American Journal of Case Reports最新文献

BACKGROUND Trichoblastic carcinoma (TC) is a rare adnexal tumor that usually occurs on the scalp and the face. Clinical presentation is nonspecific and can simulate basal cell carcinoma (BCC). Optimal treatment and outcomes remain unclear. Surgery remains the main treatment but can be difficult or impossible in locally-advanced situations. Thus, conservative options must be developed as TC affects elder populations, and radiotherapy may be a good conservative option but its use in TC is poorly documented. CASE REPORT We report a case of an 86-year-old man with an asymptomatic 5-cm tumor of the nose, extending to the right cheek and inner canthus. He was immunodeficient on kidney transplant and was treated by prednisone and tacrolimus. Biopsy confirmed the TC diagnosis. Surgery and brachytherapy were not possible due to clinical extension, advanced age, and comorbidities. External beam radiation with intensity modulated radiotherapy on tomotherapy was attempted. Seventy Gy was delivered in 35 fractions over 2 months 5/7 days. A bolus with ball (Seemed®) was used each treatment day to deliver an adequate dose of radiotherapy to the skin. Clinical examination at 1 month did not reveal any residual lesion and the patient remains in complete remission after 9 months of follow-up. Tolerance was characterized by grade 2 (NCI CTC AE V4) radiodermatitis, mucositis, and conjunctivitis, managed by local medications. CONCLUSIONS We report a rare case of locally-advanced TC, successfully treated by external beam radiation, which may be an acceptable alternative treatment for unresectable tumors.

BACKGROUND Surgery was once considered the only possible treatment for uterine fibroids. However, a more conservative treatment approach can preserve women's reproductive capacity. In recent years, uterine artery embolization (UAE) and medical treatments have been introduced as a minimally or non-invasive therapeutic option. Relugolix is a non-peptide gonadotrophin-releasing hormone (GnRH) receptor antagonist used to reduce the release of luteinizing hormone (LH) and follicle-stimulating hormone (FSH). This report presents the case of a 40-year-old woman with inoperable uterine fibroids managed with combined uterine artery embolization and Relugolix, a non-peptide gonadotrophin-releasing hormone (GnRH) receptor antagonist. CASE REPORT A woman in her 40s presented with recurrent menometrorrhagia and an 80-mm uterine fibroid causing bleeding and anemia. Due to her medical history and previous surgeries, surgery was deemed risky. Instead, a conservative approach involving UAE followed by Relugolix combination therapy (Relugolix-CT) was pursued before performing minimally invasive surgery. Following bilateral UAE, the ultrasound scan showed there had already been a reduction in the fibroid size. Right after the UAE, the patient was discharged with Relugolix-CT, which reduced the symptoms and helped further reduce the fibroid's size and vascularity. Six months later, a mini-resectoscopic myomectomy was performed under local anaesthesia. CONCLUSIONS This case underscores the effectiveness of UAE and Relugolix-CT as a pre-surgical strategy for large uterine fibroids in patients who are not immediately suitable candidates for major surgery. This new combined approach can lead to improved patient outcomes and reduced surgical risks.

