{"title":"B-Acute Lymphoblastic Leukemia/Lymphoblastic Lymphoma Mimicking Fibrosing Mediastinitis: A Case Report and Diagnostic Insight.","authors":"Aya Kitamura, Shigehisa Yanagi, Kotaro Shide, Yuichiro Sato, Ayako Kamiunten, Yasuhiro Yamanari, Akiko Kitamura, Makoto Sumiyoshi, Yasuharu Oda, Hironobu Tsubouchi, Kazuya Shimoda, Taiga Miyazaki","doi":"10.12659/AJCR.945804","DOIUrl":"10.12659/AJCR.945804","url":null,"abstract":"<p><p>BACKGROUND Fibrosing mediastinitis (FM) is a rare, fibroproliferative disorder within the mediastinum. It is extremely rare for hematologic malignancies to develop as FM. CASE REPORT A 32-year-old Japanese man with a 1-month history of headache and 2-week history of facial swelling underwent chest computed tomography (CT); a diffuse mass-like lesion was revealed in the anterior mediastinum with severe stenosis of vital mediastinal organs. After a surgical biopsy, an initial diagnosis of idiopathic FM was made. The FM lesions responded mildly to corticosteroids but recurred repeatedly. Sixteen months after the treatment initiation, blasts appeared in the peripheral blood (PB), and the patient was diagnosed with B-acute lymphoblastic leukemia/lymphoblastic lymphoma (B-ALL/LBL). Chemotherapy led to complete remission of the B-ALL/LBL and almost complete disappearance of FM-like lesions. Immunohistochemistry of the mediastinal biopsy specimen taken before the blasts' appearance in PB demonstrated a CD34/CD7/terminal deoxynucleotidyl transferase-positive population, an identical pattern of expression common to the blasts in the patient's PB and bone marrow. CONCLUSIONS This is the first case report of B-ALL/LBL presenting as FM. This case underscores the importance of considering the possibility of latent hematologic malignancy even in the absence of new symptoms other than those caused by FM lesions for a long period of time. This is the first demonstration that leukemia cells may be present in the FM lesions from the initial stage of disease onset. Even if a diagnosis of idiopathic FM is confirmed, continued suspicion of the presence of hematologic malignancy is vital for improving patient outcomes.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"25 ","pages":"e945804"},"PeriodicalIF":1.0,"publicationDate":"2024-12-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11694771/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142903748","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Mohammed Imran Quraishi, Mark Andrew Rowley, Daniel Fulks, Alexandria Atkins
{"title":"Multiple Gas-Containing Renal Stones: A Case Report.","authors":"Mohammed Imran Quraishi, Mark Andrew Rowley, Daniel Fulks, Alexandria Atkins","doi":"10.12659/AJCR.946317","DOIUrl":"10.12659/AJCR.946317","url":null,"abstract":"<p><p>BACKGROUND Emphysematous urinary tract infections are rare and serious conditions that are often multifactorial in etiology and may be associated with the presence of renal stones. Diagnosis can be made by finding gas within the renal collecting system or parenchyma. However, the radiographic finding of gas within a renal stone is rare and little has been published to describe the significance of this finding, its promoting factors, and management. While finding a single gas-containing renal stone is rare, we present a patient with multiple gas-containing stones. CASE REPORT A 63-year-old woman with a history of diabetes and recurrent nephrolithiasis was found to have multiple gas-containing renal stones during a workup of gross hematuria. She was currently being treated for a urinary tract infection. Imaging revealed multiple stones with central encapsulated air and hydronephrosis. She underwent subsequent lithotripsy and stent placement due to this concerning finding, but developed sepsis 2 days following treatment. Cultures from the lithotripsy isolated Proteus mirabilis. It is hypothesized that lithotripsy resulted in endotoxin-mediated sepsis. CONCLUSIONS Proper management of gas-containing renal stones in the setting of urinary tract infections includes broad-spectrum antibiotics (carbapenem plus vancomycin if obstruction is present) followed by drainage via percutaneous nephrostomy and then stone removal. Immediate lithotripsy should be avoided in cases of emphysematous pyelonephritis as it can result in endotoxin-mediates sepsis.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"25 ","pages":"e946317"},"PeriodicalIF":1.0,"publicationDate":"2024-12-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11694769/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142956430","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Carolina Maya-González, Teresita Díaz De Ståhl, Sandra Wessman, Fulya Taylan, Bianca Tesi, Kristina Lagerstedt-Robinson, Giorgio Tettamanti, Milena Dukic, Anna Poluha, Gustaf Ljungman, Ann Nordgren
