scholarly journals Transient congenital hyperinsulinism and hemolytic disease of a newborn despite rhesus D prophylaxis: a case report

2021 ◽  
Vol 15 (1) ◽  
Author(s):  
Sandra Simony Tornoe Riis ◽  
Marianne Hoerby Joergensen ◽  
Kristina Fruerlund Rasmussen ◽  
Steffen Husby ◽  
Jane Preuss Hasselby ◽  
...  
Keyword(s):  
Case Report ◽  
Rare Complication ◽  
Blood Type ◽  
Congenital Hyperinsulinism ◽  
Hemolytic Disease ◽  
Fetomaternal Hemorrhage ◽  
Elevated Liver Enzymes ◽  
Conjugated Hyperbilirubinemia ◽  
Immunoglobulin Prophylaxis ◽  
Positive Direct

Abstract Background In neonates, rhesus D alloimmunization despite anti-D immunoglobulin prophylaxis is rare and often unexplained. Rhesus D alloimmunization can lead to hemolytic disease of the newborn with anemia and unconjugated hyperbilirubinemia. In past reports, transient congenital hyperinsulinism has been described as a rare complication of rhesus D alloimmunization. Our case report illustrates that rhesus D alloimmunization can result in a pseudosyndrome with severe congenital hyperinsulinism, anemia, and conjugated hyperbilirubinemia, despite correctly administered anti-D immunoglobulin prophylaxis. Case presentation We report of a 36-year-old, Caucasian gravida 1, para 1 mother with A RhD negative blood type who received routine antenatal anti-D immunoglobulin prophylaxis. Her full term newborn boy presented with severe congenital hyperinsulinism, anemia, and conjugated hyperbilirubinemia up to 295 µmol/L (ref. < 9), accounting for 64% of the total bilirubin. Syndromic congenital hyperinsulinism was suspected. Examinations showed a positive direct antiglobulin test, initially interpreted as caused by irregular antibodies; diffuse congenital hyperinsulinism by 18F-DOPA positron emission tomography/computed tomography scan; normal genetic analyses for congenital hyperinsulinism; mildly elevated liver enzymes; delayed, but present bile excretion by Tc99m-hepatobiliary iminodiacetic acid scintigraphy; and cholestasis and mild fibrosis by liver biopsy. The maternal anti-D titer was 1:16,000 day 20 postpartum. Y-chromosome material in the mother’s blood could not be identified. This could, however, not exclude late intrapartum fetomaternal hemorrhage as the cause of immunization. No causative genetic findings were deetrmined by trio whole exome sequencing. The child went into clinical remission after 5.5 months. Conclusion Our case demonstrates that rhesus D alloimmunization may present as a pseudosyndrome with transient congenital hyperinsulinism, anemia, and inspissated bile syndrome with conjugated hyperbilirubinaemia, despite anti-D immunoglobulin prophylaxis, possibly due to late fetomaternal hemorrhage.

Anti-D-induced severe hemolytic disease of the newborn in an Omani newborn born a rhesus-positive mother

2018 ◽  
Vol 7 (1) ◽  
Author(s):  
Hanan F. Nazir ◽  
Huda Al Harrasi ◽  
Saif Al Hosni ◽  
Abdulhakeem Al Rawas ◽  
Arwa Al-Riyami ◽  
...  
Keyword(s):  
Literature Review ◽  
Neonatal Jaundice ◽  
Rare Complication ◽  
Blood Transfusions ◽  
Hemolytic Disease ◽  
Positive Mother ◽  
Antiglobulin Test ◽  
Rh Incompatibility ◽  
Positive Direct ◽  
Hb Concentration

