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Amniotic fl uid embolism: analysis of the national registry... In the National Registry analysis, the authors suggested that the term “ amniotic fl uid embolism ” be discarded and the sy

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AFE Although disputed on statistical grounds by Morgan [1] , this misconception persisted in some writings until recently The historic anecdotal association between hypertonic uterine con-tractions and the onset of symptoms in AFE was further clarifi ed

by the analysis of the National Registry [5] These data demon-strated that the hypertonic contractions commonly seen in asso-ciation with AFE appear to be a result of the release of catecholamines into the circulation as part of the initial human hemodynamic response to any massive physiologic insult Under these circumstances, norepinephrine, in particular, acts as a potent uterotonic agent [5,55] Thus, while the association of hypertonic contractions and AFE appears to be valid, it is the physiologic response to AFE that causes the hypertonic uterine activity rather than the converse Indeed, there is a complete ces-sation of uterine blood fl ow in the presence of even moderate uterine contractions; thus, a tetanic contraction is the least likely time during an entire labor process for any exchange between maternal and fetal compartments [56] Oxytocin is not used with increased frequency in patients suffering AFE compared with the general population, nor does oxytocin - induced hyperstimulation commonly precede this condition [5] Thus, several authorities, including the American College of Obstetricians and Gynecologists, have concluded that oxytocin use has no relationship to the occurrence of AFE [1,5,57] A recent population - based cohort study of approximately 3 million deliveries in Canada reported a statistically signifi cant association of labor induction with AFE [58] , however the same investigators did not observe this associa-tion in another populaassocia-tion - based cohort of approximately 3 million deliveries in the US [59] , casting doubt regarding any clinically signifi cant relationship between labor stimulation and AFE

The syndrome of AFE appears to be initiated after maternal intravascular exposure to various types of fetal tissue Such expo-sure may occur during the course of normal labor and delivery; after potentially minor traumatic events, such as appropriate intrauterine pressure catheter placement; or during cesarean section Because fetal - to - maternal tissue transfer is virtually uni-versal during the labor and delivery process, actions by healthcare providers, such as intrauterine manipulation or cesarean delivery, may affect the timing of the exposure No evidence exists, however, to suggest that exposure itself can be avoided by altering clinical management Simple exposure of the maternal circula-tory system to even small amounts of amniotic fl uid or other fetal tissue may, under the right circumstances, initiate the syndrome

of AFE This understanding explains the well - documented occur-rence of fatal AFE during fi rst - trimester pregnancy termination

at a time when neither the volume of fl uid nor positive intrauter-ine pressure could be contributing factors [11] Whereas much has been written about the importance to the fetus of an immu-nologic barrier between the mother and the antigenically differ-ent products of conception, little attdiffer-ention has been paid to the potential importance of this barrier to maternal well - being The observations of the National Registry as well as cumulative data for the past several decades suggest that breaches of this barrier

identical; fever is unique to septic shock, and cutaneous

manifes-tations are more common in anaphylaxis Nevertheless, the

marked similarities of these conditions suggest similar

patho-physiologic mechanisms

Detailed discussions of the pathophysiologic features of septic

shock and anaphylactic shock are presented elsewhere in this text

Both of these conditions involve the entrance of a foreign

sub-stance (bacterial endotoxin or specifi c antigens) into the

circula-tion, which then results in the release of various primary and

secondary endogenous mediators (Figure 35.3 ) Similar

patho-physiology has also been proposed in non - pregnant patients with

pulmonary fat embolism It is the release of these mediators that

results in the principal physiologic derangements characterizing

these syndromes These abnormalities include profound

myocar-dial depression and decreased cardiac output, described in both

animals and humans; pulmonary hypertension, demonstrated in

lower primate models of anaphylaxis; and disseminated

intervas-cular coagulation, described in both human anaphylactic

reac-tions and septic shock [44 – 53] Further, the temporal sequence

of hemodynamic decompensation and recovery seen in

experi-mental AFE is virtually identical to that described in canine

ana-phylaxis [49] An anaphylactoid response is also well described in

humans and involves the non - immunologic release of similar

mediators [44] It is also intriguing that, on admission to hospital,

41% of patients in the AFE registry gave a history of either drug

allergy or atopy [5]

