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Brief report
Ovarian fibromatosis: “The black garland sign”
Fibromatosis ovárica: “signo de la guirnalda negra”
M. Santos Urios
Corresponding author
msantosurios@gmail.com

Corresponding author.
, C. García Espasa, L. Concepción Aramendía
Servicio de Radiodiagnóstico, Hospital General Universitario de Alicante, Alicante, Spain
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The only laboratory finding of note was slight elevation of the CA-125 marker &#40;46<span class="elsevierStyleHsp" style=""></span>U&#47;mL&#59; normal &#60;35<span class="elsevierStyleHsp" style=""></span>U&#47;mL&#41;&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">The patient&#8217;s physical examination included a pelvic examination in which a firm&#44; immobile mass at the base of the Pouch of Douglas was palpated&#46; An emergency transvaginal ultrasound detected two heterogeneous solid lesions at the base of the Pouch of Douglas &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; With this discovery&#44; a decision was made to contact the diagnostic imaging department to complete the study&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">An abdominal and pelvic computed tomography &#40;CT&#41; scan with intravenous contrast &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>A&#41; confirmed the presence of two well-delimited parauterine lesions&#44; with slight thick peripheral enhancement and central hypouptake&#46; The study was completed with pelvic magnetic resonance imaging &#40;MRI&#41; &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>B&#8211;E&#41;&#44; which identified two polylobulated lesions measuring approximately 6<span class="elsevierStyleHsp" style=""></span>cm on the left side and 5<span class="elsevierStyleHsp" style=""></span>cm on the right side&#46; Both images corresponded to enlarged ovaries with a thickened&#44; markedly hypointense cortex on T2-weighted sequences in relation to the presence of peripheral fibrous tissue&#44; known as the &#8220;black garland sign&#8221;&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> The central ovarian stroma was intact&#44; with at least one identifiable follicle&#46; Following intravenous contrast administration&#44; slight enhancement of the peripheral cortical region was seen with hypointensity in the central region&#46; On diffusion-weighted sequences with a high <span class="elsevierStyleItalic">b</span> value&#44; both adnexal lesions showed a low peripheral signal and a central area of high signal intensity&#46; The findings reported were highly suggestive of ovarian fibromatosis&#46; None of the imaging tests showed associated findings raising suspicion of malignancy &#40;lymphadenopathy&#44; peritoneal carcinomatosis&#44; etc&#46;&#41;&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">As the patient suffered from prolonged amenorrhoea and was experiencing discomfort&#44; in accordance with her wishes&#44; surgical removal was chosen&#46; The operation consisted of a bilateral salpingo-oophorectomy&#44; yielding an intraoperative finding of bilateral pearly&#44; very firm ovarian masses&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">The macroscopic anatomopathological analysis &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>A&#41; of the surgical specimens showed ovaries with a peripheral ridge of whitish tissue exhibiting an elastic consistency&#44; consistent with fibrotic tissue&#44; and a yellowish centre with vessels inside in relation to preserved ovarian stroma&#46; The microscopic analysis &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>B&#41; showed replacement of the peripheral ovarian parenchyma featuring fusocellular proliferation with areas of collagen and a central stromal oedema&#46; All this confirmed the diagnosis of bilateral ovarian fibromatosis&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0035" class="elsevierStylePara elsevierViewall">Ovarian fibromatosis is a very uncommon benign condition&#44; first described by Scully and Young<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> in 1984&#44; which usually affects young women&#44; with a mean age of 25 years&#46; It presents with menstrual abnormalities as well as abdominal pain in most cases&#44; although it may also be asymptomatic&#46; Its involvement is predominantly unilateral&#44; although there are some examples of bilateral involvement&#44; as in our case&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Histologically&#44; ovarian fibromatosis is characterised by proliferation of collagen-producing fusiform cells around normal ovarian structures&#46; Although its pathophysiology is not entirely clear&#44; it seems that the lesion could be secondary to partial or intermittent torsion leading to venous and lymphatic obstruction&#46; The disease bears resemblances to massive ovarian oedema&#44; since both involve enlarged ovaries with preservation of internal ovarian structures&#46; However&#44; they differ in terms of the nature of the abnormal tissue&#44; which is fibrotic in fibromatosis and oedematous in massive ovarian oedema&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> However&#44; as seen in the microscopic analysis in our case&#44; it is possible to detect foci of stromal oedema in ovaries with fibromatosis&#46; In fact&#44; there are reports in the scientific literature of foci of stromal oedema similar to foci of