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April 1982, Vol 136, No. 4 >
Article | April 1982

Familial Occurrence of Renal and Intestinal Disease Associated With Tissue Autoantibodies FREE

Demetrius Ellis, MD; Stanley E. Fisher, MD; William I. Smith, MD; Ronald Jaffe, MB, BCh
[+] Author Affiliations

Reprint requests to Department of Pediatrics, Children's Hospital of Pittsburgh, 125 DeSoto St, Pittsburgh, PA 15213 (Dr Ellis).


Am J Dis Child. 1982;136(4):323-326. doi:10.1001/archpedi.1982.03970400041013.
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• Chronic tubulointerstitial renal disease and villous atrophy of the small intestine occurred in two first cousins. Both had protracted diarrhea with malabsorption and died despite intensive parenteral alimentation. In one patient signs of generalized proximal tubular dysfunction developed, followed by nephrotic syndrome and progressive renal insufficiency. A renal biopsy specimen disclosed severe tubulointerstitial disease and membranous glomerulopathy. In this patient, circulating immune complexes were detected and granular deposits of IgG and C3 were seen in the intestinal epithelial cells by direct immunofluorescence. Antiintestinal antibodies (IgG class) were demonstrated by indirect immunofluorescence. The other patient had interstitial nephritis but no glomerular abnormality. On direct immunofluorescence, both patients had confluent granular staining of the renal tubular basement membranes. These immunopathologic studies suggest a common immunologic mechanism in the pathogenesis of the renal and gastrointestinal disorders in these infants.

(Am J Dis Child 1982;136:323-326)

