AJP Reports最新文献

筛选
英文 中文
Wounds to Wisdom: Exploring Risk Factors and Outcomes for Surgically Managed Postcesarean Wound Infections. 从伤口到智慧:探索剖宫产术后伤口感染的危险因素和结果。
IF 0.6
AJP Reports Pub Date : 2026-02-09 eCollection Date: 2026-01-01 DOI: 10.1055/a-2789-1655
Janice Wong, Emma E H Peek, Ysaac Zegeye, Ashley Cummings, Mia Grayson, Caitlin Moran, Sara Bernate Angulo, Carmen Rauh Garrido, Jennifer Okunbor, Rachel Wood, Sarah K Dotters-Katz
{"title":"Wounds to Wisdom: Exploring Risk Factors and Outcomes for Surgically Managed Postcesarean Wound Infections.","authors":"Janice Wong, Emma E H Peek, Ysaac Zegeye, Ashley Cummings, Mia Grayson, Caitlin Moran, Sara Bernate Angulo, Carmen Rauh Garrido, Jennifer Okunbor, Rachel Wood, Sarah K Dotters-Katz","doi":"10.1055/a-2789-1655","DOIUrl":"10.1055/a-2789-1655","url":null,"abstract":"<p><strong>Objective: </strong>Postcesarean surgical site infections (SSIs) contribute substantially to morbidity and healthcare costs, yet understanding of their management remains limited.</p><p><strong>Study design: </strong>Retrospective cohort study of patients delivering via cesarean at a single healthcare system from June 2013 to July 2022 with SSIs within 30 days of delivery. Rates and risk factors for surgical intervention were examined as the primary outcome. Secondary analysis evaluated outcomes in those who required surgical versus conservative management.</p><p><strong>Results: </strong>Of 533 patients, 69 (12.9%) required surgical management; this population was less likely to have private insurance and more likely to have diabetes than patients managed conservatively. Factors independently associated with surgical intervention included body mass index (BMI) 40 to 49.9, BMI ≥ 50, hypertensive disorders of pregnancy, blood transfusion, general anesthesia, and penicillin allergy. Among 297 patients evaluated, patients requiring surgical intervention( <i>n</i>  = 69, 23.2%) experienced higher rates of morbidity, including sepsis, acute kidney injury, and fascial dehiscence. Patients requiring surgical intervention had higher rates of inpatient admission, intensive care unit admission, and longer readmissions.</p><p><strong>Conclusion: </strong>Patients with higher BMI, hypertensive disorders of pregnancy, general anesthesia, blood transfusion, and penicillin allergies may warrant closer monitoring for wound infection. Furthermore, patients requiring surgical intervention for postpartum wound infections had higher morbidity and longer, more complex hospitalizations.</p>","PeriodicalId":7645,"journal":{"name":"AJP Reports","volume":"16 1","pages":"e43-e49"},"PeriodicalIF":0.6,"publicationDate":"2026-02-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12885764/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146155628","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}
引用次数: 0
Risk Stratification of Postpartum Patients with Hypertensive Disorders of Pregnancy to Telehealth Follow-Up: A Quality Improvement Project. 妊娠期高血压疾病产后患者远程健康随访的风险分层:质量改进项目
IF 0.6
AJP Reports Pub Date : 2026-02-02 eCollection Date: 2026-01-01 DOI: 10.1055/a-2788-9872
Ariana M Banuelos, Kalin Ellison, Weirui Xiao, Patricia Chavez, Diana S Wolfe, Anna E Bortnick, Kavita Vani
{"title":"Risk Stratification of Postpartum Patients with Hypertensive Disorders of Pregnancy to Telehealth Follow-Up: A Quality Improvement Project.","authors":"Ariana M Banuelos, Kalin Ellison, Weirui Xiao, Patricia Chavez, Diana S Wolfe, Anna E Bortnick, Kavita Vani","doi":"10.1055/a-2788-9872","DOIUrl":"10.1055/a-2788-9872","url":null,"abstract":"<p><strong>Background: </strong>Guidelines recommend a 3-day follow-up for severe hypertensive disorders of pregnancy (HDP) and a 7- to 10-day follow-up for nonsevere HDP, but implementation varies.