massive ovarian oedema in up to 50&#37; of cases of ovarian fibromatosis&#46; This would point to a possible link between the two conditions&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">In 2003&#44; Bazot et al&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> reported the case of homogeneous&#44; low-signal bilateral ovarian masses on T2-weighted sequences corresponding to fibrous infiltration of the ovary&#46; They stressed that partial preservation of normal ovarian structures distinguishes this from other ovarian masses&#46; However&#44; this characteristic image of markedly hypointense fibrous tissue on T2-weighted sequences around the residual ovary was finally described by Takeuchi et al&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> as the &#8220;black garland sign&#8221; &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>B&#41;&#46; Although it is not always present&#44; the &#8220;black garland sign&#8221; is very specific to ovarian fibromatosis and was subsequently reported by other authors&#46;<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6&#44;7</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">The differential diagnosis based on its hypointensity on T1 and T2 sequences should be made with other entities that also show it&#44; such as fibromas&#44; fibrothecomas&#44; Brenner tumours and Krukenberg tumours&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> However&#44; imaging of preserved central ovarian structures surrounded by fibrous tissue is not observed in these lesions and may reflect the specific characteristics of ovarian fibromatosis&#46; In addition&#44; Krukenberg tumours show strong contrast uptake on both CT and MRI imaging&#44;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> whereas reported cases of ovarian fibromatosis show weak uptake on post-contrast T1-weighted sequences&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Aggressive pelvic fibromatosis secondarily affecting the ovaries is another differential diagnosis that must be considered&#44; but it is distinguished by extraovarian involvement which is not typical in ovarian fibromatosis&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> In addition&#44; an association with other diseases such as abdominal fibromatosis&#44; sclerosing peritonitis and Meigs syndrome has been reported&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">In conclusion&#44; ovarian fibromatosis is a disease with a low prevalence&#44; which may interfere with radiological interpretation and cause it to be initially omitted from the differential diagnoses of solid ovarian masses&#46; Visualisation of the &#8220;black garland sign&#8221; on MRI&#44; given its specificity&#44; should lead to it being considered as a possible diagnosis&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Authorship</span><p id="par0115" class="elsevierStylePara elsevierViewall"><ul class="elsevierStyleList" id="lis0005"><li class="elsevierStyleListItem" id="lsti0005"><span class="elsevierStyleLabel">1</span><p id="par0060" class="elsevierStylePara elsevierViewall">Responsible for study integrity&#58;</p></li><li class="elsevierStyleListItem" id="lsti0010"><span class="elsevierStyleLabel">2</span><p id="par0065" class="elsevierStylePara elsevierViewall">Study concept&#58; MSU&#44; CGE&#44; LCA&#46;</p></li><li class="elsevierStyleListItem" id="lsti0015"><span class="elsevierStyleLabel">3</span><p id="par0070" class="elsevierStylePara elsevierViewall">Study design&#58; MSU&#44; CGE&#44; LCA&#46;</p></li><li class="elsevierStyleListItem" id="lsti0020"><span class="elsevierStyleLabel">4</span><p id="par0075" class="elsevierStylePara elsevierViewall">Data collection&#58; MSU&#44; CGE&#44; LCA&#46;</p></li><li class="elsevierStyleListItem" id="lsti0025"><span class="elsevierStyleLabel">5</span><p id="par0080" class="elsevierStylePara elsevierViewall">Data analysis and interpretation&#58; CGE&#44; LCA&#46;</p></li><li class="elsevierStyleListItem" id="lsti0030"><span class="elsevierStyleLabel">6</span><p id="par0085" class="elsevierStylePara elsevierViewall">Statistical processing&#58; N&#47;A&#46;</p></li><li class="elsevierStyleListItem" id="lsti0035"><span class="elsevierStyleLabel">7</span><p id="par0090" class="elsevierStylePara elsevierViewall">Literature search&#58; N&#47;A&#46;</p></li><li class="elsevierStyleListItem" id="lsti0040"><span class="elsevierStyleLabel">8</span><p id="par0095" class="elsevierStylePara elsevierViewall">Drafting of the article&#58; MSU&#46;</p></li><li class="elsevierStyleListItem" id="lsti0045"><span class="elsevierStyleLabel">9</span><p id="par0100" class="elsevierStylePara elsevierViewall">Critical review of the manuscript with intellectually significant contributions&#58; CGE&#44; LCA&#46;</p></li><li class="elsevierStyleListItem" id="lsti0050"><span class="elsevierStyleLabel">10</span><p id="par0105" class="elsevierStylePara elsevierViewall">Approval of the final version&#58; MSU&#44; CGE&#44; LCA&#46;</p></li></ul></p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Conflicts of interest</span><p id="par0110" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest&#46;</p></span></span>"
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          "identificador" => "xres1701796"
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          "titulo" => "Introduction"
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        "titulo" => "Abstract"
