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observed acute renal failure in 7.4% 32/435 of patients with HELLP syndrome, and approximately one - third of these patients required hemodialysis [32].. HELLP syndrome associated with a

Trang 1

fl exion and then compresses the bladder [19,20] Other risk factors for urinary obstruction in pregnancy include pyelonephri-tis, renal calculi, ureteral narrowing, and low abdominal wall compliance [21]

Renal ultrasound is the fi rst step in the evaluation of possible urinary tract obstruction, although results may be inconclusive due to the physiologic dilation of the collecting system often seen

in pregnancy due to both the effects of progesterone and the mechanical pressure of the gravid uterus Thus, anterograde or retrograde pyelography may be necessary for defi nitive diagnosis Relief of the obstruction may be accomplished by ureteral stent placement, percutaneous nephrostomy, manual reduction of an incarcerated uterus, or amnioreduction in the case of polyhy-dramnios If the fetus is signifi cantly premature, correcting the obstruction should allow for a substantial delay in delivery

as well as recovery of renal function If the patient is near term, however, delivery may be indicated to remove both the mechani-cal and hormonal causes of the obstruction It should be noted that the fetal mortality rate for reversible obstructive uropathy with associated renal failure has been reported to be as high as 33% [22]

Pyelonephritis

Pyelonephritis is an important cause of ARF during pregnancy

As a result of the normal physiologic changes that accompany pregnancy, the urinary collecting system is prone to dilation and urinary stasis In addition, there is an increased sensitivity to bacterial endotoxin - induced tissue damage These normal changes result in an increased incidence in both upper and lower tract infections The incidence of pyelonephritis in pregnancy is approximately 2% and it is one of the most common causes of sepsis during pregnancy [23] Presenting symptoms generally include fever, fl ank pain, nausea, vomiting, and possibly urinary frequency, dysuria, and urgency The most common causative

organism is E coli , which accounts for nearly 75% of cases [24] Other potential pathogens include Proteus mirabilis , Klebsiella pneumoniae , group B streptococci, enterococci, and Pseudomonas aeruginosa Prompt and appropriate antibiotic treatment is

gen-erally very effective in treating pyelonephritis during pregnancy, with improvement seen in the fi rst 24 – 48 hours After resolution

of the initial infection, suppressive antibiotic treatment through-out pregnancy should be considered as the recurrence rate is as high as 20%

Although pyelonephritis rarely results in a signifi cant decline

in renal function in non - pregnant patients, Gilstrap and col-leagues demonstrated a substantial decrease in creatinine clear-ance among gravidas with pyelonephritis, with a return to normal

or near normal renal function in the majority of women re -evaluated following antibiotic therapy [25,26] As mentioned previously, it has been postulated that this decline in renal func-tion is related to an increased vascular sensitivity to bacterial endotoxins and vasoactive mediator release in pregnancy [1] It

ATN may occur in cases of rhabdomyolysis or massive hemolysis

More commonly in pregnancy, however, ATN is ischemic in

nature, as a result of a hemodynamic insult with hypotension and

impaired renal perfusion This is commonly due to a hemorrhage

during pregnancy, which may be the result of either placental

abruption or a postpartum hemorrhage which complicates

approximately 1% and 4 – 6% of pregnancies respectively [16]

In those patients with pre - eclampsia who develop renal failure,

ATN appears to be the underlying renal lesion Clinically, it may

be diffi cult to distinguish between severe prerenal azotemia and

ATN, although urinary indices and urinalysis may be helpful

(Table 28.2 ) Urinalysis typically reveals muddy brown granular

casts and renal tubular epithelial cells In light of impaired renal

tubular function, laboratory evaluation reveals a high urinary

sodium excretion as well as urine that is neither concentrated nor

dilute Acute tubular necrosis may be either oliguric (urine

output < 400 mL/day) or non - oliguric ( > 400 mL/day), depending

on the mechanism of injury and the severity Treatment of ATN

is supportive and necessitates optimization of hemodynamics,

avoidance of potential nephrotoxin exposure, nutritional support

with careful monitoring of fl uids and electrolytes and,

occasion-ally, dialysis Renal function typically recovers in 7 – 14 days with

appropriate treatment

Urinary o bstruction

Although urinary obstruction is a relatively uncommon cause of

ARF in pregnancy, it is readily reversible and, therefore, must be

considered in the differential Obstruction may occur at any level

of the urinary tract due to a wide variety of causes, many of which

are not unique to pregnancy (Table 28.6 ) Additionally, gravidas

with an abnormally confi gured or overdistended uterus, such as

those with uterine leiomyomata, polyhydramnios, or multiple

gestations, may be particularly susceptible Ureteral compression

by the gravid uterus, with resultant ARF and hypertension, has

been reported [17] and large leiomyomata have even been

reported to cause ureteral obstruction in the fi rst trimester [18]

Another cause unique to pregnancy is an incarcerated uterus,

which may cause urinary retention as the gravid uterus enlarges

but becomes trapped in the pelvis secondary to signifi cant

Table 28.6 Causes of urinary obstruction

Blood clots Blood clots

Sloughed papillae Neuropathic bladder

Ureteral stricture or ligation Urethral stricture

Retroperitoneal fi brosis

Extrinsic compression by tumor, gravid uterus

Trang 2

of underlying chronic renal disease, presumably unmasked by pregnancy and/or pre - eclampsia [5]