{"title":"Pediatric Soft Tissue Sarcoma in Limb-Girdle Muscular Dystrophy: Molecular Findings and Clinical Implications.","authors":"Carolina Maya-González, Teresita Díaz De Ståhl, Sandra Wessman, Fulya Taylan, Bianca Tesi, Kristina Lagerstedt-Robinson, Giorgio Tettamanti, Milena Dukic, Anna Poluha, Gustaf Ljungman, Ann Nordgren","doi":"10.12659/AJCR.945715","DOIUrl":"10.12659/AJCR.945715","url":null,"abstract":"<p><p>BACKGROUND Limb-girdle muscular dystrophy recessive 1 (LGMDR1) is an autosomal recessive degenerative muscle disorder characterized by progressive muscular weakness caused by pathogenic variants in the CAPN3 gene. Desmoplastic small round cell tumors (DSRCT) are ultra-rare and aggressive soft tissue sarcomas usually in the abdominal cavity, molecularly characterized by the presence of a EWSR1::WT1 fusion transcript. Mouse models of muscular dystrophy, including LGMDR1, present an increased risk of soft tissue sarcomas. However, the DSRCT risk and general cancer risk in patients with LGMD is unknown. Here, we delineate the clinical, molecular, and genetic findings of a patient with LGMDR1 who developed a DSRCT. CASE REPORT The patient was a boy who was diagnosed at the age of 9 years with LGMDR1, caused by the biallelic pathogenic variants NP_000061.1:p.(Arg448Cys) and NP_000061.1:p.(Thr184ArgfsTer36) in CAPN3. At 17 years of age, a pathologic soft tissue mass was found in the right pelvis. Immunostaining was positive for Desmin and negative for Myogenin and MyoD1, and RNA sequencing showed a EWSR1::WT1 fusion transcript, confirming the diagnosis of DSRCT. The patient relapsed after 1 year and, following a second relapse, he was started on palliative treatment. No germline variants in childhood cancer predisposition genes were detected by whole genome sequencing. CONCLUSIONS We describe a patient with LGMDR1 who developed a DSRCT. Since associations between LGMD and pediatric cancer are hitherto unknown, further studies are warranted, as little information is currently published about the pediatric cancer risk in this patient group.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"25 ","pages":"e945715"},"PeriodicalIF":1.0,"publicationDate":"2024-12-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11694770/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142898877","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Norihito Yoshida, Sadamu Takahashi, Mai Hitaka, Yasushi Ohashi, Ryo Ichibayashi
{"title":"Role of Renal Replacement Therapy in Managing Toluene-Induced Acidosis.","authors":"Norihito Yoshida, Sadamu Takahashi, Mai Hitaka, Yasushi Ohashi, Ryo Ichibayashi","doi":"10.12659/AJCR.945657","DOIUrl":"10.12659/AJCR.945657","url":null,"abstract":"<p><p>BACKGROUND Toluene poisoning can occur as a result of occupational exposure in industries such as painting, as well as through misuse, leading to complications such as neurological symptoms due to the accumulation of the metabolic byproduct of hippuric acid and metabolic acidosis. However, the exact mechanisms remain unclear. Hippuric acid is not removed by dialysis, so urinary excretion plays a central role. Symptomatic treatment, primarily involving fluid replacement, remains the standard approach for managing toluene toxicity. Nonetheless, the effectiveness of hemodialysis as a supplementary treatment for toluene poisoning has not yet been conclusively determined. CASE REPORT We present a case of toluene poisoning with prerenal injury in a 52-year-old man with a long history of painting work, which occurred during the summer months. Due to insufficient response to fluid therapy and the persistence of metabolic acidosis, hemodialysis was initiated. Urinary hippuric acid excretion increased after dialysis, suggesting a renoprotective effect of dialysis despite the high protein binding and low dialysability of hippuric acid. This indicates that hemodialysis is a viable treatment option. Following treatment, a furosemide + fludrocortisone loading test was performed to evaluate the reversibility of tubular damage caused by hippuric acid. The test revealed no impairment in acidification, suggesting that the renal tubular acidosis induced by toluene toxicity is likely reversible. CONCLUSIONS This case highlights the diagnosis of renal tubular acidosis secondary to acute toluene poisoning, which was unresponsive to conservative fluid therapy and necessitated hemodialysis. Furthermore, a furosemide and fludrocortisone loading test confirmed the reversibility of the toluene-induced renal tubular acidosis.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"25 ","pages":"e945657"},"PeriodicalIF":1.0,"publicationDate":"2024-12-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11687328/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142898880","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Jose A Ontiveros-Sánchez de la Barquera, Luis Alberto De La Garza García, Silvia Viridiana Esquivel García, Guillermo Sánchez Torres, Grecia Alejandra Perez Jalomo
