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1.
Conservative Management of Gestational Hypercalcemia Due to Primary Hyperparathyroidism with Lack of Complications.
Di Bari, F, Vita, R, Marini, H, Borrielli, I, Lo Re, C, Panebianco, V, Benvenga, S
Endocrine, metabolic & immune disorders drug targets. 2021;(8):1512-1517
Abstract
INTRODUCTION Primary hyperparathyroidism (PHPT) is rare in pregnancy. PHPT and hypercalcemia are associated with negative maternofetal outcomes. Therefore, an early diagnosis and adequate treatment are essential. CASE PRESENTATION We described the case of a pregnant woman complaining of nausea, vomiting and weight loss. Diagnosis of gestational PHPT (GPHPT) was made based on elevated serum calcium and parathyroid hormone levels (3.4 mmol/L and 41.6 pmol/L). Neck ultrasound documented a nodule suggestive of enlarged parathyroid, whereas the abdomen ultrasound revealed renal microlithiasis. Conservative treatment was started with oral hydration and a low-calcium diet. Clinical and biochemical monitoring was weekly and multidisciplinary. Despite our suggestion, the patient refused parathyroidectomy in the second trimester. Additional intravenous fluid rehydration from the 15th to the 25th week of gestation ameliorated the symptoms rapidly, and reduced calcium levels progressively from the 23rd week. At week 40, the woman gave birth to a healthy girl. At month 8 postpartum, calcemia and PTH were still elevated, and accompanied by osteoporosis and nephrocalcinosis. Surgery was accepted, and a parathyroid adenoma was removed. CONCLUSION In the absence of guidelines for GPHPT management, its treatment should be individualized. In our case, despite high calcium levels, conservative treatment with strict monitoring led to a positive outcome of pregnancy.
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2.
Preeclampsia and low sodium (PALS): A case and systematic review.
Powel, JE, Rosenthal, E, Roman, A, Chasen, ST, Berghella, V
European journal of obstetrics, gynecology, and reproductive biology. 2020;:14-20
Abstract
Normal physiologic changes in pregnancy include mild hyponatremia. In some cases of preeclampsia, more significant hyponatremia has been associated with syndrome of inappropriate antidiuretic hormone secretion and hypervolemic hyponatremia. A 45-year-old gravida 2, para 0010 with a dichorionic twin gestation was diagnosed with preeclampsia at 30 weeks 6 days and noted to have concomitant hyponatremia of 125 mEq/L at our institution. Her hyponatremia was initially managed with furosemide and water restriction. She was delivered at 33 weeks 5 days due to worsening preeclampsia and continued significant hyponatremia despite treatment. Her hyponatremia resolved within 48 h after delivery. Our objectives were to discuss trends, treatment, and outcomes of cases with hyponatremia in preeclampsia. We performed a systematic review of the literature using Ovid Medline (1963-2017), Scopus (1962-2017), and PubMed (1963-2017, including Cochrane database). Relevant articles describing any case report of hyponatremia in preeclampsia were identified from the above databases without any time, language, or study limitations. Studies were deemed eligible for inclusion if they described a case of hyponatremia in the setting of preeclampsia. 18 manuscripts detailing 55 cases were identified. Pertinent demographic data and laboratory values were extracted. Maternal management strategy, diagnosis, delivery, and neonatal outcome data were also collected. Mean, range, standard deviation, and percentage calculations were used as applicable. Advanced maternal age (46 %), nulliparity (79 %), and multifetal gestation (34 %) were noted in patients with preeclampsia and low sodium. Hyponatremia was detected on average at 34 weeks gestation. 64 % were diagnosed with preeclampsia with severe features. When reported, diagnoses related to hyponatremia were syndrome of inappropriate antidiuretic hormone secretion (41 %) or hypervolemic hyponatremia (59 %). Indications for delivery included severe hyponatremia unresponsive to conservative measures in addition to other known obstetric or preeclamptic indications. Hyponatremia resolved within 48 h on average in cases where postpartum resolution was reported. It may be prudent to screen women with preeclampsia for electrolyte disturbances as part of their evaluation, especially in the setting of severe features. Initially, hyponatremia may be treated with medical management. In addition to established obstetric or preeclamptic indications, delivery may be considered if severe hyponatremia no longer responds to conservative measures.
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3.
Increased creatine demand during pregnancy in Arginine: Glycine Amidino-Transferase deficiency: a case report.