HELLP s yndrome

HELLP is an acronym used to describe a constellation of fi ndings,

including h emolysis, e levated l iver enzymes, and l ow p latelets

Nausea, epigastric or right upper quadrant pain, and tenderness may be present at the time of diagnosis, as well as proteinuria and renal dysfunction Coagulation studies including fi brinogen, pro-thrombin time, and partial thromboplastin time may be useful in distinguishing this disorder from others associated with dissemi-nated intravascular coagulation (DIC), in that they are often normal in patients with HELLP syndrome in the absence of pla-cental abruption

HELLP syndrome has been described in 4 – 12% of patients with severe pre - eclampsia [30] and is considered to represent

a variant of severe pre - eclampsia However, in a small study

by Krane, in which patients with HELLP syndrome underwent renal biopsy, less than half had the glomerular endotheliosis classic for pre - eclampsia [31] Sibai et al observed acute renal failure in 7.4% (32/435) of patients with HELLP syndrome, and approximately one - third of these patients required hemodialysis [32] Evidence of disseminated intravascular coagulation was present in 84% of these patients, and 44% had abruptio placen-tae HELLP syndrome associated with acute renal failure in this study carried a maternal mortality rate of 13% and perinatal mortality rate of 34% The poor prognoses described by Sibai likely refl ect the severity of disease seen in his patient population

Generally, treatment of HELLP syndrome consists of expeditious delivery once the diagnosis is established, as well as magnesium sulfate for seizure prophylaxis as discussed earlier, with rapid recovery of renal function expected In a group of 23 patients with HELLP syndrome who were normotensive prior to pregnancy, no residual renal impairment was observed following delivery However, 40% of patients with chronic hypertension and subsequent HELLP syndrome eventually required chronic dialysis [32,33]

Acute f atty l iver of p regnancy

Acute fatty liver of pregnancy is another uncommon cause of ARF

in pregnancy, with an incidence reported as between 1 in 6700 and 1 in 13 000 deliveries [34,35] The disease exhibits a slight predominance in nulliparas; it has been diagnosed as early as 24 weeks of gestation and as late at 7 days postpartum [4,35] , but usually occurs in the last few weeks of gestation Initial manifesta-tions are non - specifi c, including nausea, vomiting, headache, malaise, and abdominal pain Laboratory evaluation reveals mild elevation of serum transaminase levels, hyperbilirubinemia, and leukocytosis as well as hypoglycemia Renal failure develops in

is this sensitivity to endotoxin that may account for the greater

incidence of septic shock and adult respiratory distress syndrome

from pyelonephritis during pregnancy

Pre - e clampsia

Among those causes of ARF unique to pregnancy, pre - eclampsia/

eclampsia accounts for the majority One study of ARF in

preg-nancy performed in Uruguay, which included patients from 1976

to 1994, reported that pre - eclampsia was the cause of ARF in

approximately 47% of cases [27] Another retrospective study

conducted at an inner city hospital in Georgia described pre

eclampsia in more than one - third of 21 cases of ARF diagnosed

at their institution from 1986 to 1996 [7]

Classically, pre - eclampsia is defi ned as the development of

hypertension, proteinuria, and edema after the 20th week of

ges-tation (It should be noted, however, that severe pre - eclampsia

may occur earlier than 20 weeks in the presence of gestational

trophoblastic disease, also called a molar pregnancy.) Elevated

liver enzymes, coagulation abnormalities, and microangiopathic

hemolytic anemia may be seen in severe pre - eclampsia as well

The diagnosis is established clinically and rarely confi rmed by

renal biopsy

Pathologically, pre - eclampsia is characterized by swollen

glo-merular capillary endothelial cells or gloglo-merular endotheliosis,

with resultant capillary obstruction and glomerular ischemia

[28] Importantly, the extent of the morphologic lesion does

not necessarily correspond to the degree of renal functional

impairment [4] In addition, the presence of subtle volume

depletion and enhanced sensitivity of the renal vasculature to

vasoconstriction may contribute to superimposed ATN, which

many believe to be the lesion associated with signifi cant ARF in

pre - eclampsia

Treatment of severe pre - eclampsia and the associated renal

failure ultimately depends on delivery of the infant and seizure

prophylaxis with magnesium sulfate during the delivery and for

at least 24 hours postpartum This is accomplished regardless of

the gestational age of the fetus, though consultation with a

mater-nal - fetal medicine specialist is recommended with a premature

fetus to determine if it is possible to delay delivery long enough

to administer corticosteroids in an attempt to improve fetal lung

maturity It is important to monitor fl uid administration closely

while magnesium sulfate is given as patients with impaired renal

function will not clear the medication as well and dose reductions

may be necessary

Recovery of renal function is usually seen within days to weeks

after delivery with isolated pre - eclampsia, although up to 20%

may have some degree of residual impairment [29] In contrast,

when patients with chronic hypertension and underlying renal

disease experience ARF in pregnancy, approximately 80% will

require long - term renal replacement therapy [15] Histologic

evaluation in those patients with persistent renal impairment,

proteinuria, or hypertension postpartum has revealed evidence

Trang 3

those receiving such treatment Additional therapeutic interven-tions varied, including aspirin, dipyridamole, and corticosteroids Greater than 50% of all patients had evidence of renal dysfunc-tion, although those with severe ARF or anuria were excluded from the Canadian multicenter trial

