Pub Date : 2025-02-13DOI: 10.1016/j.hmedic.2025.100177
Rabia Akram , Shahid Mahmood Baig , Ikram Ullah Khan , Javed Iqbal , Henry Houlden , Tao Sun , Brijesh Sathian , Ghulam Hussain , Javed Iqbal
Ataxia with oculomotor apraxia type 1 (AOA1) is an uncommon genetic condition characterized by dysarthria and gait abnormalities. The predominant characteristic of AOA1 is childhood-onset progressive cerebellar ataxia. Our study aimed to determine the clinical and molecular spectrum of AOA1. We investigated a consanguineous Pakistani family with four affected individuals with early-onset progressive ataxia by using whole-exome sequencing (WES) and co-segregation analysis. Sequencing analysis revealed a novel homozygous variant c.527 T > G (p.Val176Gly) in exon 5 of the aprataxin (APTX) gene. The prominent clinical features of affected individuals include wide-based ataxic gait, difficulties walking, dysmetria, and limb ataxia. The findings of this study broaden the genotypic spectrum of APTX mutations.
{"title":"Genetic insights into ataxia with ocular apraxia type 1: Unraveling a mutation in APTX in a case report of Pakistani family","authors":"Rabia Akram , Shahid Mahmood Baig , Ikram Ullah Khan , Javed Iqbal , Henry Houlden , Tao Sun , Brijesh Sathian , Ghulam Hussain , Javed Iqbal","doi":"10.1016/j.hmedic.2025.100177","DOIUrl":"10.1016/j.hmedic.2025.100177","url":null,"abstract":"<div><div>Ataxia with oculomotor apraxia type 1 (AOA1) is an uncommon genetic condition characterized by dysarthria and gait abnormalities. The predominant characteristic of AOA1 is childhood-onset progressive cerebellar ataxia. Our study aimed to determine the clinical and molecular spectrum of AOA1. We investigated a consanguineous Pakistani family with four affected individuals with early-onset progressive ataxia by using whole-exome sequencing (WES) and co-segregation analysis. Sequencing analysis revealed a novel homozygous variant c.527 T > G (p.Val176Gly) in exon 5 of the aprataxin (<em>APTX)</em> gene. The prominent clinical features of affected individuals include wide-based ataxic gait, difficulties walking, dysmetria, and limb ataxia. The findings of this study broaden the genotypic spectrum of <em>APTX</em> mutations.</div></div>","PeriodicalId":100908,"journal":{"name":"Medical Reports","volume":"10 ","pages":"Article 100177"},"PeriodicalIF":0.0,"publicationDate":"2025-02-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143421544","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}
Case Diagnosis: Bell’s palsy is a neuropathy involving the seventh cranial nerve, that, can be caused by traumatic, infective, inflammatory, and idiopathic. Presentation of Bell’s palsy in pediatric population is quite rare. Appropriate therapy must be given to prevent sequelae that affect their function, social life, and psychology.Case Description: We present a case of a six-year-old girl with Bell’s palsy. She came with five days of left facial nerve paralysis. Patient couldn’t raise her left eyebrow, unable to close her left eye completely, asymmetrical on the left nose and lip, absence of left forehead creases and nasolabial fold, and synkineses. To quantify the outcome, Facial Grading System (FGS) was calculated, and obtained score 5/100 on the first day of therapy. The patient was prescribed a course of methylprednisolone for eleven days and mecobalamine for seven days. The physical rehabilitation program was electrical stimulation and infrared therapy once a week, and Proprioceptive Neuromuscular Facilitation (PNF). On the seventh day, the FGS score was 10/100.This happened because her parents, as fulltime workers, not performed PNF appropriately and the patient couldn’t perform it independently. Therefore, patient was trained with easier facial exercises that could be performed independently in front of mirror. Then, the FGS score significantly improved, the score was 58/100 on the 13th day, and 96/100 on the 16th day.Discussion: Facial exercises in front of mirror can provide a biofeedback effect that make visual stimulation to the brain to give signals in moving the facial muscles repeatedly to prevent potential facial muscle contractures and improve facial function.Conculsion: Facial exercises in front of mirror can help patient with Bell’s Palsy to recover in shorter duration and increase muscle strength, prevent synkinesis, dyskinesis, and improve facial functionality in children with Bell’s Palsy.
