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1.
Sci Rep ; 11(1): 8393, 2021 04 16.
Artigo em Inglês | MEDLINE | ID: mdl-33863937

RESUMO

Childhood-onset polyarteritis nodosa (PAN) is a rare and systemic necrotising vasculitis in children affecting small- to medium-sized arteries. To date, there have been only a few reports because of its rarity. Thus, we aimed to investigate the clinical manifestations, laboratory findings, treatment, and long-term outcomes in patients with childhood-onset PAN and to evaluate the usefulness of the paediatric vasculitis activity score (PVAS). We retrospectively analysed the data of nine patients with childhood-onset PAN from March 2003 to February 2020. The median ages at symptom onset, diagnosis, and follow-up duration were 7.6 (3-17.5), 7.7 (3.5-17.6), and 7.0 (1.6-16.3) years, respectively. All patients had constitutional symptoms and skin manifestations, while five exhibited Raynaud's phenomenon. Organ involvement was observed in one patient. The median PVAS at diagnosis was 7 (range: 2-32). Prednisolone was initially used for induction in all patients, and other drugs were added in cases refractory to prednisolone. All patients survived, but three patients with high PVAS at diagnosis experienced irreversible sequelae, including intracranial haemorrhage and digital amputation. In conclusion, early diagnosis and treatment may minimise sequelae in patients with childhood-onset PAN. This study suggests that high PVAS score at diagnosis may be associated with poor prognosis.


Assuntos
Poliarterite Nodosa/patologia , Vasculite/patologia , Adolescente , Criança , Pré-Escolar , Progressão da Doença , Feminino , Humanos , Masculino , Poliarterite Nodosa/epidemiologia , Poliarterite Nodosa/cirurgia , Prognóstico , República da Coreia/epidemiologia , Estudos Retrospectivos , Vasculite/epidemiologia , Vasculite/cirurgia
5.
J Cutan Pathol ; 47(11): 1076-1079, 2020 Nov.
Artigo em Inglês | MEDLINE | ID: mdl-32761868

RESUMO

Polyarteritis nodosa (PAN) is a necrotizing vasculitis. The clinical manifestations are determined by the location of the compromised arteries. Cutaneous PAN can present as nodular lesions similar to erythema nodosum, palpable purpura, livedo reticularis, and ulceration. It often affects the lower limbs but other anatomical sites can also be involved. However, concomitant facial edema is an extremely rare manifestation. It has been more than 20 years since the last case report describing this unusual presentation of PAN. Furthermore, our patient is the first case presenting with hemifacial edema fluctuating every second or third day due to PAN confirmed by skin biopsy.


Assuntos
Edema/etiologia , Face/patologia , Poliarterite Nodosa/patologia , Dermatopatias/patologia , Humanos , Masculino , Poliarterite Nodosa/complicações , Dermatopatias/etiologia , Adulto Jovem
6.
Immunol Res ; 68(3): 152-160, 2020 06.
Artigo em Inglês | MEDLINE | ID: mdl-32578130

RESUMO

The etiology of polyarteritis nodosa (PAN) and localized PAN, including cutaneous arteritis (CA), remains unknown; however, initial endothelial damage has been implicated. The intima of the vasculitis lesions is predominantly infiltrated by innate-like bystander-activated CD8 T cells, in addition to the macrophages. Macrophages are among the major inflammatory cells involved in innate immunity and are classified into M1 and M2 subtypes. M1-type macrophages kill pathogens and cause inflammation, while M2-type macrophages promote the repair of tissues. Macrophage subtypes infiltrating in PAN and localized PAN vasculitis lesions have not yet been investigated. Innate immune response to a triggering factor on the endothelial cell surface may initiate CA pathogenesis. Thus, many M1-type macrophages may infiltrate in the intima during early CA. We assessed this hypothesis by immunohistochemical observation of macrophage phenotypes and polarization. Twenty-seven skin biopsy specimens from patients with CA were retrieved. Based on histology, we classified CA into four phases. The phenotypes of infiltrating macrophages in CA were evaluated by immunohistochemistry using antibodies against Iba-1, a pan-macrophage marker, and CD163, an M2-type macrophage marker. Our results showed that the ratio of CD163-positive M2-type macrophages to Iba1-positive macrophages was lower in the intima in the early stage of CA than in the later stage. In the media to adventitia, there was no significant difference in the ratios between these stages. These findings indicate that innate immunity is involved in the intima in the early stage of CA, suggesting that a trigger for CA might exist in endothelial cells.