The ability of arachidonic acid metabolites to cause the same

physiologic and hemodynamic changes observed in human AFE

has been noted [54] Further, in the rabbit model of AFE,

pre-treatment with an inhibitor of leukotriene synthesis has been

shown to prevent death [28] These experimental observations

further support the clinical conclusions of the National AFE

Registry analysis that this condition involves the anaphylactoid

release of endogenous mediators, including arachidonic acid

metabolites, which result in the devastating pathophysiologic

sequence seen in clinical AFE [5]

Earlier anecdotal reports suggested a possible relationship

between hypertonic uterine contractions or oxytocin use and

Figure 35.3 Proposed pathophysiologic relation between AFE, septic shock,

and anaphylactic shock Each syndrome also may have specifi c direct physiologic

effects (Reproduced by permission from Clark SL, Hankins GVD, Dudley DA et al

Amniotic fl uid embolism: analysis of the national registry Am J Obstet Gynecol

1995; 172: 1158 – 1169.)

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opment of a consumptive coagulopathy, which may lead to exsanguination, even if attempts to restore hemodynamic and respiratory function are successful It must be emphasized, however, that in any individual patient any of the three principal phases (hypoxia, hypotension, or coagulopathy) may either dom-inate or be entirely absent [5,38,61] Clinical variations in this syndrome may be related to variations in either the nature of the antigenic exposure or maternal response The differential diag-nosis is summarized in Table 35.3

Maternal outcome is dismal in patients with AFE syndrome

In documented “ classic ” cases, the overall maternal mortality rate appears to be 60 – 80% [5,32] Only 15% of patients survive neu-rologically intact In a number of cases, following successful hemodynamic resuscitation and reversal of disseminated intra-vascular coagulation, life - support systems were withdrawn because of brain death resulting from the initial profound hypoxia In patients progressing to cardiac arrest, only 8% survive neurologically intact [5] In the National Registry database, no form of therapy appeared to be consistently associated with improved outcome A large series of patients in whom the diag-nosis of AFE was obtained from the discharge summary reported

a 26% mortality rate Notably, however, many patients in this series lacked one or more potentially lethal clinical manifestation

of the disease classically considered mandatory for diagnosis, thus casting the diagnosis into doubt However, if one assumes that the discharge diagnosis of these patients was accurate, these data suggest improved outcome for those women with milder forms

of the disease [61] Samuelsson and colleagues [62] , using the Swedish Cause of Death Register, found that the case fatality rate for AFE was high and remained unaltered (42 – 48%) during the decades spanning the 1970s through 1990s Reported maternal mortality rates are summarized in Table 35.4

Neonatal outcome is similarly poor If the event occurs prior

to delivery, the neonatal survival rate is approximately 80%; only half of these fetuses survive neurologically intact [5] Fetuses sur-viving to delivery generally demonstrate profound respiratory acidemia Although at the present time no form of therapy appears to be associated with improved maternal outcome, there is a clear relationship between neonatal outcome and event - to - delivery interval in those women suffering cardiac

may, under certain circumstances and in susceptible maternal –

fetal pairs, be of immense signifi cance to the mother as well [5]

Previous experimental evidence in animals and humans

unequivocally demonstrates that the intravenous administration

of even large amounts of amniotic fl uid per se is innocuous

[21,24,60] Further, the clinical fi ndings described in the National

Registry are not consistent with an embolic event as commonly

understood (Table 35.2 ) Thus, the term “ amniotic fl uid

embo-lism ” itself appears to be a misnomer In the National Registry

analysis, the authors suggested that the term “ amniotic fl uid

embolism ” be discarded and the syndrome of acute peripartum

hypoxia, hemodynamic collapse, and coagulopathy should be

designated in a more descriptive manner, as “ anaphylactoid

syn-drome of pregnancy ”

Clinical p resentation

Clinical signs and symptoms noted in patients with AFE are

described in Table 35.2 In a typical case, a patient in labor, having

just undergone cesarean delivery or immediately following

vaginal delivery or pregnancy termination, suffers the acute onset

of profound hypoxia and hypotension followed by

cardiopulmo-nary arrest The initial episode often is complicated by the

Table 35.2 Signs and symptoms noted in patients with amniotic fl uid

embolism

Sign or symptom No of patients (%)

