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platelets 634&#44;000<span class="elsevierStyleHsp" style=""></span>k&#47;&#956;l &#40;150&#44;000&#8211;400&#44;000&#41;&#44; GTO&#58; 144<span class="elsevierStyleHsp" style=""></span>U&#47;l &#40;up to 38&#41;&#44; TGP&#58; 192<span class="elsevierStyleHsp" style=""></span>U&#47;l &#40;up to 46&#41;&#44; alkaline phosphatase 448<span class="elsevierStyleHsp" style=""></span>U&#47;l &#40;35&#8211;105&#41;&#44; direct bilirubin &#46;31<span class="elsevierStyleHsp" style=""></span>mg&#47;dl &#40;up to &#46;20&#41;&#44; CPK 1232<span class="elsevierStyleHsp" style=""></span>IU&#47;l &#40;&#60;135&#41;&#44; LDH 233 &#40;&#60;460&#41;&#44; aldolase 10<span class="elsevierStyleHsp" style=""></span>U&#47;l &#40;&#60;3&#41;&#44; ESR 119<span class="elsevierStyleHsp" style=""></span>mm&#47;1<span class="elsevierStyleHsp" style=""></span>h &#40;up to 20&#41;&#44; CRP 317<span class="elsevierStyleHsp" style=""></span>mg&#47;l &#40;0&#8211;5&#41;&#46; Renal function and serologies for hepatitis B and C&#59; HIV&#44; ANF &#40;HeP-2&#41;&#44; nDNA <span class="elsevierStyleItalic">&#40;Crithidia&#41;</span>&#44; ENA&#44; p-ANCA and c-ANCA and complements&#58; negative or normal&#46; MRI showed hyperintensity in the twin muscles compatible with myositis&#44; while electromyography of the upper and lower limbs showed a motor-sensitive polyneuropathy with decreased sural nerve conduction speed&#46; Inflammatory infiltration&#44; atrophy and focal perifascicular regeneration was found on muscle biography &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Biopsy of the sural nerve showed moderate myelinated fibre depletion and signs of axonal degeneration along with extensive lymphocytic infiltration around the small epineurial vessels and the wall of the median artery&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">The skin biopsy showed acute and chronic inflammatory infiltration of septal distribution and medium-sized vessels with vasculitic phenomena and fibrinoid necrosis of the septal wall with erythrocyte extravasation in the hypodermis&#46; Aortography and total bone scan were normal&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">With a diagnosis of PAN &#40;Chapell Hill and ACR criteria&#58; weight loss of more than 4<span class="elsevierStyleHsp" style=""></span>kg&#44; testicular pain&#44; myalgia&#44; polyneuritis demonstrated by electromyography&#44; diastolic blood pressure higher than 90<span class="elsevierStyleHsp" style=""></span>mmHg and medium-sized artery biopsy compatible with vasculitis&#41; 3 pulses of intravenous methylprednisolone 1<span class="elsevierStyleHsp" style=""></span>g&#47;day were given&#44; continuing with 60<span class="elsevierStyleHsp" style=""></span>mg&#47;day of oral meprednisone and monthly intravenous cyclophosphamide&#46; The patient made good progress&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0040" class="elsevierStylePara elsevierViewall">PAN is a vasculitis that predominantly affects the small and medium-sized arteries&#46; It affects the skin &#40;20&#37;&#41;&#44; gastrointestinal tract &#40;50&#37;&#41;&#44; kidneys &#40;40&#37;&#8211;60&#37;&#41; and peripheral nerves &#40;60&#37;&#41;&#46; Muscle compromise is not a feature of this disease&#46; Although myalgia&#44; swelling and impossibility of walking can present in 51&#37; of patients&#44;<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> myositis is found in 19&#37; of these diseases&#44;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> and there are only 14 cases reporting this compromise as a form of onset of this vasculitis&#46;<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2&#44;6&#44;7</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The muscle enzymes are elevated in only 5&#37; of cases&#44; as occurred in the case we describe&#46; The CPK enzymes &#40;5&#37;&#8211;25&#37;&#41; are the most altered&#44; and elevated LDH and aldolase have been reported in some cases&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">These cases must be differentiated