AJP Reports最新文献

{"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}

{"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}

{"title":"Aggressive Chemotherapy and Antepartum Management of Small-Cell Carcinoma of the Ovary of Hypercalcemic Type: A Novel Case Report with Ethical and Medical Decision-Making Consideration.","authors":"Hind N Moussa, Dana Rector, Wesley Gherman, Eric Shuffle, Robert Fresch, Thomas Reid","doi":"10.1055/a-2764-3698","DOIUrl":"10.1055/a-2764-3698","url":null,"abstract":"<p><strong>Background: </strong>Small-cell carcinoma of the ovary, hypercalcemic type (SCCOHT) is a rare, aggressive, and highly fatal gynecologic malignancy with few treatment guidelines offered in nonpregnant patients.</p><p><strong>Case report: </strong>A 32-year-old woman presented for initial prenatal care at 9 weeks and her dating ultrasound was notable for a large complex adnexal mass. Magnetic resonance imaging performed 2 weeks later revealed accelerated interval growth with possible malignant etiology. She underwent surgical evaluation with salpingo-oophorectomy. Pathology noted signature features of SCCOHT. The patient desired expectant management and declined termination of pregnancy or chemotherapy. Four weeks later, she developed severe abdominal pain, nausea, and vomiting, and she was diagnosed with extensive metastatic disease. After multidisciplinary and multi-institutional counseling, she elected for continuation of her pregnancy and 6-agent chemotherapy including Vinblastine, Cisplatin, Cyclophosphamide, Bleomycin, Doxorubicin, Etoposide (VPCBAE) with close antepartum surveillance. The patient completed multiple rounds of chemotherapy and subsequently delivered via primary cesarean at 27 ^2/7 weeks due to maternal sepsis and nonreassuring fetal status. Maternal and neonatal death occurred approximately 1-week postpartum.</p><p><strong>Conclusion: </strong>This is the first known case of SCCOHT in which the pregnancy was continued through an aggressive chemotherapy regimen.</p>","PeriodicalId":7645,"journal":{"name":"AJP Reports","volume":"15 4","pages":"e175-e180"},"PeriodicalIF":0.6,"publicationDate":"2025-12-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12747791/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145861643","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":"Giant Cervicothoracic Cystic Lymphangioma in a Neonate: Clinical Challenges and Management Considerations.","authors":"Marina A Bustamante-Ordoñez, Gianelli S Curi-Vilchez, Eduardo Marchand-Bayona, Carlos Zavaleta-Corvera, Angel Samanez-Obeso","doi":"10.1055/a-2739-1840","DOIUrl":"10.1055/a-2739-1840","url":null,"abstract":"<p><strong>Background: </strong>Lymphangiomas are benign vascular malformations of the lymphatic system, commonly affecting the head and neck. Neonatal cases pose significant clinical and surgical challenges.</p><p><strong>Case presentation: </strong>We report a premature female neonate (35 weeks of gestation) with a large cervicothoracic cystic lymphangioma (8 × 7 cm), causing respiratory distress and vascular compression. Imaging revealed an extensive lymphatic malformation (160 × 67 × 87 mm), affecting the sternocleidomastoid muscle, salivary glands, and adjacent vasculature. Surgical resection achieved 90% tumor removal while preserving neurovascular structures. Postoperative complications included transient neuromotor deficit of the right upper limb and <i>Candida lusitaniae</i> sepsis. Despite intensive care, tumor progression led to respiratory failure, and the patient succumbed 12 days' postsurgery.</p><p><strong>Discussion: </strong>Extensive cervicothoracic lymphangiomas in neonates require complex management. Imaging aids in surgical planning, delineating tumor extent, and complications. While surgical resection is the standard treatment, it carries risks in neonates with large lesions. Sclerotherapy has shown efficacy in macrocystic lymphangiomas but remains debated for extensive neonatal cases. A multidisciplinary approach is crucial to optimize outcomes.