis is crucial, as recurrence is likely with re-exposure to the precipitating agent. CASE REPORT A 37-year-old woman presented with acute dyspnea, lethargy, and hypoxia after exposure to severe oxidizing agents, including first-time use of trimethoprim-sulfamethoxazole and phenazopyridine, as well as lidocaine during a recent dental procedure (not her first exposure). Shortly thereafter, she developed tachycardia, tachypnea, and hypoxia, with concurrent hemolytic anemia. Despite oxygen therapy, she had a persistent elevated saturation gap. Arterial blood gas confirmed dyshemoglobinemia, and further testing identified sulfhemoglobinemia (1.3%). She was treated with blood transfusion and discontinuation of oxidizing agents to prevent further hypoxia. CONCLUSIONS Sulfhemoglobinemia is a rare and often missed diagnosis, as most blood gases analyzers do not measure sulfhemoglobin. Despite this, this diagnosis requires great clinical suspicion, as patients are at high risk for repeated exposure to offending agents and recurrence of life-threatening hypoxia.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e947587"},"PeriodicalIF":1.0,"publicationDate":"2025-05-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12067997/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144051354","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}

{"title":"Iron Deficiency Anemia Linked to Severe Pediculosis Capitis in a 12-Year-Old Boy.","authors":"Kaylie N Nichols, Spencer B Price, Jonathan D Duhamell, Megan E Williams, Amruta Padhye, Alicia Bach, Tyler Severance","doi":"10.12659/AJCR.947139","DOIUrl":"https://doi.org/10.12659/AJCR.947139","url":null,"abstract":"<p><p>BACKGROUND Head lice is a common condition in school-age children caused by Pediculus humanus capitis. Although typically associated with scalp itching and discomfort, chronic or severe infestations can occasionally lead to more severe health consequences. This report describes a 12-year-old boy from Missouri, USA, presenting with iron deficiency anemia (IDA) and a severe head lice infestation. IDA is characterized by insufficient iron to meet the body's needs, typically due to blood loss, nutritional deficiencies, or malabsorption. While there is no established causative relationship between head lice and IDA, several case reports have suggested a co-occurrence, often in the absence of other identifiable causes. This raises the possibility that significant lice burden may contribute to or exacerbate IDA, particularly through blood loss from lice feeding on the host's scalp. CASE REPORT A 12-year-old boy presented to his pediatrician with fatigue, pallor, and weakness. Initial outpatient laboratory test data revealed a hemoglobin of 3.4 g/dL, necessitating urgent transportation to the emergency department (ED). Upon examination in the ED, a severe Pediculus humanus capitis infestation was incidentally discovered. Despite extensive inpatient workup, no other source of anemia was identified. The patient was treated with blood transfusions and topical pediculicides to address his anemia and head lice, respectively. Remarkably, his anemia and symptoms resolved without recurrence following these interventions. CONCLUSIONS Prolonged pediculosis capitis with high lice burden should be considered as a potential etiologic factor in patients with unexplained IDA and a negative anemia workup. Further research is warranted to explore the mechanisms linking these 2 conditions.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e947139"},"PeriodicalIF":1.0,"publicationDate":"2025-05-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12065385/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144052560","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}