Alessandrì, MG, Strigini, F, Cioni, G, Battini, R
BMC pregnancy and childbirth. 2020;(1):506
Abstract
BACKGROUND Creatine (Cr), an amino acid derivative, is one of the most important sources of energy acting as both a spatial and temporal energy buffer through its phosphorylated analogue phosphocreatine (PCr) and creatine kinase (CK). Maternal Cr biosynthesis and metabolism seem to play an important role in pregnancy, as shown in preclinical and in healthy human pregnancy studies. Patients with Arginine:Glycine Amidino-Transferase deficiency (AGAT-d), due to the deficit of the first enzyme involved in Cr synthesis, are at a disadvantage due to their failure to synthesize Cr and their dependence on external intake, in contrast to normal subjects, where changes in Cr biosynthesis supply their needs. We report the outcomes of a pregnancy in an AGAT-d woman, and the challenge we faced in managing her treatment with oral Cr to ensure optimal conditions for her fetus. CASE PRESENTATION A 22-year-old AGAT-d woman referred to our Institute for the management of her first conception at 11 weeks of fetal gestational age. Sonographic monitoring at 20 w GA indicated a reduction of fetal growth, in particular of the head circumference that was below the 3rd centile. Biochemical monitoring of Cr in biological fluids of the mother revealed a decline of the Cr concentrations, in particular in the urine sample, requiring prompt correction of the Cr dose. At 35 weeks of gestation the patient delivered a male infant, heterozygous for GATM mutation, with normal brain Cr levels; at one year the baby achieved typical developmental milestones. CONCLUSIONS This rare pregnancy demonstrates that Cr levels in the blood and urine of the mother with AGAT-d decreased since the first months of gestation. The increase of the Cr daily dose administered to the mother seems to have produced beneficial effects also on the fetus.
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4.
Brief report: Metabolic acidosis in newborn infants following maternal use of acetazolamide during pregnancy.
Ibrahim, A, Hussain, N
Journal of neonatal-perinatal medicine. 2020;(3):419-425
Abstract
The information regarding fetal effects of acetazolamide use during pregnancy and lactation is sparse. We report the clinical and pharmacodynamic characteristics of maternal acetazolamide use and the timing of its effects on acid-base balance in three cases who presented with metabolic acidosis in the newborn period. We found that the infants' clinical status soon after birth was inconsistently correlated with maternal drug dose and concentrations of medication in maternal serum. However, there was low transfer of the drug in breast milk and its use did not affect clinical symptomatology. We also present a review of literature on this subject to help consolidate our current knowledge on this topic.
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5.
A case of pregnancy and lactation-associated osteoporosis and a review of the literature.
Jia, P, Wang, R, Yuan, J, Chen, H, Bao, L, Feng, F, Tang, H
Archives of osteoporosis. 2020;(1):94
Abstract
INTRODUCTION The pregnancy and lactation-associated osteoporosis (PLO) is a rare disease whose precise pathophysiological mechanisms remain mostly unknown. CASE REPORT We reported here a case of PLO that occurred in the early postpartum period and led to multiple compression fractures. Combination therapy with alendronate, calcium carbonate, and vitamin D was used to treat the patient and a marked but gradual increase in the density of bone mineral was observed. Moreover, no further fractures have occurred. CONCLUSION PLO is a very rare type of osteoporosis associated with severe chronic back pain. Increased bone resorption significantly increases the risk of bone fractures in women with PLO. Early diagnosis, stopping breastfeeding, treatment of calcium and vitamin D, bisphosphonates, or other antiosteoporosis medicine and regular follow-ups of these cases are particularly important in the prevention of fractures and to increase the quality of life of patients.
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6.
Neuroleptic malignant syndrome in pregnancy: case report and literature review.
Escobar-Vidarte, MF, Loaiza-Osorio, S, Messa, AA, Macías, GE
The journal of maternal-fetal & neonatal medicine : the official journal of the European Association of Perinatal Medicine, the Federation of Asia and Oceania Perinatal Societies, the International Society of Perinatal Obstetricians. 2019;(14):2438-2441
Abstract
BACKGROUND Neuroleptic malignant syndrome (NMS) is a serious complication associated with the use of drugs that affect dopaminergic system neurotransmission. The occurrence of NMS during pregnancy or gestation is considered a life-threatening obstetric emergency. CASE We are reporting the first case in Latin America of NMS in one pregnant women with acute psychotic episode. One day after starting with antipsychotic therapy, she developed a fever higher than 39.0 °C with tachycardia, tachypnea, generalized muscle rigidity and somnolence, with creatine kinase (CPK) levels evidencing a result of 2800 U/L. She was treated successfully with levetiracetam, biperiden and quetiapine. DISCUSSION A search in PubMed, Embase and Ovid from 1988 to 2016 resulted in seven cases reported in either pregnant or puerperal women. In general, NMS resolves within 3-14 days; most NMS cases reported during pregnancy have involved the use of haloperidol (5 case reports) which is concordant with this report. The obstetric results were good in cases reported, only two women showed signs, among them: hyperemesis gravidarum and preterm delivery. Most of the pregnant women who had NMS presented other associated comorbidities, being mostly of infectious origin. In other investigations, it has been affirmed that NMS can become lethal in adults; however, in our search for pregnant women with this disease, no associated mortality was found. CONCLUSIONS NMS is seen infrequently during pregnancy. The clinical diagnosis requires high suspicion by the examiner. It is important that obstetricians timely recognize the condition.
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7.
Successful pregnancy in maple syrup urine disease: a case report and review of the literature.
