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Polymyositisdermatomyositis and pregnancy.

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29 1
POLYMYOSITIS/DERMATOMYOSITIS AND
PREGNANCY
GASPAR GUTIERREZ, ROGELIO DAGNINO, and GREGORIO MINTZ
Eighteen women with polymyositis/dermatomyositis (PM/DM) were studied to determine the possible
influences of pregnancy on the disease and the influence
of the disease on pregnancy. Before the onset of PM/DM
there were 77 pregnancies: 7 (9%) ended in abortion, 2
(2.5%) in perinatal deaths, with a total fetal loss of
11.5%. There were 3 (3.8%) premature newborns that
survived. These figures are equivalent to those of the
general population. There were 10 pregnancies in 7
patients coinciding with PM/DM, 1 of them with twins; 3
(30%) ended in abortion, 3 (25%) in perinatal deaths,
with a total fetal loss of 55%, and 5 (50%) pregnancies
ended prematurely. Four of the 7 women had onset of
PM/DM during pregnancy, and 3 others with previously
inactive disease had an exacerbation during pregnancy.
There were no maternal deaths, nor was there any
correlation between activity of PM/DM and fetal loss.
These results together with those previously reported
suggest that pregnancy in PM/DM should be considered
high-risk for both the mother and the baby.
In recent years with the development of modern
fetal and neonatal monitoring and care, and with
maternal control (1,2), it has been possible to reduce
From the Department of Rheumatology, Hospital General
del Centro MCdico Nacional, IMSS, Mexico.
Gaspar Gutikrrez, MD: Associate; Rogelio Dagnino, MD:
Resident; Gregorio Mintz, MD: Profcssor and Chairman, Department of Rheumatology, Hospital General del Centro MCdico Nacional, IMSS. Mexico.
Address reprint requests to Gregorio Mintz, MD, Professor
and Chairman, Department of Rheumatology, Hospital General del
Centro MCdico Nacional, IMSS, Av. Cuauhtemoc 330, Mexico 7,
D. F. Mexico.
Submitted for publication May 17. 1983; accepted in revised
form August 12, 1983.
Arthritis and Rheumatism, Vol. 27, No. 3 (March 1984)
the high morbidity and mortality that accompany highrisk pregnancies (3,4). It is therefore very important to
define the possible risk that a pregnancy may pose to
the mother’s health and to the survival of the baby.
The most common causes of high-risk pregnancies are
social, economic, and cultural factors; biologic characteristics of the pregnant female; and the presence of
medical complications during previous or current
pregnancies (5).
Women with systemic lupus erythematosus
(SLE) and progressive systemic sclerosis (PSS) have
frequently been merPtioned as having high-risk pregnancies (6-8); however, only 6 case reports (9-13) of
polymyositis/dermatomyositis(PM/DM) and pregnancy can be found in the literature. The purpose of this
study is to better define the maternal and fetal risks
during pregnancy in the presence of this disease.
PATIENTS AND METHODS
All female patients who attended our outpatient
clinic and fulfilled the criteria of definite PM/DM (14) were
studied. Women who had typical adult dermatomyositis and
polymyositis, and childhood myositis were included, but
women with associated neoplasms or other connective tissue
diseases, except secondary Sjogren’s syndrome, were excluded from this study.
All patients were interviewed by one of the authors
who used a previously constructed questionnaire that recorded information regarding number of pregnancies before
and after the onset of the disease, age at termination of each
pregnancy, stillbirths and neonatal deaths, evolution of
PMIDM during pregnancy and 8 weeks postpartum, and
causes of secondary sterility. All this information was considered with the review of the clinical record of the patient.
For the purpose of clarification, we define “abortion” as the spontaneous interruption of pregnancy before
the 20th week. (There were no induced abortions in these
patients.) “Prematurity” is the spontaneous termination of
GUTIERREZ ET AL
292
gestation between the 21st and the 37th weeks. “At term” is
delivery between weeks 38 and 40. “Stillbirth” is intrauterine fetal death after week 21 and before birth. “Neonatal
death” is the death of a newborn in the period between birth
and 30 days of age. “Perinatal death” is the sum of stillbirths
and neonatal deaths. “Total fetal loss” is the sum of
abortions and perinatal deaths.
