Histiocytoid Sweet syndrome: a novel association with relapsing polychondritis

Arima, Yumi; Namiki, Takeshi; Ueno, Makiko; Kato, Kohei; Tokoro, Shown; Takayama, Kaoru; Miura, Keiko; Yokozeki, Hiroo

doi:10.1111/bjd.14229

Cited by 10 publications

(9 citation statements)

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“…Herein, the set of immunolabelled cells showed a mixed-inflammatory infiltrate characterised by the predominance of macrophages, T lymphocytes, some B lymphocytes and neutrophils. Our findings are similar to those reported in mice (Kitagaki and Hirota, 2007) and humans (Arima et al, 2016) with AC or RP. Most cells also presented immunolabelling by lysozyme, which can be attributed to their histiocytic and polymorphonuclear origins.…”

Section: Tablesupporting

confidence: 92%

“…Most cells also presented immunolabelling by lysozyme, which can be attributed to their histiocytic and polymorphonuclear origins. Strong lysozyme positivity was previously detected in the auricular perichondrium of humans with RP (Arima et al, 2016;Ahn et al, 2017).…”

Section: Tablementioning

confidence: 71%

“…In cats and cattle with AC or RP, the inflammatory infiltrate in the perichondrium is predominantly lymphocytic (Gerber et al, 2002;Baba et al, 2009;Adissu et al, 2014), unlike the histiocytic and neutrophilic characteristics in the case reported here. CD4-positive T lymphocyte subsets associated with cytokine Acta Veterinaria Hungarica 67, 2019 production such as IFN-γ, IL-2, and TNF-α are considered responsible for triggering the inflammatory process and cartilage injury (Ouchi et al, 2011;Arima et al, 2016). Therefore, the individual interpretation of the set of inflammatory cells in the few cases of AC in cats reported in the literature does not allow the establishment of a single pattern for the inflammatory response in the species.…”

Section: Tablementioning

confidence: 99%

“…The pathogenesis of AC is unknown; however, it is speculated to be similar to that of human RP, with antibody production and reaction against type II collagen, matrilin-1 (cartilage-specific protein), as well as proteoglycan-rich tissues (Arnaud et al, 2014;Cantarini et al, 2014). Although the mechanisms of the disease are not completely known, the immune profile of inflammatory cells in the damaged cartilaginous tissues in humans with RP (Ouchi et al, 2011;Arima et al, 2016), and cattle (Adissu et al, 2014) and mice with AC (Kitagaki and Hirota, 2007) predominantly involves CD3+ and CD4+ T lymphocytes, CD68+ or Mac-1+ macrophages and a few neutrophils. Herein, we describe the immunopathological findings of an AC case in a cat.…”

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confidence: 99%

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Immunopathological findings in a cat with auricular chondritis

Wilson¹,

Machado²,

Sousa³

et al. 2019

Acta Veterinaria Hungarica

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At clinical examination, a 5-year-old male domestic short-haired cat exhibited painful swelling and erythema of the pinnae of both ears. Microscopically, the lesions on both pinnae were composed of diffuse granulomatous chondritis with degeneration and necrosis of the pinnal cartilage. Numerous mast cells were also observed within and surrounding the inflammatory lesion. Immunohistochemistry showed a mixed inflammatory infiltrate characterised by the predominance of macrophages (CD68+, MAC 387+ and Lysozyme+), T lymphocytes (CD3+), some B lymphocytes (CD79α+) and neutrophils. Immunopathological characterisation of the lesion showed a granulomatous inflammation profile and suggests that the morphological changes and immunopathogenesis of auricular chondritis in cats presents a similarity with relapsing polychondritis in humans.

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Section: Tablesupporting

confidence: 92%

Section: Tablementioning

confidence: 71%

Section: Tablementioning

confidence: 99%

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confidence: 99%

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Immunopathological findings in a cat with auricular chondritis

Wilson¹,

Machado²,

Sousa³

et al. 2019

Acta Veterinaria Hungarica

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“… 9 , 10 , 17 , 18 The remaining cases were described in case reports. 11 , 13 , 14 , 19 – 38 In Vignon-Pennamen et al series, the 9 H-SS cases studied were all associated to MDS. Overall, 40% (40/100) of these previously published cases of H-SS were associated to a hematological malignancy, and 24% (24/100) with a MDS, while these associations were of 55.5% (12/22) and 31.8% (7/22), respectively, in our series.…”

