Unveiling the Uncommon: Fallopian Tube Cavernous Hemangioma in pregnancy with endometriosis background – a case report and literature review

In: Research Square · 2024 · doi:10.21203/rs.3.rs-4521730/v1 · W4400344992
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This case report details a rare fallopian tube cavernous hemangioma discovered during a cesarean section in a pregnant patient with a history of endometriosis.

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This paper reports a pre-peer-reviewed case of a 39-year-old pregnant woman in whom a rare fallopian tube cavernous hemangioma was discovered incidentally during an emergency caesarean section after spontaneous rupture of membranes, despite no radiologic findings during routine pregnancy scans. Histology showed an encapsulated lesion composed of variably sized, dilated thin-walled vessels lined by flattened endothelial cells with focal fibrin thrombi, consistent with cavernous hemangioma; the authors note a limitation of suboptimal specimen preservation. The report discusses proposed hormonal influences, including estrogen-driven vascular growth, and provides a literature review emphasizing that fallopian tube hemangiomas are usually asymptomatic and sporadically documented. Relevance to endometriosis: the patient’s history explicitly includes endometriosis and adenomyosis, and the discussion links estrogen- and hormone-related mechanisms and related pelvic conditions to possible influences on vascular lesion development, though the main focus is a fallopian tube cavernous hemangioma case report and literature review rather than endometriosis itself.

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Unveiling the Uncommon: Fallopian Tube Cavernous Hemangioma in pregnancy with endometriosis background – a case report and literature review | Research Square window.SnipcartSettings = { analytics: { enabled: false } }; (function() { var accessVector = localStorage.getItem('access_vector') || ''; window.dataLayer = window.dataLayer || []; if (accessVector) { window.dataLayer.push({ user: { profile: { profileInfo: { snid: accessVector } } } }); } })(); (function(w,d,s,l,i){w[l]=w[l]||[];w[l].push({'gtm.start':new Date().getTime(),event:'gtm.js'});var f=d.getElementsByTagName(s)[0],j=d.createElement(s),dl=l!='dataLayer'?'&l='+l:'';j.async=true;j.src='https://www.googletagmanager.com/gtm.js?id='+i+dl;f.parentNode.insertBefore(j,f);})(window,document,'script','dataLayer','GTM-K279D39R'); Browse Preprints In Review Journals COVID-19 Preprints AJE Video Bytes Research Tools Research Promotion AJE Professional Editing AJE Rubriq About Preprint Platform In Review Editorial Policies Our Team Advisory Board Help Center Sign In Submit a Preprint Cite Share Download PDF Case Report Unveiling the Uncommon: Fallopian Tube Cavernous Hemangioma in pregnancy with endometriosis background – a case report and literature review Konstantinos Malligiannis Ntalianis, Gladys Hulipas, Hemapriya Mohanraj, and 3 more This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-4521730/v1 This work is licensed under a CC BY 4.0 License Status: Posted Version 1 posted You are reading this latest preprint version Abstract This case report presents a fallopian tube cavernous hemangioma, a highly rare benign tumour, which was found during a caesarean section. Previous medical and gynaecology histories involved endometriosis, adenomyosis, and irritable bowel syndrome. Fallopian tube hemangiomas are sporadic benign vascular tumours, with only 16 cases documented in the literature. These tumours are typically small, ranging from 5 mm to 30 mm in size. The aetiology of hemangiomas is still unclear, but it is commonly believed that the growth of fallopian tube hemangiomas is stimulated by oestrogens, which promote blood vessel growth. Most hemangiomas, which are mainly asymptomatic, are discovered incidentally during surgeries for other conditions or complications. To the best of our knowledge, we report the first ever case of cavernous haemangioma of the fallopian tube identified in pregnancy. Figures Figure 1 Figure 2 Figure 3 Figure 4 Figure 5 Introduction Hemangiomas are tumours of vascular origin and are commonly found in soft tissues and the skin [ 1 ]. Vascular malformations are a diverse group of lesions typically resulting from congenital errors in vessel morphogenesis, characterised by cellular turnover without true proliferation [ 2 ]. The International Society for the Study of Vascular Anomalies (ISSVA) classifies vascular lesions based on morphological and immunohistochemical markers. Capillary hemangiomas are considered "difficult to classify" because their morphological characteristics do not align with any specific type in the ISSVA classification [ 3 ]. Vascular tumours in female pelvic organs are exceedingly are [ 4 ]. Despite most of these lesions being discovered incidentally, they can clinically and radiologically resemble a variety of benign and malignant conditions [ 5 ]. Clinically, hemangiomas may present with a bluish discoloration of the skin and a history of size fluctuations. Pain can occur post-exercise due to the diversion of blood flow from surrounding tissues into the hemangioma. Imaging reveals hemangiomas containing serpentine vessels, fat, smooth muscle, hemosiderin, and phlebolith [ 6 , 7 ]. Histologically, hemangiomas are categorised by the dominant type of vascular channel: capillary, cavernous, arteriovenous, and venous. Cavernous hemangiomas are found in the skin, liver, kidneys, breast, brain, bone, and skeletal muscle. They are characterised by cavern-like vascular spaces larger than those in capillary hemangiomas, lined with non-atypical endothelial cells. Hemangiomas are rare in female genital organs except for the vulva [ 1 , 8 ]. Hemangiomas in female reproductive organs, especially in the fallopian tubes, are extremely rare. The first description was provided by Ragins and Crane in 1947 [ 9 ]. Katiyar et al. reported on capillary hemangiomas, noting eleven documented cases of cavernous hemangiomas in the fallopian tubes of patients aged 13 to 77 years up to 2016 [ 10 ]. Since then, four additional cases of fallopian tube hemangiomas have been published, including three capillary hemangiomas [ 11 ]. Here, we present a case of a fallopian tube cavernous hemangioma in a 39-year-old woman. To the best of our knowledge, we report the first ever case of cavernous haemangioma of the fallopian tube identified in pregnancy. Case Report A white, 39-year-old, nulliparous, pregnant woman was admitted to our unit following spontaneous rupture of membranes during outpatient induction of labour for postdates. The gestational age, on the day of admission, was 42 weeks + 0 days. The background history involved an IVF pregnancy and a positive vaginal swab for Group B Streptococcus. From a medical and gynaecological history point of view, endometriosis, adenomyosis, irritable bowel syndrome (IBS), and asthma were reported. Endometriosis was confirmed in the past radiologically and had laparoscopic left ovarian cystectomy (endometrioma). The patient stayed on the delivery suite for approximately 14 hours and had an oxytocin infusion due to inadequate progress. After 8 hours on oxytocin infusion, in view of the delayed 1st stage of labour and maternal request, an emergency Caesarean section was performed for the delivery. An uneventful caesarean section was done under spinal anesthesia. The baby was born in very good condition with no concerns. Intraoperatively, the uterus was found to have a small subserosal 1x2 cm fibroid; the ovaries were normal, as was the right tube as well. On the left tube, at the fimbrial end, there was a brown-coloured nodule measuring 1 x 1 cm, which was ligated, excised with the patient's consent, and sent to the histology laboratory. The patient recovered well post-operatively and was discharged the following day. Initially, during the routine obstetric scans, there were no radiological findings to suggest the existence of the haemangioma. The patient was asymptomatic throughout pregnancy. The histopathological report showed, macroscopically, a segment of fallopian tube measuring 14 x 5 mm. Upon slicing, no focal lesion was identified. Microscopically, the sections showed a circumscribed and encapsulated lesion (Fig. 1 ) composed of proliferating, variably sized, dilated, and thin-walled vessels (Fig. 2 ). These were lined by a single layer of flattened endothelial cells (Fig. 5 ). Focal fibrin thrombi could be seen in some of the vessels (Figs. 3 and Fig. 4). The preservation of the specimen was not optimal, but there was no evidence of cytologic atypia or mitotic activity. These findings were consistent with a fallopian tubal cavernous haemangioma. Discussion Fallopian tube hemangiomas are rare benign vascular tumours, with only 16 documented cases in the literature. Most of these tumours, ranging from 2 to 10 mm in size, were asymptomatic and discovered incidentally during surgeries for other conditions. Therefore, it is likely that the actual incidence of fallopian tube hemangiomas is much higher [ 11 ]. Most of these vascular lesions appear during the second and third decades of life, and parity does not seem to have a significant impact [ 12 ]. The aetiology of hemangiomas remains unclear. Although hemangiomas are generally considered to be of congenital origin, they can also develop in adults from developmental remnants that retain the capacity for later proliferation [ 13 ]. The growth of hemangiomas in the genital tract is also believed to be influenced by hormonal effects, such as the growth-stimulating effects of ooestrogen on blood