A two-month-old male was brought to the Infectology service of INP to seek medical care, because he had dysmorphias. Also, maternal acute toxoplasmosis was suspected at six weeks of gestation (WG) due to abnormal evolution and presence of IgM anti-T. gondii antibodies (abs). The nineteen-year-old woman harbored intrauterine dichorionic diamniotic twins of 7.4 (gestational sac of 2.4 cm) and 7.3 (2.3 cm) WG as calculated from the ultrasound (USG). She had miscarriage threat at 11 WG. At the 14th WG, only one apparently normal fetus was apparent in the USG, so there was involution of the other twin. A unilateral complete palatal defect was seen in the USG performed during the last trimester. A male product was born by cesarean at 38 WG; with a weight of 2.78 kg (P10), a height of 48 cm (P10–25), an Apgar score of 8/9 and a Silverman score of 0 points. The newborn lacked the left eye and presented ipsilateral oblique oro-orbital cleft. The diagnostic approach began four days after birth with a TORCH profile. IgG was positive for T. gondii, Rubella and Cytomegalovirus, but IgM abs were negative against the three agents. The blood count was normal. The baby presented bilateral sensorineural hearing loss as demonstrated by auditive screening. The metabolic screening results were normal. When the patient arrived to INP, no evidence of psychomotor retardation was found and the Ophthalmology service discarded alterations in the healthy eye. Cytomegalovirus and T. gondii infections were searched in blood by serology and real-time PCR. Congenital toxoplasmosis was confirmed by the presence of IgM abs and IgG neoabs in western blot, as well as by real time PCR in blood, with a parasite load of 6903 parasites/mL [, ]. In the cranial CT scan, the left oblique oro-orbital and parasagittal cleft defect were observed, as well as ipsilateral lack of osseous tissue and soft palate. Left choanal atresia and narrow-left vestibule were discovered in the nasal cavity. The right nasal cavity was communicated with the oropharynx through a narrow choanal passage. The left turbinates were hypoplastic. A nasal endoscopy was performed which confirmed CT scan findings. The left eyeball and the optic nerve were absent; however, hypoplastic extraocular muscles were present in the orbit. The brain parenchyma and the right side of the face were normal. According to the data described above, diagnosis was type 3 left craniofacial cleft (Tessier classification), ipsilateral anophthalmia and congenital toxoplasmosis. Anti-T. gondii therapy with pyrimethamine, trimethoprim/sulfamethoxazole and folinic acid was started according to the international recommendations []. At the beginning, the mother did not adhere to treatment, because as she argued, there was a long distance between her home and the hospital, but mainly because she had difficulty to understand the benefits of prophylactic chemotherapy; the physicians perseverated, so the second semester of life the baby received proper treatment. Surgical management was focused in maximizing actual vision and improving aesthetics, through simultaneous stimulation of soft tissues and bony orbital growth. Anophthalmia requires additional remodeling by soft tissue reconstruction and endo-orbital volume replacement (with implants, expanders and dermis-fat grafts). A first surgery was performed to start correction of the anatomical defect. During the surgery, a biopsy containing mucosa and skin was taken from the anophthalmia site. Images compatible with T. gondii pseudocysts and free parasites were observed by histopathology, as well as an active chronic inflammatory reaction, composed by lymphocytes, plasma cells, eosinophils and neutrophils. Prognosis is favorable since the baby had no neurodevelopment disorders, no right eye or intracranial lesions and is following a protocol for reconstructive plastic surgery including eyeball prosthesis.