Section: Discussionmentioning

confidence: 89%

Histiocytoid Sweet Syndrome Is More Frequently Associated With Myelodysplastic Syndromes Than the Classical Neutrophilic Variant

et al. 2016

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Histiocytoid Sweet syndrome (H-SS) is a histological variant of Sweet syndrome (SS) differing from classical neutrophilic SS (N-SS) by a dermal infiltrate mainly composed of lymphocytes and histiocytoid myeloperoxidase-positive cells. We aimed to report a large series of H-SS and compare the frequency and type of hematological malignancies associated to H-SS and N-SS. We included 62 patients with a coding histopathologic diagnosis of SS prospectively registered between 2005 and 2014 in the database of our Department of Pathology. Overall, 22 (35.5%) and 40 (64.5%) patients had a histological diagnosis of H-SS and N-SS, respectively. Median age, sex ratio, and cutaneous lesions were similar in the 2 groups. The frequency of extra-cutaneous manifestations was similar (50% vs 37.5%, P = 0.42). Recurrent forms were significantly more frequent in H-SS than in N-SS patients (21% vs 2.5%, P = 0.01). A hematological malignancy was diagnosed in 22 patients, 12 (55.5%) with H-SS and 10 (25%) with N-SS (P = .019). Hematological malignancy was of myeloid origin in 8/22 (36.3%) H-SS and 5/40 (12.5%) N-SS patients (P = .02), and of lymphoid origin without myeloid component in 4/22 (18.1%) H-SS and 4/40 (10%) N-SS patients (P = .35), respectively. One N-SS patient had a hematological malignancy of mixed (myeloid and lymphoid) phenotype. A myelodysplastic syndrome (MDS) was diagnosed in 7/22 (31.8%) H-SS and 1/40 (2.5%) N-SS patients (P < .001). Hematological disease was diagnosed before (in 8 H-SS and 3 N-SS patients) or at the time of the occurrence of the cutaneous lesions (in 1 H-SS and 7 N-SS patients). However, in 3 H-SS patients, all with MDS, cutaneous lesions preceded the hematological disease by ≤6 months. In conclusion, H-SS was associated with MDS in one third of patients but also with lymphoid malignancies, and cutaneous lesions could precede the hematological diagnosis in patients with MDS. A complete hematological assessment is mandatory at diagnosis, and monitoring blood cell counts should be recommended for at least 6 months after the diagnosis of H-SS.

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Clinicopathologic, Immunohistochemical, and Molecular Features of Histiocytoid Sweet Syndrome

et al. 2017

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syndrome is a rare histopathologic variant of Sweet syndrome. The nature of the histiocytoid infiltrate has generated considerable controversy in the literature. OBJECTIVE The main goal of this study was to conduct a comprehensive overview of the immunohistochemical phenotype of the infiltrate in histiocytoid Sweet syndrome. We also analyze whether this variant of Sweet syndrome is more frequently associated with hematologic malignancies than classic Sweet syndrome. DESIGN This is a retrospective case series study of the clinicopathologic, immunohistochemical, and molecular features of 33 patients with a clinicopathologic diagnosis of histiocytoid Sweet syndrome was conducted in the dermatology departments of 5 university hospitals and a private laboratory of dermatopathology. MAIN OUTCOME AND MEASURES The clinical, histopathological, immunohistochemical, and follow-up features of 33 patients with histiocytoid Sweet syndrome were analyzed. In some cases, cytogenetic studies of the dermal infiltrate were also performed. We compare our findings with those of the literature. RESULTS The dermal infiltrate from the 33 study patients (20 female; median age, 49 years; age range, 5-93 years; and 13 male; median age, 42 years; age range, 4-76 years) was mainly composed of myeloperoxidase-positive immature myelomonocytic cells with histiocytoid morphology. No cytogenetic anomalies were found in the infiltrate except in 1 case in which neoplastic cells of chronic myelogenous leukemia were intermingled with the cells of histiocytoid Sweet syndrome. Authentic histiocytes were also found in most cases, with a mature immunoprofile, but they appeared to be a minor component of the infiltrate. Histiocytoid Sweet syndrome was not more frequently related with hematologic malignancies than classic neutrophilic Sweet syndrome. CONCLUSIONS AND RELEVANCE The dermal infiltrate of cutaneous lesions of histiocytoid Sweet syndrome is composed mostly of immature cells of myeloid lineage. This infiltrate should not be interpreted as leukemia cutis.

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Histiocytoid Sweet syndrome: a novel association with relapsing polychondritis

Cited by 10 publications

References 10 publications

Immunopathological findings in a cat with auricular chondritis

Immunopathological findings in a cat with auricular chondritis

Histiocytoid Sweet Syndrome Is More Frequently Associated With Myelodysplastic Syndromes Than the Classical Neutrophilic Variant

Clinicopathologic, Immunohistochemical, and Molecular Features of Histiocytoid Sweet Syndrome

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