{"title":"The importance of mirror facial exercises in a child with Bell’s palsy: A case report","authors":"Zahraqumi Halimati , Henny Luthfianingrum , Ghani Ikhsan Majid","doi":"10.1016/j.hmedic.2025.100179","DOIUrl":"10.1016/j.hmedic.2025.100179","url":null,"abstract":"<div><div>Case Diagnosis: Bell’s palsy is a neuropathy involving the seventh cranial nerve, that, can be caused by traumatic, infective, inflammatory, and idiopathic. Presentation of Bell’s palsy in pediatric population is quite rare. Appropriate therapy must be given to prevent sequelae that affect their function, social life, and psychology.Case Description: We present a case of a six-year-old girl with Bell’s palsy. She came with five days of left facial nerve paralysis. Patient couldn’t raise her left eyebrow, unable to close her left eye completely, asymmetrical on the left nose and lip, absence of left forehead creases and nasolabial fold, and synkineses. To quantify the outcome, Facial Grading System (FGS) was calculated, and obtained score 5/100 on the first day of therapy. The patient was prescribed a course of methylprednisolone for eleven days and mecobalamine for seven days. The physical rehabilitation program was electrical stimulation and infrared therapy once a week, and Proprioceptive Neuromuscular Facilitation (PNF). On the seventh day, the FGS score was 10/100.This happened because her parents, as fulltime workers, not performed PNF appropriately and the patient couldn’t perform it independently. Therefore, patient was trained with easier facial exercises that could be performed independently in front of mirror. Then, the FGS score significantly improved, the score was 58/100 on the 13th day, and 96/100 on the 16th day.Discussion: Facial exercises in front of mirror can provide a biofeedback effect that make visual stimulation to the brain to give signals in moving the facial muscles repeatedly to prevent potential facial muscle contractures and improve facial function.Conculsion: Facial exercises in front of mirror can help patient with Bell’s Palsy to recover in shorter duration and increase muscle strength, prevent synkinesis, dyskinesis, and improve facial functionality in children with Bell’s Palsy.</div></div>","PeriodicalId":100908,"journal":{"name":"Medical Reports","volume":"10 ","pages":"Article 100179"},"PeriodicalIF":0.0,"publicationDate":"2025-02-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143421523","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}
Pub Date : 2025-02-12DOI: 10.1016/j.hmedic.2025.100175
Wael A. Labib , Reem.M.A. Ebrahim
A 38-year-old male dental patient experienced an out-of-hospital cardiac arrest (OHCA) due to a significant anterior myocardial infarction. After collapsing and losing responsiveness, he received advanced life support and intubation. An electrocardiogram confirmed the myocardial infarction, leading to an urgent coronary angiography that revealed a total occlusion in the proximal left anterior descending artery. Emergency revascularization with a drug-eluting stent was performed, restoring blood flow. Although he remained comatose after the procedure, echocardiography indicated preserved systolic function. Mechanical ventilation and enteral feeding were initiated to support recovery. This study highlights coronary revascularization's complexities and potential benefits for critically ill patients who have experienced an out-of-hospital cardiac arrest (OHCA).
{"title":"Mortality benefits of coronary revascularization in a 38-year-old post-out-of-hospital cardiac arrest patient: Case report","authors":"Wael A. Labib , Reem.M.A. Ebrahim","doi":"10.1016/j.hmedic.2025.100175","DOIUrl":"10.1016/j.hmedic.2025.100175","url":null,"abstract":"<div><div>A 38-year-old male dental patient experienced an out-of-hospital cardiac arrest (OHCA) due to a significant anterior myocardial infarction. After collapsing and losing responsiveness, he received advanced life support and intubation. An electrocardiogram confirmed the myocardial infarction, leading to an urgent coronary angiography that revealed a total occlusion in the proximal left anterior descending artery. Emergency revascularization with a drug-eluting stent was performed, restoring blood flow. Although he remained comatose after the procedure, echocardiography indicated preserved systolic function. Mechanical ventilation and enteral feeding were initiated to support recovery. This study highlights coronary revascularization's complexities and potential benefits for critically ill patients who have experienced an out-of-hospital cardiac arrest (OHCA).</div></div>","PeriodicalId":100908,"journal":{"name":"Medical Reports","volume":"10 ","pages":"Article 100175"},"PeriodicalIF":0.0,"publicationDate":"2025-02-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143421524","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}
Pub Date : 2025-02-10DOI: 10.1016/j.hmedic.2025.100176
Emanuel-Youssef Dib , Jessy Fadel , Karam Karam , Sarah Saleh , Houssein Chebbo , Elias Fiani
Background
Eosinophilic esophagitis (EoE) is a chronic, immune-mediated esophageal disorder marked by eosinophil infiltration and symptoms that frequently resemble gastroesophageal reflux disease (GERD). It is usually accompanied by atopic conditions such as asthma, eczema, allergic rhinitis, and food allergies. Steakhouse syndrome is characterized by acute esophageal food impaction, which is more frequent in older people and can be caused by various underlying diseases, including EoE. Accurate diagnosis and management of EoE are critical for symptom relief and to prevent complications.
