David R Bayless, Trevor M Caldarera, Hassan M Harirah
{"title":"Autoimmune polyglandular syndrome type 2 in pregnancy: a case report and review of the literature.","authors":"David R Bayless, Trevor M Caldarera, Hassan M Harirah","doi":"10.1515/crpm-2020-0058","DOIUrl":"10.1515/crpm-2020-0058","url":null,"abstract":"<p><strong>Objectives: </strong>Autoimmune polyglandular syndromes are uncommon heterogeneous conditions characterized by the association of two or more organ-specific endocrinopathies. Very few cases of these syndromes have been described during pregnancy. Here we report a case of autoimmune polyglandular syndrome type-2 presenting during pregnancy and complicated by preeclampsia with severe features.</p><p><strong>Case presentation: </strong>The patient is a 35-year-old G7P0242 woman with a history of adrenal insufficiency, type 1 diabetes mellitus, and chronic lymphocytic thyroiditis. She was admitted to our institution at 34 weeks' gestation for preterm contractions, nausea, and lower extremity edema for a few weeks prior to admission. At 35 weeks' gestation, she developed preeclampsia with severe features requiring repeat cesarean section with good maternal and fetal outcomes. Recognizing the occurrence of this rare syndrome during pregnancy, adequate replacement of the deficient hormones, and close maternal and fetal surveillance are essential to achieving favorable outcomes.</p><p><strong>Conclusions: </strong>To our knowledge, this is the first reported case of a pregnant woman with autoimmune polyglandular syndrome type-2 complicated by preeclampsia with severe features.</p>","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":"295 1","pages":"20200058"},"PeriodicalIF":0.1,"publicationDate":"2022-02-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11800655/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"86434497","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":"Didelphys uterus: a successful induction of labor in a case of intrauterine fetal death near term.","authors":"Algeri Paola, D'Oria Patrizia, Toto Valentina, Fenili Paola, Ermito Santina, Bonalumi Silvia, Rinaldo Denise, Ciammella Massimo","doi":"10.1515/crpm-2021-0078","DOIUrl":"10.1515/crpm-2021-0078","url":null,"abstract":"<p><strong>Objectives: </strong>Müllerian anomalies are associated with infertility and worse pregnancy outcomes.</p><p><strong>Case presentation: </strong>A 34-years-old primigravida patient affected by didelphys uterus and type 2 diabetes mellitus was admitted at 36.4 weeks with intrauterine fetal death. Labor was induced with oral Mifepristone and vaginal Dinoprostone. She had an uneventful vaginal delivery.</p><p><strong>Conclusions: </strong>Pre-gestational evaluation should be recommended in each woman, in order to optimize clinical conditions in case of a chronic disease; moreover, if the patient is infertile Müllerian malformations should be excluded. In a didelphys uterus, the combination of Mifepristone and Dinoprostone could be a safe option for labor induction.</p>","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":"11 3 1","pages":"20210078"},"PeriodicalIF":0.1,"publicationDate":"2022-02-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11800674/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"78357272","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":"Postpartum sepsis-like illness and rash associated with Hansen's disease.","authors":"Jinai Bharucha, Lynne Saito-Tom","doi":"10.1515/crpm-2021-0046","DOIUrl":"10.1515/crpm-2021-0046","url":null,"abstract":"<p><strong>Objectives: </strong>Hansen's disease (HD) often manifests during pregnancy and the postpartum. Patients with HD may experience reactions that mimic other conditions making diagnosis challenging.</p><p><strong>Case presentation: </strong>We present a case of a patient from Chuuk, a state of the Federated States of Micronesia with a sepsis-like illness and worsening painful rash immediately postpartum. Antepartum, the patient noted a pruritic rash on her legs. Four hours after delivery, the patient became febrile and later developed systemic inflammatory response syndrome (SIRS). The rash rapidly spread to other areas of her body and became painful and edematous. Eight weeks after delivery, a skin biopsy revealed tuberculoid granulomatous dermatitis consistent with HD.</p><p><strong>Conclusions: </strong>HD and its associated reactions are easily misdiagnosed. Performing a skin biopsy of unusual skin lesions or common skin lesions with severe illness in a pregnant patient can expedite diagnosis of rare conditions such as HD. Early initiation of treatment for HD and its reactions are critical to prevent serious nerve damage and permanent disability.</p>","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":"109 1","pages":"20210046"},"PeriodicalIF":0.1,"publicationDate":"2022-02-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11800671/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"85688983","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}
