American Journal of Case Reports最新文献

BACKGROUND The configuration of the hepatic arteries is known to vary substantially between individuals. Here, we report a rare retroperitoneal configuration of an accessory hepatic artery existing alongside a left and right hepatic artery branching from the proper hepatic artery. During routine dissection, we discovered an anomalous configuration of the hepatic arteries that does not fit the commonly used categorizations for abnormal hepatic vasculature. We briefly discuss potential implications of this finding. CASE REPORT The left and right hepatic arteries branch directly from the common hepatic artery and supply the liver. The accessory hepatic artery arises directly from the superior mesenteric artery, travels posterior to the portal triad, and enters the porta hepatis. It was found to travel posterior to the neck of the pancreas, duodenum, and portal triad at the same depth as the inferior vena cava, suggesting that the accessory right hepatic artery was traveling retroperitoneally, posterior to the opening of the lesser sac. The accessory hepatic artery exclusively supplies the cystic arteries. Considering existing classification schemes, our case most closely resembles Michels's type 6 and Hiatt's type 3, but most specifically López-Andújar's type 6. To the best of our knowledge, this is one of the first reported cases of a retroperitoneal accessory right hepatic artery. CONCLUSIONS Knowledge of the possible variations of the hepatic arteries is important to reduce the risk of iatrogenic injury and improve patient outcomes in surgery, especially in laparoscopic cases or pancreatoduodectomies where more unique variants may be especially difficult to identify or easier to injure. Future research should explore the frequency of retroperitoneal accessory right hepatic arteries, as their unique course may be easily missed.

BACKGROUND ROS1 fusion-positive locally-advanced lung adenocarcinoma is a rare malignant tumor with no clear neoadjuvant therapy guidelines and a poor prognosis. This report describes a 49-year-old man with a ROS1 fusion-positive locally-advanced lung adenocarcinoma with a pathological complete response (pCR) to the tyrosine kinase inhibitor crizotinib combined with chemotherapy. CASE REPORT A 49-year-old Chinese man visited the hospital with a cough and phlegm that began over 20 days ago. Computed tomography (CT) revealed a 4.5-cm diameter mass in the lower lobe of the left lung with enlarged lymph nodes fused together in the left hilum, staging stage IIIA (cT2bN2aM0). Given the pathological diagnosis of adenocarcinoma of lung from the transbronchial lung biopsy (TBLB), the patient subsequently underwent chemotherapy with the lobaplatin and paclitaxel regimen. Subsequently, genetic tests using fluorescence quantitative polymerase chain reaction (PCR) assay for biopsy pathology showed ROS1 fusion-positivity. Based on this, after completing 1 cycle of chemotherapy, the patient continued with daily oral treatment with 500 mg of crizotinib. A follow-up CT after 30 days of crizotinib therapy showed the tumor had vanished. Radical surgery confirmed pCR, and the patient continues crizotinib maintenance therapy with no signs of recurrence on subsequent chest CTs. CONCLUSIONS This case serves to underscore the excellent efficacy of neoadjuvant therapy in a patient with ROS1 fusion-positive locally-advanced lung adenocarcinoma. Neoadjuvant lobaplatin/paclitaxel combined with crizotinib can be considered for such patients, but attention should be paid to the difficulty of surgery, timing selection, and formulation of management guidelines.

BACKGROUND Hepatic lesion in a young woman can lead to multiple diagnostic hypotheses, mainly infection and tumor. Crohn's disease (CD) is hardly evoked by clinicians but is reportedly associated with liver damage, especially diffuse granulomas and aseptic abscess. IgA deficiency has been associated with celiac disease or inflammatory bowel disease, including CD. In this report, we present the diagnosis of CD in a fit 23-year-old woman following detection of aseptic liver abscess associated with a previously unknown selective IgA deficiency. CASE REPORT A young 23-year-old woman with no previous medical history other than appendicitis 1 year ago was hospitalized with persistent fever for 2 weeks associated with C-reactive protein increase (142 mg/L). Abdominal computed tomodensitometry and MRI showed a 4-cm liver abscess (segment IV). Biopsy revealed an aseptic epithelioid gigantocellular granuloma with caseous-free necrosis and granulomas rich in eosinophilic polynuclei. Furthermore, colonoscopy detected an inflammation in the colonic and ileal mucosa, with focal ulcerations, suggestive of CD. Immunological assessment led to the diagnosis of selective IgA deficiency. Anti-TNF and immunosuppressor therapies led to a rapid recovery and regression of hepatic lesions. CONCLUSIONS CD should be considered in aseptic liver abscess cases. Considering the association between IgA deficiency and CD, IgA (and IgG/IgM) should be assessed in patients with CD. Further research is necessary to confirm if specific manifestations such as aseptic liver abscess frequently occur in patients with CD related to IgA deficiency.

