Tailgut cyst of retrorectal space is uncommon and its classification and prognosis are unclear. The lesion usually consists of a multiloculated cyst lined by squamous, transitional, and glandular epithelium. Disorganized fascicles of smooth muscle may be seen in the wall. Glomus coccygeum, normally located at the tip of coccyx, is incidentally discovered in the presacral mass including tailgut cyst. We report a case of tailgut cyst in a 35 year old female who had internal hemorrhoid and intermittent anal pain for 3 years. Pelvic computerized tomography revealed a 3x2 cm sized mass in the presacral area. The mass was multicystic and lined by squamous, transitional, and peudostratified ciliated columnar epithelium with inflammatory cells and scattered smooth muscle in the wall. Dermal adnexal structures were not present, which excluded dermoid cyst. A glomus body was identified in the surrounding soft tissue.
Female ; Humans ; Cysts

PURPOSE: Recent studies have proved that bladder compliance is closely related to the collagen content of the bladder. It has been shown through human fibroblast, condrocyte, and rat myofibroblasts that interferon(IFN)-gammadecreased collagen synthesis. The objective of this study was to investigate the alteration of collagen deposition and gene expressions in collagen types I and III by partial bladder outlet obstruction and to compare the changes during IFN-gamma treatment. MATERIALS AND METHODS: Following 4 weeks of partial bladder outlet obstruction in 200-250gm. SD rats, the bladder tissues were evaluated for collagen deposition by an immunohistochemical staining using the polyclonal antibodies and the messenger ribonucleic acid(mRNA) contents of the collagen type I and III were evaluated by Northern hybridization. Recombinant IFN-gamma(100,000 units) was administered once a day for 4 weeks by subcutaneous injection. RESULTS: In the control bladders, the immunohistochemical staining for type I and type III collagen protein showed an intense localization in the lamina propria with modest localization in the inter-fascicular region. With obstruction type I and type III collagen staining intensified, especially in the inter-fascicular region. The IFN-gamma administration reduced the inter-fascicular deposition of collagen type I and III. There was an up-regulation of collagen type I and III gene expression after the obstruction. In the obstructed bladders proalpha1(I) and proalpha1(III) collagen mRNA intensity was increased 3.4 and 5.1 folds than the control groups, respectively. This up-regulation was down-regulated by IFN-gamma administration. CONCLUSIONS: The down regulation of collagen types I and III gene expression, and decreased protein deposition after IFN-gamma treatment demonstrated that the effect of IFN-gamma was, in part, transcriptionally regulated. These results suggest that the recombinant IFN-gamma may be beneficial in the treatment of patients with poorly compliant bladder.
Animals ; Antibodies ; Collagen Type I ; Collagen Type III ; Collagen* ; Compliance ; Down-Regulation ; Fibroblasts ; Gene Expression ; Humans ; Injections, Subcutaneous ; Interferon-gamma ; Interferons* ; Mucous Membrane ; Myofibroblasts ; Rats* ; RNA, Messenger ; Up-Regulation ; Urinary Bladder Neck Obstruction ; Urinary Bladder*

PURPOSE: To evaluate the NT in the first trimester associated with fetal chromosomal abnormality, fetal structural abnormality and adverse pregnancy outcome. METHODS: From Mar. 1993 to Dec. 1997, 133 cases of increased NT were reviewed retrospectively. All these fetues were taken a karyotyping and/or a high resolution ultrasonography for associated anomaly. The NT was measured by transvaginal ultrasonography(TVS) and transabdominal ultrasonography(TAS) if difficulty was encountered with TVS in the period of the 10th-14th week of gestation. The increased NT is approved when it is over 3mm. RESULTS: Of these 133 cases, 102 cases(76.7%) revealed normal outcomes and 31 cases(23.3%) revealed abnormal outcomes such as chromosomal abnormality, structural abnormality and adverse pregnancy outcome, The mean value of NT was 3.44+/-0.68mm in the normal group and 4.93+/-2.70mm in the abnormal group, respectively. The chromosomal abnormalities were found in 16 of the 31 cases: nine Down SD, two Patau syndrome, one Tuner synrome, one triploidy, one 46, XX,i(18q), one 47,iso(Xp)/46,XX one confined placental mosaicism. In 12 cases of the 31 cases, the structural anomalies were detected. Of these 12 cases, 3 cases were eventually developed to cytsic hygroma, 3 cases were cardiac anomaly, 3 cases were renal anomaly, 1 case was congenital diaphragmatic hernia, 1 case was skeletal dysplasia and 1 case was suspected syndrome. The adverse pregnancy outcome such as intrauterine growth restriction, intrauterine fetal death and preterm labor was revealed in 3 cases. In the cases of NT over 7mm, the result was 100% abnormal outcome. CONCLUSION: Increased NT in the first trimester could be a highly efficient the predictor for detection of chromosomal abnormality, structural anomaly and adverse pregnancy outcome.
Chromosome Aberrations ; Female ; Fetal Death ; Hernia, Diaphragmatic ; Humans ; Karyotyping ; Lymphangioma, Cystic ; Mosaicism ; Nuchal Translucency Measurement ; Obstetric Labor, Premature ; Pregnancy ; Pregnancy Outcome* ; Pregnancy Trimester, First ; Pregnancy* ; Retrospective Studies ; Triploidy ; Ultrasonography

