Infliximab Treatment in Pachydermoperiostosis

Costa, Flávia Nunes; Fialho, Sônia Cristina de Magalhães Souza; Zimmermann, Adriana Fontes; Neves, Fabrício Souza; Castro, Gláucio Ricardo Werner de; Pereira, Ivânio Alves

doi:10.1097/rhu.0b013e3181df91c6

Cited by 23 publications

(9 citation statements)

References 11 publications

(6 reference statements)

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“…Other publications described inflammation and bone metabolism were also involved in PDP 23 , 45 . IL-6, TNFα and RANKL are three of the most important factors.…”

Section: Discussionmentioning

confidence: 99%

“…It is produced by lots of cell types involved in inflammation. TNFα can stimulate PGE 2 synthesis and can also increase RANKL expression 23 , 48 . RANKL is a member of the TNF superfamilies.…”

Section: Discussionmentioning

confidence: 99%

“…Besides the PGE 2 metabolism, inflammation and unbalance between the bone resorption and osteogenesis also play important roles in PDP 3 , 4 , 7 . Several cytokines and cell receptors, such as interleukin-6 (IL-6), tumor necrosis factor-alpha (TNFα), receptor activator of nuclear factor Kappa ligand (RANKL), were reported increasing in serum and synovial fluid of PDP patients 21 – 23 . We detected the IL-6, TNFα and RANKL expression in hypertrophic gastric mucosa of PDP patients for the first time.…”

Section: Introductionmentioning

confidence: 99%

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Interleukin-6, tumor necrosis factor-alpha and receptor activator of nuclear factor kappa ligand are elevated in hypertrophic gastric mucosa of pachydermoperiostosis

Huang

Wang

Cao

et al. 2017

Sci Rep

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Pachydermoperiostosis (PDP) is a rare inherited multisystem disease characterized with digital clubbing, pachydermia and periostosis. Variants in either HPGD or SLCO2A1 that interrupt the prostaglandin E2 (PGE2) pathway have been shown to be involved in PDP. Here, in addition to six confirmed variants in HPGD or SLCO2A1, we identified four novel SLCO2A1 variants in eight PDP patients from seven Chinese Han families. In addition, gastric mucosa hyperplasia was observed in all affected individuals and interleukin-6 (IL-6), tumor necrosis factor-alpha (TNFα) and receptor activator of nuclear factor kappa ligand (RANKL) expression were elevated in hypertrophic gastric mucosa. Two of eight patients who had severe arthralgia were treated with celecoxib. After three months, their arthralgia was partly relieved and IL-6, TNFα and RANKL expression were decreased in accordance with their relieved hypertrophic gastric mucosa. Our study broadens the variation spectrum of SLCO2A1 and suggests that the gastric mucosa hyperplasia might be a common characteristic of PDP. Moreover, celecoxib would be a considerable choice for PDP patients. We also revealed that IL-6, TNFα and RANKL may play important roles in the molecular mechanisms of gastric mucosa hyperplasia in PDP for the first time.

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“…Other publications described inflammation and bone metabolism were also involved in PDP 23 , 45 . IL-6, TNFα and RANKL are three of the most important factors.…”

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Interleukin-6, tumor necrosis factor-alpha and receptor activator of nuclear factor kappa ligand are elevated in hypertrophic gastric mucosa of pachydermoperiostosis

Huang

Wang

Cao

et al. 2017

Sci Rep

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“…Retinoids have been shown to reduce procollagen production by diminishing procollagen mRNA in fi broblasts, thus inhibiting the production of collagenase and improving skin lesions in pachydermia: fi broblast biosynthetic activity in the skin lesions of pachydermoperiostosis demonstrates an alteration which may be responsible, at least partly, for the patient's phenotype (1a). Infl iximab, tumor necrosis factor alpha (TNF-alpha) inhibitor, may be eff ective in the resorption of newly formed bones (18). Surgical treatment has been mainly focused on cosmetic and functional improvement (19).…”

Section: Discussionmentioning

confidence: 99%

Congenital Primary Pachydermoperiostosis and Striate Palmoplantar Keratoderma - a Case Report

Stojanović

Vuckovic

Jovanović

et al. 2014

Serbian Journal of Dermatology and Venereology

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The authors present a rare case of congenital pachydermoperiostosis associated with striate palmoplantar keratoderma in a 55-year-old female. Pachydermoperiostosis (PDP) is a heterogeneous syndrome characterized by hypertrophic changes involving predominantly the skin and bones of the extremities: pachydermia, clubbing of the fi ngers and toes, and hypertrophic osteoarthropathy. Primary pachydermoperiostosis (Touraine-Solente-Gole syndrome) (PPDP) or primary hypertrophic osteoarthropathy (PHO) is a rare congenital disorder and is one of two types of hypertrophic osteoarthropathy. In addition to the three main criteria, which are confi rmed clinically, histologically, and by X-ray, there may be other additional clinical features. Hyperhidrosis of the hands and feet may be troublesome. The skin of the face, forehead and scalp becomes grossly thickened and thrown into folds. The folding of the scalp produces a form of cutis verticis gyrata. Additional clinical features include hypohidrosis, seborrhea, sebaceous gland hyperplasia and folliculitis, carpal and tarsal tunnel syndrome, chronic leg ulcers and calcifi cation in the Achilles tendon. Our patient presented with most of these additional clinical features, such as acro-osteolysis of the fi ngers and toes, which generally occurs occasionally. In regard to palmoplantar keratoderma, we have not found reports of its association with PPDP in the available literature. Unlike PPDP, secondary pachydermoperiostosis (secondary hypertrophic osteoarthropathy -SHO) occurs in association with severe pulmonary disease such as bronchiectasis, abscess, bronchial carcinoma, pleural mesothelioma, or thymic, esophageal or stomach cancer, which were all excluded in our patient. In conclusion, this paper presents a congenital form of pachydermoperiostosis in a female also suffering from striate keratoderma. According to the available literature, this is the fi rst case report of comorbidity between these two dermatoses.

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“…Важная роль в патогенезе ГОА принадлежит различ-ным цитокинам и клеточным рецепторам, в том числе ин-терлейкину 6 и компонентам системыRANKL-RANK-ос-теопротегерин. Считается, что избыточная продукция этих цитокинов сопровождается повышением уровней продук-тов деградации С-концевых телопептидов коллагена I ти-па, соотношения гидроксипролин/креатинина в моче вме-сте со сниженными уровнями костной щелочной фосфата-зы; таким образом, предполагается, что ГОА характеризу-ется повышенной костной резорбцией [19]. Активация сигнального Wnt-пути при первичной ГОА способствует развитию пахидермии путем усиления функции дермаль-ных фибробластов [20].…”

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Primary idiopathic hypertrophic osteoarthropathy in a child: characteristics of the disease with a case report

Любимова¹,

Snegireva²,

Терентьев³

et al. 2017

RJTAO

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Infliximab Treatment in Pachydermoperiostosis

Cited by 23 publications

References 11 publications

Interleukin-6, tumor necrosis factor-alpha and receptor activator of nuclear factor kappa ligand are elevated in hypertrophic gastric mucosa of pachydermoperiostosis

Interleukin-6, tumor necrosis factor-alpha and receptor activator of nuclear factor kappa ligand are elevated in hypertrophic gastric mucosa of pachydermoperiostosis

Congenital Primary Pachydermoperiostosis and Striate Palmoplantar Keratoderma - a Case Report

Primary idiopathic hypertrophic osteoarthropathy in a child: characteristics of the disease with a case report

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