Renal Tubular Acidosis, Sjögren Syndrome, and Bone Disease

Fulop, Milford; Mackay, Meggan

doi:10.1001/archinte.164.8.905

Cited by 50 publications

(18 citation statements)

References 28 publications

(23 reference statements)

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“…3 A 2004 survey by Fulop and co-workers, based on the analysis of 250 patient records, has concluded that the occurrence of osteomalacia in patients with type 1 RTA need not be associated with Sjögren's syndrome. 4 This finding concurs with the present case.…”

Section: Case Reportsupporting

confidence: 93%

Renal tubular acidosis presenting as osteomalacia

Haridas¹,

Haridas²,

Mudrabettu³

2017

IJRCI

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Section: Case Reportsupporting

confidence: 93%

Renal tubular acidosis presenting as osteomalacia

Haridas¹,

Haridas²,

Mudrabettu³

2017

IJRCI

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“…-ATPase in the cortical collecting tubules was observed in the kidney biopsy specimen of a patient with distal RTA with SjS. Fulop and Mackay [12] documented that there seemed to be no better explanation for the bone disease in SjS than some combination of acidosis and hypophosphatemia. Coexisting vitamin D deficiency, when present, may be an aggravating factor.…”

Section: Discussionmentioning

confidence: 99%

“…Since the first report [14] of the case of SjS with osteomalacia was published in 1977, as far as we investigated, only 13 cases have been reported in English and Japanese so far [4][5][6][7][8][9][10][11][12][13][14][15][16]. Previous published case reports are summarized in Table 1.…”

Section: Discussionmentioning

confidence: 99%

Osteomalacia secondary to renal tubular acidosis due to Sjögren’s syndrome: a case report and review of the literature

et al. 2012

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A 43-year-old woman was admitted to our hospital because of generalized bone pain. Arterial blood gas showed pH 7.266, HCO 3 -13.5 mEq/l, and anion gap (AG) 12. Since her urine pH was 7.0 despite metabolic acidosis with normal AG, we diagnosed distal renal tubular acidosis (RTA). Serum phosphate was 2.5 mg/dl, the level of b2 microglobulin was 41100 lg/l, and aminoaciduria was present. These results indicated proximal tubular dysfunction. The radiograph showed pseudofracture in the pubic bone, indicating osteomalacia. Bone scintigram showed abnormal accumulations of 99mTc-HMDP in multiple joints. Then, her generalized bone pain was considered to be a symptom of osteomalacia. Despite the absence of overt Sicca syndrome, the evaluation of Sjögren's syndrome (SjS) as a cause of distal RTA was performed. Antibodies to the SS-A level was 127U/ml. Tear break-up time was 3 s bilaterally and salivary gland scintigraphy showed low uptake of 99mTc in the submandibular glands and the parotids. Thus, we diagnosed SjS finally. Gallium scintigraphy showed mild abnormal uptake in bilateral kidneys, suggesting acute tubulointerstitial nephritis. After treatment with prednisolone, alfacalcidol, and sodium bicarbonate, bone pain was remarkably relieved. Additionally, aminoaciduria disappeared and the level of b2 microglobulin decreased. We speculated that the coincidence of proximal tubular dysfunction and distal RTA cause a severe manifestation of osteomalacia.

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“…Mucosal biopsy from lower lip for lymphocytic infiltration of the minor salivary glands showed chronic sialadenitis with evidence of epimyoepithelial islands and fibrosis strongly suggestive of Sjogren's disease. Dry eye was confirmed by Schirmer's test which was strongly positive (1mm at 5mins).As the literature suggests the association of dRTA (also Primary Sjogren's) with MBD [8][9][10][11][12][13][14], skeletal survey was also performed which showed moderate anterior wedging of the vertebral bodies(Lund 's criteria) and USG picture of bilateral renal microliths. DEXA Scan was not performed as this facility was not available at that time.…”

Section: Discussionmentioning

confidence: 99%