* Includes all live fetuses in utero at time of event

† Eighteen patients did not survive long enough for these diagnoses to be

confi rmed

‡ Eight patients did not survive long enough for this diagnosis to be confi rmed

§ One patient was intubated at the time of the event and could not be assessed

¶ Diffi cult ventilation was noted during cardiac arrest in six patients, and

wheezes were auscultated in one patient

Reproduced by permission from Clark SL, Hankins GVD, Dudley DA Amniotic

fl uid embolism: analysis of a national registry Am J Obstet Gynecol 1995; 172:

1158 – 1169

Table 35.3 Differential diagnosis of amniotic fl uid embolism

Air embolus Anaphylaxis Anesthetic toxicity Myocardial infarction Peripartum cardiomyopathy Placental abruption Pulmonary aspiration Septic shock Transfusion reaction Venous thromboembolism

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cance of histologic fi ndings in patients with pulmonary fat embolism [68]

Other putative markers for AFE, such as serum tryptase [69] , pulmonary mast cell antitryptase [70] , serum TKH - 2 antibody to fetal antigen sialyl Tn [71] , pulmonary TKH - 2 antibody to fetal antigen sialyl Tn [72] , serum complement [69] , and plasma zinc coproporphyrin I [73] , have been studied but as of yet provide

no defi nitive means of diagnosing or excluding AFE

Treatment

For the mother, the end - result of therapy remains disappointing, with a high mortality rate In the National Registry, we noted no

arrest (Table 35.5 ) [5] Similar fi ndings were reported by Katz et

al [63] in patients suffering cardiac arrest in a number of

differ-ent clinical situations

Diagnosis

In the past, histologic confi rmation of the clinical syndrome

of AFE was often sought by the detection of cellular debris of

presumed fetal origin either in the distal port of a pulmonary

artery catheter or at autopsy [32] Several studies conducted

during the past decade, however, suggest that such fi ndings

are commonly encountered, even in normal pregnant women

(Figure 35.4 ) [64 – 67] In the analysis of the National AFE

Registry, fetal elements were found in roughly 50% of cases in

which pulmonary artery catheter aspirate was analyzed and in

roughly 75% of patients who went to autopsy [5] The frequency

with which such fi ndings are encountered varies with the number

of histologic sections obtained In addition, multiple special

stains often are required to document such debris [32] Thus, the

diagnosis of AFE remains a clinical one; histologic fi ndings are

neither sensitive nor specifi c It is interesting to note that similar

conclusions have been drawn regarding the diagnostic signifi

Table 35.4 Summary of published amniotic fl uid embolism case series

Series Methodology Period AFE Incidence (1 per x births) Maternal Mortality

Gilbert 1999 [61] California population database of 1,094,248 singleton births 1994 – 1995 20,646 14/53 (26%)

Kramer 2006 [58] Canadian population based cohort of 3,018,781 deliveries 1991 – 2002 16,667 24/180 (13%) Samuelsson 2007 [62] Swedish Cause of Death Register from 2,961,000 deliveries 1973 – 1999 51,947 25/57 (44%) Abenhaim 2008 [59] U.S population based cohort study of 2,940,362 births 1998 – 2003 12,987 49/227 (22%)

* Incidence of fatal cases

* * years 1972 – 1980

Table 35.5 Cardiac arrest - to - delivery interval and neonatal outcome

Interval (min) Survival Intact survival

Reproduced by permission from Clark SL, Hankins GVD, Dudley DA Amniotic

fl uid embolism: analysis of the national registry Am J Obstet Gynecol 1995; 172:

1158 – 1169

Figure 35.4 Squamous cells recovered from the pulmonary arterial circulation

of a pregnant patient with class IV rheumatic mitral stenosis (magnifi cation,

× 1000) From Clark 1986 [66]

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to the mother and deliver the baby [5,63] For the pregnant patient, the standard ABC of cardiopulmonary resuscitation should be modifi ed to include a fourth category, D: delivery New modalities for the treatment of AFE, such as high - dose steroids [5] , extracorporeal membrane oxygenation with intra aortic balloon counterpulsation [74] , continuous hemodiafi ltra-tion [75,76] , cardiopulmonary bypass [77,78] , recombinant factor VIIa [79,80] , and nitric oxide [81] have been reported in survivors but are thus far of limited cumulative experience or demonstrated benefi t