BACKGROUND Despite global vaccination efforts, COVID-19 still necessitates effective treatments for severe cases that can quickly escalate to life-threatening complications, such as acute respiratory distress syndrome (ARDS) and secondary pulmonary arterial hypertension (PAH). Here, we present the clinical journey of a 73-year-old Ecuadorian man who developed severe COVID-19 pneumonia complicated by an opportunistic Candida krusei infection and ARDS, subsequently progressing to long-term PAH, managed with bosentan, an endothelin 1 (ET-1) antagonist. CASE REPORT The patient, vaccinated with 2 doses of CoronaVac, experienced severe COVID-19 complications, including ARDS and secondary PAH, further complicated by a C. krusei infection. Despite prompt mechanical ventilation and intensive care, his condition rapidly deteriorated. Clinical evaluation confirmed COVID-19-associated ARDS, secondary PAH, and C. krusei infection through bronchoalveolar lavage. The therapeutic approach combined bosentan (125 mg twice daily) with dual antifungal therapy, leading to significant stabilization and eventual discharge. Post-discharge assessments showed persistent cardiopulmonary dysfunction, consistent with post-COVID-19 syndrome. CONCLUSIONS This case highlights critical COVID-19 complications in a vaccinated patient. While vaccination may provide substantial protection, COVID-19 pneumonia treated with corticosteroids can increase the risk of opportunistic infections like C. krusei, and ARDS can lead to pulmonary fibrosis and PAH. This case underscores the need for research on therapeutic strategies for complex COVID-19 cases and emphasizes comprehensive, personalized care for managing COVID-19 complications and sequelae.

BACKGROUND Melanoma is a malignant transformation of melanocytes known for its capacity to metastasize, necessitating early diagnosis and intervention. While over 97% of melanomas are diagnosed with a known primary site, such as the skin, eye, or mucous membranes, rare cases like melanoma of unknown primary (MUP) can present with atypical manifestations, highlighting the need for comprehensive examinations. CASE REPORT In January 2023, a 68-year-old woman presented with a 4-week history of swelling and discoloration in her left upper eyelid, revealing a hard, livid mass indicative of malignancy. Surgery and subsequent histopathology confirmed epithelioid cell melanoma in the lacrimal gland. Although no other primary melanoma sites were found, imaging and biopsy in August 2023 identified metastatic spread to her lungs. Despite proton radiotherapy, progression led to thoracoscopic surgery for metastasis resection in September 2023. Further re-evaluation revealed metastases in both lungs, leading to her enrolment in a trial comparing PD-1 (programmed death ligand 1) antibody plus LAG-3 (lymphocyte activation gene 3) antibody with PD-1 monotherapy. Subsequent progression required treatment with ipilimumab and nivolumab, which resulted in near complete regression of the orbital tumor. Unfortunately, she later developed serious complications, including hypophysitis, lung infection, renal failure, and viral meningitis, which ultimately led to her death in April 2024. CONCLUSIONS This case report highlights the diagnostic challenge in distinguishing between melanoma of known and unknown primary origins. It emphasizes the importance of thorough histological work-up and heightened suspicion for melanoma of the lacrimal gland, despite its rarity, to ensure prompt diagnosis and treatment due to its metastatic potential.

BACKGROUND Neuroendocrine tumors (NETs) of the middle ear are exceptionally rare, representing less than 2% of primary ear tumors in adults. These neoplasms originate from neuroendocrine cells within the middle ear cavity, posing significant diagnostic and therapeutic challenges. CASE REPORT We present a 27-year-old man with a 2-month history of left-sided hearing loss, tinnitus, and ear fullness. Comprehensive assessments, including tuning fork tests, pure tone audiometry, computed tomography (CT) scan of the temporal bone, and magnetic resonance imaging (MRI), were conducted. Differential diagnoses, included cholesteatoma and other middle ear masses, but chronic suppurative otitis media was not considered due to the intact tympanic membrane. Intraoperative findings revealed a pale pink mass encasing the ossicular chain, confirmed as a neuroendocrine tumor (CK [+], Syn [+++], CK18 [+], EMA [++], CD56 [-], CgA [+], Ki67 [2%+]) by immunohistochemistry. The tumor was excised via an endoscopic tympanotomy. Postoperative follow-up over 2 years showed no recurrence or metastasis, and the patient reported no significant hearing impairment affecting quality of life. CONCLUSIONS Middle ear neuroendocrine tumors require meticulous diagnostic workup and precise surgical intervention. This case adds to the limited body of evidence supporting that middle ear neuroendocrine tumors, although rare, can be managed effectively with tailored surgical approaches. Continued case reporting will be vital to enhance understanding and to refine treatment guidelines for optimal patient outcomes.