Delivery in cases of TTP/HUS is not necessarily indicated, especially at very early gestational ages, which is why care must be taken to differentiate this disease from severe pre - eclampsia

Nine of the 76 women seen at Johns Hopkins presented in their third trimester of pregnancy, although there was no comment as

to the degree of renal impairment in this subset of patients A recent report of three patients with postpartum HUS at the Rhode Island hospital who were treated with frequent plasma exchange and prednisone reported survival in all three patients [41] Additionally, Hayward and colleagues described nine preg-nant women presenting between the fi rst trimester of gestation and 1 month postpartum with TTP - HUS [42] Of these 21 women from three institutions, all but one survived, and none required renal replacement therapy With respect to future preg-nancies, one recent report cites only an 18% recurrence risk in subsequent pregnancies in patients with a history of postpartum TTP/HUS [43]

Postpartum r enal f ailure

Idiopathic postpartum renal failure, also referred to as postpar-tum HUS, is a unique cause of pregnancy - associated ARF that typically develops in the puerperium following an uncomplicated pregnancy and delivery Women may present up to several months following delivery with severe hypertension, microangio-pathic hemolytic anemia, and oliguric renal failure, often with congestive heart failure and CNS manifestations A prodromal

fl u - like illness or initiation of oral contraceptives may be associ-ated with postpartum renal failure as well as with idiopathic HUS, suggesting a toxic or hormonal infl uence

Pathologically, the disease is often indistinguishable from the thrombotic microangiopathies, idiopathic HUS and TTP, with arteriolar injury, fi brin deposition, and microvascular (arteriolar and glomerular capillary) thrombosis The major pathologic involvement is renal, as opposed to CNS involvement seen in TTP The pathogenesis of the thrombotic microangiopathies remains unclear, although intravascular coagulation, disordered platelet aggregation, endothelial damage, and alterations in pros-taglandins have been suggested [44] Therapies have been chosen

in an attempt to intervene in one or more of these processes, including plasma exchange, plasma infusion, antiplatelet agents, and anticoagulation In addition, acute and long - term dialytic support is often necessary, with approximately 12 – 15% of patients developing end - stage renal disease The maternal mortal-ity rate was estimated at between 46% and 55% in the 1980s [45,46] but appears to be improving with the use of plasma exchange and other treatments

the majority of cases and, left untreated, patients may progress to

fulminant hepatic failure with jaundice, encephalopathy,

dis-seminated intravascular coagulopathy, gastrointestinal

hemor-rhage, and death Maternal and fetal mortality rates as high as

85% were seen in the past, although with earlier diagnosis and

treatment a recent analysis of 28 consecutive cases reported no

maternal deaths [35]

Diagnosis of fatty liver may be established by liver biopsy

revealing microvesicular fatty infi ltration Computed

tomogra-phy (CT) may reveal decreased hepatic attenuation A report by

Usta and colleagues described their experience with 13 patients

(14 cases) of AFLP over an 8 - year period, all of whom had ARF

on presentation [36] They reported 100% maternal survival,

with 13% perinatal mortality Although nine of 14 cases were

initially diagnosed as pre - eclampsia, the diagnosis of AFLP was

subsequently confi rmed either by liver biopsy (10/14), CT of the

liver (2/14), or clinically One patient experienced a recurrence

of AFLP in a subsequent pregnancy Although CT revealing

hepatic density below the normal range of 50 – 70 Hounsfi eld

units has been reported as suggestive of AFLP, Usta ’ s study

dem-onstrated a high false - negative rate with only two of 10 abnormal

scans, including nine biopsy - proven cases [36] Contributing to

the diagnostic dilemma in these women is the frequent

occurrence of hypertension, edema, and proteinuria suggestive of pre

eclampsia, although renal pathology has failed to reveal evidence

of glomerular endotheliosis As is the case with severe pre

-eclampsia, expeditious delivery is warranted, with prompt

improvement in both hepatic and renal failure noted in nearly all

cases [34,35]

Thrombotic t hrombocytopenic

p urpura/ h emolytic u remic s yndrome

Thrombotic thrombocytopenic purpura/hemolytic uremic

syn-drome (TTP/HUS) is an uncommon disorder during pregnancy

with an incidence of approximately 1 in 25 000 births [37] It is

characterized by the classic pentad of thrombocytopenia,

hemo-lytic anemia, fever, neurologic abnormalities, and some degree of

renal dysfunction During pregnancy, the disorder tends to

present earlier than pre - eclampsia, with a median gestational age

of onset of 23 weeks [38] The underlying pathophysiology of the

disorder is apparently due to intravascular thrombi that result in

fragmentation of red blood cells, platelet consumption, and

varying degrees of systemic ischemia

Although treatment guidelines are not well established, plasma

exchange is recommended due to an apparent benefi t in survival

in a small number of patients Due to the continuum of disease,

both HUS and TTP have been considered together in most

clini-cal trials The Canadian Apheresis Study Group and a group at

Johns Hopkins University examined therapeutic outcomes in

TTP and TTP/HUS, respectively [39,40] Both reported the

supe-riority of plasma exchange therapy in terms of clinical response

and survival, with mortality rates of 22% and 9% respectively, in

Trang 4

tion recovers Close attention to fl uid balance is critical because either superimposed volume depletion or fl uid overload may exacerbate ARF or necessitate earlier dialytic intervention In addition, magnesium sulfate administration in cases of pre eclampsia may also increase the patient ’ s risk for fl uid overload

