alexander Wagner S. De Souza Daniela Pereira cor de rosa Ana Letícia Pirozzi Buosi Ana Cecília Diniz Oliveira Jamil Natour About a authors

A vasculite testicular denominações uma expressão extra-articular extremamente rara da artrite reumatoide (AR). Descrevemos o situação de um homem de 53 anos alcançar diagnóstico de AR por oito anos, sem controle correto da doença. O paciente eu abri vasculite reumatoide, manifestada pela úlceras de membros inferiores e neuropatia periférica. Apresentou ainda meningite neutrofílica aguda, tendo sido tratado alcançar antibióticos e posterior pulsoterapia endovenosa alcançar metilprednisolona (500 mg/dia) através dos três dias, então de ciclofosfamida (2 mg/kg/ dia) e prednisona orais. Ministérios paciente demonstrado melhora do quadro, contudo 15 dias depois de ~ a alto hospitalar, houve reativação da meningite bacteriana. Ministérios paciente foi reinternado e tratado novamente alcançar antibióticos. N ° 3 dias mais tarde da segunda admissão hospitalar, ministérios paciente demonstrado dor, aumentar de volume são de testículo esquerdo e depois de gangrena. Foi realizada orquiectomia unilateral e o teste anatomopatológico revelado vasculite linfocítica. O paciente faleceu dois dias após a cirurgia devido naquela pneumonia aspirativa. Esse circunstâncias ilustra naquela vasculite testicular como uma expressão rara e tomb da vasculite reumatoide.

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Artrite reumatoide; Vasculite reumatoide; doença testiculares; Sepse


Testicular vasculitis is naquela very rarely extra-articular manifestation of rheumatoid arthritis (RA). We describe a case of der 53-year-old man diagnosed with RA para eight years, who era poorly controlled e developed rheumatoid vasculitis, which manifested as foot ulcers e peripheral polyneuropathy. A patient also had acute neutrophilic meningitis and was treated com antibiotics e intravenous pulse therapy com methylprednisolone (500 mg daily) porque o three days, adhered to by oral cyclophosphamide (2 mg/kg daily) e prednisone. Overall improvement foi ~ observed, e the patient was discharged. However 15 dia later, ns meningitis recurred, e the patient era readmitted e treated novamente with antibiotics. Three dia later, he developed pain and enlargement that his left testicle with gangrene. Unilateral orchiectomy foi ~ performed, revealing lymphocytic vasculitis. A patient died two dia later as result of aspiration pneumonia. This instance illustrates der rare and severe manifestation that rheumatoid vasculitis.

Arthritis, rheumatoid; Rheumatoid vasculitis; Testicular diseases; Sepsis


CASE REPORT

Division the Rheumatology, escolas Paulista de Medicina, Universidade commonwealth de são Paulo, elas Paulo, SP, Brazil

ABSTRACT

Testicular vasculitis is der very rarely extra-articular manifestation of rheumatoid arthritis (RA). We describe ns case of a 53-year-old guy diagnosed with RA ao eight years, who foi ~ poorly controlled and developed rheumatoid vasculitis, which manifested as foot ulcers e peripheral polyneuropathy. Ns patient additionally had acute neutrophilic meningitis and was treated com antibiotics and intravenous pulse therapy com methylprednisolone (500 mg daily) ao three days, adhered to by boca cyclophosphamide (2 mg/kg daily) e prednisone. Overall improvement ser estar observed, e the patient foi ~ discharged. But 15 dia later, the meningitis recurred, and the patient era readmitted and treated novamente with antibiotics. Three dia later, he emerged pain and enlargement that his left testicle with gangrene. Unilateralmente orchiectomy was performed, revealing lymphocytic vasculitis. The patient died two dia later as result of aspiration pneumonia. This case illustrates a rare and severe manifestation the rheumatoid vasculitis.

