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A case of synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO)
syndrome complicated by IgA nephropathy with nephrotic syndrome
#MMPMID28509162
Morimoto K
; Nakatani K
; Asai O
; Mondori K
; Tomiwa K
; Mondori T
; Nakagawa Y
; Iwano M
; Shiiki H
CEN Case Rep
2016[May]; 5
(1
): 26-30
PMID28509162
show ga
A 62-year-old man visited our hospital with a mild sore throat, high-grade fever,
and clavicular pain. Seven years earlier, he had been diagnosed with synovitis,
acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome. His clavicles were
tender and remarkably swollen. Also noted was marked pitting edema in the lower
extremities and pustulosis on the palms and soles of the feet. Laboratory studies
on admission showed an elevated white cell count (23,400/?l) and serum C-reactive
protein level (24.4 mg/dl). Urinalysis revealed proteinuria (2+) and occult blood
(3+) with numerous dysmorphic red blood cells and hyalin casts. The patient was
diagnosed with recurrence of his SAPHO syndrome and started on oral
glucocorticoid therapy. By day 9 after admission, he had gained 16 kg in body
weight, and his proteinuria (6.4 g/day) and serum creatinine level (2.3 mg/dl)
were elevated. Renal biopsy revealed mesangial proliferative glomerulonephritis
with deposition of IgA and C3 in the mesangial area and along the capillary
walls. The patient was diagnosed with IgA nephropathy accompanied by nephrotic
syndrome. With oral prednisolone therapy, his fever, clavicular pain, and
proteinuria were gradually relieved. The clinical course in this case suggests
the onset of nephrotic syndrome with IgA nephropathy was associated with the
recurrence of the patient's SAPHO. To our knowledge, this is the first reported
case of SAPHO-associated IgA nephropathy.
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