or toxicity from the medication and should be monitored closely Correction of the metabolic acidosis seen with ARF may require bicarbonate therapy or dialysis, if it remains refractory to medical therapy or occurs in the setting of congestive heart failure Prevention of hyperphosphatemia includes dietary phosphate restriction and non - absorbable or calcium - containing phosphate binders given with meals Dietary potassium restriction also is imperative to avoid potentially life - threatening hyperkalemia A cation - exchange resin, such as kayexalate, can be used for mild hyperkalemia or until dialysis is available For hyperkalemia with associated electrocardiographic changes, acute therapy includes intravenous calcium gluconate to stabilize the cardiac membrane, infusion of glucose and insulin or inhaled β - agonists to transiently shift potassium intracellularly, and acute dialysis Additional conservative measures include avoiding further neph-rotoxic exposure and hypotension, control of hypertension, and medication dose adjustment according to the degree of renal impairment

In patients with severe metabolic abnormalities that are unre-sponsive to conservative medical management, volume overload and pulmonary congestion that cannot be corrected with diuret-ics, or signs and symptoms of uremia including pericarditis and encephalopathy, dialysis is indicated

As discussed previously, if the underlying etiology is deter-mined to be severe pre - eclampsia, then delivery may be indicated, even at very early gestational ages as there is no other way to prevent progression of the disease

Prognosis

The prognosis for return of renal function depends on multiple variables, including baseline renal status, duration of renal failure,

Bilateral r enal c ortical n ecrosis

Acute, bilateral renal cortical necrosis is a pathologic entity

con-sisting of partial or complete destruction of the renal cortex, with

sparing of the medulla While not unique to pregnancy, this rare

and catastrophic form of ARF occurs most commonly in

preg-nancy, with obstetric causes accounting for 50 – 70% of cases [47]

Although BRCN represents less than 2% of cases of ARF in the

non - pregnant population, it has been reported to account for

10 – 38% of obstetric cases of renal failure, perhaps secondary to

the hypercoagulable state and altered vascular sensitivity of

preg-nancy [31,48] Patients typically present between 30 and 35 weeks

of gestation in association with profound shock and renal

hypo-perfusion, such as that seen with abruptio placentae, placenta

previa, and other causes of obstetric hemorrhage Acute BRCN has

also been observed early in pregnancy associated with septic

abor-tion Abruption placentae, with either overt or concealed

hemor-rhage, appears to be the most common antecedent event [47]

Patients with BRCN present with severe and prolonged

oliguria or anuria (urine output < 50 mL/day), fl ank pain, gross

hematuria, and urinalysis demonstrating RBC and granular casts

Diagnosis is established by renal arteriogram demonstrating

virtual absence of cortical blood fl ow (interlobular arteries),

despite patency of the renal arteries Diagnosis may also be

estab-lished by ultrasonography, contrast - enhanced CT demonstrating

areas of cortical lucency, and MRI [49] The prognosis for patients

with BRCN is extremely poor, again likely related to the severity

of illness, with one study of 15 cases during pregnancy reporting

a mortality rate of 93% [48]

Management of a cute r enal f ailure

Management of ARF in pregnancy is similar to that in the non

pregnant patient, including supportive therapy as well as dialysis

General principles include treating the underlying cause,

preven-tion of further renal injury, and supportive care until renal

Table 28.7 Classifi cation of pregnancy - associated acute renal failure

Pre - eclampsia HELLP syndrome Acute fatty Postpartum Pyelonephritis Bilateral renal

liver of pregnancy (HUS) renal failure cortical necrosis

Proteinuria RUQ pain Elevated LFTs Occurring postpartum Positive urine culture Hemorrhage

Hypertension Proteinuria Hyperbilirubinemia MAHA Fever Hypotension/shock

Edema Hemolysis Coagulopathy Oliguria Oliguria/anuria

Elevated LFTs Oliguria Severe HTN Flank pain Thrombocytopenia Nausea Prodromal illness Gross hematuria Normal coags Abdominal pain Thrombocytopenia

HTN, hypertension; LFTs, liver function tests; MAHA, microangiopathic hemolytic anemia; RUQ, right upper quadrant

Trang 5

13 Turton P , Hughes P , Bolton H , Sedgwick P Incidence and demo-graphic correlates of eating disorder symptoms in a pregnant

popula-tion Int J Eat Disord 1999 ; 26 : 448 – 452

14 Grunfeld JP , Ganeval D , Bournerias F Acute renal failure in

preg-nancy Kidney Int 1980 ; 18 : 179 – 191

15 Sibai BM , Villar MA , Mabie BC Acute renal failure in hypertensive disorders of pregnancy Pregnancy outcome and remote prognosis in

thirty - one consecutive cases Am J Obstet Gynecol 1990 ; 162 ( 3 ):