from polymyositis and myositis of other origins&#44; and therefore diagnosis relies on anatomical pathology&#46; It is appropriate to choose the biopsy site by MRI&#44; which shows increased signal intensity on the T2 and STIR sequences&#44; indicating inflammatory changes&#46;<a class="elsevierStyleCrossRefs" href="#bib0070"><span class="elsevierStyleSup">4&#8211;7</span></a> Almost all the patients reported underwent a muscular biopsy&#44; compatible with vasculitis&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3&#44;9&#44;10</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">This is the first described case with myositis demonstrated by histology&#46; These histological signs might have gone unnoticed in previous cases&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conclusion</span><p id="par0060" class="elsevierStylePara elsevierViewall">Perhaps we ought to broaden the spectrum when analysing muscle biopsies for PAN and look for signs of vasculitis as well as myositis&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Ethical Disclosures</span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Protection of people and animals</span><p id="par0065" class="elsevierStylePara elsevierViewall">The authors declare that no experiments were performed on humans or animals for this study&#46;</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Confidentiality of data</span><p id="par0070" class="elsevierStylePara elsevierViewall">The authors declare that no patient data appear in this article&#46;</p></span><span id="sec0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Right to privacy and informed consent</span><p id="par0075" class="elsevierStylePara elsevierViewall">The authors declare that no patient data appear in this article&#46;</p></span></span><span id="sec0045" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0065">Conflict of Interest</span><p id="par0085" class="elsevierStylePara elsevierViewall">The authors have no conflict of interest to declare&#46;</p></span></span>"
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        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A 47-year-old man presented with weight loss&#44; bilateral calf pain&#44; fever&#44; hypertension&#44; orchitis and oligoarthritis&#46; Lab tests&#58; anemia and elevated muscle enzymes&#46; Resonance magnetic imaging&#58; hyperintensity in gastrocnemius muscles &#40;myositis&#41;&#46; Histologic exam of the muscles&#58; inflammatory infiltrate with atrophy and perifascicular regeneration&#46; Treatment&#58; methylprednisone &#40;bolus&#41; and cyclophosphamide&#46; Muscle pain and swelling and difficulty in walking are common in panarteritis nodosa &#40;PAN&#41;&#44; whereas histologically demonstrated myositis is not&#46; Even more rare is myositis as the initial presentation of this vasculitis&#46;</p></span>"
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        "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Var&#243;n de 47 a&#241;os que consulta por p&#233;rdida de peso&#44; dolor con tumefacci&#243;n en pantorrillas&#44; fiebre&#44; hipertensi&#243;n arterial&#44; orquitis y oligoartritis&#46; Laboratorio&#58; anemia y aumento de enzimas musculares&#46; Resonancia magn&#233;tica&#58; hiperintensidad en gemelos &#40;miositis&#41;&#46; Histolog&#237;a de m&#250;sculo&#58; infiltrado inflamatorio con atrofia y regeneraci&#243;n perifascicular&#46; Tratamiento&#58; pulsos de metilprednisolona y ciclofosfamida&#46; Mialgias&#44; tumefacci&#243;n muscular y deambulaci&#243;n dificultosa son hallazgos comunes en poliarteritis nodosa &#40;PAN&#41;&#44; no as&#237; la miositis demostrada histol&#243;gicamente y m&#225;s infrecuente a&#250;n como forma de inicio de esta vasculitis&#46;</p></span>"
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Case report
Myositis as the Initial Presentation of Panarteritis Nodosa
Miositis como forma de presentación de panarteritis nodosa
Romina Calvo, Melina Negri, Alberto Ortiz, Susana Roverano, Sergio Paira
Corresponding author
pairasergio@hotmail.com

Corresponding author.