BACKGROUND: Ebstein's anomaly is characterized by various degrees of apical displacement of the proximal attachment of the tricuspid valve. The disease has an extremely variable course in presenting itself. Therefore, we examined the morphologic and clinical features of Ebstein's anomaly as presented in the fetus to define the factors that determine its outcome. METHODS: We reviewed the history and echocardiographic studies of 10 fetuses (mean gestational age 28.0 weeks) diagnosed in utero with Ebstein's anomaly. The reason for referral was cardiomegaly on routine obstetric scanning in 5 cases, fetal arrhythmia in 3 cases, small main pulmonary artery in 3 cases, vessel view in 1, and twin pregnancy complicated by polyhydramniosis in 1. RESULTS: Of the 10 cases, pregnancy was terminated after diagnosis in 5, 2 cases were carried to term, and 1 case was at 34 weeks of gestation. We were unable to follow-up 2 cases. There were no intrauterine deaths and no misdiagnoses. Massive cardiomegaly and severe tricuspid regurgitation were detected in 7 cases; mild cases were detected in two, and one was nearly normal. Associated cardiac lesions included pulmonary artresia in 4 cases, pulmonary stenosis in 3, and pulmonary hypoplasia in 1. The cases with right ventricular outflow tract (RVOT) obstruction showed a tendency to develop more cardiomegaly and tricuspid regurgitation than with normal RVOT. There was no abnormality in other organ systems and karyotyping. CONCLUSION: Although increased cardiothoracic ratio and associated lesions of the right ventricular outflow tract contribute to the poor outcome in the prenatally detected cases, the absence of these features does not always indicate a good prognosis because progression of the disease can occur with advancing gestational age. No absolute measurement or single echocardiographic feature emerged as a consistent predictive factor for prognosis.
Arrhythmias, Cardiac ; Cardiomegaly ; Diagnosis ; Diagnostic Errors ; Ebstein Anomaly* ; Echocardiography* ; Fetus* ; Follow-Up Studies ; Gestational Age ; Karyotyping ; Pregnancy ; Pregnancy, Twin ; Prognosis ; Pulmonary Artery ; Pulmonary Valve Stenosis ; Referral and Consultation ; Tricuspid Valve ; Tricuspid Valve Insufficiency

As the number of people vaccinated increases, people who complain of adverse reactions continue to occur. We experienced a case characterized by low blood pressure, persistent fever, edema due to increased systemic vascular permeability, and systemic inflammation confirmed by image and laboratory examinations after ChAdOx1 coronavirus disease 2019 (COVID-19) vaccination. The diagnostic criteria for multisystem inflammatory syndrome (MIS) in adults are known as fever of 3 days or more in adults, 2 or more mucocutaneous/ gastrointestinaleurologic symptoms, elevation of inflammatory markers, and clinical/ imaging diagnosis of heart failure. A 67-year-old man who was medicated for hypertension and diabetes was admitted complaining of fever, maculopapular rash, diarrhea, headache, chills, and dizziness 6 days after the first vaccination of ChAdOx1 nCoV-19 in Korea.The COVID-19 test was negative but with low blood pressure, leukocytosis, skin rash, pulmonary edema, and increased inflammation markers. His lab findings and clinical course were consistent with those of MIS after COVID-19 vaccination. He was medicated with methylprednisolone 1 mg/kg and diuretics and recovered rapidly. He was discharged after 2 weeks and confirmed cure at outpatient clinic. We report an MIS case after COVID-19 vaccination in Korea.

