American Indians and Alaska Natives (AI/AN) are the Indigenous peoples of the United States. According to the U.S. Census Bureau, approximately 9.7 million people self-identified as AI/AN (alone or in combination with other races), representing 2.9% of the total U.S. population. These people represent diverse groups of discrete Tribes, each with their language, culture, and geographic home. As part of the conquest and settlement of North America, some Indigenous peoples signed treaties with the U.S. government, surrendering their lands in return for various government commitments, including health care. The Indian Health Service (IHS) was born out of these agreements. The IHS is an agency in the U.S. Department of Health and Human Services under the U.S. Public Health Service. The IHS provides a comprehensive health service delivery system for approximately 2.7 million AI/AN who belong to 574 federally recognized Tribes/nations in 37 states. The aim of this paper is to make recommendations regarding the initiation of sustainable neurology care in marginalized or underserved populations by reviewing 40 years of neurology care provision within the IHS. We will discuss (1) the IHS, (2) neurological care provided within the IHS, including midlevel provider extension of neurology care and traditional medical care, and (3) select neurological diagnoses within AI/AN populations. Marginalized populations, including those in the United States that are rural, remote, or low socioeconomic status, lack access to specialty neurology care. This includes many AI/AN. The IHS has developed novel solutions to promote specialty care, including neurology. Notably, initial IHS investments in full-time neurology providers have led to more robust neurology care, often receiving attention from university programs. This suggests that an initial investment in stable on-site full-time neurology services provides a path to potential sustainable care for marginalized populations.