Novel association of metastatic Crohn's disease and Wolman disease

Alasmari, Amal; Alessa, Reema; Alajroush, Waleed; Alkhodair, Rayan; AlHaddad, Sukainah

doi:10.1016/j.jdcr.2021.12.014

Cited by 2 publications

(3 citation statements)

References 8 publications

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“…Topical tacrolimus can improve the lesions, but there is no case report where topical tacrolimus could induce remission [ 3 , 5 , 9 , 24 , 39 , 57 , 61 ], and can currently not be recommended as a single therapeutic option. It might be useful to support other therapies.…”

Section: Discussionmentioning

confidence: 99%

Fistulising skin metastases in Crohn’s disease: a case report and review of the literature

Elger,

Loibl,

Buechler

et al. 2024

J Med Case Reports

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Background Metastatic Crohn’s disease is a rare disorder characterized by various granulomatous skin lesions that occur independently of gastrointestinal tract involvement. However, currently there is no standardized care or specific treatment. Therapeutic approaches include immunosuppressive agents, such as corticosteroids, azathioprine, and monoclonal antibodies targeting inflammatory cytokines like tumor necrosis factor (TNF). Case presentation We present a case of a 29-year-old western European woman with significant blind ending abdominal subcutaneous fistulas and abscesses, who sought evaluation in the dermatology department. Histological examination revealed multiple epithelioid cell granulomas. There was no evidence of infectious or rheumatologic diseases such as sarcoidosis. The tentative diagnosis was metastatic Crohn’s disease, which was not related to an intestinal manifestation of the disease. The patient responded to infliximab but had to discontinue it due to an allergic reaction. Subsequent adalimumab treatment failed to induce clinical remission; thus, therapy was switched to ustekinumab, resulting in a positive response. Written informed consent for publication of their clinical details and clinical images was obtained from the patient. For our study more than 1600 publications were screened for cases of metastatic Crohn’s disease on PubMed database. 59 case reports with 171 patients were included in the analysis and evaluated for localization, diagnostic and therapeutic approaches, and complications and were summarized in this review. Conclusion The successful ustekinumab treatment of a patient with metastatic Crohn's disease underscores the potential of this minimally investigated therapeutic option, highlighting the need for future treatment guidelines given the increasing prevalence of such cases.

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Section: Discussionmentioning

confidence: 99%

Fistulising skin metastases in Crohn’s disease: a case report and review of the literature

Elger,

Loibl,

Buechler

et al. 2024

J Med Case Reports

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“…Two reported cases of MCD involving the external genitalia had complete recovery on treatment with oral and topical steroids along with azathioprine [ 9 , 10 ]. Further, a recent case of MCD involving the genital area and Wolman disease was reported to have clinical improvement beginning with a 14-day treatment of topical miconazole and hydrocortisone ointment used twice daily, followed by 0.1% topical tacrolimus ointment applied twice daily [ 13 ]. Since there are no established guidelines for the treatment of MCD, the drugs we tried include doxycycline 100 mg PO daily for four weeks with worsening of the lesions.…”

Section: Discussionmentioning

confidence: 99%

“…Due to a scarcity of randomized control trials, approaches to therapy for MCD remain inconsistent [2]. Different treatment modalities have been used with successful responses for MCD in the literature, including a variety of chemotherapeutic agents and surgical procedures combined with oral zinc sulfate [8][9][10][11][12][13]. Two reported cases of MCD involving the external genitalia had complete recovery on treatment with oral and topical steroids along with azathioprine [9,10].…”

Section: Discussionmentioning

confidence: 99%

Metastatic Crohn’s Disease in External Genitalia With Good Outcome on Adalimumab: A Rare Case of a Saudi Female and a Short Review

Alotaibi,

Fathaddin,

AlMutairi

et al. 2023

Cureus

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Crohn’s disease (CD), an inflammatory bowel disease that involves the gastrointestinal tract, is observed in daily hospital practice. On the other hand, metastatic Crohn's disease (MCD) is a rare entity in which cutaneous lesions are found in regions apart from the digestive system. This article describes a rare case of cutaneous CD in a Saudi female, which manifested initially as vulvar and perianal skin lesions. The diagnosis was proven by skin biopsy, and adalimumab offered effective treatment. Although cutaneous MCD is rare, it is an important cutaneous manifestation, as early detection creates the possibility of accessing effective management.

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Novel association of metastatic Crohn's disease and Wolman disease

Cited by 2 publications

References 8 publications

Fistulising skin metastases in Crohn’s disease: a case report and review of the literature

Fistulising skin metastases in Crohn’s disease: a case report and review of the literature

Metastatic Crohn’s Disease in External Genitalia With Good Outcome on Adalimumab: A Rare Case of a Saudi Female and a Short Review

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