Uzerk KM, Buyukaksoy M, Candar C, Koksal CS, Meryem KS, Haberal G, Hacı YH, Sağlam A

Language: English
References: 13
Page: 153-160
PDF size: 294.23 Kb.

ABSTRACT

Monoclonal gammopathy is a disorder characterized by the proliferation of plasma cells or B lymphocytes and overproduction of monoclonal immunoglobulin. Renal involvement in monoclonal gammopathy can manifest in various ways, but histiocytes obstructing glomerular capillaries is exceptionally rare. We report on a case of a patient presenting with renal impairment, proteinuria, and anemia. Kidney biopsy revealed numerous histiocytes plugging glomerular capillaries and renal tubular lumina, containing crystalline monoclonal immunoglobulin deposits. The bone marrow aspirate demonstrated an increase in atypical plasma cells with cytoplasmic inclusions, consistent with a diagnosis of multiple myeloma. This case highlights an unusual presentation of monoclonal gammopathy with renal involvement, emphasizing the importance of considering this diagnosis in patients with unexplained renal impairment and proteinuria. This is, to our knowledge, the fourth reported case of crystalstoring histiocytosis involving glomerular capillary loops and the first to describe its coexistence with light chain cast nephropathy.

REFERENCES

1. Sethi S, Rajkumar SV, D’Agati VD. The Complexityand Heterogeneity of Monoclonal Immunoglobulin-Associated Renal Diseases. J Am Soc Nephrol. 2018;29(7):1810-23.

2. Nasr SH, Valeri AM, Sethi S, Fidler ME, Cornell LD,Gertz MA, et al. Clinicopathologic Correlations inMultiple Myeloma: A Case Series of 190 Patientswith Kidney Biopsies. American Journal of KidneyDiseases. 2012;59(6):786-94.

3. Mobarki M, Papoudou-Bai A, Dumollard JM,Alhazmi AH, Musawi S, Madkhali MA, et al. Crystal-Storing Histiocytosis: The Iceberg of More SeriousConditions. Diagnostics. 2023;13(2):271.

4. Zhu L, Wang L, Shi H, Jiang L, Li X, Shao C, et al.Combined crystal-storing histiocytosis, light chainproximal tubulopathy, and light chain crystallinepodocytopathy in a patient with multiple myeloma: acase report and literature review. Ren Fail.2023;45(1):2145970.

5. Bladé J, Fernández-Llama P, Bosch F, Montolíu J,Lens XM, Montoto S, et al. Renal failure in multiplemyeloma: presenting features and predictors ofoutcome in 94 patients from a single institution. ArchIntern Med. 1998;158(17):1889-93.

6. Gupta RK, Rosenberg AZ, Bagnasco SM, Arend LJ.Renal crystal-storing histiocytosis involvingglomeruli - A comprehensive clinicopathologicanalysis. Ann Diagn Pathol. 2019;43:151403.

7. Fang H, Chiu A, Reichard KK. Crystal-StoringHistiocytosis in Bone Marrow: A ClinicopathologicStudy of Eight Cases and Review of Literature. Am JClin Pathol. 2018;149(2):148-63.

8. Sethi S, Cuiffo BP, Pinkus GS, Rennke HG. Crystalstoringhistiocytosis involving the kidney in a lowgradeB-cell lymphoproliferative disorder. Am JKidney Dis. 2002;39(1):183-8.

9. Stokes MB, Aronoff B, Siegel D, D’Agati VD.Dysproteinemia-related nephropathy associated withcrystal-storing histiocytosis. Kidney Int.2006;70(3):597-602.

10. Shah S, Sethi S, Arend L, Geetha D. Crystal-storinghistiocytosis. Kidney Int. 2016;89(2):507.

11. Messiaen T, Deret S, Mougenot B, Bridoux F,Dequiedt P, Dion JJ, et al. Adult Fanconi syndromesecondary to light chain gammopathy.Clinicopathologic heterogeneity and unusualfeatures in 11 patients. Medicine (Baltimore).2000;79(3):135-54.

12. Lebeau A, Zeindl-Eberhart E, Müller EC, Müller-Höcker J, Jungblut PR, Emmerich B, Löhrs U.Generalized crystal-storing histiocytosis associatedwith monoclonal gammopathy: molecular analysis ofa disorder with rapid clinical course and review ofthe literature. Blood. 2002;100(5):1817-27.

13. Jones D, Bhatia VK, Krausz T, Pinkus GS. Crystalstoringhistiocytosis: a disorder occurring inplasmacytic tumors expressing immunoglobulinkappa light chain. Hum Pathol. 1999;30(12):1441-8.