American Journal of Case Reports最新文献

BACKGROUND Conjoined twins are a rare congenital anomaly with various types, depending on the connection site. Managing these cases requires a structured approach leading to separation surgery. This report provides a detailed description of the anesthetic strategies and challenges of pygopagous conjoined female twins who underwent 2 surgeries before separation at 17 weeks of age. CASE REPORT The female twins were conjoined at the sacral level (S4), with fused thecal sacs at L5 and cord tethering, with the conus terminating below L3 in both patients. They had separate rectal ampullae that fused into a single anal canal in the midline in a Y formation. The babies underwent 2 surgeries prior to separation: meningocele repair for twin B and anal canal dilation and sphincter mapping for both twins, followed by a ventriculoperitoneal shunt insertion for twin B. The separation surgery included dividing the bony vertebral fusions and dural sacs, untethering the spinal cords and nerves, and correctly allocating the intrapelvic muscles, guided by sphincter muscle mapping. Anesthesia was managed by 2 distinct teams each time, with duplicated equipment and color-coded medications to prevent errors. Due to the critical condition of twin B, general anesthesia was administered to her first. The report also addresses the challenges faced during the 3 surgical procedures. CONCLUSIONS Anesthetic management for pygopagus twins presents complex challenges. Despite limited experience with similar cases, successful management was achieved through planning, effective communication, and rehearsal of unfamiliar setups. Attention to detail and involvement of highly experienced teams were crucial to the success of the procedures.

BACKGROUND The extraction of impacted supernumerary teeth requires precision and accuracy to mitigate iatrogenic damage to crucial anatomical structures during dental surgical procedures, thereby enhancing postoperative healing outcomes. Dynamic navigation systems (DNS) have been applied in dentistry in maxillofacial fractures, orthognathic surgery, root canal treatment, and endodontic surgery. CASE REPORT A 22-year-old female patient visited our department to assess and manage unerupted third molars. An initial cone beam computed tomography (CBCT) scan was obtained. Radiographic and clinical examinations showed the presence of a supernumerary tooth impacted on the lingual side between the root of the lower second premolar and the lower first molar and bilateral lower impacted third molars. The patient agreed to removal of these teeth. To perform the treatment planning of this case and to guide the surgeon intraoperatively, a dynamic surgical navigation system was recommended for surgical extraction of a supernumerary tooth and the impacted third molars. CONCLUSIONS The dynamic navigation system coupled with a high-speed contra-angle handpiece for the extraction of supernumerary teeth is a personalized, digitally-driven, precise, minimally invasive, and efficient treatment approach. In this case, the DNS and the high-speed contra-angle handpiece were seamlessly integrated to facilitate visualization of the surgical procedure, thereby safeguarding of surrounding vital anatomical structures while enhancing patient comfort.

BACKGROUND Hyperthermia is strongly associated with a worse neurological outcome in traumatic brain injury (TBI) and can be exacerbated by shivering. However, effective treatments for uncontrolled shivering have yet to be established. We successfully treated a patient with severe TBI and repeated episodes of shivering using dantrolene sodium hydrate (dantrolene). CASE REPORT A 28-year-old healthy male sustained an acute subdural hematoma with a midline shift following a traffic accident. He underwent emergency evacuation of the hematoma and craniectomy and was admitted to the Intensive Care Unit. The patient experienced severe shivering on the first day of hospitalization. Neuroprotective drugs were administered, and targeted temperature management at normothermia was initiated, initially controlling the shivering. On day 9 of hospitalization, shivering recurred, with a high fever. Despite the reintroduction of general anesthesia and targeted temperature management, the shivering was intractable. We administered dantrolene, which successfully eliminated the shivering. After the intervention, the patient's body temperature and intracranial pressure were well managed. The patient was subsequently transferred to a rehabilitation hospital, with a favorable neurological outcome 70 days after the injury. CONCLUSIONS This case report demonstrates that dantrolene is an effective option for managing uncontrollable shivering in the context of intracranial pressure control after severe brain injury. This finding suggests the potential for the broader use of dantrolene in similar clinical scenarios and supports further investigation of its efficacy and mechanisms of action in TBI care.

