Abstract
Background: Accessory and cavitated uterine masses (ACUM) are rare Mullerian anomalies, defined as an isolated accessory cavitated mass lined with endometrial epithelium. Objectives: This article explores ACUM lesions from two aspects and integrates a case report of a successful laparoscopic-assisted intervention for ACUM in a 24-year-old woman with refractory dysmenorrhea with a review of current literature on laparoscopic surgical techniques. Unique to this case was the process of undergoing targeted genetic sequencing via the Find ITTM Panel Version 3.4 on the ACUM, looking for mutations in KRAS, PIK3CA, and FGFR2. A process that was inspired by recent reports that indicate that even normal endometrium can harbor cancer-associated mutations. Methods: A comprehensive search of Ovid MEDLINE and Embase was performed. Studies were selected if they explored laparoscopic surgery’s impact on ACUM patients’ outcomes related to fertility, menorrhagia, or dysmenorrhea. Risk of bias assessment was performed using the JBI Critical Appraisal Checklist. Outcome: From 160 articles identified, 25 full-text articles were analyzed with a total of 75 unique patients discussed. Dysmenorrhea was present in 100% of cases (n = 75/75), and laparoscopic resection improved patient symptoms in 84% (n = 63/75) of cases. The mass excised in the case was positive for somatic missense mutations in RET (R813W) and HRAS (G12S) genes, identified at low variant allele frequencies. Conclusions and Outlook: These results demonstrated that laparoscopic surgical approaches are effective and frequently the first surgical approach chosen for the treatment of ACUM, but that techniques to treat these conditions are not standardized. This case is the first to demonstrate mutations in ACUM, suggesting a potential role for cancer-associated somatic mutations in their genesis. Future developments in this area may include sending more of these samples for genetic analysis, improving our understanding of how these lesions are formed, while also working to standardize how they are removed.
Journal Section:
Narrative Review
References
1.
Acién
P
, Acién
M
, Fernández
F
, José Mayol
M
, Aranda
I
. The cavitated accessory uterine mass: a Müllerian anomaly in women with an otherwise normal uterus
. Obstet Gynecol
. 2010
;116
(5
):1101
–9
.2.
Acién
P
, Bataller
A
, Fernández
F
, Acién
MI
, Rodríguez
JM
, Mayol
MJ
. New cases of accessory and cavitated uterine masses (ACUM): a significant cause of severe dysmenorrhea and recurrent pelvic pain in young women
. Hum Reprod
. 2012
;27
(3
):683
–94
. 3.
Peyron
N
, Jacquemier
E
, Charlot
M
, Devouassoux
M
, Raudrant
D
, Golfier
F
, et al. Accessory cavitated uterine mass: MRI features and surgical correlations of a rare but under-recognised entity
. Eur Radiol
. 2019
;29
(3
):1144
–52
. 4.
Kyo
S
, Sato
S
, Nakayama
K
. Cancer-associated mutations in normal human endometrium: surprise or expected
. Cancer Sci
. 2020
;111
(10
):3458
–67
. 5.
Timmerman
S
, Stubbe
L
, Van den Bosch
T
, Van Schoubroeck
D
, Tellum
T
, Froyman
W
. Accessory cavitated uterine malformation (ACUM): a scoping review
. Acta Obstet Gynecol Scand
. 2024
;103
:1036
–45
. 6.
Protopapas
A
, Kypriotis
K
, Chatzipapas
I
, Kathopoulis
N
, Sotiropoulou
M
, Michala
L
. Juvenile cystic adenomyoma vs blind uterine horn: challenges in the diagnosis and surgical management
. J Pediatr Adolesc Gynecol
. 2020
;33
(6
):735
–8
. 7.
Paul
PG
, Chopade
G
, Das
T
, Dhivya
N
, Patil
S
, Thomas
M
. Accessory cavitated uterine mass: a rare cause of severe dysmenorrhea in young women
. J Minim Invasive Gynecol
. 2015
;22
(7
):1300
–3
. 8.
Lac
V
, Nazeran
TM
, Tessier-Cloutier
B
, Aguirre-Hernandez
R
, Albert
A
, Lum
A
, et al. Oncogenic mutations in histologically normal endometrium: the new normal
. J Pathol
. 2019
;249
(2
):173
–81
. 9.
10.
Shamseer
L
, Moher
D
, Clarke
M
, Ghersi
D
, Liberati
A
, Petticrew
M
, et al. Preferred reporting items for systematic review and meta-analysis protocols (PRISMA-P) 2015: elaboration and explanation
. BMJ Br Med J (Clin Res Ed)
. 2015
;349
(1
):g7647
. 11.
