Abstract
The hypereosinophilic syndrome (HES) is a set of rare blood diseases defined by a persistent blood eosinophil count of ≥ 1.5 × 109 cells/l, with clinical features and organ dysfunction attributable to eosinophilia. The most frequently involved organ in HES is the heart. Kidney damage is considered to be rare. To the best of our knowledge, this is the first documented case of acute kidney injury secondary to the T-lymphocytic variant of HES reported in Pakistan. A 16-year-old boy presented with complaints of generalized body swelling, decreased urine output, and two episodes of cola-colored urine. His renal function was deteriorated with 3 + albuminuria and a protein: creatinine ratio of 6.58. The patient underwent a series of investigations (serum protein electrophoresis, computed tomography, lymph node and bone marrow biopsy, and peripheral blood flow cytometry) to know the actual cause of acute kidney injury. Eosinophilia was noticed on peripheral blood smear and bone marrow biopsy. Kidney biopsy showed acute tubular necrosis, while abdominal computed tomography exhibited hepatosplenomegaly and lymphadenopathy. Peripheral blood flow cytometry revealed the CD3 + T cells count of 4422 cells/µl, CD4 + T cell count of 1925 cells/µl, and CD8 + T cell count of 2195 cells/µl. The patient was diagnosed with lymphocytic variant hypereosinophilic syndrome (L-HES) and was effectively managed with prednisolone. L-HES is a recognized subtype of HES that can rarely present with early renal manifestations. Physicians should be vigilant of such uncommon presentations for appropriate management of these cases.
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References
Shomali W, Gotlib J. World Health Organization-defined eosinophilic disorders: 2022 update on diagnosis, risk stratification, and management. Am J Hematol. 2022;97(1):129–48. https://doi.org/10.1002/ajh.26352.
Simon HU, Rothenberg ME, Bochner BS, et al. Refining the definition of hypereosinophilic syndrome. J Allergy Clin Immunol. 2010;126(1):45–9. https://doi.org/10.1016/j.jaci.2010.03.042.
Valent P, Klion AD, Horny HP, et al. Contemporary consensus proposal on criteria and classification of eosinophilic disorders and related syndromes. J Allergy Clin Immunol. 2012;130(3):607-612.e9. https://doi.org/10.1016/j.jaci.2012.02.019.
Bagot M, Bodemer C, Wechsler J, et al. Non epidermotropic T lymphoma preceded for several years by hypereosinophilic syndrome. Ann Dermatol Venereol. 1990;117(11):883–5. https://europepmc.org/article/med/2099714. Accessed 16 Sept 2021
Roufosse F, Cogan E, Goldman M. Lymphocytic variant hypereosinophilic syndromes. Immunol Allergy Clin North Am. 2007;27(3):389–413. https://doi.org/10.1016/j.iac.2007.07.002.
Lefèvre G, Copin MC, Staumont-Sallé D, et al. The lymphoid variant of hypereosinophilic syndrome: study of 21 patients with CD3-CD4+ aberrant T-cell phenotype. Medicine (Baltimore). 2014;93(17):255–66. https://doi.org/10.1097/MD.0000000000000088.
Garella G, Marra L. Hypereosinophilic syndrome and renal insufficiency. Minerva Urol Nefrol. 1990;42(2):135–6. https://europepmc.org/article/med/2392741. Accessed 16 Sept 2021
Navarro I, Torras J, Gomà M, Cruzado JM, Grinyó JM. Renal involvement as the first manifestation of hypereosinophilic syndrome: a case report. NDT Plus. 2009;2(5):379–81. https://doi.org/10.1093/ndtplus/sfp092.
Choi YJ, Lee JD, Yang KH, et al. Immunotactoid glomerulopathy associated with idiopathic hypereosinophilic syndrome. Am J Nephrol. 1998;18(4):337–43. https://doi.org/10.1159/000013362.
Liapis H, Ho AK, Brown D, Mindel G, Gleich G. Thrombotic microangiopathy associated with the hypereosinophilic syndrome. Kidney Int. 2005;67(5):1806–11. https://doi.org/10.1111/j.1523-1755.2005.00278.x.
Giudicelli CP, Didelot F, Duvic C, Desrame J, Herody M, Nedelec G. Eosinophilie et pathologie renale [Eosinophilia and renal pathology]. Med Trop (Mars). 1998;58(4 Suppl):477–81.
Lanfranchi J, Meyrier A, Sachs RN, Guillevin L. Syndrome d’hyperéosinophilie sanguine avec atteinte cardiaque et glomérulopathie extra-membraneuse [Blood hypereosinophilia syndrome with cardiac involvement and extramembranous glomerulopathy]. Ann Med Interne (Paris). 1986;137(2):133–7.
Chusid MJ, Dale DC, West BC, Wolff SM. The hypereosinophilic syndrome: analysis of fourteen cases with review of the literature. Medicine (Baltimore). 1975;54(1):1–27. https://europepmc.org/article/med/1090795. Accessed 16 Sept 2021
Klion A. Hypereosinophilic syndrome: current approach to diagnosis and treatment. Annu Rev Med. 2009;60:293–306. https://doi.org/10.1146/annurev.med.60.062107.090340.
Klion AD, Bochner BS, Gleich GJ, et al. Approaches to the treatment of hypereosinophilic syndromes: a workshop summary report. J Allergy Clin Immunol. 2006;117(6):1292–302. https://doi.org/10.1016/j.jaci.2006.02.042.
Pallesen KAU, Herlin T, Holm M, et al. Idiopathic hypereosinophilic syndrome: a rare diagnosis in children. Clin Case Rep. 2020;8(10):2013–6. Published 2020 Sep 14. https://doi.org/10.1002/ccr3.3165.
Simon H, Klion A. Therapeutic Approaches to Patients With Hypereosinophilic Syndromes. Semin Hematol. 2012;49(2):160–70. https://doi.org/10.1053/j.seminhematol.2012.01.002.
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Abdul Saboor Khan, Sidra J. Barry, and Fatima Memon did the literature search, drafted the initial manuscript, approved the final version of the manuscript, and agreed to be accountable for all aspects in ensuring that questions related to accuracy or integrity of any part of the work are appropriately investigated and resolved. Abdul Saboor Khan and Sidra J. Barry were actively involved in the patient’s care.
Taha Bin Arif and Anum Khalid contributed to the conception of the work, revised the manuscript for important intellectual content, approved the final version of the manuscript, and agreed to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.
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Khan, A.S., Barry, S.J., Memon, F. et al. A Case of T-Lymphocytic Variant Hypereosinophilic Syndrome with Acute Kidney Injury as an Early Manifestation. SN Compr. Clin. Med. 4, 56 (2022). https://doi.org/10.1007/s42399-022-01139-8
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DOI: https://doi.org/10.1007/s42399-022-01139-8