Assuntos
Endotélio Vascular/patologia , Macrófagos/imunologia , Poliarterite Nodosa/imunologia , Pele/irrigação sanguínea , Adulto , Idoso , Antígenos CD/metabolismo , Antígenos de Diferenciação Mielomonocítica/metabolismo , Biomarcadores/metabolismo , Biópsia , Proteínas de Ligação ao Cálcio/metabolismo , Endotélio Vascular/citologia , Endotélio Vascular/imunologia , Feminino , Humanos , Imunidade Inata , Macrófagos/metabolismo , Masculino , Proteínas dos Microfilamentos/metabolismo , Pessoa de Meia-Idade , Poliarterite Nodosa/patologia , Receptores de Superfície Celular/metabolismo , Pele/imunologia , Pele/patologia , Adulto Jovem
8.
J Dermatol ; 47(5): 534-537, 2020 May.
Artigo em Inglês | MEDLINE | ID: mdl-32096266

RESUMO

We developed a questionnaire to examine the findings of cutaneous arteritis among dermatological specialists experienced in vasculitis as certified by the Committee for guidelines for the management of vasculitis and vascular disorders of the Japanese Dermatological Association. We sent a questionnaire to 12 dermatological facilities identified through the revised Committee for guidelines for the management of vasculitis and vascular disorders of the Japanese Dermatological Association. Retrospective data obtained from 84 patients at the 12 dermatological facilities between 2012 January 2016 December were evaluated. The 84 patients were categorized into two groups, a systemic steroid treatment group (group 1, n = 52) and a no systemic steroid treatment group (group 2, n = 32). C-reactive protein in group 1 patients was significantly higher than that in group 2 patients. Frequency of fever, arthritis, myalgia- and peripheral neuropathy in group 1 was significantly higher than that in group 2. We propose that these symptoms could serve as early markers for the transfer from cutaneous arteritis to systemic polyarteritis nodosa. We further suggest that patients who are subsequently associated with cerebral hemorrhage and infarction, who are originally diagnosed as having cutaneous arteritis, could progress to systemic polyarteritis nodosa. The study demonstrated that it is important for dermatologists to detect these findings early in order to establish an accurate diagnosis and a timely treatment.


Assuntos
Dermatologistas/estatística & dados numéricos , Poliarterite Nodosa/etiologia , Pele/patologia , Adulto , Biomarcadores/análise , Proteína C-Reativa/análise , Feminino , Seguimentos , Glucocorticoides/uso terapêutico , Humanos , Japão , Masculino , Pessoa de Meia-Idade , Poliarterite Nodosa/sangue , Poliarterite Nodosa/tratamento farmacológico , Poliarterite Nodosa/patologia , Estudos Retrospectivos , Pele/irrigação sanguínea , Inquéritos e Questionários/estatística & dados numéricos
9.
J Am Acad Dermatol ; 83(2): 501-508, 2020 Aug.
Artigo em Inglês | MEDLINE | ID: mdl-32044177

RESUMO

BACKGROUND: Lymphocytic thrombophilic arteritis (LTA), or macular lymphocytic arteritis, is defined by a primary lymphocytic vasculitis. However, the nosology of LTA has been controversial, with speculation that it may represent an indolent non-nodule-forming variant of cutaneous polyarteritis nodosa (cPAN). OBJECTIVE: This study compares the clinicopathologic features of patients with LTA or cPAN to assess if these conditions should be considered distinct entities. METHODS: This is a cross-sectional study of all LTA and cPAN cases at a single tertiary center using prospectively collected clinical data and blinded histologic assessment. RESULTS: The study included 17 patients with LTA and 13 patients with cPAN. Clinically, cases of LTA were distinguished by a more widespread pattern of livedo racemosa, which was noninfiltrated and asymptomatic. In contrast, cPAN was associated with localized starburst livedo, purpura, and episodic features including nodules, pain, and large inflammatory ulcers. When patients were separated according to the presence (>5%) or paucity (≤5%) of neutrophils on blinded histology review, they had distinct clinical features and differences in disease course. LIMITATIONS: This was a single-center study. CONCLUSION: Our data support the classification of LTA and cPAN as separate entities rather than a spectrum of the same disorder and highlight the importance of clinicopathologic correlation in distinguishing these conditions.


