International Medical Case Reports Journal最新文献

{"title":"Delayed Intrathoracic Textile Foreign Body Following Penetrating Shrapnel Injury Presenting with Expectoration of Fabric-Like Material and Chronic Middle-Lobe Destruction: A Case Report.","authors":"Mohammad Alaa Aldakak, Hamza Hdeoa, Raneem Ahmad, Youssef Abbas, Yousef Al Jabban, Asmaa Dandashy, Hussain Chaban","doi":"10.2147/IMCRJ.S604561","DOIUrl":"https://doi.org/10.2147/IMCRJ.S604561","url":null,"abstract":"<p><strong>Background: </strong>Retained intrathoracic and tracheobronchial foreign bodies are uncommon but important causes of chronic respiratory symptoms. Diagnosis may be delayed because presentations are nonspecific and retained textile material is often radiolucent. Such material can lead to chronic infection, pleural adhesions, and progressive lung destruction.</p><p><strong>Case presentation: </strong>A 31-year-old Syrian male military serviceman and active smoker (20 pack-years) presented with a long-standing intermittent cough that had worsened over one week and become productive of white sputum. He also reported a single episode of expectorating suspected textile material, along with fatigue, weakness, and night sweats, but no fever, dyspnea, hemoptysis, or chest pain. Years earlier, he had sustained a penetrating shrapnel injury complicated by pneumothorax and later purulent pleural effusion requiring tube thoracostomy. On admission, examination suggested right basal involvement; laboratory studies were unremarkable, and tuberculosis testing was negative. Chest CT showed a heterogeneous right-sided lesion with right middle-lobe atelectatic change and tree-in-bud opacities, suggesting chronic inflammatory or infectious disease. Bronchoscopy revealed friable, bleeding mucosa and distortion at the right middle-lobe bronchial orifice.</p><p><strong>Case discussion: </strong>Due to persistent symptoms and chronic localized abnormalities, right thoracotomy was performed. Dense pleural adhesions were found, and an intrathoracic textile foreign body was removed, most likely implanted during the prior traumatic injury. The right middle lobe was extensively destroyed with purulent change, requiring right middle lobectomy. Histopathology showed fibrosis, architectural destruction, fibroblastic foci, honeycomb change, and chronic inflammation consistent with a UIP pattern; adhesion fragments showed acute-on-chronic foreign body-associated inflammation.</p><p><strong>Conclusion: </strong>Radiolucent textile foreign bodies may remain undetected for years after penetrating thoracic trauma and later present with chronic respiratory symptoms and localized destructive infection. Once irreversible damage is established, thoracotomy with foreign body removal and lobectomy may be necessary.</p>","PeriodicalId":14337,"journal":{"name":"International Medical Case Reports Journal","volume":"19 ","pages":"604561"},"PeriodicalIF":0.7,"publicationDate":"2026-04-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13134679/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147815295","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":"Gastric Adenocarcinoma Presenting as Pseudoachalasia: A Case Report from a Low-Resource Setting.","authors":"Esmail Husein Mohamud, Liban Hussein Ahmed, Ali Ahmed Nor, Abdullahi Ali Muse, Abdirafi Said Elmi","doi":"10.2147/IMCRJ.S603996","DOIUrl":"https://doi.org/10.2147/IMCRJ.S603996","url":null,"abstract":"<p><strong>Background: </strong>Pseudoachalasia is an uncommon cause of dysphagia, often due to malignancy at the gastroesophageal junction (GEJ). It is crucial to distinguish idiopathic achalasia from pseudoachalasia due to tumor infiltrations of the lower esophageal sphincter.</p><p><strong>Aim: </strong>The aim of this case is to highlight the importance of clinicopathological features that distinguish pseudoachalasia from achalasia and the most appropriate diagnostic workup as well as the management challenges in low-resource settings.</p><p><strong>Case: </strong>We present the case of a 76-year-old man with a 40-year smoking history presented with a one-month history of progressive dysphagia, first to solids and then to liquids, associated with epigastric discomfort, regurgitation, early satiety, and 10 kg unintentional weight loss over 4 months. Given the patient's advanced age, alarming GI symptoms, and smoking history, a malignant upper gastrointestinal pathology was highly suspected. Esophagogastroduodenoscopy (EGD) revealed an infiltrative thickened mass at the gastric cardia with reduced distensibility and irregular ulceration on retroflexion, which resulted in subtle difficulty when passing the scope across the GE junction. Computed Tomography (CT) showed circumferential irregular gastric wall thickening involving the gastric cardia, fundus, and body with regional lymphadenopathy, loss of fat plane to the pancreas concerning for invasion, and multiple small hypodense liver lesions. Histopathological examination confirmed the presence of discohesive pleomorphic cells with invasion into the lamina propria with no lymphovascular or perineural invasion was observed in the examined sections, consistent with diffuse-type gastric adenocarcinoma.