{"title":"Miliary Patterns on Computerized Tomography Imaging of Chest in Immunocompromised Patient: Unveiling Disseminated Histoplasmosis.","authors":"Sravani Kamatam, Anusha Akepati, Divya Shivakumar","doi":"10.12659/AJCR.946631","DOIUrl":"https://doi.org/10.12659/AJCR.946631","url":null,"abstract":"<p><p>BACKGROUND Disseminated histoplasmosis is a rare fungal infection caused by dimorphic fungus Histoplasma capsulatum. Immunocompetent patients are usually asymptomatic or have self-limiting disease; however, immunocompromised patients present as disseminated infection. Our case report includes an immunocompromised host with atypical presentation and imaging findings consistent with a miliary pattern that did not fit the classical clinical picture of disseminated histoplasmosis, causing a delay in the diagnosis. CASE REPORT We report a case of a patient who presented with shortness of breath, nonproductive cough, low-grade fevers, and weight loss, who received a diagnosis of disseminated histoplasmosis, with an underlying immunocompromised state. Due to the ineffectiveness of initial management with empiric antibiotics, imaging with computed tomography (CT) of the chest was done, revealing miliary disease, and tuberculosis was first in the differential diagnosis. She had a positive histoplasma urine antigen, and bronchial alveolar lavage fluid tested positive for histoplasma antigen, confirming miliary histoplasmosis. Her CT of the abdomen/pelvis showed multiple lesions in the spleen and adrenal glands bilaterally, concerning for disseminated histoplasmosis, which was treated with amphotericin B, followed by itraconazole. CONCLUSIONS Disseminated histoplasmosis is a rare and challenging diagnosis; there should be a low suspicion for histoplasmosis in patients treated with tumor necrosis factor inhibitors, as mortality is high in such cases. Despite the inability to detect antibodies in immunocompromised patients, definitive diagnostic tests must be considered through culture and histopathology, the criterion standard tests, before excluding infectious causes. This case highlights the challenges faced in diagnosing and treating miliary histoplasmosis in immunocompromised patients.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e946631"},"PeriodicalIF":1.0,"publicationDate":"2025-05-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12065387/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144033385","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}

{"title":"Methimazole-Induced Pancytopenia with ANCA Positivity: Diagnostic and Management Challenges.","authors":"Sanhitha Valasareddy, Farina Tariq, Muhammad Zaheer","doi":"10.12659/AJCR.947323","DOIUrl":"https://doi.org/10.12659/AJCR.947323","url":null,"abstract":"<p><p>BACKGROUND Pancytopenia is an exceedingly rare adverse effect of antithyroid medications. It can be associated with both propylthiouracil and methimazole. While agranulocytosis is a more common adverse effect, pancytopenia has unique diagnostic and management issues due to its potential severity. CASE REPORT We present the case of a 36-year-old woman who presented to the Emergency Department with pancytopenia 2 months after starting methimazole for Grave's disease. Her case was complicated by severe candidemia and Serratia bacteremia. Laboratory workup revealed positive cytoplasmic and perinuclear anti-neutrophil cytoplasmic antibodies (ANCA) on immunofluorescence testing, with negative results on ELISA for MPO and PR3 antibodies. These findings were attributed to methimazole-induced ANCA positivity rather than primary ANCA-associated vasculitis. Methimazole was discontinued, and the patient was managed with aggressive antimicrobial therapy and supportive care, including blood transfusions and antifungal treatment. Despite the complexity of her condition, she survived after several months of intensive care and showed gradual improvement in blood counts and overall clinical status. CONCLUSIONS This case underscores the importance of recognizing potential iatrogenic causes of pancytopenia in patients receiving antithyroid medications. Early identification, prompt discontinuation of the offending agent, and comprehensive supportive care are essential for positive outcomes. This case also highlights the necessity for vigilant monitoring, patient education, and regular follow-up in individuals on antithyroid medications to prevent severe complications, morbidity, and mortality associated with rare but serious adverse effects like pancytopenia.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e947323"},"PeriodicalIF":1.0,"publicationDate":"2025-05-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12063506/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144052585","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}