Assuntos
Arterite/diagnóstico , Linfócitos/patologia , Poliarterite Nodosa/diagnóstico , Pele/patologia , Trombofilia/diagnóstico , Adulto , Arterite/sangue , Arterite/complicações , Arterite/patologia , Estudos Transversais , Diagnóstico Diferencial , Progressão da Doença , Feminino , Humanos , Livedo Reticular/etiologia , Masculino , Pessoa de Meia-Idade , Neutrófilos/patologia , Poliarterite Nodosa/complicações , Poliarterite Nodosa/patologia , Estudos Prospectivos , Púrpura/etiologia , Pele/irrigação sanguínea , Pele/citologia , Trombofilia/sangue , Trombofilia/complicações , Trombofilia/patologia , Adulto Jovem
10.
An Bras Dermatol ; 95(1): 32-39, 2020.
Artigo em Inglês | MEDLINE | ID: mdl-31889596

RESUMO

BACKGROUND: Macular lymphocytic arteritis most commonly presents as hyperpigmented macules on the lower limbs. The pathogenesis of this disease is still unclear and there is an ongoing debate regarding whether it represents a new form of cutaneous vasculitis or an indolent form of cutaneous polyarteritis nodosa. OBJECTIVE: To describe clinical, histopathological, and laboratory findings of patients with the diagnosis of macular lymphocytic arteritis. METHODS: A retrospective search was conducted by reviewing cases followed at the Vasculitis Clinic of the Dermatology Department, School of Medicine, University of São Paulo, between 2005 and 2017. Seven patients were included. RESULTS: All cases were female, aged 9-46 years, and had hyperpigmented macules mainly on the legs. Three patients reported symptoms. Skin biopsies evidencing a predominantly lymphocytic infiltrate affecting arterioles at the dermal subcutaneous junction were found, as well as a typical luminal fibrin ring. None of the patients developed necrotic ulcers, neurological damage, or systemic manifestations. The follow-up ranged from 18 to 151 months, with a mean duration of 79 months. STUDY LIMITATIONS: This study is subject to a number of limitations: small sample of patients, besides having a retrospective and uncontrolled study design. CONCLUSIONS: To the best of the authors' knowledge, this series presents the longest duration of follow-up reported to date. During this period, none of the patients showed resolution of the lesions despite treatment, nor did any progress to systemic vasculitis. Similarities between clinical and skin biopsy findings support the hypothesis that macular lymphocytic arteritis is a benign, incomplete, and less aggressive form of cutaneous polyarteritis nodosa.


Assuntos
Arterite/patologia , Dermatopatias Vasculares/patologia , Adolescente , Adulto , Biópsia , Criança , Feminino , Seguimentos , Humanos , Hiperpigmentação/patologia , Imuno-Histoquímica , Linfócitos/patologia , Pessoa de Meia-Idade , Poliarterite Nodosa/patologia , Estudos Retrospectivos , Adulto Jovem
11.
Vet Pathol ; 57(2): 281-285, 2020 03.
Artigo em Inglês | MEDLINE | ID: mdl-31763954

RESUMO

This report presents a novel canine condition in 32 dogs in which aberrant migration of Spirocerca lupi larvae through mesenteric arteries, instead of gastric arteries, led to small or large intestinal infarction. This form of spirocercosis was first recognized in Israel in 2013 and is currently ongoing. Typical clinical signs were anorexia and weakness of 3 to 4 days and, less frequently, vomiting and diarrhea, followed by collapse, bloody diarrhea, and severe vomiting. Exploratory laparotomy showed 1 or more infarcted and often perforated intestinal segments in all cases. Microscopically, there was intestinal mucosal to transmural coagulative necrosis and mesenteric multifocal necrotizing eosinophilic arteritis, thrombosis, hemorrhage, and early fibroplasia. Third-stage S. lupi larvae were identified by morphologic features in 9 of 32 (28%) cases, and the species was confirmed by polymerase chain reaction in 4 cases. Nearly 50% of the dogs had been receiving prophylactic therapy, which did not prevent this form of spirocercosis.


