Article information
Abstract
The primary hypertrophic osteoarthropathy (pachydermoperiostosis) is a hereditary disease characterized by skin thickening (pachydermia), finger clubbing, and proliferation of periosteum (periostitis) with subperiosteal new bone formation. We describe the cases of 2 brothers of 30 and 24 years, who consulted due to bone pain, arthralgia, and oligoarthritis. Pachydermia, hyperhidrosis, seborrhea, digital clubbing, periostosis, and non-inflammatory effusions of the knees. The first had been diagnosed with juvenile idiopathic arthritis at age 15, while the youngest also presented with a thoracic scoliosis, hypertrophic gastritis, iron deficiency anemia, and glucose intolerance by pancreatic endocrine dysfunction. In both patients, symptoms were controlled satisfactorily with etoricoxib (90mg/day) and risedronate (35mg/week).
Keywords:
Primary hypertrophic osteoarthropathy
Pachydermoperiostosis
Arthritis
Hypertrophic gastritis
Digital clubbing
Bisphosphonates
Resumen
La osteoartropatía hipertrófica primaria (paquidermoperiostosis) es una enfermedad hereditaria caracterizada por engrosamiento cutáneo (paquidermia), dedos en palillo de tambor y proliferación perióstica (periostosis) con neoformación ósea subcortical. Describimos los casos de 2 hermanos de 30 y 24 años, que consultaron por dolores óseos, artralgias y oligoartritis. En ambos casos se objetivó paquidermia, hiperhidrosis, seborrea, hipocratismo digital, periostosis y derrame articular en las rodillas de características no inflamatorias. El primero había sido diagnosticado de artritis idiopática juvenil a los 15 años, mientras que el más joven también presentaba una escoliosis dorsal, gastritis hipertrófica, anemia ferropénica crónica e intolerancia a la glucosa por disfunción endocrina pancreática. Los síntomas se controlaron satisfactoriamente en ambos enfermos con etoricoxib (90mg/día) y risedronato (35mg/ semana).
Palabras clave:
Osteoartropatía hipertrófica primaria
Paquidermoperiostosis
Artritis
Gastritis hipertrófica
Dedos en palillo de tambor
Bisfosfonatos
Full text is only aviable in PDF
References
[1.]
M. Mattuci-Cerinic, T. Lotti, I. Jajic, A. Pignole, C. Bussani, M. Cagnoni.
The clinical spectrum of pachydermoperiostosis (primary hypertrophic osteoarthropathy).
Medicine (Balt), 70 (1991), pp. 208-214
[2.]
Z. Jajic, I. Jajic, T. Nemcic.
Primary hypertrophic osteoarthropathy: Clinical, radiologic, and scintigraphic characteristics.
Arch Med Res, 32 (2001), pp. 136-142
[3.]
J.A. Cavallasca, V.A. Malah, D.E. Fernández, S.G. Garbia, G.G. Nasswetter.
Paquidermoperiostosis (osteoartropatía hipertrófica primaria).
Medicina (Buenos Aires), 66 (2006), pp. 147-149
[4.]
J.C. Santos, M. Yuste, O. Martínez, M.T. Alonso, J. Sánchez.
Paquidermoperiostosis (síndrome de Touraine-Solente-Golé) Descripción de un caso.
Actas Dermosifiliogr, 98 (2007), pp. 116-120
[5.]
M. Auger, N. Stavrianeas.
Pachydermoperiostosis.
Orphanet Enciclopedia, (2004), pp. 1-8
[6.]
G.P. Sinha, P. Curtis, D. Haigh, G.T. Lealman, W. Dods, C.P. Bennett.
Pachydermoperiostosis in childhood.
Br J Rheumatol, 36 (1997), pp. 1224-1227
[7.]
N. Friedreich.
Hyperostose des gesammten Skelettes.
Virchows Arch Anat, 43 (1868), pp. 83-87
[8.]
A. Touraine, G. Solente, L. Golé.
Un syndrome ostéodermatopathique: la pachydermie plicaturée avec pachyériostose des extrémités.
Press Med, 43 (1935), pp. 1820-1824
[9.]
Z. Jajic, I. Jajic.
Prevalence of primary hypertrophic osteoarthropathy in selected population.
Clin Exp Rheum, 10 (1992), pp. 73
[10.]
I.R. Mahy, J. Wiggins.
Pachydermoperiostosis mimicking acromegaly.
J R Soc Med, 85 (1992), pp. 46-47
[11.]
D. Shari, M. Momeni.
Primary hypertrophic osteoarthropathy in a patient with rheumatoid arthritis.
J C R, 13 (2007), pp. 242-243
[12.]
M. Martínez-Lavin, C. Pineda, T. Valdez, J.C. Cajigas, M. Weisman, N. Gerber, et al.
Primary hypertrophic osteoarthropathy.
Sem Arthritis Rheum, 17 (1988), pp. 156-162
[13.]
Kerimovic-Morina DJ, Mladenovic V. Primary hyperthrophic osteoarthropathy in 32 patients. Clin Exp Rheumatol. 1002;10:51–6.