Cytologic finding of 2 cases of metastatic leiomyosarcoma diagnosed by fine needle aspirtion cytology are reported. Case 1 is pleomorphic leiomyosarcoma which had metastsized to the liver from the stomach of a 54-year-old male patient. The cytologic features showed highly cellular aspirates with nuclear pleomorphism and interlacing patern. Case 2 is low grade leiomyowsarcoma that occurred in the uterus of a 43-year female patient and metastsized to both lungs. The aspirates were less cellular than that of case 1, and showed spindle cells with minimal pleomorphism, but cell block revealed interlacing patterns of smooth muscle cells with occasional mitosis.
Biopsy, Fine-Needle* ; Female ; Humans ; Leiomyosarcoma* ; Liver ; Lung ; Male ; Middle Aged ; Mitosis ; Myocytes, Smooth Muscle ; Needles ; Neoplasm Metastasis ; Stomach ; Uterus

In small children with left ventricular outflow tract obstruction, a few methods of surgical treatment could be considrred. The pulmonary autogrart provides a promising options for aortic valve replacement as part of the aortoventriculoplasty procedure in children. We report a successfully treated congenital aortic stcnoinsufricicncy with severe left ventricular dysfunction in an early infant with the aortoventriculoplasty using thc pulmonary autograft (the Ross-Konno procedure).
Aortic Valve ; Autografts* ; Child ; Dronabinol ; Humans ; Infant ; Transplantation, Autologous ; Ventricular Dysfunction, Left ; Ventricular Outflow Obstruction

Esophageal involvement in tuberculosis is rare, and the clinical presentation may mimic an esophageal submucosal tumor. A 30-year-old woman presented with dysphagia for 1 month. At esophagoscopy, a 3-cm subepithelial mass with normal covering mucosa was found 28~25 cm from the upper incisors. We diagnosed the lesion as a submucosal tumor of the esophagus and performed endoscopic ultrasonography 1 week after the first examination. The second endoscopy showed a large, linear ulceration on the same subepithelial mass. The histologic examination obtained following an endoscopic biopsy revealed chronic granulomatous inflammation, and a molecular nested PCR study for Mycobacterium tuberculosis was positive. The final diagnosis was mediastinal tuberculous lymphadenitis with invasion into the esophagus. We suggest the use of molecular biology techniques when there is a strong clinical suspicion of tuberculosis and difficulty in arriving at a definite diagnosis.
Adult ; Biopsy ; Deglutition Disorders ; Endoscopy ; Endosonography ; Esophagoscopy ; Esophagus ; Female ; Humans ; Hydrazines ; Incisor ; Inflammation ; Mediastinum ; Molecular Biology ; Mucous Membrane ; Mycobacterium tuberculosis ; Polymerase Chain Reaction ; Tuberculosis ; Tuberculosis, Lymph Node ; Ulcer

Behcet's disease is chronic, relapsing, multi-systemic inflammatory vasculitis. Unlikely the other autoimmune diseases, gastric non-Hodgkin's lymphoma has rarely been reported in a patient with Behcet's disease. We report here on a 40-year-old man who had Behcet's disease for several years and who suffered recurrent oral ulcer, genital ulcer and skin lesion. The patient was treated with azathioprine and intermittent oral steroid. He complained of epigastric pain for 1 month, and he had a large ulcerative lesion seen on endoscopic evaluation. We confirmed the lesion was a gastric, diffuse, large B cell lymphoma by endoscopic biopsy. The patient was treated with the rituximab-CHOP regimen, and he has achieved a complete remission for 3 years. We report here on a case of gastric non-Hodgkin's lymphoma in a patient with Behcet's disease and who also had an ileocecal valve ulcer.
Adult ; Autoimmune Diseases ; Azathioprine ; Biopsy ; Humans ; Ileocecal Valve ; Lymphoma ; Lymphoma, B-Cell ; Lymphoma, Non-Hodgkin ; Oral Ulcer ; Skin ; Stomach ; Ulcer ; Vasculitis

Paradoxical embolism is a kind of stroke caused by embolism of thrombus of venous origin through a lateral opening in the heart, such as a patent foramen ovale (PFO). Although the most frequent manifestation of paradoxical embolism is cryptogenic stroke, noncerebral paradoxical embolism is also associated with PFO. We experienced a case of cryptogenic renal infarction in a previously healthy 70-year-old man. He had no cardiac thrombus on transthoracic echocardiography and electrocardiogram revealed a normal sinus rhythm. Because it was cryptogenic renal infarction, we performed transesophageal echocardiography with microbubble test. Microbubble test using agitated saline proved the presence of right-to-left shunt and patent foramen ovale was diagnosed. We also performed lower leg doppler ultrasonogram, but there was no evidence of deep vein thrombosis. Although only the presence of a right-to-left shunt is not enough to establish the diagnosis of paradoxical embolism, it is uncommon for the source of the embolism to be identified. In this case, we concluded that paradoxical embolism is the cause of renal embolism. We report paradoxical renal embolism through PFO with review of relevant literatures.
Aged ; Dihydroergotamine ; Echocardiography ; Echocardiography, Transesophageal ; Electrocardiography ; Embolism ; Embolism, Paradoxical ; Foramen Ovale, Patent ; Heart ; Humans ; Infarction ; Kidney ; Leg ; Microbubbles ; Stroke ; Thrombosis ; Venous Thrombosis