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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">First patient&#46; <span class="elsevierStyleItalic">Clinical presentation&#58;</span> A&#44; Multiple brownish-red papules on the left cheek&#46; B and C&#44; Lesions on the right cheek that took on a more erythematous and edematous appearance on rubbing&#46; D-F&#44; Clinical deterioration of the lesions on the facial area and appearance of lesions on the upper back and arms&#46;</p> <p id="spar0010" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleItalic">Histology&#58;</span> G&#44; Discrete mononuclear infiltrate in the superficial dermis with disperse eosinophils&#46; H&#44; The vast majority of infiltrate cells are positive for CD163&#46; I&#44; Numerous intraepidermal and dermal cells positive for CD1a&#46; J&#44; Many of these are negative for langerin&#46; K&#44; A discrete increase in perivascular mast cells can be observed&#46; &#40;G&#44; HE&#8239;&#215;&#8239;200&#59; H&#44; CD163&#8239;&#215;&#8239;100&#59; I&#44; CD1a &#215;100&#59; J&#44; langerin &#215;400&#59; K&#44; c-kit &#215;200&#41;&#46;</p>"
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which had appeared 3 months earlier &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Figs&#46; 1</a>A and 1B&#41;&#46; The mother reported no other symptoms&#44; except for a change in color of the lesions on rubbing &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B and 1C&#41;&#46; A follow-up visit found an increase in number and intensity of the lesions&#44; and the appearance of new elements on the upper part of the back and the arms &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>D-1F&#41;&#46; A skin biopsy revealed a dense inflammatory infiltrate in the superficial dermis&#44; consisting predominantly of macrophages&#44; with few eosinophils&#46; Immune staining was positive for CD163 and CD68 in most cells&#46; Fifteen percent of cells were positive for CD1a and negative for S100 and langerin in the dermal infiltrate&#44; and intraepidermal levels were also very high&#46; Furthermore&#44; the c-KIT marker revealed a discrete increase in the predominantly perivascular mast-cell count &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>G-1&#8239;K&#41;&#46; Additional tests &#40;abdominal ultrasound&#44; blood test and urine sediment&#41; revealed a reduction in hemoglobin &#40;10&#46;1&#8239;g&#47;dL&#41; and a discrete increase in the size of the spleen&#59; for this reason&#44; the patient was sent to the pediatric hematologist for evaluation&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">Case 2</span>&#58; A 1-year-old child with no past history of interest visited with yellowish-red maculopapular lesions that had appeared on the face and upper part of the torso 2 months earlier &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Figs&#46; 2</a>A and 2B&#41;&#46; The lesions took on a redder color on crying&#46; Histopathology revealed a mononuclear inflammatory infiltrate in the superficial dermis&#46; Immune staining of the infiltrate was positive for CD163 and CD68 and negative for CD1a&#44; S100&#44; and langerin &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>C-2F&#41;&#46; As in the previous case&#44; a discrete increase in the number of mast cells was observed&#46; Blood tests revealed an elevated white blood cell count &#40;20450&#47;L&#41; with no other infection data&#59; the patient was therefore referred to the pediatric hematologist for evaluation&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">A diagnosis of iron deficiency anemia was made in the pediatric hematology consultation in the first case and blood-test and ultrasound results were normal a few months later&#46; The white blood cell count was normal in follow-up visits in the second patient&#46; Given the epidemiologic clinical context and after ruling out systemic involvement&#44; both children were diagnosed with BCH&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Non-Langerhans cell histiocytoses located in the skin and mucosa include a variety of entities that are classified depending on clinical signs and symptoms&#44; immunophenotype&#44; and the presence or absence of systemic involvement&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> According to a recently published classification&#44; BCH belongs to the group of juvenile xanthogranulomas &#40;in group C of non-Langerhans cell histiocytoses&#41;&#44; together with other entities that do not have systemic involvement&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> The term <span class="elsevierStyleItalic">cephalic</span> is subject to debate&#44; as involvement of the torso and upper extremities has been observed in a high percentage of cases&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Histologically&#44; it is characterized by a histiocyte infiltrate in the superficial and middle dermis&#44; with positive staining for CD68 and CD163&#44; and negative staining for CD1a&#44; S100&#44; and langerin&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;3</span></a> Cases of BCH have been published&#44; however&#44; with immunophenotypes positive for S100<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> and