Case Presentation
A 27-year-old man with lactose, nut, and cabbage allergies and a history of recurrent food impactions complained of chest pain after eating meat. Initial examinations, including chest and spine X-rays and laboratory testing, were normal, with no elevated eosinophils in the CBC differential. An emergency gastroscopy revealed a stenotic esophagus with rings and furrows, and food residue, favoring steakhouse syndrome. Biopsies revealed more than 15 eosinophils per high-powered field, indicating EoE, while malignancy was ruled out. Omeprazole, fluticasone, and a six-food elimination diet were used for treatment. A follow-up endoscopy after one month revealed both endoscopic and histologic remission.
Conclusion
This case emphasizes the significance of identifying EoE as a possible cause of recurring dietary impacts that manifest as steakhouse syndrome. A thorough examination and correct diagnosis are essential for effective treatment. The patient’s successful resolution emphasizes the importance of clinical vigilance and comprehensive treatment strategies for esophageal disorders. Further research is warranted to improve diagnostic and therapeutic approaches for EoE.
{"title":"Eosinophilic esophagitis masquerading as steakhouse syndrome: A case report","authors":"Emanuel-Youssef Dib , Jessy Fadel , Karam Karam , Sarah Saleh , Houssein Chebbo , Elias Fiani","doi":"10.1016/j.hmedic.2025.100176","DOIUrl":"10.1016/j.hmedic.2025.100176","url":null,"abstract":"<div><h3>Background</h3><div>Eosinophilic esophagitis (EoE) is a chronic, immune-mediated esophageal disorder marked by eosinophil infiltration and symptoms that frequently resemble gastroesophageal reflux disease (GERD). It is usually accompanied by atopic conditions such as asthma, eczema, allergic rhinitis, and food allergies. Steakhouse syndrome is characterized by acute esophageal food impaction, which is more frequent in older people and can be caused by various underlying diseases, including EoE. Accurate diagnosis and management of EoE are critical for symptom relief and to prevent complications.</div></div><div><h3>Case Presentation</h3><div>A 27-year-old man with lactose, nut, and cabbage allergies and a history of recurrent food impactions complained of chest pain after eating meat. Initial examinations, including chest and spine X-rays and laboratory testing, were normal, with no elevated eosinophils in the CBC differential. An emergency gastroscopy revealed a stenotic esophagus with rings and furrows, and food residue, favoring steakhouse syndrome. Biopsies revealed more than 15 eosinophils per high-powered field, indicating EoE, while malignancy was ruled out. Omeprazole, fluticasone, and a six-food elimination diet were used for treatment. A follow-up endoscopy after one month revealed both endoscopic and histologic remission.</div></div><div><h3>Conclusion</h3><div>This case emphasizes the significance of identifying EoE as a possible cause of recurring dietary impacts that manifest as steakhouse syndrome. A thorough examination and correct diagnosis are essential for effective treatment. The patient’s successful resolution emphasizes the importance of clinical vigilance and comprehensive treatment strategies for esophageal disorders. Further research is warranted to improve diagnostic and therapeutic approaches for EoE.</div></div>","PeriodicalId":100908,"journal":{"name":"Medical Reports","volume":"10 ","pages":"Article 100176"},"PeriodicalIF":0.0,"publicationDate":"2025-02-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143378902","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}
<div><h3>Introduction</h3><div>Acute hemorrhagic leukoencephalitis (AHLE) is a rare and severe form of acute disseminated encephalomyelitis (ADEM), characterized by rapid progression and inflammation of the brain's white matter. It's often preceded by an upper respiratory infection, bacterial or viral illness, or even vaccination. An opercular syndrome is characterized by the triad of facial weakness, dysarthria, and difficulty in swallowing due to lesions in the operculum, the part of the brain that covers the insula. When AHLE is associated with opercular syndrome and scrub typhus, it presents a unique and challenging clinical condition. Here, we are presenting a case of AHLE (a subtype of ADEM), which is very rare but is also associated with scrub typhus and opercular syndrome (a rare syndrome). In this way, the presenting case becomes the rarest of rare or most exceptional.