SAPHO Syndrome in an Adult Male with Ulcerative Colitis

Sayeed, Sana; Imtiaz, Areeba; Naz, Faiza

doi:10.29271/jcpsp.2019.07.671

“…In recent literature, SS has been reported to mimic cutaneous T-cell lymphoma [101], leprosy [102][103][104], leukemia cutis [105,106], Mycobacterium haemophilum infection [107], RAS-associated autoimmune leukoproliferative disease, rheumatoid neutrophilic dermatoses [108], secondary syphilis [109], and synovitis acne pustulosis hyperostosis syndrome [110]. Conversely, SS has been reported to be mimicked by cutaneous metastasis [111] and rosacea-like dermatitis [63].…”

Section: Differential Diagnosismentioning

confidence: 99%

New Practical Aspects of Sweet Syndrome

Joshi

¹

,

Friske

²

,

Hsiou

³

et al. 2022

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Sweet syndrome (SS), or acute febrile neutrophilic dermatosis, is an inflammatory, non-infectious skin reaction characterized clinically by tender, erythematous papules/plaques/pustules/nodules commonly appearing on the upper limbs, trunk, and head and neck; histologically, SS is characterized by dense neutrophilic infiltrate in the dermis. SS is accompanied by fever; an elevation of inflammatory markers (e.g., erythrocyte sedimentation rate, C reactive protein) in serum may also be observed. Although most cases of SS are idiopathic, SS also occurs in the setting of malignancy or following administration of an associated drug. SS has also been reported in association with pregnancy and a burgeoning list of infectious (most commonly upper respiratory tract infections) and inflammatory diseases; likewise, the litany of possible iatrogenic triggers has also grown. Over the past several years, a wider spectrum of SS presentation has been realized, with several reports highlighting novel clinical and histological variants. Corticosteroids continue to be efficacious first-line therapy for the majority of patients with SS, although novel steroid-sparing agents have been recently added to the therapeutic armamentarium against refractory SS. New mechanisms of SS induction have also been recognized, although the precise etiology of SS still remains elusive. Here, we catalogue the various clinical and histological presentations of SS, summarize recently reported disease associations and iatrogenic triggers, and review treatment options. We also attempt to frame the findings of this review in the context of established and emerging paradigms of SS pathogenesis.

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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

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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.

show abstract

“…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.

show abstract

SAPHO Syndrome in an Adult Male with Ulcerative Colitis

Cited by 3 publications

References 7 publications

New Practical Aspects of Sweet Syndrome

New Practical Aspects of Sweet Syndrome

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

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