American Journal of Case Reports最新文献

BACKGROUND Ureaplasma urealyticum (UU) is a common microorganism that has been associated with a variety of obstetric and neonatal complications, such as infertility, stillbirth, histologic chorioamnionitis, neonatal sepsis, respiratory infections, and central nervous system infections. However, it is rare for it to cause severe neonatal asphyxia. This rarity is the focus of our case report, which aims to highlight the potential severity of UU infections in newborns. CASE REPORT A male neonate was delivered vaginally at 40+5 weeks of gestation, with a history of premature rupture of membranes at 7 hours and low amniotic fluid volume. After birth, he had no spontaneous breathing at birth, with cyanosis around the mouth and extremities and flaccid limbs. The Apgar score was a low 3, indicating severe asphyxia. Immediate medical interventions were undertaken, including cardiopulmonary resuscitation, tracheal intubation, and airway clearing. Despite these efforts, the newborn required transfer to our hospital with the assistance of an invasive ventilator. Upon admission, UU DNA was detected in the sputum at 3.25×10⁴ copies/mL. He received erythromycin for infection, mechanical ventilation, milrinone combined with sildenafil to reduce pulmonary arterial hypertension, and glycerol fructose to reduce intracranial pressure. Although these treatments successfully controlled the infection, the infant suffered significant neurological damage. Tragically, the family decided to cease treatment, and the neonate died at 12 days of age. CONCLUSIONS Ureaplasma urealyticum infection can cause severe neonatal asphyxia. We report the present case to raise awareness about the importance of early detection and intervention for UU in pregnant women to improve maternal and neonatal outcomes.

BACKGROUND Extensor pollicis longus (EPL) tendon rupture is a potential complication following distal radius fracture, typically occurring several weeks after injury. Herein, we present a rare case of acute extensor pollicis longus tendon rupture associated with a distal radius fracture. CASE REPORT A 35-year-old woman visited our hospital with a distal radius fracture. At the time of examination, the thumb could be moved, although there was pain in the wrist. Computed tomography (CT) revealed a fracture line extending to the ulnar side of Lister's tubercle. During volar locking plate fixation, visual confirmation of the EPL tendon injury was conducted. According to the intraoperative finding, the EPL tendon was 70% ruptured and elongated, and the extension mechanism of the thumb was disrupted. Based on these findings, direct suture of the EPL tendon, including elongation of the ruptured tendon, was performed. One year after surgery, the EPL tendon excursion was smooth, with no limitations in thumb mobility, and the patient had returned to work. CONCLUSIONS This case highlights the importance of recognizing that EPL tendon injury can occur in conjunction with distal radius fractures. The risk of the EPL tendon injury is particularly high when the fracture line extends to the ulnar side of Lister's tubercle, which serves as the EPL tendon floor. When an EPL tendon injury is suspected based on preoperative examination and fracture type, it is important to obtain adequate informed consent and to prepare for potential EPL tendon reconstruction before surgery.

BACKGROUND Cleidocranial dysplasia (CCD) is a rare (1: 1 000 000) autosomal dominant congenital skeletal dysplasia characterized by widely patent calvarial sutures, clavicular hypoplasia, supernumerary teeth, and short stature. Only a minority of the cases are diagnosed early after birth. We present another case of proven CCD presenting with typical neonatal phenotype to promote awareness of this rare disorder. CASE REPORT A male term neonate presented clinically with unusual soft skull, extremely large fontanels, and palpable right clavicular discontinuity. Cranial computed tomography revealed severe calvarian ossification defect leading to widely enlarged sutures. Right-sided clavicular dysplasia with discontinuity in the middle part were shown on a chest X-ray. Whole-exome sequencing and Sanger sequencing of the RUNX2 gene confirmed the diagnosis of CCD following a single-nucleotide mutation (NM_001024630.3: c.569G>A) in the child as well as in his mother. His additional family members in the maternal line had also different degrees of clavicular and cranial hypoplasia or multiple dental anomalies. CONCLUSIONS In clinical practice, in a newborn presenting with severe defects in ossification of the skull and widely enlarged sutures and/or hypoplasia or aplasia of the clavicles, CCD should be considered based on a combination of clinical and radiological genetic criteria. Family history and genetic testing are crucial since the mutation follows autosomal dominant inheritance.