</p><p><strong>Conclusion: </strong>Neonatal cervicothoracic lymphangiomas pose significant challenges. Surgery remains primary, but sclerotherapy may be considered in selected cases. Multidisciplinary management is essential to improve prognosis and reduce morbidity.</p>","PeriodicalId":7645,"journal":{"name":"AJP Reports","volume":"15 4","pages":"e169-e174"},"PeriodicalIF":0.6,"publicationDate":"2025-11-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12623118/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145547831","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":"Confirming Rupture of Membranes with Intra-Amniotic Fluorescein Dye Test.","authors":"Katherine Freedy, Sarah K Dotters-Katz, Bobby May, Sloane Mebane, Virginia Watkins, Matthew R Grace, Jennifer Gilner","doi":"10.1055/a-2724-5458","DOIUrl":"10.1055/a-2724-5458","url":null,"abstract":"<p><strong>Introduction: </strong>We describe two patients presenting with preterm loss of fluid and inconclusive evaluations requiring further assessment. Patient 1 was a 41-year-old G3P1011 at 21 ^6/7 weeks; Patient 2 was a 22-year-old G2P1001 at 31 ^5/7 weeks. In both, preterm prelabor rupture of membranes (PPROM) workups yielded mixed results, prompting intra-amniotic dye testing. Due to a national indigo carmine shortage, sodium fluorescein was used. We present photographs of tampons examined under UV light, confirming PPROM in both cases.</p><p><strong>Methods: </strong>Under ultrasound guidance, 5 cc of sodium fluorescein was injected into the amniotic cavity. Patients wore a tampon for 15 minutes while ambulating. Both provided consent for publication.</p><p><strong>Results: </strong>On direct visualization, tampons appeared normal. Under UV light (Wood's lamp), the fluorescein emitted a bright neon green fluorescence. Both specimens demonstrated photoluminescence, confirming PPROM.</p><p><strong>Conclusion: </strong>Although the use of 2 to 5 cc of fluorescein for intra-amniotic dye testing is described in the literature, visual documentation of positive results is limited. These images may guide clinicians in confirming PPROM when indigo carmine is unavailable, supporting fluorescein as a viable diagnostic alternative.</p><p><strong>Key points: </strong>Fluorescein dye can be used to confirm PPROM when tampons are examined under UV light.We share unique images of positive fluorescein dye tests to confirm PPROM diagnosis.Images may aid diagnosis when indigo carmine is unavailable and fluorescein is used.</p>","PeriodicalId":7645,"journal":{"name":"AJP Reports","volume":"15 4","pages":"e166-e168"},"PeriodicalIF":0.6,"publicationDate":"2025-11-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12606055/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145511227","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":"A Neonate with FNAIT Supported by Placental Chronic Histiocytic Intervillositis and Confounded by Maternal Preeclampsia: A Case Report.","authors":"Hannah White, Amelia Sybenga","doi":"10.1055/a-2703-4068","DOIUrl":"10.1055/a-2703-4068","url":null,"abstract":"<p><strong>Introduction: </strong>Fetal and neonatal alloimmune thrombocytopenia (FNAIT) occurs in the setting of maternal anti-human platelet antigen (anti-HPA) antibodies against paternally derived fetal platelet antigens. Recent studies have also demonstrated an association between chronic placental inflammation and FNAIT, specifically low-grade chronic histiocytic intervillositis (CHI). We present a neonate with profound thrombocytopenia after delivery with co-occurring CHI, whose platelet counts recovered rapidly with platelet transfusions, born to a primigravida mother with late-onset preeclampsia.