A 34-year-old parous woman developed high fever and threatened preterm labor after a 1-day trip, for which she was receiving prenatal care at a hospital. Three days after onset, at 24 4/7 weeks of gestation, she was transferred to our hospital in an emergency. Soon after the woman's arrival at our hospital, the infant was spontaneously stillborn via a transvaginal delivery. Laboratory tests revealed severe maternal disseminated intravascular coagulation with renal and liver insufficiency. Histopathologic examination of the placenta revealed vast fibrin deposition and remarkable neutrophilic infiltration in the intervillous space, suggesting a rare bacterial infection caused by Arthrobacter spp. The bacteria were predominantly detected in the placenta and maternal blood serum by common bacterial 16S rRNA sequencing after polymerase chain reaction amplification. We report the first case, to our knowledge, of bacteremia with Arthrobacter spp., which may lead to maternal disseminated intravascular coagulation and intrauterine fetal death.
Malignant transformation is rarely seen in the disease course of mature cystic teratoma (MCT) of the ovary. Adenocarcinoma arising from MCT is especially rare. We herein present the case of a premenopausal woman with a mucinous borderline-like tumor arising from a MCT. Based on the histological transition between the borderline-like tumor and gastrointestinal elements of the MCT, we consider that the tumor derived from teratomatous gastrointestinal epithelium. Immunohistochemistry showed that the proliferating mucinous cells were diffusely positive for cytokeratin 20 and partially positive for cytokeratin 7. MUC5AC was partially positive, whereas MUC2 and MUC6 were positive in a small number of tumor cells. The immunophenotype of cytokeratins and mucins in the present case was compatible with malignant transformation of the teratomatous gastrointestinal epithelium.
Lymphocytic adenohypophysitis is a very rare autoimmune disease characterized by an anterior pituitary infiltration of lymphocytes that cause various symptoms, such as headache, visual disturbance, and hypopituitarism. Lymphocytic adenohypophysitis is more common in women and has a remarkable association with the perinatal period. Recently, we experienced a case of lymphocytic adenohypophysitis shortly after delivery. A 37-year-old primiparous woman delivered her baby at 38 weeks' gestation. The next morning, she presented symptoms of hypoglycemia and hypothermia. Blood testing revealed hypoadrenalism and hypothyroidism. Pituitary magnetic resonance imaging showed characteristic findings consistent with lymphocytic adenohypophysitis. We immediately initiated medication with steroids and levothyroxine. Currently, her pituitary size has normalized and its function has fully recovered. Physical stress resulting from labor and delivery may unmask subclinical lymphocytic adenohypophysitis. Practitioners should keep in mind the possibility of lymphocytic adenohypophysitis in any pregnant women with symptoms of hypoglycemia and hypothermia after delivery.
Cystic adenomyosis, a cystic lesion in the myometrium, is a rare disease that is characterized by intense dysmenorrhea. Here we report a case of a woman diagnosed with adnexa chocolate cyst that was found to be cystic adenomyosis during laparoscopic surgery. The case is of a 37-year-old woman, gravida 0 and para 0. When she was 32 years old, she visited a nearby hospital for dysmenorrhea. She was diagnosed with adnexa chocolate cyst in both sides and was followed up with oral low dose of estrogen-progestin (LEP). Five years later, she was suggested to undergo surgery because of the increasing size of the cysts. During the laparoscopic surgery, we found that the cystic lesion was not typical, and the connection with the uterus was clear. The pathological findings were consistent with the diagnosis of cystic adenomyoma. We suggest that when cystic lesions are identified with severe dysmenorrhea, the connection with the uterus and the possibility of cystic adenomyoma should be considered.
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