Assuntos
Doenças do Cão/parasitologia , Hemorragia/veterinária , Infarto/veterinária , Poliarterite Nodosa/veterinária , Infecções por Spirurida/veterinária , Thelazioidea/isolamento & purificação , Trombose/veterinária , Animais , Doenças do Cão/patologia , Cães , Feminino , Hemorragia/parasitologia , Hemorragia/patologia , Infarto/parasitologia , Infarto/patologia , Intestinos/parasitologia , Intestinos/patologia , Israel , Larva , Masculino , Artérias Mesentéricas/parasitologia , Artérias Mesentéricas/patologia , Poliarterite Nodosa/parasitologia , Poliarterite Nodosa/patologia , Reação em Cadeia da Polimerase/veterinária , Infecções por Spirurida/parasitologia , Infecções por Spirurida/patologia , Thelazioidea/genética , Trombose/parasitologia , Trombose/patologia
12.
Rev Colomb Psiquiatr (Engl Ed) ; 48(4): 261-265, 2019.
Artigo em Inglês, Espanhol | MEDLINE | ID: mdl-31779877

RESUMO

INTRODUCTION: Excoriation (skin picking) disorder is included in the DSM-5 in the obsessive compulsive and related disorders category. It is defined as the recurrent urge to touch, scratch, scrape, scrub, rub, squeeze, bite or dig in the skin, leading to skin lesions. It is a rare disorder (1.4-5.4% of the population) and occurs mainly in women. CASE REPORT: this article reports the case of a 31-year-old female patient, initially assessed by dermatology and orthopaedics for the presence of infected ulcerated lesions on her lower limbs, with other superficial lesions from scratching on her chest, arms, forearms, back and head. The patient also reported symptoms of anxiety, so was assessed by consultation-liaison psychiatry. DISCUSSION: skin picking, normal behaviour in mammals, becomes pathological from a psychiatric point of view when it is repetitive and persistent, as in the case of excoriation disorder. In view of the reported relationship with the obsessive-compulsive spectrum, use of selective serotonin reuptake inhibitors and cognitive behavioural therapy are recommended.


Assuntos
Transtorno Obsessivo-Compulsivo/diagnóstico , Poliarterite Nodosa/patologia , Comportamento Autodestrutivo/diagnóstico , Dermatopatias/diagnóstico , Adulto , Ansiedade/psicologia , Feminino , Humanos , Transtorno Obsessivo-Compulsivo/patologia , Transtorno Obsessivo-Compulsivo/terapia , Comportamento Autodestrutivo/patologia , Dermatopatias/patologia , Dermatopatias/terapia
13.
Adv Rheumatol ; 59(1): 35, 2019 08 01.
Artigo em Inglês | MEDLINE | ID: mdl-31370889

RESUMO

The panniculitides remain as one of the most challenging areas for clinicians, as they comprise a heterogeneous group of inflammatory diseases involving the subcutaneous fat with potentially-shared clinical and histopathological features. Clinically, most panniculitides present as red edematous nodules or plaques. Therefore, in addition to a detailed clinical history, a large scalpel biopsy of a recent-stage lesion with adequate representation of the subcutaneous tissue is essential to specific diagnosis and appropriate clinical management. Herein we review the panniculitides of particular interest to the rheumatologist.


