SAPHO syndrome in pediatric patients with inflammatory bowel disease treated with infliximab

Marrani, Edoardo; Belli, Gilda; Simonini, Gabriele; Trapani, Sandra; Caproni, Marzia; Lionetti, Paolo

doi:10.1016/j.dld.2018.09.001

“…The association of SAPHO syndrome with IBD was first reported by Kahn et al4 in 1992, and subsequently in several case reports and case series. [16][17][18][19] The prevalence of IBD among patients with SAPHO has been estimated to be from 8-13% in two large studies, with higher prevalence in ileocolic CD compared to ulcerative colitis. Typically, SAPHO occurs in young patients with CD who have colonic involvement; however, prevalence of this association might be underestimated among patients with IBD because of clinical similarities with other EIMs, and drug-related side effects.…”

Section: Discussionmentioning

confidence: 99%

SAPHO Syndrome in Crohn’s Disease Successfully Treated with Ustekinumab: Case Report and Review of the Literature

Caporuscio,

Maggi,

Aratari

et al. 2023

EMJ Gastroenterol

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The authors report a case of synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome in a patient with Crohn’s disease (CD). SAPHO syndrome is a rare disease characterised by the association, even if not simultaneous, of joint and skin manifestations. A young patient with CD was admitted to the authors’ hospital for the onset of bloody diarrhoea, persistent chest pain, headache, and fever while on maintenance therapy with vedolizumab. At visit, sternocostoclavicular and temporomandibular joints were tender and painful. Magnetic resonance was performed, and showed bone oedema of involved joints, while ileocolonoscopy revealed ulcers in the transverse colon. At laboratory, tests marked phlogosis and Campylobacter jejuni infection was observed. A challenge in differential diagnosis arose: atypical drug-induced extraintestinal manifestations, reactive arthritis, or extraintestinal manifestation directly associated with intestinal flare? In relation to the patient’s age, the involved joints, and magnetic resonance findings, SAPHO syndrome was diagnosed. Systemic steroids were used with a rapid clinical improvement; vedolizumab was withdrawn and ustekinumab was started with sustained clinical response.

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“…The association of SAPHO syndrome with IBD was first reported by Kahn et al4 in 1992, and subsequently in several case reports and case series. [16][17][18][19] The prevalence of IBD among patients with SAPHO has been estimated to be from 8-13% in two large studies, with higher prevalence in ileocolic CD compared to ulcerative colitis. Typically, SAPHO occurs in young patients with CD who have colonic involvement; however, prevalence of this association might be underestimated among patients with IBD because of clinical similarities with other EIMs, and drug-related side effects.…”

Section: Discussionmentioning

confidence: 99%

SAPHO Syndrome in Crohn’s Disease Successfully Treated with Ustekinumab: Case Report and Review of the Literature

Caporuscio,

Maggi,

Aratari

et al. 2023

EMJ Gastroenterol

0

View full text Add to dashboard Cite

The authors report a case of synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome in a patient with Crohn’s disease (CD). SAPHO syndrome is a rare disease characterised by the association, even if not simultaneous, of joint and skin manifestations. A young patient with CD was admitted to the authors’ hospital for the onset of bloody diarrhoea, persistent chest pain, headache, and fever while on maintenance therapy with vedolizumab. At visit, sternocostoclavicular and temporomandibular joints were tender and painful. Magnetic resonance was performed, and showed bone oedema of involved joints, while ileocolonoscopy revealed ulcers in the transverse colon. At laboratory, tests marked phlogosis and Campylobacter jejuni infection was observed. A challenge in differential diagnosis arose: atypical drug-induced extraintestinal manifestations, reactive arthritis, or extraintestinal manifestation directly associated with intestinal flare? In relation to the patient’s age, the involved joints, and magnetic resonance findings, SAPHO syndrome was diagnosed. Systemic steroids were used with a rapid clinical improvement; vedolizumab was withdrawn and ustekinumab was started with sustained clinical response.

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“…Yet, according to the Kahn criteria, a diagnosis of SAPHO (synovitis, acne, pustulosis, hyperostosis, osteomyelitis) syndrome can also be made, since this condition includes among its manifestations sacroiliitis (usually unilateral), IBD and psoriasis. [6][7][8] Overall, considering all the manifestations presented by our patient, the diagnostic criteria for several diseases were fulfilled, namely undifferentiated arthritis (considering ILAR criteria), IBD-associated chronic arthritis (not included in ILAR criteria), and SAPHO syndrome. 2,6 In the past several diagnostic criteria have been proposed trying to categorize this wide range of phenotypes.…”

Section: Discussionmentioning

confidence: 99%

The conundrum of juvenile spondyloarthritis classification: Many names for a single disease? Lesson learned from an instructive clinical case

Maniscalco

¹

,

Marrani

²

,

Lamot

³

et al. 2020

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Pediatric rheumatic diseases are often characterized by an evolving phenotype, resulting in diagnostic dilemma for physicians involved in their management. Although several classification criteria are used in childhood to uniform patients’ diagnoses, several conditions share similar clinical features and therefore their classifications may overlap or be ambiguous. This is particularly paradigmatic for the classification of juvenile spondyloarthritis (JSpA), as the currently available criteria do not encompass their complexity. The differential diagnosis of sacroiliitis is often challenging for clinicians and requires considering several conditions, which include infective, neoplastic and rheumatic diseases. We report the case of a 13‐year‐old boy with an evolving clinical phenotype; its progression shows the wide differential diagnosis required in pediatric rheumatic diseases and emphasizes the issues of the actual classification system.

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SAPHO syndrome in pediatric patients with inflammatory bowel disease treated with infliximab

Cited by 8 publications

References 10 publications

SAPHO Syndrome in Crohn’s Disease Successfully Treated with Ustekinumab: Case Report and Review of the Literature

SAPHO Syndrome in Crohn’s Disease Successfully Treated with Ustekinumab: Case Report and Review of the Literature

SAPHO Syndrome in Crohn’s Disease Successfully Treated with Ustekinumab: Case Report and Review of the Literature

The conundrum of juvenile spondyloarthritis classification: Many names for a single disease? Lesson learned from an instructive clinical case

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