BACKGROUND This report presents the case of a 49-year-old man with pulmonary aspergilloma 6 months after hospitalization and mechanical ventilation for COVID-19 who was treated with robot-assisted lobectomy. COVID-19-associated pulmonary aspergillosis is a life-threatening complication of SARS-CoV-2 infection. Aspergilloma, a delayed complication of COVID-19, is rare. Major risk factors for pulmonary aspergilloma include neutropenia, solid organ transplantation, prolonged high-dose corticosteroid therapy, hematological malignancy, cytotoxic therapy, acquired immunodeficiency syndrome, and chronic granulomatous disease. Common symptoms include cough, shortness of breath, fatigue, wheezing, weight loss, fever, and chest pain. Hemoptysis is the most severe complication of pulmonary aspergilloma. Treatment options include antifungals, bronchial artery embolization, and surgery. Surgical treatment is considered definitive for patients who do not respond to antifungal medications. CASE REPORT We describe the case of a 49-year-old man with pulmonary aspergilloma who developed a delayed sequela of SARS-CoV-2 infection. He presented to a pulmonary clinic 6 months after a severe COVID-19-related hospitalization, with symptoms of mild hemoptysis, cough, and shortness of breath. A computed tomography scan of the chest revealed a right upper lobe cavitary lesion approximately 9.6×6.1 cm in size. Bronchoalveolar lavage during bronchoscopy revealed Aspergillus fumigatus. The patient did not respond to antifungal treatment and was successfully treated with a robotic-assisted lobectomy. CONCLUSIONS Aspergillus infection should be considered in COVID-19 survivors with pulmonary symptoms. Minimally invasive robotic lobectomy is a feasible option for high-risk patients with post-COVID-19 aspergilloma and hemoptysis who are resistant to medical treatment.

BACKGROUND Juxtapapillary retinal capillary hemangioma (JRCH) is a rare vascular hamartoma that, when present, can cause many complications. We report a case of branch retinal artery occlusion (BRAO) in the setting of progressive exophytic JRCH, which, to the best of our knowledge, has not been reported until now. CASE REPORT The patient was 34-year-old woman who presented with visual blurring and visual field defects of the left eye and with no significant medical history. Fundoscopy and other auxiliary examinations revealed a red, elevated mass at the optic disc with surrounding hemorrhage and vascular abnormalities. JRCH was then diagnosed, and she underwent conservative treatment. However, 3 days later, during the follow-up, she developed a worsened best corrected visual acuity (BCVA) and visual field defect. The optical coherence tomography angiography found swelling of the tumor, macular edema, and vascular occlusion of the inferior macula; therefore, secondary BRAO was diagnosed. With emergency treatments, the tumor size reduced during the follow-up; however, the vision field, BCVA, and vascular occlusion were not improved. CONCLUSIONS JRCH are rare benign tumors associated with progressive vision loss due to complications such as macular exudate and retinal detachment. Various treatments, including laser, radiotherapy, and anti-angiogenic intravitreal (anti-VEGF) injections, have been suggested; however, the optimal approach is unclear. JRCH complicated with BRAO has not been reported before. Our report suggests that the tumor was closely related to the retinal arteriovenous system, and BRAO could be a vision-threatening complication of JRCH that deserves more attention.

