Iatrogenic Osteomalacia: Report of Two Cases

Yamamoto, Setsu; Okada, Yosuke; Mori, Hiroko; Kurozumi, Akira; Torimoto, Keiichi; Arao, Tadashi; Tanaka, Yoshiya

doi:10.7888/juoeh.35.25

Cited by 10 publications

(34 citation statements)

References 17 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…However, to our knowledge, azathioprine has not been linked with metabolic bone diseases, including osteosclerosis, although rats given azathioprine acquire inhibited bone formation and mineralization. (17) Saccharose iron administered intravenously has caused FGF-23-mediated hypophosphatemic osteomalacia, (18-19) but our patient's plasma FGF-23 level was normal or only slightly increased when her eGFR was 48 mls/min, urinary excretion of phosphorus was consistently normal, and bone formation was active on iliac crest biopsy. BTMs usually showed elevated values (formation higher than resorption), but were normal in the one evaluation by SMBP.…”

Section: V) Discussionmentioning

confidence: 75%

Idiopathic Acquired Osteosclerosis in a Middle-Aged Woman With Systemic Lupus Erythematosus

Guañabens

Mumm

Gifre

et al. 2016

Journal of Bone and Mineral Research

View full text Add to dashboard Cite

Widely distributed osteosclerosis is an unusual radiographic finding with multiple causes. A 42-year-old premenopausal Spanish woman gradually acquired dense bone diffusely affecting her axial skeleton and focally affecting her proximal long bones. Systemic lupus erythematosus (SLE) diagnosed in adolescence had been well controlled. She had not fractured or received antiresorptive therapy, and she was hepatitis C virus antibody negative. Family members had low bone mass. Lumbar spine bone mineral density (BMD) measured by dual-photon absorptiometry (DPA) at age 17 years, while receiving glucocorticoids, was 79% the average value of age-matched controls. From ages 30 to 37 years, dual-energy X-ray absorptiometry (DXA) BMD Z-scores steadily increased in her lumbar spine from þ3.8 to þ7.9, and in her femoral neck from -1.4 to -0.7. Serum calcium and phosphorus levels were consistently normal, 25-hydroxyvitamin D (25OHD) <20 ng/mL, and parathyroid hormone (PTH) sometimes slightly increased. Her reduced estimated glomerular filtration rate (eGFR) was 38 to 55 mL/min. Hypocalciuria likely reflected positive mineral balance. During increasing BMD, turnover markers (serum bone-specific alkaline phosphatase [ALP], procollagen type 1 N propeptide [P1NP], osteocalcin [OCN], and carboxy-terminal cross-linking telopeptide of type 1 collagen [CTx], and urinary amino-terminal cross-linking telopeptide of type 1 collagen [NTx and CTx]) were 1.6-to 2.8-fold above the reference limits. Those of bone formation seemed increased more than those of resorption. FGF-23 was slightly elevated, perhaps from kidney disease. Serum osteoprotegerin (OPG) and TGFb1 levels were normal, but sclerostin (SOST) and receptor activator of nuclear factor kappa-B ligand (RANKL) were elevated. Serum multiplex biomarker profiling confirmed a high level of SOST and RANKL, whereas Dickkopf-1 (DKK-1) seemed low. Matrix metalloproteinases-3 (MMP-3) and -7 (MMP-7) were elevated. Iliac crest biopsy revealed tetracycline labels, no distinction between thick trabeculae and cortical bone, absence of peritrabecular fibrosis, few osteoclasts, and no mastocytosis. Then, for the past 3 years, BMD Z-scores steadily decreased. Skeletal fluorosis, mastocytosis, myelofibrosis, hepatitis C-associated osteosclerosis, multiple myeloma, and aberrant phosphate homeostasis did not explain her osteosclerosis. Mutation analysis of the LRP5, LRP4, SOST, and osteopetrosis genes was negative. Microarray showed no notable copy number variation. Perhaps her osteosclerosis reflected an interval of autoimmune-mediated resistance to SOST and/or RANKL.

show abstract

Section: V) Discussionmentioning

confidence: 75%

Idiopathic Acquired Osteosclerosis in a Middle-Aged Woman With Systemic Lupus Erythematosus

Guañabens

Mumm

Gifre

et al. 2016

Journal of Bone and Mineral Research

View full text Add to dashboard Cite

show abstract

“…( 20 ) Sixteen patients were females ( 14,16‐18,21‐23,25,26,33,35,36,38 ) and 14 males. ( 11‐13,15,19,20,24,27‐30,32,34,37 )…”