</p><p><strong>Conclusion: </strong>Diffuse-type gastric adenocarcinoma can present with pseudoachalasia due to GEJ involvement. In older patients with rapid-onset dysphagia and systemic red flags, prompt EGD and cross-sectional imaging are essential. Early recognition is critical but challenging in low-resource settings. Treatment of pseudoachalasia depends on the underlying cause and stage of the disease.</p>","PeriodicalId":14337,"journal":{"name":"International Medical Case Reports Journal","volume":"19 ","pages":"603996"},"PeriodicalIF":0.7,"publicationDate":"2026-04-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13134552/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147815311","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 Report: Resolution of Insomnia in a 32-Year-Old Male with Generalized Anxiety Disorder Following Discontinuation of Mint-Flavored Toothpaste.","authors":"Fahad A Alateeq","doi":"10.2147/IMCRJ.S592710","DOIUrl":"https://doi.org/10.2147/IMCRJ.S592710","url":null,"abstract":"<p><strong>Background: </strong>Insomnia is a common sleep disorder frequently comorbid with anxiety and is often attributed to psychophysiological hyperarousal. While internal and behavioral factors are typically emphasized, environmental sensory exposures are less commonly recognized as potential contributors to sleep disturbance. Menthol, a widely used compound in oral hygiene products, activates transient receptor potential melastatin 8 (TRPM8) receptors, which are known to influence acute sensory and arousal responses. However, their role in chronic sleep disturbance remains unclear.</p><p><strong>Aim: </strong>This case report aims to describe a possible association between nightly menthol exposure from mint-flavored toothpaste and insomnia, and to highlight the potential role of sensory environmental factors in sleep disturbance among susceptible individuals.</p><p><strong>Case presentation: </strong>A 32-year-old male with a history of generalized anxiety disorder presented with a 7-month history of difficulty initiating and maintaining sleep. The onset of insomnia occurred shortly after the patient began using a mint-flavored toothpaste exclusively at night. No other precipitating factors, including medication changes, psychosocial stressors, or poor sleep hygiene, were identified. Following discontinuation of the mint-flavored toothpaste and substitution with a non-mint formulation, the patient experienced rapid and sustained improvement in sleep latency, nighttime awakenings, and overall sleep quality.</p><p><strong>Conclusion: </strong>This case describes a possible association between menthol exposure from mint-flavored toothpaste and insomnia in a patient with anxiety. While causality cannot be established from a single case, the temporal relationship and symptom resolution following discontinuation suggest that menthol-containing products may contribute to sleep disturbances in susceptible individuals. These findings highlight the importance of considering environmental and sensory factors, including menthol exposure, in the evaluation of unexplained insomnia, particularly among individuals with heightened sensory sensitivity. Further research is warranted to explore this potential relationship.</p>","PeriodicalId":14337,"journal":{"name":"International Medical Case Reports Journal","volume":"19 ","pages":"592710"},"PeriodicalIF":0.7,"publicationDate":"2026-04-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13134558/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147815234","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":"Human Coenurosis Caused by <i>Taenia multiceps</i> Mimicking a Thyroglossal Duct Cyst in a Child: A Case Report.","authors":"Charles Malisaba Posite, Henry Wabinga","doi":"10.2147/IMCRJ.S601445","DOIUrl":"https://doi.org/10.2147/IMCRJ.S601445","url":null,"abstract":"<p><strong>Background: </strong>Coenurosis is a rare zoonotic parasitic infection in humans, caused by the larval stage (coenurus) of <i>Multiceps multiceps</i>, typically acquired through ingestion of ova shed in the feces of infected canids. While the central nervous system is the most commonly affected site, extracranial involvement, particularly in the head and neck region, is exceedingly uncommon. In pediatric patients, such presentations may closely mimic congenital cystic lesions, leading to potential delays in diagnosis and management.