There are limited data on risk of recurrence in a subsequent pregnancy for women who experience AFE; fewer than a dozen cases are reported in the published literature [82 – 87] At present,

it appears that the risk of recurrence is low

Despite many advances in the understanding of this condition, AFE or anaphylactoid syndrome of pregnancy remains enigmatic and in most cases is associated with dismal maternal and fetal outcomes, regardless of the quality of care rendered Thus, AFE remains unpredictable, unpreventable, and, for the most part, untreatable Further insight into this rare, but lethal, disorder may be forthcoming from the UK Obstetric Surveillance System (UKOSS), a joint initiative of the Royal College of Obstetricians and Gynaecologists and the National Perinatal Epidemiology Unit, which goal is to describe the epidemiology of a variety of uncommon disorders of pregnancy [88]

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difference in survival among patients suffering initial cardiac

arrest in small rural hospitals attended by family practitioners

compared with those suffering identical clinical signs and

symp-toms in tertiary - level centers attended by board - certifi ed

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1 The initial treatment for AFE is supportive Cardiopulmonary

resuscitation is performed if the patient is suffering from a

lethal dysrhythmia Oxygen should be provided at high

concentrations

2 In the patient who survives the initial cardiopulmonary insult,

it should be remembered that left ventricular failure is commonly

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performed, and if the patient remains signifi cantly hypotensive,

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sur-vival is extremely unlikely, regardless of the therapy rendered In

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properly performed cardiopulmonary resuscitation (diffi cult at

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normal cardiac output Under these circumstances, it is fair to

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cardiac arrest, even during ideal performance of

cardiopulmo-nary resuscitation Because the interval from maternal arrest to

delivery is directly correlated with newborn outcome,

perimor-tum cesarean delivery should be initiated immediately on the

diagnosis of maternal cardiac arrest in patients with AFE,

assum-ing suffi cient personnel are available to continue to provide care

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72 Oi H , Kobayashi H , Hirashima Y , Yamazaki T , Kobayashi T , Terao

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Critical Care Obstetrics, 5th edition Edited by M Belfort, G Saade,

M Foley, J Phelan and G Dildy © 2010 Blackwell Publishing Ltd.

Antiphospholipid Syndrome

T Flint Porter 1 & D Ware Branch 2

1 Department of Obstetrics and Gynecology, University of Utah Health Science UT and Maternal - Fetal Medicine, Urban Central

Region, Intermountain Healthcare, Salt Lake City, UT, USA

2 Department of Obstetrics and Gynecology, University of Utah Health Sciences Center and Women and Newborns Services,

Intermountain Healthcare, Salt Lake City, UT, USA

Introduction

Systemic lupus erythematosus (SLE) is a chronic infl ammatory

condition that affects virtually every organ system With an

increased prevalence among women of reproductive age, it is the

autoimmune disease most commonly encountered during

preg-nancy The majority of women with stable, uncomplicated SLE

tolerate pregnancy well with relatively few serious obstetric

com-plications However, women with poorly controlled disease and/

or serious SLE - related end - organ disease are at substantial risk

for maternal morbidity and even mortality, as well as several

adverse obstetric outcomes Antiphospholipid syndrome (APS)

is another autoimmune condition associated with several adverse

pregnancy outcomes Anticoagulation prophylaxis reduces the

risk of thromboembolism and fetal death but the incidence of

pre - eclampsia, uteroplacental insuffi ciency, and preterm birth

remain high

Management of both SLE and APS during pregnancy requires

vigilance for signs and symptoms of disease exacerbation,

aggres-sive immunosuppresaggres-sive treatment when needed, and careful

assessment of fetal well - being A multidisciplinary approach is

essential and should include the rheumatologist, obstetrician, and

if renal disease is present, the patient ’ s nephrologist

Systemic l upus e rythematosus in p regnancy

Background

The prevalence of SLE varies depending on the population under

study but generally ranges between 5 and 125 per 100 000 and

affects women 5 – 10 times more often than men [1,2] The peak

age of onset in young women occurs between their late teens and

early 40s [3] The prevalence among ethnic groups, such as those

with African or Asian ancestry, is highest and the disease appears

to be more severe when compared to white patients Familial studies indicate that genetic susceptibility to lupus involves several complex gene polymorphisms Russell and colleagues [4] reported a linkage between susceptibility to lupus in family members and disequilibrium in polymorphisms located on the long arm of chromosome 1, 1q23 – 24 Genes coding for C - reactive protein (CRP), actively involved in apoptosis, have been mapped

to this area and CRP levels are commonly low in patients with lupus Another familial study suggested that a single nucleotide

polymorphism within the programmed cell death 1 gene ( PDCD1 )

is associated with the development of the disease in both European and Mexican populations [5]