</p><p><strong>Objective: </strong>To improve adherence to guideline-recommended postpartum follow-up by targeting provider discharge recommendations. A risk stratification tool incorporating HDP severity, maternal symptoms, and discharge blood pressure guided providers to recommend telehealth follow-up at 3, 5, or 7 days. We aimed to maintain 3-day recommendations for severe HDP and reduce unnecessary 3-day recommendations for nonsevere HDP.</p><p><strong>Study design: </strong>This quality improvement project was conducted at a single urban academic institution. The risk stratification tool was integrated into discharge workflows, and demographic, clinical, and telehealth follow-up data for pre- and post-intervention cohorts were abstracted from the electronic medical record and compared using summary statistics and bivariate analyses.</p><p><strong>Results: </strong>Cohorts were similar at baseline. After implementation, all patients with severe HDP continued to receive 3-day follow-up instructions. Among patients with nonsevere HDP patients, 3-day recommendations decreased from 100 to 31.1% ( <i>p</i>  < 0.001), with a corresponding decrease in 3-day telehealth scheduling. Scheduling for severe HDP did not improve.</p><p><strong>Conclusion: </strong>The tool improved guideline-aligned provider recommendations for postpartum HDP. Scheduling changes were limited, suggesting future efforts should target workflow and system processes.</p>","PeriodicalId":7645,"journal":{"name":"AJP Reports","volume":"16 1","pages":"e36-e42"},"PeriodicalIF":0.6,"publicationDate":"2026-02-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12863972/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146111938","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}
引用次数: 0
Thyroid Storm in a Twin Pregnancy Presenting as Severe Hypertension and Preterm Labor. 甲状腺风暴在双胎妊娠表现为严重高血压和早产。
IF 0.6
AJP Reports Pub Date : 2026-01-29 eCollection Date: 2026-01-01 DOI: 10.1055/a-2788-1788
Adwoa A Baffoe-Bonnie, Lillian J Dubiel, Lillian B Boettcher, Maria Martinez Cruz, Amanda M Craig
{"title":"Thyroid Storm in a Twin Pregnancy Presenting as Severe Hypertension and Preterm Labor.","authors":"Adwoa A Baffoe-Bonnie, Lillian J Dubiel, Lillian B Boettcher, Maria Martinez Cruz, Amanda M Craig","doi":"10.1055/a-2788-1788","DOIUrl":"10.1055/a-2788-1788","url":null,"abstract":"<p><strong>Background: </strong>Thyroid storm is a rare, life-threatening endocrine emergency in pregnancy with significant maternal and neonatal implications. Clinical symptoms may mimic those of hypertensive disorders in pregnancy. Prompt diagnosis and multidisciplinary management are critical for reducing maternal and neonatal morbidity and mortality.</p><p><strong>Case: </strong>A 31-year-old woman with a dichorionic diamniotic twin pregnancy, chronic hypertension, short cervix, and poorly controlled Graves' disease presented at 22 weeks' gestation with vaginal bleeding and severe-range blood pressures. She was diagnosed with thyroid storm and admitted to the intensive care unit. She failed to respond to maximal medical management, ultimately requiring plasmapheresis. She later developed preterm labor and spontaneously delivered periviable twins. She eventually underwent total thyroidectomy for definitive management of thyroid storm.</p><p><strong>Conclusion: </strong>This case illustrates the severe consequences of poorly controlled Graves' disease in pregnancy. Although thyroid storm can mimic preeclampsia, abnormal thyroid function tests, persistent tachycardia, fevers, and hypertension with wide pulse pressures may help distinguish it. Timely recognition and multidisciplinary care are imperative to decrease morbidity and mortality associated with thyroid storm.</p>","PeriodicalId":7645,"journal":{"name":"AJP Reports","volume":"16 1","pages":"e30-e35"},"PeriodicalIF":0.6,"publicationDate":"2026-01-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12854848/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146103625","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}
