Dataset Information

Treatment of Severe Japanese Encephalitis Complicated With Hashimoto's Thyroiditis and Guillain-Barre Syndrome With Protein A Immunoadsorption: A Case Report.

ABSTRACT: A severely comatose female patient was diagnosed with Japanese encephalitis (JE). Her condition was complicated by Hashimoto's thyroiditis (HT) and Guillain-Barré syndrome (GBS). After antiviral, glucocorticoid, and immunoglobulin treatment, the patient's consciousness was restored, and she could breathe spontaneously. Following this, new-onset, primarily demyelinating GBS developed, which progressed to demyelination combined with axonal injury. The patient was switched to protein A immunoadsorption (PAIA) therapy, and her Hughes score decreased rapidly, from 4 to 1 after 6 months. This patient is the first to receive PAIA combined with an antiviral-glucocorticoid-immunoglobulin regimen to treat encephalitis, meningitis, HT, and GBS caused by JE infection, thereby reflecting the importance of clinical application of PAIA in the treatment of immunological complications of JE.

SUBMITTER: Zang Q

PROVIDER: S-EPMC8777188 | biostudies-literature | 2021

REPOSITORIES: biostudies-literature

ACCESS DATA

Publications

Treatment of Severe Japanese Encephalitis Complicated With Hashimoto's Thyroiditis and Guillain-Barré Syndrome With Protein A Immunoadsorption: A Case Report.

Zang Qiuling Q Wang Yating Y Guo Junshuang J Long Liyang L Zhang Shuyu S Cui Can C Song Dandan D Yu Boguang B Tang Fenlan F Teng Junfang J Miao Wang W

Frontiers in immunology 20220107

A severely comatose female patient was diagnosed with Japanese encephalitis (JE). Her condition was complicated by Hashimoto's thyroiditis (HT) and Guillain-Barré syndrome (GBS). After antiviral, glucocorticoid, and immunoglobulin treatment, the patient's consciousness was restored, and she could breathe spontaneously. Following this, new-onset, primarily demyelinating GBS developed, which progressed to demyelination combined with axonal injury. The patient was switched to protein A immunoadsorp ...[more]

PMID: 35069593

Similar Datasets

Project description:BACKGROUND:Thyroid-associated orbitopathy (TAO) constitutes an immune-mediated inflammation of the orbital tissues of unclear etiopathogenesis. TAO is most prevalent in hyperthyroid patients with Graves' disease (GD); however, severe cases of orbitopathy associated with Hashimoto's thyroiditis (HT) have rarely been described. CASE PRESENTATION:Herewith we report an unusual case of a middle-aged clinically and biochemically euthyroid woman with a stable HT, who developed a severe unilateral left-sided TAO. Thyrotropin receptor antibodies (TRAb) concentration was negative. Intraocular pressure in the left eye was mildly elevated (24?mmHg), while vision acuity was not compromised. Abnormal positioning of the eyeball suggested the extraocular muscles involvement. Unilaterally, von Graefe's, Stellwag's, Kocher's and Moebius' signs were positive. Conjunctival erythema, redness and edema of the eyelid and an enlarged, swollen lacrimal caruncle were visible. She received 4/7 points in the Clinical Activity Scale (CAS) and class IV in the NO SPECS severity scale for the left eye (I-0, II-a, III-0, IV-b, V-0, VI-0). Magnetic resonance imaging (MRI) revealed thickening of the left medial rectus muscle with an increase in T2 signal intensity and prolonged T2 relaxation indicating an active form of TAO. The patient received therapy with glucocorticosteroids intravenously, followed by intramuscular injections with a cumulative dose of 3.24?g of methylprednisolone during a 9-week period with good tolerance. The applied therapy, combined with adequate L-thyroxine substitution, as well as vitamin D and selenium supplementation, resulted in a complete remission of ophthalmic symptoms. CONCLUSIONS:Unilateral exophthalmos in TRAb-negative patients with HT is not a typical manifestation of the disease, and requires a wider differential diagnosis with MRI of the orbits. Scheme of three iv. pulses of methylprednisolone intravenously and the continuation of treatment with im. injections seems to be an effective and safe method of treatment in this group of patients. What is more, adequate vitamin D supplementation and the maintenance of biochemical euthyroidism may help to achieve an ultimate therapeutic effect. Patients with TAO in the course of HT need a careful and continued interdisciplinary approach both ophthalmological and endocrinological. Further studies are needed to elucidate the etiopathogenesis of TAO in TRAb-negative patients.

