Received Date: 22-Sep-2016

Revised Date: 01-Nov-2016

Accepted Article
Accepted Date: 06-Feb-2017

Article Type: Brief Communication

Subheading: Obstetrics

BRIEF COMMUNICATION

Case report of a pregnant patient with systemic lupus erythematosus with

uterine atony and very thin myometrium with uterine fibrosis

Yu Tokushige 1*, Shuichiro Iwami 1, Takafumi Nonogaki 1, Takahiro Shibayama 2,

Toshihide Shimada 2, Sachiko Minamiguchi 3

1
Department of Obstetrics Gynaecology, Osaka Redcross Hospital, Osaka, Japan.
2
Department of Pathology, Osaka Redcross Hospital, Osaka, Japan.
3
Department of Diagnostic Pathology, Kyoto University Hospital, Kyoto, Japan.

*
Corresponding author: Yu Tokushige

#102 Akurosuuemachihigashi-2 1-10-10 Tsuruhashi, Ikuno Ward, Osaka, Osaka

Prefecture 544-0031, Japan. Tel.: +81 906 988 5192.

E-mail address: re_remini@emobile.ne.jp

Keywords: Pregnancy; Systemic lupus erythematosus; Thin uterine myometrium;

Uterine fibrosis; Uterine atony.

This article has been accepted for publication and undergone full peer review but has not
been through the copyediting, typesetting, pagination and proofreading process, which may
lead to differences between this version and the Version of Record. Please cite this article as
doi: 10.1002/ijgo.12118
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 Synopsis: A pregnant patient with systemic lupus erythematosus experienced

hypertension and underwent cesarean delivery. They experienced uterine atony and
Accepted Article
a very thin myometrium with uterine fibrosis.

The present case report describes a pregnant patient with systemic lupus

erythematosus (SLE) who presented at the Osaka Redcross Hospital, Osaka, Japan

with uterine atony and a very thin myometrium with uterine fibrosis; to the best of our

knowledge, this is the first published report of this kind.

A nulliparous woman aged 35 years, who had been diagnosed with SLE aged 30

years, became pregnant using in vitro fertilization with frozen embryo transfer. The

patient’s anti-Ro antibody test result was positive, and the anti-cardiolipin antibody

test result was negative. The patient received 15 mg of oral prednisone daily for the

treatment of SLE and was in remission during pregnancy, which is considered

normal until the third trimester. The patient described in the present case report

provided written informed consent for the use of their data in the present study.

After 32 weeks of pregnancy, the patient’s serum uric acid level was elevated, and

reached 6.8 mg at 36 weeks of pregnancy; the patient’s blood pressure was normal.

At 39 weeks of pregnancy, the patient experienced abdominal pain and had elevated

systolic blood pressure (140 mm Hg). Fetal heart rate monitoring demonstrated sinus

tachycardia (170–180 beats per minute) and the patient’s cervix was dilated to 3 cm.

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 Labor was induced owing to pregnancy-induced hypertension using oxytocin;

however, uterine contraction did not occur. The patient underwent cesarean delivery
Accepted Article
for obstructed labor; uterine atony and a very thin myometrium were observed

following delivery.

B-Lynch suturing and oxytocin injection into the myometrium did not improve uterine

atony. The patient experienced blood loss of 4500 mL and a total abdominal

hysterectomy was performed for hemorrhagic shock.

The neonate was male (2742 g), with a 5-minute Apgar score of 8 and an umbilical

cord blood pH of 7.307. The uterus and placenta were 17 cm × 9.5 cm × 0.8 cm and

16 cm × 18 cm × 1.5 cm in size, respectively. Pathological assessment (using the

Masson trichrome stain) demonstrated a corpus myometrium–fibrosis ratio of 4:6

(Figure 1A); comparatively, a uterus removed from a control patient for hemorrhagic

shock caused by placenta previa exhibited a myometrium–fibrosis ratio of 9:1 (Figure

1B). It was not possible to contact the patient the control image was obtained from;

consequently, written informed consent for the use of the image was not obtained.

The uterine fibrosis in the present patient’s myometrium was similar to that observed

in the cervix. Chorioamnionitis (Blanc 2) was also detected. Postoperative amniotic

fluid embolism workup detected high interleukin-8 (52.3 pg/mL), low complement

component 3 (39.0 mg/dL), and low complement component 4 (7.0 mg/dL) levels.

There have been six reports of patients with SLE experiencing uterine atony and a

very thin myometrium after postpartum hysterectomy in Japan [1–5]; four of these

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 patients did not experience uterine contractions following oxytocin administration and

all were diagnosed with placenta accreta.

