American Journal of Case Reports最新文献_第10页

Surgical Management of Pulmonary Artery Sling in a Pediatric Patient 儿科患者肺动脉吊带的手术治疗

IF 1

American Journal of Case Reports Pub Date : 2024-07-01 DOI: 10.12659/ajcr.944683

Haiyuan Liu, Xiaofan Yang, Shuaipeng Zhang, Yang Li, Wenpeng Dong

{"title":"Surgical Management of Pulmonary Artery Sling in a Pediatric Patient","authors":"Haiyuan Liu, Xiaofan Yang, Shuaipeng Zhang, Yang Li, Wenpeng Dong","doi":"10.12659/ajcr.944683","DOIUrl":"https://doi.org/10.12659/ajcr.944683","url":null,"abstract":"Pulmonary artery sling (PAS) is an anatomical vascular anomaly due to the origin of the left pulmonary artery from the right pulmonary artery, which runs posteriorly between the esophagus and trachea, resulting in compression of adjacent structures. Accurate evaluation for malformation of the pulmonary artery and severity of airway obstruction is essential to surgical strategy. This report presents the diagnosis and surgical management of pulmonary artery sling in a 12-year-old boy. Case Report: A 12-year-old boy had chest tightness and wheezing after exercise for 6 years. He was diagnosed with PSA based on findings from imaging tests, demonstrating the left pulmonary artery originated from the middle of the right pulmonary artery and the tracheal carina was located at the site of the T6 thoracic vertebra. The main bronchus and esophagus were compressed by the left pulmonary artery due to its ectopic origin. Then, after comprehensive preoperative assessment, the patient underwent surgical repair of PAS. Conclusions: This report highlights the importance of pulmonary artery sling diagnosis, imaging, and surgical planning, and the role of a multidisciplinary team in preoperative and postoperative patient management. An individualized strategy based on the preoperative assessment, intraoperative coordination among cardiologists, surgeons, and perfusionists, and careful postoperative management are the core elements for successful PAS repair.","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":null,"pages":null},"PeriodicalIF":1.0,"publicationDate":"2024-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141698563","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

Voriconazole-Induced Periostitis: A Mimicker of Skeletal Coccidioidomycosis 伏立康唑诱发的骨膜炎：骨骼球孢子菌病的模仿者

IF 1

American Journal of Case Reports Pub Date : 2024-07-01 DOI: 10.12659/ajcr.944102

Priyal J Shah, Janis Blair

引用次数: 0

Virtual Reality-Guided Simulation for Percutaneous Coronary Intervention in a Patient with Anatomical Anomalies: A Case Report. 虚拟现实引导模拟对解剖异常患者进行经皮冠状动脉介入治疗：病例报告。

IF 1

American Journal of Case Reports Pub Date : 2024-06-19 DOI: 10.12659/AJCR.944485

Jun Goto, Takeshi Niizeki, Tadateru Iwayama, Toshiki Sasaki, Hirooki Higami, Masafumi Watanabe

{"title":"Virtual Reality-Guided Simulation for Percutaneous Coronary Intervention in a Patient with Anatomical Anomalies: A Case Report.","authors":"Jun Goto, Takeshi Niizeki, Tadateru Iwayama, Toshiki Sasaki, Hirooki Higami, Masafumi Watanabe","doi":"10.12659/AJCR.944485","DOIUrl":"10.12659/AJCR.944485","url":null,"abstract":"BACKGROUND Virtual reality (VR)-guided GC simulation for patients with anatomical anomalies using cardiac computed tomography (CT) has been recently reported. Rotational atherectomy (RA) for the left circumflex (LCX) ostium is challenging due to the tortuous anatomy, acute angulation, and variable vessel size compared to other lesions. The appropriate positioning and coaxiality of the guide catheter (GC) are key factors for safely performing RA. It would be beneficial if it could be simulated prior to percutaneous coronary intervention (PCI). CASE REPORT We treated a 55-year-old man with angina. We performed coronary angiography and detected an ostial calcified lesion of the LCX. We needed RA for this lesion, but PCI was very difficult and challenging. CT revealed right-sided aortic arch with stenosis of left subclavian artery from the Kommerell diverticulum at the distal part of the aortic arch. Therefore, the approach site for PCI was limited. We simulated the appropriate guide catheter and approach site for PCI by VR. PCI was successfully performed with RA, as in the VR simulation. CONCLUSIONS We successfully performed PCI for an ostial calcified lesion of the LCX in a patient with a right-sided aortic arch. Use of VR-guided GC simulation is a useful new option that can help visualize the anatomy and ensure safe procedures for complex lesions.","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":null,"pages":null},"PeriodicalIF":1.0,"publicationDate":"2024-06-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11334098/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141421343","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