</p><p><strong>Case presentation: </strong>We present the case of a 4-year-old male from the pastoralist Karamoja region of Northeastern Uganda who presented with a painless, slow-growing midline cystic neck mass of unspecified duration. The mass was mobile, located in the subcutaneous plane superficial to the strap muscles, and lacked communication with the deeper neck structures or oral cavity. Initially presumed to be a thyroglossal duct cyst, the lesion was managed via total surgical excision through a transverse neck incision. Histopathological examination revealed a thin-walled cyst containing multiple protoscolices with characteristic hooklets, consistent with a coenurus cyst caused by <i>Multiceps multiceps</i>. The patient's postoperative recovery was uneventful.</p><p><strong>Conclusion: </strong>This case, the first of its kind reported in Uganda, demonstrates that <i>Multiceps multiceps</i> can clinically mimic a thyroglossal duct cyst. It underscores the necessity of histopathologic evaluation for all pediatric neck masses in endemic regions. Definitive management requires a high index of suspicion for multi-organ involvement, necessitating postoperative systemic anthelmintics and screening for occult central nervous system or ocular disease to ensure comprehensive care.</p>","PeriodicalId":14337,"journal":{"name":"International Medical Case Reports Journal","volume":"19 ","pages":"601445"},"PeriodicalIF":0.7,"publicationDate":"2026-04-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13127446/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147815302","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":"Intraoperative Ventilation Failure Due to a Fenestrated Tracheostomy Tube in a Three-Month-Old Infant with a Laryngeal Mass: A Case Report and Literature Review.","authors":"Mohammad Al Hazaymeh, Khayria Hamid Tahir, Atef Hulliel, Anas Alrusan, Diab Bani Hani","doi":"10.2147/IMCRJ.S598746","DOIUrl":"https://doi.org/10.2147/IMCRJ.S598746","url":null,"abstract":"<p><p>Pediatric tracheostomy is a common procedure, with a substantial proportion of cases performed on infants, often due to chronic respiratory failure or complex airway anomalies. This case report details a critical intraoperative event involving a three-month-old infant undergoing tracheostomy for a laryngeal mass. The patient experienced immediate and profound ventilation failure following the insertion of a fenestrated tracheostomy tube. Mechanical obstruction was ruled out by passing a suction catheter through the tube. Furthermore, switching ventilatory modes to pressure-controlled volume-guaranteed (PC-VG) or volume-controlled ventilation (VCV) was also ineffective in restoring adequate ventilation. This was managed using a completely unfenestrated tube. The tube was placed and the breathing circuit connected, a large portion of the air leak was successfully eliminated, and appropriate ventilation parameters were restored. Fenestrated tracheostomy tubes have established benefits in adults; however, this case highlights a cautionary observation in ventilated infants due to potential air leak and ventilatory failure, underscoring careful tube selection.</p>","PeriodicalId":14337,"journal":{"name":"International Medical Case Reports Journal","volume":"19 ","pages":"598746"},"PeriodicalIF":0.7,"publicationDate":"2026-04-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13118648/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147771432","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":"Ultrasound-Guided Superior Laryngeal Nerve Block Combined with Cricothyroid Membrane Puncture for Awake Tracheal Intubation in a Patient with a Laryngeal Tumor: A Case Report and Literature Review.","authors":"Mingming Chen, Jin Luan, Danqi Chen, Ling Hu, Hui Long","doi":"10.2147/IMCRJ.S589800","DOIUrl":"https://doi.org/10.2147/IMCRJ.S589800","url":null,"abstract":"<p><strong>Background: </strong>Airway management for laryngeal tumor surgery is challenging because conventional induction of general anesthesia may worsen dynamic obstruction, whereas prophylactic tracheostomy is more invasive. We describe a minimally invasive awake airway strategy using ultrasound-guided superior laryngeal nerve block (SLNB) combined with cricothyroid membrane puncture.</p><p><strong>Case presentation: </strong>A 59-year-old man with a laryngeal tumor, hypertension, diabetes mellitus, and coronary artery stenosis underwent successful awake flexible bronchoscopy-guided tracheal intubation after ultrasound-guided bilateral SLNB combined with cricothyroid membrane puncture, with a front-of-neck surgical airway prepared as backup. At predefined peri-intubation time points, systolic/diastolic blood pressure ranged from 131-183/68-102 mmHg, heart rate from 44 to 78 beats/min, respiratory rate from 14 to 22 breaths/min, and SpO₂ from 96% to 100%. No hypoxemia or airway-related complication occurred.</p><p><strong>Conclusion: </strong>In selected patients with high-risk laryngeal tumors, ultrasound-guided SLNB combined with cricothyroid membrane puncture may facilitate awake tracheal intubation while avoiding unnecessary surgical airway creation. This technique may serve as a useful alternative when conventional induction is considered hazardous.