{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,8,28]],"date-time":"2025-08-28T10:10:03Z","timestamp":1756375803095,"version":"3.44.0"},"reference-count":14,"publisher":"BMJ","issue":"7","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Rep"],"accepted":{"date-parts":[[2024,6,18]]},"published-print":{"date-parts":[[2024,7]]},"abstract":"<jats:p>Sjogren\u2019s syndrome is a known cause of renal tubular acidosis (RTA). However, osteomalacia associated with Sjogren\u2019s syndrome is rare and seldom reported in literature. We report a case of pseudofractures of both femora due to osteomalacia as a result of RTA secondary to Sjogren\u2019s syndrome, which was initially misdiagnosed as a stress fracture. A man in his 30s presented with hip pain and was initially misdiagnosed to have stress fractures because of the \u2018through and through\u2019 extension of the \u2018fracture\u2019 lines at the neck of both femora. The patient had a normal serum biochemistry profile except for elevated alkaline phosphatase levels. On further evaluation, he was found to have distal RTA secondary to Sjogren\u2019s syndrome. The patient responded to sodium bicarbonate therapy with clinical, biochemical and radiological improvement. A high index of suspicion for RTA should be kept in a patient with osteomalacia with a normal calcium profile and vitamin D level.<\/jats:p>","DOI":"10.1136\/bcr-2023-256661","type":"journal-article","created":{"date-parts":[[2024,7,2]],"date-time":"2024-07-02T23:50:25Z","timestamp":1719964225000},"page":"e256661","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":1,"title":["Unusual presentation of Sjogren\u2019s syndrome"],"prefix":"10.1136","volume":"17","author":[{"given":"Soham","family":"Mukherjee","sequence":"first","affiliation":[{"name":"Endocrinology, Post Graduate Institute of Medical Education and Research, Chandigarh, India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Durairaj","family":"Arjunan","sequence":"additional","affiliation":[{"name":"Endocrinology, Post Graduate Institute of Medical Education and Research, Chandigarh, India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0410-8778","authenticated-orcid":false,"given":"Sanjay","family":"Bhadada","sequence":"additional","affiliation":[{"name":"Endocrinology, Post Graduate Institute of Medical Education and Research, Chandigarh, India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Amal","family":"Shaharyar","sequence":"additional","affiliation":[{"name":"Endocrinology, Post Graduate Institute of Medical Education and Research, Chandigarh, India"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"239","published-online":{"date-parts":[[2024,7,2]]},"reference":[{"key":"2025082802515817000_17.7.e256661.1","doi-asserted-by":"publisher","DOI":"10.1097\/01.ASN.0000013882.73122.2B"},{"key":"2025082802515817000_17.7.e256661.2","doi-asserted-by":"crossref","first-page":"72","DOI":"10.1159\/000087537","article-title":"Distal renal tubular acidosis: alkali heals osteomalacia and increases net production of 1, 25-dihydroxyvitamin D","volume":"101","author":"Wrong","year":"2005","journal-title":"Nephron Physiol"},{"key":"2025082802515817000_17.7.e256661.3","doi-asserted-by":"crossref","unstructured":"Sabatini S , Kurtzman NA . Overall acid-base regulation by the kidney. In: Seldin DW , Giebisch G , eds. Regulation of Acid-Base Balance. New York: Raven Press, 1989: 185\u2013210.","DOI":"10.1007\/978-1-4613-0567-5_29"},{"key":"2025082802515817000_17.7.e256661.4","first-page":"525","article-title":"Review of the diagnostic evaluation of renal tubular acidosis","volume":"16","author":"Yaxley","year":"2016","journal-title":"Ochsner J"},{"key":"2025082802515817000_17.7.e256661.5","doi-asserted-by":"crossref","first-page":"123","DOI":"10.1007\/s13730-012-0027-0","article-title":"Osteomalacia secondary to renal tubular acidosis due to Sj\u00f6gren\u2019s syndrome: a case report and review of the literature","volume":"1","author":"Nagae","year":"2012","journal-title":"CEN Case Rep"},{"key":"2025082802515817000_17.7.e256661.6","doi-asserted-by":"crossref","first-page":"35","DOI":"10.1002\/art.39859","article-title":"American college of rheumatology\/European league against rheumatism classification criteria for primary Sj\u00f6gren\u2019s syndrome: a consensus and data-driven methodology involving three international patient cohorts","volume":"69","author":"Shiboski","year":"2017","journal-title":"Arthritis Rheumatol"},{"key":"2025082802515817000_17.7.e256661.7","doi-asserted-by":"publisher","DOI":"10.1001\/archinte.1996.00440140045004"},{"key":"2025082802515817000_17.7.e256661.8","doi-asserted-by":"crossref","first-page":"679","DOI":"10.1007\/s12098-007-0120-0","article-title":"Evaluation of renal tubular acidosis","volume":"74","author":"Bagga","year":"2007","journal-title":"Indian J Pediatr"},{"key":"2025082802515817000_17.7.e256661.9","doi-asserted-by":"publisher","DOI":"10.1172\/JCI119694"},{"key":"2025082802515817000_17.7.e256661.10","doi-asserted-by":"publisher","DOI":"10.1136\/bmj.2.4723.91"},{"key":"2025082802515817000_17.7.e256661.11","first-page":"268","article-title":"Fatigue fractures","volume":"108","author":"Morris","year":"1968","journal-title":"Calif Med"},{"key":"2025082802515817000_17.7.e256661.12","doi-asserted-by":"crossref","first-page":"392","DOI":"10.12659\/AJCR.907503","article-title":"Sj\u00f6gren\u2019s syndrome associated with fanconi\u2019s syndrome and osteomalacia","volume":"19","author":"Gutierrez","year":"2018","journal-title":"Am J Case Rep"},{"key":"2025082802515817000_17.7.e256661.13","doi-asserted-by":"publisher","DOI":"10.1172\/JCI110586"},{"key":"2025082802515817000_17.7.e256661.14","doi-asserted-by":"crossref","first-page":"271","DOI":"10.5001\/omj.2011.66","article-title":"Distal renal tubular acidosis, hypokalemic paralysis, nephrocalcinosis, primary hypothyroidism, growth retardation, osteomalacia and osteoporosis leading to pathological fracture: a case report","volume":"26","author":"Basak","year":"2011","journal-title":"OMJ"}],"container-title":["BMJ Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bcr-2023-256661","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,8,28]],"date-time":"2025-08-28T09:52:06Z","timestamp":1756374726000},"score":1,"resource":{"primary":{"URL":"https:\/\/casereports.bmj.com\/lookup\/doi\/10.1136\/bcr-2023-256661"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,7]]},"references-count":14,"journal-issue":{"issue":"7","published-online":{"date-parts":[[2024,7,2]]},"published-print":{"date-parts":[[2024,7]]}},"alternative-id":["10.1136\/bcr-2023-256661"],"URL":"https:\/\/doi.org\/10.1136\/bcr-2023-256661","relation":{},"ISSN":["1757-790X"],"issn-type":[{"type":"electronic","value":"1757-790X"}],"subject":[],"published":{"date-parts":[[2024,7]]}}}