CNS vasculitis and stroke in neonatal lupus erythematosus: A case report and review of literature

Saini, Arushi Gahlot; Sankhyan, Naveen; Bhattad, Sagar; Vyas, Sameer; Saikia, Biman; Singhi, Pratibha

doi:10.1016/j.ejpn.2014.01.007

Cited by 23 publications

(7 citation statements)

References 17 publications

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“…Most reported CNS manifestations were diagnosed as asymptomatic and transient hydrocephalus and macrocephaly, based on computed tomography and brain ultrasonography [ 18 , 19 , 20 ]. Some studies have reported ischemic stroke and vasculitis with discernible symptoms and severe sequelae [ 21 , 22 , 23 ], but there have been few case reports of aseptic meningitis in NLE [ 5 ]. In this study, neurologic diseases were present in six patients (22.2%), where two patients (22.2%) presented with aseptic meningitis and three patients (11.1%) had bilateral GMH grade I with cystic change, all of which had no symptoms and sequalae, but one patient (3.7%) had bilateral GMH grade II with IVH, who died due to complications of prematurity, including respiratory distress syndrome with pulmonary hypertension and PDA.…”

Section: Discussionmentioning

confidence: 99%

Neonatal Lupus Erythematosus as a Rare Cause of Fever in Young Infants

Song

Park

Byun

et al. 2021

JCM

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Neonatal lupus erythematosus (NLE) is a rare disease caused by passively transmitted autoantibodies from the mother. NLE is a multi-organ system disease characterized by cutaneous, cardiac, hematological, hepatobiliary, and neurological manifestations. This study aimed to review the various symptoms and clinical manifestations in young infants with NLE and their mothers. We conducted a retrospective review of medical records of patients with NLE who were both examined and treated at Pusan National University Children’s Hospital between January 2009 and December 2020 and their mothers. Twenty-seven patients with NLE comprising 13 male patients (48.1%) and 14 female patients (51.9%) were included. The most common symptom was rash (40.7%), followed by fever (25.9%), arrhythmia (14.8%), splenomegaly (11.1%), and intrauterine growth retardation (7.4%). Seven patients with fever had various organ system manifestations, including cutaneous (100%), hematological (71.4%), hepatobiliary (57.1%), and central nervous system (CNS; 28.6%) manifestations. Two of the febrile patients had aseptic meningitis. Cutaneous, cardiac, hematological, hepatobiliary, and CNS involvement were noted in 44.4%, 18.5%, 51.9%, 40.7%, and 22.2% of the patients, respectively. Systemic lupus erythematosus (SLE) was the most common maternal disease (14/27, 51.9%). Ten mothers (37.0%) had not been diagnosed with any autoimmune disease until their babies were diagnosed. Among them, three were subsequently diagnosed with SLE, five were diagnosed with the Sjögren’s syndrome, and two of them still had no known diagnosis of any autoimmune disorder. Fever is a common symptom of NLE; thus, when there is no clear focus of fever in infants, NLE needs to be considered, especially in cases with skin rashes.

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Section: Discussionmentioning

confidence: 99%

Neonatal Lupus Erythematosus as a Rare Cause of Fever in Young Infants

Song

Park

Byun

et al. 2021

JCM

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“…In contrast to these more common causes, immune-mediated disorders manifesting with neurological symptoms (such as seizures) in newborns are extremely rare [ 13 , 14 ]. Several cases of NLE-associated seizures are reported [ 15 - 20 ]. NLE is an acquired autoimmune disease caused by maternal antibodies to SSA /Ro and SSB/La, ANAs, anti-ds DNA or anti-Sm antibodies and in rare cases anti-RNP antibodies [ 21 ].…”

Section: Discussionmentioning

confidence: 99%

A newborn with grouped facial skin lesions and subsequent seizures

Döring

Rohrer

Tsiflikas³

et al. 2014

BMC Pediatr

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BackgroundCongenital grouped skin lesions are alarming signs of a variety of threatening diagnoses of quite different origin. The present case report shows an impressive clinical pattern of a neonate and illustrates the difficulty in differential diagnosis of mixed connective tissue disease and neonatal lupus erythematosus in newborns. This reported case is to our knowledge the first description of an unrecognized mixed connective tissue disease in the mother with an unusual clinical manifestation in the newborn, comprising skin lesions, neurological damage and non-typical antibody constellation.Case presentationWe report on a Caucasian female neonate from a perinatally asymptomatic mother, who presented with grouped facial pustular-like skin lesions, followed by focal clonic seizures caused by multiple ischemic brain lesions. Herpes simplex virus infection was excluded and both the mother and her infant had the antibody pattern of systemic lupus erythematosus and neonatal lupus erythematosus, respectively. However, clinical signs in the mother showed overlapping features of mixed connective tissue disease.ConclusionThis case report emphasizes congenital Lupus erythematosus and mixed connective tissue disease as important differential diagnoses of grouped skin lesions in addition to Herpes simplex virus-infection. The coexistence of different criteria for mixed connective tissue disease makes it difficult to allocate precisely maternal and congenital infantile disease.

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“…In isolation, CNS involvement is rare and is described as transient vasculopathy. One case report in the literature described ischemic infarction secondary to CNS vasculitis (7) . In the present study, we describe the case of a neonate with convulsive seizures after birth and signs of acute ischemia on MRI (Figure 3).…”

Section: Systemic Lupus Erythematosusmentioning

confidence: 99%

Spectrum of central nervous system involvement in rheumatic diseases: pictorial essay

Vieira

Nascimento

Júnior³

et al. 2018

Radiol Bras

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The rheumatic diseases, which include systemic lupus erythematosus, rheumatoid arthritis, Behçet's disease, scleroderma, and ankylosing spondylitis, are characterized by involvement of connective tissue, with multiple manifestations. In those diseases, there can be involvement of the peripheral or central nervous system, and that involvement can be primary, presenting as a major feature of the clinical presentation, or secondary, as an effect of the drugs used in order to control a given disease or its complications. Knowledge of the wide variety of imaging findings is crucial to the diagnosis of a rheumatic disease, especially in the early stages, enabling effective treatment and minimizing disability. This pictorial essay, presenting cases from the records of two tertiary teaching hospitals, encompasses cases of patients diagnosed with rheumatic disease and illustrates the neuroradiological findings on magnetic resonance imaging and computed tomography, in order to emphasize the importance of these methods for properly diagnosing rheumatic diseases.

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CNS vasculitis and stroke in neonatal lupus erythematosus: A case report and review of literature

Cited by 23 publications

References 17 publications

Neonatal Lupus Erythematosus as a Rare Cause of Fever in Young Infants

Neonatal Lupus Erythematosus as a Rare Cause of Fever in Young Infants

A newborn with grouped facial skin lesions and subsequent seizures

Spectrum of central nervous system involvement in rheumatic diseases: pictorial essay

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