SLE e xacerbation ( fl are) d uring p regnancy

Based on published reports, fl are occurs in about 30 – 60% of pregnant patients with lupus Renal disease and disease activity appear to increase the rate of fl are during pregnancy There is a split of opinion about whether pregnancy itself predisposes to lupus fl are [2] All studies are hampered by the fact that many of the most common signs and symptoms of lupus fl are also occur

in normal pregnancy Some studies show higher rates of fl are in pregnant women compared to non - pregnant controls, even when disease is inactive at the time of conception [6 – 11] Others report

no difference in rates of fl are in women with well - controlled disease, whether treated or untreated, and non - pregnant women [12 – 18] Importantly, in nearly all studies, fl ares during preg-nancy are reported to be mild to moderate in nature and easily treated with glucocorticoids

Women with pre - existing renal disease, even when inactive, are undoubtedly at greatest risk for SLE fl are during pregnancy [3,9] Pregnancy may predispose to deterioration of renal function, especially for women with active lupus nephritis (LN) and/or renal insuffi ciency before conception [19 – 21] Tandon and col-leagues [22] reported that LN patients with inactive renal disease showed changes in disease activity and deterioration in renal function during pregnancy that were similar to those in non pregnant patients with active LN Overall, about one - third of

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[21] and in 40% with pre - existing proteinuria as defi ned by

> 300 mg/24 h or a creatinine clearance < 100 mL/min [30] Not surprisingly, the most important risk factor for pregnancy loss in women with SLE is coexisting APS In one series of preg-nant women with SLE, the presence of antiphospholipid antibod-ies had a positive predictive value for pregnancy loss of 50% [40]

In another, positive predictive value increased to over 85% if women with SLE also had a fetal death in a prior pregnancy [32]

Neonatal l upus e rythematosus

Neonatal lupus erythematosus (NLE) is a rare condition of the fetus and neonate, occurring in 1 of 20 000 of all live births and

in fewer than 5% of all women with SLE [41] Dermatological NLE is most common and is described as erythematous, scaling annular or elliptical plaques occurring on the face or scalp, analo-gous to the subacute cutaneous lesions in adults Lesions appear

in the fi rst weeks of life, probably induced by exposure of the skin

to ultraviolet light, and may last for up to 6 months [42] Hypopigmentation may persist for up to 2 years A small percent-age of affected infants will go on to have other autoimmune diseases later in life [42] Hematological NLE is rare and may be manifest as autoimmune hemolytic anemia, leukopenia, throm-bocytopenia and hepatosplenomegaly

Cardiac NLE lesions include congenital complete heart block (CCHB) and the less frequently reported endocardial fi broelas-tosis Endomyocardial fi brosis caused by NLE leads to interrup-tion of the conducinterrup-tion system, especially in the area of the atrioventricular node The diagnosis is typically made around 23 weeks of gestation [43] when a fi xed bradycardia, in the range of

60 – 80 beats/min, is detected during a routine prenatal visit Fetal echocardiography reveals complete atrioventricular dissociation with a structurally normal heart The prognosis varies but in the

most severe cases, hydrops fetalis develops in utero Because the

endomyocardial damage is permanent, a pacemaker may be nec-essary for neonatal survival In the largest series of 113 cases diagnosed before birth, 19% died, of which 73% died within 3 months of delivery [43] In that same series, the 3 - year survival was 79% Cutaneous manifestations of NLE have also been reported in infants with CCHB [42]

Not all women who give birth to babies with NLE have been previously diagnosed with an autoimmune disorder [42,44] However, in one study, 7 of 13 previously asymptomatic mothers who delivered infants with dermatologic NLE were later diagnosed with one of several autoimmune disorders [42] Surprisingly, asymptomatic women who deliver infants with CCHB are less likely to later develop an autoimmune disorder than those with dermatologic manifestations alone [44] Fetal immunologic damage is probably caused by maternal autoantibodies that cross the placenta and bind to fetal tissue [45 – 49] Anti - Ro/SSA antibodies are found in 75 – 95% mothers who deliver babies with NLE [43,45,50] A smaller percentage have anti - La/SSB, and some have both [50] Dermatological NLE has also been associated with anti - U1RNP without anti - Ro/SSA

or anti - La/SSB [50,51] Of mothers with SLE who are

serologi-women with renal disease experience fl are during pregnancy,

fewer than 25% have worsening renal function, and only 10% of

have permanent deterioration Renal deterioration appears to be

less severe in women with inactive LN in the 6 months before

conception [9,14,19,21,23,24]