vessels, which have been confirmed through immunohistochemistry demonstrating ooestrogen receptors. Also, Bonetti et al. hypothesised that ooestrogen might play a role in the development of hemangiomas due to the presence of ooestrogen receptors in endothelial cells. In their study, they found that all three tumours were immunoreactive for CD31, CD34, and factor VIII-related antigens [ 14 ]. Additionally, ovarian hemangiomas can exert a mass effect on the ovary, causing stromal luteinization and the production of androgens. These androgens are then converted to oestrogen peripherally, potentially affecting the endometrium and myometrium. This mechanism could be one reason for adenomyosis in our case [ 5 ]. Furthermore, pregnancy and contraceptive oral pills may promote the growth or exacerbate the symptoms of an existing lesion, suggesting a hormonal influence. There is a possibility that these changes can lead to actual blood vessel growth or angiogenesis in preexisting vascular anomalies [ 13 , 15 ]. The patient in our case report had a history of adenomyosis, IBS, and endometriosis. Endometriosis is linked to nearly a three-fold increase in the risk of irritable bowel syndrome, with over 20% of women with endometriosis also having irritable bowel syndrome. Endometriosis is a chronic, oestrogen-dependent inflammatory condition characterised by the presence of endometrial tissue outside the uterine cavity. The relationship between endometriosis and irritable bowel syndrome remains unclear, with several theories having been proposed to explain it. One theory suggests an immunological link through the increased activation of mast cells observed in both conditions. Key features of this immunological connection include abnormal levels of inflammatory cytokines and immune cell activation in the peritoneal cavity. Retrograde menstruation, which involves the flow of menstrual blood containing endometrial cells into the pelvic cavity, is another plausible explanation, potentially triggering irritable bowel syndrome symptoms. Another theory for the increased association between these two disorders is a hormonal linkage. This connection involves gonadotropin-releasing hormone-containing neurons and receptors for luteinizing hormone within the pelvic organs and the enteric nervous system. It is hypothesised that the pain experienced by some women with irritable bowel syndrome may result from female sex hormones, as studies have shown that IBS symptoms often worsen during menstruation [ 16 , 17 ]. Differential diagnoses for fallopian tube hemangiomas include lymphangiomas, vascular leiomyomas, mesothelioma, endometriosis, and adenomatoid tumors. Histologically, distinguishing hemangiomas from these neoplasms and fallopian tube cancer is not challenging. However, an immunohistochemical study is recommended for reliable confirmation of the diagnosis. Immunohistochemically, hemangiomas test positive for CD31, CD34, and smooth muscle actin (SMA). Lymphangiomas show positivity for LYVE-1. Both adenomatoid tumours and mesotheliomas exhibit positivity for mesothelial markers, including calretinin, D2-40, and WT-1 protein. Vascular leiomyomas are positive for SMA, desmin, and caldesmon. [ 10 , 11 , 18 , 19 ]. In most instances, hemangiomas are small, asymptomatic, and discovered incidentally. Larger lesions can present with an ovarian torsion, acute abdomen, or rupture leading to hemoperitoneum. In our case, the hemangioma was found incidentally and did not produce any specific signs or symptoms due to its size and location. Most fallopian tube hemangiomas have been incidentally discovered in the left fallopian tube. However, three cases of cavernous hemangiomas have presented with an acute abdomen due to hemoperitoneum. Histologically, cavernous hemangiomas are characterized by irregularly dilated, thick-walled vascular spaces lined with endothelium, whereas capillary hemangiomas consist of proliferating endothelial cells forming lumina with a single layer of endothelial lining and red blood cells [ 10 ]. Considering sparing surgery is an appropriate treatment for fallopian tube hemangioma, as no relapses have been documented in previous cases. Diagnosing before surgery is nearly impossible, and macroscopic diagnosis during surgery is unlikely. Hence, conducting a frozen section examination or taking a biopsy, especially for small lesions, is necessary. Misinterpretations of the tumor’s origin can lead to inadequate surgeries. Misinterpreting a benign tumour in youth may result in unnecessary radical surgery, causing fertility loss, while overlooking a malignant process can endanger life [ 5 , 10 , 11 ]. Conclusion This case report presents a fallopian tube cavernous hemangioma, a highly rare benign tumour, which was found during a caesarean section. Previous medical and gynaecology