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Acute meningoencephalomyelitis in an exacerbation of primary Sjögren's syndrome. 急性脑膜脑脊髓炎在原发性Sjögren综合征的恶化。
IF 0.6
BMJ Case Reports Pub Date : 2025-09-28 DOI: 10.1136/bcr-2025-267370
Franco Emanuel Appiani, Francesca Sanchez Ato, Pilar Brito-Zerón, Antonio Jesus Donaire Pedraza
{"title":"Acute meningoencephalomyelitis in an exacerbation of primary Sjögren's syndrome.","authors":"Franco Emanuel Appiani, Francesca Sanchez Ato, Pilar Brito-Zerón, Antonio Jesus Donaire Pedraza","doi":"10.1136/bcr-2025-267370","DOIUrl":"https://doi.org/10.1136/bcr-2025-267370","url":null,"abstract":"<p><p>A middle-aged woman with long-standing primary Sjögren's syndrome presented with a 2 month history of persistent headaches that acutely worsened over 5 days, accompanied by new-onset gait instability, bulbar symptoms, renal impairment and a recurrent cutaneous flare in sun-exposed areas. Neurological examination revealed bradyphrenia, flaccid dysarthria, an absent gag reflex, generalised hyperreflexia and axial ataxia. Neuroimaging demonstrated diffuse brainstem and subcortical hyperintensities, pachymeningeal enhancement, arterial calibre changes and a diffusion-restricted lesion. Cerebrospinal fluid analysis revealed mild protein elevation without pleocytosis and negative results on infectious and autoimmune panels. After excluding alternative aetiologies, central nervous system involvement secondary to Sjögren's syndrome was suspected. A 3-days intravenous methylprednisolone bolus was administered, followed by an initial cycle of cyclophosphamide. Neurological improvement was achieved in 10 days. Complete clinical and imaging resolution was observed in 1 month, with stable outcomes at 1 year follow-up. This case emphasises the importance of early recognition of severe Sjögren's manifestations.</p>","PeriodicalId":9080,"journal":{"name":"BMJ Case Reports","volume":"18 9","pages":""},"PeriodicalIF":0.6,"publicationDate":"2025-09-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145190815","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Visualising anterior hip dislocation: contrasting obturator and pubic types. 髋前位脱位的显像：对比闭孔型和耻骨型。
IF 0.6
BMJ Case Reports Pub Date : 2025-09-28 DOI: 10.1136/bcr-2025-267883
Hirokazu Takai, Kengo Hirata, Kensuke Miyazaki, Yuto Nonoue
{"title":"Visualising anterior hip dislocation: contrasting obturator and pubic types.","authors":"Hirokazu Takai, Kengo Hirata, Kensuke Miyazaki, Yuto Nonoue","doi":"10.1136/bcr-2025-267883","DOIUrl":"https://doi.org/10.1136/bcr-2025-267883","url":null,"abstract":"","PeriodicalId":9080,"journal":{"name":"BMJ Case Reports","volume":"18 9","pages":""},"PeriodicalIF":0.6,"publicationDate":"2025-09-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145190928","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Early-onset vasculitis: a toddler with ADA2 deficiency. 早发性血管炎：ADA2缺乏的幼儿。
IF 0.6
BMJ Case Reports Pub Date : 2025-09-28 DOI: 10.1136/bcr-2025-265538
Mai M Abd Elhamed, Nermeen M Galal, Yasser Wali, Ilham Youssry
{"title":"Early-onset vasculitis: a toddler with ADA2 deficiency.","authors":"Mai M Abd Elhamed, Nermeen M Galal, Yasser Wali, Ilham Youssry","doi":"10.1136/bcr-2025-265538","DOIUrl":"https://doi.org/10.1136/bcr-2025-265538","url":null,"abstract":"<p><p>Deficiency of adenosine deaminase 2 (DADA2) is an autoinflammatory disease caused by autosomal recessive inherited loss of function mutations in the adenosine deaminase 2 (ADA2) encoding gene, which reduces plasma ADA2 concentration levels.ADA2 has a major role in the maturation and differentiation of the immune system. It ensures immune homeostasis with the help of adenosine receptors on the cell surface because ADA2 acts as an extracellular enzyme that has an important role in regulating extracellular adenosine concentration and mediates cellular immune interactions. We report a fatal outcome of an infant with DADA2 as a result of delayed diagnosis and definitive treatment with bone marrow transplantation.</p>","PeriodicalId":9080,"journal":{"name":"BMJ Case Reports","volume":"18 9","pages":""},"PeriodicalIF":0.6,"publicationDate":"2025-09-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145190836","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Recovery from immobilisation-associated osteoporosis with anti-resorptive and anabolic therapy. 通过抗吸收和合成代谢治疗从固定相关骨质疏松症中恢复。
IF 0.6
BMJ Case Reports Pub Date : 2025-09-28 DOI: 10.1136/bcr-2025-267469
Maryell Urroz Lopez, Gregory Kline