Akar
ME
, Leezer
KH
, Yalcinkaya
TM
. Robot-assisted laparoscopic management of a case with juvenile cystic adenomyoma
. Fertil Steril
. 2010
;94
(3
):e55
–7
. 12.
Bedaiwy
MA
, Henry
DN
, Elguero
S
, Pickett
S
, Greenfield
M
. Accessory and cavitated uterine mass with functional endometrium in an adolescent: diagnosis and laparoscopic excision technique
. J Pediatr Adolesc Gynecol
. 2013
;26
(4
):e89
–91
. 13.
Blank
C
, van Vliet
HAAM
, Schoot
BC
. A novel surgical laparoscopic approach of cystic uterine adenomyomas using a monopolar electrosurgical LEEP device. ESGE (European Society for Gynecological Endoscopy)
. Annual Congress
. 2016
.14.
Calagna
G
, Cucinella
G
, Tonni
G
, Gregorio
RD
, Triolo
O
, Martorana
A
, et al. Cystic adenomyosis spreading into subserosal-peduncolated myoma: How to explain it
. Int J Surg Case Rep
. 2015
;8
C:29
-31
. 15.
Gnade
CM
, Pickett
CM
, Oshinowo
AE
. Laparoscopic Management of a Juvenile Cystic Adenomyoma
. J Minim Invasive Gynecol
. 2022
;29
(11
suppl):S42
-3
. 16.
Takeuchi
H
, Kitade
M
, Kikuchi
I
, Kumakiri
J
, Kuroda
K
, Jinushi
M
. Diagnosis, laparoscopic management, and histopathologic findings of juvenile cystic adenomyoma: a review of nine cases
. Fertil Steril
. 2010
;94
(3
):862
–8
. 17.
Jain
N
, Goel
S
. Cystic Adenomyoma simulates uterine malformation: a diagnostic dilemma: case report of two unusual cases
. J Hum Reprod Sci
. 2012
;5
(3
):285
–8
. 18.
Kerbage
Y
, Dericquebourg
S
, Collinet
P
, Verpillat
P
, Giraudet
G
, Rubod
C
. Cystic adenomyoma surgery
. Hum Reprod
. 2022
;51
(3
):102313
. 19.
Kiyak
H
, Seckin
KD
, Karakis
L
, Karacan
T
, Ozyurek
ES
, Resit Asoglu
M
. Decidualized juvenile cystic adenomyoma mimicking a cornual pregnancy
. Fertil Steril
. 2020
;113
(2
):463
–5
. 20.
Kriplani
A
, Mahey
R
, Agarwal
N
, Bhatla
N
, Yadav
R
, Singh
MK
. Laparoscopic management of juvenile cystic adenomyoma: four cases
. J Minim Invasive Gynecol
. 2011
;18
(3
):343
–8
. 21.
Kriplani
I
, Kriplani
A
, Jain
N
, Lata
K
, Sodhi
M
. Laparoscopic excision of large ACUM masquerading as degenerated myoma: a case report
. Italian JOG
. 2023
;35
(1
). 22.
Kumakiri
J
, Kikuchi
I
, Sogawa
Y
, Jinushi
M
, Aoki
Y
, Kitade
M
, et al. Single-incision laparoscopic surgery using an articulating monopolar for juvenile cystic adenomyoma
. Minim Invasive Ther Allied Technol
. 2013
;22
(5
):312
–5
. 23.
Mollion
M
, Host
A
, Faller
E
, Garbin
O
, Ionescu
R
, Roy
C
. Report of two cases of Accessory Cavitated Uterine Mass (ACUM): Diagnostic challenge for MRI
. Radiol Case Rep
. 2021
;16
(11
):3465
–9
. 24.
Nabeshima
H
, Murakami
T
, Nishimoto
M
, Sugawara
N
, Sato
N
. Successful total laparoscopic cystic adenomyomectomy after unsuccessful open surgery using transtrocar ultrasonographic guiding
. J Minim Invasive Gynecol
. 2008
;15
(2
):227
–30
. 25.
Nabeshima
H
, Murakami
T
, Terada
Y
, Noda
T
, Yaegashi
N
, Okamura
K
. Total laparoscopic surgery of cystic adenomyoma under hydroultrasonographic monitoring
. J Am Assoc Gynecol Laparosc
. 2003
;10
(2
):195
–9
. 26.