Abstract Hemolytic disease of the fetus and newborn (HDFN) due to anti-D antibodies is a well-known complication of rhesus (Rh) incompatibility, encountered in D-positive babies born to alloimmunized D-negative mothers who have been sensitized during previous labor or abortion. Here, we report a case of significant hemolytic disease of the newborn due to the presence of anti-D antibodies in an Rh-positive baby born to an Rh-positive mother. The boy presented at day 1 of life with neonatal jaundice and required intensive phototherapy. His hemoglobin (Hb) concentration gradually dropped from 17 g/dL to 6.6 g/dL. The blood bank workup revealed O Rh-positive blood group, with a positive direct antiglobulin test (DAT) and confirmed the presence of anti-D antibodies. His mother was typed as O Rh positive, with a negative DAT and positive anti-D. He required two blood transfusions, and his Hb stabilized at the age of 7 weeks. Anti-D HDFN is a rare complication of Rh-positive and Rh-weak positive pregnancies. The lack of awareness of this phenomenon is often a source of confusion for clinicians. A literature review of similar cases and possible explanations are discussed.

scholarly journals Serological profile and hemolytic disease in term neonates with ABO incompatibility

2009 ◽  
Vol 49 (4) ◽  
pp. 219 ◽  
Author(s):  
Desiana Dharmayani ◽  
Djajadiman Gatot ◽  
Rinawati Rohsiswatmo ◽  
Bambang Tridjaja
Keyword(s):  
Blood Type ◽  
Hemolytic Disease ◽  
Chi Square ◽  
Cross Sectional ◽  
Term Neonates ◽  
Serological Profile ◽  
Abo Incompatibility ◽  
Elution Method ◽  
Positive Direct ◽  
Chi Square Analysis

Background Hemolytic disease of the newborn (HDN) due toABO blood type incompatibility is one of the most commoncause of neonatal hyperbilirubunemia that potentially leads tobilirubin encephalopathy. Data on ABO-hemolytic disease of thenewborn (ABO-HDN), especially regarding umbilical cord bloodserological profile, are limited.Objective To identify the serological profile and hemolytic disease in term neonates with ABO incompatibility.Methods This was a cross-sectional descriptive study, conductedat RSIA Budi Kemuliaan Jakarta.Results We found 68 healthy term neonates with ABOincompatibility, nine of them had positive direct antiglobulintest (OAT) result, and 38 subjects had a positive result onOAT with elution method. The highest titer of IgG was 1:8.Hyperbilirubinemia was found in 30 ( 44%) subjects, andABO-HDN was diagnosed in 28 (41 %) subjects. Within thepositive OAT group, eight out of nine subjects had sufferedfrom hyperbilirubinemia and ABO-HDN. Meanwhile, withinthe positive OAT with elution method group; 24 subjects hadsuffered from hyperbilirubinemia with 23 of them having ABOHDN. Based on the chi-square analysis; those with positive OAT with elution method had 3.2 times higher risk of suffering from ABO-HDN. Furthermore, there was a 3.6 times higher risk ofsufferring from hyperbilirubinemia.Conclusion In healthy term neonates with ABO incompatibility,the incidence of ABO-HDN is 41%. OAT serological examinationwith elution method is better than OAT in assessing riskfor hyperbilirubinemia and ABO-HDN.

A case report on management of a rare complication of abdominal sling surgerypresacral venous bleeding

2019 ◽  
Vol 10 (2) ◽  
pp. 19-21
Author(s):  
Shirish S Dulewad ◽  
◽  
Pooja Chandak ◽  
Madhura Pophalkar ◽  
◽  
...  
Keyword(s):  
Case Report ◽  
Rare Complication ◽  
Venous Bleeding

scholarly journals A227 UMBILICAL HERNIA INCARCERATION IN LIVER CIRRHOSIS: A RARE COMPLICATION OF PARACENTESIS

2021 ◽  
Vol 4 (Supplement_1) ◽  
pp. 263-265
Author(s):  
A LAGROTTERIA ◽  
A Aruljothy ◽  
K Tsoi
Keyword(s):  
Liver Cirrhosis ◽  
Abdominal Pain ◽  
Case Report ◽  
Small Bowel ◽  
Large Volume ◽  
Umbilical Hernia ◽  
Rare Complication ◽  
Abdominal Wall Hernia ◽  
Refractory Ascites ◽  
Large Volume Paracentesis