Sara Bernardes da Cunha, Maria Carolina Fortuna Carneiro, Inês Falcão Reis, Cátia Rasteiro, Augusta Pinto, Teresa Paula Teles
{"title":"Fetal neonatal alloimmune thrombocytopenia treatment with intravenous immunoglobulin: a challenge in pregnancy management and infection assessment ‒ case report.","authors":"Sara Bernardes da Cunha, Maria Carolina Fortuna Carneiro, Inês Falcão Reis, Cátia Rasteiro, Augusta Pinto, Teresa Paula Teles","doi":"10.1515/crpm-2021-0095","DOIUrl":"10.1515/crpm-2021-0095","url":null,"abstract":"<p><strong>Objectives: </strong>Fetal and neonatal alloimmune thrombocytopenia is a rare condition associated with fetal and neonatal morbimortality. Prevention of recurrence includes intravenous immunoglobulin. One challenge in pregnancy surveillance remains the fact that maternal intravenous immunoglobulins therapy can result in false-positive infectious markers. The goal of this case report is to highlight the possible serological misdiagnosed infection associated with intravenous immunoglobulins therapy in pregnancy, and the difficulty of management in this time of a women's life.</p><p><strong>Case presentation: </strong>We report a case of a 38-year-old pregnant woman, with a previous affected child with fetal neonatal alloimmune thrombocytopenia. To prevent recurrence, intravenous immunoglobulin treatment was administered in early second trimester. In the second trimester routine analysis, a positive anti-treponemal test and a toxoplasmosis seroconversion occurred. Infection suspicion based on test positivity of some infectious agents, after passive acquired antibodies, can lead to anxiety and subsequent unnecessary treatment.</p><p><strong>Conclusions: </strong>Clinicians and pathologists must be aware of the possible acquisition of these antibodies during treatment and be able to counsel patients receiving intravenous immunoglobulin. Managing possible infectious intercurrences in pregnancy remains a challenge.</p>","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":"1 1","pages":"20210095"},"PeriodicalIF":0.1,"publicationDate":"2022-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11800660/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"76867659","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}
Diletta Fumagalli, Tommaso Bignardi, Angelo Vanzulli, Paola Francesca Corbella, Mario Giuseppe Meroni, Maria Lieta Interdonato
{"title":"Expectant management of placenta accreta after a mid-trimester pregnancy loss: a case report and a short review.","authors":"Diletta Fumagalli, Tommaso Bignardi, Angelo Vanzulli, Paola Francesca Corbella, Mario Giuseppe Meroni, Maria Lieta Interdonato","doi":"10.1515/crpm-2021-0008","DOIUrl":"10.1515/crpm-2021-0008","url":null,"abstract":"<p><strong>Objectives: </strong>Placenta accreta spectrum (PAS) disorders are a significant cause of maternal morbidity and mortality. Traditionally women with PAS are offered surgery, while expectant management is still considered investigational.</p><p><strong>Case presentation: </strong>We present a case of expectant management of PAS after pregnancy loss at 19-weeks. PAS was suspected at sonography and confirmed by MRI. Patient was offered expectant management to preserve fertility. This consisted of leaving the placenta <i>in situ</i>, followed by in- and out-patient clinical and sonographic examinations and blood tests. After five weeks placental detachment occurred without major complications.</p><p><strong>Conclusions: </strong>Our report suggests that expectant management could be a safe option in selected cases of PAS after mid-trimester pregnancy loss. We recommend expectant management should be offered in referral centers for PAS.