引用次数: 0
Pregnancy and Poikiloderma with Neutropenia. 妊娠和嗜中性粒细胞减少的千皮病。
IF 0.6
AJP Reports Pub Date : 2026-01-22 eCollection Date: 2026-01-01 DOI: 10.1055/a-2764-3829
Jessica Nelson, Hailey Cox, Ira Hamilton, Nauman Khurshid, Nikolina Docheva
{"title":"Pregnancy and Poikiloderma with Neutropenia.","authors":"Jessica Nelson, Hailey Cox, Ira Hamilton, Nauman Khurshid, Nikolina Docheva","doi":"10.1055/a-2764-3829","DOIUrl":"10.1055/a-2764-3829","url":null,"abstract":"<p><strong>Background: </strong>Poikiloderma with neutropenia is an autosomal recessive condition characterized by postinflammatory poikiloderma and chronic neutropenia. To date, there is no published literature reporting on the impact of pregnancy on this rare genetic disorder.</p><p><strong>Case: </strong>This case highlights a successful pregnancy outcome in a 26-year-old gravida 1 para 0 female with poikiloderma with neutropenia. Her pregnancy was complicated by thrombocytopenia, low fetal fraction, and high-risk cell-free DNA result.</p><p><strong>Discussion: </strong>The findings of low fetal fraction and high-risk aneuploidy were unexpected given normal neonatal outcome. It is not yet well understood whether these findings are related to the genetic condition itself or use of medications used to manage her condition.</p>","PeriodicalId":7645,"journal":{"name":"AJP Reports","volume":"16 1","pages":"e27-e29"},"PeriodicalIF":0.6,"publicationDate":"2026-01-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12826880/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146049940","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}
引用次数: 0
Associated Factors Relating to the Success of Labor Induction among Pregnant Women Diagnosed with Fetal Intrauterine Growth Restriction: A Retrospective Cohort Study from France. 诊断为胎儿宫内生长受限的孕妇引产成功的相关因素:来自法国的回顾性队列研究
IF 0.6
AJP Reports Pub Date : 2026-01-16 eCollection Date: 2026-01-01 DOI: 10.1055/a-2764-2369
Phuc Nhon Nguyen, Anna Ramos
{"title":"Associated Factors Relating to the Success of Labor Induction among Pregnant Women Diagnosed with Fetal Intrauterine Growth Restriction: A Retrospective Cohort Study from France.","authors":"Phuc Nhon Nguyen, Anna Ramos","doi":"10.1055/a-2764-2369","DOIUrl":"10.1055/a-2764-2369","url":null,"abstract":"<p><strong>Objective: </strong>This study aimed to identify factors related to the successful induction of labor (IOL) among pregnancies complicated with fetal growth restriction (FGR).</p><p><strong>Methods: </strong>We performed a retrospective cohort study between January and December 2024 at Orléans University Hospital, Orléans, France. The study enrolled all singleton pregnancies complicated by FGR and underwent IOL. Logistic regression was used to reveal the associated factors relating to the success of IOL, following two criteria, including Bishop score after IOL greater than 7 points (criterion 1) and vaginal birth (criterion 2).</p><p><strong>Results: </strong>Bishop score had a negative correlation with IOL duration. This study did not find statistically significant clinical factors related to the success of the Bishop score change of more than 7 points. However, absence of Doppler abnormalities and IOL at term, more than 37 weeks, were related to the success rate of vaginal delivery (OR [95% CI]: 22.5 [3.240-156.269] and OR [95% CI]: 22.5 [3.240-156.269], respectively; <i>p</i>  < 0.05).</p><p><strong>Conclusion: </strong>Bishop score before IOL helps in reducing the duration of IOL. In FGR pregnancies with a normal Doppler ultrasound scan and gestational age at IOL greater than 37 weeks, these are good prognostic factors for vaginal delivery; however, further research is needed to explore more associated factors.</p>","PeriodicalId":7645,"journal":{"name":"AJP Reports","volume":"16 1","pages":"e18-e26"},"PeriodicalIF":0.6,"publicationDate":"2026-01-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12811059/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145996999","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}