Smoking-Associated Endotracheal Hair Growth: A Case Report on Tracheal Complications. 与吸烟相关的气管内毛发生长：气管并发症病例报告。

IF 1

American Journal of Case Reports Pub Date : 2024-06-18 DOI: 10.12659/AJCR.943909

Paul Thöne, Roland Kropfmüller, Daniela Gompelmann, Bernd Lamprecht, David Lang

{"title":"Smoking-Associated Endotracheal Hair Growth: A Case Report on Tracheal Complications.","authors":"Paul Thöne, Roland Kropfmüller, Daniela Gompelmann, Bernd Lamprecht, David Lang","doi":"10.12659/AJCR.943909","DOIUrl":"10.12659/AJCR.943909","url":null,"abstract":"BACKGROUND A 52-year-old male patient presented with symptoms of chronic cough and persistent tracheal irritation 26 years after surgical closure of a tracheostoma, supported by an autologous auricular cartilage graft and cutaneous transplant. At the initial clinical presentation, the patient was an active smoker, with a cumulative dose of 31 pack years. CASE REPORT Bronchoscopy revealed endotracheal hair growth and local inflammation at the graft site. Initial anti-inflammatory, antimycotic, and antibacterial therapy was administered, followed by endoscopic structure remodeling. There were multiple recurrences with similar symptoms, showing isolated hair growth, without inflammation. Annual endoscopic restructuring sessions were indicated, and the patient experienced them as highly relieving. Recurrent hair growth was finally terminated by argon plasma laser-coagulation and after smoking cessation. We hypothesize that the onset of hair growth was triggered by the patient's cigarette smoking. CONCLUSIONS Endotracheal hair growth is a potential complication of autograft-supported tracheal restructuring. The initial administration of antimicrobial and anti-inflammatory medication, combined with endoscopic restructuring, could have contained the active inflammation; the application of argon plasma laser-coagulation finally stopped the hair growth. Smoking is associated with the upregulation of molecular signaling pathways in the respiratory epithelium, which can stimulate hair follicles, such as sonic hedgehog protein, WNT-1/ß-catenin, and epidermal growth factor receptor.","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":null,"pages":null},"PeriodicalIF":1.0,"publicationDate":"2024-06-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11196209/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141421342","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

Extended Left Hemihapatectomy with Right Hepatic Artery Reconstruction for Primary Hepatic Neuroendocrine Neoplasm: A Brief Report. 原发性肝脏神经内分泌肿瘤的扩展左半肝切除术与右肝动脉重建术：简要报告。

IF 1.2

American Journal of Case Reports Pub Date : 2024-06-18 DOI: 10.12659/AJCR.943721

Yin Jiang, Joseph Mugaanyi, Shi Wei Zhang, Gao Qing Wang, Yong Fei Hua, Ye-Ming Zhou, Caide Lu

{"title":"Extended Left Hemihapatectomy with Right Hepatic Artery Reconstruction for Primary Hepatic Neuroendocrine Neoplasm: A Brief Report.","authors":"Yin Jiang, Joseph Mugaanyi, Shi Wei Zhang, Gao Qing Wang, Yong Fei Hua, Ye-Ming Zhou, Caide Lu","doi":"10.12659/AJCR.943721","DOIUrl":"https://doi.org/10.12659/AJCR.943721","url":null,"abstract":"BACKGROUND rimary hepatic neuroendocrine neoplasms (PHNEN) are exceedingly rare tumors with atypical clinical manifestations, accounting for less than 0.5% of all neuroendocrine tumors. Currently, there is a lack of consensus on their management, and guidelines do not recommend postoperative chemotherapy for patients with stage G1/G2 disease after curative resection. We present a case report of PHNEN, outlining its diagnostic challenges, treatment strategy, and clinical outcomes. CASE REPORT A 31-year-old man presented with jaundice and was initially diagnosed with suspected IgG4-related disease, which initially appeared to respond to steroid therapy, but manifested worsening jaundice 4 months after initial treatment. Subsequent evaluation revealed a PHNEN NET G2 with lymph node metastasis and invasion of the right hepatic artery; and involvement of the hepatic duct at the hepatic hilum, primarily the left hepatic duct. The patient underwent extended left hemi-hepatectomy with caudate lobe resection, bile duct resection, and lymphadenectomy, followed by reconstruction of the right hepatic artery. Postoperatively, the patient received adjuvant chemotherapy consisting of capecitabine (1000 mg bid D1-14) and temozolomide (200 mg qn D10-14) for 6 cycles. Currently, the patient remains disease free 43 months after treatment. CONCLUSIONS PHNEN presents diagnostic challenges due to its rarity and lack of specific markers. Surgical resection remains the cornerstone of treatment, with chemotherapy being considered in select cases with high-risk features. Further research is needed to refine treatment approaches and improve outcomes for patients with PHNEN.","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":null,"pages":null},"PeriodicalIF":1.2,"publicationDate":"2024-06-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141421341","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