REFERENCES

1 +
Stokke KT, Teisberg PA, Mybre E, et al:  Nephrotic syndrome in ulcerative colitis . Scand J Gastroenterol 1976;;11:571-576.
2 +
Freundlich M, Gallego S, Scovino R:  Membranoproliferative glomerulonephritis (MPGN) associated with malabsorption and diabetes mellitus , abstracted. Kidney Int 1978;;14:651.
3 +
Martini A, Mariani G, Binda S:  Extramembranous glomerulopathy associated with villus atrophy of the small intestine: Presence of circulating antibodies directed against renal tubular basement membrane and cytoplasm of intestinal epithelium , abstracted. Pediatr Res 1980;; 14:1014.
4 +
Eckhardt R, Heinisch M, Meyer zum Buschefelde KH:  Cellular immune reactions against common antigen, small intestine, and colon antigen in patients with Crohn's disease, ulcerative colitis and cirrhosis of the liver . Scand J Gastroenterol 1976;;11:49-54.
5 +
Marcussen H:  Anti-colon antibodies in ulcerative colitis: A clinical study . Scand J Gastroenterol 1976;;11:763-767.
6 +
Van Thiel DH, Smith WI, Rabin BS, et al:  A syndrome of immunoglobulin A deficiency, diabetes mellitus, malabsorption, and a common HLA haplotype . Ann Intern Med 1977;;86:10-19.
7 +
Fisher SE, Boyle JT, Holtzapple P:  Chronic protracted diarrhea and jejunal atrophy in an infant: Cimetidine-associated stimulation of jejunal mucosal growth . Dig Dis Sci 1981;;26:181-186.
8 +
Peterson PA, Evrin PE, Berggard I:  Differentiation of glomerular, tubular, and normal proteinuria: Determinations of urinary excretion of B2-microglobulin, albumin, and total protein . J Clin Invest 1969;;48:1189-1198.
9 +
Mancini G, Carbonara AO, Heremans JF:  Immunochemical quantitation of antigens by single radial immunodiffusion . Int J Immunochem 1965;;2:235-254.
10 +
Zubler RH, Lange G, Lambert PH:  Detection of immune complexes in unheated sera by a modified 125I-C1q binding test . J Immunol 1976;;116:232-235.
11 +
Sunshine P, Sinatra FR, Mitchell CH:  Intractable diarrhea of infancy . Clin Gastroenterol 1977;;6:445-461.
12 +
Larcher VF, Shepherd R, Francis DEM, et al:  Protracted diarrhea in infancy: Analysis of 82 cases with particular reference to diagnosis and management . Arch Dis Child 1977;;52:597-605.
13 +
Davidson GP, Cutz E, Hamilton JR, et al:  Familial enteropathy: A syndrome of protracted diarrhea from birth, failure to thrive, and hypoplastic villus atrophy . Gastroenterology 1978;; 75:783-790.
14 +
Andres GA, McCluskey RT:  Tubular and interstitial renal disease due to immunologic mechanisms . Kidney Int 1975;;7:271-289.
15 +
Strife CF, McDonald BM, Ruley EJ, et al:  Shunt nephritis: The nature of the serum cryoglobulins and their relation to the complement profile . J Pediatr 1976;;88:403-413.
16 +
Powell DE:  Non-suppurative lesions in staphylococcal septicemia . J Pathol Bacteriol 1961;;82:141-149.
17 +
Gutman RA, Striker GE, Gilliland BC, et al:  The immune complex glomerulonephritis of bacterial endocarditis . Medicine 1972;;51:1-25.
18 +
Baldwin DS, Levine BB, McCluskey RT, et al:  Renal failure and interstitial nephritis due to penicillin and methicillin . N Engl J Med 1968;;279:1245-1252.
19 +
Ryan JA, Abel RM, Abbott WM, et al:  Catheter complications in total parenteral nutrition: A prospective study of 200 consecutive patients . N Engl J Med 1974;;290:757-761.
20 +
Sanders RA, Sheldon GF:  Septic complications of total parenteral nutrition: A five-year experience . Am J Surg 1976;;132:214-220.
21 +
McDonald R, Wiggelinkhuizen J, Kaschula OC:  The nephrotic syndrome in very young infants . Am J Dis Child 1971;;122:507-512.
22 +
Row PG, Cameron JS, Turner DR, et al:  Membranous nephropathy: Long-term follow-up and association with neoplasia . Q J Med 1975;;44:207-239.
23 +
Glassock RJ, Bennett CM:  The glomerulopathies , in Brenner BM, Rector RC (eds): The Kidney . Philadelphia, WB Saunders Co, 1976;, p 1003.
24 +
Baldwin DS, Gluck MC, Lowenstein J, et al:  Lupus nephritis: Clinical course as related to morphologic forms and their transitions . Am J Med 1977;;62:12-30.
25 +
Levy M, Gagnadoux MF, Beziau A, et al:  Membranous glomerulonephritis associated with anti-tubular and anti-alveolar basement membrane antibodies . Clin Nephrol 1978;;10:158-165.
26 +
Tung KSK, Black WC:  Association of renal glomerular and tubular immune complex disease and antitubular basement antibody . Lab Invest 1975;;32:696-700.
27 +
Buckley RH, Becker WG:  Abnormalities in the regulation of human IgE synthesis . Immunol Rev 1978;;41:288-314.
28 +
Gatti RA, Kersey JH, Yunis EJ, et al:  Graft-versus-host disease . Prog Clin Pathol 1973;;5:1-181.