引用次数: 0
Group B Streptococcus Sepsis with Cardiac Abscesses in a Neonate with Migrated Umbilical Catheter: Literature Review. B组脓毒症合并心脏脓肿的新生儿与移动脐带导管:文献回顾。
IF 0.6
AJP Reports Pub Date : 2026-01-15 eCollection Date: 2026-01-01 DOI: 10.1055/a-2770-5011
Trenton Judd, Mimily Harsono, Jie Zhang, Fatima Mir, Massroor Pourcyrous
{"title":"Group B <i>Streptococcus</i> Sepsis with Cardiac Abscesses in a Neonate with Migrated Umbilical Catheter: Literature Review.","authors":"Trenton Judd, Mimily Harsono, Jie Zhang, Fatima Mir, Massroor Pourcyrous","doi":"10.1055/a-2770-5011","DOIUrl":"10.1055/a-2770-5011","url":null,"abstract":"<p><p>Group B <i>Streptococcus</i> (GBS) is the leading cause of sepsis, pneumonia, and/or meningitis in neonates. Insertion of an umbilical catheter (UC) is a common practice in neonatal intensive care for primary central vascular access in extreme premature neonates. UC is used for the administration of intravenous medications, parenteral nutrition, blood samplings, and continuous central blood pressure monitoring. Malposition or migration of UC tends to occur in extreme premature infants with risks of multiple complications. We present a case of an extreme premature neonate who developed fatal GBS sepsis with autopsy findings of multiple cardiac abscesses in the myocardium but not in any other organ. GBS sepsis with isolated multifocal myocardial abscesses leading to sudden fatal clinical deterioration has not been described previously. In this review, we describe the plausible pathological mechanism of this rare presentation. Intracardiac migrated UC, in conjunction with rhythmic heart contractions and intracardiac blood flow dynamics, can cause direct trauma to the endocardium. Damaged endocardium can be a potential nidus for bacterial overgrowth and abscess formation that ultimately may lead to cardiac failure. Therefore, the correct aseptic technique of securing and management of UC, and daily assessment of UC position are recommended to prevent complications associated with catheter migration.</p>","PeriodicalId":7645,"journal":{"name":"AJP Reports","volume":"16 1","pages":"e10-e17"},"PeriodicalIF":0.6,"publicationDate":"2026-01-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12807581/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145996991","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}
引用次数: 0
Successful Use of Recombinant Tissue Plasminogen Activator in an Extremely Low Birth Weight Premature Infant for the Resolution of Multiple Thrombi. 重组组织型纤溶酶原激活剂在极低出生体重早产儿中用于解决多发性血栓的成功应用。
IF 0.6
AJP Reports Pub Date : 2026-01-14 eCollection Date: 2026-01-01 DOI: 10.1055/a-2780-3344
Kartik Mody, Christine Wade, Becky Micetic, Myleanna Vernon, Natalie Wade
{"title":"Successful Use of Recombinant Tissue Plasminogen Activator in an Extremely Low Birth Weight Premature Infant for the Resolution of Multiple Thrombi.","authors":"Kartik Mody, Christine Wade, Becky Micetic, Myleanna Vernon, Natalie Wade","doi":"10.1055/a-2780-3344","DOIUrl":"10.1055/a-2780-3344","url":null,"abstract":"<p><strong>Introduction: </strong>The literature is limited regarding the use of recombinant tissue plasminogen activator (r-tPA) in premature infants. We describe the use of r-tPA to treat life-threatening intra- and extracardiac thrombi in a very low birth weight patient born at 23 weeks of gestational age.</p><p><strong>Case report: </strong>An extremely premature infant was diagnosed with multiple thrombi at 4 weeks of age. The acute phase of treatment was managed with an infusion of r-tPA followed by unfractionated heparin, then low-molecular-weight heparin for continued anticoagulation. The patient did not experience any side effects associated with the therapies and was discharged home.