Intramural pregnancy is a rare and potentially life-threatening form of ectopic pregnancy, often associated with prior uterine surgery. This report describes a case of twin intramural pregnancy following early conception after adenomyomectomy. A 36-year-old nulliparous woman conceived via in vitro fertilization five months after open adenomyomectomy. At 8 weeks of gestation, transvaginal ultrasound and magnetic resonance imaging revealed two gestational sacs implanted within the myometrium at the previous surgical site, without communication with the endometrial cavity. Surgical excision was performed through the prior uterine incision using intraoperative ultrasound guidance and Foley catheter placement to delineate the endometrial cavity. Despite dense adhesions and a thinned myometrium, both sacs were successfully removed without complications, and histopathology confirmed intramural pregnancy. After postoperative gonadotropin-releasing hormone agonist therapy and adequate healing, conception was achieved again via in vitro fertilization one year later. The subsequent pregnancy progressed without evidence of myometrial thinning on mid-trimester imaging, and a healthy male infant was delivered via elective cesarean section at 35 weeks and 6 days of gestation. Early conception following adenomyomectomy, particularly in the setting of assisted reproductive technology, may increase the risk of intramural implantation. Fertility-preserving surgical management using intraoperative ultrasound guidance and cavity delineation appears feasible and can result in a successful subsequent pregnancy. • Early conception after adenomyomectomy may increase the risk of intramural pregnancy, particularly following in vitro fertilization. • Twin intramural implantation at a prior surgical scar is an extremely rare clinical presentation. • Magnetic resonance imaging is valuable for accurate diagnosis and surgical planning in atypical ectopic pregnancies. • Intraoperative ultrasound with endometrial cavity delineation enables safe, fertility-preserving surgical management. • Successful subsequent pregnancy demonstrates the feasibility of this approach after adequate healing.
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