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and erythematous lesions with adhered crusts on the nose&#44; forehead&#44; and cheeks &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The biopsy of the cervical lymph nodes revealed a sarcoid granulomatous lymphadenitis&#44; with negative staining for Ziehl-Neelsen&#44; Grocott&#44; and PAS&#46; Blood cultures and urine tests were negative for aerobes&#44; anaerobes&#44; and <span class="elsevierStyleItalic">Mycobacterium</span> species were negative&#46; The histologic study of the skin lesions on the face and right foot revealed the existence of naked granulomas&#44; with no central necrosis or lymphocytic corona&#44; in the deep and middle dermis &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Blood tests showed increased levels of angiotensin-converting enzyme and acute phase reactants&#44; and serology for syphilis was negative&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">The x-ray study revealed destruction of the distal phalanx of the first toe&#44; with signs of bone resorption and tendinitis&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">The CT scan showed a small number of enlarged hilar&#44; subcarinal mediastinal&#44; and retroperitoneal lymph nodes associated with bilateral micronodules in the upper third of the lung fields&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">A diagnosis of sarcoid dactylitis in the context of systemic sarcoidosis was made and treatment was initiated with oral corticosteroids associated with hydroxychloroquine&#59; a slight improvement was achieved and this treatment was therefore suspended&#46; Treatment was begun with 150&#160;mg&#47;d of azathioprine&#46; After 6 months of treatment&#44; the swelling of the toes had been reduced and the crusts of the facial lesions had disappeared &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; After a year of treatment only the erythematous lesions persist&#44; with no crust or swelling&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0040" class="elsevierStylePara elsevierViewall">Bone involvement in sarcoidosis usually manifests in the form of joint pain&#44; whereas deforming arthritis is rare&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Two patterns of appearance of sarcoid joint disease exist&#58; an early form of presentation that occurs in the first 6 months and is usually observed in L&#246;fgren syndrome&#44; and a late-onset form that affects multiple joints&#44; generally those of the small bones of the hands and feet and which appears after 6 months&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> They are often associated with nail disorders&#44; acropachy with racket nails&#44; or acrosclerosis&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">Radiologically&#44; sarcoid dactylitis is characterized by trabecular abnormalities with a honeycomb or barred pattern&#44; which may be associated with cystic formations and a periosteal reaction&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Histologically&#44; in bone lesions&#44; noncaseating granulomas are found that invade the phalanges and the surrounding soft tissue&#46; This granulomatous dactylitis is associated with a worse outcome and greater risk of systemic involvement and of lupus pernio in chronic sarcoidosis&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Lupus pernio is one of the most characteristic lesions of sarcoidosis&#44; which is more frequent in Caucasian women with long-term disease&#44; as in our case&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> It is characterized by pink or violaceous papular lesions on the nose&#44; cheeks&#44; ears&#44; lips&#44; or scalp&#46; It tends to be associated with chronic sarcoidosis and involvement of the upper respiratory apparatus&#46; If the lupus pernio lesions are located on the fingers or toes&#44; cystic bone lesions on the phalanges of the affected members are often observed&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">Bone involvement tends to be associated with a fourfold higher mortality&#59; however&#44; patients do not usually have symptoms&#44; despite the presence of abnormal radiologic findings&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">Treatment of sarcoid dactylitis requires the use of high-dose corticosteroids associated with antimalarial drugs and methotrexate&#46; In our patient&#44; we ruled out the use of methotrexate owing to the patient&#8217;s stated desire to have children and we therefore used azathioprine for some time&#44; 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Case and Research Letters
Granulomatous Dactylitis: An Unusual Finding in Sarcoidosis
Dactilitis granulomatosa como manifestación inusual de la sarcoidosis
I. García Moralesa,
Corresponding author
irenegmorales@gmail.com

Corresponding author.