        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">The main objective in the imaging differential diagnosis of an ovarian mass is to establish whether it is cystic or solid&#59; solid lesions are less common&#46; Ovarian fibromatosis is a benign disease of the ovary that is rarely included in the differential diagnosis of solid ovarian lesions&#46; Characteristic features of masses that have a fibrous component are low signal in T1-weighted MRI sequences and especially in T2-weighted MRI sequences&#46; The presence of peripheral fibrotic tissue around the residual ovarian tissue is specific to ovarian fibromatosis&#59; on MRI&#44; this results in marked hypointensity on T2-weighted images that has been dubbed the &#8220;black garland sign&#8221;&#46; This sign&#44; together with slight peripheral enhancement after the administration of contrast material and the preservation of the ovarian architecture&#44; facilitates the diagnosis&#44; making it possible to avoid unnecessary surgical interventions&#46;</p></span>"
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        "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">El primer objetivo del diagn&#243;stico diferencial por imagen ante una masa ov&#225;rica es establecer su naturaleza qu&#237;stica o&#44; menos frecuente&#44; s&#243;lida&#46; La fibromatosis ov&#225;rica es una patolog&#237;a benigna del ovario rara&#44; incluida en el diagn&#243;stico diferencial ante el hallazgo de lesiones ov&#225;ricas s&#243;lidas&#46; Caracter&#237;sticas propias de las masas que presentan componente fibr&#243;tico son su baja se&#241;al en secuencias de resonancia magn&#233;tica potenciadas en T1 y especialmente en T2&#46; La presencia de tejido fibr&#243;tico perif&#233;rico alrededor del tejido ov&#225;rico residual es una caracter&#237;stica espec&#237;fica en la fibromatosis ov&#225;rica y que tiene como traducci&#243;n en la RM una marcada hipointensidad en T2 conocida como &#8220;signo de la guirnalda negra&#8221;&#46; Este signo&#44; junto con una d&#233;bil captaci&#243;n poscontraste perif&#233;rica&#44; as&#237; como la preservaci&#243;n de la arquitectura ov&#225;rica&#44; nos facilita el diagn&#243;stico y puede evitar cirug&#237;as innecesarias&#46;</p></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Santos Urios M&#44; Garc&#237;a Espasa C&#44; Concepci&#243;n Aramend&#237;a L&#46; Fibromatosis ov&#225;rica&#58; &#8220;signo de la guirnalda negra&#8221;&#46; Radiolog&#237;a&#46; 2022&#59;64&#58;164&#8211;168&#46;</p>"
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Image from transvaginal ultrasound showing one of the two solid masses &#40;callipers&#41; dependent on the right ovary&#46; The lesion is heterogeneous and has a lobulated contour&#44; with an acoustic shadow&#46;</p>"
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          "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A&#41; Axial image from abdominal and pelvic computed tomography with intravenous contrast identifying two parauterine masses with a hypodense centre and a peripheral margin with a density similar to muscle &#40;hollow arrows&#41;&#46; They are associated with scant free fluid at the base of the Pouch of Douglas &#40;white arrow&#41;&#46; B&#41; Coronal T2-weighted magnetic resonance imaging &#40;MRI&#41; of the pelvis showing that both ovaries are enlarged&#44; with follicles in the central region &#40;white arrows&#41;&#46; Thickened&#44; well-delimited and markedly hypointense ridges consistent with the &#8220;black garland sign&#8221; &#40;asterisks&#41; can be observed&#46; C&#41; Axial T1-weighted MRI of the pelvis with no intravenous contrast identifying enlarged ovaries with a slightly lesser intensity on their periphery &#40;arrow tips&#41; compared to the central stroma &#40;white arrows&#41;&#46; D&#41; Axial T1-weighted MRI with fat suppression acquired following administration of intravenous contrast showing weak peripheral uptake by the fibrous part of both ovaries &#40;arrow tips&#41; with hypointensity of the central part &#40;hollow arrow&#41;&#46; Scant free fluid is identified at the base of the Pouch of Douglas &#40;white arrow&#41;&#46; E&#41; Axial diffusion-weighted MRI &#40;b 800&#41;&#46; Both adnexal lesions show a low peripheral signal &#40;arrow tips&#41;&#44; with central hyperintensity corresponding to areas of stromal oedema &#40;white arrow&#41;&#44; subsequently confirmed in the pathology analysis&#46;</p>"
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          "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">A&#41; Macroscopic specimens from both ovaries following surgery&#46; The right ovary &#40;left side of image&#41; and the left ovary &#40;right side of image&#41;&#44; previously fragmented&#44; enabling visualisation of a peripheral ridge of homogeneous whitish tissue exhibiting an elastic consistency&#44; consistent with fibrotic tissue &#40;asterisks&#41;&#46; The centre is composed of yellowish stroma with vessels &#40;arrow&#41;&#46; The macroscopic findings are consistent with bilateral ovarian fibromatosis&#46; B&#41; Haematoxylin-eosin staining at 40x magnification of the ovary showing replacement of more than 90&#37; of the parenchyma with fusocellular proliferation &#40;black arrow&#41; forming intertwined bundles with variable areas of collagen and stromal oedema &#40;hollow arrow&#41;&#46;</p>"
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