BACKGROUND The management of unstable atlas fractures remains a subject of ongoing debate and controversy. The conservative surgical treatment commonly involves fusion, resulting in severe loss of cervical spine mobility, and a large incisions and extensive tissue dissection are required. We aim to introduce a novel concept and surgical approach for treating atlas fracture, one that involves minimizing trauma while maintaining mobility of the upper cervical spine without resorting to fusion. CASE REPORT We present the case of a 48-year-old woman with an atlas fracture who underwent robot-assisted pedicle screw placement via biportal endoscopy technique. We seek to introduce a novel concept and surgical approach for treating atlas fracture, one that involves minimizing trauma while maintaining mobility of the upper cervical spine without resorting to fusion. The patient's preoperative VAS score for neck pain was 7, which improved to 3 postoperatively. The right and left cervical rotation improved from 15° preoperatively to 50° postoperatively. Preoperative CT and MRI scans demonstrated satisfactory reduction of the anterior and posterior arch fractures and satisfactory fixation position of the pedicle screws. She was discharged 4 days later, and she had good range of flexion, extension, and rotation of the cervical spine 2 months after surgery. CONCLUSIONS Robot-assisted percutaneous atlas pedicle screw placement via biportal endoscopy is a beneficial surgical treatment for atlas fractures. This technique offers satisfactory alignment and union of the fractures and preservation of atlanto-axial joint motion while having the advantages of minimal invasiveness, rapid postoperative recovery, and fewer complications.

BACKGROUND Extracorporeal blood purification strategies were recently developed as adjunctive treatments for sepsis. CytoSorb® is an approved medical device designed to reduce blood levels of inflammatory cytokines. The oXiris high-adsorption membrane filter is used in continuous hemofiltration adsorption. We describe the case of a 67-year-old man with recurrent septic shock, requiring treatment with antibiotics, vasopressors, inotropes, mechanical ventilation, continuous renal replacement therapy (CRRT), and adjunctive treatment with an oXiris filter and hemadsorption using the CytoSorb device. CASE REPORT A 67-year-old man was admitted to the Intensive Care Unit (ICU) with septic shock. He received antibiotics, fluids, vasopressors, and inotropes and was mechanically ventilated. Acute kidney failure was treated with CRRT. His condition improved, and he was transferred to the general ward. On day 3 in the ward, he developed a new episode of septic shock and was readmitted to ICU. Standard therapy with fluids, vasopressors, and empiric antibiotics was started. Despite treatment, his condition deteriorated dramatically. CRRT with an oXiris filter in combination with hemoadsorption using CytoSorb was started. After CRRT initiation and integration of the CytoSorb adsorber, rapid improvement in the patient's condition was observed. Daily laboratory test results showed significant decreases in procalcitonin and CRP. The patient was discharged from ICU on day 5 after initiation of CytoSorb therapy. CONCLUSIONS Although no guidelines and large clinical trial data are yet available to support the use of CytoSorb and oXiris, this report supports the findings from previous reports and small studies, that they can be considered as adjunctive treatments for patients with sepsis.

BACKGROUND Post-transplant lymphoproliferative disorder (PTLD) encompasses a group of disorders ranging from hyperplastic to malignant lymphoid proliferations in the post-transplant period owing to immunosuppression, often in the setting of EBV transformation. PTLD is a rare complication of immunosuppression that, like lymphomas, can have a variable presentation based on disease localization. We report a case of PTLD mass effect at the porta hepatis for the first time in the literature, resulting in hepatic artery stenosis (HAS) and common hepatic duct obstruction. CASE REPORT A 54-year-old woman presented with cholestasis 4 months after receiving a deceased donor liver transplant. MRCP revealed a mass at the porta hepatis causing biliary compression, which resolved with subsequent biliary stenting. The woman presented again 3 weeks later with a similar presentation. CT revealed that the mass had enlarged, causing HAS, which resolved with subsequent hepatic artery stenting. The biopsy revealed PTLD, and the patient was successfully managed with rituximab and a reduction of her immunosuppressive regimen. CONCLUSIONS This case report describes a rare occurrence of PTLD, which particularly impacting the porta hepatis, resulting in HAS and compression of the bile duct. PTLD should be considered in the differential diagnosis for obstructive jaundice and hepatic artery compression, even in the early post-transplant months, when these symptoms are often attributed to surgical factors.

BACKGROUND Acute urinary retention (AUR) is relatively rare in non-pregnant women and is usually associated with lower urinary tract dysfunction, neurological issues, or pelvic organ compression. Adenomyosis is a common gynecologic condition characterized by the invasion of endometrial glands and stroma into the myometrium, often accompanied by symptoms such as dysmenorrhea and heavy menstrual periods. Although adenomyosis is common, its involvement in causing urinary retention is rare but deserves clinical attention. CASE REPORT We report the case of a patient with recurrent urinary retention due to adenomyosis, who had a 20-year history of dysmenorrhea and menorrhagia. Imaging revealed significant thickening of the posterior uterine wall and a globular increase in size. Due to the ineffectiveness of conservative treatment, the patient ultimately underwent a total hysterectomy via laparoscopy. After the operation, the patient's urinary retention symptoms completely disappeared, her urinary function quickly returned to normal, and her postoperative follow-up showed no recurrence of urinary retention or other urinary-related symptoms. CONCLUSIONS Although adenomyosis is common in women, acute urinary retention remains rare. This case report suggests that clinicians should consider adenomyosis as a potential cause alongside other common causes of recurrent or unexplained urinary retention in female patients, particularly in those presenting with classic symptoms such as prolonged dysmenorrhea and menorrhagia. The successful treatment of this case further highlights the importance of an individualized treatment plan, as total hysterectomy can effectively relieve symptoms and improve the patient's quality of life.