BACKGROUND Human Papilloma Virus (HPV)-associated Vulvar Squamous Cell Carcinomas (VSCC) present more frequently in young women than HPV- independent tumors. Due to its association with HPV infection, the incidence of vulvar cancer is increasing in young women; however, during pregnancy, it is still extremely rare. CASE REPORT We present the case of a 36-year-old pregnant woman at 23 weeks of pregnancy, diagnosed with HPV 16-associated VSCC, Federation of Gynecology and Obstetrics (FIGO) stage IB. Information on the coexistence of VSCC with pregnancy is unique, so it seems extremely important to disseminate it to develop the most effective treatment regimen. Additionally, making any decisions regarding therapeutic methods during pregnancy encounters great ethical problems. The size of the tumor was 0.5 cm with a depth of invasion 0.3 cm. The patient underwent therapy and gave birth by cesarean section at 38 weeks of pregnancy because of orthopedic indications. Surgical removal of the vulvar tumor was performed, including a margin of 1.5 cm of healthy tissue. Due to the patient's lack of consent, the sentinel node biopsy was not performed. No recurrence has been observed for 9 years. CONCLUSIONS The poorer prognosis of HPV-associated VSCC is independent of age and stage, with worse outcomes even in early-stage disease. For this reason, it is essential to sensitize clinicians to the possibility of such a diagnosis and to pay attention to the possibility of taking effective treatment during pregnancy, but safe for the fetus.

BACKGROUND Endometriosis is a condition where uterine lining tissue grows outside the uterine cavity, commonly on the ovaries and pelvic peritoneum, but can also occur in rare locations such as the cervix, lungs or pleura. Cervical endometriosis is typically diagnosed retrospectively through post-surgical pathology as it was in this case. This article presents a case of cervical endometriosis with recurrent vaginal bleeding, reviews recent literature to aid in clinical diagnosis and treatment. CASE REPORT A 42-year-old woman presented with a 5-year history of recurrent vaginal bloody discharge. Clinical evaluations revealed a cyst on the posterior wall of the cervix, suspected to be an endometriotic cyst, along with a uterine diverticulum and an endometrial polyp. The patient underwent surgical excision of the cyst, repair of the uterine diverticulum, and removal of the endometrial polyp. Postoperative pathology confirmed the diagnosis of an endometriotic cyst of the cervix. The patient was subsequently treated with oral Dienogest. An 8-month follow-up showed no complaints of discomfort, with gynecological examinations indicating good recovery and no abnormalities detected on ultrasound. CONCLUSIONS Cervical endometriotic cysts are rare but can lead to serious complications if not properly diagnosed and managed. Clinicians should remain vigilant for abnormal symptoms such as recurrent vaginal bleeding and pelvic pain. Timely and appropriate treatment measures, including surgical intervention and hormonal therapy, should be implemented to reduce the risk of complications and monitor for potential malignant transformation. Regular follow-ups are crucial to ensure early detection of any recurrence or progression.

BACKGROUND Ranulas are typical causes of sublingual cysts in children. However, our case was histopathologically confirmed to be a dermoid cyst. Epidermoid and dermoid cysts of the floor of the mouth account for <0.01% of all oral cavity lesions and 0.29% of head and neck tumors in children. Salivary congestive cysts (ranulas) involve the sublingual salivary glands or the common duct of the sublingual and submandibular salivary glands. This report describes a 13-year-old boy presenting with a sublingual cyst, diagnosed by histopathology as a dermoid cyst. Treatment is based on surgical removal of the cyst, sometimes along with the altered salivary gland. CASE REPORT A 13-year-old boy was admitted to the Department of Otolaryngology with the Subdivision of Maxillofacial Surgery for the diagnosis of a tumor localized under the tongue. A significant growth of the tumor during a 3-month period was noticed, with appearance of a mass effect, speech disorders, and difficulties in eating. Significant elevation of the floor of the mouth and tongue was shown. The presence a ranula was indicated. Surgical excision was performed using intra-oral excision. Histopathological examination revealed a diagnosis of dermatoid cyst. CONCLUSIONS This case highlights the importance of detailed histopathological diagnosis of lesions and the usefulness of imaging methods like magnetic resonance imaging (MRI), ultrasound (US) or computed tomography (CT). Our patient had a dermoid cyst, which appears rarely among children in the floor of the mouth. This shows the significance of their proper differentiation, as some may be misdiagnosed as ranula.

BACKGROUND IgG4-related disease (IgG4-RD) is a systemic fibro-inflammatory disease potentially affecting every part of the human body. Because of variability in clinical presentation, IgG4-RD can be challenging to diagnose. Untreated disease can lead to irreversible organ damage such as fibrosis. Early recognition and therapy are therefore essential. The first step in the treatment of IgG4-RD is glucocorticoid therapy, but the relapse rate after tapering is high. Other agents that are commonly used are disease-modifying antirheumatic drugs (DMARDs) and rituximab. Hydroxychloroquine is not common in the treatment of IgG4-RD, but can be promising. CASE REPORT We here describe a case of a patient with systemic IgG4-RD with manifestation in the pancreas, lung, and salivary glands. Initial treatment consisted of prednisone, with good response. Because of relapse after tapering, prednisone was restarted in combination with azathioprine. However, azathioprine had to be discontinued because of adverse effects. While tapering prednisone, new pulmonary manifestations emerged and hydroxychloroquine was started. This led to an excellent clinical response with no additional adverse effects. CONCLUSIONS This case report on IgG4-RD demonstrates a good response to treatment with hydroxychloroquine. Hydroxychloroquine is believed to have anti-inflammatory and anti-fibrotic effects, which may favorably influence the treatment of IgG4-RD. Therefore, hydroxychloroquine may be a good treatment option in IgG4-RD. Larger cohorts are required to study the efficacy of hydroxychloroquine in IgG4-RD.

