ACR Meeting Abstracts

ACR Meeting Abstracts

Abstract Number: 425

Impact of in Utero Hydroxychloroquine Exposure on Age of Onset of Cutaneous Neonatal Lupus

Julie Barsalou1, Nathalie Costedoat-Chalumeau2, Cesar Fors-Nieves3, Ummara Shah4, Patrick Brown5, Carl Laskin6, Nathalie Morel2, Kateri Levesque7, Jill P. Buyon8, Earl Silverman9 and Peter M. Izmirly10, 1Pediatric Rheumatology, CHU Sainte-Justine, Université de Montréal, Montréal, QC, Canada, 2Internal Medicine Department, Cochin Hospital, “René-Descartes Paris V” University, Paris, France, 3Division of Rheumatology, NYU School of Medicine, New York, NY, 4Medicine, Strong Memorial Hospital, University of Rochester, Rochester, NY, 5University of Toronto, Toronto, ON, Canada, 6Medicine, Rheumatology and Obstetrics and Gynecology, University of Toronto and LifeQuest Centre for Reproductive Medicine, Toronto, ON, Canada, 7Internal Medicine, CHU Ste-Justine, Montréal, QC, Canada, 8NYU School of Medicine, New York, NY, 9Division of Rheumatology, Hospital for Sick Children and University of Toronto, Toronto, ON, Canada, 10Rheumatology, NYU School of Medicine, New York, NY

Meeting: 2015 ACR/ARHP Annual Meeting

Date of first publication: September 29, 2015

Keywords: , , ,

Session Information

Date: Sunday, November 8, 2015

Title: Pediatric Rheumatology – Clinical and Therapeutic Aspects Poster I: Lupus, Scleroderma, JDMS

Session Type: ACR Poster Session A

Session Time: 9:00AM-11:00AM

Background/Purpose: Biopsy specimens of cutaneous
neonatal lupus (cNL) lesions usually show interface dermatitis.
Hydroxychloroquine (HCQ) is an effective treatment for interface dermatitis
seen in connective tissue diseases. It may also be of benefit for cNL. Due to
the transplacental passage of HCQ, fetuses of treated mothers are exposed to
HCQ. Neonates are not continued on HCQ after delivery, therefore HCQ levels are
expected to decline after birth. The aim of this study was to asses if in utero
exposure to HCQ delayed the onset of cNL.

Methods: A multicenter retrospective cohort
was assembled that included all cases of cNL found in 3 data sources. Inclusion
criteria were: infant born to a woman positive for anti-Ro ± anti-La antibodies, infant diagnosed with cNL, known age of onset of cNL
and documentation of maternal medications during pregnancy. Kaplan-Meier
survival analysis was used to assess if HCQ delayed the onset of cNL. Proportions
of infants developing cNL within distinct time frames were compared between those
HCQ exposed and non exposed.

Results: A total of 327 cases of cNL were
included: N=190 from the Research Registry for Neonatal Lupus, N=107 from the
SickKids Neonatal Lupus cohort and N=30 from the French Registry of Neonatal Lupus.
Twenty-eight (8.6%) infants were exposed to HCQ. Infants exposed to HCQ had overall
similar baseline characteristics to those not exposed (Table 1). Survival
analysis showed no statistically significant difference in the median number of
weeks to cNL onset: HCQ exposed 6.0 (95% CI: 5.7-6.3) vs non exposed 4.4 (95%
CI: 3.8-5.0) weeks; p=0.452 (Figure 1). Although no statistically significant
difference was seen, the survival curves showed a delayed onset of rash during the first postnatal
month in infants exposed to HCQ in utero. The frequency of
infants developing cNL within specific time frames did not significantly segregate
by HCQ exposure (Table 2).

Conclusion: Although in utero HCQ exposure did
not significantly delay the time of onset of cNL, survival curves suggest later
onset of cNL in those exposed to HCQ perhaps due to protection conferred by HCQ
when neonatal HCQ blood levels still remain within a  detectable range.

Table 1. Baseline characteristics of the 327 patients

Patients

(N)

Exposed to HCQ

(N=28)

Non exposed to HCQ

(N=299)

p value

Maternal characteristics

Diagnosis, N (%)

326

<0.001

  Asymptomatic/Undifferentiated autoimmune

  syndrome

3 (10.7)

151 (50.5)

  SLE

13 (46.4)

65 (21.8)

  Cutaneous lupus

2 (7.2)

3 (1.0)

  Sjogren’s syndrome

4 (14.3)

47 (15.8)

  Sjogren’s syndrome and SLE

6 (21.4)

25 (8.4)

  Sjogren’s syndrome and RA

0

1 (0.4)

  Dermatomyositis

0

1 (0.4)

  Overlap connective tissue disease

0

1 (0.4)

  RA or juvenile idiopathic arthritis

0

4 (1.3)

Antibody status, N (%)

  Anti-Ro positive

325

28 (100)

297 (100)

–

  Anti-La positive

317

23 (85.2)

222 (76.6)

0.306

Medication intake, N (%)

  Fluorinated steroids

326

3 (10.7)

38 (12.8)

0.999

  Non-fluorinated steroids

327

12 (42.9)

32 (10.7)

<0.001

  Azathioprine

327

1 (3.6)

2 (0.7)

0.236

  IVIG

326

0

6 (2.0)

0.999

Children characteristics

Sex, female:male, N

326

15:13

172:126

0.671

Age of onset of cNL, weeks (median (IQR))

327

6.0 (4.0-8.0)

4.4 (2.0-8.9)

0.435


Table 2. Proportion of infants developing cNL within specific time frames as per exposure status to HCQ

Cutoffs of age of onset of cNL

Exposed to HCQ

N (%)

Non exposed to HCQ   N (%)

p value

A.

Rash present at birth

3 (10.7)

59 (19.7)

0.244

B.

Rash onset <2 weeks

5 (17.9)

90 (30.1)

0.172

C.

Rash onset <4 weeks

11 (39.3)

144 (48.2)

0.368

Disclosure: J. Barsalou, None; N. Costedoat-Chalumeau, None; C. Fors-Nieves, None; U. Shah, None; P. Brown, None; C. Laskin, None; N. Morel, None; K. Levesque, None; J. P. Buyon, None; E. Silverman, None; P. M. Izmirly, None.

To cite this abstract in AMA style:

Barsalou J, Costedoat-Chalumeau N, Fors-Nieves C, Shah U, Brown P, Laskin C, Morel N, Levesque K, Buyon JP, Silverman E, Izmirly PM. Impact of in Utero Hydroxychloroquine Exposure on Age of Onset of Cutaneous Neonatal Lupus [abstract]. Arthritis Rheumatol. 2015; 67 (suppl 10). https://acrabstracts.org/abstract/impact-of-in-utero-hydroxychloroquine-exposure-on-age-of-onset-of-cutaneous-neonatal-lupus/. Accessed .

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