Congenital Lupus with Multiorgan Involvement: A Case Report and Review of Literature

Padilla-España, L.; Cabrera, Rocío Díaz; Boz, J. del; Calero, Carmen Lozano

doi:10.1111/pde.12450

Cited by 10 publications

(4 citation statements)

References 13 publications

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“… 16 It is, however, extremely rare in neonates, with very few cases reported. 12 , 17 , 18 While interstitial lung disease usually progresses slowly over time, presenting decades after SLE diagnosis, LP, another lupus-related parenchymal disease, is an acute disease that may occur as the initial presentation of SLE. 16 It is one of the rarest pulmonary manifestations in the adult population and case series describe clinical presentation as similar to a respiratory infection, with fever, cough, dyspnea, hypoxemia and pleuritic chest pain, it may also be challenging to distinguish from severe infection and acute respiratory distress.…”

Section: Discussionmentioning

confidence: 99%

Lupus Pneumonitis: Case Report of a Rare Manifestation of Neonatal Lupus

Mota

Santos

Éden

et al. 2022

Journal of Investigative Medicine High Impact Case Reports

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Neonatal lupus is a rare entity, secondary to placental transfer of antibodies from mothers with immune-mediated conditions. While pulmonary involvement is common in lupus, its incidence in neonates is extremely rare, with very few cases being reported in the literature. The authors report a case of a neonate whose mother was diagnosed with systemic lupus erythematosus, with a prenatal diagnosis of third-degree atrioventricular block. While initially admitted in the neonatal intensive care unit with no need for organ support, he presented progressive respiratory failure, initially attributed to sepsis. Favorable clinical progression after pacemaker placement allowed ventilatory weaning, but respiratory failure was again apparent. Chest computer tomography revealed areas of ground-glass lesions, raising the suspicion for lupus pneumonitis. He was started on immunoglobulin and corticosteroids, with clinical improvement. The authors consider this case to be relevant due to the rarity of acute lupus pneumonitis in neonates, alerting that it must be considered a differential diagnosis in neonates with prolonged ventilator dependency.

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Section: Discussionmentioning

confidence: 99%

Lupus Pneumonitis: Case Report of a Rare Manifestation of Neonatal Lupus

Mota

Santos

Éden

et al. 2022

Journal of Investigative Medicine High Impact Case Reports

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“…Neonatal lupus erythematosus (NLE) [2131][2132][2133][2134][2135][2136] may cause neonatal hepatitis syndrome. Hepatic involvement may occur with NLE, which is caused by passage of maternal anti-Ro/SSA and anti-La/SSA antibodies, or occasionally U1-ribonucleoprotein, across the placenta.…”

Section: Neonatal Lupus Erythematosusmentioning

confidence: 99%

Developmental and Inherited Liver Disease

Quaglia¹,

Roberts²,

Torbenson³

2018

Macsween's Pathology of the Liver

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Diagnostic approach to histology 113 Neonatal and early infantile liver disease 113 Postneonatal, childhood and adulthood presentation 118 Neonatal hepatitis 119 Histopathological features 120 Biliary atresia 121 Classification and aetiopathogenesis 121 Pathological features at surgical intervention 124 Pathology of intrahepatic changes 126 Paucity of intrahepatic bile ducts 129 Alagille syndrome (arteriohepatic dysplasia) 129 Nonsyndromic duct paucity disorders 131 Bile duct anomalies, congenital dilations and ductal plate malformation disorders 131 Choledochal cyst 132 Hereditary fibropolycystic disease (ductal plate malformation) 133 Cystic fibrosis 141 Hereditary disorders of bile acid synthesis and bilirubin metabolism 143 Primary disorders of bile acid synthesis 143 Disorders of bile canalicular transporters 146 Other diseases causing intrahepatic cholestasis 150 Hereditary defects of bilirubin metabolism 151 Disorders of porphyrin metabolism 153 Porphyria cutanea tarda 154 Erythropoietic protoporphyria 155 Disorders of carbohydrate metabolism and related conditions 156 Glycogen storage diseases (glycogenoses) 156 Myoclonus epilepsy, Lafora type (Lafora disease) 161 Galactosaemia Hereditary fructose intolerance Disorders of glycoprotein and glycolipid metabolism Mucopolysaccharidoses Aspartylglucosaminuria α-Mannosidosis Fucosidosis Mucolipidoses Congenital disorders of glycosylation (carbohydrate-deficient glycoprotein syndrome) Endoplasmic reticulum storage diseases α1-Antitrypsin deficiency α1-Antichymotrypsin deficiency Afibrinogenaemia and hypofibrinogenaemia Antithrombin III deficiency Disorders of amino acid metabolism Hereditary tyrosinaemia type 1 Congenital hyperammonaemia syndromes and urea cycle disorders Cystinosis Homocystinuria (cystathionine β-synthase deficiency) Disorders of lipoprotein and lipid metabolism Abetalipoproteinaemia Familial hypobetalipoproteinaemia Familial high-density lipoprotein deficiency (Tangier disease) Familial hypercholesterolaemia Wolman disease and cholesteryl ester storage disease Gangliosidoses α-Galactosidase A deficiency (Fabry disease) Sulphatide lipidosis (metachromatic leucodystrophy) Chapter 3 Developmental and Inherited Liver Disease

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“…In 2015, in this journal, we reported a case of an infant of Senegalese descent with congenital lupus erythematosus with multiorgan involvement . We now report on a follow‐up of this infant and the subsequent findings in his younger brother.…”

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confidence: 99%