</p><p><strong>Case report: </strong>A male neonate was born at 40 weeks to a mother who had no known history of pregnancies, miscarriages, or transfusions. The mother developed severe preeclampsia during the induction of labor. Shortly after delivery, a physical exam of the infant showed inappropriate bruising on the heels of both feet, scattered petechiae on the hard palate, a hematoma on the left thigh after a vitamin K shot, and a bruise on the upper left abdomen. His platelet count was found to be 7,000. Platelet count rose to 94K by day 3 of life following transfusions. Placental pathology confirmed CHI. Maternal testing revealed anti-HPA-1 antibodies supporting FNAIT.</p><p><strong>Conclusion: </strong>This case highlights a potential relationship between maternal alloimmune response and preeclampsia. It also highlights the importance of considering FNAIT as a diagnosis in a neonate presenting with thrombocytopenia regardless of maternal preeclampsia, and the importance of submitting the placenta for a pathology exam.</p>","PeriodicalId":7645,"journal":{"name":"AJP Reports","volume":"15 3","pages":"e163-e165"},"PeriodicalIF":0.6,"publicationDate":"2025-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12488233/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145211322","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":"Corrigendum: Fetal Cardiac Collapse Diagnosed By Umbilical Venous Flow Volume After Thoraco-Amniotic Shunting for Severe Pleural Effusion.","authors":"Yuichiro Takahashi, Shigenori Iwagaki, Kazuhiko Asai, Masako Matsui, Ryuichi Shimaoka, Hitomi Ono, Saki Inuzuka","doi":"10.1055/a-2706-6895","DOIUrl":"https://doi.org/10.1055/a-2706-6895","url":null,"abstract":"<p><p>[This corrects the article DOI: 10.1055/a-2697-2212.].</p>","PeriodicalId":7645,"journal":{"name":"AJP Reports","volume":"15 3","pages":"e162"},"PeriodicalIF":0.6,"publicationDate":"2025-09-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12473515/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145184546","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":"Corrigendum: Noninvasive Maternal-Fetal Hemodynamic Monitoring as A Predictor of Severe Preeclampsia in Low-Resource Settings: A Case Report.","authors":"Lilian Toledo-Jaldin, Richard Gomez, Litzi Lazo-Vega, Alison Larrea, Adolfo Vásquez, Wilson Ormachea-Orellana, Valquiria Miranda-Garrido, Colleen G Julian","doi":"10.1055/a-2698-8526","DOIUrl":"https://doi.org/10.1055/a-2698-8526","url":null,"abstract":"<p><p>[This corrects the article DOI: 10.1055/a-2689-2550.].</p>","PeriodicalId":7645,"journal":{"name":"AJP Reports","volume":"15 3","pages":"e154"},"PeriodicalIF":0.6,"publicationDate":"2025-09-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12445976/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145111918","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":"Fetal Cardiac Collapse Diagnosed By Umbilical Venous Flow Volume After Thoraco-Amniotic Shunting for Severe Pleural Effusion.","authors":"Yuichiro Takahashi, Shigenori Iwagaki, Kazuhiko Asai, Masako Matsui, Ryuichi Shimaoka, Hitomi Ono, Saki Inuzuka","doi":"10.1055/a-2697-2212","DOIUrl":"10.1055/a-2697-2212","url":null,"abstract":"<p><strong>Objective: </strong>Although thoraco-amniotic shunting (TAS) for severe pleural effusion is an effective fetal treatment, there are some cases in which it deteriorates, showing circulatory collapse. To evaluate the usefulness of umbilical venous blood flow volume (UVFV) for predicting deterioration, we analyzed the fetal low UVFV situation.</p><p><strong>Methods: </strong>In 22 cases of fetal severe pleural effusion, we measured UVFV/fetal estimated birth weight (mL/minute/kg) prospectively before and after TAS by ultrasonography. We defined low UVFV/kg as < 50 mL/minute/kg (2.5 percentile) and compared subgroups based on their UVFV value and analyzed the outcome after birth.</p><p><strong>Results: </strong>Total survival rate was 59% at 6 months. Seven cases in the low group before delivery (UVFV/kg 19.5) showed poor prognoses, such as fetal/neonatal death and longer neonatal intensive care unit management (100% vs. the normal UVFV group 40%, <i>p</i>  = 0.017). The low group also showed umbilical artery absent end-diastolic velocity (71%); edema resolved in 50%, suggesting hypo inflow from the placenta and fetal hypocardiac output status, revealing fetal cardiac collapse.