Pulmonary hypertension arises in up to 14% of patients with

lupus, and even mildly raised pulmonary artery pressures can be

seen in 37% of patients [25] Though fortunately rare during

pregnancy, pulmonary hypertension confers an unacceptably

high risk of maternal death and patients should be counseled

accordingly

Obstetric c omplications in w omen with SLE

Women with SLE are at risk for several obstetric complications,

sometimes resulting in serious maternal and perinatal

morbid-ity Between 20 and 30% of women with SLE have pregnancies

complicated by pre - eclampsia [10,21,26] Uteroplacental

insuf-fi ciency resulting in intrauterine growth restriction (IUGR) or

small for gestational age neonates occurs in 12 – 40% of lupus

pregnancies [6,9,12,26,27] The risk of IUGR is highest for

women with renal insuffi ciency and/or hypertension [28,29]

Preterm birth is also more common in pregnancies complicated

by SLE [7 – 10,12,26] Most of preterm deliveries in women with

SLE probably occur iatrogenically because of disease

exacerba-tion and/or obstetric complicaexacerba-tions, though a higher risk of

preterm premature rupture of membranes has been reported

The likelihood of serious obstetric complications is highest for

women with poorly controlled disease, renal disease and/or

chronic hypertension, and APS [10,21,27,28,31,32] Chronic

steroid use may also contribute to higher rates of pre - eclampsia

and IUGR

Pregnancy loss is thought to be more prevalent among women

with SLE, with rates ranging from 10 to 50% [11,16,17,29,33]

First - trimester loss occurs in about 20% of lupus pregnancies, not

markedly higher than the general population [34] However,

still-birth (after 20 weeks of gestation) rates are elevated in several

studies [9,14,21,35,36] In one series, 20% of losses occurred

during the second or third trimester [35] Disease activity

increases the likelihood of pregnancy loss [9,14] with one study

reporting live births in 64% of women with active disease within

6 months of conception, compared to 88% in women with

qui-escent disease [21] In the Hopkins Lupus Pregnancy Cohort,

increased lupus activity did not increase the risk for miscarriage,

but the stillbirth rate was threefold higher [36] The timing of

lupus activity affects the pregnancy loss rate, with activity early

in pregnancy being the most dangerous Proteinuria,

thrombo-cytopenia, and hypertension in the fi rst trimester are each

inde-pendent risk factors for pregnancy loss Women with these risk

factors have a 30 – 40% chance of suffering a pregnancy loss [37]

Accordingly, pregnancy loss is more likely if SLE is diagnosed

during the index pregnancy [38,39]

Renal insuffi ciency is also important; one group reported fetal

loss in 50% of pregnancies complicated by moderate to severe

renal insuffi ciency as defi ned by serum creatinine > 1.5 mg/dL

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cally positive for anti - Ro/SSA antibodies, 15% will have infants

affected with dermatological SLE; the proportion who deliver

infants with CCHB is much smaller However, once a woman

with SLE and anti - Ro/SSA antibodies delivers one infant with

CCHB, her risk for recurrence is at least two - to threefold higher

than other women with anti - SSA/Ro - SSB/La antibodies who have

never had an affected child [45]

There is no known in utero therapy that completely reverses

fetal CCHB secondary to SLE However, there is some evidence

that treatment with glucocorticoids, plasmapheresis, intravenous

immune globulin or some combination thereof, may slow the

progression of prenatally diagnosed CCHB or at least prevent

recurrence in a future pregnancy [52] In utero treatment with

dexamethasone was felt to slow disease progression in one case

report of hydrops secondary to CCHB [53] In one retrospective

study, maternally administered dexamethasone appeared to

prevent progression from second - degree block to third - degree

block [54] In a large series of 87 pregnancies at risk for NLE,

mothers who received corticosteroids before 16 weeks of

gesta-tion were less likely to deliver infants with CCHB compared to

mothers who received no therapy [55] However, there was no

benefi t to treatment when CCHB was diagnosed in utero In utero

treatment with digoxin is not benefi cial for prenatally diagnosed

CCHB [56]