RESULTS
Of a total of 22 patients entering the study, 4
were excluded: 2 because they were not sexually
active, 1 because of primary sterility, and 1 because of
sterility secondary to constant use of oral contraceptives. Of the remaining 18 patients included in this
study, 2 were diagnosed as having adult polymyositis,
15 as having adult dermatomyositis (1 developed a
secondary Sjogren’s syndrome), and 1 as having childhood myositis. The age at onset of PM/DM ranged
from 10-65 years with an average of 38.4 years.
Fertility. With the exception of the patient with
childhood myositis whose onset of disease was at age
10, the remaining 17 patients had 1 or more pregnancies before the onset of PM/DM. There were a total of
77 pregnancies with a fertility rate of 4.5.
Simultaneous with the disease or after its onset,
there were 10 pregnancies in 7 patients, which shows a
drop in fertility rate to I .7 ( P < 0.01). The average age
at onset of PM/DM in these 7 patients was 23 years
(range 10-33).
The causes for secondary sterility were: menopause in 11, contraception methods in 4, recent delivery in 1, and in the 2 remaining patients with active
sexual lives for more than 1 year after their last
delivery, no cause could be found.
Fetal loss. Of the 77 pregnancies that occurred
before the onset of PM/DM, 7 (9%) ended in spontaneous abortions and 2 (2.5%) were neonatal deaths, with
a total fetal loss of 11.5%. There were 3 (3.8%)
premature newborns, all of whom survived (Table 1).
Of the 10 pregnancies occuring after the onset
of PM/DM, 1 resulted in twins. Three pregnancies
(30%) ended in spontaneous abortion and 5 (50%) were
premature deliveries of which I was a stillbirth and 2
were neonatal deaths. Total fetal loss was 55%: 6 of 1 1
Table 1.
babies. Of these 10 pregnancies, 5 coincided with
active PM/DM, 1 of which ended in a spontaneous
abortion, 1 in a stillbirth, and 1 with a neonatal death.
The total fetal loss was 60%, and the remaining 2
pregnancies were premature deliveries with surviving
newborns.
Of the 5 pregnancies during inactive PM/DM, 2
ended in spontaneous abortion, the twin pregnancy
had a premature delivery with neonatal death of I of
the newborns (weight 900 gm). The remaining 2 pregnancies were at term with surviving newborns. Total
fetal loss was 50%: 3 of 6 babies.
Evolution of PWDM during pregnancy. Four of
the 18 patients (22%) began their PM/DM in relation to
a pregnancy: 3 during the first trimester and 1 after a
cesarean section (Table 2). Seven pregnancies occurred during inactive phases of PM/DM, and 3
(42.8%) women had an exacerbation: 2 during the third
trimester and 1 in the postabortion period.
Treatment. Prednisone was administered in the
same doses given to non-pregnant patients when the
diagnosis of PM/DM was established or when it was
considered to be active. When the disease was judged
to be inactive, no prednisone was given. None of the
pregnant patients received immunosuppressant drugs.
DISCUSSION
PM/DM is a group of inflammatory and degenerative conditions of striated muscle of unknown etiology that produce symmetric muscle weakness and
atrophy (15). Criteria for diagnosis, as well as a 5group classification system, have been proposed
(14,16), and these are widely used for the study and
definition of prognosis and treatment.
We excluded from this study those patients
with myositis associated with neoplasms (17) and with
other connective tissue diseases (5-7) to avoid an
overlap of these conditions on pregnancy, which
would produce unreliable results. Since a personal
interview was a requisite for inclusion in this study, it
is evident that many patients who have died or abandoned our clinic, and those who could not be located,
cannot be included. However, the interview format
Fetal loss in polyrnyositis/dermatornyositis (PM/DM)
KO.pregnancies
Before disease onset
After disease onset
Active PM/DM
Inactive PM/DM
77
10 (1 twin)
5
5 ( I twin)
% spontaneous
abortion
9
30
20
40
Yo stillbirth
0
9
20
0
70neonatal
death
2.5
18.0
20.0
25.0
%, total
fetal
loss
% premature
11.5
55.0
60.0
50.0
3.8
50.0
40.0
20.0
293
PM/DM AND PREGNANCY
Table 2. Obstetric history and evolution of polymyositis/dermatomyositis(PMIDM)
Previous pregnancies
Patient
1
2 normal
2
4 normal (1
neonatal
death)
3
0
I normal
I cesareadlive
Disease
evolution
Diagnosis
DM began 1st
trimester 3rd
pregnancy
DM began 1st
trimester 5th
pregnancy
Childhood DM
PM began 1st
trimester 2nd
pregnancy
DM began I5
days after
cesarean
2 ( I abortion)
6
3 normal
DM
7
2 normal
DM
3 (1 premature)
will produce more reliable results despite the relatively
small sample.