Genome Sequencing Analysis of a Rare Case of Blood Infection Caused by Flavonifractor plautii. 黄癣菌引起的罕见血液感染病例的基因组测序分析

IF 1

American Journal of Case Reports Pub Date : 2024-06-17 DOI: 10.12659/AJCR.943920

Xingying Chen, Wei Bi, Xinyi Ruan, Limin Jin, Nenghua Zhang

{"title":"Genome Sequencing Analysis of a Rare Case of Blood Infection Caused by Flavonifractor plautii.","authors":"Xingying Chen, Wei Bi, Xinyi Ruan, Limin Jin, Nenghua Zhang","doi":"10.12659/AJCR.943920","DOIUrl":"10.12659/AJCR.943920","url":null,"abstract":"BACKGROUND Flavonifractor plautii belongs to the clostridium family, which can lead to local infections as well as the bloodstream infections. Flavonifractor plautii caused infection is rarely few in the clinic. To understand better Flavonifractor plautii, we investigated the drug sensitivity and perform genome sequencing of Flavonifractor plautii isolated from blood samples in China and explored the drug resistance and pathogenic mechanism of the bacteria. CASE REPORT The Epsilometer test method was used to detect the sensitivity of flavonoid bacteria to antimicrobial agents. PacBio sequencing technology was employed to sequence the whole genome of Flavonifractor plautii, and gene prediction and functional annotation were also analyzed. Flavonifractor plautii displayed sensitivity to most drugs but resistance to fluoroquinolones and tetracycline, potentially mediated by tet (W/N/W). The total genome size of Flavonifractor plautii was 4,573,303 bp, and the GC content was 59.78%. Genome prediction identified 4,506 open reading frames, including 9 ribosomal RNAs and 66 transfer RNAs. It was detected that the main virulence factor-coding genes of the bacteria were the capsule, polar flagella and FbpABC, which may be associated with bacterial movement, adhesion, and biofilm formation. CONCLUSIONS The results of whole-genome sequencing could provide relevant information about the drug resistance mechanism and pathogenic mechanism of bacteria and offer a basis for clinical diagnosis and treatment.","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":null,"pages":null},"PeriodicalIF":1.0,"publicationDate":"2024-06-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11196211/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141332097","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

Bartter Syndrome Presenting as Arginine-Vasopressin Resistance: A Report of 2 Cases. 表现为精氨酸-加压素抵抗的巴特综合征：2 例报告

