A 2-month-old female infant presented with a single hemorrhagic crusted papule on the chin present since birth. No visceral involvement could be demonstrated. The lesion involuted spontaneously with scarring in 3 months. Mononuclear cells in the cutaneous infiltrate were Langerhans cells with typical Birbeck granules which positively stained with S-100 protein. This case is the solitary type of congenital self-healing reticulohistiocytosis.
Case Report ; Female ; Histiocytosis, Langerhans-Cell/*congenital/pathology ; Human ; Infant ; Skin Diseases/*congenital/pathology ; Support, Non-U.S. Gov't

An autopsy case of disseminated HSV type 2 infection occurring in a neonate at 32 weeks' gestation, delivered by cesarean section after premature rupture of membrane of 7 days duration, is presented. Herpes simplex virus type 2 was isolated from the vesicular skin lesion. The mother and patient had specific antibody to type 2 herpes simplex virus. Patient's parents had denied any herpetic orolabial or genital lesion during or before this pregnancy. Cultures from the cervical and vaginal swabs of the mother were negative for HSV. Postmortem examination showed hepatic necrosis, skin vesicle, devastating necrotizing inflammation of the brain, chorioretinitis and interstitial pneumonitis.
Autopsy ; Brain/pathology ; Encephalitis/*etiology ; Herpes Simplex/*congenital/pathology ; Humans ; Infant, Newborn ; Infant, Premature, Diseases/*pathology ; Liver/pathology ; Male ; Necrosis ; Skin/pathology

We report an autopsy case of a male fetus with Adams-Oliver syndrome. His mother was a healthy, 31-year-old woman and her family and past histories were unremarkable. Therapeutic termination was done at 28() weeks gestational age due to oligohydramnios detected by antenatal ultrasonography. Chromosomal study revealed normal karyotype. On autopsy, characteristic transverse terminal defect of four extremities was found. Both feet were short and broad. All toes were rudimentary with no nails and fingers were irregularly short. On infantogram, all toe-bones were stubby and rudimentary. The middle and terminal phalanges of 2nd, 3rd +ACY- 5th fingers and the terminal phalange of 4th finger on the right hand were absent. The middle and terminal phalanges of 2nd +ACY- 5th fingers and terminal phalange of 3rd finger were defected on the left hand. His abnormalities were consistent with features of Adams-Oliver syndrome, which has not been reported in Korea.
Abnormalities, Multiple/pathology+ACo- ; Abnormalities, Multiple/embryology ; Abortion, Habitual ; Abortion, Therapeutic ; Adult ; Autopsy ; Case Report ; Female ; Fetal Diseases/pathology+ACo- ; Foot Deformities, Congenital/radiography ; Foot Deformities, Congenital/pathology+ACo- ; Foot Deformities, Congenital/embryology ; Genes, Dominant ; Hand Deformities, Congenital/radiography ; Hand Deformities, Congenital/pathology+ACo- ; Hand Deformities, Congenital/embryology ; Human ; Male ; Oligohydramnios ; Pregnancy ; Scalp/embryology ; Scalp/abnormalities+ACo- ; Skin Abnormalities/pathology+ACo- ; Skin Abnormalities/embryology ; Syndrome

We report an autopsy case of a male fetus with Adams-Oliver syndrome. His mother was a healthy, 31-year-old woman and her family and past histories were unremarkable. Therapeutic termination was done at 28() weeks gestational age due to oligohydramnios detected by antenatal ultrasonography. Chromosomal study revealed normal karyotype. On autopsy, characteristic transverse terminal defect of four extremities was found. Both feet were short and broad. All toes were rudimentary with no nails and fingers were irregularly short. On infantogram, all toe-bones were stubby and rudimentary. The middle and terminal phalanges of 2nd, 3rd +ACY- 5th fingers and the terminal phalange of 4th finger on the right hand were absent. The middle and terminal phalanges of 2nd +ACY- 5th fingers and terminal phalange of 3rd finger were defected on the left hand. His abnormalities were consistent with features of Adams-Oliver syndrome, which has not been reported in Korea.
Abnormalities, Multiple/pathology+ACo- ; Abnormalities, Multiple/embryology ; Abortion, Habitual ; Abortion, Therapeutic ; Adult ; Autopsy ; Case Report ; Female ; Fetal Diseases/pathology+ACo- ; Foot Deformities, Congenital/radiography ; Foot Deformities, Congenital/pathology+ACo- ; Foot Deformities, Congenital/embryology ; Genes, Dominant ; Hand Deformities, Congenital/radiography ; Hand Deformities, Congenital/pathology+ACo- ; Hand Deformities, Congenital/embryology ; Human ; Male ; Oligohydramnios ; Pregnancy ; Scalp/embryology ; Scalp/abnormalities+ACo- ; Skin Abnormalities/pathology+ACo- ; Skin Abnormalities/embryology ; Syndrome