</div></div><div><h3>Case report</h3><div>A complex clinical case involving a 12-year-old girl, who had the symptoms of fever, vomiting, headache, focal seizure, and altered sensorium for the last 25 days, she was initially diagnosed as tuberculous meningitis (TBM) in an outside hospital, but there was no significant improvement with standard anti-tubercular therapy (ATT). Due to no notable improvement, she was brought to our institute (IMS, BHU) for further evaluation suggests a need for a comprehensive approach to uncover any underlying conditions that may be contributing to her symptoms. The additional and necessary investigations, such as fever workup, repeat CSF analysis, routine blood tests, and MRI brain, were crucial steps in this process. The serum of the patient was positive for IgM scrub typhus suggesting an active infection. The presence of lymphocytic pleocytosis, elevated protein, and reduced sugar in the CSF indicate inflammation and dysfunction within the central nervous system (CNS). The MRI findings of extensive subcortical and white matter changes with haemorrhage further suggest AHLE. Symptoms and MRI features also projected the diagnosis of opercular syndrome. CSF neuro-infection panel was negative. Stopping ATT and initiating pulse steroid therapy along with doxycycline was a prudent decision. Doxycycline is the treatment of choice for scrub typhus, and the addition of pulse steroid therapy aims to reduce inflammation and prevent further neurological damage.</div></div><div><h3>Conclusion</h3><div>AHLE (Acute Hemorrhagic Leukoencephalitis) is indeed a severe and often fatal condition, and its association with scrub typhus and opercular syndrome presents a unique and challenging clinical scenario. Given its rarity and the potential for misdiagnosis or lack of recognition, highlighting such cases can be crucial in increasing awareness among healthcare professionals. Moreover, documenting such rare occurrences can contribute significantly to the medical literature, providing valuable insights into the clinical features, pathophysiology, and management str
{"title":"Acute hemorrhagic leukoencephalitis (AHLE): An exceptional clinical presentation with the association of scrub typhus and opercular syndrome","authors":"Arpan Mitra , Niraj Kumar Srivastava , Subhraneel Paul , Anand Kumar , Deepika Joshi","doi":"10.1016/j.hmedic.2025.100166","DOIUrl":"10.1016/j.hmedic.2025.100166","url":null,"abstract":"<div><h3>Introduction</h3><div>Acute hemorrhagic leukoencephalitis (AHLE) is a rare and severe form of acute disseminated encephalomyelitis (ADEM), characterized by rapid progression and inflammation of the brain's white matter. It's often preceded by an upper respiratory infection, bacterial or viral illness, or even vaccination. An opercular syndrome is characterized by the triad of facial weakness, dysarthria, and difficulty in swallowing due to lesions in the operculum, the part of the brain that covers the insula. When AHLE is associated with opercular syndrome and scrub typhus, it presents a unique and challenging clinical condition. Here, we are presenting a case of AHLE (a subtype of ADEM), which is very rare but is also associated with scrub typhus and opercular syndrome (a rare syndrome). In this way, the presenting case becomes the rarest of rare or most exceptional.</div></div><div><h3>Case report</h3><div>A complex clinical case involving a 12-year-old girl, who had the symptoms of fever, vomiting, headache, focal seizure, and altered sensorium for the last 25 days, she was initially diagnosed as tuberculous meningitis (TBM) in an outside hospital, but there was no significant improvement with standard anti-tubercular therapy (ATT). Due to no notable improvement, she was brought to our institute (IMS, BHU) for further evaluation suggests a need for a comprehensive approach to uncover any underlying conditions that may be contributing to her symptoms. The additional and necessary investigations, such as fever workup, repeat CSF analysis, routine blood tests, and MRI brain, were crucial steps in this process. The serum of the patient was positive for IgM scrub typhus suggesting an active infection. The presence of lymphocytic pleocytosis, elevated protein, and reduced sugar in the CSF indicate inflammation and dysfunction within the central nervous system (CNS). The MRI findings of extensive subcortical and white matter changes with haemorrhage further suggest AHLE. Symptoms and MRI features also projected the diagnosis of opercular syndrome. CSF neuro-infection panel was negative. Stopping ATT and initiating pulse steroid therapy along with doxycycline was a prudent decision. Doxycycline is the treatment of choice for scrub typhus, and the addition of pulse steroid therapy aims to reduce inflammation and prevent further neurological damage.