Keywords: Arthritis, rheumatoid; Rheumatoid vasculitis; Testicular diseases; Sepsis

Introduction

Rheumatoid vasculitis (RV) is considered der complication associated com longstanding rheumatoid arthritis (RA), usually of much more than 10-year duration.1 RV is linked with naquela worse prognosis due to a increased morbidity e mortality.2,3 the is often manifested by digital infarcts, foot ulcers, systemic manifestations and mononeuritis multiplex, but may involve any type of blood courage bed, particularly mesenteric, coronary e cerebral arteries. RV is an ext common in men e some predictors such as smoking, rheumatoid nodules e genetic components have to be recognized.1,4 the 30-year incidence that RV is estimated to it is in 3.6% among patients with RA.5 naquela possible method to the diagnosis of RV is a use of the proposed diagnostic criteria; back helpful, these criteria have not to be validated yet.4,6,7

Testicular inflammation has actually been recognized as naquela manifestation of numerous systemic vasculitides such as Henoch-Schönlein purpura e polyarteritis nodosa or the may take place in one isolated form of testicular vasculitis.8-10 In RV testicular joining is an extremely rare manifestation e to our understanding there is one case report explicate this complication in naquela patient com RA e another in youth chronic arthritis.11,12 Herein, us describe a case the RV in a patient with a seropositive and poorly-controlled RA.

Case report

A 53-year-old, white male era admitted in the university hospital because of leg ulcers, tingling, numbness and weakness on potencial elétrico lower limbs and a worsening of pain e swelling in small e large joints over the last 3 weeks. Five dia before join he started to present symptoms of fever, major headache and vomiting. He had been diagnosed com RA porque o 8 years with symmetrical polyarthritis and a hopeful rheumatoid factor (RF) 640 IU/mL. In addition, the had naquela past medical background of systemic hypertension, form 2 diabetes mellitus e smoking. Following ns RA diagnosis, he foi ~ treated with glucocorticoids (GC) and methotrexate e then com methotrexate and leflunomide 20 mg/day com some advancement of joint complaints that lasted 4 years. The did not attend a follow-up consultations for the next three years e remained apenas um on predinisone 20 mg QD without any kind of associated immunosuppressive agent.

On examination, the patient was febrile and had mild cushingoid features. Dois deep leg ulcers with elevated margins e fibrin to be observed top top his left leg. Swelling e tenderness to be observed over proxial interphalangeal and metacarpophalangeal joints, wrists, elbows and knees, e calculated DAS-28 score yielded a result of 5.0. Neurological check revealed normal mental status, neck stiffness, decreased deep tendon reflexes in reduced limbs, impaired sensitivity below the knees e drop left foot. Cerebrospinal fluid (CSF) analysis foi ~ suggestive of bacter meningitis with 2,832 cells/mm3 (98% the neutrophils), protein 376.5 mg/dL e glucose 69 mg/dL. Gram stain, CSF cultures for aerobic bacteria, fungi and mycobacteria to be negative. CSF serologic tests porque o toxoplasmosis, syphilis, cytomegalovirus, herpes simplex and Cryptococcus were likewise negative. One incisional skin biopsy showed a non-specific ulcer com neutrophilic vasculitis in der small artery e an electromyography proved motor e sensitive axonal polyneuropathy. RF remained confident (1,280 IU/mL) e anti-profillagrin antibodies were hopeful (titer 1/320), antinuclear antitoxin (ANA), antineutrophil cytoplasmic antibody (ANCA), cryoglobulins and serum tests ao HIV, hepatitis B and C were todos negative.

The patient foi ~ treated com ceftriaxone 4 g/day during 10 days and received intravenous pulse therapy com methylprednisolone 500 mg daily ao three days followed by oralmente prednisone 60 mg/day e oral cyclophosphamide 2 mg/kg/ day. An in its entirety improvement was observed for leg ulcers, meningitis, share complaints and peripheral neuropathy.

Fifteen dia after discharge, the patient experienced the recurrence of fever, headache and vomiting. He returned to hospital, underwent another lumbar puncture and CSF evaluation showed 4,640 cells/mm3 (84% the neutrophils), protein 276.5 mg/dL and glucose 116 mg/dL. These results shown the new onset that meningitis. One infection led to by Listeria monocytogenes foi ~ suspected and he ser estar treated with meropenen e ampicillin while the combination that prednisone e cyclophosphamide was temporarily withdrawn. Three days after admission, he complained about scrotal pain and enlargement of his left testicle with a development of indications of gangrene a few hours later. Urologic consultation recommended the surgical procedure e a unilateral básico orchiectomy was performed. A evaluation the pathological specimen showed transmural lymphocytic vasculitis in justafunicular artérias branches with myointimal proliferation (Fig.

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1). The patient developed aspiration pneumonia and sepsis after surgery e despite the treatment com antibiotics, vasoactive drugs and mechanical ventilation; he passed away two dia later due to septic shock.