Multicentric Reticulohistiocytosis Successfully Treated With Infliximab

Kalajian, Andrew H.; Callen, Jeffrey P.

doi:10.1001/archderm.144.10.1360

Cited by 48 publications

(17 citation statements)

References 23 publications

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“…Treatment of the primary malignancy, when associated with MRH, results in complete remission only in few cases. Successful treatment of arthritis has been reported with methotrexate, cyclophosphamide, chlorambucil, alone or in combination with systemic steroids, bisphosphonates (alendronate, zoledronate), TNF- inhibitors (infl iximab, etanercept) (4,8,12). Surgical excision is a therapeutic procedure for individual lesions of solitary or multiple RHs.…”

Section: Discussionmentioning

confidence: 99%

Multiple Reticulohistiocytomas in an 88-year-old Man: a Case Report

Gajic¹,

Roš²,

Arapović³

et al. 2013

Serbian Journal of Dermatology and Venerology

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Solitary and multiple reticulohistiocytomas, often referred to as diffuse cutaneous reticulohistiocytosis, that are generally restricted to skin, must be differentiated from multicentric reticulohistiocytosis. Multicentric reticulohistiocytosis is a rare histiocytic proliferative disease affecting joints, skin and mucous membranes, while arthropathy usually precedes skin and mucosal involvement. Other organs may also be involved, and 20% of patients have an associated internal malignancy. Multicentric reticulohistiocytosis has been reported to be associated with autoimmune diseases and malignancies. We present an 88-year-old man, with a sudden eruption of asymptomatic, fi rm, skin to brown colored papules and nodules, 6 to 17 mm in diameter, localized on forehead, arms, legs, and buttocks. There were no symptoms of joint or other organ involvement. Clinical and dermoscopic characteristics pointed to reticulohystiocytomas. Multiple excision biopsies of different tumors were performed and histopathology reports confi rmed the diagnosis. In the case presented, reticulohistiocytosis was limited to the skin, exhibiting multiple reticlohistiocytomas with typical clinical, dermoscopic and rather peculiar histopathology presentation. Apart from this, some histologic features were seen in early lesions of multicentric reticulohistiocytosis, such as multinuclear cells dissociating collagen fi bers with pale eosinophilic and foamy cytoplasm. Besides surgical excisions, no other treatment options were done. Laboratory and other tests showed no presence of extracutaneous illness, and no autoimmune or paraneoplastic processes. At one year follow up, the remaining tumors were of the same size, but there were no recurrences at excision sites, no signs of disease progression or systemic involvement. Since diffuse cutaneous reticulohistiocytosis without arthropathy as well as isolated reticulohistiocytomas have been described, in some cases of multiple reticulohistiocytomas even without systemic symptoms and signs, multicentric reticulohistiocytosis should be considered with an appropriate follow up. In such cases, skin lesions have the same histological features as lesions in multicentric reticulohistiocytosis, but they are not associated with joint problems or neoplasms.

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Section: Discussionmentioning

confidence: 99%

Multiple Reticulohistiocytomas in an 88-year-old Man: a Case Report

Gajic¹,

Roš²,

Arapović³

et al. 2013

Serbian Journal of Dermatology and Venerology

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“…All patients had biopsies consistent with MRH. Using these methods, 27 cases of MRH with dermatomyositis-like features were identified (table 1) [4,5,6,7,8,9,10,11,12,13,14,15,16,17,18,19,20,21,22,23,24,25,26,27]. …”

Section: Discussionmentioning

confidence: 99%

“…Recent literature on the treatment of MRH has focused on the promising results seen in patients treated with tumor necrosis factor α inhibitors [12,35,36,37,38]. In comparison, tumor necrosis factor α inhibitors are increasingly recognized as exacerbating or causing dermatomyositis, further supporting the need for diagnostic accuracy [39,40,41,42].…”

Section: Discussionmentioning

confidence: 99%

Multicentric Reticulohistiocytosis with Dermatomyositis-Like Features: A More Common Disease Presentation than Previously Thought

Fett

Liu

2011

Dermatology

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Multicentric reticulohistiocytosis (MRH) is a rare form of non-Langerhans histiocytosis that presents with erosive arthritis and skin nodules. Approximately 25% of patients with MRH have an associated malignancy. Dermatomyositis is an inflammatory autoimmune condition that has also been associated with malignancy. To date, 7 cases of MRH have been reported to present with cutaneous features of dermatomyositis. We describe an eighth patient with MRH who presented with dermatomyositis-like features (V-neck erythema, shawl sign, Gottron’s papules and periungual erythema), who developed metastatic breast cancer 1 year after diagnosis. We hypothesized that clinical overlap between MRH and dermatomyositis was not as uncommon as review of the literature suggested. Careful review of the physical exam findings and photographs of the 234 papers reporting MRH revealed 27 cases of MRH with dermatomyositis-like features. Of these 27 cases, 7 (26%) were associated with a malignancy. Skin biopsies of the cutaneous features mimicking dermatomyositis revealed pathologic features of MRH. This is a descriptive analysis of published case reports. Based on a review of published case reports, MRH presenting with dermatomyositis-like features is likely fairly common. Histological examination of skin biopsies allows for disease differentiation. Differentiating MRH from dermatomyositis is important for management decisions and comorbidity screening.

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“…Whole body CT scanning was performed in 2007 and 2008 with no malignancy identified DISCUSSION MRH is not thought to be a true paraneoplastic syndrome; however, careful malignancy screening is indicated at diagnosis and throughout the disease course. 2 Many treatments have been trialled, including systemic corticosteroids and methotrexate, 3 bisphosphonate drugs, 4 and, more recently, tumour necrosis factor inhibitors. 2 One case report suggests a role for antituberculous therapy in the treatment of MRH.…”

Section: Case Reportmentioning

confidence: 99%

“…2 Many treatments have been trialled, including systemic corticosteroids and methotrexate, 3 bisphosphonate drugs, 4 and, more recently, tumour necrosis factor inhibitors. 2 One case report suggests a role for antituberculous therapy in the treatment of MRH. 5 Azathioprine is a pro-drug of 6-mercaptopurine (6-MP) that, through a series of enzymatic conversions, can be incorporated into DNA synthesis and thereby halt the de novo purine synthesis pathway.…”

Section: Case Reportmentioning

confidence: 99%

A case of multicentric reticulohistiocytosis responsive to azathioprine in a patient with no underlying malignancy

Rudd¹,

Dolianitis²,

Varigos³

et al. 2011

Australasian Journal of Dermatology

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Multicentric reticulohistiocytosis (MRH), a rare histiocytic systemic condition characterized by mutilating arthritis and multiple cutaneous nodules, has been associated with malignancy including that of the breast, thyroid and colon. An unsubstantiated link with infectious agents such as mycobacterium tuberculosis has been described. Many treatments have been used with varying success. We describe the case of a 60-year-old man with MRH and no underlying malignancy who initially responded well to azathioprine and whose disease recurred upon cessation. A second course of azathioprine administered in conjunction with antituberculous treatment resulted in disease control. Broader associations and features of this rare disease are discussed.

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Multicentric Reticulohistiocytosis Successfully Treated With Infliximab

Cited by 48 publications

References 23 publications

Multiple Reticulohistiocytomas in an 88-year-old Man: a Case Report

Multiple Reticulohistiocytomas in an 88-year-old Man: a Case Report

Multicentric Reticulohistiocytosis with Dermatomyositis-Like Features: A More Common Disease Presentation than Previously Thought

A case of multicentric reticulohistiocytosis responsive to azathioprine in a patient with no underlying malignancy

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