777 – 783

16 Ovelese Y , Ananth CV Placental abruption Obstet Gynecol 2006 ; 108 :

1005 – 1016

17 Satin AJ , Seiken GL , Cunningham FG Reversible hypertension in

pregnancy caused by obstructive obstetric uropathy Obstet Gynecol

1993 ; 81 : 823 – 825

18 Courban D , Blank S , Harris MA , Bracy J , August P Acute renal failure

in the fi rst trimester resulting from uterine leiomyomas Am J Obstet Gynecol 1997 ; 177 ( 2 ): 472 – 473

19 Myers DL , Scotti RJ Acute urinary retention and the incarcerated,

retroverted, gravid uterus A case report J Reprod Med 1995 ; 40 ( 6 ):

487 – 490

20 Nelson MS Acute urinary retention secondary to an increased gravid

uterus Am J Emerg Med 1986 ; 4 ( 3 ): 231 – 232

21 Brandes JC , Fritsche C Obstructive acute renal failure by a gravid uterus: a case report and review Am J Kidney Dis 1991 ; 18 :

398 – 401

22 Khanna N , Nguyen H Reversible acute renal failure in association with bilateral ureteral obstruction and hydronephrosis in pregnancy

Am J Obstet Gynecol 2001 ; 184 ( 2 ): 239 – 240

23 Cunningham FG , Lucas MJ Urinary tract infections complicating

pregnancy Bailli è re ’ s Clin Obstet Gynaecol 1994 ; 8 : 353 – 373

24 Davison JM , Lindheimer MD Renal disorders In: Creasy RK , Resnick

R , eds Maternal Fetal Medicine , 4th edn Philadelphia : WB Saunders ,

1999 : 873 – 894

25 Whalley PJ , Cunningham FG , Martin FG Transient renal dysfunction

associated with acute pyelonephritis of pregnancy Obstet Gynecol

1975 ; 46 : 174 – 177

26 Gilstrap LC , Cunningham FG , Whalley PJ Acute pyelonephritis

during pregnancy: an anterospective study Obstet Gynecol 1981 ; 57 :

409 – 413

27 Ventura JE , Villa M , Mizraji R , Ferreiros R Acute renal failure in

pregnancy Ren Fail 1997 ; 19 ( 2 ): 217 – 220

28 Antonovych TT , Mostofi FK Atlas of Kidney Biopsies Washington,

DC : Armed Forces Institute of Pathology , 1981 : 266 – 275

29 Suzuki S , Gejyo F , Ogino S Post - partum renal lesions in women

with pre - eclampsia Nephrol Dial Transplant 1997 ; 12 : 2488 –

2493

30 Martin JN , Blake PG , Perry KG , et al The natural history of HELLP

syndrome: patterns of disease progression and regression Am J Obstet Gynecol 1991 ; 164 : 1500 – 1513

31 Krane NK Acute renal failure in pregnancy Arch Intern Med 1988 ;

148 : 2347 – 2357

32 Sibai BM , Ramadan MK Acute renal failure in pregnancies

compli-cated by hemolysis, elevated liver enzymes, and low platelets Am J Obstet Gynecol 1993 ; 168 : 1682 – 1690

33 Nakabayashi M , Adachi T , Itoh S , Kobayashi M , Mishina J , Nishida

H Perinatal and infant outcome of pregnant patients undergoing

chronic hemodialysis Nephron 1999 ; 82 : 27 – 31

34 Kaplan MM Acute fatty liver of pregnancy N Engl J Med 1985 ; 313 :

367 – 370

and the etiology of the ARF For instance, if the patient had

normal renal function before ARF from an acute obstructive

process that is relieved in a timely manner, then a full recovery

should be expected On the other hand, as previously discussed,

studies have demonstrated that, of patients with compromised

renal function who develop pre - eclampsia with ARF, up to 80%

may require long - term dialysis [15]

Summary

Evaluation of the pregnant patient with ARF encompasses a

broad range of disorders, some of which are unique to pregnancy

Prerenal azotemia, intrinsic renal disease, including ATN, GN,

and interstitial nephritis, and urinary obstruction should be

con-sidered based on clinical presentation Evaluation of ARF during

pregnancy is similar to that in the non - pregnant patient,

includ-ing urinalysis and urinary diagnostic indices, and in some cases,

renal biopsy In addition, diseases unique to pregnancy and those

more common during pregnancy must be considered, including

pre - eclampsia, HELLP syndrome, AFLP, postpartum renal

failure, and BRCN (Table 28.7 ) Treatment may necessitate

prompt delivery of the infant, even at early gestational ages when

issues of prematurity may exist

References

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3 Stratta P , Besso L , Canavese C , et al Is pregnancy - related acute renal

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4 Lindheimer MD , Katz AI , Ganeval D , et al Acute renal failure in

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Trang 6

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in patients with adult thrombotic thrombocytopenic purpura -

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45 Weiner CP Thrombotic microangiopathy in pregnancy and the

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28 consecutive cases Am J Obstet Gynecol 1999 ; 181 ( 2 ): 389 – 395

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hemolytic uremic syndrome Mayo Clin Proc 2001 ; 76 : 1154 – 1162

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thrombotic thrombocytopenic purpura - hemolytic uremic syndrome

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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.