Project description:BackgroundMalaria is an infectious disease caused by Plasmodium parasites, transmitted to humans by infected female Anopheles mosquitoes. Five Plasmodium species infect humans: P. vivax, P. falciparum, P. ovale, P. malariae, and P. knowlesi. Guillain-Barré Syndrome (GBS) is an inflammatory condition that can lead to paralysis, autonomic dysfunction, respiratory failure, and sensory symptoms. GBS typically follows an infection with Campylobacter bacteria, commonly found in undercooked poultry, but is rarely associated with malaria.Clinical presentationA 16-year-old female patient presented to our emergency department with a 1-day history of altered mentation. She had experienced a severe global headache and fever for 3 days prior to presentation. The patient tested positive for falciparum malaria and was admitted to the ward, where she received IV artesunate and other supportive management. After 3 days of admission, she noticed weakness and numbness in her lower extremities. Subsequently, the weakness progressed upward to involve her upper extremities. After extensive workup, the patient was managed with consideration of Guillain-Barré Syndrome (GBS), and she made a complete recovery after 12 weeks.DiscussionGuillain-Barré Syndrome (GBS) is an acute paralytic illness often triggered by infections, particularly viral ones. It is the leading cause of sudden muscle weakness, typically following respiratory or gastrointestinal infections, with Campylobacter jejuni being the most common cause. This patient's neurological symptoms pointed to paralysis of the lower motor neurons. Guillain-Barré Syndrome is also suggested by elevated protein levels and a lack of cells in the cerebrospinal fluid. This clinical picture emerged following a Plasmodium falciparum infection. Although the specific subtype (demyelinating or axonal) was not determined in this case due to the absence of a nerve conduction study, demyelinating subtypes have been found in GBS following Plasmodium infection.ConclusionIn conclusion, while malaria is an exceptionally rare cause of Guillain-Barré Syndrome (GBS), it should be considered in patients with recent malaria infection who present with symptoms of lower motor neuron lesions.

Project description:BackgroundInflammatory myofibroblastic tumors (IMTs) of the thyroid are extremely rare soft-tissue tumors. In the literature, IMTs are sometimes called plasma cell granulomas (PCGs) or inflammatory pseudotumors, which often causes ambiguity. To date, 17 cases of PCGs and five cases of thyroid IMTs have been reported. These cases reveal that IMTs of the thyroid are often negative for the anaplastic lymphoma kinase (ALK-1) gene. To provide further information on this rare lesion, we present a case of an ALK-1-positive thyroid IMT and a review of IMTs of the thyroid.Case presentationA 34-year-old Chinese woman presented with a painless neck mass that had persisted for over a month. Ultrasonography revealed a 4.28 × 2.53 cm2 hypoechoic mass, in the left lobe of the thyroid gland. Serum levels of thyroglobulin and anti-thyroglobulin antibodies were high. Subsequently, left lobectomy was performed. Macroscopically, the lesion was a gray-brown nodular mass with a partial envelope. Histologically, two different lesion types were observed. The first lesion showed classic spindle cell proliferation, with spindle cells arranged in fascicles, accompanied by mature inflammatory cells. The other lesion showed a large number of infiltrating lymphocytes, with lymphoid follicles in the remaining thyroid gland, which was atrophic. Immunohistochemical staining showed that the spindle cells were negative for CK19, CyclinD1, Gelectin-3, EMA, CD34, S100, Bcl-2, and STAT-6, but strongly positive for ALK-1, vimentin, and TTF1. CK was focally expressed, and the Ki-67 index was 5%. A diagnosis of IMT was proposed according to immunohistochemical findings and morphology. Hashimoto's thyroiditis was confirmed according to serum levels of thyroglobulin and anti-thyroglobulin antibodies and morphology. The patient did not receive adjuvant therapy. She remained alive without disease recurrence for 10 months after lobectomy.ConclusionsIMTs should be considered in the diagnosis when spindle cell proliferation accompanied by mature inflammatory cells is observed, spindle cells are mildly atypical, and myofibroblast differentiation is present in the thyroid. A uniform diagnostic term is crucial to avoid ambiguity. Clinicians and pathologists should be aware of the necessity for long-term follow-up, especially in ALK-positive cases. The therapeutic potential of ALK-1 positivity should be explored further.

Dataset Information

Treatment of Severe Japanese Encephalitis Complicated With Hashimoto's Thyroiditis and Guillain-Barre Syndrome With Protein A Immunoadsorption: A Case Report.

Publications

Treatment of Severe Japanese Encephalitis Complicated With Hashimoto's Thyroiditis and Guillain-Barré Syndrome With Protein A Immunoadsorption: A Case Report.

Similar Datasets

OmicsDI is part of the ELIXIR infrastructure

Tweets