</p><p><strong>Conclusion: </strong>Thrombotic events in neonates, though rare, are being increasingly identified due to improved survival of premature infants, enhanced diagnostic modalities, and the widespread use of central venous catheters. Heparin remains the standard of care in neonates for anticoagulation. Thrombolytic therapy with agents such as r-tPA, though less frequently employed, may be lifesaving in certain instances, such as the one presented in this case report.</p>","PeriodicalId":7645,"journal":{"name":"AJP Reports","volume":"16 1","pages":"e1-e6"},"PeriodicalIF":0.6,"publicationDate":"2026-01-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12803789/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145987719","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}
引用次数: 0
Pregnancy-Induced Isolated Severe Vaginal Varicosities: A Case Report and Literature Review. 妊娠引起的孤立性严重阴道静脉曲张1例报告及文献复习。
IF 0.6
AJP Reports Pub Date : 2026-01-14 eCollection Date: 2026-01-01 DOI: 10.1055/a-2765-7948
Hanna Brancaccio, Itishree Panda, Isabelle Crary, Asha Rijhsinghani
{"title":"Pregnancy-Induced Isolated Severe Vaginal Varicosities: A Case Report and Literature Review.","authors":"Hanna Brancaccio, Itishree Panda, Isabelle Crary, Asha Rijhsinghani","doi":"10.1055/a-2765-7948","DOIUrl":"10.1055/a-2765-7948","url":null,"abstract":"<p><strong>Objective: </strong>During pregnancy, total blood volume increases by about 40%. As gestation advances, blood volume to the pelvic region increases significantly. Due to the enlarging uterus, the venous return from the lower body decreases with advancing gestational age. The pooling and stasis of blood in the lower body can lead to the formation of varices, affecting the lower extremity, vulva, and vaginal regions. Varicosities in the lower extremities and vulva are not uncommon and often asymptomatic. Vulvar varicosities are more common in women with varicosities in the lower extremities. On the other hand, vaginal varicosities are extremely rare and mentioned only in case reports. Although asymptomatic, vaginal varicosities could become a concern for hemorrhage risk during vaginal delivery. There is little information in the current medical literature about the diagnosis and management of large vaginal varicosities during pregnancy.</p><p><strong>Study: </strong>Design We present a case of a primigravida with very large prolapsing vaginal varicosities that presented as a large external mass at 36 weeks of gestation.</p><p><strong>Results: </strong>The patient was managed expectantly during the antenatal period. She was delivered via an elective scheduled cesarean section. An almost complete resolution of the vaginal varices was noted at 6 hours postoperatively, with complete resolution reported on postoperative day 22.</p><p><strong>Conclusion: </strong>Due to the rarity of the condition, we hope to add our experience to the literature.</p>","PeriodicalId":7645,"journal":{"name":"AJP Reports","volume":"16 1","pages":"e7-e9"},"PeriodicalIF":0.6,"publicationDate":"2026-01-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12803788/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145987766","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}
引用次数: 0
Fatal Neonatal Echovirus 11 Infection Following Maternal Peripartum Illness: A Case Report with Literature Review. 围产期疾病后新生儿致死性埃可病毒11型感染1例并文献复习。
IF 0.6
AJP Reports Pub Date : 2025-12-30 eCollection Date: 2025-10-01 DOI: 10.1055/a-2764-2405
Ming-Ju Wang, Chia-Chen Lee, Hung-Yang Chang, Chie-Pein Chen
