Citation: Sarkissian S, Kahlon K, Walavalkar V, Siaghani P, Tran MH, et al. Renal Manifestations of Alternate Complement Pathway Dysregulation. J Urol
Nephrol. 2015;2(1): 4.
J Urol Nephrol
August 2015 Vol.:2, Issue:1
© All rights are reserved by Harley et al.
Renal Manifestations of
Alternate Complement Pathway
Dysregulation
Abstra c t
Irre g ula ritie s o f the a lte rna te c o mp le me nt p a thwa y c a n le a d
to b o th re na l-limite d a nd syste mic p a tho lo g y. We p re se nt the c a se
o f a tra nsp la nt re c ip ie nt with re na l d ise a se o f unkno wn e tio lo g y who
d e ve lo p e d a c ute kid ne y injury in the we e ks fo llo wing tra nsp la nt a nd
ma nife ste d d iffe re nt histo lo g ic a nd c linic a l p a tte rns o f a lte rna te
c o mp le me nt p a thwa y me d ia te d immuno lo g ic injury a t d iffe re nt p o ints
in he r p o st-tra nsp la nt c o urse . We se a rc he d Me d line fo r c a se s a nd
stud ie s p e rta ining to a lte rna te c o mp le me nt p a thwa y re na l-a sso c ia te d
d ise a se s, the ir p a tho lo g ic me c ha nisms, p o te ntia l tre a tme nt o p tio ns,
and report our fndings.
Introduction
Alternate complement pathway (ACP) dysregulation and its varied
clinical manifestations are an area of intense interest in contemporary
nephrology research. Diagnoses such as atypical hemolytic uremic
syndrome, membranoproliferative glomerulonephritis type 2,
atypical post-infectious glomerulonephritis, and the relatively new
entity known as C3 glomerulopathy, have been linked to aberrancies
in the normal immunologic cascade following alternate complement
pathway activation. Advances in ACP genetics have revealed
heritable predispositions to such diagnoses and furthered the drive to
develop targeted, accessible treatment options. Te growing number
of patients on kidney transplant waiting lists with primary diagnoses
among these entities has also spurned a need to carefully evaluate
recipient candidacy given the signifcant possibility for disease
recurrence and the limited treatment options currently available in
such situations. We report a complicated case of ACP dysregulation
in a kidney transplant recipient and provide a discussion of
manifestations related to this pathology.
Case
A 47 year-old Hispanic female sufered from uncontrolled, chronic
hypertension since her late 20s, had limited access to medical care,
and presented to a nephrologist with advanced symptomatic CKD
she was started on hemodialysis. As a result of her late presentation,
her nephrologist determined a native biopsy would yield little data
other than advanced glomerulosclerosis and thus one was deemed
unwarranted and not performed. Afer a few years on hemodialysis,
she underwent a living unrelated kidney transplant. She received the
IL-2 inhibitor basiliximab for induction, was put on a rapid steroid
taper, and started on maintenance tacrolimus and mycophenolic
acid. She was discharged home with a nadir creatinine of 1.1 mg/dl.
Two weeks later her creatinine had rebounded to 1.9 mg/dl.
She had 3.5 grams proteinuria and an active urine sediment. Her
peripheral smear was unremarkable and platelet count preserved. An
allograf biopsy was done and showed proliferative glomerulonephritis
without acute rejection. Immunofuorescence revealed granular,
mesangial positive C3 staining and negative immunoglobulin and
light chain staining. Electron microscopy demonstrated electron
dense subepithelial and mesangial deposits. A preliminary pathologic
diagnosis of post-infectious glomerulonephritis was made without
any clinical evidence of antecedent infection.
In the following weeks, labs revealed an IgG lambda monoclonal
gammopathy in the serum and urine. A bone marrow biopsy was
performed showing 2-3% plasma cells. Subsequently three doses of
weekly cyclophosphamide and bortezomib with dexamethasone
was administered and subsequent low dose steroid maintenance
therapy was added and continued. Tereafer, her creatinine rose to
4.4 mg/dl. Other than a few rare schistocytes, evidence for a systemic
microangiopathic process was decidedly unimpressive with stable
hemoglobin levels and absence of hyperbilirubinemia throughout.
Although the platelet count dropped on a single occasion to 139
K/mcL, all other counts were 150 K/mcL. A second renal allograf
biopsy was performed. (see Table 1 for diagnostic test summary)
Te second biopsy revealed evidence of glomerular thrombotic
microangiopathy(TMA) with mesangiolysis, glomerular shrinking,
and fbrin positive deposits by immunofuorescence. C3 mesangial
staining persisted. Tubular lambda light chain staining was now
positive. Electron microscopy showed subepithelial deposit is had
decreased in size and now demonstrated occluded capillary lumens
Sarmen Sarkissian
1
, Kanwarpal Kahlon
1
, Vighnesh
Walavalkar
2
, Parwiz Siaghani
2
, Minh-Ha Tran
3
, and
Kevin Harley
4
*
1
Department of Medicine, Divison of Hematology-Oncology, UC
Irvine School of Medicine, 101 The City Dr S, Orange, CA 92868,
USA
2
Department of Pathology, UC Irvine School of Medicine, 101 The
City Dr S, Orange, CA 92868, USA
3
Departments of Pathology and Medicine, UC Irvine Health School
of Medicine, 101 The City Dr S, Orange, CA 92868, USA
4
Department of Medicine, Division of Nephrology & Hypertension,
UC Irvine Health School of Medicine, 101 The City Dr S, Orange,
CA 92868, USA
Address for Correspondence
Kevin Harley, Department of Medicine, Division of Nephrology &
Hypertension, UC Irvine Health School of Medicine, 101 The City Dr S,
Orange, CA 92868, USA, Tel: 714-456-5142; E-mail: kharley@uci.edu
Submission: 28 May 2015
Accepted: 04 August 2015
Published: 10 August 2015
Copyright: © 2015 Sarkissian S, et al. This is an open access article
distributed under the Creative Commons Attribution License, which
permits unrestricted use, distribution, and reproduction in any medium,
provided the original work is properly cited.
Reviewed & Approved by: Dr. Yasin Aydogmus, Specialist/
urologist, Etimesgut Military Hospital, Ankara, Turkey
Case Report Open Access
Journal of
Urology &
Nephrology