Accepted Article
The patient in the present study did not have placenta accreta and amniotic fluid

embolism could not sufficiently explain the thin myometrium and uterine atony before

delivery. SLE causes fibrosis in many organs, including the skin, kidney, bladder,

and heart; however, the pathophysiological basis of this is unknown [6–9]. It is

suggested that uterine fibrosis of SLE, completed before delivery, caused the

postpartum hemorrhage in the patient.

Conflicts of interest

The authors have no conflicts of interest.

References

[1] Youhei M, Yuuki T, Koutaro D, Seishi F, Hiroshi S, Tsuyomu I, Takatsugu M,

Masato K. Two cases of pregnant patients with systemic lupus erythematosus, with

uterine atony and placenta accrete [in Japanese]. 47th Annual Congress of Japan

Society of Perinatal and Neonatal Medicine. 2011 July

[2] Tsuyoshi O, Tsuyoshi Y, Takashi H, Norio I, Atsumu T, Akitaka K, Jyunji I.

Case report of a pregnant patient with systemic lupus erythematosus with very thin

myometrium who underwent hysterectomy for placenta accreta [in Japanese]. 63rd

Annual Congress of The Japan Society of Obstetrics and Gynecology. 2011 Aug

[3] Nao M, Takashi S, Erina T, Yakashi U, Yuki K, Tomoaki I. Case report of a

pregnant patient with systemic lupus erythematosus with postpartum hemorrhage

This article is protected by copyright. All rights reserved.

 and placenta accreta [in Japanese]. Journal of Japan Society of Perinatal and

Neonatal Medicine 2012;48(2):560

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[4] Kenta Y, Yuki K, Mikiko N, Jyunko W, Erina T, Nao M, Takashi U, Takashi S,

Tomoaki I. Case report of a pregnant patient with systemic lupus erythematosus,

with uterine atony and placenta accreta, who underwent hysterectomy [in Japanese].

The Tokai journal of obstetrics and gynecology;49:366

[5] Yukiko H, Mika S, Rikiya S, Takahito M, Yakuya M, Takuji T, Yuichiro N,

Takafumi N, Mitsuru S, Daisuke Y, Souhachi F, Naoki K, Hirotake N, Takuya M,

Koichiro S. Case report of a pregnant patient with systemic lupus erythematosus,

had no uterine contraction with oxytocin, diagnosed with placenta accreta [in

Japanese]. 22th Annual Congress of The Japanese Branch Of International

Federation of Placental Association. 2014 Oct

[6] Seneviratne MG, Grieve SM, Figtree GA, Garsia R, Celermajer DS, Adelstein

S, Puranik R. Prevalence, distribution and clinical correlates of myocardial fibrosis in

systemic lupus erythematosus: a cardiac magnetic resonance study. Lupus 2016

May;25(6):573-81

[7] Sole C, Gimenez Barcons M, Ferrer B, Ordi Ros J, Cortes Hernandez J.

Microarray study reveals a transforming growth factor-β-dependent mechanism of

fibrosis in discoid lupus erythematosus. Br J Dermatol 2016;175(2):302-13

[8] Yung S, Chan TM. Mechanisms of Kidney Injury in Lupus Nephritis-the Role

of Anti-dsDNA Antibodies. Front Immunol 2015 Sep 15;6:475

[9] Koh JH, Lee J, Jung SM, Ju JH, Park SH, Kim HY, Kwok SK. Lupus cystitis in

Korean patients with systemic lupus erythematosus: risk factors and clinical

outcomes. Lupus 2015 Oct;24(12):1300-7

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 Figure 1 (A) Uterine corpus of tthe patient described in the present report (Masson

trichrome stain at ×200 magnificcation). (B) Uterine corpus from a control patient
p who
Accepted Article
experienced hemorrhagic shockk caused by placenta previa (Masson trichrome stain

at ×200 magnification).

Author contributions

YT was the physician in charge, managing and treating the patient, and prepared the

initial draft of the manuscript. SII participated in the treatment of the patient and

diagnosing the patient’s conditio

on, and contributed to manuscript writing. TN
T

contributed to analysis and interpretation of data, and assisted in drafting the

manuscript. TShib and TShim ccontributed to the analysis and interpretatio

on of clinical

data, and assisted in revising th

he manuscript. SM participated in diagnosin
ng the

patient, contributed to the analyysis and interpretation of clinical data, and assisted
a in

writing the manuscript. All autho

ors approved the final manuscript.

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