Orlando
MS
, Carey-Love
A
, Attaran
M
, King
CR
. Surgical techniques for excision of juvenile cystic adenomyoma
. Fertil Steril
. 2022
;118
(4
):810
–1
. 27.
Peters
A
, Rindos
NB
, Guido
RS
, Donnellan
NM
. Uterine-sparing Laparoscopic Resection of Accessory Cavitated Uterine Masses
. J Minim Invasive Gynecol
. 2018
;25
(1
):24
–5
. 28.
Rekeda
L
, Zwayn
N
, Williamso
A
. Juvenile cystic adenomyoma managed surgically and medically: a case series
. J Pediatr Adolesc Gynecol
. 2022
;35
(2
):215
–6
. 29.
Said
MR
, Afaneh
H
, Zaghmout
O
, Moses
K
, Young
OJ
, Abuzeid
MI
. Minimally invasive management of juvenile cystic adenomyoma: report of three cases
. Facts Views Vis Obgyn
. 2021
;13
(3
):267
–72
. 30.
Strelec
M
, Banović
M
, Banović
V
, Sirovec
A
. Juvenile cystic adenomyoma mimicking a Mullerian uterine anomaly successfully treated by laparoscopic excision
. Int J Gynaecol Obstet
. 2019
;146
(2
):265
–6
. 31.
Takeda
A
, Sakai
K
, Mitsui
T
, Nakamura
H
. Laparoscopic management of juvenile cystic adenomyoma of the uterus: report of two cases and review of the literature
. J Minim Invasive Gynecol
. 2007
;14
(3
):370
–4
. 32.
Tokgoz
VY
, Tekin
AB
. A rare case of the new entity of müllerian anomalies mimicking the noncommunicating rudimentary cavity with hemi-uterus: accessory cavitated uterine mass
. Fertil Steril
. 2022
;117
(3
):646
–8
. 33.
Green
LJ
, Sokhi
T
, Balogun
M
, Ganesan
R
. Myometrial cysts: a case series
. Case Rep Womens Health
. 2019
;24
:e00155
. 34.
Grimbizis
GF
, Gordts
S
, Di Spiezio Sardo
A
, Brucker
S
, De Angelis
C
, Gergolet
M
, et al. The ESHRE/ESGE consensus on the classification of female genital tract congenital anomalies
. Hum Reprod
. 2013
;28
(8
):2032
–44
. 35.
Di Spiezio Sardo
A
, Campo
R
, Gordts
S
, Spinelli
M
, Cosimato
C
, Tanos
V
, et al. The comprehensiveness of the ESHRE/ESGE classification of female genital tract congenital anomalies: a systematic review of cases not classified by the AFS system
. Hum Reprod
. 2015
;30
(5
):1046
–58
. 36.
Landrum
MJ
, Lee
JM
, Benson
M
, Brown
GR
, Chao
C
, Chitipiralla
S
, et al. ClinVar: improving access to variant interpretations and supporting evidence
. Nucleic Acids Res
. 2018
;46
(D1
):D1062
–7
. 37.
Tate
JG
, Bamford
S
, Jubb
HC
, Sondka
Z
, Beare
DM
, Bindal
N
, et al. COSMIC: the Catalogue of somatic mutations in cancer
. Nucleic Acids Res
. 2019
;47
(D1
):D941
–7
. 38.
Roly
ZY
, Backhouse
B
, Cutting
A
, Tan
TY
, Sinclair
AH
, Ayers
KL
, et al. The cell biology and molecular genetics of Müllerian duct development
. Wiley Interdiscip Rev Dev Biol
. 2018
;7
(3
):e310
. 39.
Jain
S
. The many faces of RET dysfunction in kidney
. Organogenesis
. 2009
;5
(4
):177
–90
. 40.
Qian
W
, Zhang
M
, Huang
H
, Chen
Y
, Park
G
, Zeng
N
, et al. Costello syndrome with special cutaneous manifestations and HRAS G12D mutation: a case report and literature review
. Mol Genet Genomic Med
. 2021
;9
(6
):e1690
. 41.
Do
H
, Dobrovic
A
. Sequence artifacts in DNA from formalin-fixed tissues: causes and strategies for minimization
. Clin Chem
. 2015
;61
(1
):64
–71
. 42.
Anglesio
MS
, Papadopoulos
N
, Ayhan
A
, Nazeran
TM
, Noë
M
, Horlings
HM
, et al. Cancer-associated mutations in endometriosis without cancer
. N Engl J Med
. 2017
;376
(19
):1835
–48
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