Introduction: Early repair in patients affected by myelomeningocele (MMC) is of paramount importance in order to prevent infection, minimize neural tissue damage, and reduce mortality. Treatment must include duraplasty and possibly an adequate soft tissue coverage. Delayed surgery in MMC patients can be more tedious due to the less clear borders between the placode and the skin. Moreover, the risks of wound infection and breakdown increase significantly. Case Presentation: We present the unusual case of a large MMC in a 3-year-old patient treated by combining the recently described cryopreserved amniotic membrane (AM) as homograft for dural reconstruction and a bilateral Keystone flap for soft tissue reconstruction. Discussion: Thanks to its anti-inflammatory and elastic proprieties, the AM can play an important role in preventing adhesion between the reconstructed layers, thus reducing the risk of spinal cord tethering. The Keystone flap, at the same time, allows the wound tension to be distributed widely over the flap margins and not only along the midline, which overlies the duraplasty, enhancing the scar quality and lowering the risk of cerebrospinal fluid recurrence and wound dehiscence, with no donor site morbidity.

1.
Greenberg
F
,
James
LM
,
Oakley
GP
 Jr
.
Estimates of birth prevalence rates of spina bifida in the United States from computer-generated maps
.
Am J Obstet Gynecol
.
1983
Mar
;
145
(
5
):
570
3
.
[PubMed]
0002-9378
2.
Katzen
M
.
The total care of spina bifida cystica
.
Surg Annu
.
1981
;
13
:
325
39
.
[PubMed]
0081-9638
3.
Raghavan
A
,
Wright
JM
,
Huang Wright
C
,
Sajatovic
M
,
Miller
J
.
Effect of Dural Substitute and Technique on Cranioplasty Operative Metrics: A Systematic Literature Review
.
World Neurosurg
.
2018
Nov
;
119
:
282
9
.
[PubMed]
1878-8750
4.
Saad
H
,
Krisht
KM
,
Yang
WH
,
Lopez-Gonzalez
MA
,
Aboud
E
,
Krisht
AF
.
Biocompatible Amniotic Sac Implant Maintains a Scar-Free Brain Surface During Recurrent Glioma Surgery
.
World Neurosurg
.
2017
Nov
;
107
:
308
13
.
[PubMed]
1878-8750
5.
Hasegawa
M
,
Fujisawa
H
,
Hayashi
Y
,
Yamashita
J
.
Autologous amnion graft for repair of myelomeningocele: technical note and clinical implication
.
J Clin Neurosci
.
2004
May
;
11
(
4
):
408
11
.
[PubMed]
0967-5868
6.
Marton
E
,
Giordan
E
,
Gioffrè
G
,
Canova
G
,
Paolin
A
,
Mazzucco
MG
, et al
Homologous cryopreserved amniotic membrane in the repair of myelomeningocele: preliminary experience
.
Acta Neurochir (Wien)
.
2018
Aug
;
160
(
8
):
1625
31
.
[PubMed]
0001-6268
7.
Patterson
TJ
,
Till
K
.
The use of rotation flaps following excision of lumbar myelo-meningoceles: an aid to the closure of large defects
.
Br J Surg
.
1959
May
;
46
(
200
):
606
8
.
[PubMed]
0007-1323
8.
Behan
FC
.
The Keystone Design Perforator Island Flap in reconstructive surgery
.
ANZ J Surg
.
2003
Mar
;
73
(
3
):
112
20
.
[PubMed]
1445-1433
9.
Attenello
FJ
,
Tuchman
A
,
Christian
EA
,
Wen
T
,
Chang
KE
,
Nallapa
S
, et al
Infection rate correlated with time to repair of open neural tube defects (myelomeningoceles): an institutional and national study
.
Childs Nerv Syst
.
2016
Sep
;
32
(
9
):
1675
81
.
[PubMed]
0256-7040
10.
Mehta
VA
,
Bettegowda
C
,
Ahmadi
SA
,
Berenberg
P
,
Thomale
UW
,
Haberl
EJ
, et al
Spinal cord tethering following myelomeningocele repair
.
J Neurosurg Pediatr
.
2010
Nov
;
6
(
5
):
498
505
.
[PubMed]
1933-0707
11.
Talamonti
G
,
D’Aliberti
G
,
Collice
M
.
Myelomeningocele: long-term neurosurgical treatment and follow-up in 202 patients
.
J Neurosurg
.
2007
Nov
;
107
(
5
Suppl
):
368
86
.
[PubMed]
0022-3085
12.
Inoue
HK
,
Kobayashi
S
,
Ohbayashi
K
,
Kohga
H
,
Nakamura
M
.
Treatment and prevention of tethered and retethered spinal cord using a Gore-Tex surgical membrane
.
J Neurosurg
.