Grünert, SC, Rosenbaum-Fabian, S, Schumann, A, Schwab, KO, Mingirulli, N, Spiekerkoetter, U
Nutrition journal. 2018;(1):51
Abstract
BACKGROUND Maple syrup urine disease (MSUD) is an autosomal recessive disorder of branched-chain amino acid metabolism. Patients with MSUD are at risk of life-threatening metabolic decompensations with ketoacidosis and encephalopathy. These episodes are often triggered by physiological stress. Only few cases of pregnancies in MSUD mothers have been reported so far. CASE PRESENTATION We present the favorable outcome of a pregnancy in a woman with classical MSUD. She presented in the metabolic outpatient clinic in week 7 of gestation. Branched-chain amino acid concentrations were measured at least weekly to adjust dietary leucine intake. Despite excellent compliance, leucine concentrations frequently exceeded the target value of < 300 μmol/L during the first trimester. From the second trimester until delivery, protein and leucine intake increased continuously to about threefold compared to pre-pregnancy values. To maximize patient safety during delivery and the postpartum period, a detailed plan including peripartal infusion therapy, dietary recommendations and monitoring parameters was developed. Primary Caesarean section was performed in week 38 of gestation, and the patient gave birth to a healthy girl. Lactation was successfully implemented. Leucine levels were maintained within the target range throughout the complete postpartum period. In addition to our case, we give an overview about all cases of pregnancies in MSUD mothers published so far. CONCLUSIONS Management of pregnancy, delivery, postpartum period and lactation may be challenging in patients with MSUD. Careful monitoring and interdisciplinary collaboration is essential to minimize the risk of metabolic crisis, especially after delivery.
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8.
The medical management of Cushing's syndrome during pregnancy.
Lim, WH, Torpy, DJ, Jeffries, WS
European journal of obstetrics, gynecology, and reproductive biology. 2013;(1):1-6
Abstract
Cushing's syndrome during pregnancy is a rare metabolic condition that is associated with high maternal and foetal morbidity. Clinical symptoms may mimic those of normal pregnancy. A diagnosis is best made based on clinical presentation, laboratory and imaging findings as well as a high index of suspicion. Medical management with anti-steroidogenic agents such as metyrapone has been shown to be effective, but surgery is usually the recommended treatment option. Its main limitation is optimal timing of the procedure in late first trimester or early second trimester to prevent spontaneous termination of pregnancy. We describe our experience and management of a 39-year-old patient with uncontrolled hypertension at 25 weeks gestation which was later diagnosed as ACTH independent Cushing's syndrome and had a favourable pregnancy outcome. The role of medical therapy and its challenges, as well as its impact on pregnancy outcomes, were explored by a literature search conducted through Pubmed and Medline databases. A total of 12 patients with Cushing's syndrome during pregnancy were reported to have been managed with metyrapone, with ketoconazole being studied to a significant degree in three cases. Of these women, 53% delivered close to term and 20% developed pre-eclampsia. Despite two neonatal deaths and one stillborn reported, medical management appeared effective in controlling hypercortisolemia during pregnancy with strict monitoring of blood pressure and foetal surveillance. It remains the only active management in the setting of pregnancy-induced Cushing's syndrome, and has shown to be a viable option in controlling serum cortisol levels especially as an adjunct to surgery as reflected in four cases. A multidisciplinary approach towards an individualised management process is warranted with medical management to ensure a safe maternal and foetal outcome.
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9.
Maternal antibody-associated fetal second-degree heart block and atrial flutter: case report and review.
Sacks, JH, Samai, C, Gomez, K, Kanaan, U
Pediatric cardiology. 2013;(8):2040-3
Abstract
Exposure to maternal anti-Ro (SS-A) and anti-La (SS-B) antibodies is a well-described risk factor for the development of fetal atrioventricular (AV) block. The role of maternal fluorinated steroids in the treatment and prevention of antibody-mediated fetal AV block is controversial. Fetal atrial flutter has rarely been described in association with maternal antibodies. This report describes a case of fetal exposure to maternal anti-Ro antibodies with associated second-degree AV block and atrial flutter. Interestingly, the reported patient had 2:1 AV conduction during both normal atrial rates (consistent with AV node conduction disease) and episodes of flutter (consistent with physiologic AV node functionality). The fetus was treated with transplacental digoxin and dexamethasone, which resolved both rhythm disturbances. The case report is followed by a brief discussion of AV block and atrial flutter associated with maternal antibody exposure.
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10.
Differing obstetric outcomes of rosacea fulminans in pregnancy: report of three cases with review of pathogenesis and management.
Jarrett, R, Gonsalves, R, Anstey, AV
Clinical and experimental dermatology. 2010;(8):888-91
Abstract
Rosacea fulminans (RF) is a rare facial dermatosis characterized by the sudden onset of severe facial inflammation consisting of numerous pustules, cystic swellings and coalescing sinuses. The standard treatment is the retinoid drug isotretinoin in combination with systemic corticosteroids or with high-dose oral tetracycline antibiotics. We report three recent cases of RF in pregnancy with differing obstetric outcomes: an intrauterine death, a termination of pregnancy, and a normal vaginal delivery. The pathogenesis of RF is considered and therapeutic options in pregnancy are reviewed.