Fatal Subacute Cytomegalovirus Encephalitis Associated With Hypogammaglobulinemia and Thymoma

Cucchiara, Brett; Forman, Mark S.; McGarvey, Michael L.; Kasner, Scott E.; King, David C.

doi:10.4065/78.2.223

Cited by 25 publications

(14 citation statements)

References 23 publications

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“…In this case, no paraneoplastic syndromes were observed and even the weak positive ANA test was considered non‐specific and inconclusive as no obvious compatible clinical signs were observed. In agreement with our observations, in human pathology, thymic carcinomas are very rarely associated with myasthenia gravis or other immune‐mediated systemic diseases when compared with their benign counterpart (Tarr and others 2001, Cucchiara and others 2003, Montella and others 2003).…”

Section: Discussionsupporting

confidence: 92%

Ectopic cervical thymic carcinoma in a dog

Faísca

Henriques

Dias³

et al. 2011

Journal of Small Animal Practice

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A 10-year-old male German shepherd dog was referred for evaluation of a cranial cervical mass causing progressively worsening respiratory distress. A fine-needle aspirate of the mass was obtained and the cytology results were compatible with a carcinoma. The dog underwent chemotherapy without clinical improvement and was ultimately euthanased because of clinical deterioration. At post-mortem examination, an irregular multi-lobated mass in the cranial cervical region was observed causing ventro-lateral tracheal deviation. Histopathology and immunohistochemistry revealed a mixed population of CD3 lymphocytes and macrophages in an exuberant fibrous stroma, associated with dispersed cytokeratin-positive epithelial cells with marked eosinophilic cytoplasm. Some of the epithelial cells were arranged in concentric clusters that were interpreted as Hassall's corpuscles. Histopathological examination of the thyroid gland revealed several neoplastic emboli composed of epithelial cells similar to those observed in the cervical mass. To the authors' knowledge, this is the first report of an ectopic cervical thymic carcinoma in a dog.

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Section: Discussionsupporting

confidence: 92%

Ectopic cervical thymic carcinoma in a dog

Faísca

Henriques

Dias³

et al. 2011

Journal of Small Animal Practice

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“…Whether thymoma resection should be performed for GS patients is still controversial. Although it can relieve the local symptoms caused by thymoma compression and prevent the local infiltration and distant metastasis of thymoma, there are no reported cases of reversal of immunodeficiency after thymoma resection [ 17 ]. Our patient underwent thymectomy many years ago, which seemed to confirm that the operation did not improve immune function.…”

Section: Discussionmentioning

confidence: 99%

Case report of Ureaplasma urealyticum meningitis in a patient with thymoma and hypogammaglobulinaemia

Zhang

Hou

et al. 2021

BMC Infect Dis

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Background Ureaplasma urealyticum (UU) is found among the normal vaginal flora in a considerable proportion of asymptomatic women; however, adult central nervous system (CNS) infection of UU is extremely rare. Good's syndrome (GS) is an adult-onset immunodeficiency characterized by thymoma, hypogammaglobulinaemia, low or absent B‑cells, and an inverted CD4+/CD8+ T‑cell ratio. Patients with GS usually have severe or recurrent infections. Case presentation We describe the case report of a 49-year-old woman who developed UU meningitis. Initial routine anti-viral and anti-bacterial therapy showed no improvement in the patient's condition. Next-generation sequencing (NGS) of cerebrospinal fluid (CSF) identified the UU DNA sequence. Accordingly, a diagnosis of UU meningitis was made, and minocycline therapy was initiated. The patient responded favourably, with no signs of disease at subsequent follow-up. According to the severity and rarity of the case, secondary immunodeficiency was suspected. Flow cytometry found hypogammaglobulinaemia. Combined with the previous history of thymoma, the patient was diagnosed with immune deficiency disease of GS. Conclusions This case may be the first adult case report in the literature describing UU meningitis in a patient with GS. The diagnosis of GS should be considered in patients presenting with unexplained antibody deficiency and thymoma.

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“…2 Encephalitis is very uncommon in GS, and only three patients with CMV encephalitis have been reported. 4,5 On the other hand, the association between thymoma and PML is very unusual, with only two cases described. 7,8 This index case represents the first reported association between GS and PML.…”

Section: Discussionmentioning

confidence: 99%

“…The humoral and cellular immunodeficiency in GS is responsible for recurrent respiratory, gastrointestinal, and urinary infections from common and opportunistic bacterial and fungal pathogens; 2,3 40% of patients develop opportunistic viral infections, and three patients with cytomegalovirus encephalitis have been reported. 4,5 PML, an infection caused by JC virus (JCV), usually occurs in patients infected with HIV, or in patients on treatment with immunosuppressive or immunomodulatory drugs. Herein we describe the first unusual association between GS and PML.…”

Section: Introductionmentioning

confidence: 99%

Progressive multifocal leukoencephalopathy in a patient with Good's syndrome

Squintani¹,

Ferrari

Bazzoli

et al. 2010

International Journal of Infectious Diseases

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Good's syndrome (GS) is an immunodeficiency characterized by thymoma, hypogammaglobulinemia, and impaired T-cell function. The clinical manifestations of GS include recurrent or chronic infections from common or opportunistic pathogens. Encephalitis is a rare event, with only anecdotal reports of cytomegalovirus infection. Herein we report the case of a 79-year-old woman with GS who developed subacute motor deficits and cognitive changes. Magnetic resonance imaging (MRI) of the brain disclosed white- and gray-matter lesions, mostly in the right frontal and parietal areas. Polyoma virus JC, the agent of progressive multifocal encephalopathy (PML), was identified in cerebrospinal fluid samples and brain biopsy specimens. After diagnosis, the disease had a rapid fatal course. The present case represents the first reported association between GS and PML.

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Fatal Subacute Cytomegalovirus Encephalitis Associated With Hypogammaglobulinemia and Thymoma

Cited by 25 publications

References 23 publications

Ectopic cervical thymic carcinoma in a dog

Ectopic cervical thymic carcinoma in a dog

Case report of Ureaplasma urealyticum meningitis in a patient with thymoma and hypogammaglobulinaemia

Progressive multifocal leukoencephalopathy in a patient with Good's syndrome

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