Bamboo Spine Mimicking Bekhterev's Disease Caused by Long-term Acitretin Treatment

Abstract

Sir.During the last 20 years several papers have been published regarding the skeletal toxicity of long-term retinoid therapy. The literature has been conflicting. Soineeentres repori development of skeletal hyperostosis due to oral retinoids (1-6). Other investigators do not report radiographic changes during either short-term retinoid therapy (7) or prolonged treatment in adults (8). Osteoporosis caused by acitretin (9, 10) has also been discussed. We here report on 3 patients with lamellar iehthyosis who have been treated with acitretin lor several years and developed spinal hyperostoses. CASE REPORTSPatient 1 is a 60-year-old woman with lamellar iehthyosis. She was given etretinate 0.75 tng/kg/day from 1979 and later switehed to aeilretin 0.5 tng/kg/day continuously, and she is still using this medication. In 2001 she complained of increased stiffness in her spine and lower back pain. X-ray examination ofthe spine showed extensive hyperostosis-like bamboo spine and initially the diagnosis of Bckhterev's disease (spondylitis ankylopoetica) was suspected. However, X-ray of her iliosaeral joints was normal and she was HLA B27 negative. The diagnosis of Bekhtcrev's disease was therefore ruled out by her rheumatologist.Patient 2 is a 69-year-old man with lamellar iehthyosis. He has used retinoids approximately 0.5 mg/kg/day continuously since I97S. During the last years he has complained of lower baek pain and increased stiffness of his spine. X-ray examination showed extensive hyperostosis-like batnboo spine. His iliosaeral joints were radiographically normal and he was HLA B27 negative.Patient 3 is a 45-year-old wotnan with lamellar iehthyosis. Since 1978 she has used etretinate 0.7 mg/kg/day and, from 1992, acitretin 0.5 mg/kg/day continuously. She experienced increased stiffness of her spine since 2001 and X-ray examination showed hyperostosis-like bamboo spine (Fig. I). She was HLA B27 negative and her iliosaeral joints were normal. DISCUSSIONDifferent dermatologieal centres monitor patients on long-term retinoid therapy differently regarding bone toxieity. Sotne centres do not make X-ray skeletal investigations, while others do. Interestingly several Fig. /, Conventional X-ray imjigeoflumharspincuuinlero-posteriorprojeetion in patieni 3. Syndesmopiiyles similar to those seen in Hekliterev's disease lateral to the intervcrlehral discs {arrows).centres have not detected hyperostosis in their patients on long-term retinoid therapy. Whether this is explained by the lack of skeletal monitoring or genetic differences between groups of patients is not known. In our departtnent we have, during the last 20 years, routinely perfortned X-ray surveys ofthe skeleton at baseline and every second year in patients on long-term retinoid treattnent. We experienced two clearly abnortnal calcifieations (5) in the forearm ofa patient with X-Iinked recessive iehthyosis and a latnlnar hyperostotic process in the forearm in a 66-year old woman with pustulosis palmoplantaris.In our previous study (5) radiographs ofthe spine were of ...

Hiperceratose epidermolítica: um seguimento de 23 anos de uso de retinoides orais

“…8 By contrast, a study by Mork et al with three patients dosed with 0.5 to 0.75 mg/kg day for 22, 28 and 14 years, spinal hyperostosis was observed in all three 3 cases. 9 These findings are consistent with those of Bondson et al who report that in a case where the retinoid dose was 25 mg/day for 20 years, changes in lipid levels and transaminases were observed. 10 Reports of use of the medication for shorter periods (6 months to 2 years), no significant systemic adverse events even at higher dosage (1mg/kg/day) were noted.…”

British Association of Dermatologists guidelines on the efficacy and use of acitretin in dermatology

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Spondyloarthritic Changes During Acitretin Treatment

“…5 Nevertheless, arthralgia, myalgia, arthritis, skeletal hyperostosis and extraosseous calcification may emerge during maintenance treatment or over time. 6,7 Bone pain, arthralgia and myalgia are common but new hyperostotic lesions and extraskeletal calcification may occur very rare. As distinct from this, nr-axSpA occurred in this case without other significant rheumatological manifestations.…”