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AJP Reports Apr 2024Fetal hyperthyroidism is a rare prenatal disease and can be life-threatening. The diagnosis is based on ultrasound in mothers with a history of Basedow-Graves' disease...
Fetal hyperthyroidism is a rare prenatal disease and can be life-threatening. The diagnosis is based on ultrasound in mothers with a history of Basedow-Graves' disease and elevation of thyrotropin receptor antibodies (TRAbs) levels. The treatment consists of antithyroid drugs. We present a mother with Basedow-Graves' disease, treated with radioactive iodine 16 years ago. She had an unplanned pregnancy at the age of 29 years, and an elevation of TRAbs (21 U/L) was found at the sixth week of pregnancy. At 22 weeks of gestation, fetal ultrasound displayed tachycardia, goiter, exophthalmos, and suspicion of craniosynostosis, hence methimazole was started. Concomitantly, suppressed maternal thyroid-stimulating hormone (TSH) was found. Her daughter was born at 33 + 6 weeks showing clinical and laboratory findings of hyperthyroidism. Consequently, treatment with methimazole was prescribed. Normal thyroid function was documented in the mother after giving birth. Clear explanation has not been found for the alteration of maternal TSH during pregnancy.
PubMed: 38827649
DOI: 10.1055/a-2317-9431 -
Clinical Medicine Insights. Case Reports 2024Infratemporal fossa (ITF) tumors are rare in children and may present with a variety of symptoms. Teratomas are neoplasms derived from the 3 germ layers and...
Infratemporal fossa (ITF) tumors are rare in children and may present with a variety of symptoms. Teratomas are neoplasms derived from the 3 germ layers and approximately 6% to 10% are within the head and neck. Our study discusses one of the first reported cases of teratoma in the ITF in a pediatric patient. A 3-year-old girl presents with 2 years of recurrent monthly left periorbital swelling accompanied by fevers, skin discoloration, and pain. Prior episodes were treated with antibiotics with incomplete resolution. Imaging revealed a cystic lesion centered in the ITF. She was taken for endoscopic endonasal biopsy of the lesion and had no complications. Pathology revealed a mature teratoma composed primarily of pancreatic tissue. Providers should consider masses such as teratoma in the differential for ITF tumors and periorbital edema unresponsive to typical treatment.
PubMed: 38827640
DOI: 10.1177/11795476241255563 -
Cureus May 2024Periorbital necrotizing fasciitis (NF) is a devastating bacterial infection associated with irreversible inflammatory destruction of soft tissues. Outcomes include...
Periorbital necrotizing fasciitis (NF) is a devastating bacterial infection associated with irreversible inflammatory destruction of soft tissues. Outcomes include disfigurement, vision loss, septic shock, and death within hours to days. We describe two cases of periorbital NF that presented to our unit within a three-month period. We aim to highlight the key clinical features of periorbital NF, demonstrate the rapid progression of the disease, and the need for prompt identification and decisive intervention. Both patients presented with fever and left-sided periorbital swelling and showed rapid progression of swelling and gangrenous changes to the periorbital skin with worsening proptosis. They were treated with broad-spectrum intravenous antibiotics and underwent emergency surgical debridement of necrotic tissue followed by reconstruction. We propose a formal protocol that we recommend to aid the diagnosis and management of periorbital NF in an acute setting.
PubMed: 38826916
DOI: 10.7759/cureus.59501 -
The American Journal of Case Reports Jun 2024BACKGROUND Orbital metastasis originating from hepatocellular carcinoma (HCC), particularly as an initial manifestation in patients without a known history of HCC, is... (Review)
Review
Orbital Metastasis as the First Manifestation of Hepatocellular Carcinoma, and Its Effective Treatment with Combined Dual Immunotherapy: A Case Report and Review of the Literature.
