The coronavirus disease 2019 (COVID-19) pandemic has claimed nearly 5.5 million lives worldwide. Adenovirus-based vaccines are safe and effective, but they are rarely associated with vaccine-induced thrombosis and thrombocytopenia (VITT) as well as cerebral venous sinus thrombosis (CVST). We conducted a systematic literature search of intracerebral hemorrhage (ICH) secondary to CVST associated with VITT from the Ad26.COV2.S vaccine, and we present the first case of this pathology in the reviewed literature of a patient who required neurosurgical decompression. The systematic literature review was completed on December 19, 2021, by searching PubMed and Ovid for articles with primary data on CVST associated with VITT following the Ad26.COV2.S vaccine. We also specifically searched for cases that required neurosurgical intervention. Articles were independently screened by two authors, and both secondary and tertiary searches were done as well. Descriptive statistics were collected and presented in table form. Nine studies were identified that met inclusion criteria. There were no cases identified of patients who underwent neurosurgical decompression after developing this pathology. We thus present the first case in the reviewed literature of a patient who developed ICH after receiving the Ad26.COV2.S vaccine and underwent decompressive hemicraniectomy. Despite severe thrombocytopenia and prolonged intensive care, the patient was discharged to neurorehabilitation. There is a much greater risk of CVST and ICH during COVID-19 infections than from the vaccines. However, as booster vaccines are approved and widely distributed, it is critical to make prompt, accurate diagnoses of this vaccine-related complication and consider neurosurgical decompression.