{"title":"Remimazolam Use in Awake Craniotomy for Patient with Morbid Obesity and Recurrent Glioma.","authors":"Małgorzata Małłek-Grabowska, Bartosz Kozlowski, Robert Włodarski, Piotr B Grabowski, Szymon Hoppe, Jacek Furtak","doi":"10.12659/AJCR.946483","DOIUrl":"https://doi.org/10.12659/AJCR.946483","url":null,"abstract":"<p><p>BACKGROUND Awake craniotomy is a neurosurgical procedure that is used when it is necessary for a patient to be conscious during resection of a tumor. In neurosurgical operations conducted within the eloquent cortex, it is essential to administer anesthesia to ensure that the patient does not experience pain or fear during the procedure and to ensure their full cooperation with the neurophysiologist and neurosurgeon in stimulating this cortex. The asleep-awake-asleep method was used in this case of awake craniotomy. In this case report, we evaluate remimazolam, a drug with ultra-short-acting properties and suitability for use in anesthesia during awake craniotomies, for its exceptional utility in this type of treatment. CASE REPORT The case report describes the anesthetic management of a 32-year-old man with morbid obesity (body mass index, 44 kg/m²) who underwent resection of a recurrent tumor (low-grade glioma) in the temporoparietal region. The patient was anesthetized with remimazolam, fentanyl, and remifentanil and underwent a scalp block. The patient did not experience any pain during the surgical procedure and was able to actively cooperate with the neuropsychologist during the crucial phase of tumor resection, once the effects of the anesthetic wore off. The patient's circulatory parameters remained within normal ranges, and no signs of respiratory depression were observed during 225 min of anesthesia. CONCLUSIONS Remimazolam offers an intriguing alternative to conventional sedatives used for sedation during awake craniotomies, particularly for patient groups at high risk of airway obstruction when heavily sedated, such as individuals with severe obesity.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e946483"},"PeriodicalIF":1.0,"publicationDate":"2025-05-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12063507/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144003329","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}

{"title":"Refractory Pleuritis in a Patient with Silicosis, Systemic Sclerosis, and Sjögren's Syndrome: Considering the Potential Role of Adjuvant-Induced Autoimmunity.","authors":"Julia Morimoto, Yasuhito Suzuki, Ryuichi Togawa, Natsumi Watanabe, Takahiro Kumanaka, Ryutaro Tanaka, Kentaro Kazama, Koshi Saito, Rina Harigane, Ryuki Yamada, Riko Sato, Hikaru Tomita, Takashi Umeda, Mami Rikimaru, Yuki Sato, Hiroyuki Minemura, Takefumi Nikaido, Junpei Saito, Kenya Kanazawa, Xintao Wang, Yoshinori Tanino, Yoko Shibata","doi":"10.12659/AJCR.947856","DOIUrl":"https://doi.org/10.12659/AJCR.947856","url":null,"abstract":"<p><p>BACKGROUND Silicosis, which is caused by the inhalation of crystalline silica, is known to be associated with a variety of autoimmune diseases. Recently, a new pathogenesis called autoimmune/inflammatory syndrome induced by adjuvants (ASIA) has been reported, which occurs after exposure to substances with adjuvant activity, including silica, and shares clinical features observed in several autoimmune diseases. CASE REPORT A 65-year-old man with silicosis was admitted to our hospital due to fever and chronic fatigue. Symptoms such as sicca and Raynaud's phenomenon and pleural effusion appeared as new findings on admission. Examination led to a diagnosis of systemic scleroderma (SSc) and Sjögren's syndrome (SjS). Considering that SjS was the main cause of the disease, corticosteroid therapy was initiated. However, the patient's general condition deteriorated, leading finally to his death. Silica acts as an adjuvant, inducing chronic inflammatory cytokine release. Thus, prolonged exposure to silica can contribute to the development of autoimmune diseases such as SSc and SjS. In this case, the refractory pleuritis may have been related to the pathogenesis of ASIA. ASIA is difficult to manage if the causative adjuvant cannot be eliminated. CONCLUSIONS We described a case of newly diagnosed SSc and SjS with therapy-resistant pleuritis in a patient with silicosis. Silicosis complicated with corticosteroid-resistant autoimmune disease suggests that ASIA, an adjuvant disease, is involved in the pathogenesis. Therefore, not only SSc and SjS but also the pathogenesis of ASIA should be considered in such cases. Since adjuvant exposure is a causative factor in ASIA, avoiding such exposure is crucial.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e947856"},"PeriodicalIF":1.0,"publicationDate":"2025-05-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12060736/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144016346","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}