Letters to the Editor—Brief Communication / European Journal of Obstetrics & Gynecology and Reproductive Biology 164 (2012) 234–238 237

Table 1fFN results and shortest CL between 22 and 24 weeks gestation with onset of labour and delivery gestation (CS, caesarean section).

Patient number fFN test 22–24 weeks Shortest CL (mm) 22–24 weeks Onset of labour Gestation at delivery

1 Negative 38.0 Spontaneous 35 + 0

2 Negative 44.0 Prelabour CS 33 + 6

3 Negative 47.0 Prelabour CS 35 + 6

4 Negative 33.0 Prelabour CS 36 + 2

5 Negative 36.0 Spontaneous 35 + 3

6 Negative 20.0 Prelabour CS 34 + 2

7 Negative 8.5 Prelabour CS 33 + 5

8 Negative 38.0 Prelabour CS 33 + 4

9 Negative 47.0 Prelabour CS 32 + 3

erend

al

s-

eo

al

*nn,

l,

9)

2

yaad,

nsl

length (CL) measurement in the prediction of delivery of tripletpregnancies.

A significant factor contributing to the rising incidence ofpreterm births is the increase in multiple births due to assistedconception [1]. The management of triplets involves admissionto hospital, timely administration of antenatal corticosteroidsand in utero transfer to optimise neonatal care. These interven-tions can improve outcome but are difficult to time appropri-ately and therefore resources are often misspent. CLmeasurement and fFN testing are increasing utilised to predictpreterm delivery.

The use of CL measurement in triplet pregnancies is wellreported. One study reported that a CL � 25 mm at 15–20 weeks’gestation has a positive predictive value (PPV) of 100% forspontaneous delivery at <28 weeks gestation [2].

For asymptomatic, high risk women tested at 22–30 weeks’gestation, a positive fFN confers a 20% and 39% risk of delivery anda negative fFN confers a 0% and 4% risk of delivery before 30 and 34weeks’, respectively [3]. This study showed that a positive fFN withCL > 25 mm had more than double the risk of delivery at 37 weeksand six times the risk of delivery at 34 weeks compared to aCL � 25 mm and negative fFN [3]. There are, however, no reports ofoutcome following a negative fFN test with a short CL in tripletpregnancies.

In the present series (Table 1), patients 1–9 had a negative fFNat initial testing and all delivered after 30 weeks’ gestation,irrespective of CL, including those with a spontaneous onset oflabour. Patient 10 had a positive fFN and had a spontaneous labouronset before 30 weeks, confirming the excellent predictive value offFN in asymptomatic pregnancies and suggesting the reliability ofits use in triplet pregnancies. Further fFN tests in patient 11 after 24weeks were negative and the delivery at 33 + 1 weeks suggests thatthose patients who test negative after an initial positive fFN thengo on to have a lower risk of early delivery.

Patient 7 had a CL of 8.5 mm and negative fFN but did not have aspontaneous onset of labour before 30 weeks’ gestation, comparedto patient 10 with a CL of 23.0 mm and positive fFN, whospontaneously delivered at 28 + 3 weeks, which suggests thesuperiority of fFN testing to predict early delivery.

The CL of 8.5 mm in patient 7 alarmed her clinicians and despitethe negative fFN she was admitted and given corticosteroids at24 + 1 weeks, due to the absence of literature supporting fFNprediction in triplets. However, due to consistently negative fFNshe was considered safe for discharge at 28 + 0 weeks. Patient 10had a CL of 23 mm and a positive fFN. By 25 + 2 weeks her CL hadshortened to 7 mm and she delivered at 28 + 3 weeks. The positivefFN demonstrated the high risk of very early delivery andmanagement was administered at an appropriate time.

10 Positive 23.0

11 Positive 25.0

In conclusion, this case series indicates that fFN testing could bean extremely useful tool in the management of triplet pregnanciesand is likely a stronger predictor than CL. We suggest that fortnightly

fFN testing after 22 weeks’ gestation could be an integral part of thmanagement of triplet pregnancies. It may be possible fotransvaginal ultrasound, which many women find intrusive, to breserved for positive fFN patients only. The use of fFN testing itriplet pregnancies could lead to fewer unnecessary admissions anobstetric interventions and a reduction in the associated costs.

References

[1] Shennan A. Why should preterm births be rising. British Medical Journ2006;332:924–5.

[2] Guzman ER. Use of cervical ultrasonography in prediction of spontaneoupreterm birth in triplet gestations. American Journal of Obstetrics and Gynaecology 2000;183:1108–13.

[3] Bolt LA, Chandiramani M, De Greeff A, Seed P, Kurtzman J, Shennan AH. Thvalue of combined cervical length measurement and fetal fibronectin testing tpredict spontaneous preterm birth in asymptomatic high-risk women. Journof Maternal-Fetal and Neonatal Medicine 2011;24:928–32.

