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The efficacy of adalimumab in psoriatic arthritis concomitant to overlapping primary biliary cholangitis and primary sclerosing cholangitis: a case report

View Article: PubMed Central - PubMed

ABSTRACT

Background: The overlap syndrome of primary biliary cholangitis (formerly called primary biliary cirrhosis) and primary sclerosing cholangitis is an extremely rare condition that has never been described in association with other immune-mediated diseases, including psoriatic arthritis. While treatment with anti-Tumour Necrosis Factor-alpha (TNF-α) agents has proved to be effective in inflammatory arthropathies such as psoriatic arthritis, they have been employed in only a limited number of patients with autoimmune hepatitis, and their effectiveness is unclear.

Case presentation: We report the case of a 51-year-old female affected with psoriatic arthritis concomitant to overlapping primary biliary cholangitis and primary sclerosing cholangitis in whom 28 months of adalimumab treatment improved the symptoms of the inflammatory arthropathy as well as those of both cholangiopathies.

Conclusion: Our results suggest that further studies examining the therapeutic role of this particular TNF-α blocker are warranted in cholestatic autoimmune hepatitis patients, and in particular in those individuals in whom the disease is associated with inflammatory arthropathies.

No MeSH data available.


Related in: MedlinePlus

Right hand: arthritis of the fourth distal interphalangeal joint with severe nail psoriasis (a). Radiograph of the right hand: erosions in the fourth metacarpophalangeal and distal interphalangeal joints (b)
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Fig1: Right hand: arthritis of the fourth distal interphalangeal joint with severe nail psoriasis (a). Radiograph of the right hand: erosions in the fourth metacarpophalangeal and distal interphalangeal joints (b)

Mentions: In October 2012, arthritis of the fourth right DIP joint and severe nail psoriasis were two new symptoms that were added to the clinical picture (Fig. 1a). Hand X-rays showed new erosions in the fourth right MCF and DIP (Fig. 1b). Adalimumab treatment (40 mg fortnightly), which was begun at that time, led to notable relief of the pain and stiffness. Three months later the painful swellings of the second and fourth right DIP joints showed marked improvement, and the nail of the fourth right finger appeared normal (Fig. 2a). The patient’s Health Assessment Questionnaire (HAQ) disability index score fell from 0.750 to 0.375, the Disease Activity Score 28 (DAS28) fell from 4.25 to 3.18 and the Disease Activity in Psoriatic Arthritis (DAPSA) score fell from 17.37 to 4.29. Liver function tests improved, the ALP normalized, and the γGT and IgM improved (respectively falling to 124 IU/L and 5.35 g/L). Twelve months later, the HAQ continued to fall reaching 0.25, the DAS 28 fell to 1.80, and the DAPSA to 2.69. Hand X-rays showed that the bone erosion infourth right DIP joint seemed to have disappeared while the erosion at the base of the first phalanx persisted (Fig. 2b). Unfortunately, we do not have Computerized Tomography (CT) or MRI confirmation of these findings. Twenty-eight months later, the patient’s HAQ and DAS 28 values were stable, the DAPSA was 5.29 (low disease activity), the γGT and serum IgM had fallen even further (respectively reaching 98 IU/ml, and 4.93 g/L), and the ALP remained at normal levels (107 IU/ml) (Fig. 3). At present, the patient continues to receive Adalimumab and enjoys good health.Fig. 1


The efficacy of adalimumab in psoriatic arthritis concomitant to overlapping primary biliary cholangitis and primary sclerosing cholangitis: a case report
Right hand: arthritis of the fourth distal interphalangeal joint with severe nail psoriasis (a). Radiograph of the right hand: erosions in the fourth metacarpophalangeal and distal interphalangeal joints (b)
© Copyright Policy - OpenAccess
Related In: Results  -  Collection

License 1 - License 2
Show All Figures
getmorefigures.php?uid=PMC5120445&req=5

Fig1: Right hand: arthritis of the fourth distal interphalangeal joint with severe nail psoriasis (a). Radiograph of the right hand: erosions in the fourth metacarpophalangeal and distal interphalangeal joints (b)
Mentions: In October 2012, arthritis of the fourth right DIP joint and severe nail psoriasis were two new symptoms that were added to the clinical picture (Fig. 1a). Hand X-rays showed new erosions in the fourth right MCF and DIP (Fig. 1b). Adalimumab treatment (40 mg fortnightly), which was begun at that time, led to notable relief of the pain and stiffness. Three months later the painful swellings of the second and fourth right DIP joints showed marked improvement, and the nail of the fourth right finger appeared normal (Fig. 2a). The patient’s Health Assessment Questionnaire (HAQ) disability index score fell from 0.750 to 0.375, the Disease Activity Score 28 (DAS28) fell from 4.25 to 3.18 and the Disease Activity in Psoriatic Arthritis (DAPSA) score fell from 17.37 to 4.29. Liver function tests improved, the ALP normalized, and the γGT and IgM improved (respectively falling to 124 IU/L and 5.35 g/L). Twelve months later, the HAQ continued to fall reaching 0.25, the DAS 28 fell to 1.80, and the DAPSA to 2.69. Hand X-rays showed that the bone erosion infourth right DIP joint seemed to have disappeared while the erosion at the base of the first phalanx persisted (Fig. 2b). Unfortunately, we do not have Computerized Tomography (CT) or MRI confirmation of these findings. Twenty-eight months later, the patient’s HAQ and DAS 28 values were stable, the DAPSA was 5.29 (low disease activity), the γGT and serum IgM had fallen even further (respectively reaching 98 IU/ml, and 4.93 g/L), and the ALP remained at normal levels (107 IU/ml) (Fig. 3). At present, the patient continues to receive Adalimumab and enjoys good health.Fig. 1

View Article: PubMed Central - PubMed

ABSTRACT

Background: The overlap syndrome of primary biliary cholangitis (formerly called primary biliary cirrhosis) and primary sclerosing cholangitis is an extremely rare condition that has never been described in association with other immune-mediated diseases, including psoriatic arthritis. While treatment with anti-Tumour Necrosis Factor-alpha (TNF-α) agents has proved to be effective in inflammatory arthropathies such as psoriatic arthritis, they have been employed in only a limited number of patients with autoimmune hepatitis, and their effectiveness is unclear.

Case presentation: We report the case of a 51-year-old female affected with psoriatic arthritis concomitant to overlapping primary biliary cholangitis and primary sclerosing cholangitis in whom 28 months of adalimumab treatment improved the symptoms of the inflammatory arthropathy as well as those of both cholangiopathies.

Conclusion: Our results suggest that further studies examining the therapeutic role of this particular TNF-α blocker are warranted in cholestatic autoimmune hepatitis patients, and in particular in those individuals in whom the disease is associated with inflammatory arthropathies.

No MeSH data available.


Related in: MedlinePlus