BACKGROUND Extragastrointestinal stromal tumors (E-GISTs) are a rare subtype of gastrointestinal stromal tumors (GISTs) that develop outside of the gastrointestinal tract from interstitial cells of Cajal, exhibiting specific markers such as CD117 and DOG1. These tumors often present diagnostic challenges, particularly when their clinical manifestations mimic other abdominal conditions, such as acute appendicitis. CASE REPORT A 75-year-old male patient with a history of multiple comorbidities presented to the Emergency Department with symptoms of chronic pain in the right lower quadrant. Imaging studies, including computed tomography scans, revealed a large heterogeneous density mass measuring 11.3×9.2 cm in the right lower quadrant and a dilated appendix with wall thickening. Subsequent surgical resection with right hemicolectomy and primary anastomosis was performed, and postoperative considerations included adjuvant therapy with imatinib, due to spindle cell morphology and high mitotic activity. Histopathological examination and immunohistochemical staining confirmed the diagnosis, showing positive CD117, DOG1, Bcl-2, D2-40, and WT1 markers. CONCLUSIONS This case report aims to highlight the complexities in diagnosing E-GISTs with atypical presentations and emphasizes the critical role of comprehensive imaging and histopathological assessments in achieving an accurate diagnosis and guiding appropriate management strategies. The successful diagnosis and management highlight the critical role of imaging modalities and immunohistochemical analysis in guiding treatment decisions, while postoperative care, including targeted therapy, is crucial for reducing the risk of recurrence and improving patient outcomes. Future research should focus on optimizing postoperative management strategies and investigating the potential of intraoperative biopsies for tumors abutting adjacent structures.

BACKGROUND Cut throat injury (CTI) is a type of penetrating neck injury that is an emergency in otorhinolaryngology and head and neck surgery. CASE REPORT We report 2 cases of CTI in zone 2. Patient 1 had severe injury to the larynx, trachea, and esophagus due to attempted suicide, whereas Patient 2 had a ruptured larynx due to attempted homicide, without injury to the vocal cords. The emergency doctor only used gauze to compress the neck rather than inserting a tracheal cannula through the opening immediately to maintain airway patency. Patient 1 had dyspnea at 1 point. Fortunately, Patient 2 had a stable neck wound did not bleed much, with few blocking blood clots. After performing a low tracheotomy in these 2 patients, the neck injury was repaired, gastric tubes were placed, and gastric enteral feeding diet was commenced. Both patients received anti-infection treatment and professional psychiatric therapy after surgery, and both recovered well. CONCLUSIONS Airway management is the critical first step, and the tracheal cannula should be inserted through the opening in the injured larynx for the first time to maintain airway patency; patients whose condition is unstable should undergo immediate surgical exploration, and those whose condition is stable without hard signs should undergo enhanced neck CT to assess the injury; prompt psychiatric care is also critical.

BACKGROUND Simple (unicameral) bone cysts (SBCs) are benign, fluid-filled bone lesions that are typically solitary and can be unicameral (single chamber) or septated. Most commonly affecting the long bones of children and adolescents, SBCs can be asymptomatic or associated with pain or fractures. Multifocal SBCs, featuring multiple cysts across different bones, are exceptionally rare and pose unique diagnostic and therapeutic challenges. Here, we report an exceptional case of a 20-year-old woman with multifocal SBCs affecting 9 known anatomical sites and discuss the unique diagnostic and therapeutic challenges. CASE REPORT A 20-year-old woman presented with right proximal tibia pain after a fall. Radiographs and MRI revealed radiolucent lesions in the proximal tibia, distal tibia, and fibula, with additional lesions identified on whole-body scintigraphy in the left proximal and distal tibia, left proximal humerus, and pelvis. Initial biopsy was suggestive of SBCs but inconclusive, and surgical biopsy was required for definitive diagnosis. Histologic evaluation confirmed SBCs, demonstrating cyst walls, cholesterol clefts, and fibrin-like deposits. Treatment included curettage, local adjuvant therapy with argon beam and doxycycline, bone grafting, and doxycycline sclerotherapy for pelvic and distal extremity lesions. At 17 months after treatment, the patient remained asymptomatic, with imaging showing sclerosis and near-complete resolution of the lesions. CONCLUSIONS This report presented a rare case of a young woman with multiple SBCs across 9 anatomical sites. Overall, the management of multifocal SBCs requires a multidisciplinary approach, involving orthopedic surgeons, radiologists, and pathologists for optimal outcomes.