Abstract Background Patients with decompensated liver cirrhosis with ascites frequently have umbilical hernias with a prevalence of 20% and are managed with large volume paracentesis (LVP). Common complications of LVP include hemorrhage, infection, and bowel perforation that occur infrequently with a frequency of less than 1%. However, incarceration of umbilical hernias has been reported as a rare complication of LVP and is speculated to be from ascitic fluid decompression that reduces the umbilical hernia ring diameter resulting in entrapment of the hernia sac. It is unclear whether the quantity or the fluid removal rate increases the herniation risk. Based on case series, this rare complication occurs within 48 hours of the LVP and requires emergent surgical repair and involves a high risk of morbidity and mortality due to potential infection, bleeding, and poor wound healing. Aims We describe a case report of an incarcerated umbilical hernia following a bedside large-volume paracentesis. Methods Case report Results A 59-year-old Caucasian male presented to the emergency department with a 24-hour history of acute abdominal pain following his outpatient LVP. His medical history included Child-Pugh class C alcoholic liver cirrhosis with refractory ascites managed with biweekly outpatient LVP and a reducible umbilical hernia. He reported the onset of his abdominal pain 2-hours after his LVP with an inability to reduce his umbilical hernia. Seven liters of clear, straw-coloured asitic fluid was drained. Laboratory values at presentation revealed a hemoglobin of 139 g/L, leukocyte count of 4.9 x109 /L, platelet count of 110 xo 109 /L, and a lactate of 2.7 mmol/L His physical exam demonstrated an irreducible 4 cm umbilical hernia and bulging flanks with a positive fluid wave test. Abdominal computed tomography showed a small bowel obstruction due to herniation of a proximal ileal loop into the anterior abdominal wall hernia, with afferent loop dilation measuring up to 3.4 cm. He was evaluated by the General Surgery consultation service and underwent an emergent laparoscopic hernia repair. There was 5 cm of small bowel noted to be ecchymotic but viable, with no devitalized tissue. He tolerated the surgical intervention with no post-operative complications and was discharged home. Conclusions Ultrasound-guided bedside paracentesis is a common procedure used in the management of refractory ascites and abdominal wall hernia incarceration should be recognized as a potential rare complication. To prevent hernia incarceration, patients with liver cirrhosis should be examined closely for hernias and an attempt should be made for external reduction prior to LVP. A high index of suspicion for this potential life-threatening condition should be had in patients who present with symptoms of bowel obstruction following a LVP. Funding Agencies None

scholarly journals Hydrops fetalis with isolated massive ascites in a preterm neonate with rhesus disease

2021 ◽  
Author(s):  
Nasenien Nourkami-Tutdibi ◽  
Martina Geipel ◽  
Gabriele Meyberg-Solomayer ◽  
Zoltan Takacs ◽  
Sascha Meyer
Keyword(s):  
Intensive Care ◽  
Case Report ◽  
Neonatal Intensive Care ◽  
Preterm Neonate ◽  
Fluid Collection ◽  
Hemolytic Disease ◽  
Massive Ascites ◽  
Rhesus Incompatibility ◽  
Trained Staff ◽  
Clinical Stabilization

SummarySignificant progress in prenatal care has decreased the incidence of rhesus incompatibility, which may result in hemolytic disease of the fetus and newborn (HDFN). This case report describes an unusual presentation of HDFN in a preterm infant delivered by caesarean section with isolated massive abdominal fluid collection as the leading clinical sign in addition to severe anemia. The immediate drainage of ascites provided transient clinical stabilization with improved pulmonary function in the delivery suite. After admission to the neonatal intensive care unit (NICU), HDFN treatment was initiated. This case report shows the importance of adequately trained staff including neonatologists, pediatricians and NICU nurses in the delivery suite to provide neonatal intensive care for HDFN.

scholarly journals A case report of colouterine fistula due to colonic diverticulitis: a rare complication

2020 ◽  
Vol 51 (1) ◽  
Author(s):  
Shweta Pandey ◽  
Saurabh Maheshwari ◽  
Uddandam Rajesh ◽  
Darshan Singh Grewal ◽  
Vibhuti Maria
Keyword(s):  
Case Report ◽  
Rare Complication ◽  
Colonic Diverticulitis ◽  
Colouterine Fistula
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