Search

Background: Immunoglobulin G4-related disease (IgG4-RD) includes several immune-mediated fibro-inflammatory conditions affecting multiple organs. Increased IgG4 serum levels support the diagnosis of IgG4-RD and characteristic histopathology of fibrous infiltrates or masses containing IgG4-positive plasma cells. We present the case of a 61-year-old woman with low back pain who was diagnosed with IgG4-RD involving the thoracoabdominal aorta and retroperitoneum. Case Report: A 61-year-old woman who had persistent low back pain was referred to a university hospital in South Korea. Computed tomography (CT) and 2-[18F]-fluoro-2-deoxy-D-glucose positron emission tomography/CT (F-18-FDGPET/CT) demonstrated diffuse circumferential infiltrates from the thoracoabdominal aorta, iliac vessels, and retroperitoneum associated with right obstructive uropathy. The serum IgG4 concentration was 418.0 mg/L (reference range: 30-2000). She underwent laparoscopic retroperitoneal biopsy and received a cystoscopic double- J ureteral stent. IgG4-positive plasma cells, lymphoplasmacytic infiltration, and fibrosis were observed on histopathological examination. She was diagnosed with IgG4-RD and treated with glucocorticoids (GCs) for 6 months. She underwent femoral-to-femoral bypass graft surgery for revascularization due to occlusion of the right iliac arteries. She experienced relapse after GC discontinuation, and GC administration was resumed. She had difficulty tapering GC use owing to persistent low back pain, which improved with combined treatment of GC and immunosuppressant. Conclusions: We present a case of IgG4-RD involving the thoracoabdominal aorta and retroperitoneum based on the 2019 classification criteria. The importance of radiological studies of IgG4-RD has increased, and F-18-FDG-PET/CT, which is a functional imaging modality of the whole body, is a valuable evaluation method for diagnosis and clinical outcomes. [ABSTRACT FROM AUTHOR]

Objective: Unusual clinical course Background: Hemangiomas are relatively rare, slow-growing, benign neoplasms that can cause necrosis, ulceration, and infection leading to airway obstruction or intractable hemorrhage. Controversy persists regarding the treatment options for these tumors, which include active observation, corticosteroids, sclerotherapy, laser treatment, and surgical resection. Case Report: A 61-year-old man presented with a 6-month history of persistent throat clearing and foreign body sensation in the throat. He was receiving medical treatment and psychotherapy for major depressive disorder and anxiety disorder. Laryngoscopy and computed tomography revealed a large, irregular, lobulated mass covered by bluish mucosa in the hypopharynx and larynx on the right without involvement of the true vocal fold or subglottis. Orotracheal intubation was performed under general anesthesia. The hemangioma abutting the epiglottis and arytenoid was dissected by CO2 laser. The hemangioma in the pharyngoepiglottic fold and aryepiglottic fold was resected using an ultrasonic scalpel. The hemangioma in the ventricle, false vocal fold, and paraglottic space was treated by potassium-titanyl-phosphate (KTP) laser photocoagulation. Pathological examination confirmed hemangioma. There has been no recurrence during 18 months of follow-up. Conclusions: The treatment of pharyngolaryngeal hemangiomas is challenging. It is important to select treatment options considering the characteristics of the treatments and the anatomical and functional relationship between the hemangioma and the surrounding structures. Single-session KTP laser photocoagulation combined with surgical resection using an ultrasonic scalpel via a transoral approach according to anatomic site could be an effective treatment for pharyngolaryngeal hemangioma. [ABSTRACT FROM AUTHOR]

Patient: Female, 57-year-old Final. Diagnosis: Rosai-Dorfman disease. Symptoms: Cough Medication: -- Clinical. Procedure: -- Specialty: Otolaryngology. Objective: Rare disease. Background: Rosai-Dorfman disease (RDD), is a rare, benign, proliferative, histiocytic disorder characterized by persistent massive lymphadenopathy, which mimics malignant tumors. Diagnosis of extranodal RDD without lymphadenopathy is difficult due to its unusual clinical manifestation and lack of typical histopathologic features. Hence, it requires both a high degree of clinical suspicion and careful histopathologic examination. Case Report: A 57-year-old woman presented with an isolated mass on the base of the tongue (BOT) without lymphadenopathy. Laryngoscopic examination revealed a mass on the midline of the BOT. The patient underwent complete surgical excision via suspension laryngoscopy with a CO2 laser. Based on the histopathologic features, including numerous histiocytic infiltrations with emperipolesis and cytoplasmic expression of S100 and CD68 in histiocytes, the diagnosis was confirmed as extranodal RDD. No further treatment was required, and followup evaluation revealed no evidence of recurrence. Conclusions: Because no ideal therapeutic approach is available for RDD, treatment should be tailored to the clinical manifestations. To prevent airway obstruction and recurrence, surgery is considered an appropriate option in cases of localized RDD arising on the upper respiratory tract. We report an extremely rare case of extranodal RDD without lymphadenopathy in the BOT, and provide a detailed discussion of its clinical and histopathologic features and treatment with a brief review of the relevant literature. [ABSTRACT FROM AUTHOR]