BACKGROUND Hemophagocytic lymphohistiocytosis (HLH) is a rare and life-threatening syndrome that can occur either in genetically predisposed individuals (primary HLH) or in particular conditions in immunocompromised patients (secondary HLH). Secondary HLH is very rare among solid organ transplant recipients, especially in lung transplant recipients, for whom its prognosis is dismal. CASE REPORT We report an exceptional case of HLH occurring unusually late following lung transplantation. At 11 years after transplantation, the patient, aged 67 years, presented with pancytopenia, fever, hyperferritinemia, and hypertriglyceridemia, along with splenomegaly. Exhaustive serological and PCR tests ruled out active infection. Bone marrow aspirates showed signs of hemophagocytosis, and bone marrow biopsy was suggestive of post-transplant lymphoproliferative disorder (PTLD). Timely treatment with etoposide and corticosteroids led to a transient improvement in the patient's clinical condition, and rituximab was initiated as a treatment for PTLD. Unfortunately, pancytopenia persisted for weeks, and the patient died from refractory septic shock, despite appropriate intravenous antibiotics. Autopsy revealed lymphoid infiltration of the mediastinal lymph nodes, liver and bone marrow, with some lymphocytes expressing CD3. A final diagnosis of Ann-Arbor stage IV non-EBV-mediated monomorphic T-cell PTLD was established. CONCLUSIONS This case report highlights a very unusual and fatal presentation of HLH in a lung transplant recipient, secondary to a T-cell PTLD. Indeed, HLH is typically seen as infection-related and reported to occur in the initial months following transplantation. To date, no guidelines or consensus exist regarding the management of immunosuppression regimen in solid organ transplantation.

BACKGROUND Managing unique anatomical structures, such as the nasopalatine canal, remains key for successful implant placement and long-term functionality. Topical oral oxygen therapy (TOOT) has gained attention for its antibacterial, regenerative properties, and ability to accelerate wound healing. This report presents a case of postoperative palatal necrosis successfully treated with TOOT oxygen-active gel (blue®m) CASE REPORT A 33-year-old male patient presented with the primary concern of needing rehabilitation of the anterior maxillary region. Clinical and imaging examinations revealed a deficient maxillary ridge and an enlarged incisive foramen. The treatment plan involved nasopalatine deflation with guided bone regeneration using particulate synthetic bone graft and a collagen membrane. Seven days after surgery, he returned with minimal pain but had necrotic tissue on the palate. The necrotic tissue and a portion of the contaminated biomaterial were removed, and the area was thoroughly cleansed with a saline solution. A thick layer of active oxygen gel (blue®m) was then applied to the palatal lesion. The patient was instructed to apply the gel 3 times daily for 30 days and attended follow-up appointments every 2 days. After 12 days, rapid healing and significant clinical improvement were observed, with the patient reporting no pain or sensitivity. By day 34, the lesion had fully closed, and re-epithelialization was achieved. CONCLUSIONS Our patient had complete resolution of palatal necrosis after nervus deflation using TOOT with active oxygen gel (blue®m), and this therapy seemed to accelerate the healing process.

BACKGROUND Refeeding syndrome (RFS) and starvation-associated injuries are significant complications in malnourished patients. Severe weight loss after obesity surgery is frequently associated with malnutrition, consequently increasing the likelihood of RFS and starvation-related injuries as postoperative complications. RFS and starvation-induced injury in a single patient has rarely been reported. In this paper, we present, for the first time, a case of hepatic injury attributed to both refeeding syndrome and starvation-induced hepatic injury in a malnourished woman following bariatric surgery. CASE REPORT A 27-year-old female patient was admitted to the hospital for severe malnutrition after sleeve gastrectomy. Her body mass index (BMI) dropped from 37.2 kg/m² to 12.4 kg/m² 1 year after surgery. After nutritional supplementation, her liver enzymes levels increased significantly, with severe hypophosphatemia suggesting the development of RFS. During the calorie restriction treatment for RFS, the patient unexpectedly exhibited the recurrent increase of liver enzyme levels and severe reduction in body weight, albumin, and hemoglobin, which is considered to be caused by starvation-induced injury during the treatment of RFS. Following precise nutritional re-supplementation, her liver enzyme levels were dramatically decreased, with significant elevated hemoglobin and albumin levels at discharge and during the follow-up visit. CONCLUSIONS Chronic malnutrition and extreme weight loss can occur following bariatric surgery. Our report highlights the potential for RFS and starvation-related liver injuries as postoperative complications for high-risk patients after bariatric surgery. Liver injury can occur in both RFS and starvation-induced hepatitis. Nutrition initiation and supplementation should be carefully balanced in high-risk patients during nutritional treatments.

BACKGROUND Adult colonic intussusceptions are relatively rare and are mostly caused by organic structures that serve as lead points. However, the pathogenesis of adult intussusception is not fully understood, and no cases of retrograde colonic intussusception without pathological abnormalities or associations with colonoscopy have been reported. CASE REPORT A 74-year-old woman presented with abdominal distension and constipation. Abdominal computed tomography (CT) revealed marked dilatation of the right and sigmoid colon, initially suggesting volvulus of the sigmoid colon. Observation of the left colon revealed no abnormal findings on the colonoscopy. Due to the persistence of abdominal symptoms from right colon dilatation, another colonoscopy was performed, and a transanal drainage tube was inserted into the transverse colon. Enterography showed a steep contrast interruption in the descending colon, which was missed at this time. The patient's abdominal pain worsened 3 days after removal of the drainage tube. Retrograde intussusception of the sigmoid colon was discovered on abdominal CT, and a laparoscopic left hemicolectomy was performed. Pathological examination revealed multiple ulcers in the superimposed area, but no abnormal organic findings that could be considered as a lead point were found. In this case, the stretching technique and/or shear stress on the sigmoid colon by a second colonoscopy may have contributed to the development of this condition. CONCLUSIONS This is the first report of colonoscopy-associated retrograde colonic intussusception without organic abnormalities. Although much is unknown about the pathogenesis in this case, it may provide new insights into the pathogenesis of intussusception.