</div></div><div><h3>Conclusion</h3><div>AHLE (Acute Hemorrhagic Leukoencephalitis) is indeed a severe and often fatal condition, and its association with scrub typhus and opercular syndrome presents a unique and challenging clinical scenario. Given its rarity and the potential for misdiagnosis or lack of recognition, highlighting such cases can be crucial in increasing awareness among healthcare professionals. Moreover, documenting such rare occurrences can contribute significantly to the medical literature, providing valuable insights into the clinical features, pathophysiology, and management str","PeriodicalId":100908,"journal":{"name":"Medical Reports","volume":"10 ","pages":"Article 100166"},"PeriodicalIF":0.0,"publicationDate":"2025-02-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143095202","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}
Pub Date : 2025-02-01DOI: 10.1016/j.hmedic.2024.100152
Abdul Qadir , Amal Wael Abdellatif , Jamal Sajid , Mamunul Islam , Dana Al-Sayegh
Streptococcus gordonii, a Gram-positive bacterium found in the human oral cavity, skin, intestines, and respiratory tract, can occasionally cause serious infections, including periodontitis, bacteremia, and infective endocarditis. Rarely, it can lead to infectious spondylitis, a severe spinal infection. We report a case of a 49-year-old male presenting with neck pain and fever. Examination revealed cervical spine tenderness and elevated inflammatory markers. MRI indicated spondylitic changes and cervical disc degeneration with mild disc bulge at C4-C5. Blood cultures identified S. gordonii. Hence, making a definitive diagnosis of S. gordonii causing infectious spondylodiscitis. A 6-week course of ceftriaxone resolved his symptoms and normalized inflammatory markers. Previous literature reports only few similar cases, mainly linked to dental or spinal surgeries. This is the only case reported unusual as the patient had no recent dental or spinal procedures yet predisposing to infectious spondylitis. This highlights the need for spinal imaging in patients with S. gordonii bacteremia and neck or back pain to ensure proper diagnosis and treatment.
{"title":"Spondylodiscitis by Streptococcus Gordonii in an immunocompetent adult: A case report","authors":"Abdul Qadir , Amal Wael Abdellatif , Jamal Sajid , Mamunul Islam , Dana Al-Sayegh","doi":"10.1016/j.hmedic.2024.100152","DOIUrl":"10.1016/j.hmedic.2024.100152","url":null,"abstract":"<div><div><em>Streptococcus gordonii</em>, a Gram-positive bacterium found in the human oral cavity, skin, intestines, and respiratory tract, can occasionally cause serious infections, including periodontitis, bacteremia, and infective endocarditis. Rarely, it can lead to infectious spondylitis, a severe spinal infection. We report a case of a 49-year-old male presenting with neck pain and fever. Examination revealed cervical spine tenderness and elevated inflammatory markers. MRI indicated spondylitic changes and cervical disc degeneration with mild disc bulge at C4-C5. Blood cultures identified <em>S. gordonii</em>. Hence, making a definitive diagnosis of <em>S. gordonii</em> causing infectious spondylodiscitis. A 6-week course of ceftriaxone resolved his symptoms and normalized inflammatory markers. Previous literature reports only few similar cases, mainly linked to dental or spinal surgeries. This is the only case reported unusual as the patient had no recent dental or spinal procedures yet predisposing to infectious spondylitis. This highlights the need for spinal imaging in patients with <em>S. gordonii</em> bacteremia and neck or back pain to ensure proper diagnosis and treatment.</div></div>","PeriodicalId":100908,"journal":{"name":"Medical Reports","volume":"9 ","pages":"Article 100152"},"PeriodicalIF":0.0,"publicationDate":"2025-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143144380","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}
Pub Date : 2025-02-01DOI: 10.1016/j.hmedic.2025.100160
John Peters , Tyler Lackland , Mohamed M.G. Mohamed , Christopher Barrios
Necrotizing pneumonia is a relatively rare complication of bacterial pneumonia. It is characterized by pulmonary inflammation associated with vascular compromise causing necrosis and is mostly triggered by common respiratory pathogens. Atypical pathogens might cause necrotizing pneumonia in immunocompromised hosts. We are presenting an atypical case of necrotizing pneumonia caused by Delftia acidovorans and Stenotrophomonas maltophilia in an immunocompetent host. We aim to highlight the potential role of these bacteria as causative pathogens of necrotizing pneumonia and to facilitate early consideration and recognition by health care providers.