Lauren A. BoltKiki Morriso

Andrew H. ShennaMaternal and Fetal Research Unit, King’s College London

Division of Reproduction and Endocrinology, St Thomas’ Hospita

London SE1 7EH, United Kingdom

*Corresponding author. Tel.: +44 020 7188 363E-mail address: lauren.bolt@gmail.com (L.A. Bolt

21 May 201

http://dx.doi.org/10.1016/j.ejogrb.2012.06.012

Pregnancy in a patient with anterior sacral meningocele andbicornuate uterus

Dear Editor,

We report a case of a 32 year-old woman, gravida 2, affected banterior sacral meningocele (ASM) and bicornuate uterus. ASM is

congenital lesion containing cerebrospinal fluid that results from

failed closure and abnormal differentiation of the neural tube ancan lead to obstructive labour. Possible complications are rupturemeningitis, abscess and progressive nerve root damage that calead to death [1,2]. ASM usually has sporadic presentation and it ioften associated with urogenital and rectal defects [1]. Sacra

Spontaneous 28 + 3

Prelabour CS 33 + 1

defect with anterior meningocele (SDAM) is an autosomaldominant disorder, depending on mutations in the VANGL1 gene.Currarino syndrome is a triad composed by partial sacral agenesis,

prtioas

dunesethcoGeafm

coboreThledu

codeevsuanof(Fdyhirelamab

dege

veprscnech

dehoantiowafrepr

wboAchembycaar

Re

[1]

[2]

[3]

[4]

Fig35

co

Letters to the Editor—Brief Communication / European Journal of Obstetrics & Gynecology and Reproductive Biology 164 (2012) 234–238238

esacral mass and anorectal malformations. Genital malforma-ns, such as bicornuate uterus and septate vagina, are sometimes

sociated [3].In this case ASM was diagnosed when she was 20 years old,ring a laparotomic procedure for a 7 cm pelvic mass. The patientver reported symptoms. When she presented at our ambulatoryrvice for preconception counselling, we suggested, according toe literature [3], administration of folic acid 5 mg daily beforenception in order to avoid foetal neural tube malformations.netic counselling revealed that the patient’s mother was also

fected by ASM, but all genetic investigations for knownutations were negative.

The pregnancy started in the right horn and had a physiologicalurse. At 12 weeks, nuchal translucency was 1.2 mm with nosene detection. We performed close ultrasound scans that nevervealed the presence of ASM or other malformations in the foetus.e mother’s ASM appeared as well-defined, unilocular, fluid-filled

sion in the retrorectal space, with no volumetric US changes

. 1. MRI SE T2W image on the sagittal plane shows the presacral mass at

weeks’ pregnancy. The presence of a stalk between the sacral canal and the mass

nfirms the meningeal origin of the lesion.

ring the whole of the pregnancy.At 35 weeks of pregnancy, the patient presented with

mplaints of back pain. We excluded uterine contractions andcided for MRI scan, which is the gold standard procedure foraluation of ASM. MRI examination was carried out using aperconductive 0.5T scanner (SIGNA GE, Milwaukee, WI, USA)d the integrated body coil. The imaging protocol used consisted

SE and T2W sequences acquired on sagittal and axial planesig. 1). MR images confirmed the presence of anterior sacralsraphism, better showing the presence of a pelvic mass, withgh signal intensity on the T2W sequences, extending to thetroperitoneal presacral space, with a prevalent left-side antero-teral extension. Moreover, it allowed us to assess the absence ofacroscopically evident nervous structures passing through thisnormal communication.Foetal weight estimation by ultrasound was 2750 g, so we

cided on caesarean section which was performed underneral anesthesia, with a Pfannenstiel skin incision and a ht

rtical uterine incision in the right horn at 36 weeks ofegnancy. The female newborn weighting 2700 g had Apgarores of 9 and 10 at 1 and 5 min, respectively. Both mother andwborn had no postoperative complications and were dis-arged four days after surgery.Sixteen months after delivery, the patient presented to our

partment carrying a spontaneous pregnancy in the rightrn. We adopted the same management as already describedd at 36 weeks of pregnancy performed, without complica-ns, a caesarean section delivering a healthy male baby

eighting 2800 g. Although the patient’s mother wasfected by ASM, both children, at 3 and 5 years follow up,spectively, show no symptoms of ASM, confirming theenatal diagnoses.In the literature are reported about 18 cases of AMS associated

ith pregnancy. Only one case of pregnancy in a patient affected byth ASM and bicornuate uterus has previously been reported [4].cording to the literature [2], caesarean section is the best for thealth of women affected by ASM because prevents possibleeningocele rupture during labour. Pregnancy in patients affected

anterior sacral meningocele must be carefully followed andesarean section must be performed as soon as compression signse identified.

ferences

Gardner PA, Albright AL. ‘‘Like mother, like son’’: hereditary anterior sacralmeningocele. Journal of Neurosurgery 2006;104:138–42.

Manson F, Comalli-Dillon K, Moriaux A. Anterior sacral meningocele: manage-ment in gynecological practice. Ultrasound in Obstetrics and Gynecology2007;30:893–6.

Chen CP. Syndromes disorders and maternal risk factors associated with neuraltube defects (II). Taiwanese Journal of Obstetrics and Gynecology 2008;47:283–90.

Kontopoulos EV, Oyelese Y, Nath C, Schwebel M, Smulian JC, Vintzileos AM.Maternal anterior sacral meningocele in pregnancy. Journal of Maternal-Fetaland Neonatal Medicine 2005;17:423–5.

Daniela SuricoRoberta Amadori*

Department of Obstetrics and Gynaecology,

University of Eastern Piedmont, Novara, Italy

Roberta AmbrosiniDepartment of Radiology, University of Eastern Piedmont,

Novara, Italy

Alessandro VigoneNicola Surico

Department of Obstetrics and Gynaecology,

University of Eastern Piedmont, Novara, Italy

*Corresponding author at: Department of Obstetricsand Gynaecology, ‘‘Maggiore della Carita’’ Hospital,

Viale Mazzini 18, 28100 Novara, Italy.Tel.: +39 0321 3733665/39 3381993125;

fax: +39 0321 3733659E-mail address: ierama@tin.it (R. Amadori)

14 February 2012

tp://dx.doi.org/10.1016/j.ejogrb.2012.06.028