The 10 patients in this study with PM/DM and
pregnancy were seen over the course of almost 20
years. This confirms the rarity of this association that
is implied by there having been reports in the literature
of only 6 isolated cases since 1958 (9-13). These cases
are summarized in Table 3. We have excluded from
Table 3.
Active during
pregnancy
Abortion
Active during
pregnancy
Premature
stillbirth
Inactive during
pregnancy,
exacerbation
postabortion
Exacerbation
in 3rd
trimester
Active during
pregnancy
Abortion
Cesareanllive
premature
Premature
neonatal death
Inactive during
pregnancy
Abortion
Inactive during
pregnancy
Inactive during
pregnancy
Exacerbation
during 3rd
trimester
Inactive during
pregnancy
Cesarean at term
I,ive/normal
delivery
Premature
Twins premature/l
neonatal death
this review the patient reported by Le Coulant et al
(18) because the paticnt does not fulfill diagnostic
criteria, and there is no mention of the outcome of the
newborn. The patient reported by Spellacy (19) can be
better diagnosed as having a combination of PSS and
DM and has also been excluded.
Fertility rates in our patients arc statistically
different before and after the beginning of PM/DM.
Previous reports of pregnancy in polymyositis/dermatomyositis(PM/DM)
Author, year*
Diagnosis, age
(years)
Glickman, 1958
DM, 27
Improved
Healthy
Masse, 1962
DM, 26
Improved
Healthy
DM. 27
Inactive
Tsai et al, 1973
DM, 33
Bauer et at,
1979
PM, 25
DM began 1st
trimester
PM began 1st
trimester
Abortion 2nd
trimester
Neonatal death
Katz, 1980
PM, 26
* See references
t ACTH
Fetal outcome
=
Disease
evolution
PM began 1st
trimester
Fetal outcome
Stillbirth
Healthy
9-13, respectively, for additional information.
Adrenocorticotropic hormone.
Commentst
Prednisone 20-30 mg daily
during pregnancy and
postpartum
ACTH during pregnancy and
postpartum
ACTH during pregnancy and
postpartum
No treatment
Prednisone up to 160 mg
daily, improved postabortion
Prednisone (dosage not
recorded), improved postabortion
GUTIERREZ ET AL
294
However, the number of years at risk is widely variable in both pre- and post-disease onset periods, and
the late age a t onset of PM/DM plus the frequent use of
contraceptives preclude a more accurate evaluation of
the possible influence of PM/DM on fertility.
Pregnancy evolution and total fetal loss before
the onset of PM/DM in our study are similar to that in
the general population (8), but different from that
reported in SLE, PSS, and mixed connective tissue
disease (MCTD), where total fetal loss is high even
before the onset of disease (8,20,21).
Once PM/DM had begun, the frequency of
spontaneous abortions, prematurity, and perinatal
deaths increased dramatically, showing up t o 55% fetal
loss, similar t o the 50% fetal loss of the 6 previously
reported cases. This is higher than the frequencies
reported in SLE, PSS, and MCTD (6-8,21).
Fetal loss in women with active PM/DM during
their pregnancies is not significantly different from that
observed in women with inactive disease during pregnancy. This suggests that treatment of maternal disease probably does not modify fetal prognosis and that
this modification may be achieved with vigilant obstetric and neonatal care as we have previously demonstrated in SLE (22).
