Multicentric reticulohistiocytosis with dermatomyositis‐like features: A case report with dermoscopy and reflectance confocal microscopy findings

Farabi, Banu; Jamgochian, Marielle; Rao, Babar

doi:10.1111/cup.14170

J Cutan Pathol

2021

DOI: 10.1111/cup.14170

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Multicentric reticulohistiocytosis with dermatomyositis‐like features: A case report with dermoscopy and reflectance confocal microscopy findings

Banu Farabi

Marielle Jamgochian

Babar Rao

Abstract: Multicentric reticulohistiocytosis (MRH) is an insidious‐onset, non‐Langerhans‐cell histiocytosis (NLCH) affecting the joints and skin. Early diagnosis is important to prevent destructive arthritis and disease‐related complications. Reflectance confocal microscopy (RCM) is a technique that allows the visualization of the epidermis and superficial dermis noninvasively on a horizontal plane with quasi‐histologic images of the skin. RCM features of NLCH including Rosai‐Dorfman disease, adult xanthogranuloma (AXG)… Show more

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2024

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Cited by 3 publications

References 24 publications

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Multicentric reticulohistiocytosis with oral and laryngeal involvement in association with autoimmune/inflammatory syndrome induced by adjuvants: Expanding the spectrum of two uncommon entities

Girolami,

Kluge,

Gallmetzer

et al. 2024

Modern Rheumatology Case Reports

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Multicentric reticulohistiocytosis (MRH) is the most frequent entity in the group of reticulohistiocytoses. It is usually accompanied by a symmetrical erosive polyarthritis and is frequently associated with cancer and autoimmune disorders. Autoimmune syndrome induced by adjuvants (ASIA) is an inflammatory syndrome triggered by adjuvants such as those contained in vaccines or by silicone implants. Here we report a 71-years old female with a history of breast cancer treated with surgery and subsequent prosthesis who developed a systemic hyperinflammatory syndrome including seronegative symmetric polyarthritis, multiple skin lesions and two large nodular lesions in the oral cavity and larynx. Clinical picture was consistent with a clinical diagnosis of ASIA, with breast implant rupture and/or vaccination against SARS-CoV-2 as possible triggers. Histopathology of skin, oral and laryngeal nodules revealed cutaneous/mucous and submucosal infiltration of large epithelioid mononuclear or binucleated cells with fine granular ground glass-like cytoplasm and round to kidney-shaped nuclei with prominent nucleoli, without atypical features or relevant pleomorphism, accompanied by sparse giant cells and lymphocytes. These cells stained positive for CD68 and CD45 and negative for S100, CD1a, and markers of epithelial or neural/melanocytic differentiation, altogether consistent with a diagnosis of reticulohistiocytosis. Clinic-pathological correlation allowed the final diagnosis of MRH. To our knowledge, this is the first report of a co-occurrence of MRH with ASIA and this is relevant to broaden the spectrum of those both rare diseases.

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Multicentric reticulohistiocytosis with oral and laryngeal involvement in association with autoimmune/inflammatory syndrome induced by adjuvants: Expanding the spectrum of two uncommon entities

Girolami,

Kluge,

Gallmetzer

et al. 2024

Modern Rheumatology Case Reports

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Analysis of the characteristics of reflectance confocal microscopy images of xanthogranuloma and xanthoma

Sun,

Wang

2024

Int J Dermatology

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BackgroundBoth xanthogranuloma and xanthoma clinically manifest as yellowish skin lesions. Historically, the gold standard for diagnosis was skin histopathological examination. Currently, the advent of reflectance confocal microscopy (RCM) offers additional diagnostic support for these diseases by revealing their microscopic features, thereby enhancing the theoretical foundation for diagnosis.ObjectiveThis study aimed to elucidate the distinctive characteristics of RCM images in xanthogranuloma and xanthoma, assess their diagnostic value, and investigate the relationship between RCM and histopathological features, ultimately boosting diagnostic accuracy.MethodsRCM and histopathological examinations were conducted on 13 patients with xanthogranuloma and 12 with xanthoma, recruited from our Dermatology Clinic between August 2022 and November 2023. The study involved analyzing RCM image features and correlating them with histopathological findings.ResultsThe RCM images of 13 xanthogranuloma and 12 xanthoma cases showed similar features. Xanthogranuloma predominantly exhibited epidermal atrophy and thinning in 6 cases (46.15%). Additionally, in 69.23% of cases, scattered small mononuclear inflammatory cells were infiltrated in the superficial and middle dermis layers. Medium to high refractive cells, predominantly vacuolated and resembling foam, were observed in 61.54% of cases. All cases demonstrated high refractive cells with distinct target‐shaped, disc‐shaped, horseshoe‐like, and flower‐ring structures. Concordance rates with histopathological examinations were 69.23, 92.31, 92.31, and 100%, respectively. Regarding xanthoma, epidermal atrophy and thinning occurred in two cases (16.67%), and mononuclear inflammatory cell infiltration was observed in 25% of cases. High refractive cells with the previously mentioned shapes were present in 100% of cases, though only 16.67% displayed these characteristics exclusively. The concordance rates were 66.67, 91.67, 100, and 91.67%, respectively.ConclusionRCM imaging of xanthogranuloma and xanthoma presents distinctive, highly consistent features with their histopathology, offering valuable insights for clinicians in diagnosing and differentiating these conditions.

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Solitary nodule of cutaneous reticulohistiocytosis: A case report

Fournier

Shao

Popovic

et al. 2022

J Case Rep Images Pathol

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Introduction: Solitary cutaneous reticulohistiocytosis represents a rare form of benign monocyte/macrophage proliferation. On routine histology, these lesions are typically described as large cells with cytoplasm showing ground glass appearance and giant cells. They grow up to 1 cm in size with rare cases exceeding this size. Case Report: This case report of a 28-year-old male demonstrated a nodule of reticulohistiocytosis measuring 2.2 cm in size. Microscopic features showed a well-demarcated nodule in the dermis with large histiocytes with ground-glass eosinophilic cytoplasm, giant cells, and foamy macrophages in a background of mixed inflammatory cells. Immunohistochemical staining showed positive staining for vimentin, CD68, CD31, with focal and patchy positivity for S100, CD43, and CD45 and negative staining for CD1a, langerin, CD21, CD23, CD30, CD34, ERG, D2-40, AE1/AE3, epithelial membrane antigen (EMA), smooth muscle actin (SMA), myogenin, desmin, SOX10, HMB-45, tyrosinase, and MelanA. Conclusion: The microscopic and immunohistochemical findings are characteristic of this entity but it is important to recognize for proper management and differentiation from other malignant lesions.

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Multicentric reticulohistiocytosis with dermatomyositis‐like features: A case report with dermoscopy and reflectance confocal microscopy findings

Cited by 3 publications

References 24 publications

Multicentric reticulohistiocytosis with oral and laryngeal involvement in association with autoimmune/inflammatory syndrome induced by adjuvants: Expanding the spectrum of two uncommon entities

Multicentric reticulohistiocytosis with oral and laryngeal involvement in association with autoimmune/inflammatory syndrome induced by adjuvants: Expanding the spectrum of two uncommon entities

Analysis of the characteristics of reflectance confocal microscopy images of xanthogranuloma and xanthoma

Solitary nodule of cutaneous reticulohistiocytosis: A case report

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