Recurrent granulomatous cheilitis associated with Crohn’s disease successfully treated with ustekinumab: case report and literature review

Taxonera, Carlos; Alba, Cristina; Colmenares, Michel; Olivares, David; Rey, Enrique

doi:10.1177/1756284820934327

Cited by 10 publications

(6 citation statements)

References 34 publications

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“…This appears to be the first reported case of VDZ leading to remission, and adds to the two recently published cases of UST inducing remission in OFG + CD. 23,24 This shows the importance of targeting the underlying intestinal CD in treating OFG. Interestingly, UST has also recently been shown to be useful for another extraintestinal granulomatous condition, metastatic CD [MCD]: nine cases of MCD were treated with UST, and all responded with the first or second dose, with five achieving remission in a median time of 5 months.…”

Section: Discussionmentioning

confidence: 99%

Orofacial Granulomatosis Associated with Crohn’s Disease: a Multicentre Case Series

Phillips

Verstockt

Sładek

et al. 2021

Journal of Crohn's and Colitis

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Background Orofacial granulomatosis (OFG) is a rare syndrome that may be associated with Crohn’s disease (CD). We aimed to characterise this relationship and the management options in the biologic era. Methods This multicentre case series was supported by the European Crohn’s and Colitis Organisation (ECCO), and performed as part of the Collaborative Network of Exceptionally Rare case reports (CONFER) project. Clinical data were recorded in a standardised collection form. Results This report includes 28 patients with OFG associated with CD: 14 males (mean age of 32 years, ±12.4 SD) and 14 females (40.3 years, ±21.0 SD). Non-oral upper gastrointestinal tract involvement was seen in 6 cases and perianal disease in 11. The diagnosis of OFG was made prior to CD diagnosis in 2 patients, concurrently in 8 and after CD diagnosis in 18. The distribution of OFG involved the lips in 16 cases and buccal mucosa in 18. Pain was present in 25 cases, with impaired swallowing or speaking in 6. Remission was achieved in 23 patients, notably with the use of anti-TNFs in 9 patients, vedolizumab in 1, ustekinumab in 1 and thalidomide in 2. A further 5 cases were resistant to therapies including anti-TNFs. Conclusion OFG associated with CD may occur before, concurrently or after the diagnosis of CD. Perianal and UGI disease are common associations and there is a significant symptom burden in many. Remission can be obtained with a variety of immunosuppressive treatments, including several CD approved biologicals.

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Section: Discussionmentioning

confidence: 99%

Orofacial Granulomatosis Associated with Crohn’s Disease: a Multicentre Case Series

Phillips

Verstockt

Sładek

et al. 2021

Journal of Crohn's and Colitis

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“…More recently, ustekinumab, a monoclonal antibody targeting the p40 subunit of IL-12/IL-23, has shown efficacy in managing OCD as seen in the current case. 19,20 The variable response of CD to classical or biologic treatment options reflects a complex immunopathological nature of the disease and entails further research into this area.…”

Section: Discussionmentioning

confidence: 99%

Crohn's disease initially presenting with oral manifestations and managed with ustekinumab: A case report

Alrashdan

Safadi

2021

Special Care in Dentistry

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Oral Crohn's disease (OCD) refers to the spectrum of oral manifestations seen in Crohn's disease (CD) patients and could precede other signs of the disease elsewhere, which requires dental practitioners to be vigilant for oral mucosal changes that may accompany such systemic disorders. Classical treatment modalities are based on the use of corticosteroids, immune-modulators and more recently, biologics. Ustekinumab is a novel agent from this last group that has shown efficacy in a limited number of case reports. A case of a debilitating CD presenting initially with oral manifestations and ultimately managed with ustekinumab is reported.

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“…In patients who have had a prior infusion reaction to infliximab, certolizumab combined with methotrexate has recently been shown to resolve the cutaneous lesions. Recent studies indicated treatment with an anti-TNF agent (mainly ustekinumab) as the most recommended therapeutic option for granulomatous cheilitis (with or without CD) after failure of conventional treatments 11…”

Section: Treatmentmentioning

confidence: 99%

Granulomatous cheilitis: differential diagnosis in patients with macrocheilia

et al. 2023

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Macrocheilia, or lip enlargement, has a varied aetiology, but granulomatous conditions, both infective and non-infective, comprise a significant proportion of patients. Diagnosis starts with clinical investigations while histological examination is required for a definitive diagnosis. In the case presented, a young man presented with painless swelling of upper lip over the past 3 months. Given the clinical history and biopsy results, the diagnosis of granulomatous cheilitis was made which is considered a rare manifestation of metastatic Crohn’s disease. Treatment options remain debated, though in the situation presented a conservative approach was adopted, consisting of antibiotics and corticosteroid therapy, which resulted in significant remission in lip swelling without recurrence after a 3-month follow-up.

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Recurrent granulomatous cheilitis associated with Crohn’s disease successfully treated with ustekinumab: case report and literature review

Cited by 10 publications

References 34 publications

Orofacial Granulomatosis Associated with Crohn’s Disease: a Multicentre Case Series

Orofacial Granulomatosis Associated with Crohn’s Disease: a Multicentre Case Series

Crohn's disease initially presenting with oral manifestations and managed with ustekinumab: A case report

Granulomatous cheilitis: differential diagnosis in patients with macrocheilia

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