One-fourth of our patients began PM/DM during pregnancy, and approximately one-half of those
patients with initially inactive disease had an exacerbation. A full 50% of the previously reported cases
began PM/DM during the first trimester of pregnancy.
These data allow us to consider pregnancy as a precipitating factor in the onset and in the exacerbation of
PM/DM, and t o strongly advise a close followup of the
mother for early detection of disease flare and timely
treatment.
In spite of the retrospective nature of this study
and the relatively small sample, the results we obtained, together with those in the literature, clearly
show that pregnancy in PM/DM must be classified and
managed as a high-risk pregnancy.
ACKNOWLEDGMENT
The authors express their appreciation to Ms Margarita Sedeno for her expert secretarial services.
REFERENCES
Biggs JS: Progress in fetal assessment. Obstet Gynecol
45:227-233, 1975
Farahani G, Vasudeva K, Petrie K , Fenton AN: Oxytocin challenge test in high risk pregnancy. Obstet GyneC O ~47: 159-168, 1976
Hack M , Fanaroff AA, Merkatz IR: ‘The low birth
weight infant: evolution of a changing outlook. N Engl J
Med 301:1162-1165, 1979
4. Spellacy WN: Management of the High Risk Pregnancy.
Baltimore, University Park Press, 1976
5. Donnely JF, Flowers CE, Creadick RN, Greenberg BG,
Wells HB: Parental, fetal and environmental factors in
perinatal mortality. Am J Obstet Gynecol74: 1245-1256,
1975
6. Cecere FA, Persellin KH: The interaction of pregnancy
and the rheumatic diseases. Clin Rheum Dis 7:747-767,
1981
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and neonatal risks. Clin Obstet Gynecol 21:385-396,
1978
8. Fraga A, Mintz G, Orozco J, Orozco JH: Fertility and
sterility rates, fetal wastage and maternal morbidity in
systemic lupus erythematosus. J Rheumatol 1 :293-298,
1974
9. Glickman FS: Dermatomyositis associated with pregnancy. US Armed Forces M J 9:417-425, 1958
10. Masse MR: Grossesses et dermatomyosite. Bull SOC
Franc Derm Syph 69:921-923, 1962
11. Tsai A, Lindheimer MD, Lamberg SI: Dermatomyositis
complicating pregnancy. Obstet Gynecol 41 570-573,
1973
12. Bauer KA, Siegler M, Lindheimer MA: Polymyositis
complicating pregnancy. Arch Intern Med 139:449, 1979
13. Katz AL: Another case of polymyositis in pregnancy
(letter). Arch Intern Med 140:1123, 1980
14. Bohan A, Peter JB: Polymyositis and dermatomyositis.
N Engl J Med 292:344-347, 403-407, 1975
15. Pearson CM: Polymyositis and dermatomyositis, Arthritis and Allied Conditions. Ninth edition. Edited by DJ
McCarty. Philadelphia, Lea & Febiger, 1979, pp 742-761
16. Bohan A, Peter JB, Bowman RS, Pearson CM: Computer assisted analysis of 153 patients with polymyositis and
dermatomyositis. Medicine 56:255-286, 1977
17. Blatt J, Mulvihill JJ, Ziegler JL, Young RC, Poplack
DG: Pregnancy outcome following cancer chemotherapy. Am J Med 69:828-832, 1980
18. Le Coulant, Texier, Maleville, Beylot, Denef Dermatomyosite et dysembryome de l’ovaire: dermatomyosite et
grossesse. Bull SOCFranc Derm Syph 69:639-642, 1964
19. Spellacy WN: Scleroderma and pregnancy: report of a
case. Obstet Gynecol 23:297-300, 1964
20. Grigor RR, Sherrington PC, Hughes GRV, Hawkins DF:
Outcome of pregnancy in systemic lupus erythematosus.
Proc R SOCMed 70:99-100, 1977
21. Kaufman RL, Kitridou RC: Pregnancy in mixed connective tissue disease: comparison with systemic lupus
erythematosus. J Rheumatol 9549-555, 1982
22. Gutierrez G, Jimenez J, Mintz G: Results of a prospective multidisciplinary approach to pregnancy in systemic
lupus erythematosus (abstract). Arthritis Rheum (suppl)
24:S107, 1981
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