Section: Resultsmentioning

confidence: 99%

“…(14,32) Only 1 case report reported TRACP-5b level and it was high. (22) Urine NTX was measured twice and was high in 1 case (37) and normal in the other. (36) Normal levels of urine pyridinoline and deoxypyridinoline were also reported once.…”

Section: Laboratory Testsmentioning

confidence: 99%

“…(27) Osteomalacia signs and symptoms Bone or muscle pain was reported in 28 of the 30 cases. (11,(13)(14)(15)(16)(17)(18)(19)(20)(21)(23)(24)(25)(26)(27)(28)(29)(30)(31)(32)(33)(34)(35)(36)(37)(38) In 21 cases, the pain was associated with fractures or pseudofractures, (11,(13)(14)(15)(16)18,19,21,23,24,26,(28)(29)(30)(32)(33)(34)(36)(37)(38) in 7 cases there was pain despite no fractures or pseudofractures reported, (17,20,25,…”

Section: Laboratory Testsmentioning

confidence: 99%

“…Twenty patients had pseudofractures (14 of the chest, (12,14,15,19,21,22,24,26,29,30,32,(36)(37)(38) 14 of the lower limbs, (11,15,18,21,22,24,26,(28)(29)(30)32,33,37,38) 7 of the pelvis (15,16,21,22,24,26,29) , and 1 of the upper limb (26) ). Nine patients had fractures (7 of the lower limbs, (11,13,34,36) 6 of the pelvis, (11,13,14,16) 4 of the ribs, (12,14,19) and 3 of the vertebra (14,16,37) ). Six patients had both fractures and pseudofractures.…”

Section: Laboratory Testsmentioning

confidence: 99%

See 2 more Smart Citations

Osteomalacia as a Complication of Intravenous Iron Infusion: A Systematic Review of Case Reports

Vilaça

Velmurugan

Smith

et al. 2020

Journal of Bone and Mineral Research

View full text Add to dashboard Cite

Randomized control trials (RCTs) have shown that certain intravenous iron preparations can induce high levels of fibroblast growth factor 23 (FGF-23) and persistent hypophosphatemia. Repeated iron infusions may lead to prolonged hypophosphatemia and osteomalacia events not captured by RCTs. Several previous case reports have described skeletal adverse effects after repeated iron infusions. To characterize these effects, we conducted a systematic review of case reports. MEDLINE, Embase, Web of Science, and Cochrane databases were searched in March 2021. We selected case reports of patients ≥16 years old. Study quality was assessed using the tool from Murad and colleagues. We report the results in a narrative summary. We identified 28 case reports, reporting 30 cases. Ages ranged from 28 to 80 years (median 50 years). Most patients (n = 18) received ferric carboxymaltose (FCM), whereas 8 received saccharated ferric oxide (SFO) and 3 received iron polymaltose (IPM). All but 2 cases had more than five infusions (range 2 to 198, median 17). The lowest phosphate levels ranged from 0.16 to 0.77 mmol/L (median 0.36 mmol/L). Intact FGF-23 (iFGF-23) was high when measured. Serum 25OH vitamin D was low in 10 of 21 cases measured and 1,25(OH) 2 vitamin D in 12 of 18. Alkaline phosphatase was high in 18 of 22 cases. Bone or muscle pain was reported in 28 of the 30 cases. Twenty patients had pseudofractures, 9 had fractures, and 6 patients had both. All 15 available bone scans showed focal isotope uptake. Case reports tend to report severe cases, so potential reporting bias should be considered. Osteomalacia is a potential complication of repeated iron infusion, especially in patients with gastrointestinal disorders receiving prolonged therapy. Pain and fractures or pseudofractures are common clinical findings, associated with low phosphate, high iFGF-23, high alkaline phosphatase, and abnormal isotope bone scan. Discontinuing or switching the iron formulation was an effective intervention in most cases.

show abstract

Osteomalazie

Scharla¹

2018

DGIM Innere Medizin

View full text Add to dashboard Cite

Iatrogenic Osteomalacia: Report of Two Cases

Abstract: When a patient is diagnosed with unexplained pain, it is important to pay attention to the possibility of an iatrogenic etiology.

Cited by 10 publications

References 17 publications

Idiopathic Acquired Osteosclerosis in a Middle-Aged Woman With Systemic Lupus Erythematosus

Idiopathic Acquired Osteosclerosis in a Middle-Aged Woman With Systemic Lupus Erythematosus

Osteomalacia as a Complication of Intravenous Iron Infusion: A Systematic Review of Case Reports

Osteomalazie

Contact Info

Product

Resources

About