</p>","PeriodicalId":14337,"journal":{"name":"International Medical Case Reports Journal","volume":"19 ","pages":"589800"},"PeriodicalIF":0.7,"publicationDate":"2026-04-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13118702/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147771445","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":"Recessive Dystrophic Epidermolysis Bullosa in a Resource-Limited Setting: A Case Report From Mohamed Adam Sheikh Children's Teaching Hospital Hargeisa, Somaliland.","authors":"Mohamed Osman Aw Hashi, Sadam Ismail Ahmed, Ahmed M Derie, Radia Mohamed Abdirahman, Riham Rashid Abdullahi","doi":"10.2147/IMCRJ.S600282","DOIUrl":"https://doi.org/10.2147/IMCRJ.S600282","url":null,"abstract":"<p><strong>Introduction: </strong>Epidermolysis Bullosa (EB) is a rare, genetically determined mechanobullous disorder characterized by profound skin and mucosal fragility. The management of EB is complex, multidisciplinary, and palliative, posing extreme challenges in resource-limited settings where diagnostic capabilities and specialized care are scarce. This report highlights the utility of clinical acumen and the devastating impact of socioeconomic barriers on disease progression.</p><p><strong>Case presentation: </strong>A 4-year-old male from rural Somaliland presented to a tertiary children's hospital with a lifelong history of trauma-induced blistering pseudohypertrophic scarring, and digital contractures. Born to consanguineous parents, who presented with a lifelong history of trauma-induced fragile blisters, erosions, and crustations affecting his limbs, abdomen, and oral mucosa. Examination revealed flaccid bullae, erosions, hemorrhagic crusts, secondary bacterial infection, and disabling finger contractures. A sibling had died in infancy with similar symptoms. A clinical diagnosis of severe, recessive dystrophic EB was made. Diagnostic confirmation via skin biopsy was declined by the caregivers. Management with oral antibiotics, topical wound care, and education was initiated, however, the patient was lost to follow-up due to extreme poverty and geographical isolation. The patient was lost to follow-up after discharge due to socioeconomic barriers and a return to a remote village, highlighting the fragility of care continuity.</p><p><strong>Conclusion: </strong> This case epitomizes the profound challenges of managing a severe genodermatosis within a fragile health system. It underscores the critical reliance on clinical acumen in the absence of diagnostic tools and illustrates how poverty, social disruption, and geographical barriers converge to exacerbate morbidity and preclude long-term management. The report calls for context-specific strategies, including training in basic EB wound care, development of low-cost care protocols, and enhanced support systems for patients and caregivers in underserved regions.</p>","PeriodicalId":14337,"journal":{"name":"International Medical Case Reports Journal","volume":"19 ","pages":"600282"},"PeriodicalIF":0.7,"publicationDate":"2026-04-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13118639/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147771421","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":"Development of <i>Pseudomonas aeruginosa</i> Drug Resistant Due to Prolonged Use of Broad-Spectrum Antibiotics in Severe Viral Infection/Candidemia Cases.","authors":"Masafumi Seki","doi":"10.2147/IMCRJ.S595233","DOIUrl":"https://doi.org/10.2147/IMCRJ.S595233","url":null,"abstract":"<p><p>Pre-multidrug-resistant <i>Pseudomonas aeruginosa</i> (Pre-MDRP) defined as resistance to any two of the three antibiotics, such as usually carbapenems, fluoroquinolones and aminoglycosides, is usually induced by inappropriate use of these broad spectrum antibiotics. We experienced two such cases: Case 1 is a 13-years old female patient with recurrent respiratory syncytial virus (RSV) infection. The laboratory data and chest X-ray findings mimicked the bacterial pneumonia, and tazobactam/piperacillin (TAZ/PIPC), levofloxacin (LVFX), and meropenem (MEPM). However, her condition did not improve, and pre-MDRP was isolated and treated by ciprofloxacin (CPFX). Case 2 is 66 years old male with the history with <i>P. aeruginosa</i> was previously isolated and LVFX was used because bacterial pneumonia was suspected. However, <i>Candida albicans</i> from blood followed by pre-MDRP was isolated from sputum, and he was treated by micafungin and CPFX. The cases suggested that prolonged and/or repeated exposure to anti-pseudomonas agents without appropriate microbial diagnosis and de-escalation of antibiotics might be induce the resistant pathogens and the relevance of pre-MDRP might be an early warning stage in antimicrobial resistance.