Uterine rupture due to adenomyosis in an adolescent:A case report and review of literature

BACKGROUND Uterine rupture is a fatal medical complication with a high mortality rate.Most cases of uterine rupture occur in late pregnancy or during labor and are mainly related to uterine scarring due to previous surgical procedures.Adenomyosis is a possible risk factor for uterine rupture.However,spontaneous uterine rupture due to severe adenomyosis in a non-gravida-teenaged female has not been reported in the literature to date.CASE SUMMARY A 16-year-old girl was referred to our hospital for acute abdominal pain and hypovolemic shock with a blood pressure of 90/50 mmHg.Radiologic studies revealed a huge endometrial mass with multiple nodules in the lung,suggesting lung metastasis.The patient underwent an emergency total hysterectomy and wedge resection of the lung nodules.Histologically,the uterus showed diffuse adenomyosis with glandular and stromal dissociation.Lung nodules were endometrioma with massive hemorrhage.Immunohistochemistry demonstrated that the tumor cells were positive for PAX8,ER,and PR expression,leading to a final diagnosis of pulmonary endometriosis and uterine adenomyosis.Following surgery,the patient remains in good condition without recurrence.CONCLUSION This is the first case of spontaneous uterine rupture due to adenomyosis in a nongravida adolescent.

Chorioamnionitis caused by Serratia marcescens in a healthcare worker: A case report

BACKGROUND Healthcare workers(HCWs)are at an increased risk for exposure to infections.Serratia marcescens(S.marcescens)is a gram-negative,opportunistic and nosocomial pathogen belonging to the Enterobacterieae family.A few case reports have been published of chorioamnionitis caused by S.marcescens infection.Immunological changes during pregnancy can also affect the risk of infection.However,few studies have examined hospital-acquired bacterial infection in pregnant HCWs.CASE SUMMARY A 33-year-old woman,a resident in anesthesiology,was admitted at 14 wk gestation for fever with chills.She had no medical history other than contact dermatitis of both hands that started from the beginning of the trainee.There was no obvious infection focus and no bacterial growth in blood cultures.She was discharged after 1 wk of empirical antibiotic treatment.At three weeks before the fever started,she had a blister on the site of contact dermatitis on both hands,she applied antibiotic ointment for three days and the blisters had healed.At 19 wk gestation,she had a high fever and was readmitted.Physical examination and image studies were nonspecific and the patient had no other symptoms.S.marcescens grew in blood cultures at 19 wk gestation.Treatment with intravenous antibiotics was started.However,she suffered a miscarriage at 224/7 wk gestation.Pathologically,the amniotic membrane showed chorioamnionitis with a focal infarct.Subsequently,a placenta tissue culture grew S.marcescens.CONCLUSION HCWs can be exposed to pathogens that can cause opportunistic infections such as S.marcescens.Pregnancy affects the immune system,making it susceptible to opportunistic infections.Therefore,pregnant HCWs may require more preventive measures,including hand hygiene and avoid risk factors(ex.wrapping the skin).

Primary duodenal dedifferentiated liposarcoma:A case report and literature review

BACKGROUND Dedifferentiated liposarcoma(DDLPS)is an extremely rare neoplasm that exhibits various morphologies.The tumor is characterized by immunoreactivity to MDM2 and CDK4 and can be confirmed by detecting MDM2 amplification via fluorescence in situ hybridization(FISH).Herein,we report an unusual case of DDLPS arising from the duodenum.CASE SUMMARY A 64-year-old man presented with repeated abdominal pain and weight loss.Radiologic studies revealed a mass of the duodenum involving the pancreas.The patient was treated with pylorus-preserving pancreaticoduodenectomy.Histologically,the tumor showed a high-grade sarcoma.Immunohistochemistry demonstrated that the tumor cells were positive for MDM2 and CDK4 expression.MDM2 amplification was detected via FISH,leading to the final diagnosis of DDLPS.Following surgery,the patient was treated in the intensive care unit due to peritonitis,and died 60 d after surgery.CONCLUSION To the best of the authors’knowledge,this is the first case of primary duodenal DDLPS in Korea and the third case in the English-language literature.Care must be taken not to misdiagnose DDLPS as another high-grade tumor.Liposarcoma should be in the differential diagnosis list.

TFE3-expressing malignant perivascular epithelioid cell tumor of the mesentery:A case report and review of literature

BACKGROUND Perivascular epithelioid cell tumor(PEComa)is a rare mesenchymal tumor that exhibits an epithelioid and spindle cell morphology.The tumor is characterized by immunoreactivity for melanocytic and myogenic markers but can be misdiagnosed as more common tumors with similar characteristics,including gastrointestinal stroma tumors or leiomyosarcomas.Recently,a subset of PEComas has been reported to harbor a transcription factor binding to TFE3 fusion.Herein,we report a rare case of TFE3-expressing malignant PEComa arising from the mesentery.CASE SUMMARY A 50-year-old woman presented with abdominal discomfort for 3 months.Results of laboratory tests were all within the normal ranges,and the patient had no notable medical history.Magnetic resonance imaging revealed a large tumor on the right side of the pelvic floor,which was originally suspected to be a primary ovarian tumor.However,during surgery,the tumor was revealed to have originated from the mesentery.Histologically,the tumor was composed of bundles of spindle cells and sheets of epithelioid cells.Extensive coagulative necrosis and numerous mitotic figures were observed.Immunohistochemistry revealed that the tumor cells were positive for smooth muscle actin,HMB-45,and TFE3 expression.Tumor involvement of the rectal serosa was identified,leading to a final diagnosis of malignant PEComa of the mesentery.Surgical resection was followed by adjuvant chemotherapy.No recurrence or metastasis was observed over a 6-month follow-up period.CONCLUSION Malignant PEComa of the mesentery is extremely rare and should be distinguished from morphological mimics through differential diagnosis and immunohistochemistry.

Cytomegalovirus colitis induced segmental colonic hypoganglionosis in an immunocompetent patient: A case report

BACKGROUND Cytomegalovirus(CMV)colitis is usually seen in immunocompromised patients with risk factors such as human immunodeficiency virus infection,solid organ transplant,inflammatory bowel disease,or malignancy.Therefore,many clinicians usually do not consider the possibility of CMV colitis in immunocompetent patients.We reported a rare case of segmental colonic hypoganglionosis associated with CMV colitis in an immunocompetent patient.CASE SUMMARY A 48-year-old woman with no underlying disease was admitted to our hospital for severe abdominal pain and constipation.Computed tomography of the abdomen showed diffuse dilatation of the small intestine and the entire colon.Initial sigmoidoscopic findings and result of polymerase chain reaction(PCR)for CMV revealed the compatible findings of CMV colitis,the patient was treated with intravenous ganciclovir.After treatment,sigmoidoscopic findings and CMV PCR results improved.However the patient continued to suffered from constipation.Eight months after the initial admission,patient visited the emergency department with severe abdominal pain and imaging revealed aggravation of fecal impaction and bowel dilatation.We performed subtotal colectomy to control patient’s symptom.Histological examination of the resected specimen showed significantly reduced number of mature ganglion cells in the sigmoid colon compared to that in the proximal colon.CONCLUSION Our case demonstrates that CMV colitis can develop even in patients with no other underlying disease,and that CMV colitis can be one of the causes for developing colonic hypoganglionosis.