eng Science and Education Publishing American Journal of Medical Case Reports 2374-216X 2018-06-08 6 5 81 86 10.12691/ajmcr-6-5-2 AJMCR2018652 article Tariq Al-Saadi t.dhiyab@hotmail.com 1 Ahmed Al Habsi 2 Zahra Al Hajri 3 Neurosurgical Resident, Montreal Neurological Institute and Hospital - McGill University, Montreal, Canada Department of Neurosurgery, Khoula Hospital, Muscat, Oman Department Radiology, Khoula Hospital, Muscat, Oman Background: Congenital dermal sinus tract (DST) is a rare entity of cranial or spinal dysraphism that may occur anywhere from nasion and along the midline neuraxis from occipital to sacral regions. Craniospinal infection caused by occipital DST is even rarer. Because of their scarcity, these lesions are not well documented in the literature, often mixed with dermal sinuses in other location or other dysmorphic features. This paper reports a unique case of an infant presented with craniospinal abscesses resulting from occipital dermal sinus tract. Methods and materials: In this paper, we report a case of a 16-month-old girl presented with high grade fever, vomiting and lethargy. She had a discharging occipital skin lesion. Her diagnosis was "Occipital DST with Cerebellospinal Abscess", which was treated successfully by excision of the DST and cerebellar abscess. Histopathological examination revealed a dermoid cyst. She received 8 weeks of parenteral antibiotic treatment with a good outcome. Results: Occipital DST is a rare condition. Its clinical presentation varies and clinical suspicion is required. Early neurosurgical intervention is important to prevent the risk of potential complications such as abscess and bacterial meningitis. Conclusion: This case highlights the importance of early recognition and evaluation of midline craniospinal cutaneous stigmata in infant. Further neurosurgical assessment with radiological investigations are recommended for early detection and management. Once diagnosed is made, surgical intervention and appropriate antibiotic therapy are the mainstay of treatment. http://pubs.sciepub.com/ajmcr/6/5/2/ajmcr-6-5-2.pdf occipital dermal sinus tract abscess dermoid meningitis