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References

1 +
Stokke KT, Teisberg PA, Mybre E, et al:  Nephrotic syndrome in ulcerative colitis . Scand J Gastroenterol 1976;;11:571-576.
2 +
Freundlich M, Gallego S, Scovino R:  Membranoproliferative glomerulonephritis (MPGN) associated with malabsorption and diabetes mellitus , abstracted. Kidney Int 1978;;14:651.
3 +
Martini A, Mariani G, Binda S:  Extramembranous glomerulopathy associated with villus atrophy of the small intestine: Presence of circulating antibodies directed against renal tubular basement membrane and cytoplasm of intestinal epithelium , abstracted. Pediatr Res 1980;; 14:1014.
4 +
Eckhardt R, Heinisch M, Meyer zum Buschefelde KH:  Cellular immune reactions against common antigen, small intestine, and colon antigen in patients with Crohn's disease, ulcerative colitis and cirrhosis of the liver . Scand J Gastroenterol 1976;;11:49-54.
5 +
Marcussen H:  Anti-colon antibodies in ulcerative colitis: A clinical study . Scand J Gastroenterol 1976;;11:763-767.
6 +
Van Thiel DH, Smith WI, Rabin BS, et al:  A syndrome of immunoglobulin A deficiency, diabetes mellitus, malabsorption, and a common HLA haplotype . Ann Intern Med 1977;;86:10-19.
7 +
Fisher SE, Boyle JT, Holtzapple P:  Chronic protracted diarrhea and jejunal atrophy in an infant: Cimetidine-associated stimulation of jejunal mucosal growth . Dig Dis Sci 1981;;26:181-186.
8 +
Peterson PA, Evrin PE, Berggard I:  Differentiation of glomerular, tubular, and normal proteinuria: Determinations of urinary excretion of B2-microglobulin, albumin, and total protein . J Clin Invest 1969;;48:1189-1198.
9 +
Mancini G, Carbonara AO, Heremans JF:  Immunochemical quantitation of antigens by single radial immunodiffusion . Int J Immunochem 1965;;2:235-254.
10 +
Zubler RH, Lange G, Lambert PH:  Detection of immune complexes in unheated sera by a modified 125I-C1q binding test . J Immunol 1976;;116:232-235.
11 +
Sunshine P, Sinatra FR, Mitchell CH:  Intractable diarrhea of infancy . Clin Gastroenterol 1977;;6:445-461.
12 +
Larcher VF, Shepherd R, Francis DEM, et al:  Protracted diarrhea in infancy: Analysis of 82 cases with particular reference to diagnosis and management . Arch Dis Child 1977;;52:597-605.
13 +
Davidson GP, Cutz E, Hamilton JR, et al:  Familial enteropathy: A syndrome of protracted diarrhea from birth, failure to thrive, and hypoplastic villus atrophy . Gastroenterology 1978;; 75:783-790.
14 +
Andres GA, McCluskey RT:  Tubular and interstitial renal disease due to immunologic mechanisms . Kidney Int 1975;;7:271-289.
15 +
Strife CF, McDonald BM, Ruley EJ, et al:  Shunt nephritis: The nature of the serum cryoglobulins and their relation to the complement profile . J Pediatr 1976;;88:403-413.
16 +
Powell DE:  Non-suppurative lesions in staphylococcal septicemia . J Pathol Bacteriol 1961;;82:141-149.
17 +
Gutman RA, Striker GE, Gilliland BC, et al:  The immune complex glomerulonephritis of bacterial endocarditis . Medicine 1972;;51:1-25.
18 +
Baldwin DS, Levine BB, McCluskey RT, et al:  Renal failure and interstitial nephritis due to penicillin and methicillin . N Engl J Med 1968;;279:1245-1252.
19 +
Ryan JA, Abel RM, Abbott WM, et al:  Catheter complications in total parenteral nutrition: A prospective study of 200 consecutive patients . N Engl J Med 1974;;290:757-761.
20 +
Sanders RA, Sheldon GF:  Septic complications of total parenteral nutrition: A five-year experience . Am J Surg 1976;;132:214-220.
21 +
McDonald R, Wiggelinkhuizen J, Kaschula OC:  The nephrotic syndrome in very young infants . Am J Dis Child 1971;;122:507-512.
22 +
Row PG, Cameron JS, Turner DR, et al:  Membranous nephropathy: Long-term follow-up and association with neoplasia . Q J Med 1975;;44:207-239.
23 +
Glassock RJ, Bennett CM:  The glomerulopathies , in Brenner BM, Rector RC (eds): The Kidney . Philadelphia, WB Saunders Co, 1976;, p 1003.
24 +
Baldwin DS, Gluck MC, Lowenstein J, et al:  Lupus nephritis: Clinical course as related to morphologic forms and their transitions . Am J Med 1977;;62:12-30.
25 +
Levy M, Gagnadoux MF, Beziau A, et al:  Membranous glomerulonephritis associated with anti-tubular and anti-alveolar basement membrane antibodies . Clin Nephrol 1978;;10:158-165.
26 +
Tung KSK, Black WC:  Association of renal glomerular and tubular immune complex disease and antitubular basement antibody . Lab Invest 1975;;32:696-700.
27 +
Buckley RH, Becker WG:  Abnormalities in the regulation of human IgE synthesis . Immunol Rev 1978;;41:288-314.
28 +
Gatti RA, Kersey JH, Yunis EJ, et al:  Graft-versus-host disease . Prog Clin Pathol 1973;;5:1-181.

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