{"title":"Recovery from immobilisation-associated osteoporosis with anti-resorptive and anabolic therapy.","authors":"Maryell Urroz Lopez, Gregory Kline","doi":"10.1136/bcr-2025-267469","DOIUrl":"https://doi.org/10.1136/bcr-2025-267469","url":null,"abstract":"<p><p>The detrimental effects of immobilisation on bone health are well known and have been studied primarily in inpatient and long-term care settings. We describe a case seen in the osteoporosis clinic involving a patient with profound bone loss and hypercalcaemia secondary to chronic immobilisation in the setting of a debilitating systemic inflammatory myopathy. While undergoing treatment with bisphosphonate, her hypercalcaemia resolved, and no further bone loss was found based on bone turnover markers. Treatment of the underlying myopathy permitted significant recovery and return to ambulation, but with persistent very low bone density. To try to regain the lost bone mass, she was started on romosozumab. After 1 year of treatment, repeated measurements of bone mineral density showed an increase of 6% in her lumbar spine and 11% in her hip, respectively. Romosozumab use showed a significant recovery of nearly half of her original bone loss after her initial treatment with bisphosphonates.</p>","PeriodicalId":9080,"journal":{"name":"BMJ Case Reports","volume":"18 9","pages":""},"PeriodicalIF":0.6,"publicationDate":"2025-09-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145190845","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Tuberculous monoarthritis in a rituximab-treated patient with rheumatoid arthritis: diagnostic and therapeutic challenges in a tuberculosis-endemic region. 利妥昔单抗治疗的类风湿性关节炎患者的结核性单关节炎：结核病流行地区的诊断和治疗挑战。
IF 0.6
BMJ Case Reports Pub Date : 2025-09-28 DOI: 10.1136/bcr-2025-267999
Joydeep Samanta, Jhasaketan Meher, Vinay Pandit
{"title":"Tuberculous monoarthritis in a rituximab-treated patient with rheumatoid arthritis: diagnostic and therapeutic challenges in a tuberculosis-endemic region.","authors":"Joydeep Samanta, Jhasaketan Meher, Vinay Pandit","doi":"10.1136/bcr-2025-267999","DOIUrl":"https://doi.org/10.1136/bcr-2025-267999","url":null,"abstract":"<p><p>A woman in her 60s with seropositive rheumatoid arthritis developed tuberculous arthritis of the wrist following rituximab therapy, despite negative latent tuberculosis (TB) screening. Although rituximab is considered safer than anti-TNF agents in terms of TB risk, this case highlights a rare yet serious complication. Diagnosis was confirmed via synovial fluid analysis after persistent monarthritis unresponsive to standard RA therapy. Management was complicated by drug-induced liver injury and ototoxicity, leading to multiple modifications of anti-tubercular therapy. The therapeutic dilemma centred on balancing effective TB treatment with drug tolerability, necessitating a cautious, stepwise reintroduction of first-line agents. Despite these challenges, the patient achieved full recovery and later resumed low-dose methotrexate for RA. This case underscores the importance of considering TB even with negative screening and demonstrates that individualised therapy can overcome complex treatment barriers in immunosuppressed patients in TB-endemic settings.</p>","PeriodicalId":9080,"journal":{"name":"BMJ Case Reports","volume":"18 9","pages":""},"PeriodicalIF":0.6,"publicationDate":"2025-09-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145190914","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Percutaneous caecostomy in the management of malignant large bowel obstruction. 经皮结肠切除术治疗恶性大肠梗阻。
IF 0.6
BMJ Case Reports Pub Date : 2025-09-28 DOI: 10.1136/bcr-2025-265986
Anek Jena, Victoria V Barbee, Daniel Kashani, Karthik Gnanapandithan
{"title":"Percutaneous caecostomy in the management of malignant large bowel obstruction.","authors":"Anek Jena, Victoria V Barbee, Daniel Kashani, Karthik Gnanapandithan","doi":"10.1136/bcr-2025-265986","DOIUrl":"https://doi.org/10.1136/bcr-2025-265986","url":null,"abstract":"","PeriodicalId":9080,"journal":{"name":"BMJ Case Reports","volume":"18 9","pages":""},"PeriodicalIF":0.6,"publicationDate":"2025-09-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145190808","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Rare tumour presenting as a cystic lesion in an infant. 在婴儿中表现为囊性病变的罕见肿瘤。
IF 0.6
BMJ Case Reports Pub Date : 2025-09-28 DOI: 10.1136/bcr-2025-267276
Jonathan M Stern, Gurinder Kumar, Aparna Roy, Timothy Pittinger
{"title":"Rare tumour presenting as a cystic lesion in an infant.","authors":"Jonathan M Stern, Gurinder Kumar, Aparna Roy, Timothy Pittinger","doi":"10.1136/bcr-2025-267276","DOIUrl":"https://doi.org/10.1136/bcr-2025-267276","url":null,"abstract":"<p><p>A previously healthy male infant presented with a painless skin-coloured subcutaneous upper midline back mass overlying the spine. Initial differential diagnosis included dermoid cyst and pilomatrixoma. He underwent wide excision, with histopathology confirming a plexiform fibrohistiocytic tumour with positive margins. Re-excision achieved clear margins. This case highlights a rare presentation and emphasises the importance of including this tumour in the differential diagnosis of paediatric cystic masses.</p>","PeriodicalId":9080,"journal":{"name":"BMJ Case Reports","volume":"18 9","pages":""},"PeriodicalIF":0.6,"publicationDate":"2025-09-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145190816","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Pulse deficits resulting in apparent bradycardia in atrial bigeminy. 房颤患者脉搏不足导致明显的心动过缓。