[14.]
A. Latos-Bielenska, I. Marik, M. Kuklic, A. Materna-Kiryluk, K. Kozlowski.
Pachydermoperiostosis: Critical analysis with report of five unusual cases.
J Pediatric, 166 (2007), pp. 1237-1243
[15.]
M. Castori, L. Sinibaldi, R. Mingarelli, R.S. Lachman, D.L. Rimoin, B. Dallapiccola.
Pachydermoperiostosis: An update.
Clin Genet, 68 (2005), pp. 477-486
[16.]
I. Jajic, Z. Jajic, M. Ozie-Bebek.
HLA antigens and primary hypertrophic osteoarthropathy.
Clin Exp Rheum, 10 (1992), pp. 74
[17.]
A. Ángel-Moreno, E. Martínez, L. Suárez, J.L. Pérez.
Painful hypertrophic osteoarthropathy treated with octreotide. The pathogenic role of vascular endothelial growth factor (VEGF).
Rheumatology, 44 (2005), pp. 1327-1328
[18.]
S. Uppal, C.P. Diggle, I.M. Carr, C.W. Fishwick, M. Ahmed, G.H. Ibrahim, et al.
Mutations in 15-hydroxyprostaglandin dehydrogenase cause primary hypertrophic osteoarthropathy.
Nat Genet, 40 (2008), pp. 789-793
[19.]
D. Rendina, G. de Filippo, R. Viceconti, E. Soscia, C. Sirignano, M. Salvatore, et al.
Interleukin (IL)-6 and receptor activator of nuclear factor (NF)-KappaB ligand (RANKL) are increased in the serum of a patient with primary pachydermoperiostosis.
Scand J Rheumatol, 37 (2008), pp. 225-229
[20.]
T. Seyhan, Ö.R. Özerdem, C. Aliagaoglu.
Severe complete pachydermoperiostosis (Touraine-Solente-Golé syndrome).
Dermatol Surg, 31 (2005), pp. 1465-1467
[21.]
E. Monteiro, P. Carvalho, A. Silva, A. Ferraro.
Frontal rhytidectomy: A new approach to improve deep wrinkles in a case of pachydermoperiostosis.
Plast Reconstr Surg, 112 (2003), pp. 1189-1190
[22.]
S.K. Shinjo, E.F. Borba, C.R. Gonc-alves, M. Levy Neto.
Ankylosing spondylitis in a patient with primary hypertrophic osteoarthropathy: Association or coincidence?.
J Clin Rheumatol, 13 (2007), pp. 175
[23.]
S.K. Shinjo, M. Levy Neto, E.F. Borba.
Palindromic rheumatism associated with primary hypertrophic osteoarthropathy.
Clinics, 61 (2006), pp. 581-583
[24.]
P. Fietta, P. Manganelli.
Pachydermoperiostosis and psoriatic onychopathy: An unusual association.
JEADV, 17 (2003), pp. 73-76
[25.]
P. Fietta, P. Manganelli, Carnitine:.
A therapeutic option for childhood psoriatic onycho-pachydermo-periostitis.
Clin Rheum, 26 (2007), pp. 93-94
[26.]
F. Gaston-Garrete, L. Porteau-Cassard, V. Marc, L. Zabraniecki, E. Ginesty, V. Andrieu, et al.
A case of primary hypertrophic osteoarthropathy without skin involvement (Currarino's disease).
Rev Rheum Engl Ed, 65 (1998), pp. 591-593
[27.]
R. Seggewiss, T. Hess, C. Fiehn.
A family with a variant form of primary hypertrophic osteoarthropathy (HOA) restricted to the lower extremities.
Joint Bone Spine, 70 (2003), pp. 230-233
[28.]
B.G. Akdeniz, T. Seckin.
Periodontal and alveolar bone abnormalities associated with pachydermoperiostosis.
Periodontal Clin Investig, 23 (2001), pp. 5-10
[29.]
A.J. Reginato, V. Shiapachasse, R. Guerrero.
Familial idiopathic hypertrophic osteoarthropathy and craneal suture defects in children.
Skel Radiol, 8 (1982), pp. 105-109
[30.]
I. Jajic.
Femoral head osteonecrosis in primary hypertrophic osteoarthropathy.
Ann Rheum Dis, 52 (1993), pp. 386
[31.]
Z. Jajic, I. Jajic.
Radiological changes of short and flat bones in primary hypertrophic osteoarthropathy.
Ann Rheum Dis, 57 (1998), pp. 747-750
[32.]
S.K. Lam, W.K. Hui, J. Ho, K.P. Wong, J.I. Rotter, I.M. Samloff.
Pachydermoperiostosis, hypertrophic gastropathy, and peptic ulcer.
Gastroenterology, 84 (1983), pp. 834-839
[33.]
A. Pignone, A. Calabro, J.I. Rotter.
Gastric abnormalities in pachydermoperiostosis.
Clin Exp Rheumatol, 10 (1992), pp. 72
[34.]