histories involved endometriosis, adenomyosis, and irritable bowel syndrome. The aetiology of hemangiomas is still unclear, but it is commonly believed that the growth of fallopian tube hemangiomas is stimulated by oestrogens, which promote blood vessel growth. Most hemangiomas, which are mainly asymptomatic, are discovered incidentally during surgeries for other conditions or complications. To the best of our knowledge, we report the first ever case of cavernous haemangioma of the fallopian tube identified in pregnancy. Declarations Funding No specific funding was sought for this publication. Consent for publication Ethics approval and consent to participate Written informed consent was obtained from the patient. A copy of the written consent is available for review by the editor of this journal. Availability of data and materials The datasets used and/or analyzed during the current publication are available from the corresponding author upon reasonable request. Competing interests The authors declare no competing interests. Author Contribution KMN - wrote the main manuscript GH - contributed in the writing of the case report from the obstetric point of viewHM - contributed in the writing of the case report from the histopathology point of view and prepared the figuresCAR- contributed in the writing of the literature reviewAN - reviewed the manuscript from the histopathology point of view and prepared the figuresSS - reviewed the manuscript from the obstetric point of viewAll authors reviewed the manuscript. References Yoon G, & KHS. Characterization of clinicopathological features of tubal cavernous hemangioma. Journal of Clinical and Experimental Pathology, International 2016; 9(7), 7476-7481. Merrow AC, Gupta A, Patel MN et al. 2014 Revised Classification of Vascular Lesions from the International Society for the Study of Vascular Anomalies: Radiologic-Pathologic Update. RadioGraphics 2016; 36 :1494–516. Karpathiou G, Chauleur C, Da Cruz V et al. Vascular lesions of the female genital tract: Clinicopathologic findings and application of the ISSVA classification. Pathophysiology 2017; 24 :161–7. Vilanova JC, Barceló J, Smirniotopoulos JG et al. Hemangioma from Head to Toe: MR Imaging with Pathologic Correlation. RadioGraphics 2004; 24 :367–85. Kalaivanan C, Subbarayan D, Kanna NRR et al. Capillary hemangioma: A concurrent presentation in ovary and fallopian tube. J Midlife Health 2019; 10 :93. Wu JS, Hochman MG. Soft-Tissue Tumors and Tumorlike Lesions: A Systematic Imaging Approach. Radiology 2009; 253 :297–316. Kransdorf MJ, Jelinek JS, Moser RP. Imaging of soft tissue tumors. Radiol Clin North Am 1993; 31 :359–72. Wenger DE, Wold LE. Benign vascular lesions of bone: radiologic and pathologic features. Skeletal Radiol 2000; 29 :63–74. Ragins AB, Crane RD. Cavernous Hemangioma of the Fallopian Tube. Am J Obstet Gynecol 1947; 54 :883–6. Katiyar R. Capillary Hemangioma of the Fallopian Tube. JOURNAL OF CLINICAL AND DIAGNOSTIC RESEARCH 2016, DOI: 10.7860/JCDR/2016/16774.7570. SUMTSOV D, SUMTSOV G, HYRIAVENKO N et al. Fallopian Tube Hemangioma Discovered on Follow-up for Uterine Leiomyoma. Medeni Med J 2024:0–0. Kondi-Pafiti A, Kairi-Vassilatou E, Spanidou-Carvouni H et al. Vascular tumors of the female genital tract: a clinicopathological study of nine cases. Eur J Gynaecol Oncol 2003; 24 :48–50. Kouach J. Haemangioma of the Cervix Micking Cervical Endometriosis: A Case Report. Journal of Gynecology and Obstetrics 2015; 3 :66. Reggiani Bonetti L, Boselli F, Lupi M et al. Expression of estrogen receptor in hemangioma of the uterine cervix: reports of three cases and review of the literature. Arch Gynecol Obstet 2009; 280 :469–72. Cherkis RC, Kamath CP. Hemangioma of the uterine cervix and pregnancy. A case report. J Reprod Med 1988; 33 :393–5. Nabi MY, Nauhria S, Reel M et al. Endometriosis and irritable bowel syndrome: A systematic review and meta-analyses. Front Med (Lausanne) 2022; 9 , DOI: 10.3389/fmed.2022.914356. Resta C, Moustogiannis A, Chatzinikita E et al. Gonadotropin-Releasing Hormone (GnRH)/GnRH Receptors and Their Role in the Treatment of Endometriosis. Cureus 2023, DOI: 10.7759/cureus.38136. Wojnar A, Drożdż K, Dzięgiel P. Cavernous haemangioma of the oviduct. Pol J Pathol 2010; 61 :103–4. Hourani R, Sarkis R, Atallah D et al. Rectal hemangioma mimicking uterosacral endometriosis on transvaginal ultrasound examination. Ultrasound in Obstetrics & Gynecology 2008; 31 :363–5. Additional Declarations No competing interests reported. 