BACKGROUND Orbital metastasis originating from hepatocellular carcinoma (HCC), particularly as an initial manifestation in patients without a known history of HCC, is rare. Few reports exist on the treatment of patients having HCC with orbital metastasis using targeted therapy or immunotherapy. CASE REPORT We report a case of advanced-stage HCC in a 65-year-old man who first presented with progressive, painless blurred vision and proptosis of the right eye for 2 weeks. The patient had no history of chronic liver disease or cancer. Computed tomography revealed an enhancing hyperdense extraconal mass in the right orbit; a biopsy revealed metastatic HCC. Abdominal CT, which was performed to investigate the primary cancer, revealed a 1.2×1.6-cm arterial-enhancing nodule with venous washout in hepatic segment 5, associated with liver cirrhosis. The patient's serum alpha-fetoprotein level was 70.27 ng/dL. Chest computed tomography revealed lung metastasis. Thus, first-line systemic therapy combining durvalumab and tremelimumab was initiated alongside palliative radiotherapy targeting the right orbit, which began 1 week after the first dose of dual immunotherapy. The patient had significant clinical improvement, reduced proptosis, and serum alpha-fetoprotein levels. CONCLUSIONS Although orbital metastasis is a rare manifestation of HCC, physicians should recognize and consider aggressive investigations for early diagnosis, especially in patients with existing risk factors for HCC. Dual immunotherapy with durvalumab and tremelimumab in combination with radiotherapy can be considered a potential treatment option for managing advanced HCC with orbital metastasis.
Topics: Humans; Male; Carcinoma, Hepatocellular; Liver Neoplasms; Aged; Orbital Neoplasms; Antibodies, Monoclonal, Humanized; Antibodies, Monoclonal; Immunotherapy; Antineoplastic Combined Chemotherapy Protocols; Tomography, X-Ray Computed; Antineoplastic Agents, Immunological
PubMed: 38825807
DOI: 10.12659/AJCR.944002 -
PloS One 2024While several studies have proposed a connection between the gut microbiome and the pathogenesis of Graves's disease (GD), there has been a lack of reports on alteration... (Comparative Study)
Comparative Study
While several studies have proposed a connection between the gut microbiome and the pathogenesis of Graves's disease (GD), there has been a lack of reports on alteration in microbiome following using anti-thyroid drug treatment (ATD) to treat GD. Stool samples were collected from newly diagnosed GD patients provided at baseline and after 6 months of ATD treatment. The analysis focused on investigating the association between the changes in the gut microbiome and parameter including thyroid function, thyroid-related antibodies, and the symptom used to assess hyperthyroidism before and after treatment. A healthy control (HC) group consisting of data from 230 healthy subjects (110 males and 120 females) sourced from the open EMBL Nucleotide Sequence Database was included. Twenty-nine GD patients (14 males and 15 females) were enrolled. The analysis revealed a significant reduction of alpha diversity in GD patients. However, after ATD treatment, alpha diversity exhibited a significant increase, restored to levels comparable to the HC levels. Additionally, GD patients displayed lower levels of Firmicutes and higher levels of Bacteroidota. Following treatment, there was an increased in Firmicutes and a decrease in Bacteroidota, resembling levels found in the HC levels. The symptoms of hyperthyroidism were negatively associated with Firmicutes and positively associated with Bacteroidota. GD had significantly lower levels of Roseburia, Lachnospiraceaea, Sutterella, Escherichia-shigella, Parasuterella, Akkermansia, and Phascolarctobacterium compared to HC (all p < 0.05). Post-treatment, Subdoligranulum increased (p = 0.010), while Veillonella and Christensenellaceaea R-7 group decreased (p = 0.023, p = 0.029, respectively). Anaerostipes showed a significant association with both higher smoking pack years and TSHR-Ab levels, with greater abundantce observed in smokers among GD (p = 0.16). Although reduced ratio of Firmicutes/Bacteroidetes was evident in GD, this ratio recovered after treatment. This study postulates the involvement of the gut microbiome in the pathogenesis of GD, suggesting potential restoration after treatment.
Topics: Humans; Graves Disease; Gastrointestinal Microbiome; Male; Female; Adult; Antithyroid Agents; Middle Aged; Feces; Case-Control Studies
PubMed: 38820506
DOI: 10.1371/journal.pone.0300678 -
Journal of Surgical Case Reports May 2024Orbital abscesses are caused by infection within or near the orbit and show obvious signs of pain, proptosis and raised inflammatory markers. Diagnosis is based on...