BACKGROUND Infective endocarditis (IE) is a severe, life-threatening, and relatively common complication after valve replacement operations, with incidence rates varying between 1.8% and 5.8%, with an in-hospital mortality rate of up to 20%. Common microorganisms are (listed by decreasing incidence) Streptococci, Staphylococcus aureus, Enterococci, bacteria of the HACEK group, and fungi. Treatment of IE is complex, typically involving prolonged courses of antibiotics. However, in cases of aortic prosthetic valve endocarditis, root abscess formation with involvement of the aorto-mitral skeleton is not uncommon and complex surgical intervention is required. One of the notable advancements in surgical management is the use of homografts for aortic root endocarditis. CASE REPORT We report the first case series of 8 patients successfully operated on for prosthetic valve endocarditis with extensive aortic root abscess formation in Greece at Onassis Cardiac Surgery Center with the use of aortic homograft. All cases were redo surgeries and had good outcomes. Interestingly, one of the cases had extensive aortic root involvement with abscess formation extending to the aorto-mitral fibrous skeleton, requiring aortic root replacement with homograft, aorto-mitral skeleton reconstruction with bovine pericardium and mitral valve replacement with a mechanical prosthesis. Two other patients required concomitant coronary bypass grafting of the right coronary artery with reversed saphenous vein grafts. CONCLUSIONS Aortic root replacement with aortic homograft is the preferred choice for prosthetic valve endocarditis with aortic root abscess formation. Despite the technical complexity needed for implantation, this option offers a second chance for survival in patients with this challenging condition.

BACKGROUND Pseudomelanosis duodeni (PD) is a rare incidental endoscopic finding characterized by flat, discrete speckles of dark pigment, usually in the proximal duodenum. PD is associated with chronic conditions, including end-stage renal disease, hypertension, and diabetes, and with certain medications, including oral iron supplements and sulfur-containing antihypertensives. CASE REPORT A 56-year-old woman presented with lower abdominal pain, intermittent rectorragia, and a history of peptic ulcer disease and iron-deficiency anemia, treated with oral iron supplements. She was hemodynamically stable, and laboratory test results were pertinent only for microcytic anemia. In the workup of iron-deficiency anemia, esophagogastroduodenoscopy was performed, showing findings suspicious for PD, which was confirmed by pathology. Colonoscopy revealed large internal hemorrhoids, and hemorroidectomy was scheduled during the same admission. Duodenum biopsies showed edematous villosities and large clusters of pigmented macrophages, with golden-brown cytoplasm positively stained with Perl's Prussian blue stain, indicating the presence of iron inside the macrophages. These findings confirmed the PD diagnosis. The pigment in PD is composed primarily of iron and sulfur, with iron being the main component, as seen in staining. In our case, we present findings of PD along with lower gastrointestinal bleeding manifesting as hemmoroidal bleeding. Giving the anatomical nature of hemorrhoids and that our patient was on oral iron therapy, the most likely mechanism behind the development of PD in our case was related to the oral iron therapy. CONCLUSIONS PD is a benign disorder of the duodenum. Further studies are needed to investigate its long-term outcomes and to formulate optimal management strategies.

BACKGROUND Recurrent heterotopic pregnancy is a serious and rare pregnancy complication that occurs after in vitro fertilization and embryo transfer, posing a great threat to the safety of the mother and intrauterine fetus. We report a case of recurrent heterotopic pregnancy after in vitro fertilization and embryo transfer. We also reviewed the literature to explore the causes, diagnosis, treatment, and prevention of recurrent heterotopic pregnancy. CASE REPORT A 32-year-old woman with tubal factor infertility underwent in vitro fertilization and embryo transfer. Oocyte extraction was performed twice, while embryo transfer was performed 4 times, with 2 embryos transferred each time. No pregnancies occurred during the 2 fresh embryo transfer cycles; however, heterotopic pregnancies occurred during both frozen-thawed embryo transfer cycles. Ultrasonography detected only the first heterotopic pregnancy, at 6 weeks after embryo transfer. As the intrauterine embryo had stopped developing, laparoscopically monitored dilatation and curettage and removal of the affected fallopian tubes were performed. The second heterotopic pregnancy was detected 3 weeks after embryo transfer, guided by the ultrasound examination conducted earlier. After timely surgical treatment, a live birth occurred. CONCLUSIONS This case and review of the literature elucidates the importance of considering the possibility of a heterotopic pregnancy during ultrasound examinations, especially in patients who utilized assisted reproductive technology to transfer multiple embryos.