SAGE Open Medical Case Reports最新文献

{"title":"Could we preserve reproductive function in complete vaginal agenesis and cervical agenesis? A case report and clinical perspective.","authors":"Alfonsus Zeus Suryawan, Widyani Rachim, Marshal Wenas, Mochamad Rizkar Arev Sukarsa","doi":"10.1177/2050313X251324073","DOIUrl":"10.1177/2050313X251324073","url":null,"abstract":"<p><p>Early studies show hysterectomy is a main surgical choice for cervical agenesis with excellent outcomes. However, as time went on more literature showed successful uterovaginal anastomosis performed in cervical agenesis. This case report wishes to highlight the management of the patient with complete vaginal and cervical agenesis, which presented with acute abdominal pain due to severe extension of the uterus. We aim to explore options and dilemmas in preserving reproductive function in such cases. A 13-year-old child was presented with acute abdominal pain and primary amenorrhea due to suspected vaginal agenesis. Physical examination revealed fundal height equal to 20 weeks of pregnancy. The vagina has no introitus and the external urethral meatus was normal. Abdominal ultrasound revealed a hematometra with the size of 8.84 × 3.12 cm accompanied by hematosalpinx on both fallopian tubes. The patient then undergoes an emergency hysterectomy. During the operation, it was found the cervix was substituted with the muscular part of the uterus with a little part of connective tissue part with no cardinal ligaments. Earlier articles showed hysterectomy is the first line of treatment in cervical agenesis. However, with the advancement of surgical techniques conservative treatment was made possible and reported in several case reports. According to European Society of Human Reproduction and Embryology (ESHRE) classification, our patient is classified as U0-C4-V4 (complete vaginal and cervix agenesis). We opted for a hysterectomy in this patient due possibility of fertility being slim and acute abdominal pain relief. Performing such a difficult operation in our setting requires expert meetings, and tool preparation also an amniotic graft for neovagina which in our case we could not afford due to the acute abdominal pain the patient experienced, and the patient's adamant wish for a hysterectomy. Hysterectomy remains a safer approach in low-resource settings with excellent postoperative outcomes. However, reconstructive surgery is an option that should be offered with its own risks and benefits.</p>","PeriodicalId":21418,"journal":{"name":"SAGE Open Medical Case Reports","volume":"13 ","pages":"2050313X251324073"},"PeriodicalIF":0.6,"publicationDate":"2025-05-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12085751/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144094604","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":"The role of immunotherapy in a refractory case of acute hemorrhagic leukoencephalitis in a pediatric patient.","authors":"Richard Johnson, Mohammad B Khan","doi":"10.1177/2050313X251332459","DOIUrl":"https://doi.org/10.1177/2050313X251332459","url":null,"abstract":"<p><p>Acute hemorrhagic leukoencephalitis (AHLE) is a rare neurologic inflammatory disorder characterized by severe and rapidly progressive encephalopathy; typically considered a severe variant of acute demyelinating encephalomyelitis (ADEM). There is limited pediatric data on treatment modalities for refractory illness. We report here the case of an 8-year-old girl who presented with acute concern for encephalopathy, ataxia, and seizures in the context of preceding infectious symptoms and neuroimaging findings highly suspicious for ADEM. Treatment was promptly initiated with high-dose glucocorticoids followed by addition of intravenous immunoglobulin therapy. Despite ongoing treatment and permissive hypernatremia and hyperosmolar therapy to treat signs of increased intracranial pressure, she showed no clinical signs of improvement, so plasmapheresis was initiated. Follow-up magnetic resonance imaging (MRI) brain showed progression of lesions with more hemorrhagic conversion concerning for AHLE, so anakinra was started. With the combination of pheresis and anakinra, the patient began to show signs of neurologic recovery. Her course was complicated by delirium and physical deconditioning, requiring inpatient rehab, although her neurologic function showed daily improvement. Final MRI brain to date reflected these improvements. Our case highlights the importance of rapid escalation of care to improve mortality rates and reduce neurologic sequelae of AHLE. Future studies are needed to address the safety profile and efficacy of anakinra in refractory disease.</p>","PeriodicalId":21418,"journal":{"name":"SAGE Open Medical Case Reports","volume":"13 ","pages":"2050313X251332459"},"PeriodicalIF":0.6,"publicationDate":"2025-05-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12078970/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144079972","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":"Transient left ventricular function deterioration after patent ductus arteriosus closure: A case series.","authors":"Mohammad Reza Sabri, Chehreh Mahdavi, Hamid Bigdelian, Ali Reza Ahmadi, Mehdi Ghaderian, Bahar Dehghan, Davood Ramezani Nezhad","doi":"10.1177/2050313X251336987","DOIUrl":"https://doi.org/10.1177/2050313X251336987","url":null,"abstract":"<p><p>There is growing evidence of left ventricular decompensation following the closure of the patent ductus arteriosus, which most often happens early in the post-surgical time and improves on its own a few months after. Here, we aim to present nine cases whose left ventricular ejection fraction deteriorated early or late after the