Orbital abscesses are caused by infection within or near the orbit and show obvious signs of pain, proptosis and raised inflammatory markers. Diagnosis is based on clinical features and radiological imaging, and requires early antibiotics and often surgical drainage to save vision. Sub-Tenon's injections of triamcinolone acetonide (TA) have caused localized infections in previous reports, which have responded to therapeutic interventions. Here we report a case where a delayed presentation of an orbital abscess secondary to sub-Tenon's TA for persistent post-operative cystoid macular oedema, without obvious signs of infection, rapidly progressed to cause orbital compartment syndrome. Despite treatment, the patient lost complete vision in the affected eye. This case discusses the rare and unusual cause of abscess formation and a diagnostic dilemma.
PubMed: 38817784
DOI: 10.1093/jscr/rjae339 -
Frontiers in Immunology 2024Thyroid eye disease (TED) is a disfiguring autoimmune disease characterized by changes in the orbital tissues and is caused by abnormal thyroid function or... (Review)
Review
Thyroid eye disease (TED) is a disfiguring autoimmune disease characterized by changes in the orbital tissues and is caused by abnormal thyroid function or thyroid-related antibodies. It is the ocular manifestation of Graves' disease. The expression of thyroid-stimulating hormone receptor (TSHR) and the insulin-like growth factor-1 receptor (IGF-1 R) on the cell membrane of orbital fibroblasts (OFs) is responsible for TED pathology. Excessive inflammation is caused when these receptors in the orbit are stimulated by autoantibodies. CD34 fibrocytes, found in the peripheral blood and orbital tissues of patients with TED, express immune checkpoints (ICs) like MHC II, B7, and PD-L1, indicating their potential role in presenting antigens and regulating the immune response in TED pathogenesis. Immune checkpoint inhibitors (ICIs) have significantly transformed cancer treatment. However, it can also lead to the occurrence of TED in some instances, suggesting the abnormality of ICs in TED. This review will examine the overall pathogenic mechanism linked to the immune cells of TED and then discuss the latest research findings on the immunomodulatory role of ICs in the development and pathogenesis of TED. This will offer fresh perspectives on the study of pathogenesis and the identification of potential therapeutic targets.
Topics: Humans; Graves Ophthalmopathy; Animals; Immune Checkpoint Inhibitors; Immune Checkpoint Proteins; Autoantibodies; Receptor, IGF Type 1; Receptors, Thyrotropin
PubMed: 38817600
DOI: 10.3389/fimmu.2024.1392956 -
Journal of Medicine and Life Feb 2024The present report describes for the first time a case of diffuse hyperthyroidism in a 30-year-old female patient who had normal levels of thyroid-stimulating hormone...
The present report describes for the first time a case of diffuse hyperthyroidism in a 30-year-old female patient who had normal levels of thyroid-stimulating hormone receptor antibodies (TSHR-Ab), slightly elevated plasma levels of thyroid hormones, and slightly increased thyroid blood flow. Seven years before, after severe stress, she had Graves' disease with elevated plasma levels of TSHR-Ab. The patient's recent medical history included mental stress and autonomic dysfunction. This report describes a mild form of hyperthyroidism in terms of elevated plasma levels of thyroid hormones and Doppler ultrasonography data; this condition was first defined as 'minor hyperthyroidism'. The examination data suggest a probable secondary role of the immune system and primary role of the autonomic nervous system in the pathogenesis of Graves' disease.
Topics: Humans; Female; Adult; Hyperthyroidism; Receptors, Thyrotropin; Autoantibodies; Graves Disease; Immunoglobulins, Thyroid-Stimulating; Thyroid Hormones
PubMed: 38813368
DOI: 10.25122/jml-2023-0458 -
Acta Medica Philippina 2024A carotid-cavernous sinus fistula (CCF) is an abnormal communication between the internal carotid artery and/or the external carotid artery and the cavernous sinus....