BACKGROUND Hypertriglyceridemia (HTG) is associated with circulating free fatty acids that can cause acute pancreatitis. Therapeutic plasma exchange (TPE) is a rapid and effective method of reducing triglyceride levels. This case series presents 6 cases of acute pancreatitis associated with hypertriglyceridemia with a rapid response to therapeutic plasma exchange. CASE REPORT Six patients diagnosed with hypertriglyceridemia-induced acute pancreatitis (HTG-AP) were hospitalized and received therapeutic plasma exchange at the Affiliated Hospital of Shandong University of Traditional Chinese Medicine. Upon admission, laboratory tests and abdominal computed tomography (CT) were performed, and all signs and test results were consistent with the diagnosis of hypertriglyceridemia and acute pancreatitis (AP). Among them, 4 patients were discharged after therapeutic plasma exchange. Anaphylaxis and ketoacidosis occurred in 2 cases during therapeutic plasma exchange. CONCLUSIONS High levels of triglyceride can lead to acute pancreatitis events. After therapeutic plasma exchange treatment for hypertriglyceridemia-induced acute pancreatitis, triglyceride levels decrease significantly, and adverse reactions during therapeutic plasma exchange should be actively watched for. However, there are no clear criteria for applying therapeutic plasma exchange, and more studies are needed to assess the value and risks of this treatment option. This case series shows the importance of evaluating triglyceride levels in patients with acute pancreatitis and the role of therapeutic plasma exchange.

BACKGROUND Second-degree atrioventricular (AV) block is a frequently encountered conduction abnormality on surface electrocardiogram (ECG). However, it does not always imply a block at the AV nodal level. In rare cases, this block can occur below the bundle of His, within the infra-Hisian region of the His-Purkinje system. While the incidence of infra-Hisian block is generally low in the general population, it becomes more common in specific high-risk groups, such as older adults and individuals with structural heart disease. Infra-Hisian block carries a significant risk of progressing to complete heart block, particularly if the patient shows a markedly prolonged His-ventricular (HV) interval or evidence of a bi-fascicular block. CASE REPORT We present the case of a 65-year-old woman who experienced recurrent episodes of syncope. Her surface ECG revealed a bi-fascicular block along with Wenckebach AV block, and 24-h Holter monitoring showed no other significant abnormalities. A baseline short PR interval with second-degree AV block indicated a possible infra-nodal block. An electrophysiology study confirmed an atypical Wenckebach AV block with second-degree infra-Hisian AV block. The patient subsequently underwent permanent pacemaker implantation. CONCLUSIONS Given its potential to develop into more severe forms of heart block, infra-Hisian block is a critical condition that requires accurate identification and management. Several ECG indicators can help diagnose second-degree AV block. For example, RP-dependent PR interval or RP-PR reciprocity is characteristic of Wenckebach AV block. A short PR interval at baseline and minimal amount of PR interval lengthening before block in type 1 second-degree AV block suggest involvement of the His-Purkinje system.

BACKGROUND In several studies, the presence of Howell-Jolly body-like inclusions within neutrophils has been observed in cases of HIV infection, SARS-CoV-2 infection, post-transplant immunosuppression, and during chemotherapy or antiviral therapy. The phenomenon of neutrophils exhibiting Howell-Jolly body-like inclusions on peripheral blood smears can be attributable to viral infections or the pharmacological effects of medications. CASE REPORT A 14-year-old male patient who had received a diagnosis of lymphoblastic leukemia a year ago underwent hematopoietic stem cell transplantation and was readmitted due to a recurrence of gastrointestinal graft-versus-host disease (GVHD). During treatment, tacrolimus was administered in conjunction with steroids to treat GVHD, yet the patient's diarrhea worsened. Examination of a peripheral blood smear revealed neutrophils, with cytoplasmic inclusions resembling Howell-Jolly bodies. Despite an increased dosage of tacrolimus, control of GVHD was suboptimal, and there was a risk of gastrointestinal infection. During further treatment, the patient developed symptoms of gastrointestinal bleeding, which, upon gastroscopy and colonoscopy, led to the diagnosis of extensive gastrointestinal mucormycosis. The therapeutic regimen was escalated to include oral amphotericin B, while continuing tacrolimus and discontinuing all other immunosuppressants. CONCLUSIONS The present case highlights the presence of Howell-Jolly body-like inclusions within the cytoplasm of neutrophils observed in the peripheral blood smear of a patient after hematopoietic stem cell transplantation. Morphological analysis suggests that the emergence of these inclusions is highly likely to be induced by medications used in the treatment of GVHD.