{"title":"Fatal Neonatal Echovirus 11 Infection Following Maternal Peripartum Illness: A Case Report with Literature Review.","authors":"Ming-Ju Wang, Chia-Chen Lee, Hung-Yang Chang, Chie-Pein Chen","doi":"10.1055/a-2764-2405","DOIUrl":"10.1055/a-2764-2405","url":null,"abstract":"<p><p>Neonatal Echovirus 11 (E-11) infection acquired perinatally often manifests as a severe, sepsis-like illness with rapid clinical deterioration within the first week of life. The infection is characterized by fulminant multiorgan failure, commonly involving severe hepatitis with coagulopathy, myocarditis, and meningoencephalitis. Adverse outcomes are strongly associated with prematurity and vertical transmission from a mother with an acute peripartum infection, which precludes the transfer of protective serotype-specific antibodies. We present a fatal case of E-11 sepsis in a late preterm neonate following maternal peripartum illness.</p>","PeriodicalId":7645,"journal":{"name":"AJP Reports","volume":"15 4","pages":"e181-e185"},"PeriodicalIF":0.6,"publicationDate":"2025-12-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12753186/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145877350","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}
引用次数: 0
Sirolimus for Recurrent Chylothorax and Edema in an Infant with Noonan Syndrome after Resolved Hydrops Fetalis: A Case Report. 西罗莫司治疗Noonan综合征患儿胎儿水肿后复发性乳糜胸和水肿1例。
IF 0.6
AJP Reports Pub Date : 2025-12-30 eCollection Date: 2025-10-01 DOI: 10.1055/a-2770-4952
Mariko Hattori, Kei Tamai, Hirokazu Watanabe, Akihito Takeuchi, Makoto Nakamura, Michio Ozeki, Misao Kageyama
{"title":"Sirolimus for Recurrent Chylothorax and Edema in an Infant with Noonan Syndrome after Resolved Hydrops Fetalis: A Case Report.","authors":"Mariko Hattori, Kei Tamai, Hirokazu Watanabe, Akihito Takeuchi, Makoto Nakamura, Michio Ozeki, Misao Kageyama","doi":"10.1055/a-2770-4952","DOIUrl":"10.1055/a-2770-4952","url":null,"abstract":"<p><strong>Background: </strong>Noonan syndrome (Online Mendelian Inheritance in Man #163950) is a RASopathy caused by germline mutations in the RAS/RAF/mitogen-activated protein kinase signaling pathway and characterized by distinctive facial features, musculoskeletal abnormalities, and congenital heart defects. A subset of patients with <i>RIT1</i> mutations present with hypertrophic cardiomyopathy and generalized lymphatic anomalies. While sirolimus, a mammalian target of rapamycin inhibitor (mTOR), has been shown to suppress lymphangiogenesis, its efficacy in Noonan syndrome remains unclear.</p><p><strong>Case presentation: </strong>We report the case of an infant with Noonan syndrome and <i>RIT1</i> mutation who developed recurrent refractory chylothorax and edema. Despite multiple interventions, including corticosteroids, octreotide, thoracic duct ligation, and pleurodesis, the patient's condition remained critical and refractory. Skin biopsy revealed lymphatic malformations. Sirolimus (0.6 mg/day) was initiated at 220 days of age, but was discontinued due to a markedly elevated serum level (88.2 ng/mL) and a lack of therapeutic effect. The patient died of <i>Escherichia coli</i> sepsis at 235 days of age.</p><p><strong>Conclusion: </strong>Although sirolimus was ineffective in this case, initiation of treatment at a lower dose may be advisable for patients with compromised hepatic function or concurrent infections. Further studies are warranted to clarify the appropriate indications, dosage, and timing of sirolimus therapy for Noonan syndrome.</p>","PeriodicalId":7645,"journal":{"name":"AJP Reports","volume":"15 4","pages":"e186-e188"},"PeriodicalIF":0.6,"publicationDate":"2025-12-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12753187/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145877320","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}
引用次数: 0
0
×
引用
GB/T 7714-2015
复制
MLA
复制
APA
复制
导出至
BibTeX EndNote RefMan NoteFirst NoteExpress
×
提示
您的信息不完整,为了账户安全,请先补充。
现在去补充
×
提示
您因"违规操作"
具体请查看互助需知
我知道了
×
提示
确定
请完成安全验证×
相关产品
×
本文献相关产品
联系我们:info@booksci.cn Book学术提供免费学术资源搜索服务,方便国内外学者检索中英文文献。致力于提供最便捷和优质的服务体验。 Copyright © 2023 布克学术 All rights reserved.
京ICP备2023020795号-1
ghs 京公网安备 11010802042870号
Book学术文献互助
Book学术文献互助群
群 号:604180095
Book学术官方微信
小红书