Bilateral proptosis: an unusual primary presentation of scurvy—a case report

Springer Science and Business Media LLC - Tập 52 - Trang 1-5 - 2021
Damini S.1, Rashmi Dixit1, Tamanna Khullar1, K. Rajeshwari2
1Department of Radiodiagnosis, Maulana Azad Medical College and Lok Nayak Hospital, New Delhi, India
2Department of Paediatrics, Maulana Azad Medical College and Lok Nayak Hospital, New Delhi, India

Tóm tắt

Scurvy is a nutritional vitamin C deficiency disease which shows classical signs on radiographs most commonly along long bones with subperiosteal hemorrhage being a classical finding. Primary presentation of scurvy with orbital hemorrhage presenting as proptosis is however uncommon. We report an unusual primary presentation of scurvy in a 4-year-old boy with West syndrome and global developmental delay who presented to the emergency with complaints of bilateral proptosis and eyelid swelling (right > left) which on examination revealed bluish discolouration of bilateral eyelids. On further multimodality imaging investigation, orbital subperiosteal hemorrhages were identified as the cause, along with classical radiographic features of scurvy in bilateral lower limb long bones. The diagnosis was further bolstered by rapid resolution of periorbital ecchymosis after initiating oral vitamin C supplementation. To the best of our knowledge, primary presentation of scurvy in the form of bilateral proptosis due to orbital hemorrhages although uncommon can still be a manifestation of scurvy in addition to the more routinely detected subperiosteal hemorrhages along long bones. The significance of establishing the aetiology in such cases of orbital hemorrhage cannot be overemphasised, in view of drastic clinical recovery after starting vitamin C therapy.

Tài liệu tham khảo

Nasiri J, Zamani F (2017) Periorbital ecchymosis (raccoon eye) and orbital hematoma following endoscopic retrograde cholangiopancreatography. Case Rep Gastroenterol 11(1):134–141. https://doi.org/10.1159/000456657 Büyükavci M, Yildirim ZK (2009) Recurrent bruising of lower eyelids: a presenting symptom of neuroblastoma in a child/Alt gözkapaklarinda tekrarlayan morluklar yakinmasiyla basvuran nöroblastomlu bir çocuk olgu. Turk J Haematol 26(4):210 Timmerman R (2003) Raccoon eyes and neuroblastoma. N Engl J Med 349(4):e4. https://doi.org/10.1056/ENEJMicm020675 McNab AA (2014) Nontraumatic orbital haemorrhage. survey of ophthalmology, 59(2): 166-184. Roeser HP (1983) The role of ascorbic acid in the turnover of storage iron. Semin Hematol 20(2):91–100 Cheah SC, Tang P, Matthew TJ, Ooi MH, Husain S (2020) Spontaneous orbital haematoma in a scurvy child: a forgotten diagnosis. Int J Pediatr Otorhinolaryngol 137:110224. https://doi.org/10.1016/j.ijporl.2020.110224 Snow I (1905) Eye symptoms of infantile scurvy. A case of infantile scurvy with extreme protrusion of the right eyeball, shown by autopsy to be due to a large retrobulbar hematoma. Arch Pediatr 1905(22):576e80 Rajakumar K (2001) Infantile scurvy: a historical perspective. Pediatrics 108(4):e76–e76. https://doi.org/10.1542/peds.108.4.e76 Joshi R, Gustas-French CN, Fanburg-Smith JC, Helm KF, Flemming D (2019) Scurvy: a rare case in an adult. Skeletal Radiol 48(6):977–984. https://doi.org/10.1007/s00256-018-3069-3 Pelkan F (1925) The roentgenogram in early scurvy. Am J Dis Child 30(2):174–188 Nelson WE, Doughty WM, Mitchell AG (1933) Roentgenographic visualization of subperiosteal hemorrhage in infantile scurvy. JAMA 101(1):14–17. https://doi.org/10.1001/jama.1933.02740260016004 Sloan B, Kulwin DR, Kersten RC (1999) Scurvy causing bilateral orbital hemorrhage. Arch Ophthalmol 117(6):842–843. https://doi.org/10.1001/archopht.117.6.842 Corl K, Hofmann RJ, Sutton E (2010) Non-traumatic orbital hemorrhage. J Emerg Med 38(3):378–380. https://doi.org/10.1016/j.jemermed.2007.10.021 Pavone P, Polizzi A, Marino SD, Corsello G, Falsaperla R, Marino S, Ruggieri M (2020) West syndrome: a comprehensive review. Neurol Sci 22:1–6