Xanthomatous Multicentric Reticulohistiocytosis

Camargo, Kathlyn; Pinkston, Olga; Abril, Andy; Sluzevich, Jason C.

doi:10.1097/rhu.0000000000000652

Cited by 4 publications

(5 citation statements)

References 9 publications

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“…Camargo et al reported a 33-year-old Hispanic woman with typical clinical and radiological features of MRH, but the pathology showed histiocytes with foamy rather than conventional ground-glass appearance (Table 1). 8 Andrew et al described a 32-year-old woman with destructive arthritis and progressive nodular eruption, which were most consistent with MRH, but again the histopathology showed xanthomized histiocytes (Table 1). 9 Matiz et al reported a case of a 3-year-old female who presented with xanthomatous lesions on the hands, around the nares, and forehead, associated with significant arthritis that was clinically compatible with MRH; however, the pathology showed foamy histiocytes (Table 1).…”

Section: E T T E R T O T H E E D I T O R Xanthomatous Erosive Arthrit...mentioning

confidence: 86%

“…reported a 33‐year‐old Hispanic woman with typical clinical and radiological features of MRH, but the pathology showed histiocytes with foamy rather than conventional ground‐glass appearance (Table 1). 8 Andrew et al. described a 32‐year‐old woman with destructive arthritis and progressive nodular eruption, which were most consistent with MRH, but again the histopathology showed xanthomized histiocytes (Table 1).…”

Section: Figurementioning

confidence: 86%

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Xanthomatous erosive arthritis: A new disease entity?

Fei

Sun

Zhou

et al. 2022

Pediatric Blood & Cancer

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Section: E T T E R T O T H E E D I T O R Xanthomatous Erosive Arthrit...mentioning

confidence: 86%

Section: Figurementioning

confidence: 86%

Xanthomatous erosive arthritis: A new disease entity?

Fei

Sun

Zhou

et al. 2022

Pediatric Blood & Cancer

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“…The immune disturbance we found in our patient included positive ANA, anti-Ro52, anti-SSA/Ro, anti-SSB/La antibodies, and low-positive RF. Some authors found no inflammatory syndrome or autoantibodies in their patients [ 11 , 12 , 13 ], while others reported positive ANA and anti-CCP antibodies [ 6 , 14 ]. However, the issue of MRH possibly overlapping with autoimmune rheumatic conditions remains a matter of debate [ 11 ].…”

Section: Discussionmentioning

confidence: 99%

Multicentric Reticulohistiocytosis Exhibiting Positive HLA-B07 and HLA-B08: A Case Report

et al. 2020

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Multicentric reticulohistiocytosis (MRH) is a rare cause of destructive inflammatory arthritis involving both small, as well as larger joints. We report the case of a 40-year-old Caucasian female with a family history of neoplasia who was referred to our service witha two-month history of inflammatory joint pain. On examination, the patient had inflammatory arthritis, mainly involving the peripheral joints, sacroiliac joint pain, and numerous papulonodular mucocutaneous lesions, including periungual “coral beads”. Imaging tests revealed erosive arthritis with synovitis and tenosynovitis, sacroiliac joint changes, as well as papulonodular mucosal lesions in the nasal vestibule, the oropharyngeal mucosa, and supraglottic larynx. She tested positive for HLA-B*07 (Human Leukocyte Antigen B*07) and HLA-B*08, ANA (antinuclear antibodies), RF (rheumatoid factor), anti-Ro52, anti-SSA/Ro, and anti-SSB/La antibodies. The skin biopsy was suggestive of MRH, showing a histiocyte infiltrate and frequent giant multinucleated cells. The patient exhibited favorable outcomes under Methotrexate, then Leflunomide. However, she displayed worsening clinical symptoms while under Azathioprine. To our knowledge, this is the first case of MRH to exhibit positive HLA-B*07 together with HLA-B*08. The rarity of MRH, its unknown etiology and polymorphic clinical presentation, as well as its potential neoplastic/paraneoplastic, and autoimmune nature demand extensive investigation.

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“…23 Unusual histological findings have been reported, such as xanthomized, foamy histiocytes (Fig. 4F), [24][25][26] and epidermotropism, 26 suggesting monocytemacrophage activation.…”

Section: Histopathologymentioning

confidence: 95%

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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Xanthomatous Multicentric Reticulohistiocytosis

Cited by 4 publications

References 9 publications

Xanthomatous erosive arthritis: A new disease entity?

Xanthomatous erosive arthritis: A new disease entity?

Multicentric Reticulohistiocytosis Exhibiting Positive HLA-B07 and HLA-B08: A Case Report

Skin Manifestation Associated With Multicentric Reticulohistiocytosis

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Xanthomatous Multicentric Reticulohistiocytosis

Cited by 4 publications

References 9 publications

Xanthomatous erosive arthritis: A new disease entity?

Xanthomatous erosive arthritis: A new disease entity?

Multicentric Reticulohistiocytosis Exhibiting Positive HLA-B*07 and HLA-B*08: A Case Report

Skin Manifestation Associated With Multicentric Reticulohistiocytosis

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Product

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Multicentric Reticulohistiocytosis Exhibiting Positive HLA-B07 and HLA-B08: A Case Report