Diagnosis of SLE and d etection of SLE

e xacerbation ( fl are)

Thorough and frequent clinical assessment remains essential for

the timely and accurate detection of SLE fl are [2] In pregnancy,

detection is more diffi cult because many of the typical signs and

symptoms associated with fl are are considered normal (Table

36.1 ) The SLE disease activity index (SLEDAI) has been modifi ed

Table 36.1 Common symptoms in pregnancy that may mimic lupus fl are

Constitutional • Debilitating fatigue; may worsen throughout

pregnancy

Skin • Palmar erythema

• Facial blush due to increased estrogen levels

Face • Melasma; photosensitive rash over cheeks and

forehead

Hair • Increased hair thickness and growth

• Hair loss postpartum

Pulmonary • Increased respiratory rate secondary to

increased progesterone levels

• Dyspnea

Musculoskeletal • Back pain

䊊 Relaxin loosens sacroiliac joint and pubis

䊊 Gravid uterus increases lumbar lordosis

• Joint effusions

Central Nervous System • Headache, normal in pregnancy as well as in

preeclampsia

Table 36.2 Preeclampsia versus Lupus Flare

Preeclampsia SLE Flare

• Gestational age After 20 weeks ’ Anytime

• Complement (C3 & C4) Normal Usually low

• Anti - DS DNA antibodies Negative Usually positive

for pregnancy with several caveats to rule out normal pregnancy complications and thereby more accurately identify true SLE activity [2,43] ) The most common presenting symptom in both

fl are and new onset disease is extreme fatigue Fever, weight loss, myalgia and arthralgia are also very common [57] In pregnancy, skin rashes are more frequent than musculoskeletal manifesta-tions [8] Patients with LN exhibit worsening proteinuria along with pyuria, hematuria and urinary casts Not surprisingly, SLE fl are in pregnant women with LN is easily confused with the development of pre - eclampsia/eclampsia syndromes (Table 36.2 )

Serological evaluation of SLE disease activity may be benefi cial

in confi rming fl are in confusing cases However, no study has found serial laboratory testing superior to thorough clinical assessment and if fl are is suspected, treatment should not be reserved only for women with positive serologic evaluation Even

so, the most specifi c serologic sign of SLE fl are is an elevation in anti - double - stranded DNA (anti - ds DNA) which precedes lupus

fl are in more than 80% of patients [58 – 60] In pregnancy, ele-vated anti - ds DNA titers have also been shown to correlate with the need for preterm delivery [61] and in combination with aCL antibodies, with an increased risk of fetal loss

Serial evaluation of complement levels has been suggested as method of predicting SLE fl are during pregnancy Devoe [62] reported that SLE fl are was signaled by a decline of C3 and C4 into the subnormal range and Buyon [63] reported that the phys-iologic rise in C3 and C4 levels normally seen during pregnancy did not occur in women with active disease The same group reported that activation of the alternative complement pathway

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chronic glucocorticoid therapy are the same as in non - pregnant patients and include weight gain, striae, acne, hirsutism, immu-nosuppression, osteonecrosis and gastrointestinal ulceration During pregnancy, chronic glucocorticoid therapy has also been associated with an increased risk of pre - eclampsia [35,73 – 75] , uteroplacental insuffi ciency [75] , and glucose intolerance [73,74] Women chronically treated with glucocorticoids should be screened for gestational diabetes at 22 – 24, 28 – 30 and 32 – 34 weeks of gestation

Hydroxychloroquine

Hydroxychloroquine (HCQ) has been proven to decrease the risk

of SLE fl are, improve the prognosis of SLE nephritis, and prevent death [76 – 79] It also has the lowest side - effect profi le of any medication available to treat SLE and is well tolerated by most patients In the past, many patients and their physicians discon-tinued hydroxychloroquine in pregnancy because of concerns about teratogenicity including ototoxicity [80] and eye damage [81] However, an accumulating body of evidence suggests that hydroxychloroquine can be used safely for the treatment of SLE during pregnancy [82 – 86] and is in fact superior to glucocorti-coids for women who require maintenance therapy during preg-nancy [87] An expert panel of international physicians have recommended the continuation of HCQ during pregnancy [79]