{"title":"A coinfection of Delftia acidovorans and Stenotrophomonas maltophilia as an atypical cause of necrotizing pneumonia: A case report","authors":"John Peters , Tyler Lackland , Mohamed M.G. Mohamed , Christopher Barrios","doi":"10.1016/j.hmedic.2025.100160","DOIUrl":"10.1016/j.hmedic.2025.100160","url":null,"abstract":"<div><div>Necrotizing pneumonia is a relatively rare complication of bacterial pneumonia. It is characterized by pulmonary inflammation associated with vascular compromise causing necrosis and is mostly triggered by common respiratory pathogens. Atypical pathogens might cause necrotizing pneumonia in immunocompromised hosts. We are presenting an atypical case of necrotizing pneumonia caused by <em>Delftia acidovorans</em> and <em>Stenotrophomonas maltophilia</em> in an immunocompetent host. We aim to highlight the potential role of these bacteria as causative pathogens of necrotizing pneumonia and to facilitate early consideration and recognition by health care providers.</div></div>","PeriodicalId":100908,"journal":{"name":"Medical Reports","volume":"9 ","pages":"Article 100160"},"PeriodicalIF":0.0,"publicationDate":"2025-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143144950","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}
Pub Date : 2025-02-01DOI: 10.1016/j.hmedic.2025.100163
Thomas Solomon , Dereje Dageme , Abdela Aliyi , Ibsa Mohammed , Bekalu Yirga , Chalachew Bizuye , Astawus Alemayehu
Background
Total procidentia, or complete uterine prolapse, at term pregnancy is an exceptionally rare and severe obstetric condition. This condition, when accompanied by intrauterine fetal demise (IUFD), significantly increases the risks of obstructed labor, maternal morbidity, and poor fetal outcomes. Early recognition and a comprehensive management approach are critical to minimizing adverse outcomes. This case highlights the challenges and importance of multidisciplinary care in managing such a rare and complex presentation.
Case presentation
A 30-year-old gravida 7, para 6 woman at 39 weeks and 5 days of gestation, with no antenatal care, presented with labor symptoms, including a protruding vaginal mass and a two-day history of absent fetal movements. Physical examination confirmed total uterine prolapse and absent fetal heart tones. Ultrasound revealed a third-trimester intrauterine pregnancy complicated by IUFD. Following informed consent, an emergency cesarean section was performed, delivering a 3000 g macerated male stillborn. The surgery also included a bilateral salpingectomy per the patient’s request.
Outcome
The patient’s postoperative course was complicated by partial bowel obstruction on the 2nd postoperative day, managed conservatively. By the 6th postoperative day, she developed wound dehiscence and intra-abdominal infection, necessitating a re-laparotomy and hysterectomy. Multidisciplinary coordination between surgical and anesthesiology teams was essential in managing these severe complications.
Conclusion
This case underscores the critical importance of prompt surgical intervention and rigorous postoperative care in managing total procidentia at term with IUFD. The significant complications encountered highlight the necessity of a multidisciplinary approach and This rare condition also highlight the importance of innovative surgical techniques and rigorous postoperative monitoring to minimize maternal morbidity and improve outcomes in life-threatening obstetric emergencies.