</p>","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":"29 1","pages":"20210008"},"PeriodicalIF":0.1,"publicationDate":"2022-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11800657/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"75497455","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":"Coarctation of the aorta and persistent left superior vena cava: HDlive Flow features at 14 weeks of gestation.","authors":"Toshiyuki Hata, Aya Koyanagi, Riko Takayoshi, Takahito Miyake, Yuichiro Nakai, Kazumasa Tani, Kei Hayata, Hisashi Masuyama","doi":"10.1515/crpm-2021-0066","DOIUrl":"10.1515/crpm-2021-0066","url":null,"abstract":"<p><strong>Objectives: </strong>A significant discrepancy between a large ductus arteriosus and a smaller aorta at their connection is key to diagnose coarctation of the aorta (CoA) at 14-16 weeks of gestation. CoA was associated with persistent left superior vena cava (PLSVC) in 21.3% of fetuses. HDlive Flow findings for CoA or PLSVC were obtained only in the third trimester of pregnancy. To the best of our knowledge, there has been no report on the prenatal findings of CoA and PLSVC using HDlive Flow with spatiotemporal image correlation (STIC) before 20 weeks of gestation.</p><p><strong>Case presentation: </strong>We present the trans-abdominal HDlive Flow features of CoA and PLSVC at 14 weeks of gestation. With a three-vessel trachea view on multiplanar view using color Doppler with STIC, PLSVC on the left side of the pulmonary artery was noted, and a narrowing aortic isthmus was suspected. A narrowing isthmus was also suspected with an aortic arch view. HDlive Flow clearly showed the spatial relationships among the right superior vena cava, aorta with narrowing isthmus, pulmonary artery, and PLSVC. A preductal 'shelf' was also suspected. No other fetal anomaly was noted. Neonatal echocardiography after delivery confirmed CoA and PLSVC.</p><p><strong>Conclusions: </strong>To the best of our knowledge, this is the first report on HDlive Flow features of fetal CoA and PLSVC using STIC early in the second trimester of pregnancy.</p>","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":"105 1","pages":"20210066"},"PeriodicalIF":0.1,"publicationDate":"2022-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11800681/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"80709344","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}
Leonor Castro, Ana C Ferreira, Álvaro Cohen, Israel J Macedo, Teresa Tomé
{"title":"Preterm twins with antenatal presentation of Pearson syndrome.","authors":"Leonor Castro, Ana C Ferreira, Álvaro Cohen, Israel J Macedo, Teresa Tomé","doi":"10.1515/crpm-2021-0083","DOIUrl":"10.1515/crpm-2021-0083","url":null,"abstract":"<p><strong>Objectives: </strong>Pearson syndrome is a mitochondrial cytopathy with multisystemic involvement that typically presents in infancy and has poor prognosis. We aim to present a case that is distinct due to the timing of presentation and associated anomalies.</p><p><strong>Case presentation: </strong>We report the case of preterm monochorionic twins with transfusion dependent fetal anemia that had post-natal multisystem dysfunction which led to the diagnosis of Pearson syndrome.</p><p><strong>Conclusions: </strong>This case highlights the possibility of antenatal presentation of Pearson syndrome, which should be considered in cases of severe fetal anemia without an apparent cause.</p>","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":"4 1","pages":"20210083"},"PeriodicalIF":0.1,"publicationDate":"2022-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11800656/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"73869661","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":"Stress degree demonstrated in mothers with phenylketonuria or hyperphenylalaninemia infant when requested for total or partial breastfeeding replacement","authors":"K. Schulpis, K. Iakovou","doi":"10.1515/crpm-2019-0079","DOIUrl":"https://doi.org/10.1515/crpm-2019-0079","url":null,"abstract":"Abstract Obejctives Phenylketonuria (PKU) and hyperphenylalaninemia (HPHE) are metabolic disorders of phenylalanine (phe) metabolism due to the liver enzyme phe hydroxylase deficiency resulting in mental retardation. The only treatment of these disorders is the total or the partial breastfeeding replacement with special formula, respectively. Case presentation We aimed to evaluate the maternal stress degree in the two groups of mothers and compare the stress degree between mothers who were obliged to replace breastfeeding completely with those who replaced it