Ochronotic arthropathy in the context of spondyloarthritis differential diagnosis: a case-based review

被引:5
作者
Kostova, Tsvetelina [1 ]
Batalov, Zguro [1 ]
Karalilova, Rositsa [1 ]
Batalov, Anastas [1 ]
机构
[1] Med Univ Plovdiv, Dept Propaedeut Internal Med, Plovdiv, Bulgaria
关键词
Alkaptonuria; Ochronosis; Ochronotic arthropathy; Spondyloarthritis; ALKAPTONURIA; INVOLVEMENT; JOINT;
D O I
10.1007/s00296-022-05191-4
中图分类号
R5 [内科学];
学科分类号
1002 ; 100201 ;
摘要
Alkaptonuria is a disease often forgotten because of its rarity. Its pathogenic mechanism is the deficiency of one of the enzymes of the tyrosine degradation pathway-homogentisate-1, 2-dioxygenase, which sequelae is accumulation and deposition of its metabolite homogentisic acid in connective tissues and urine. Alkaptonuria presents as a clinical triad-darkening urine upon prolonged exposure to air, pigmentation of connective tissues and debilitating arthropathy. We present a case report of a 67-year old patient with alkaptonuria who presented with the clinical triad, but was mistakenly diagnosed as having ankylosing spondylitis in the past. Currently there is no treatment for the disease hence the management strategy was focused on symptoms control with analgesics, physical therapy, dietary modification, vitamin C supplementation, and joint arthroplasty. Alkaptonuria's clinical features are extensively described in the literature and despite the fact that it is a rare disease, due to the similar radiographic changes with spondyloarthropathies, it should be included in the differential diagnosis in young patients presenting with severe joint involvement. Early recognition of the disease is necessary since its natural evolution is joint destruction leading to significant reduction in the quality of life. Alkaptonuria's articular features in the spine and peripheral tissues are well described using the classical imaging techniques. Musculoskeletal ultrasonography shows a characteristic set of findings in the soft tissues, including synovium, cartilage, tendons and entheses.
引用
收藏
页码:2277 / 2282
页数:6
相关论文
共 23 条
[1]   Alkaptonuria: Spontaneous Achilles tendon rupture: Case report [J].
Baca, Emre ;
Kural, Alev ;
Ziroglu, Nezih ;
Kural, Cemal .
EKLEM HASTALIKLARI VE CERRAHISI-JOINT DISEASES AND RELATED SURGERY, 2019, 30 (03) :325-328
[2]   Ochronotic spondyloarthropathy: spinal involvement resembling ankylosing spondylitis [J].
Balaban, Birol ;
Taskaynatan, Mehmet ;
Yasar, Evren ;
Tan, Kenan ;
Kalyon, Tunc .
CLINICAL RHEUMATOLOGY, 2006, 25 (04) :598-601
[3]   Ochronotic arthropathy [J].
Borman, P ;
Bodur, H ;
Ciliz, D .
RHEUMATOLOGY INTERNATIONAL, 2002, 21 (05) :205-209
[4]   A Mimic of Ankylosing Spondylitis, Ochronosis: Case Report and Review of the Literature [J].
Chu, Philip ;
Cuellar, Maria C. ;
Bracken, Sonali J. ;
Tarrant, Teresa K. .
CURRENT ALLERGY AND ASTHMA REPORTS, 2021, 21 (03)
[5]  
Damian LO, 2013, MED ULTRASON, V15, P321
[6]   Black joint and synovia: Histopathological evaluation of degenerative joint disease due to Ochronosis [J].
Doganavsargil, Basak ;
Pehlivanoglu, Burcin ;
Bicer, Elcil Kaya ;
Argin, Mehmet ;
Bingul, Kenan Baris ;
Sezak, Murat ;
Kececi, Burcin ;
Coker, Mahmut ;
Oztop, Fikri .
PATHOLOGY RESEARCH AND PRACTICE, 2015, 211 (06) :470-477
[7]  
Drakoulakis Emmanouil, 2012, Am J Orthop (Belle Mead NJ), V41, P80
[8]   The molecular basis of alkaptonuria [J].
FernandezCanon, JM ;
Granadino, B ;
deBernabe, DBV ;
Renedo, M ;
FernandezRuiz, E ;
Penalva, MA ;
deCordoba, SR .
NATURE GENETICS, 1996, 14 (01) :19-24
[9]   Tendon involvement in patients with ochronosis: an ultrasonographic study [J].
Filippou, G. ;
Frediani, B. ;
Selvi, E. ;
Bertoldi, I. ;
Galeazzi, M. .
ANNALS OF THE RHEUMATIC DISEASES, 2008, 67 (12) :1785-1786
[10]   Family history of ochronotic arthropathy [J].
Gomez-Lechon Quiros, L. ;
Hidalgo Calleja, C. ;
Acosta de la Vega, M. E. ;
Compan Fernandez, O. ;
Pastor Navarro, S. ;
Montilla Morales, C. .
RHEUMATOLOGY INTERNATIONAL, 2021, 41 (10) :1869-1874