CD1a&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Darier&#8217;s sign is generally negative in histiocytoses&#44; although it has presented in some cases&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> In our 2 children&#44; the lesions took on a more erythematous tone in certain situations&#46; This &#8220;pseudo-Darier&#8221; phenomenon may be due&#44; in histologic terms&#44; to the discrete increase in mast cells observed in both biopsies &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Figs&#46; 1</a>K and <a class="elsevierStyleCrossRef" href="#fig0010">2</a>G&#41;&#46; And while cases of cutaneous mastocytosis with mast-cell infiltrates positive for CD1a exist&#44;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> in our first case&#44; the mast cells observed did not correspond to those of the predominant histiocyte infiltrate &#40;CD1a positive&#41;&#44; as in the second case&#46; The differential diagnosis of BCH includes entities such as urticaria pigmentosa&#44; lichenoid sarcoidosis&#44; and other types of histiocytosis&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;3</span></a> A histologic overlap is thought to exist between BCH and other types of non-Langerhans cell histiocytoses&#44; such as juvenile xanthogranuloma and generalized eruptive histiocytosis&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;3</span></a> For this reason&#44; a correct diagnosis requires taking into account the clinical and epidemiologic characteristics and the existence of extracutaneous involvement&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> In the first case&#44; the immunophenotypic findings led us to consider a diagnosis of indeterminate cell histiocytosis&#59; however&#44; this entity tends to affect adult patients and the lesions are more frequently located on the torso&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> In our 2 children&#44; the young age of presentation&#44; the distribution of the lesions&#44; the favorable clinical outcome with attenuation of the lesions in the following months&#44; and the lack of systemic involvement led us to a final diagnosis of BCH&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">BCH is a rare entity&#44; but prognosis is good and it is self-limiting in most cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;3</span></a> Although it is not generally associated with other diseases&#44; cases of association with diabetes mellitus<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> and diabetes insipidus<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> have been reported&#46; Given the few cases of BCH published in the literature&#44; either due to its low frequency or underdiagnosis&#44; we believe that these 2 new cases with unusual immunophenotypic findings may provide new data that can help to characterize the entity histologically&#46;</p></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Silva D&#237;az E&#44; V&#225;zquez Fern&#225;ndez B&#44; Monteagudo Castro C&#44; Mart&#237;n Hern&#225;ndez JM&#46; Histiocitosis cef&#225;lica benigna simulando una mastocitosis&#46; Actas Dermosifiliogr&#46; 2022&#59;113&#58;195&#8211;198&#46;</p>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0010">This work was presented in poster format at the 30th Meeting of the Spanish Group of Pediatric Dermatology GEDP held in Bilbao on January 25 and 26&#44; 2019&#46;</p>"
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">First patient&#46; <span class="elsevierStyleItalic">Clinical presentation&#58;</span> A&#44; Multiple brownish-red papules on the left cheek&#46; B and C&#44; Lesions on the right cheek that took on a more erythematous and edematous appearance on rubbing&#46; D-F&#44; Clinical deterioration of the lesions on the facial area and appearance of lesions on the upper back and arms&#46;</p> <p id="spar0010" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleItalic">Histology&#58;</span> G&#44; Discrete mononuclear infiltrate in the superficial dermis with disperse eosinophils&#46; H&#44; The vast majority of infiltrate cells are positive for CD163&#46; I&#44; Numerous intraepidermal and dermal cells positive for CD1a&#46; J&#44; Many of these are negative for langerin&#46; K&#44; A discrete increase in perivascular mast cells can be observed&#46; &#40;G&#44; HE&#8239;&#215;&#8239;200&#59; H&#44; CD163&#8239;&#215;&#8239;100&#59; I&#44; CD1a &#215;100&#59; J&#44; langerin &#215;400&#59; K&#44; c-kit &#215;200&#41;&#46;</p>"
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Case and Research Letter
Benign Cephalic Histiocytosis Mimicking Mastocytosis
Histiocitosis cefálica benigna simulando una mastocitosis
E. Silva Díaza,
Autor para correspondencia
, B. Vázquez Fernándeza, C. Monteagudo Castrob, J.M. Martín Hernándeza
a Servicio de Dermatología, Hospital Clínico Universitario de Valencia, Universidad de Valencia, Valencia, Spain
b Servicio de Anatomía Patológica, Hospital Clínico Universitario de Valencia, Universidad de Valencia, Valencia, Spain