BACKGROUND
A carotid-cavernous sinus fistula (CCF) is an abnormal communication between the internal carotid artery and/or the external carotid artery and the cavernous sinus. There is a paucity of information on the ophthalmic outcomes of endovascular treatment for CCF in the Philippines.
OBJECTIVES
This study aimed to describe the clinical experience of CCF in our institution in order to further our understanding of the disease. This study described the demographic profile, risk factors, and clinical features of patients with CCF, and compared the data of patients according to the type of CCF. The study discussed the changes in clinical features over time in CCF patients who chose not to undergo endovascular treatment. The study also discussed the clinical outcomes of CCF patients who underwent endovascular treatment and compared the clinical outcomes to CCF patients who did not undergo endovascular treatment.
METHODS
A retrospective cohort study design was performed using a medical record review of patients clinically diagnosed with CCF from January 2011 to June 2019.
RESULTS
One hundred twenty medical records of patients diagnosed with CCF were included. Based on angiographic findings, patients were grouped according to type of CCF, with 86 patients in Group 1 or Direct CCF, 23 patients in Group 2 or Indirect CCF, and 11 patients in Group 3 or Mixed type of CCF. The patients were also grouped according to treatment, with 109 patients in Group A, or patients who did not undergo endovascular treatment, and 11 patients in Group B, or patients who underwent endovascular treatment. There was a male predominance in CCF, most occurring in the age range of 26 to 35 years. Risk factors for CCF were trauma and hypertension. Clinical features included the presence of blurring of vision, proptosis, corkscrewing of conjunctival vessels, extraocular movement limitation, diplopia, audible bruit, elevated intraocular pressure, and pulsation. Findings on Computed Tomography scan included dilated superior ophthalmic vein, proptosis, and enlarged extraocular muscles. Direct CCF (Group 1) occurred mostly in males, with mean age of 39.1 years, and with trauma as the major risk factor. Indirect CCF (Group 2) occurred mostly in females, with mean age of 52.1 years, and with hypertension as the major risk factor. Regardless of the type of CCF, patients who did not undergo endovascular treatment can exhibit spontaneous improvement or worsening of clinical features. Patients who underwent endovascular treatment generally had favorable clinical outcomes, manifesting as either improvement or no worsening of features. There were minimal ophthalmic complications associated with treatment.
CONCLUSION
Endovascular treatment is safe and effective in the improvement of visual acuity, corkscrewing of conjunctival vessels, amount of proptosis, extraocular movement limitation, diplopia, and presence of audible bruit.
PubMed: 38812769
DOI: 10.47895/amp.vi0.6550 -
Immunity, Inflammation and Disease May 2024Thyroid-associated ophthalmopathy (TAO) is the most common orbital disease in adults, potentially leading to disfigurement and visual impairment. However, the causes of...
BACKGROUND
Thyroid-associated ophthalmopathy (TAO) is the most common orbital disease in adults, potentially leading to disfigurement and visual impairment. However, the causes of TAO are not fully understood. IL-35B cells are a newly identified regulatory B cells (Bregs) in maintaining immune balance in various autoimmune diseases. Yet, the influence of IL-35Bregs in TAO remains unexplored.
METHODS
This study enrolled 36 healthy individuals and 14 TAO patients. We isolated peripheral blood mononuclear cells and stimulated them with IL-35 and CpG for 48 h. Flow cytometry was used to measure the percentages of IL-35Bregs.
RESULTS
The percentage of circulating IL-35Bregs was higher in TAO patients, and this increase correlated positively with disease activity. IL-35 significantly increased the generation of IL-35Bregs in healthy individuals. However, B cells from TAO patients exhibited potential impairment in transitioning into IL-35Breg phenotype under IL-35 stimulation.
CONCLUSIONS
Our results suggest a potential role of IL-35Bregs in the development of TAO, opening new avenues for understanding disease mechanisms and developing therapeutic approaches.
Topics: Humans; B-Lymphocytes, Regulatory; Interleukins; Female; Male; Adult; Middle Aged; Graves Ophthalmopathy; Aged
PubMed: 38804861
DOI: 10.1002/iid3.1304