IF 1

American Journal of Case Reports Pub Date : 2024-06-17 DOI: 10.12659/AJCR.942872

Maria Sousa, Regina Medeiros, Ana Luísa Rodrigues, Bernardo Dias Pereira

{"title":"Bartter Syndrome Presenting as Arginine-Vasopressin Resistance: A Report of 2 Cases.","authors":"Maria Sousa, Regina Medeiros, Ana Luísa Rodrigues, Bernardo Dias Pereira","doi":"10.12659/AJCR.942872","DOIUrl":"10.12659/AJCR.942872","url":null,"abstract":"BACKGROUND Bartter syndrome is a rare, inherited salt-wasting tubulopathy caused by mutations in 1 of 6 genes that express ion transport channels in the thick ascending limb of nephrons. Excessive prostaglandin E2 and associated hyperreninemic hyperaldosteronism occurs, causing polyhydramnios, polyuria, prematurity, failure to thrive, and characteristic physical features. Hypokalemia, hypochloremic metabolic alkalosis, and, depending on the affected gene, hypercalciuria and nephrocalcinosis are hallmarks of Bartter syndrome. CASE REPORT A 9-month-old male infant, born prematurely due to polyhydramnios, presented in the Emergency Department with dehydration due to incoercible vomiting and significant polyuria. A 6-year-old male infant with a previous history of prematurity due to polyhydramnios was referred to the Pediatric Endocrinology Department due to short stature and notable polydipsia and polyuria. Considering these marked symptoms, both cases triggered suspicion and started workup for arginine-vasopressin insufficiency/resistance. However, during the investigations, a broader clinical revision revealed that both had dysmorphic physical features (triangularly shaped face, prominent forehead, protruding ears, drooping mouth), poor growth, impaired weight gain, and typical biochemical findings (hypokalemic metabolic alkalosis, hypercalciuria, secondary hyperaldosteronism) of Bartter syndrome. Genetic testing confirmed the diagnosis of Bartter syndrome types 1 and type 2, respectively, and this diagnosis allowed proper treatment and significant clinical improvements, personalized follow-up, and genetic counseling for parents desiring further healthy pregnancies. CONCLUSIONS Here, we present clinical and follow-up findings of 2 patients with Bartter syndrome types 1 and 2 discovered upon a broader clinical revision of suspected arginine-vasopressin insufficiency/resistance. We also review pertinent data on diagnosis and management of this challenging syndrome.","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":null,"pages":null},"PeriodicalIF":1.0,"publicationDate":"2024-06-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11196210/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141421340","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

Sinus Node Dysfunction Triggered by Tonsillar Abscess: Effects of Vagal Nerve Compression. 扁桃体脓肿引发的窦房结功能障碍：迷走神经压迫的影响。

IF 1

American Journal of Case Reports Pub Date : 2024-06-16 DOI: 10.12659/AJCR.943944

Howard Yu, Sahil Zaveri, Michael Schaible, Nabeel Butt, Said Tfaili, Adam S Budzikowski

{"title":"Sinus Node Dysfunction Triggered by Tonsillar Abscess: Effects of Vagal Nerve Compression.","authors":"Howard Yu, Sahil Zaveri, Michael Schaible, Nabeel Butt, Said Tfaili, Adam S Budzikowski","doi":"10.12659/AJCR.943944","DOIUrl":"10.12659/AJCR.943944","url":null,"abstract":"BACKGROUND Compression of the vagus nerve by a pharyngeal mass is a well-documented condition that can result in sinus node dysfunction (SND). However, there is scarce literature on extrinsic vagal nerve compression from a tonsillar abscess. CASE REPORT A 59-year-old woman with a history of asthma and chronic throat discomfort presented to the Emergency Department with bradycardia, palpitations, and voice changes. Following a shellfish allergy hospitalization, an otolaryngology evaluation revealed an enlarged right tonsil, recommending tonsillectomy, but scheduling challenges persisted. The patient reported mild throat pain, dysphagia, hoarseness, rhinorrhea, and exertional dyspnea and was admitted for the evaluation of peritonsillar mass. She was found to be bradycardic with a heart rate of 47, with an electrocardiogram revealing SND. Albuterol and ipratropium nebulizers, as well as dexamethasone and pantoprazole, were initiated. With this treatment, the patient symptomatically improved with a new heart rate of 68. She was discharged with outpatient appointments, but was unfortunately lost to follow-up. CONCLUSIONS This case reveals sinus node dysfunction resulting from extrinsic vagal nerve compression by a tonsillar abscess. Pressure on the vagus nerve can trigger bradycardia and low blood pressure, possibly due to compensatory overfiring of afferent vagal nerve signals from local mass effect. Early recognition and antibiotic treatment are essential to prevent cardiac complications. Clinicians must remain vigilant for such extrinsic causes, particularly in patients with chronic sore throat and cardiac symptoms. Further research and case reports are needed to deepen our understanding of this rare yet significant association.","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":null,"pages":null},"PeriodicalIF":1.0,"publicationDate":"2024-06-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11193845/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141327968","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