Servicio de Reumatología, Hospital J.M. Cullen, Santa Fe, Argentina
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neuropathy due to involvement of the peripheral nervous system or&#44; more rarely&#44; to myositis&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3&#8211;8</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">We describe a case of PAN presenting with calf pain due to myositis as a form of onset of this vasculitis&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Clinical Observation</span><p id="par0020" class="elsevierStylePara elsevierViewall">A 47-year-old male consulted with a one-month history of weight loss &#40;10<span class="elsevierStyleHsp" style=""></span>kg&#41; and spontaneous&#44; incapacitating calf pain&#46; Physical examination&#58; fever &#40;38&#46;5<span class="elsevierStyleHsp" style=""></span>&#176;<span class="elsevierStyleSmallCaps">C</span>&#41; predominantly in the evening&#44; tachycardia&#44; blood pressure 160&#47;100<span class="elsevierStyleHsp" style=""></span>mmHg&#44; erythematous lumps in the twin muscles &#40;approximately 6<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleHsp" style=""></span>cm&#41;&#44; of hard-elastic consistency and warm&#44; and thermalgesic sensitivity was preserved &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Erythematous purpuric macules were also observed on the anterior and posterior surfaces of the legs&#44; testicular pain on palpation&#44; and arthritis in carpi&#44; metacarpophalangeal and proximal interphalangeal joints&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Laboratory tests&#58; anaemia with haemoglobin&#58; 10<span class="elsevierStyleHsp" style=""></span>g&#47;dl &#40;11&#46;9&#8211;15<span class="elsevierStyleHsp" style=""></span>g&#47;dl&#41;&#44; white cells 25&#44;160<span class="elsevierStyleHsp" style=""></span>k&#47;&#956;l &#40;4500&#8211;10&#44;000&#41;&#44; platelets 634&#44;000<span class="elsevierStyleHsp" style=""></span>k&#47;&#956;l &#40;150&#44;000&#8211;400&#44;000&#41;&#44; GTO&#58; 144<span class="elsevierStyleHsp" style=""></span>U&#47;l &#40;up to 38&#41;&#44; TGP&#58; 192<span class="elsevierStyleHsp" style=""></span>U&#47;l &#40;up to 46&#41;&#44; alkaline phosphatase 448<span class="elsevierStyleHsp" style=""></span>U&#47;l &#40;35&#8211;105&#41;&#44; direct bilirubin &#46;31<span class="elsevierStyleHsp" style=""></span>mg&#47;dl &#40;up to &#46;20&#41;&#44; CPK 1232<span class="elsevierStyleHsp" style=""></span>IU&#47;l &#40;&#60;135&#41;&#44; LDH 233 &#40;&#60;460&#41;&#44; aldolase 10<span class="elsevierStyleHsp" style=""></span>U&#47;l &#40;&#60;3&#41;&#44; ESR 119<span class="elsevierStyleHsp" style=""></span>mm&#47;1<span class="elsevierStyleHsp" style=""></span>h &#40;up to 20&#41;&#44; CRP 317<span class="elsevierStyleHsp" style=""></span>mg&#47;l &#40;0&#8211;5&#41;&#46; Renal function and serologies for hepatitis B and C&#59; HIV&#44; ANF &#40;HeP-2&#41;&#44; nDNA <span class="elsevierStyleItalic">&#40;Crithidia&#41;</span>&#44; ENA&#44; p-ANCA and c-ANCA and complements&#58; negative or normal&#46; MRI showed hyperintensity in the twin muscles compatible with myositis&#44; while electromyography of the upper and lower limbs showed a motor-sensitive polyneuropathy with decreased sural nerve conduction speed&#46; Inflammatory infiltration&#44; atrophy and focal perifascicular regeneration was found on muscle biography &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Biopsy of the sural nerve showed moderate myelinated fibre depletion and signs of axonal degeneration along with extensive lymphocytic infiltration around the small epineurial vessels and the wall of the median artery&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">The skin biopsy showed acute and chronic inflammatory infiltration of septal distribution and medium-sized vessels with vasculitic phenomena and fibrinoid necrosis of the septal wall with erythrocyte extravasation in the hypodermis&#46; Aortography and total bone scan were normal&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">With a diagnosis of PAN &#40;Chapell Hill and ACR criteria&#58; weight loss of more than 4<span class="elsevierStyleHsp" style=""></span>kg&#44; testicular pain&#44; myalgia&#44; polyneuritis demonstrated by electromyography&#44; diastolic blood pressure higher than 90<span class="elsevierStyleHsp" style=""></span>mmHg and medium-sized artery biopsy compatible with vasculitis&#41; 3 pulses of intravenous methylprednisolone 1<span class="elsevierStyleHsp" style=""></span>g&#47;day were given&#44; continuing with 60<span class="elsevierStyleHsp" style=""></span>mg&#47;day of oral meprednisone and monthly intravenous cyclophosphamide&#46; The patient made good progress&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0040" class="elsevierStylePara elsevierViewall">PAN is a vasculitis that predominantly affects the small and medium-sized arteries&#46; It affects the skin &#40;20&#37;&#41;&#44; gastrointestinal tract &#40;50&#37;&#41;&#44; kidneys &#40;40&#37;&#8211;60&#37;&#41; and peripheral nerves &#40;60&#37;&#41;&#46; Muscle