F. Ikeda, H. Okada, M. Mizuno, H. Kawamoto, N. Okano, S. Hamazaki, et al.
Pachydermoperiostosis associated with juvenile polyps of the stomach and gastric adenocarcinoma.
J Gastroenterol, 39 (2004), pp. 370-374
[35.]
Y.W. Shim, J.S. Suh.
Primary hypertrophic osteoarthropathy accompanied by Crohn's disease: A case report.
Yonsei Med J, 38 (1997), pp. 319-322
[36.]
G. Sethuraman, A.K. Malhotra, B.K. Khaitan, V.K. Sharma, R. Kumar, G.K. Makharia, et al.
Familial pachydermoperiostosis in association with protein-losing enteropathy.
Clin Exp Dermatol, 31 (2006), pp. 531-534
[37.]
S.C.H. Lee, H.J. Moon, D. Cho, D.W. Ryang, S.J. Kim, I.K. Chun, et al.
Pachydermoperiostosis with cutaneous squamous cell carcinomas.
Int J Dermatol, 37 (1998), pp. 687-700
[38.]
E.R. Farmer, G.W. Hambrick, L.E. Shulman.
Papular mucinosis. A clinicopathologic study of four patients.
Arch Dermatol, 118 (1982), pp. 9-13
[39.]
M.M. Barraud-Klenovsek, J. Lubbe, G. Burg.
Primary digital clubbing associated with palmoplantar keratoderma.
Dermatology, 194 (1997), pp. 302-305
[40.]
M.H. Han, J.K. Koh, K.C. Moon.
Hypertrophic osteoarthrophaty associated with pyoderma gangrenosum.
Br J Dermatol, 142 (2000), pp. 562-564
[41.]
D.J. Armstrong, E.M. McCausland, G.D. Wright.
Hypertrophic pulmonary osteoarthropathy (HPOA) (Pierre Marie-Bamberger syndrome): Two cases presenting as acute inflammatory arthritis. Description and review of the literature.
Rheumatol Int, 27 (2007), pp. 399-402
[42.]
X. Vandemergel, G. Decaux.
Le point sur l’osteoarthropathie hypertrophique et l’hippocratisme digital.
Rev Med Brux, 24 (2003), pp. 88-94
[43.]
M. Martínez-Lavin, A. Vargas, M. Rivera-Viñas.
Hypertrophic osteoarthropathy: A palindrome with a pathogenic connotation.
Curr Opin Rheumatol, 20 (2008), pp. 88-91
[44.]
M. Matucci-Cerinic, L. Fattorini, G. Gerini, A. Lombardi, A. Pignone, N. Petrini, et al.
Colchicine treatment in a case of pachydermoperiostosis with acroosteolisis.
Rheumatol Int, 8 (1988), pp. 185-188
[45.]
S. Beauregard.
Cutis verticis gyrate pachydermoperiostosis. Several cases in the same family Initial results of the treatment of pachyderma with isotretinoin.
Ann Dermatol Venereol, 121 (1994), pp. 134-137
[46.]
A. Okten, I. Mungan, M. Kalyoncu, Z. Orbak.
Two cases with pachydermope- riostosis and discussion of tamoxifen citrate treatment for arthalgia.
Clin Dermatol, 26 (2007), pp. 8-11
[47.]
H. Maeda, K. Kumagai, F. Konnishi, Y. Katayamma, K. Hiyama, S. Ishioka, et al.
Successful treatment of arthralgia with tamoxifen citrate in a patient with pachydermoperiostosis.
Rheumatology, 39 (2000), pp. 1158-1159
[48.]
M.H. Guyot-Drouot, E. Solau-Gervais, B. Cortet, X. Deprez, P. Chastanet, A. Cotten, et al.
Rheumatologic manifestations of pachydermoperiostosis and preliminary experience with bisphosphonates.
J Rheumatol, 27 (2000), pp. 2418-2423
[49.]
H. Jojima, K. Kinoshita, M. Naito.
A case of pachydermoperiostosis treated by oral administration of a bisphosphonate and arthroscopic synovectomy.
Mod Rheumatol, 17 (2007), pp. 330-332
[50.]
L. Bianchi, C. Lubrano, A.M. Carrozzo, S. Iraci, M. Tomassoli, G. Spera, et al.
Pachydermoperiostosis: Study of epidermal grow factor and steroid receptors.
Br J Dermatol, 132 (1995), pp. 128-133
[51.]
A. Oikarinen, R. Palatsi, M. Kymaniemi, J. Keski-Oja, J. Ristelli, M. Kallioinen.
PDP: Analysis of the connective tissue abnormalities in one family.
J Am Acad Dermatol, 31 (1994), pp. 947-953
[52.]
A. Arinci, B. Tümerdem, M.A. Karan, N. Erten, N. Büyükbabani.
Ptosis caused by pachydermoperiostosis.
Ann Plast Surg, 49 (2002), pp. 322-325
Copyright © 2009. Sociedad Española de Reumatología and Colegio Mexicano de Reumatología
