Successful Treatment of Multicentric Reticulohistiocytosis with Adalimumab, Prednisolone and Methotrexate

Motegi, Sei‐ichiro; Yonemoto, Yukio; Toki, Sayaka; Uchiyama, Akihiko; Yamada, Kazuya; Ishikawa, Osamu

doi:10.2340/00015555-2170

Cited by 9 publications

(5 citation statements)

References 11 publications

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“…The response to these agents is based on the pathophysiology that the MRH is associated with increased release and expression of pro-inflammatory cytokines such as TNF, interleukin (IL)-6, IL-1b, and IL-8 [14]. Etanercept, adalimumab, and infliximab are the anti-TNF agents that are effective in MRH [15][16][17]. Tocilizumab is an IL-6 receptor antagonist considered as a treatment option [18].…”

Section: Discussionmentioning

confidence: 99%

Multicentric Reticulohistiocytosis Presenting As Acute Pericarditis With Moderate-Sized Pericardial Effusion: A Case Report and Review of Multicentric Reticulohistiocytosis Treatment

Mandyam¹,

Onteddu²,

R³

et al. 2023

Cureus

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Multicentric reticulohistiocytosis (MRH) is a rare, class IIb non-Langerhans cell histiocytosis associated with skin and joint involvement. It is more prevalent (80%) in Caucasian females in their fifth to sixth decade of life. Patients usually demonstrate symptoms and signs of symmetric polyarthritis and papulonodular cutaneous lesions. In addition to skin and joints, multiple organs can be involved, such as the lung (pleural effusion, interstitial fibrosis, hilar lymphadenopathy), heart (pericardial effusion, myocarditis), gastrointestinal system, and urogenital system (genital tract and kidney). Pericardial involvement is a rare manifestation, and around three cases have been reported in the literature so far. Our case report is a valuable contribution to the literature, which aids clinicians in contemplating MRH as one of the differentials among patients presenting with pericardial effusion. We described the characteristics of MRH along with its differentiating features from other autoimmune conditions and management.

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Section: Discussionmentioning

confidence: 99%

Multicentric Reticulohistiocytosis Presenting As Acute Pericarditis With Moderate-Sized Pericardial Effusion: A Case Report and Review of Multicentric Reticulohistiocytosis Treatment

Mandyam¹,

Onteddu²,

R³

et al. 2023

Cureus

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“…TNF inhibitors such as etanercept, adalimumab, and infliximab, in combination with or without prednisolone, have been used with favorable effects on skin and joint manifestations in recent years. [30][31][32][33][34][35] A recent review demonstrated that 5 of 10 cases responded well to etanercept, 6 of 8 cases were successfully treated with infliximab, and 1 of 3 cases was successfully treated with adalimumab. 30 However, there have been a few cases that were unresponsive to TNF inhibitors 36 or cases showing disparate effects of TNF inhibitors between cutaneous and joint lesions, that is, favorable to the skin but not to the polyarthritis.…”

Section: Therapymentioning

confidence: 99%

“…High levels of TNF-α have been observed in the synovium and synovial fluid of affected joints in MRH patients, and TNF-α expression was abundantly detected in the skin lesion, as mentioned previously. TNF inhibitors such as etanercept, adalimumab, and infliximab, in combination with or without prednisolone, have been used with favorable effects on skin and joint manifestations in recent years 30–35 . A recent review demonstrated that 5 of 10 cases responded well to etanercept, 6 of 8 cases were successfully treated with infliximab, and 1 of 3 cases was successfully treated with adalimumab 30 .…”

Section: Therapymentioning

confidence: 99%

Skin Manifestation Associated With Multicentric Reticulohistiocytosis

Yamamoto¹

2020

J Clin Rheumatol

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Multicentric reticulohistiocytosis (MRH) is a rare multisystem disorder, primarily affecting the skin and joints. As severe joint damage is a possible symptom, early diagnosis and therapeutic intervention are required. Cutaneous lesions present with characteristic features such as discrete reddish nodules, especially on acral locations. Additionally, the face, scalp, trunk and extremities are also affected. Xanthomatous plaques are also seen on the face. The cutaneous manifestations of MRH are various, which therefore should be differentiated from several diseases. In particular, MRH has been reported to assume clinical appearances resembling Gottron papules, periungual erythema, V-neck erythema, shawl sign, and poikiloderma associated with dermatomyositis. Histopathologic features show proliferation of multinucleated giant cells with abundant eosinophilic granular ground glass-like cytoplasm in the dermis. Multicentric reticulohistiocytosis is occasionally paraneoplastic and is associated with internal malignancies. The appropriate therapies are still challenging, and oral prednisolone, immunosuppressants, and recent biologics are used. In this article, cutaneous lesions, histopathology, and pathogenesis of MRH are mainly discussed from a dermatological perspective. It is important, not only for dermatologists but also for rheumatologists and orthopedists, to be able to distinguish between the various types of skin lesions brought on by MRH. Cutaneous signs are important diagnostic clues and assessment tools for therapeutic efficacy.

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“…These cytokines are potential targets of therapy, though there is currently no standard first-line treatment regimen. The most frequently used treatments include systemic glucocorticoids, disease-modifying antirheumatic drugs such as methotrexate and cyclophosphamide, and anti-TNF biologics [1, 9, 10].…”

Section: Introductionmentioning

confidence: 99%

A Novel Case of Multicentric Reticulohistiocytosis Associated with Renal Cell Carcinoma Successfully Treated with Infliximab and Methotrexate

2023

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Multicentric reticulohistiocytosis (MRH) is categorized as a rare non-Langerhans cell histiocytosis most commonly seen in women in the fourth to fifth decade of life. This systemic inflammatory condition affects multiple organ systems and can result in severe joint destruction which can progress to arthritis mutilans. To date, various underlying malignancies have been discovered in patients with MRH including breast, gastric, thymic, hepatic, and melanoma. There has been 1 case of underlying renal cell carcinoma reported in a patient diagnosed with MRH. Additionally, there is no consistently recognized treatment for MRH described in the literature. The rarity of the disease contributes to the difficulty in defining a standardized treatment. We present the case of a patient with extensive joint and skin involvement who was successfully treated with infliximab and methotrexate, experienced clinical improvement, and was later diagnosed with clear cell renal cell carcinoma. The synergistic effects of infliximab and methotrexate, in combination with the low side-effect profile, appear to be promising in the setting of MRH and in our patient resulted in the resolution of symptoms and cutaneous manifestations. We suggest this regimen as an effective combination therapy. We emphasize thorough and continuous screening for underlying malignancy associated with MRH, despite clinical improvement or negative malignancy work-up upon initial diagnosis.

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Successful Treatment of Multicentric Reticulohistiocytosis with Adalimumab, Prednisolone and Methotrexate

Cited by 9 publications

References 11 publications

Multicentric Reticulohistiocytosis Presenting As Acute Pericarditis With Moderate-Sized Pericardial Effusion: A Case Report and Review of Multicentric Reticulohistiocytosis Treatment

Multicentric Reticulohistiocytosis Presenting As Acute Pericarditis With Moderate-Sized Pericardial Effusion: A Case Report and Review of Multicentric Reticulohistiocytosis Treatment

Skin Manifestation Associated With Multicentric Reticulohistiocytosis

A Novel Case of Multicentric Reticulohistiocytosis Associated with Renal Cell Carcinoma Successfully Treated with Infliximab and Methotrexate

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