Background: Primary neuroendocrine tumors (NETs) in the retroperitoneal space are extremely rare. We report the case of a patient diagnosed with primary presacral NET in the retroperitoneum that was initially suspected to be hepatic metastasis, who was followed up for more than 8 years. Case Report: A 78-year-old man was referred to our hospital for the treatment of a hepatic mass. Following resection, the patient was diagnosed with a grade 2 well-differentiated NET. A thorough evaluation to identify the primary tumor detected small well-demarcated presacral nodules on In-111 octreotide single-photon emission tomography/computed tomography (SPECT/CT). Metastases to other locations were not observed. Presacral nodules were difficult to remove using the surgical approach; therefore, we decided to follow up closely. After 4 years, the patient was diagnosed with recurrent hepatic metastasis and peritoneal seeding. Although combination therapy of everolimus and octreotide long-acting repeatable was administered, it was discontinued owing to disease progression. Baseline Ga-68 DOTATOC positron emission tomography-computed tomography revealed adequate avidity for the lesions observed on SPECT/CT; therefore, 5 cycles of peptide receptor radionuclide therapy (PRRT) were administered, after which stable disease was maintained. Conclusions: We identified an extremely rare primary retroperitoneal NET on In-111 octreotide SPECT/CT. During long-term follow-up, although the patient presented with recurrent hepatic metastases and peritoneal seeding, PRRT was successful in stabilizing the disease. [ABSTRACT FROM AUTHOR]

Objective: Unusual or unexpected effect of treatment Background: Robotic thyroidectomy using remote access approaches is currently regarded as the optimal surgical protocol for highly selected patients. This approach has excellent cosmetic outcomes compared with conventional open transcervical thyroidectomy. Although the remote access approach offers significant benefits, it can cause complications associated with the large working space required for surgery. Such complications can lead to unusual imaging findings. Case Report: We report a case of a 37-year-old woman with thyroid cancer who underwent robotic thyroidectomy and demonstrated unusual port-site implantation findings on post-treatment iodine-131 whole-body scintigraphy and single-photon emission computed tomography-computed tomography. Evaluation of stimulated thyroglobulin and additional imaging studies did not reveal any remarkable findings. Through a multidisciplinary discussion, we discovered that the bag had developed a tear during specimen retrieval. Our patient was administered a therapeutic dose of radioiodine, which accumulated within the target area and successfully ablated the implanted tissue. Follow-up imaging and biochemical studies were normal after a follow-up period of 7 years. Conclusions: Port-site seeding is a rare and unexpected surgical complication; however, it can be treated with radioiodine therapy involving a therapeutic dose. Meticulous surgical manipulation is essential to prevent port-site implantation related to spillage and tearing of thyroid or cancer tissue. Awareness and identification of these rare complications, which manifest as unusual imaging findings, are critical for improving the accuracy of interpretation. [ABSTRACT FROM AUTHOR]

Objective: Rare disease Background: Intravascular papillary endothelial hyperplasia (IPEH), is a vascular tumor characterized by the proliferation of endothelial cells with papillary formations. It is a rare benign disease; therefore, it is important to exclude malignant vascular neoplasm in order to prevent misdiagnosis and inappropriate overtreatment. Case Report: Herein, we describe the case of a 51-year-old male who reported changes in his voice, and who was preoperatively diagnosed with vocal cord polyp and hemorrhagic change based on laryngoscopy. He underwent laryngomicrosurgery and the lesion was completely excised via the microflap surgical method. Histopathology analysis was consistent with IPEH. Conclusions: Herein, we report an extremely rare case of IPEH arising from the true vocal cord, and we provide a brief review of the relevant literature and a detailed discussion of this rare clinical entity. [ABSTRACT FROM AUTHOR]