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">First patient&#46; <span class="elsevierStyleItalic">Clinical presentation&#58;</span> A&#44; Multiple brownish-red papules on the left cheek&#46; B and C&#44; Lesions on the right cheek that took on a more erythematous and edematous appearance on rubbing&#46; D-F&#44; Clinical deterioration of the lesions on the facial area and appearance of lesions on the upper back and arms&#46;</p> <p id="spar0010" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleItalic">Histology&#58;</span> G&#44; Discrete mononuclear infiltrate in the superficial dermis with disperse eosinophils&#46; H&#44; The vast majority of infiltrate cells are positive for CD163&#46; I&#44; Numerous intraepidermal and dermal cells positive for CD1a&#46; J&#44; Many of these are negative for langerin&#46; K&#44; A discrete increase in perivascular mast cells can be observed&#46; &#40;G&#44; HE&#8239;&#215;&#8239;200&#59; H&#44; CD163&#8239;&#215;&#8239;100&#59; I&#44; CD1a &#215;100&#59; J&#44; langerin &#215;400&#59; K&#44; c-kit &#215;200&#41;&#46;</p>"
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Benign cephalic histiocytosis &#40;BCH&#41; is a rare type of non-Langerhans cell histiocytosis &#40;NLCH&#41; of unknown etiology&#59; it affects children under 3 years of age and tends to manifest in the first year of life&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Clinical characteristics include the appearance of multiple maculopapular lesions measuring between 2 and 8&#8239;mm in diameter&#44; with a yellowish-red or brownish-red color&#44; mainly distributed on the head and neck&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">Case 1</span>&#58; A 7-month-old infant with no relevant past history was evaluated for brownish maculas on the face&#44; measuring between 2 and 4&#8239;mm in diameter&#44; which had appeared 3 months earlier &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Figs&#46; 1</a>A and 1B&#41;&#46; The mother reported no other symptoms&#44; except for a change in color of the lesions on rubbing &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B and 1C&#41;&#46; A follow-up visit found an increase in number and intensity of the lesions&#44; and the appearance of new elements on the upper part of the back and the arms &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>D-1F&#41;&#46; A skin biopsy revealed a dense inflammatory infiltrate in the superficial dermis&#44; consisting predominantly of macrophages&#44; with few eosinophils&#46; Immune staining was positive for CD163 and CD68 in most cells&#46; Fifteen percent of cells were positive for CD1a and negative for S100 and langerin in the dermal infiltrate&#44; and intraepidermal levels were also very high&#46; Furthermore&#44; the c-KIT marker revealed a discrete increase in the predominantly perivascular mast-cell count &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>G-1&#8239;K&#41;&#46; Additional tests &#40;abdominal ultrasound&#44; blood test and urine sediment&#41; revealed a reduction in hemoglobin &#40;10&#46;1&#8239;g&#47;dL&#41; and a discrete increase in the size of the spleen&#59; for this reason&#44; the patient was sent to the pediatric hematologist for evaluation&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">Case 2</span>&#58; A 1-year-old child with no past history of interest visited with yellowish-red maculopapular lesions that had appeared on the face and upper part of the torso 2 months earlier &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Figs&#46; 2</a>A and 2B&#41;&#46; The lesions took on a redder color on crying&#46; Histopathology revealed a mononuclear inflammatory infiltrate in the superficial dermis&#46; Immune staining of the infiltrate was positive for CD163 and CD68 and negative for CD1a&#44; S100&#44; and langerin &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>C-2F&#41;&#46; As in the previous case&#44; a discrete increase in the number of mast cells was observed&#46; Blood tests revealed an elevated white blood cell count &#40;20450&#47;L&#41; with no other infection data&#59; the patient was therefore referred to the pediatric hematologist for evaluation&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">A diagnosis of iron deficiency anemia was made in the pediatric hematology consultation in the first case and blood-test and ultrasound results were normal a few months later&#46; The white blood cell count was normal in follow-up visits in the second patient&#46; Given the epidemiologic clinical context and after ruling out systemic involvement&#44; both children were diagnosed with BCH&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Non-Langerhans cell histiocytoses located in the skin and mucosa include a variety of entities that are classified depending on clinical signs and symptoms&#44; immunophenotype&#44; and the presence or absence of systemic involvement&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> According to a recently published classification&#44; BCH belongs to the group of juvenile xanthogranulomas &#40;in group C of non-Langerhans cell histiocytoses&#41;&#44; together with other entities that do not have systemic involvement&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> The term <span class="elsevierStyleItalic">cephalic</span> is subject to debate&#44; as involvement of the torso and upper extremities has been observed in a high percentage of cases&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Histologically&#44; it is characterized