Congenital factor X deficiency: a case of unexplained neonatal anemia presenting with intracranial hemorrhage and hemorrhagic meningitis in the first month of life in a resource-limited setting
DOI:
https://doi.org/10.18203/2349-3291.ijcp20251113Keywords:
Factor X, Coagulation profile, Intracranial hemorrhage, Unexplained neonatal anemia, MeningitisAbstract
Factor X (FX) deficiency is an exceptionally rare inherited autosomal recessive coagulation factor disorder, posing significant challenges due to its rarity and lack of evidence-based management guidelines. It can manifest in a spectrum of hemorrhagic symptoms, including life-threatening intracranial hemorrhages. Here, we reported a case of congenital FX deficiency who presented with intracranial hemorrhage (ICH), complicated by hemorrhagic meningitis within the first month of life, following unexplained neonatal anemia. Early diagnosis and prompt intervention are crucial, especially in resource-limited settings, for managing this rare condition. This case underscores the difficulties encountered in managing rare conditions like FX deficiency and emphasizes the necessity for further research and collaboration to establish standardized management protocols. By adding to the limited literature on Factor X deficiency, this case report emphasizes considering FX deficiency in severe neonatal anemia and highlights the importance of personalized patient care in addressing rare diseases.
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References
Peyvandi F, Mannucci PM, Lak M, Abdoullahi M, Zeinali S, SharifianR, et al. Congenital factor X deficiency: spectrum of bleeding symptoms in 32 Iranian patients. Br J Haematol. 1998;102(2):626-8. DOI: https://doi.org/10.1046/j.1365-2141.1998.00806.x
Menegatti M, Peyvandi F. Factor X deficiency. Semin Thromb Hemost. 2009;35(4):407-15. DOI: https://doi.org/10.1055/s-0029-1225763
Masacupan KDT, Racho AR, Del Rosario ML, Lagaya-Aranas LM. The first reported case of factor X deficiency in a Filipino child-case study. J Haem Pract. 2023;10(1):90-4. DOI: https://doi.org/10.2478/jhp-2023-0015
World Federation of Hemophilia. Report on the Annual Global Survey 2023. October 2024. Available at: https://www1.wfh.org/publications/files/pdf-2525.pdf. Accessed on 9 March 2025.
Borboruah L, Dutta A. Management of a patient with factor X deficiency with FEIBA: a case report. J Haem Pract. 2023;10(1):68-73. DOI: https://doi.org/10.2478/jhp-2023-0012
Othman T, Abdelkarim A, Huynh K, Uche A, Lee J. Inherited moderate factor X deficiency presenting as cardiac tamponade. Case Rep Hematol. 2019;2019:9657516. DOI: https://doi.org/10.1155/2019/9657516
Ermis B, Ors R, Tastekin A, Orhan F. Severe congenital factor X deficiency with intracranial bleeding in two siblings. Brain Dev. 2004;6:137-8. DOI: https://doi.org/10.1016/S0387-7604(03)00119-0
Sandler E, Gross S. Prevention of recurrent intracranial hemorrhage in a factor X-deficient infant. Am J Pediatr Hematol Oncol. 1992;14:163-5. DOI: https://doi.org/10.1097/00043426-199205000-00012
Peyvandi F, Auerswald G, Austin SK. Diagnosis, therapeutic advances, and key recommendations for the management of factor X deficiency. Blood Rev. 2021;50:100833. DOI: https://doi.org/10.1016/j.blre.2021.100833