Assuntos
Paniculite/patologia , Reumatologistas , Síndrome de Behçet/patologia , Biópsia/métodos , Eritema Endurado/patologia , Eritema Nodoso/patologia , Etanercepte/efeitos adversos , Humanos , Imunossupressores/efeitos adversos , Infecções/patologia , Paniculite/classificação , Paniculite de Lúpus Eritematoso/patologia , Poliarterite Nodosa/patologia , Gordura Subcutânea/patologia , Tela Subcutânea/patologia , Vasculite/patologia
14.
J Med Case Rep ; 13(1): 236, 2019 Jul 31.
Artigo em Inglês | MEDLINE | ID: mdl-31362782

RESUMO

BACKGROUND: Polyarteritis nodosa is a small vessel to medium vessel vasculitis that frequently presents with multi-organ involvement, but can sometimes be limited to single organs such as the testes. Patients often require treatment with glucocorticoids, plus or minus additional immunosuppressive therapy depending on the severity of the disease. We describe a rare case of polyarteritis nodosa involving the right testis and urinary bladder without other systemic features of vasculitis. CASE PRESENTATION: A previously healthy 54-year-old First Nations Canadian man presented with intermittent gross hematuria. He underwent surgical excision of his right testis for cryptorchidism and a transurethral resection of a bladder mass. Histology showed an active medium vessel vasculitis in both organs. On extensive clinical, laboratory, and radiographic review, he had no systemic features of vasculitis. On 2-year follow-up, he has not required any systemic therapy and has not developed further symptoms. CONCLUSION: Single organ polyarteritis nodosa can sometimes be managed with surgical excision of the involved organ alone. Although our patient had two organs involved, we extrapolated the results of our literature search to guide his care. This case highlights the potential for surgical excision to cure polyarteritis nodosa despite the involvement of two organs in the absence of symptoms and signs of systemic vasculitis.


Assuntos
Poliarterite Nodosa/cirurgia , Testículo/cirurgia , Bexiga Urinária/cirurgia , Criptorquidismo/complicações , Humanos , Masculino , Pessoa de Meia-Idade , Orquiectomia , Poliarterite Nodosa/patologia , Doenças da Bexiga Urinária/patologia , Doenças da Bexiga Urinária/cirurgia
15.
J Am Podiatr Med Assoc ; 109(3): 241-245, 2019 May.
Artigo em Inglês | MEDLINE | ID: mdl-31268784

RESUMO

Polyarteritis nodosa is a progressive, often life-threatening, vasculitis affecting multiple organs, including the skin and peripheral nerves. We report a patient presenting with systemic features of the disease and with characteristic lesions in the feet 3 weeks after vaccination against hepatitis B virus infection.


Assuntos
Vacinas contra Hepatite B/efeitos adversos , Hepatite B/prevenção & controle , Poliarterite Nodosa/etiologia , Lobo Temporal/diagnóstico por imagem , Adulto , Humanos , Imageamento por Ressonância Magnética , Masculino , Poliarterite Nodosa/diagnóstico , Poliarterite Nodosa/patologia , Lobo Temporal/patologia , Dedos do Pé/patologia
16.
J Wound Care ; 28(Sup6): S1-S92, 2019 06 01.
Artigo em Inglês | MEDLINE | ID: mdl-31169055