by a histiocyte infiltrate in the superficial and middle dermis&#44; with positive staining for CD68 and CD163&#44; and negative staining for CD1a&#44; S100&#44; and langerin&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;3</span></a> Cases of BCH have been published&#44; however&#44; with immunophenotypes positive for S100<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> and CD1a&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Darier&#8217;s sign is generally negative in histiocytoses&#44; although it has presented in some cases&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> In our 2 children&#44; the lesions took on a more erythematous tone in certain situations&#46; This &#8220;pseudo-Darier&#8221; phenomenon may be due&#44; in histologic terms&#44; to the discrete increase in mast cells observed in both biopsies &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Figs&#46; 1</a>K and <a class="elsevierStyleCrossRef" href="#fig0010">2</a>G&#41;&#46; And while cases of cutaneous mastocytosis with mast-cell infiltrates positive for CD1a exist&#44;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> in our first case&#44; the mast cells observed did not correspond to those of the predominant histiocyte infiltrate &#40;CD1a positive&#41;&#44; as in the second case&#46; The differential diagnosis of BCH includes entities such as urticaria pigmentosa&#44; lichenoid sarcoidosis&#44; and other types of histiocytosis&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;3</span></a> A histologic overlap is thought to exist between BCH and other types of non-Langerhans cell histiocytoses&#44; such as juvenile xanthogranuloma and generalized eruptive histiocytosis&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;3</span></a> For this reason&#44; a correct diagnosis requires taking into account the clinical and epidemiologic characteristics and the existence of extracutaneous involvement&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> In the first case&#44; the immunophenotypic findings led us to consider a diagnosis of indeterminate cell histiocytosis&#59; however&#44; this entity tends to affect adult patients and the lesions are more frequently located on the torso&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> In our 2 children&#44; the young age of presentation&#44; the distribution of the lesions&#44; the favorable clinical outcome with attenuation of the lesions in the following months&#44; and the lack of systemic involvement led us to a final diagnosis of BCH&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">BCH is a rare entity&#44; but prognosis is good and it is self-limiting in most cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;3</span></a> Although it is not generally associated with other diseases&#44; cases of association with diabetes mellitus<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> and diabetes insipidus<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> have been reported&#46; Given the few cases of BCH published in the literature&#44; either due to its low frequency or underdiagnosis&#44; we believe that these 2 new cases with unusual immunophenotypic findings may provide new data that can help to characterize the entity histologically&#46;</p></span>"
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">First patient&#46; <span class="elsevierStyleItalic">Clinical presentation&#58;</span> A&#44; Multiple brownish-red papules on the left cheek&#46; B and C&#44; Lesions on the right cheek that took on a more erythematous and edematous appearance on rubbing&#46; D-F&#44; Clinical deterioration of the lesions on the facial area and appearance of lesions on the upper back and arms&#46;</p> <p id="spar0010" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleItalic">Histology&#58;</span> G&#44; Discrete mononuclear infiltrate in the superficial dermis with disperse eosinophils&#46; H&#44; The vast majority of infiltrate cells are positive for CD163&#46; I&#44; Numerous intraepidermal and dermal cells positive for CD1a&#46; J&#44; Many of these are negative for langerin&#46; K&#44; A discrete increase in perivascular mast cells can be observed&#46; &#40;G&#44; HE&#8239;&#215;&#8239;200&#59; H&#44; CD163&#8239;&#215;&#8239;100&#59; I&#44; CD1a &#215;100&#59; J&#44; langerin &#215;400&#59; K&#44; c-kit &#215;200&#41;&#46;</p>"
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          "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Second patient&#46; <span class="elsevierStyleItalic">Clinical presentation&#58;</span> A and B&#44; Brownish-red maculopapular lesions on the facial area&#44; upper back&#44; and arms&#46; <span class="elsevierStyleItalic">Histology&#58;</span> C&#44; Mononuclear infiltrate in the superficial dermis&#46; D&#44; The dermal population is mostly positive for CD163&#46; E&#44; An unusually high number of cells positive for CD1a is not observed&#46; F&#44; A slight increase in perivascular mast cells can be observed&#46; &#40;D&#44; HE&#8239;&#215;&#8239;200&#59; E&#44; CD163&#8239;&#215;&#8239;100&#59; F&#44; CD1a &#215;100&#59; G&#44; c-kit &#215;400&#41;&#46;</p>"
        ]
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    ]
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