</p><p><strong>Conclusion: </strong>UVFV analyses would be a new marker of fetal management of severe pleural effusion, suggesting low UVFV after TAS seems to be hypovolemic cardiac collapse and shows poor prognosis, and we had better consider immediate delivery to prevent death even after TAS.</p>","PeriodicalId":7645,"journal":{"name":"AJP Reports","volume":"15 3","pages":"155-e161"},"PeriodicalIF":0.7,"publicationDate":"2025-09-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12440646/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145079526","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":"Alloimmunization in Pregnancy: Patient-reported Quality of Care, Mental Health Effects, and Impact Upon Daily Life.","authors":"Molly R Sherwood, Bethany M Weathersby, Marion E Granger, Kathryn A Shanahan, Kara B Markham","doi":"10.1055/a-2690-9547","DOIUrl":"10.1055/a-2690-9547","url":null,"abstract":"<p><strong>Objective: </strong>The purpose of this study was to investigate mental health and impacts upon daily life in patients with a history of pregnancy alloimmunization, and secondarily to examine the relationship between disease severity and quality of care on these outcomes.</p><p><strong>Study design: </strong>This was a survey administered between November 2022 and February 2023 to U.S. adults with a history of red cell alloimmunization in pregnancy. Mental health outcomes, quality of care, and daily life impacts were reported.</p><p><strong>Results: </strong>The survey was completed by 127 alloimmunized adults. Anxiety (90.6%), guilt (74.8%), self-doubt (68.0%), isolation (71.4%), depression (68.3%), and symptoms of posttraumatic stress disorder (PTSD) (61.3%) were frequently reported. Mental health support was offered in 24.4%. Respondents reporting a high quality of care in their alloimmunized pregnancies (rating of 8/10 or higher) were significantly less likely than those who perceived receiving lower-quality care to report feelings of guilt ( <i>p</i>  = 0.0006), self-doubt ( <i>p</i>  = 0.04), depression ( <i>p</i>  = 0.03), and symptoms of PTSD ( <i>p</i>  = 0.001).</p><p><strong>Conclusion: </strong>Despite the pervasive patient burden, mental health support was infrequently offered, and patients reported low satisfaction with continuity of care and their providers' knowledge of alloimmunization. Clinicians interacting with alloimmunized patients must employ a comprehensive patient-centered approach to address the significant disease burden.</p><p><strong>Key points: </strong>The impact of alloimmunization in pregnancy on patients' mental health and daily living has not been evaluated.Females with a history of alloimmunization in pregnancy reported widespread anxiety (115/127; 90.6%), guilt (95/127; 74.8%), self-doubt (85/127; 68.0%), isolation (90/127; 71.4%), depression (86/127; 68.3%), and symptoms of PTSD (76/127; 61.3%); mental health support was offered in 24.4% of the sample (31/127). Respondents reporting a high quality of care (rating of 8/10 or higher) were significantly less likely to report feelings of guilt ( <i>p</i>  = 0.0006), self-doubt ( <i>p</i>  = 0.04), depression ( <i>p</i>  = 0.03), and symptoms of PTSD ( <i>p</i>  = 0.001). This study highlights a need to utilize patient-centered comprehensive care models to support this rare high-risk population. Bolstered clinician knowledge and referral to peer support networks, mental health counselors, and social workers may prove highly beneficial to this patient population.</p>","PeriodicalId":7645,"journal":{"name":"AJP Reports","volume":"15 3","pages":"e146-e153"},"PeriodicalIF":0.6,"publicationDate":"2025-09-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12417028/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145028702","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}