Assuntos
Ferimentos e Lesões/terapia , Vasculite Associada a Anticorpo Anticitoplasma de Neutrófilos/complicações , Vasculite Associada a Anticorpo Anticitoplasma de Neutrófilos/diagnóstico , Vasculite Associada a Anticorpo Anticitoplasma de Neutrófilos/patologia , Vasculite Associada a Anticorpo Anticitoplasma de Neutrófilos/terapia , Transtornos da Coagulação Sanguínea/complicações , Transtornos da Coagulação Sanguínea/diagnóstico , Transtornos da Coagulação Sanguínea/patologia , Transtornos da Coagulação Sanguínea/terapia , Calciofilaxia/complicações , Calciofilaxia/diagnóstico , Calciofilaxia/patologia , Calciofilaxia/terapia , Eritema Endurado/complicações , Eritema Endurado/diagnóstico , Eritema Endurado/patologia , Eritema Endurado/terapia , Transtornos Autoinduzidos/complicações , Transtornos Autoinduzidos/diagnóstico , Transtornos Autoinduzidos/terapia , Hidradenite Supurativa/complicações , Hidradenite Supurativa/diagnóstico , Hidradenite Supurativa/patologia , Hidradenite Supurativa/terapia , Humanos , /diagnóstico , /terapia , Livedo Reticular/complicações , Livedo Reticular/diagnóstico , Livedo Reticular/patologia , Livedo Reticular/terapia , Equipe de Assistência ao Paciente , Poliarterite Nodosa/complicações , Poliarterite Nodosa/diagnóstico , Poliarterite Nodosa/patologia , Poliarterite Nodosa/terapia , Pioderma Gangrenoso/complicações , Pioderma Gangrenoso/diagnóstico , Pioderma Gangrenoso/patologia , Pioderma Gangrenoso/terapia , Neoplasias Cutâneas/diagnóstico , Neoplasias Cutâneas/patologia , Neoplasias Cutâneas/secundário , Neoplasias Cutâneas/terapia , Vasculite/complicações , Vasculite/diagnóstico , Vasculite/patologia , Vasculite/terapia , Ferimentos e Lesões/diagnóstico
17.
Neurol Clin ; 37(2): 345-357, 2019 05.
Artigo em Inglês | MEDLINE | ID: mdl-30952413

RESUMO

Polyarteritis nodosa (PAN) is a necrotizing vasculitis affecting medium-sized vessels whose main manifestations are weight loss, fever, peripheral neuropathy, renal, musculoskeletal, gastrointestinal tract and/or cutaneous involvement(s), hypertension and/or cardiac failure. Peripheral neuropathy is one of the most frequent and earliest symptoms, affecting 50% to 75% of PAN patients. Central nervous system involvement affects only 2% to 10% of PAN patients, often late during the disease course. Treatment relies on combining corticosteroids and an immunosuppressant (mainly cyclophosphamide) in patients with poor prognoses. In patients with hepatitis B virus-related PAN, plasma exchanges and antiviral drugs should be combined with corticosteroids.


Assuntos
Poliarterite Nodosa/complicações , Poliarterite Nodosa/patologia , Doenças do Sistema Nervoso Central/etiologia , Humanos , Masculino , Doenças do Sistema Nervoso Periférico/etiologia , Poliarterite Nodosa/diagnóstico
18.
Rheumatol Int ; 39(8): 1447-1455, 2019 Aug.
Artigo em Inglês | MEDLINE | ID: mdl-30874872

RESUMO

Systemic vasculitis involving the breast is a rare clinical condition and may mimic breast cancer or mastitis clinically or radiographically. Here, we report a case of polyarteritis nodosa (PAN) with breast involvement and perform a literature review of published cases of systemic vasculitis affecting the breast to better understand this disorder. We report a case of PAN affecting the right breast in a young woman. A retrospective review was performed by searching Medline, Embase, Web of Science, the Cochrane Library, and Scopus for cases of systemic vasculitis involving the breast written in English up to June 1st, 2018. A 27-year-old woman presented with a painful mass in the right breast was diagnosed as PAN by the biopsy. She was treated with prednisone and methotrexate for 6 months, at which time her condition had stabilized and inflammatory markers had normalized. A total of 66 cases were identified, with granulomatosis with polyangiitis (GPA), giant cell arteritis (GCA), and PAN as the main types. The typical manifestation was mass (79.2%, 53/67) in the breast, and all diagnoses were made by the pathology of the breast biopsy. Glucocorticoid and immunosuppressant were the main therapies, and 74.6% (50/67) patients achieved remission during follow-up. Our case and a literature review of 66 cases of systemic vasculitis involving the breast reveal the importance of tissue biopsy to obtain a definitive diagnosis, because the vasculitis subtype strongly influences prognosis.