T Flint Porter

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

Introduction

Acute fatty liver of pregnancy (AFLP) is a rare, yet potentially

fatal complication of late pregnancy Also known as acute fatty

metamorphosis or acute yellow atrophy, the incidence ranges

between 1 in 7000 and 1 in 15 000 depending on the population

studied [1 – 3] Older published series reported maternal and

peri-natal mortality rates as high as 75 and 85%, respectively [4]

However, more recent experience suggests that both morbidity

and mortality can be reduced by early recognition and prompt

treatment [1,2,5]

Epidemiology

The majority of cases of AFLP occur during the third trimester

[1,5,6] , usually between 30 and 38 weeks of gestation [3] ; some

do not become clinically evident until after delivery [7] Rare

mid - trimester cases have also been reported [8,9] There are no

clear epidemiologically distinct risk factors for AFLP Neither

maternal age nor ethnicity appears to affect risk Most affected

women are in their fi rst pregnancy [7] though AFLP has been

diagnosed in multiparous women with otherwise normal

obstet-ric histories Recurrence in subsequent pregnancy has also been

reported [10,12] Additional suggested risk factors include the

presence of a male fetus [13] , and multiple gestation [7,14]

Pathogenesis

The pathogenesis of AFLP has not been fully elucidated but

abnormalities in mitochondrial fatty acid oxidation likely play an

important role Fatty acid oxidation (FAO) is the major source

of energy for skeletal and heart muscle, a process that occurs primarily in the liver during conditions of prolonged fasting, illness, and increased muscular activity [3] Hepatic FAO also plays an essential role in intermediary liver metabolism and syn-thesizes alternative sources of energy for the brain when blood glucose levels are low [13]

Mitochondrial FAO functions via a protein complex known as mitochondrial trifunction protein (MTP) It is composed of three enzymes, one of which is long - chain 3 - hydroxyacyl - CoA dehy-drogenase (LCHAD) Human defects in MTP have emerged as

an important group of metabolic errors because of their serious clinical implications (Figure 29.1 ) They are recessively inherited and result in either isolated LCHAD defi ciency or dramatically reduced functionality of all three of the MTP enzymes Most reported cases involve children with isolated LCHAD defi ciency who present within the fi rst few hours to months of life with non - ketotic hypoglycemia and hepatic encephalopathy, which progresses to coma and death if untreated [15,16] Cardiomyopathy, slowly progressing peripheral neuropathy, skeletal myopathy, or sudden, unexpected death are also reported [17,18]

Schoeman [11] and colleagues were the fi rst group to suggest

an association between recurrent maternal AFLP and a fetal fatty acid oxidation disorder in two siblings, both whom died at 6 months of age [16] Other reports of a potential causative rela-tionship followed [15,19 – 22] In one series of 12 affected preg-nancies, several offspring delivered of mothers with AFLP were diagnosed postnatally with a homozygous form of LCHAD [19,23] Parental heterozygosity was subsequently confi rmed LCHAD defi ciency was later reported in three families in associa-tion with pregnancies complicated by AFLP [20] Ibdah [15] reported that 80% of mothers who delivered babies with

con-fi rmed MTP defects developed either AFLP or HELLP during their pregnancy Three of them had a history of AFLP in a previ-ous pregnancy In a subsequent prospective study, the same group [24,25] found that in approximately 1 in 5 pregnancies complicated by AFLP, the fetus is LCHAD - defi cient These fi nd-ings support the potentially life - saving role of screening for MTP defects in children born to women with AFLP Prenatal diagnosis

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ops and leads to oliguria and acute tubular necrosis [1] In turn, damage to the proximal renal tubules results in decreased sensi-tivity to vasopressin and transient diabetes insipidus [27,28] Laboratory evidence of renal dysfunction is evident early in the disease with increased serum creatinine levels Uric acid and blood urea nitrogen concentrations are also elevated, and with the onset of jaundice, urobilinogen appears in the urine Serum electrolytes may refl ect metabolic acidosis and plasma glucose is often below 60 mg/dL suggesting reduced hepatic glycogenolysis [29] It is not uncommon for mild hypoglycemia to be masked

in subsequent pregnancies has also been performed using

chori-onic villus sampling in an effort to identify at - risk pregnancies

[26]

Clinical p resentation

The clinical presentation of AFLP is non - specifi c and most

com-monly includes nausea, vomiting, anorexia, tachycardia, and

abdominal pain (Table 29.1 ) [1,3,5,7] Symptoms may develop

suddenly or over a 2 – 3 week period Though liver size is usually

normal or small, 50% of women with AFLP are jaundiced and

complain of right upper quadrant or epigastric pain Fever,

headache, and pruritus are not uncommon [1,3] Symptoms of pre

eclampsia are present in 50% of women with AFLP including

hypertension, proteinuria, and edema [7] Some women present

with isolated obstetric complaints including contractions,

decreased fetal movement, and vaginal bleeding [1]

Systemic complications of AFLP are due to fulminant hepatic

failure and include encephalopathy, acute renal failure, infection,

pancreatitis, gastrointestinal hemorrhage, coagulopathy, and at

least mild hypoglycemia Neurological dysfunction begins early

and should immediately alert the physician to the possibility of

AFLP Symptoms may rapidly progress from restlessness,

confu-sion, and disorientation, to asterixis, seizures, psychosis, and

ulti-mately coma [1,3,5] Other systemic effects include respiratory

failure, sometimes requiring assisted ventilation [5] , ascites

[7] , and gastrointestinal bleeding from gastric ulceration and

Mallory – Weiss syndrome [2,7]

