Abstract
Anti-phospholipid syndrome (APS) is a systemic autoimmune disease causing arterial and venous thrombosis, leading to macrovascular and microvascular complications. Bone marrow necrosis (BMN) is defined as the death of hematopoietic tissue and the loss of fat cells in a bone marrow biopsy. BMN is typically associated with a poor prognosis, with most patients dying within weeks. We present a case of BMN in a 32-year-old male patient with APS admitted to our hospital. The patient presented with bilateral non-healing leg ulcers and bilateral lower limb edema. The findings of the duplex ultrasound were consistent with old bilateral deep vein thromboses in the calves. The presence of acute kidney injury and proteinuria prompted a renal biopsy, which revealed chronic thrombotic microangiopathy. Bone marrow biopsy revealed BMN. Unfortunately, the patient did not respond to immunosuppressive treatment and passed away due to septic shock. The unique features in the patient’s presentation, combined with the availability of extensive data on the patient’s history and investigations, further enhance its significance. It is not possible to establish a cause-and-effect relationship or draw conclusive findings from this case report alone. More case reports from clinicians who come across BMN in patients with APS are needed to broaden our understanding of the pathogenesis, presentation, management, and outcomes.
References
1. Truglia S, Capozzi A, Mancuso S, Manganelli V, Rapino L, Riitano G, Recalchi S, Colafrancesco S, Ceccarelli F, Garofalo T, Alessandri C, Longo A, Misasi R, Conti F, Sorice M. Relationship Between Gender Differences and Clinical Outcome in Patients With the Antiphospholipid Syndrome. Front Immunol. 2022: 932181 [PMID: 35860235, https://doi.org/10.3389/fimmu.2022.932181]
2. Sammaritano LR. Antiphospholipid syndrome. Best Pract Res Clin Rheumatol. 2020: 101463 [PMID: 31866276, https://doi.org/10.1016/j.berh.2019.101463]
3. Bustamante JG, Goyal A, Rout P, Singhal M. Antiphospholipid Syndrome. StatPearls. Treasure Island (FL): StatPearls Publishing
Copyright © 2025, StatPearls Publishing LLC.; 2025.
4. Gaspar P, Sciascia S, Tektonidou MG. Epidemiology of antiphospholipid syndrome: macro- and microvascular manifestations. Rheumatology (Oxford). 2024: SI24 [PMID: 38320589, https://doi.org/10.1093/rheumatology/kead571]
5. Vandevelde A, Devreese KMJ. Laboratory Diagnosis of Antiphospholipid Syndrome: Insights and Hindrances. J Clin Med. 2022: [PMID: 35456258, https://doi.org/10.3390/jcm11082164]
6. Garcia D, Erkan D. Diagnosis and Management of the Antiphospholipid Syndrome. N Engl J Med. 2018: 2010 [PMID: 29791828, https://doi.org/10.1056/NEJMra1705454]
7. Cohen H, Cuadrado MJ, Erkan D, Duarte-Garcia A, Isenberg DA, Knight JS, Ortel TL, Rahman A, Salmon JE, Tektonidou MG, Williams DJ, Willis R, Woller SC, Andrade D. 16th International Congress on Antiphospholipid Antibodies Task Force Report on Antiphospholipid Syndrome Treatment Trends. Lupus. 2020: 1571 [PMID: 33100166, https://doi.org/10.1177/0961203320950461]
8. Luan C, Zhao H, Ding Y. Bone marrow necrosis: Facts, controversies, and perspective. Int J Lab Hematol. 2024: 770 [PMID: 38923828, https://doi.org/10.1111/ijlh.14335]
9. Conrad ME. Bone marrow necrosis. J Intensive Care Med. 1995: 171 [PMID: 10155181, https://doi.org/10.1177/088506669501000403]
10. Wool GD, Deucher A. Bone marrow necrosis: ten-year retrospective review of bone marrow biopsy specimens. Am J Clin Pathol. 2015: 201 [PMID: 25596246, https://doi.org/10.1309/AJCP0TN1MCMOLMPK]
11. Badar T, Shetty A, Bueso-Ramos C, Cortes J, Konopleva M, Borthakur G, Pierce S, Huang X, Chen HC, Kadia T, Daver N, Dinardo C, O'Brien S, Garcia-Manero G, Kantarjian H, Ravandi F. Bone marrow necrosis in acute leukemia: Clinical characteristic and outcome. Am J Hematol. 2015: 769 [PMID: 26017166, https://doi.org/10.1002/ajh.24074]
12. Bulvik S, Aronson I, Ress S, Jacobs P. Extensive bone marrow necrosis associated with antiphospholipid antibodies. Am J Med. 1995: 572 [PMID: 7778573, https://doi.org/10.1016/s0002-9343(99)80016-3]
13. Paydas S, Kocak R, Zorludemir S, Baslamisli F. Bone marrow necrosis in antiphospholipid syndrome. J Clin Pathol. 1997: 261 [PMID: 9155683, https://doi.org/10.1136/jcp.50.3.261]
14. Morad CS, Fayez D, Mahmoud M, Aboud FM. Primary and secondary antiphospholipid syndrome characteristics in an Egyptian cohort. The Egyptian Rheumatologist. 2022: 373 https://doi.org/10.1016/j.ejr.2022.07.006]
15. El-Moniem GA, El-Garf K, Sobhy N, Elmaghraby S. Characterization of the Clinical and Laboratory Features of Primary and Secondary Antiphospholipid Syndrome in a Cohort of Egyptian Patients. Curr Rheumatol Rev. 2020: 304 [PMID: 31951187, https://doi.org/10.2174/1573397116666200116095734]
16. Elgamal BM, Rashed RA, Raslan HN. Prevalence of bone marrow necrosis in Egyptian cancer patients referring to the National Cancer Institute. J Egypt Natl Canc Inst. 2011: 95 [PMID: 22776813, https://doi.org/10.1016/j.jnci.2011.09.007]
17. Deucher A, Wool GD. How I investigate bone marrow necrosis. Int J Lab Hematol. 2019: 585 [PMID: 31424633, https://doi.org/10.1111/ijlh.13091]
18. Cabral TCS, Fernandes CM, Lage LAC, Zerbini MC, Pereira J. Bone marrow necrosis: literature review. Jornal Brasileiro de Patologia e Medicina Laboratorial. 2016: https://doi.org/10.5935/1676-2444.20160031]
This work is licensed under a Creative Commons Attribution 4.0 International License.
Copyright (c) 2025 ASIDE Case Reports