{"title":"Paradoxical Depressive Response to Intranasal Esketamine in Treatment-Resistant Depression: A Case Series.","authors":"Jose A Ontiveros-Sánchez de la Barquera, Luis Alberto De La Garza García, Silvia Viridiana Esquivel García, Guillermo Sánchez Torres, Grecia Alejandra Perez Jalomo","doi":"10.12659/AJCR.945475","DOIUrl":"10.12659/AJCR.945475","url":null,"abstract":"<p><p>BACKGROUND Esketamine is the only pharmacological agent with glutamatergic neuromodulator properties approved by the US Food and Drug Administration and European Medicines Agency to enhance the effects of serotonin selective or serotonin and norepinephrine reuptake inhibitors. Treatment-resistant depression (TRD) is a challenging and prevalent condition in the psychiatric field, in which patients often experience persistent and severe depressive symptoms, as well as a higher risk of suicidal thoughts and attempts. Esketamine has demonstrated its safety and effectiveness as a pharmacological therapy for TRD. Our report aims to present 2 cases of depressive symptom deterioration and suicide ideation in patients treated with esketamine. CASE REPORT We present 2 cases of TRD that initially responded well to intranasal esketamine but later deteriorated rapidly, with a worsening of depressive symptoms and suicidal ideation. Upon discontinuing esketamine, both patients clinically improved and showed a reduction in suicide ideation. The evaluation of affective symptoms' response to esketamine and evolution was assessed using the Montgomery-Asberg Scale and Clinical Global Impression Severity and Improvement scales. CONCLUSIONS The underlying cause for the paradoxical antidepressant reaction is not entirely clear, but we observed this phenomenon in 2 patients with TRD who were treated with esketamine. Identifications of paradoxical reactions could be difficult in TRD, with highly resistant responses to treatment and suicidal ideation. However, it is relevant to know the prevalence of this phenomenon and for clinicians to be aware of the complications of esketamine treatment.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"25 ","pages":"e945475"},"PeriodicalIF":1.0,"publicationDate":"2024-12-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11687330/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142898871","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Low-Energy Trauma: Severe Liver Injury in an Elderly Patient from a Seated Fall.","authors":"Naoko E Katsuki, Hitoshi Eguchi, Risa Hirata, Hidetoshi Aihara, Masaki Tago","doi":"10.12659/AJCR.946094","DOIUrl":"10.12659/AJCR.946094","url":null,"abstract":"<p><p>BACKGROUND Appropriate management of patients who have fallen is crucial for reducing damage and mortality. We report the case of a patient who fell from a seated position, which caused traumatic liver injury, with gastrointestinal symptoms as the primary patient concern. CASE REPORT A woman in her 80s who was living independently fell from a seated position during the daytime. She presented to our clinic the next day, reporting vomiting and loose stools. Her vital signs included blood pressure of 100/48 mmHg and a pulse rate of 76 beats/minute, with tenderness over the right chest wall. She was suspected of having a fracture in the absence of X-ray imaging and was advised to rest and treated with analgesia on demand. Subsequent laboratory results showed levels of hemoglobin, alanine transaminase, and lactate dehydrogenase of 98 g/L, 659 U/L, and 429 U/L, respectively. At the hospital, a computed tomography scan revealed severe liver injury and rib fractures. Vascular embolization or surgery was not performed, and hematogenous pleural effusion was drained once via thoracentesis. The hematoma did not enlarge, and the patient's ability to perform activities of daily living gradually improved. She was discharged after 10 days of hospitalization. CONCLUSIONS The patient presented with gastrointestinal symptoms, leading to a delayed diagnosis of severe liver injury. Low-energy trauma can cause fatal injuries, and physicians should assess patients who fall for the risk of organ injuries, while being mindful of potential biases. Additionally, non-specific symptoms after trauma can be associated with organ injury.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"25 ","pages":"e946094"},"PeriodicalIF":1.0,"publicationDate":"2024-12-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11687327/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142898864","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Shih-Hung Wang, Mei-Hui Lee, Yuarn-Jang Lee, Yung-Ching Liu