compromise is not a feature of this disease&#46; Although myalgia&#44; swelling and impossibility of walking can present in 51&#37; of patients&#44;<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> myositis is found in 19&#37; of these diseases&#44;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> and there are only 14 cases reporting this compromise as a form of onset of this vasculitis&#46;<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2&#44;6&#44;7</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The muscle enzymes are elevated in only 5&#37; of cases&#44; as occurred in the case we describe&#46; The CPK enzymes &#40;5&#37;&#8211;25&#37;&#41; are the most altered&#44; and elevated LDH and aldolase have been reported in some cases&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">These cases must be differentiated from polymyositis and myositis of other origins&#44; and therefore diagnosis relies on anatomical pathology&#46; It is appropriate to choose the biopsy site by MRI&#44; which shows increased signal intensity on the T2 and STIR sequences&#44; indicating inflammatory changes&#46;<a class="elsevierStyleCrossRefs" href="#bib0070"><span class="elsevierStyleSup">4&#8211;7</span></a> Almost all the patients reported underwent a muscular biopsy&#44; compatible with vasculitis&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3&#44;9&#44;10</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">This is the first described case with myositis demonstrated by histology&#46; These histological signs might have gone unnoticed in previous cases&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conclusion</span><p id="par0060" class="elsevierStylePara elsevierViewall">Perhaps we ought to broaden the spectrum when analysing muscle biopsies for PAN and look for signs of vasculitis as well as myositis&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Ethical Disclosures</span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Protection of people and animals</span><p id="par0065" class="elsevierStylePara elsevierViewall">The authors declare that no experiments were performed on humans or animals for this study&#46;</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Confidentiality of data</span><p id="par0070" class="elsevierStylePara elsevierViewall">The authors declare that no patient data appear in this article&#46;</p></span><span id="sec0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Right to privacy and informed consent</span><p id="par0075" class="elsevierStylePara elsevierViewall">The authors declare that no patient data appear in this article&#46;</p></span></span><span id="sec0045" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0065">Conflict of Interest</span><p id="par0085" class="elsevierStylePara elsevierViewall">The authors have no conflict of interest to declare&#46;</p></span></span>"
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        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A 47-year-old man presented with weight loss&#44; bilateral calf pain&#44; fever&#44; hypertension&#44; orchitis and oligoarthritis&#46; Lab tests&#58; anemia and elevated muscle enzymes&#46; Resonance magnetic imaging&#58; hyperintensity in gastrocnemius muscles &#40;myositis&#41;&#46; Histologic exam of the muscles&#58; inflammatory infiltrate with atrophy and perifascicular regeneration&#46; Treatment&#58; methylprednisone &#40;bolus&#41; and cyclophosphamide&#46; Muscle pain and swelling and difficulty in walking are common in panarteritis nodosa &#40;PAN&#41;&#44; whereas histologically demonstrated myositis is not&#46; Even more rare is myositis as the initial presentation of this vasculitis&#46;</p></span>"
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        "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Var&#243;n de 47 a&#241;os que consulta por p&#233;rdida de peso&#44; dolor con tumefacci&#243;n en pantorrillas&#44; fiebre&#44; hipertensi&#243;n arterial&#44; orquitis y oligoartritis&#46; Laboratorio&#58; anemia y aumento de enzimas musculares&#46; Resonancia magn&#233;tica&#58; hiperintensidad en gemelos &#40;miositis&#41;&#46; Histolog&#237;a de m&#250;sculo&#58; infiltrado inflamatorio con atrofia y regeneraci&#243;n perifascicular&#46; Tratamiento&#58; pulsos de metilprednisolona y ciclofosfamida&#46; Mialgias&#44; tumefacci&#243;n muscular y deambulaci&#243;n dificultosa son hallazgos comunes en poliarteritis nodosa &#40;PAN&#41;&#44; no as&#237; la miositis demostrada histol&#243;gicamente y m&#225;s infrecuente a&#250;n como forma de inicio de esta vasculitis&#46;</p></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Calvo R&#44; Negri M&#44; Ortiz A&#44; Roverano S&#44; Paira S&#46; Miositis como forma de presentaci&#243;n de panarteritis nodosa&#46; Reumatol Clin&#46; 2019&#59;15&#58;e24&#8211;e26&#46;</p>"
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Article information
ISSN: 21735743
Original language: English
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Idiomas
Reumatología Clínica (English Edition)
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