Renal insuffi ciency associated with AFLP is due to fatty infi

ltra-tion of the kidneys [1] Hepatorenal syndrome eventually

devel-3-ketoacyl-CoA thiolase

CoA

3-ketoacyl-CoA

Acetyl-CoA Acyl-CoA

2,3-enoyl-CoA R

3-hydroxyacyl-CoA

dehydrogenase

LCHAD deficiency block

Trifunctional protein defrciency block

Acyl-CoA dehydrogenase FAD

FADH2

3-hydroxyacyl-CoA

Enoyl-CoA hydratase

NADH + H+

NAD+ OH

C

O

S CoA C

CH2

H2O O

R CH2 CH3 C S CoA

O

R CH CH C S CoA

O

C O

S CoA

CH3

R C O

CoA S

R CH CH2 C S CoA

O

Acyl-CoA Figure 29.1 The biochemistry of mitochondrial trifunctional protein (MTP) defi ciencies

Mitochondrial fatty acid β - oxidation spiral where the MTP catalyzes long chain fatty acids substrates (see box) In isolated LCHAD defi ciency, the pathway is blocked after the enoyl Co - A hydratase reaction and before the 3 - hydroxyacyl Co - A dehydrogenase reaction, causing the accumulation of medium - and long - chain 3 - hydroxy fatty acids and their metabolites In complete MTP defi ciency, the pathway is blocked after the acyl Co - A dehydrogenase reaction and before the enoyl Co - A dehydrogenase reaction causing the accumulation of straight - chain fatty acids and their metabolites Adapted from Ibdah JA Acute fatty liver of pregnancy: an update on pathogenesis and clinical implications World J Gastroenterol 2006; 12(46):

7397 – 7404 [2]

Table 29.1 Signs and symptoms of acute fatty liver of pregnancy

Symptoms Nausea, vomiting Almost always Malaise Always Abdominal pain Almost always, may be variable in position

and severity

Physical signs Hypertension Almost always Edema Almost always Proteinuria Variable Jaundice Always Elevated liver transaminases Always Hypoglycemia Always, may be masked by administration of

glucose - containing intravenous fl uids Coagulopathy Common

Diabetes insipidus Common Encephalopathy Common, may correlate with ammonia levels

Trang 9

by the administration of dextrose solutions which often routinely

occurs at the time of admission

Virtually all women with AFLP have laboratory evidence of

coagulopathy and at least 50% require replacement of blood

components [1,2,5,30] Impaired hepatic synthesis of coagulation

factors leads to prolongation of prothrombin time (PT) and

acti-vated partial thromboplastin time (aPTT) Hypofi brinogenemia,

profound antithrombin III defi ciency, and thrombocytopenia are

common Factor VIII levels most accurately refl ect the extent of

coagulopathy and their return toward normal signals recovery

Coagulopathy may worsen in the postpartum period, most likely

secondary to low antithrombin III levels [31]

Serum transaminase concentrations are typically mildly

increased, usually between 100 and 1000 U/L Bilirubin levels are

variable but generally exceed 5 mg/dL Alkaline phosphatase is

elevated but is not helpful in making the diagnosis because of

placental production Serum albumin is usually low Ammonia

levels are elevated, due to decreased utilization by urea cycle liver

enzymes and may predict the degree of altered sensorium

Elevated amylase and lipase should raise suspicions of

concomi-tant pancreatitis [32] Liver function tests usually return to

normal 4 – 8 weeks after delivery [4]

The gold standard used for confi rmation of AFLP remains the

liver biopsy However, it is rarely necessary when other clinical

and laboratory parameters are consistent with the diagnosis

Microscopic examination of fresh specimens stained with special

fat stains, most commonly oil red, demonstrate hepatocellular

cytoplasm distended by numerous fi ne vacuoles giving the cells a

distinct foamy appearance (Figure 29.2 ) The myriad of tiny

vacuoles are separated from each other by thin eosinophilic

cyto-plasmic strands and do not coalesce to form a single large vacuole

In contrast to the cytoplasm, the cell nucleus is located centrally

and is normal in size and appearance

Histologic changes are most prominent in the central portion

of the lobule with a thin rim of normal hepatocytes at the

periph-ery The lobular architecture is usually preserved and, with rare

exceptions, necrosis and infl ammation are absent [33] This is

distinct from the periportal fi brin deposition and hemorrhagic

necrosis reported in pre - eclampsia (Figure 29.3 ) Characteristic

histologic changes may be present up to 3 weeks after the onset

of jaundice

Diagnosis

A high index of suspicion based on clinical presentation

corre-lated with correct interpretation of laboratory testing is usually

suffi cient to make the diagnosis of AFLP [1,7] Liver biopsy is

usually not necessary or even possible because of coagulopathy

Most common among the differential diagnoses are pre -

eclampsia/HELLP syndrome, viral hepatitis, and cholestasis

(Table 29.2 ) Women with AFLP or pre - eclampsia/HELLP may

have elevated serum transaminases, thrombocytopenia or

coagu-lation defects However, liver failure and jaundice are rare in

(a)

(b)

Figure 29.2 (a) Acute fatty liver of pregnancy (H & E stain; magnifi cation 200 × ) Note diffuse fatty infi ltration and absence of necrosis and infl ammation (b) Higher magnifi cation demonstrates the fi ne cytoplasmic vacuoles and centrally placed nuclei (H & E stain; magnifi cation 1000 × ) (Courtesy of Dr Patricia Latham, University of Maryland Hospital.)