, E. Sánchez Garcíab, A. Crespo Cruzc, J. Escuderoc
a Unidad de Gestión Clínica de Dermatología, Hospital Universitario Virgen Macarena, Sevilla, Spain
b Servicio de Medicina Interna, Hospital Universitario Nuestra Señora de Valme, Sevilla, Spain
c Unidad de Gestión Clínica de Dermatología, Hospital Universitario Nuestra Señora de Valme, Sevilla, Spain
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and erythematous lesions with adhered crusts on the nose&#44; forehead&#44; and cheeks &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The biopsy of the cervical lymph nodes revealed a sarcoid granulomatous lymphadenitis&#44; with negative staining for Ziehl-Neelsen&#44; Grocott&#44; and PAS&#46; Blood cultures and urine tests were negative for aerobes&#44; anaerobes&#44; and <span class="elsevierStyleItalic">Mycobacterium</span> species were negative&#46; The histologic study of the skin lesions on the face and right foot revealed the existence of naked granulomas&#44; with no central necrosis or lymphocytic corona&#44; in the deep and middle dermis &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Blood tests showed increased levels of angiotensin-converting enzyme and acute phase reactants&#44; and serology for syphilis was negative&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">The x-ray study revealed destruction of the distal phalanx of the first toe&#44; with signs of bone resorption and tendinitis&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">The CT scan showed a small number of enlarged hilar&#44; subcarinal mediastinal&#44; and retroperitoneal lymph nodes associated with bilateral micronodules in the upper third of the lung fields&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">A diagnosis of sarcoid dactylitis in the context of systemic sarcoidosis was made and treatment was initiated with oral corticosteroids associated with hydroxychloroquine&#59; a slight improvement was achieved and this treatment was therefore suspended&#46; Treatment was begun with 150&#160;mg&#47;d of azathioprine&#46; After 6 months of treatment&#44; the swelling of the toes had been reduced and the crusts of the facial lesions had disappeared &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; After a year of treatment only the erythematous lesions persist&#44; with no crust or swelling&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0040" class="elsevierStylePara elsevierViewall">Bone involvement in sarcoidosis usually manifests in the form of joint pain&#44; whereas deforming arthritis is rare&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Two patterns of appearance of sarcoid joint disease exist&#58; an early form of presentation that occurs in the first 6 months and is usually observed in L&#246;fgren syndrome&#44; and a late-onset form that affects multiple joints&#44; generally those of the small bones of the hands and feet and which appears after 6 months&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> They are often associated with nail disorders&#44; acropachy with racket nails&#44; or acrosclerosis&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">Radiologically&#44; sarcoid dactylitis is characterized by trabecular abnormalities with a honeycomb or barred pattern&#44; which may be associated with cystic formations and a periosteal reaction&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Histologically&#44; in bone lesions&#44; noncaseating granulomas are found that invade the phalanges and the surrounding soft tissue&#46; This granulomatous dactylitis is associated with a worse outcome and greater risk of systemic involvement and of lupus pernio in chronic sarcoidosis&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Lupus pernio is one of the most characteristic lesions of sarcoidosis&#44; which is more frequent in Caucasian women with long-term disease&#44; as in our case&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> It is characterized by pink or violaceous papular lesions on the nose&#44; cheeks&#44; ears&#44; lips&#44; or scalp&#46; It tends to be associated with chronic sarcoidosis and involvement of the upper respiratory apparatus&#46; If the lupus pernio lesions are located on the fingers or toes&#44; cystic bone lesions on the phalanges of the affected members are often observed&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">Bone involvement tends to be associated with a fourfold higher mortality&#59; however&#44; patients do not usually have symptoms&#44; despite the presence of abnormal radiologic findings&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">Treatment of sarcoid dactylitis requires the use of high-dose corticosteroids associated with antimalarial drugs and methotrexate&#46; In our patient&#44; we ruled out the use of methotrexate owing to the patient&#8217;s stated desire to have children and we therefore used azathioprine for some time&#44; and we even considered the use of adalimumab&#44; but this was refused by the patient&#46; The use of anti-TNF-&#945; agents&#44; infliximab&#44; or adalimumab is considered third-line treatment in sarcoidosis&#44; and is useful in refractory cases or cases with ocular or systemic involvement&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a></p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0070" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest&#46;</p></span></span>"
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Article information
ISSN: 15782190
Original language: English
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Idiomas
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