procedure. There were nine patients diagnosed with patent ductus arteriosus by echocardiography. The patients presented with a murmur, easy fatigability, exercise intolerance, and poor weight gain. The age varied from 5 months to 41 years old. Patients developed left ventricular ejection fraction deterioration, starting from the day after the operation to 3 years after the closure. Pre-closure left ventricular ejection fraction ranged from 60% to 75% and post-closure from 33% to 59%. The range for shortening fraction (SF) was from 30% to 42% and 16% to 31%, respectively. The patent ductus arteriosus was moderate to large in all patients. One patient was finally diagnosed with dilated cardiomyopathy. Patients with patent ductus arteriosus show left ventricular ejection fraction deterioration up to years after closure, whether percutaneous or operative. Large patent ductus arteriosus is associated with more complications and reduced post-surgery cardiac function due to the amount of the shunt before closure.</p>","PeriodicalId":21418,"journal":{"name":"SAGE Open Medical Case Reports","volume":"13 ","pages":"2050313X251336987"},"PeriodicalIF":0.6,"publicationDate":"2025-05-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12078958/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144079975","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":"Successful treatment of localized scleroderma using brodalumab: A case report.","authors":"Alexandra Carla Bobica, Catherine Zhu, Catherine Silotch, Hessah BinJadeed, Elena Netchiporouk","doi":"10.1177/2050313X251341561","DOIUrl":"https://doi.org/10.1177/2050313X251341561","url":null,"abstract":"<p><p>We report the first documented use of brodalumab, an IL-17 receptor antagonist, for treating severe linear localized scleroderma refractory to conventional therapies, including methotrexate, mycophenolate mofetil and corticosteroids. The patient demonstrated rapid and sustained improvement in disease activity and severity, highlighting the potential role of IL-17 signaling in the pathogenesis of localized scleroderma. This case supports further investigation of IL-17 pathway blockade in autoimmune fibrotic diseases.</p>","PeriodicalId":21418,"journal":{"name":"SAGE Open Medical Case Reports","volume":"13 ","pages":"2050313X251341561"},"PeriodicalIF":0.6,"publicationDate":"2025-05-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12078985/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144079969","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":"Percutaneous stenting as treatment for chylothorax from superior vena cava syndrome: A case report.","authors":"Arta Kasaeian, Taylor Hoffman, Mohammad Ghasemi Rad, David Wynne, David Léon","doi":"10.1177/2050313X251340792","DOIUrl":"https://doi.org/10.1177/2050313X251340792","url":null,"abstract":"<p><p>Superior vena cava syndrome is a condition that results from obstruction of the superior vena cava, the etiology of which can be benign or malignant. The impaired venous return can cause facial and upper extremity swelling, dyspnea, and neurologic symptoms. Chylothorax is a rare complication of superior vena cava syndrome. We report a case of a 69-year-old male with end-stage renal disease, who developed both a chylothorax and a contralateral simple pleural effusion secondary to superior vena cava syndrome. He was successfully treated with percutaneous endovascular stenting.</p>","PeriodicalId":21418,"journal":{"name":"SAGE Open Medical Case Reports","volume":"13 ","pages":"2050313X251340792"},"PeriodicalIF":0.6,"publicationDate":"2025-05-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12078977/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144079955","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":"Lacrimal canaliculitis related to chronic rhinosinusitis with nasal polyps: Case report and literature review.","authors":"Chen Chen, Yan Shen, Yanting Yang, Yuru Gao, Yang Bian","doi":"10.1177/2050313X251340479","DOIUrl":"https://doi.org/10.1177/2050313X251340479","url":null,"abstract":"<p><p>This paper reports an unusual case of recurrent canaliculitis who had a long history of chronic rhinosinusitis with nasal polyps. A 74-year-old woman complained of recurrent redness, purulent discharge, and occasional tearing of the left eye for 1 year. She has a 40-year untreated sinusitis history, with recurrent mucopurulent rhinorrhea, nasal obstruction and hyposmia, and loss of smell for 2 years. The patient's various symptoms, such as epiphora, purulent discharge, rhinorrhea, nasal obstruction, and anosmia, poorly responded to antibiotics and canaliculotomy. However, relief came from the unintentional usage of systemic corticosteroids. Canaliculitis is a rare form of inflammation involving the proximal lacrimal drainage system. Unfortunately, it is often misdiagnosed, has frequent recurrences, and fails to respond to therapy. This case highlights the importance of medical history, lacrimal examinations, and regular therapy in managing recurrent canaliculitis. For those canaliculitis patients with nasal symptoms, simultaneous treatment of nasal-related disorders should be considered.