{"title":"Metapneumovirus-Induced Myocarditis Complicated by Klebsiella pneumoniae Co-Infection: A Case Report.","authors":"Shih-Hung Wang, Mei-Hui Lee, Yuarn-Jang Lee, Yung-Ching Liu","doi":"10.12659/AJCR.946119","DOIUrl":"10.12659/AJCR.946119","url":null,"abstract":"<p><p>BACKGROUND Human metapneumovirus (hMPV), classified in the Pneumoviridae family, is primarily known for causing lower respiratory tract infections in children, the elderly, and immunocompromised individuals. However, rare instances have shown that hMPV can also affect other systems, such as the cardiovascular system, leading to conditions like myocarditis. CASE REPORT We describe a 68-year-old man with a medical history of diabetes, hypertension, and liver cirrhosis who presented to the Emergency Department (ED) exhibiting symptoms of fever, cough, and dyspnea. His condition deteriorated rapidly, progressing to septic shock and requiring increased oxygen support, which led to his transfer to the medical intensive care unit (MICU). Diagnostic evaluations, including cardiac echocardiography and coronary angiography (CAG), confirmed the presence of myocarditis while excluding acute myocardial infarction. Despite aggressive interventions, including extracorporeal membrane oxygenation (ECMO) and intra-aortic balloon pump (IABP) therapy, the patient's condition worsened, and he died 3 days after admission. Polymerase chain reaction (PCR) testing of a throat swab confirmed hMPV infection, with Klebsiella pneumoniae simultaneously identified via sputum culture. The bacterial susceptibility report indicated that the bacteria were sensitive to piperacillin/tazobactam, which had been administered since the patient arrived at our ED, which suggests that the bacterial infection alone cannot fully explain the patient's condition. CONCLUSIONS Compared to previously reported cases of hMPV-related myocarditis, this case is the first to demonstrate notably adverse outcomes associated with the concurrent presence of bacterial infection. The rapid progression and poor outcome despite aggressive treatment emphasize the need for early diagnosis and management of such co-infections.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"25 ","pages":"e946119"},"PeriodicalIF":1.0,"publicationDate":"2024-12-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11687334/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142898868","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Matthew J Folkman, Alexander N Hasselbach, Sarah C Porter, Hamoun Delaviz, Wendy Lackey-Cornelison, Adel Maklad
{"title":"An Accessory Right Hepatic and Cystic Arteries Derived from the Superior Mesenteric Artery: A Cadaveric Case Report.","authors":"Matthew J Folkman, Alexander N Hasselbach, Sarah C Porter, Hamoun Delaviz, Wendy Lackey-Cornelison, Adel Maklad","doi":"10.12659/AJCR.945606","DOIUrl":"10.12659/AJCR.945606","url":null,"abstract":"<p><p>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.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"25 ","pages":"e945606"},"PeriodicalIF":1.0,"publicationDate":"2024-12-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11687329/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142898857","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Efficacy of Neoadjuvant Therapy for ROS1-Positive Locally-Advanced Lung Adenocarcinoma: A Case Report.","authors":"Rong Chen, Guirong Wang, Jinlong Zhao, Yiqun Liu","doi":"10.12659/AJCR.945446","DOIUrl":"10.12659/AJCR.945446","url":null,"abstract":"<p><p>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.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"25 ","pages":"e945446"},"PeriodicalIF":1.0,"publicationDate":"2024-12-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11684347/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142883071","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Axelle Amiel, Théo Van Gucht, Marion Bolliet, Lucie Aussenac-Belle, Patrice David, Laurianne Plastaras, Martin Martinot
{"title":"Selective IgA Deficiency and Aseptic Liver Abscess as Initial Indicators of Crohn's Disease in a Young Woman: A Case Study.","authors":"Axelle Amiel, Théo Van Gucht, Marion Bolliet, Lucie Aussenac-Belle, Patrice David, Laurianne Plastaras, Martin Martinot","doi":"10.12659/AJCR.944829","DOIUrl":"10.12659/AJCR.944829","url":null,"abstract":"<p><p>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.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"25 ","pages":"e944829"},"PeriodicalIF":1.0,"publicationDate":"2024-12-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11684345/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142886167","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}