IF 0.6
BMJ Case Reports Pub Date : 2025-09-28 DOI: 10.1136/bcr-2025-267102
Andrew Wilaras, Phillip Minh Tien Karpati, Christopher Tan
{"title":"Pulse deficits resulting in apparent bradycardia in atrial bigeminy.","authors":"Andrew Wilaras, Phillip Minh Tien Karpati, Christopher Tan","doi":"10.1136/bcr-2025-267102","DOIUrl":"https://doi.org/10.1136/bcr-2025-267102","url":null,"abstract":"<p><p>Supraventricular premature beats are premature activations of the atria outside of the sinus node that is often benign. They typically present with irregular pulses and dynamic changes to pre-existing murmurs between sinus beats and ectopic beats, but rarely with bradycardia. We describe an elderly male with atrial bigeminy who presented for evacuation of a soft tissue collection in a regional hospital. On examination, he appeared bradycardic due to a significant pulse deficit, despite remaining haemodynamically stable and asymptomatic. This led to two emergency response teams being called and a brief admission to coronary care unit (CCU), primarily due to the ward's limited access to telemetry, before he underwent an uneventful surgery and was discharged. This case highlights the logistical challenges of monitoring such patients where telemetry is not readily available and emphasises the importance of recognising pulse deficits as a cause of peripheral bradycardia in otherwise stable patients with supraventricular ectopic beats.</p>","PeriodicalId":9080,"journal":{"name":"BMJ Case Reports","volume":"18 9","pages":""},"PeriodicalIF":0.6,"publicationDate":"2025-09-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145190821","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Aberrant fibromuscular common atrial band in right isomerism. 右同分异构体纤维肌性心房总带异常。
IF 0.6
BMJ Case Reports Pub Date : 2025-09-28 DOI: 10.1136/bcr-2025-268309
Damandeep Singh, Aprateem Mukherjee, Lamk Kadiyani, Niraj Nirmal Pandey
{"title":"Aberrant fibromuscular common atrial band in right isomerism.","authors":"Damandeep Singh, Aprateem Mukherjee, Lamk Kadiyani, Niraj Nirmal Pandey","doi":"10.1136/bcr-2025-268309","DOIUrl":"https://doi.org/10.1136/bcr-2025-268309","url":null,"abstract":"","PeriodicalId":9080,"journal":{"name":"BMJ Case Reports","volume":"18 9","pages":""},"PeriodicalIF":0.6,"publicationDate":"2025-09-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145190820","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Elizabethkingia miricola mediastinitis and bacteraemia. 伊丽莎白分枝杆菌纵隔炎和菌血症。
IF 0.6
BMJ Case Reports Pub Date : 2025-09-28 DOI: 10.1136/bcr-2025-267000
Grace Rothwell-Kelly, Clarice Egan, Kirsten Schaffer
{"title":"<i>Elizabethkingia miricola</i> mediastinitis and bacteraemia.","authors":"Grace Rothwell-Kelly, Clarice Egan, Kirsten Schaffer","doi":"10.1136/bcr-2025-267000","DOIUrl":"10.1136/bcr-2025-267000","url":null,"abstract":"<p><p><i>Elizabethkingia</i> species, including <i>Elizabethkingia miricola</i>, are rare opportunistic Gram-negative bacteria. We present a case of <i>E. miricola</i> bloodstream infection associated with a polymicrobial mediastinitis. A male in his 70s underwent an elective Nissen fundoplication, deteriorated on day one post surgery and was transferred to the intensive care unit. Blood cultures revealed <i>E. miricola</i> A CT scan revealed an air and fluid collection posterior to the heart, moderate bilateral pleural effusions, an extensive pneumomediastinum, pockets of free air within the upper abdomen and a splenic infarct. A mediastinal drain and pleural drains were inserted, which cultured <i>E. miricola</i>, <i>Candida albicans, Staphylococcus aureus</i> and anaerobes. The patient was treated with piperacillin-tazobactam and fluconazole with a good response. <i>E. miricola</i> is intrinsically resistant to multiple antibiotic classes, including carbapenems and aminoglycosides. The lack of published cases combined with resistance makes management challenging. Our case highlighted the need for a combination of antibiotic therapy and source control.</p>","PeriodicalId":9080,"journal":{"name":"BMJ Case Reports","volume":"18 9","pages":""},"PeriodicalIF":0.6,"publicationDate":"2025-09-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12481183/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145190823","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
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