</p>","PeriodicalId":21418,"journal":{"name":"SAGE Open Medical Case Reports","volume":"13 ","pages":"2050313X251340479"},"PeriodicalIF":0.6,"publicationDate":"2025-05-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12075961/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144079952","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":"Holothoracic spinal epidural lipomatosis: Report of a rare presentation and review of literature.","authors":"Maroua Hamadi Ben Salah, Mairi Ziaka","doi":"10.1177/2050313X251339051","DOIUrl":"https://doi.org/10.1177/2050313X251339051","url":null,"abstract":"<p><p>Spinal epidural lipomatosis refers to the abnormal accumulation of unencapsulated fat in the epidural space, pos<i>s</i>ibly associated with spinal canal narrowing and compression of adjacent nerve structures. Risk factors for spinal epidural lipomatosis include older age, male sex, obesity, and systemic corticosteroid use. While spinal epidural lipomatosis typically involves the thoracic or lumbosacral regions of the spinal cord, there are no published cases involving the entire thoracic cord. This report presents the first case of spinal epidural lipomatosis affecting the whole thoracic cord, highlighting the complexity of its diagnosis and management.</p>","PeriodicalId":21418,"journal":{"name":"SAGE Open Medical Case Reports","volume":"13 ","pages":"2050313X251339051"},"PeriodicalIF":0.6,"publicationDate":"2025-05-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12075965/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144079950","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 pediatric actinic prurigo successfully treated with upadacitinib: A case report.","authors":"Florence Morissette, Jérome Coulombe","doi":"10.1177/2050313X251341558","DOIUrl":"https://doi.org/10.1177/2050313X251341558","url":null,"abstract":"<p><p>Actinic prurigo is a rare photodermatosis, usually occurring during childhood. Pruritic papulonodules on sun-exposed areas are frequently associated with cheilitis and conjunctivitis. The most widely accepted pathophysiology to date is a type IV hypersensitivity reaction triggered by ultraviolet radiation. This case report describes a 12-year-old boy suffering from refractory actinic prurigo, who was successfully treated with the janus kinase inhibitor upadacitinib.</p>","PeriodicalId":21418,"journal":{"name":"SAGE Open Medical Case Reports","volume":"13 ","pages":"2050313X251341558"},"PeriodicalIF":0.6,"publicationDate":"2025-05-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12075963/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144079957","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":"Transient neonatal zinc deficiency in a 3-month-old infant: A case report.","authors":"Damy Horth, Isabelle Auger","doi":"10.1177/2050313X251341516","DOIUrl":"https://doi.org/10.1177/2050313X251341516","url":null,"abstract":"<p><p>Zinc deficiency can be inherited, such as in the case of acrodermatitis enteropathica or acquired. In both cases, patients will present typical acral and periorificial skin lesions, and seldom, diarrhea as well as secondary alopecia. In this report, we provide a case of transient neonatal zinc deficiency in a 3-month-old breastfed girl who presented with classical skin lesions of zinc deficiency. The diagnosis was determined following the results of blood tests, which indicated a combination of low plasma phosphatase alkaline and zinc levels. To support the diagnosis, genetic testing was performed on the mother to detect a mutation in the SLC30A2 gene. However, the results were inconclusive as a variant of unknown significance was found. After starting zinc supplementation, the skin lesions completely resolved a few weeks later without any recurrence.</p>","PeriodicalId":21418,"journal":{"name":"SAGE Open Medical Case Reports","volume":"13 ","pages":"2050313X251341516"},"PeriodicalIF":0.6,"publicationDate":"2025-05-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12075959/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144079983","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":"Case of class II lupus nephritis with nephrotic features in the context of belimumab: Intersecting clinical challenge with academic initiative to emphasize social barriers in care.","authors":"Ahyeon Cho, Joshua Shoemaker, Ahmad Matarneh, Sundus Sardar, Pankhuri Mohan, Erik Washburn, Amanda Karasinski, Gurwant Kaur, Nasrollah Ghahramani","doi":"10.1177/2050313X251336061","DOIUrl":"https://doi.org/10.1177/2050313X251336061","url":null,"abstract":"<p><p>Lupus nephritis is a severe complication of systemic lupus erythematosus that can cause significant kidney damage, leading to symptoms such as proteinuria, hematuria, and renal failure. If not managed promptly, it can progress to chronic kidney disease or even end-stage renal disease. Early diagnosis and treatment are vital to prevent such outcomes. While traditional treatments focus on immunosuppressive therapies, the introduction of belimumab offers a new treatment approach. Kidney biopsies play a crucial role in diagnosing and classifying lupus nephritis, helping guide appropriate treatment. Class II lupus nephritis typically requires supportive care, but more severe cases demand aggressive therapy. Noncompliance with treatment can worsen the condition, highlighting the need to address social challenges that impact patient care and adherence. We hereby report a 19-year-old female with systemic lupus erythematosus and nephrotic-range proteinuria, diagnosed with Class II lupus nephritis shortly after initiating belimumab, highlighting the social challenges impacting her care and follow-up. We hereby report a 19-year-old female known with systemic lupus erythematosus with nephrotic-range proteinuria and Class II lupus nephritis. Diagnosed shortly after belimumab, and highlighting the social challenges in her care and follow-up.</p>","PeriodicalId":21418,"journal":{"name":"SAGE Open Medical Case Reports","volume":"13 ","pages":"2050313X251336061"},"PeriodicalIF":0.6,"publicationDate":"2025-05-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12075971/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144079847","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}