{"title":"Challenges in management and postoperative complications of total uterine procidentia at term: Case report","authors":"Thomas Solomon , Dereje Dageme , Abdela Aliyi , Ibsa Mohammed , Bekalu Yirga , Chalachew Bizuye , Astawus Alemayehu","doi":"10.1016/j.hmedic.2025.100163","DOIUrl":"10.1016/j.hmedic.2025.100163","url":null,"abstract":"<div><h3>Background</h3><div>Total procidentia, or complete uterine prolapse, at term pregnancy is an exceptionally rare and severe obstetric condition. This condition, when accompanied by intrauterine fetal demise (IUFD), significantly increases the risks of obstructed labor, maternal morbidity, and poor fetal outcomes. Early recognition and a comprehensive management approach are critical to minimizing adverse outcomes. This case highlights the challenges and importance of multidisciplinary care in managing such a rare and complex presentation.</div></div><div><h3>Case presentation</h3><div>A 30-year-old gravida 7, para 6 woman at 39 weeks and 5 days of gestation, with no antenatal care, presented with labor symptoms, including a protruding vaginal mass and a two-day history of absent fetal movements. Physical examination confirmed total uterine prolapse and absent fetal heart tones. Ultrasound revealed a third-trimester intrauterine pregnancy complicated by IUFD. Following informed consent, an emergency cesarean section was performed, delivering a 3000 g macerated male stillborn. The surgery also included a bilateral salpingectomy per the patient’s request.</div></div><div><h3>Outcome</h3><div>The patient’s postoperative course was complicated by partial bowel obstruction on the 2nd postoperative day, managed conservatively. By the 6th postoperative day, she developed wound dehiscence and intra-abdominal infection, necessitating a re-laparotomy and hysterectomy. Multidisciplinary coordination between surgical and anesthesiology teams was essential in managing these severe complications.</div></div><div><h3>Conclusion</h3><div>This case underscores the critical importance of prompt surgical intervention and rigorous postoperative care in managing total procidentia at term with IUFD. The significant complications encountered highlight the necessity of a multidisciplinary approach and This rare condition also highlight the importance of innovative surgical techniques and rigorous postoperative monitoring to minimize maternal morbidity and improve outcomes in life-threatening obstetric emergencies.</div></div>","PeriodicalId":100908,"journal":{"name":"Medical Reports","volume":"9 ","pages":"Article 100163"},"PeriodicalIF":0.0,"publicationDate":"2025-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143144945","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}
Nasopalatine duct cysts (NPDCs) are among the most common non-odontogenic cysts, typically located in the midline of the anterior hard palate. They arise from the embryonic epithelial remnants of the nasopalatine duct. NPDCs are more frequently seen in adult males, usually during the fourth decade of life. Clinically, they present as an asymptomatic or occasionally painful, fluctuant, or discharging swelling in the hard palate. Radiographically, NPDCs appear as well-defined, corticated, inverted pear-shaped or heart-shaped radiolucencies situated between the roots of the maxillary incisors. Histologically, the epithelial lining shows considerable variation, ranging from stratified squamous to pseudostratified ciliated columnar, columnar, or cuboidal epithelium, with transitions often observed between these types. The cyst wall is fibrocollagenous and may include distinctive features such as neurovascular bundles, mucous glands, or cartilaginous remnants. Management typically involves surgical enucleation, with a low risk of recurrence. Here, we present a case of NPDC along with a brief overview of its origin, diagnostic characteristics, and treatment approach.
{"title":"A brief insight regarding Nasopalatine duct cyst – Report of a case with a review of literature","authors":"Snehanjan Sarangi, Debarati Ray, Basudev Mahato, Tathagata Bhattacharjee, Rudra Pyne","doi":"10.1016/j.hmedic.2025.100156","DOIUrl":"10.1016/j.hmedic.2025.100156","url":null,"abstract":"<div><div>Nasopalatine duct cysts (NPDCs) are among the most common non-odontogenic cysts, typically located in the midline of the anterior hard palate. They arise from the embryonic epithelial remnants of the nasopalatine duct. NPDCs are more frequently seen in adult males, usually during the fourth decade of life. Clinically, they present as an asymptomatic or occasionally painful, fluctuant, or discharging swelling in the hard palate. Radiographically, NPDCs appear as well-defined, corticated, inverted pear-shaped or heart-shaped radiolucencies situated between the roots of the maxillary incisors. Histologically, the epithelial lining shows considerable variation, ranging from stratified squamous to pseudostratified ciliated columnar, columnar, or cuboidal epithelium, with transitions often observed between these types. The cyst wall is fibrocollagenous and may include distinctive features such as neurovascular bundles, mucous glands, or cartilaginous remnants. Management typically involves surgical enucleation, with a low risk of recurrence. Here, we present a case of NPDC along with a brief overview of its origin, diagnostic characteristics, and treatment approach.</div></div>","PeriodicalId":100908,"journal":{"name":"Medical Reports","volume":"9 ","pages":"Article 100156"},"PeriodicalIF":0.0,"publicationDate":"2025-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143144948","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}