Immunosuppressants

Azathioprine (Imuran) is probably the safest immunosuppres-sant medication taken during pregnancy The fetal liver does not have the enzyme required to metabolize azathioprine into its active form [79] Series of pregnancies exposed to azathioprine for infl ammatory bowel disease or renal transplants show no signifi cant increase in fetal abnormalities among renal transplant patients Nearly 40% of the offspring were small for gestational age But this may also have been due to the underlying illness and/

or contemporaneous treatment with glucocorticoids [78,79] Most authorities recommend continuation of azathioprine treat-ment during pregnancy [57] Cyclophosphamide is reportedly teratogenic in both animal [88] and human studies [89,90] and should be avoided during the fi rst trimester Thereafter, cyclo-phosphamide should be used only in unusual circumstances such

as in women with severe, progressive proliferative glomerulone-phritis [1] Methotrexate is well known to kill chorionic villi and cause fetal death and its use should be scrupulously avoided

NSAID p reparations

The most common types of analgesics used in the treatment of SLE are non - steroidal anti - infl ammatory drugs (NSAIDs) Unfortunately, their use in pregnancy should be avoided after the

fi rst trimester because they readily cross the placenta and block prostaglandin synthesis in a wide variety of fetal tissues Though short - term tocolytic therapy with indomethacin appears to be safe [91,92] , chronic use has been associated with a number of untoward fetal effects and, when used after 32 weeks, may result

in constriction or closure of the fetal ductus arteriosus [93]

accompanies fl are during pregnancy [63] and that a combination

of low C3, C4, or CH50 levels accompanied by an elevation in

complement split products is useful in detecting fl are during

pregnancy [63] The results of other studies of complement

acti-vation in pregnant women with SLE have either been inconsistent

or not predictive of SLE fl are [11,64,65] Lockshin [66] reported

normal concentrations of the Cls – C1 inhibitor complex in

preg-nant patients with hypocomplementemia which suggests poor

synthesis of complement components rather than excessive

consumption

Laboratory confi rmation of SLE fl are is probably most helpful

in women with active LN in whom proteinuria, hypertension and

evidence of multiorgan dysfunction may easily be confused with

pre - eclampsia Both elevated anti - ds DNA titers and urinary

sedi-ment with cellular casts and hematuria weigh in weigh in favor

of active LN An increase in proteinuria in women with pre

existing LN should not necessarily raise an alarm until it doubles

[57] Pre - eclampsia is more likely in women with decreased levels

of antithrombin - III [67,68] Complement concentrations are not

helpful because activation may also occur in women with pre

eclampsia [69] In the most severe and confusing cases, the

diag-nosis can be confi rmed only with renal biopsy However, in

reality, concerns about maternal and fetal well - being often

prompt delivery, rendering the distinction between the SLE fl are

and pre - eclampsia clinically moot

Medications u sed for SLE d uring p regnancy

Glucocorticoids

The group of drugs most commonly given to pregnant women

with SLE is the glucocorticoid preparations, both as maintenance

therapy and in “ bursts ” to treat suspected SLE fl ares The doses

used in pregnancy are the same as those in non - pregnant patients

Pregnancy per se is not an indication to reduce the dose of

glu-cocorticoids, though a carefully monitored reduction in dosage

may be reasonable in appropriately selected women whose disease

appears to be in remission Some groups have recommended

prophylactic glucocorticoid therapy during pregnancy [17,24,70]

but no controlled studies have shown this practice to be prudent

or necessary in women with inactive SLE Moreover, good

mater-nal and fetal outcomes are achieved without prophylactic

treat-ment of women with stable disease [13] In contrast, glucocorticoid

treatment of women with active disease and/or elevated anti - ds

DNA titers has been shown to result in fewer relapses and better

pregnancy outcomes [14,58]

While glucocorticoids have a low potential for teratogenesis

[71] , they are not without risk during pregnancy Patients

requir-ing chronic maintenance therapy are best treated with

predniso-lone or methylprednisopredniso-lone because of their conversion to

relatively inactive forms by the abundance of 11 - ,B - ol

dehydro-genase found in the human placenta Glucocorticoids with fl

uo-rine at the 9a position (dexamethasone, betamethasone) are

considerably less well metabolized by the placenta and chronic

use during pregnancy should be avoided Both have been

associ-ated with untoward fetal effects [72] Maternal side effects of

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