partially. Mothers (n=50) were divided into two groups: n=25 with PKU and n=25 with HPHE infants. Mothers with PKU newborns were obliged to replace breastfeeding completely and feed their newborns with a common formula plus phe free medical food, whereas mothers with HPHE infants partially replaced breastfeeding with phe free medical food. Stress degrees were calculated using special questionnaire checklist by Bourne E. Psychological support was provided for both groups of mothers with stress. Conclusions Most mothers with PKU infants experienced high or severe stress, whereas mothers with HPHE newborns suffered from low and moderate symptoms and signs of stress. After psychological support most mothers with PKU newborns were ameliorated from stress symptoms; mothers with HPHE neonates experienced almost no stress symptoms. Mothers with PKU infants obliged to complete breastfeeding replacement experienced high and/or severe stress degrees as compared to mothers with HPHE newborns with partial breastfeeding replacement. Psychological support resulted in amelioration of both tested groups.","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":"1 1","pages":""},"PeriodicalIF":0.1,"publicationDate":"2021-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"90389052","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Kelly Ribeiro, S. Mahboobani, K. van Ree, K. Clifford, T. Teoh
{"title":"Postpartum ovarian vein thrombosis","authors":"Kelly Ribeiro, S. Mahboobani, K. van Ree, K. Clifford, T. Teoh","doi":"10.1515/crpm-2021-0002","DOIUrl":"https://doi.org/10.1515/crpm-2021-0002","url":null,"abstract":"Abstract Objectives Postpartum ovarian vein thrombosis (POVT) is a rare pathology that can lead to severe complications such as sepsis, extension of the thrombus leading to organ failure, and pulmonary embolism. It therefore requires early recognition and prompt treatment. Case presentation A patient with right POVT presented four days after delivery with acute right-sided abdominal pain and fever. Appendicitis was initially considered, before an abdominal-pelvic computed tomography raised the suspicion of POVT, subsequently confirmed through transabdominal ultrasound. Antibiotics and anticoagulation were initiated, with rapid clinical improvement and complete resolution of the thrombus three months later. Conclusions Diagnosing POVT is challenging as it clinically mimics other more frequent conditions. It is rare but life-threatening and should be considered in all females presenting with abdominal pain and fever in the postpartum period.","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":"131 1","pages":""},"PeriodicalIF":0.1,"publicationDate":"2021-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"74987596","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
G. Tran, A. Nguyen, Ha Xuan Nam, Tareq Mohammed Ali Al-Ahdal, Ranjit Tiwari, Hảo Nguyễn Thị, Jaffer Shah, N. Huy, T. N. Pham
{"title":"Case report: The first COVID-19 case among pregnant women at 21-week in Vietnam","authors":"G. Tran, A. Nguyen, Ha Xuan Nam, Tareq Mohammed Ali Al-Ahdal, Ranjit Tiwari, Hảo Nguyễn Thị, Jaffer Shah, N. Huy, T. N. Pham","doi":"10.1515/crpm-2020-0067","DOIUrl":"https://doi.org/10.1515/crpm-2020-0067","url":null,"abstract":"Abstract Objectives This is the report of the first pregnant woman having COVID-19 in Vietnam. The patient was at 21 weeks of gestation and a high level of serum D-dimer and fibrinogen was observed from admission day to day 5. Case presentation The patient had lived in Russia before returning to Vietnam where she was diagnosed with COVID-19. Clinical characteristics of this patient were described and there was no major derangement of laboratory parameters. She was asymptomatic when being diagnosed and then developed mild symptoms of COVID-19 after three days of admission. The patient was treated with lopinavir/ritonavir, ampicillin/sulbactam, and Lovenox and continued being hospitalized until April 25th, 2020. Conclusions We presented the clinical picture of the first COVID-19 pregnant case in Vietnam. This case report highlighted the increased risk of developing coagulopathy even in mild and asymptomatic patients.","PeriodicalId":9617,"journal":{"name":"Case Reports in Perinatal Medicine","volume":"80 1","pages":""},"PeriodicalIF":0.1,"publicationDate":"2021-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"83945519","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}