BACKGROUND Arterial hypertension in pediatric patients often presents complex diagnostic and therapeutic challenges. The diagnosis of hypertension in children is based on different guidelines than in adults, with arterial hypertension in children defined as systolic and/or diastolic blood pressure values at or above the 95th percentile for age, sex, and height. Unlike adult populations, it is predominantly secondary in etiology, with conditions such as renovascular hypertension as common causes. Fibromuscular dysplasia and Takayasu arteritis are frequent underlying causes of renal artery stenosis associated with this patient population. CASE REPORT This case report details the successful management of a 15-year-old girl with severe symptomatic hypertension. Plasma renin and aldosterone concentration analysis, Doppler ultrasonography, and angiography were crucial in assessing the severity and nature of the renal stenosis, leading to a diagnosis of renovascular hypertension associated with fibromuscular dysplasia. The patient underwent 2 successful percutaneous transluminal renal angioplasties, achieving blood pressure control without the need for long-term pharmacological therapy. CONCLUSIONS In children with elevated blood pressure, secondary hypertension should always be considered and investigated. The diagnosis of renal artery stenosis necessitates a multidisciplinary approach, combining laboratory tests and invasive and non-invasive imaging techniques. Ultrasound is often insufficient for comprehensive and detailed imaging of the renal arteries, being susceptible to error. Careful and comprehensive management of similar cases in specialized centers is essential for ensuring optimal patient care.

BACKGROUND Meckel's diverticulum is a congenital diverticulum that contains all normal layers of the gastrointestinal wall. In adults, Meckel's diverticulum can present with bowel obstruction, the most common presentation, in 35.6% of cases. In addition, complicated Meckel's diverticulum can present with small bowel obstruction. Sjögren syndrome is a rare systemic autoimmune disorder. The theory behind Sjögren syndrome is that lymphocyte-rich mononuclear cell foci infiltrate exocrine glandular tissue, and humoral factors, such as antibodies and cytokines, cause dysfunction to the exocrine glands, leading to diminished saliva and tear production and skin, tracheobronchial, and vaginal dryness. Sjögren syndrome can present with intestinal pseudo-obstruction as a rare complication. CASE REPORT A 25-year-old woman with a known case of Sjögren syndrome presented to the Emergency Department with abdominal pain. Laboratory investigations were sent, and all results were within reference values. Multiple imaging studies were done, and findings indicated the presence of ileus, rather than bowel, obstruction. Consequently, she was admitted under general surgery, and laparoscopic exploration was done, which showed Meckel's diverticulum. The patient was discharged home in a stable state. Two weeks later, a colonoscopy was done, showing a normal study. CONCLUSIONS It is important to consider Meckel's diverticulum as a possible cause of sudden abdominal pain in adults, as it can have catastrophic consequences. Furthermore, studies have shown that Meckel's diverticulum and Sjögren syndrome are distinct entities. Yet, both conditions can present with symptoms of intestinal obstruction.

BACKGROUND Internal root resorption (IRR) is a rare dental condition characterized by the progressive resorption of dentin within the root canal, often resulting from infection, trauma, or orthodontic treatment. When IRR progresses to perforation, it creates a communication pathway with periodontal tissues, necessitating effective endodontic therapy and perforation repair. Bioceramic sealers, known for their biocompatibility and flowability, have emerged as a promising alternative to traditional materials for filling and sealing the root canal system. Although commonly used in conventional root canal treatments, their application in filling resorptive defects is novel and unexplored in existing studies. CASE REPORT In the case report presented, a perforating IRR was evaluated and treated. The diagnosis was confirmed using a combination of periapical radiographs and cone-beam computed tomography (CBCT), ensuring precise lesion identification. The treatment involved root canal debridement with ultrasonic activation of irrigants and the application of calcium hydroxide as an intracanal medicament. Bioceramic sealers were utilized for obturation instead of traditional obturation techniques, effectively sealing the resorptive defects and repairing root wall perforations. CONCLUSIONS The case report highlights the novel use of bioceramic sealers in managing perforating IRR. While these sealers are gaining recognition in conventional root canal therapy, their application in resorptive defects represents an innovative approach, underscoring the need for further randomized clinical trials to validate their clinical effectiveness.