</p>","PeriodicalId":14337,"journal":{"name":"International Medical Case Reports Journal","volume":"19 ","pages":"595233"},"PeriodicalIF":0.7,"publicationDate":"2026-04-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13110802/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147771366","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":"Early US Prenatal Diagnosis and Serial Imaging Confirmation of Hydrometrocolpos with Uterine Didelphys and Cloacal Malformation: A Rare Case of Combined Müllerian Duct and Anorectal Anomaly.","authors":"Faezeh Soveyzi, Elahe Aleebrahim-Dehkordi, Jamal Jandaghi, Hourieh Soleimani","doi":"10.2147/IMCRJ.S569080","DOIUrl":"https://doi.org/10.2147/IMCRJ.S569080","url":null,"abstract":"<p><strong>Background: </strong>Hydrometrocolpos associated with uterine didelphys and cloacal malformation represents an exceptionally rare prenatal finding. These anomalies arise from complex developmental disruptions of the Müllerian ducts and anorectal structures. Early recognition through first-trimester ultrasound is critical to ensure accurate diagnosis, guide follow-up imaging, and support parental counseling and perinatal planning.</p><p><strong>Case presentation: </strong>A female fetus was identified in the first trimester with a midline cystic pelvic mass posterior to the bladder. The finding was initially suspicious for hydrometrocolpos with an underlying anorectal malformation. At 18 weeks, ultrasound confirmed a cystic lesion with thick echogenic walls extending toward the perineum, supporting the diagnosis of cloacal malformation. By 30 weeks, advanced imaging demonstrated hydrometrocolpos with a longitudinal septum, duplicated cervix, and two uteri, consistent with uterine didelphys. Additional findings included echogenic ascites due to chemical peritonitis, secondary to urine reflux into the peritoneal cavity. These results reflected a rare and complex constellation of Müllerian and cloacal anomalies.</p><p><strong>Conclusion: </strong>This case underscores the importance of systematic serial prenatal imaging and multidisciplinary evaluation in detecting rare congenital anomalies. Early identification of key sonographic features enables optimal diagnosis, facilitates individualized perinatal care, and improves prognostic counseling for families.</p>","PeriodicalId":14337,"journal":{"name":"International Medical Case Reports Journal","volume":"19 ","pages":"569080"},"PeriodicalIF":0.7,"publicationDate":"2026-04-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13110732/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147771418","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":"Prune Belly Syndrome Complicated by Congenital Urinary Fistula in a Neonate: A Case Report from Hargeisa Group Hospital, Hargeisa Somaliland.","authors":"Abdisamed Mohamoud H Ali, Khadar Jama Ibrahim, Abdinour Mohamed Hassan, Sadam Ismail Ahmed","doi":"10.2147/IMCRJ.S584785","DOIUrl":"https://doi.org/10.2147/IMCRJ.S584785","url":null,"abstract":"<p><strong>Background: </strong>Prune Belly Syndrome (PBS), also known as Eagle-Barrett syndrome, is an uncommon congenital disorder characterized by a combination of abdominal wall hypoplasia, urinary tract malformations, and bilateral cryptorchidism. The condition predominantly affects males and is frequently associated with significant neonatal morbidity and mortality related to renal and pulmonary complications. Congenital urinary fistula formation represents a particularly rare and complex manifestation of this syndrome.</p><p><strong>Case presentation: </strong>We report a case of a 2-day-old male neonate presenting to a hospital in a resource-limited setting with respiratory distress and passage of urine via the umbilicus. The patient was born at term via spontaneous vaginal delivery to a mother with no antenatal care. Physical examination revealed a critically ill neonate with a classic \"prune-like\" abdomen, bilateral cryptorchidism, urine flow from an infected umbilical cord, and associated anomalies including polydactyly and clubfoot. Ultrasonography confirmed bilateral gross hydronephrosis and a urinary fistula communicating with the umbilicus.</p><p><strong>Conclusion: </strong>This case highlights a classic presentation of PBS, unusually complicated by a congenital urinary fistula. It underscores the profound challenges in managing this complex syndrome in settings with limited resources, where definitive surgical intervention is unavailable. The report emphasizes the critical importance of prenatal diagnosis and the need for a multidisciplinary approach to improve outcomes, which was not feasible in this context. Ultimately, management was restricted to supportive care and antibiotics, illustrating the dire consequences of diagnostic and therapeutic limitations in such environments.The patient was stabilized and referred to a higher center; however, long-term outcome was not available.</p>","PeriodicalId":14337,"journal":{"name":"International Medical Case Reports Journal","volume":"19 ","pages":"584785"},"PeriodicalIF":0.7,"publicationDate":"2026-04-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13110739/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147771372","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}