Assuntos
Doenças Mamárias , Poliarterite Nodosa , Adulto , Biópsia , Doenças Mamárias/tratamento farmacológico , Doenças Mamárias/imunologia , Doenças Mamárias/patologia , Diagnóstico Diferencial , Quimioterapia Combinada , Feminino , Glucocorticoides/uso terapêutico , Humanos , Imunossupressores/uso terapêutico , Metotrexato/uso terapêutico , Poliarterite Nodosa/tratamento farmacológico , Poliarterite Nodosa/imunologia , Poliarterite Nodosa/patologia , Valor Preditivo dos Testes , Prednisona/uso terapêutico , Indução de Remissão , Resultado do Tratamento
19.
Forensic Sci Med Pathol ; 15(2): 252-257, 2019 Jun.
Artigo em Inglês | MEDLINE | ID: mdl-30810977

RESUMO

Coronary arteritis is an uncommon cause of sudden death in non-atherosclerotic coronary diseases, and is mostly associated with systemic vasculitis or systemic autoimmune diseases; therefore, sudden death due to isolated coronary arteritis rarely occurs. The case described in this report is that of a 34-year-old man with no significant personal medical history who died suddenly after presenting with nausea. Postmortem examination revealed a significant infiltration of lymphocytes predominantly on the adventitia and periadventitial tissues of the coronary arteries in the epicardium. The lymphocytic infiltrate partially extended to the thickened intima with fibrosis, destructing the media and internal elastic lamina, and the lumen was occluded by a thrombus in the left main stem and left anterior descending branch. The arterial walls exhibited focal fibrinoid necrosis with regression in the intima and fibrous scars with angiogenesis in the media and adventitia. Focal myocardial infarction was detected in the left ventricle as a fibrotic change of the myocardium. No findings associated with vasculitis were discerned in the aorta, other peripheral arteries, or major organs. Laboratory tests of postmortem blood samples returned negative results for antinuclear antibodies, cryoglobulin, immunoglobulin G4, and cytoplasmic anti-neutrophil cytoplasmic antibodies for myeloperoxidase and proteinase 3. These autopsy findings suggest that the sudden death was caused by isolated necrotizing vasculitis that is assumed to be polyarteritis nodosa localized at the coronary arteries. However, pathological characteristics may not be exactly the same between isolated necrotizing vasculitis in the coronary arteries and polyarteritis nodosa.


Assuntos
Trombose Coronária/patologia , Vasos Coronários/patologia , Morte Súbita Cardíaca/etiologia , Poliarterite Nodosa/patologia , Adulto , Humanos , Linfócitos/patologia , Masculino , Infarto do Miocárdio/patologia , Túnica Íntima/patologia , Túnica Média/patologia
20.
J Int Adv Otol ; 15(1): 156-159, 2019 Apr.
Artigo em Inglês | MEDLINE | ID: mdl-30541729

RESUMO

Polyarteritis nodosa (PAN) is a systemic vasculitis affecting the small- and medium-sized arteries that may present with hearing impairment. In rare cases, PAN may be associated with progressive labyrinthitis ossificans (LO), an otologic emergency requiring early cochlear implantation (CI) to restore hearing before the complete, irreversible cochlear ossification. We report the first case in the literature of a patient affected by PAN with bilateral sudden sensorineural hearing loss and rapid LO who underwent "emergency" bilateral simultaneous CI. This case report emphasizes the importance of an early audiological evaluation in patients with PAN when LO is suspected. Multidisciplinary approach is mandatory when facing organ-specific manifestations in patients with PAN. Detailed discussion is provided with particular regard to clinical and radiological presentation as well as CI outcomes in such a rare and challenging case.


Assuntos
Cóclea/transplante , Labirintite/cirurgia , Poliarterite Nodosa/complicações , Cóclea/diagnóstico por imagem , Cóclea/patologia , Cóclea/cirurgia , Implante Coclear/métodos , Implantes Cocleares/normas , Feminino , Fibrose , Perda Auditiva Neurossensorial/diagnóstico , Perda Auditiva Neurossensorial/etiologia , Humanos , Labirintite/etiologia , Labirintite/patologia , Livedo Reticular/diagnóstico , Livedo Reticular/etiologia , Pessoa de Meia-Idade , Poliarterite Nodosa/patologia , Janela da Cóclea/patologia , Resultado do Tratamento , Vestíbulo do Labirinto/patologia , Vestíbulo do Labirinto/cirurgia
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