1994
Apr
;
80
(
4
):
689
93
.
[PubMed]
0022-3085
13.
Sakamoto
H
,
Hakuba
A
,
Fujitani
K
,
Nishimura
S
.
Surgical treatment of the retethered spinal cord after repair of lipomyelomeningocele
.
J Neurosurg
.
1991
May
;
74
(
5
):
709
14
.
[PubMed]
0022-3085
14.
Tubbs
RS
,
Oakes
WJ
.
A simple method to deter retethering in patients with spinal dysraphism
.
Childs Nerv Syst
.
2006
Jul
;
22
(
7
):
715
6
.
[PubMed]
0256-7040
15.
Hudgins
RJ
,
Gilreath
CL
.
Tethered spinal cord following repair of myelomeningocele
.
Neurosurg Focus
.
2004
Feb
;
16
(
2
):
E7
.
[PubMed]
1092-0684
16.
Zide
B
,
Constantini
S
,
Epstein
FJ
.
Prevention of recurrent tethered spinal cord
.
Pediatr Neurosurg
.
1995
;
22
(
2
):
111
4
.
[PubMed]
1016-2291
17.
Brown
EG
,
Saadai
P
,
Pivetti
CD
,
Beattie
MS
,
Bresnahan
JC
,
Wang
A
, et al
In utero repair of myelomeningocele with autologous amniotic membrane in the fetal lamb model
.
J Pediatr Surg
.
2014
Jan
;
49
(
1
):
133
7
.
[PubMed]
0022-3468
18.
Gutman
MJ
,
Goldschlager
T
,
Fahardieh
RD
,
Ying
D
,
Xenos
C
,
Danks
RA
.
Keystone design perforator island flap for closure of myelomeningocele
.
Childs Nerv Syst
.
2011
Sep
;
27
(
9
):
1459
63
.
[PubMed]
0256-7040
19.
de Weerd
L
,
Weum
S
,
Sjåvik
K
,
Acharya
G
,
Hennig
RO
.
A new approach in the repair of a myelomeningocele using amnion and a sensate perforator flap
.
J Plast Reconstr Aesthet Surg
.
2013
Jun
;
66
(
6
):
860
3
.
[PubMed]
1748-6815
20.
Donaldson
C
,
Murday
HK
,
Gutman
MJ
,
Maher
R
,
Goldschlager
T
,
Xenos
C
, et al
Long-term follow-up for keystone design perforator island flap for closure of myelomeningocele
.
Childs Nerv Syst
.
2018
Apr
;
34
(
4
):
733
6
.
[PubMed]
0256-7040
21.
Zakaria
Y
,
Hasan
EA
.
Reversed turnover latissimus dorsi muscle flap for closure of large myelomeningocele defects
.
J Plast Reconstr Aesthet Surg
.
2010
Sep
;
63
(
9
):
1513
8
.
[PubMed]
1748-6815
22.
Pelissier
P
,
Santoul
M
,
Pinsolle
V
,
Casoli
V
,
Behan
F
.
The keystone design perforator island flap. Part I: anatomic study
.
J Plast Reconstr Aesthet Surg
.
2007
;
60
(
8
):
883
7
.
[PubMed]
1748-6815
23.
Lanni
MA
,
Van Kouwenberg
E
,
Yan
A
,
Rezak
KM
,
Patel
A
.
Applying the Keystone Design Perforator Island Flap Concept in a Variety of Anatomic Locations: A Review of 60 Consecutive Cases by a Single Surgeon
.
Ann Plast Surg
.
2017
Jul
;
79
(
1
):
60
7
.
[PubMed]
0148-7043
24.
Park
HS
,
Morrison
E
,
Lo
C
,
Leong
J
.
An Application of Keystone Perforator Island Flap for Closure of Lumbosacral Myelomeningocele Defects
.
Ann Plast Surg
.
2016
Sep
;
77
(
3
):
332
6
.
[PubMed]
0148-7043
25.
Jamjoom H, Alnoman H, Almadani Y. Closure of a Large Thoracolumbar Myelomeningocele Using a Modified Bilateral Keystone Flap. Plast Reconstr Surg Glob Open. 2016 Dec 13;4(12):e1114.
26.
Lien
SC
,
Maher
CO
,
Garton
HJ
,
Kasten
SJ
,
Muraszko
KM
,
Buchman
SR
.
Local and regional flap closure in myelomeningocele repair: a 15-year review
.
Childs Nerv Syst
.
2010
Aug
;
26
(
8
):
1091
5
.
[PubMed]
0256-7040
27.
Mustardé
JC
.
Reconstruction of the spinal canal in severe spina bifida
.
Plast Reconstr Surg
.
1968
Aug
;
42
(
2
):
109
14
.
[PubMed]
0032-1052
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