Figure 29.3 Liver section from a patient who died of complications of

pre - eclampsia (H & E stain; magnifi cation 40 × ) Note extensive hepatocellular infl ammation and necrosis (Courtesy of Dr James Kelley, Madigan AMC.)

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lutely contraindicated AFLP should not be considered an indication for cesarean, even though expeditious delivery is recommended Indeed, most hemorrhagic complications in women with AFLP occur as a result of surgical trauma [1] Attempts at induction of labor and vaginal delivery are appropri-ate as long as adequappropri-ate mappropri-aternal supportive care and fetal surveil-lance are possible Even so, fetal compromise during labor is common and cesarean delivery is often necessary [1] Women who are critically ill should not be subjected to long arduous induction of labor The ultimate decision regarding route of delivery should be individualized, based on the maternal and fetal conditions as well as the favorability of the cervical exam Anesthetic options in patients with AFLP are limited General anesthesia can further damage an already compromised liver and regional anesthetic poses a risk of hemorrhage when coagulopa-thy is present If general anesthesia must be used, inhalation agents with potential hepatotoxicity (e.g halothane ) should be avoided Isofl urane is a logical choice since it has little or no hepatotoxicity and may preserve liver blood fl ow [36,37] Epidural anesthesia is probably the best option under most circumstances because it preserves hepatic blood fl ow without hepatotoxic effects [37,38] Recognition and treatment of thrombocytopenia and coagulopathy is essential prior to neuraxial techniques

Supportive c are

Supportive care of patients with AFLP should include careful monitoring for evidence of progressive hepatic failure, hypogly-cemia, and coagulopathy This should occur in an intensive care setting and in consultation with physicians well - versed in the care

of critically ill patients Prevention of worsening hypoglycemia and reduction of endogenous production of nitrogenous wastes can be accomplished by providing approximately 2000 – 2500 calories per day, primarily in the form of glucose Most patients require solutions containing more than 5% dextrose, sometimes

as high as 25%, administered intravenously or through a

naso-pre - eclampsia/HELLP Some authorities believe that AFLP and

pre - eclampsia may occur concomitantly [1] The diagnosis of

viral hepatitis can be established quickly and with reasonable

certainty via specifi c serologic testing In addition, serum

trans-aminase levels in women with hepatitis are usually elevated well

beyond those typically seen in AFLP Women with cholestasis of

pregnancy are usually not as ill - appearing as those with AFLP,

pre - eclampsia, or viral hepatitis While liver function tests are

abnormal in cholestasis of pregnancy, concentrations of bilirubin

and transaminase are usually much lower compared to those of

AFLP or viral hepatitis and signs and symptoms typical of pre

eclampsia are rarely present

Ultrasonography, CT, and MRI are often performed as part of

the diagnostic work - up for jaundice during pregnancy Ultrasound

demonstrates echogenicities within the liver of women with AFLP

[7] While non - specifi c, ultrasound may also identify subcapsular

hematoma, cholecystitis, and/or cholangitis Both CT and MRI

may suggest AFLP based on lower density that occurs with fatty

infi ltration of the liver [34,35] However, both have high false

negative rates that limit their usefulness [30] In clinical practice,

imaging studies are complementary but not necessary to make

the diagnosis of AFLP and their performance should not delay

appropriate treatment Moreover, a normal study does not

exclude AFLP

Treatment

Women suspected of having AFLP should be hospitalized in an

intensive care setting where comprehensive supportive care can

be given and preparations for delivery can be made All published

series have reported improved maternal and perinatal outcome

when prompt delivery is accomplished [1,3 – 5,7] Most women

begin to show clinical improvement and resolution of laboratory

abnormalities by the second day postpartum [5] There are no

reported cases of AFLP resolving prior to delivery; Therefore,

once the diagnosis is established, expectant management is

Table 29.2 Differential diagnosis of acute fatty liver of pregnancy

Acute fatty liver of

pregnancy

Acute hepatitis Cholestasis of pregnancy Severe pre - eclampsia

Clinical manifestations Nausea, vomiting, malaise,

encephalopathy, abdominal pain, coagulopathy

Malaise, nausea, vomiting, jaundice, anorexia, encephalopathy

Pruritus, jaundice Hypertension, edema, proteinuria,

oliguria, CNS hyperexcitability

Bilirubin Elevated Elevated Elevated Normal or minimally elevated Transaminases Minimally elevated Markedly elevated Minimally elevated Normal or minimal to moderate

increase Alkaline phosphatase Usually normal for pregnancy Minimally elevated Moderately elevated Normal for pregnancy

Histology Fatty infi ltration, no infl ammation

or necrosis

Marked infl ammation and necrosis Biliary stasis, no infl ammation Infl ammation, necrosis, fi brin

deposition

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