Lung Lesions: Differential Diagnoses Beyond Cancer. 肺部病变：癌症之外的鉴别诊断。

IF 1

American Journal of Case Reports Pub Date : 2024-06-15 DOI: 10.12659/AJCR.943798

Paweł Jurczak, Anna Romaszko-Wojtowicz, Anna Doboszyńska

{"title":"Lung Lesions: Differential Diagnoses Beyond Cancer.","authors":"Paweł Jurczak, Anna Romaszko-Wojtowicz, Anna Doboszyńska","doi":"10.12659/AJCR.943798","DOIUrl":"10.12659/AJCR.943798","url":null,"abstract":"BACKGROUND Lung cancer is the most common malignant neoplasm diagnosed worldwide. Early diagnosis and treatment are of great importance for patient's prognosis. A wide variety of pulmonary conditions display clinical and radiological presentation similar to that of lung cancer, and the awareness of their existence can help in making correct diagnoses. CASE REPORT This article presents a description of 4 patients with an insidious type of lesions mimicking pulmonary carcinomas. The first patient was referred to Department with a tumor-like lesion in the right lung. After CT of the chest and core-needle biopsy, the lesion turned out to be an ectopic thyroid tissue. The second patient reported a dry cough and weight loss. A lung nodule mass was revealed in chest CT and the patient was diagnosed with pulmonary tuberculoma. The remaining 2 patients, despite the suspicion of lung cancer, were subsequently diagnosed with a post-traumatic pleural hematoma and diffuse large B cell lymphoma. CONCLUSIONS Low-dose computed tomography of the chest plays a significant role in the diagnosis of newly detected lesions in the lungs. However, due to the similarity of the image of cancer to that of other diseases, the ultimate diagnosis should be based on the interpretation of full imaging diagnostic tests, clinical presentation, and histopathological examination of the material obtained from the lesion. Analysis of cases enables us to expand our understanding of the diseases that need to be considered in differential diagnosis of a patient with a detected tumor-like lesion in the lungs.","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":null,"pages":null},"PeriodicalIF":1.0,"publicationDate":"2024-06-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11193846/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141321767","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

Morbihan Disease: A Rare Case of Periorbital Bilateral Edema with Histopathological Findings of Chronic Inflammation and Demodex Localization. 莫尔比昂病：一例罕见的眶周双侧水肿病例，组织病理学发现为慢性炎症和脱霉菌定位。

IF 1

American Journal of Case Reports Pub Date : 2024-06-14 DOI: 10.12659/AJCR.943421

Alessandra Di Maria, Filippo Confalonieri, Gianmaria Barone, Sofia Manara, Vanessa Ferraro

{"title":"Morbihan Disease: A Rare Case of Periorbital Bilateral Edema with Histopathological Findings of Chronic Inflammation and Demodex Localization.","authors":"Alessandra Di Maria, Filippo Confalonieri, Gianmaria Barone, Sofia Manara, Vanessa Ferraro","doi":"10.12659/AJCR.943421","DOIUrl":"10.12659/AJCR.943421","url":null,"abstract":"BACKGROUND Morbihan disease, also known as Morbihan syndrome, is a rare medical condition characterized by chronic facial edema predominantly affecting the upper two-thirds of the face. Despite being recognized in medical literature for decades, its true prevalence and underlying pathophysiology remain poorly understood. Various hypotheses, including impaired lymphatic drainage, abnormal vascular permeability, immune dysregulation, and inflammatory reactions to demodex infestation, have been proposed to explain the etiology. CASE REPORT We present a case of a 61-year-old man with organized periocular edema of the upper third of the face, ultimately leading to Morbihan disease diagnosis. The patient underwent a midface lift, allowing for tissue retrieval for histopathological examination of the eyelid edematous skin, which revealed chronic inflammation, ectasia of small lymphatic vessels, and features of demodex intrafollicular localization. These findings were not specific, but consistent with the diagnostic hypothesis. The patient was referred to a rheumatologist for further evaluation and treatment. He did not respond well to systemic corticosteroids and immunosuppressive therapy. Rather, this resulted in extension of the edema to the upper eyelid. The patient opted not to undergo further treatment. CONCLUSIONS Morbihan disease is often misdiagnosed due to its rarity and overlapping clinical features with other facial conditions. Its management is challenging and can require a combination of medical and surgical interventions. Systemic corticosteroids, immunosuppressive agents, and topical treatments have had varying success. Surgical procedures, such as blepharoplasty or laser therapy, can be considered in severe cases. Early recognition and appropriate management are crucial to improving patient outcomes and quality of life.","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":null,"pages":null},"PeriodicalIF":1.0,"publicationDate":"2024-06-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11185837/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141318530","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