Multiple cardiac rhabdomyomas not associated with tuberous sclerosis in a dizygotic twins: a case report.

BACKGROUND: Rhabdomyomas comprise the majority of cardiac tumors in fetuses and are found in association with tuberous sclerosis complex. More than 90% of fetuses and neonates with multiple cardiac rhabdomyomas have signs of tuberous sclerosis complex. However, solitary cardiac rhabdomyoma cases are largely unrelated to tuberous sclerosis complex. Here, we report a case involving multiple cardiac rhabdomyomas not associated with tuberous sclerosis complex in a dizygotic twin. CASE PRESENTATION: A 36-year-old Japanese woman was diagnosed with a dizygotic twin pregnancy in the first trimester. Consistent with dizygosity, the fetal sex was discordant (male and female). At 27 weeks of gestation, hydrops and multiple echogenic cardiac masses were noted in the male baby, with the largest mass measuring 34 × 30 mm. The female fetus appeared normal. The cardiac masses enlarged gradually with the progression of the hydrops. At 32 weeks of gestation, intrauterine death of the male fetus was confirmed. The next day, autopsy of the male fetus was performed after cesarean section. Three well-demarcated white-tan-colored nodules were formed in the ventricular walls and interventricular septum, with the largest nodule (40 × 30 mm) in the left ventricular wall. Histologically, these lesions were diagnosed as rhabdomyomas. CONCLUSIONS: We encountered a case involving multiple cardiac rhabdomyomas arising in one of dizygotic twin fetuses. Unlike most reported cases of multiple cardiac rhabdomyomas, this case was not accompanied by tuberous sclerosis complex. To the best of our knowledge, this is the first case report of multiple cardiac rhabdomyomas that developed in only one of dizygotic twins in the English literature.

Rapid Progression of Lung Cancer Following Emergency Caesarean Section Led to Postpartum Acute Respiratory Failure.

Our case patient was a 38-year-old pregnant Japanese woman who underwent emergency Caesarean section because of massive vaginal bleeding due to a low-lying placenta. Immediately after delivery, she presented with rapidly progressive dyspnea. Contrast-enhanced computed tomography revealed bilateral pleural effusion, lung nodules, multiple liver tumors, and multiple osteolytic lesions. Accordingly, epidermal growth factor receptor-mutant advanced lung adenocarcinoma was diagnosed. This report highlights the occurrence of rapid progression of lung cancer following delivery that led to postpartum acute respiratory failure, rather than due to pulmonary thromboembolism associated with the existing deep venous thrombosis of the inferior vena cava.

Rupture of hidden abnormal myometrial vessels during cesarean delivery of a patient with subserosal leiomyoma: A possible pathogenesis of sudden-onset disseminated intravascular coagulation.

We report a case of sudden-onset disseminated intravascular coagulation during cesarean delivery for a patient with a subserosal leiomyoma. Rupture of hidden anastomotic vessels resulted in a significant decrease in fibrinogen levels and uncontrolled bleeding. Uterine venous flow disturbance caused by subserosal leiomyoma compression can possibly cause such a situation.

Uterine arteriovenous malformation formed in a large uterine cervical myoma.

Arteriovenous malformation (AVM) can arise in various organs, particularly the brain, but it is rare in the uterus. Uterine AVM is potentially lethal and is generally associated with uterine trauma, such as dilatation and curettage, therapeutic abortion or uterine surgery. On the other hand, uterine myoma is the most common benign gynecological tumor, but uterine cervical myoma is rare and grows in the extraperitoneal space, with development of complex capillary networks within the tumor. Cervical myoma surgery is therefore a difficult operation with a risk of massive bleeding. We report herein a patient with uterine AVM formed within a large cervical myoma in a postmenopausal woman. The patient was a 55-year-old Japanese woman who complained of lower abdominal distension. Ultrasonography, computed tomography and magnetic resonance imaging showed an 18 × 20-cm uterine cervical tumor with dilatation of numerous vessels. Pelvic angiography was scheduled to provide accurate diagnosis and to minimize intraoperative blood loss. In fact, preoperative pelvic angiography allowed us to identify the true feeding artery and drainage veins. Occlusion of the feeding artery with a balloon device is effective in decreasing intraoperative bleeding. Abdominal total hysterectomy was performed as the surgical management of this uterine AVM. Prophylactic endovascular balloon occlusion of the ipsilateral internal iliac artery reduced the amount of hemorrhage during surgery, although blood transfusion was needed in our patient. In conclusion, preoperative embolosclerotherapy should be considered as a treatment option in patients with AVM present in a large uterine cervical myoma.