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Also discoverable on Platform About Our Team In Review Editorial Policies Advisory Board Help Center Resources Author Services Accessibility API Access RSS feed Manage Cookie Preferences © Research Square 2026 | ISSN 2693-5015 (online) Privacy Policy Terms of Service Do Not Sell My Personal Information {"props":{"pageProps":{"initialData":{"identity":"rs-4521730","acceptedTermsAndConditions":true,"allowDirectSubmit":true,"archivedVersions":[],"articleType":"Case Report","associatedPublications":[],"authors":[{"id":318115536,"identity":"b6fc9411-0bdf-4d92-845b-42aff25a3422","order_by":0,"name":"Konstantinos Malligiannis 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class=\"CitationRef\"\u003e1\u003c/span\u003e]. Vascular malformations are a diverse group of lesions typically resulting from congenital errors in vessel morphogenesis, characterised by cellular turnover without true proliferation [\u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e]. The International Society for the Study of Vascular Anomalies (ISSVA) classifies vascular lesions based on morphological and immunohistochemical markers. Capillary hemangiomas are considered \"difficult to classify\" because their morphological characteristics do not align with any specific type in the ISSVA classification [\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eVascular tumours in female pelvic organs are exceedingly are [\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e]. Despite most of these lesions being discovered incidentally, they can clinically and radiologically resemble a variety of benign and malignant conditions [\u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eClinically, hemangiomas may present with a bluish discoloration of the skin and a history of size fluctuations. Pain can occur post-exercise due to the diversion of blood flow from surrounding tissues into the hemangioma. Imaging reveals hemangiomas containing serpentine vessels, fat, smooth muscle, hemosiderin, and phlebolith [\u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e, \u003cspan citationid=\"CR7\" class=\"CitationRef\"\u003e7\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eHistologically, hemangiomas are categorised by the dominant type of vascular channel: capillary, cavernous, arteriovenous, and venous. Cavernous hemangiomas are found in the skin, liver, kidneys, breast, brain, bone, and skeletal muscle. They are characterised by cavern-like vascular spaces larger than those in capillary hemangiomas, lined with non-atypical endothelial cells. Hemangiomas are rare in female genital organs except for the vulva [\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e, \u003cspan citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eHemangiomas in female reproductive organs, especially in the fallopian tubes, are extremely rare. The first description was provided by Ragins and Crane in 1947 [\u003cspan citationid=\"CR9\" class=\"CitationRef\"\u003e9\u003c/span\u003e]. Katiyar et al. reported on capillary hemangiomas, noting eleven documented cases of cavernous hemangiomas in the fallopian tubes of patients aged 13 to 77 years up to 2016 [\u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e]. Since then, four additional cases of fallopian tube hemangiomas have been published, including three capillary hemangiomas [\u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e]. Here, we present a case of a fallopian tube cavernous hemangioma in a 39-year-old woman. To the best of our knowledge, we report the first ever case of cavernous haemangioma of the fallopian tube identified in pregnancy.\u003c/p\u003e"},{"header":"Case Report","content":"\u003cp\u003eA white, 39-year-old, nulliparous, pregnant woman was admitted to our unit following spontaneous rupture of membranes during outpatient induction of labour for postdates. The gestational age, on the day of admission, was 42 weeks\u0026thinsp;+\u0026thinsp;0 days. The background history involved an IVF pregnancy and a positive vaginal swab for Group B Streptococcus. From a medical and gynaecological history point of view, endometriosis, adenomyosis, irritable bowel syndrome (IBS), and asthma were reported. Endometriosis was confirmed in the past radiologically and had laparoscopic left ovarian cystectomy (endometrioma).\u003c/p\u003e \u003cp\u003eThe patient stayed on the delivery suite for approximately 14 hours and had an oxytocin infusion due to inadequate progress. After 8 hours on oxytocin infusion, in view of the delayed 1st stage of labour and maternal request, an emergency Caesarean section was performed for the delivery.\u003c/p\u003e \u003cp\u003eAn uneventful caesarean section was done under spinal anesthesia. The baby was born in very good condition with no concerns. Intraoperatively, the uterus was found to have a small subserosal 1x2 cm fibroid; the ovaries were normal, as was the right tube as well. On the left tube, at the fimbrial end, there was a brown-coloured nodule measuring 1 x 1 cm, which was ligated, excised with the patient's consent, and sent to the histology laboratory.\u003c/p\u003e \u003cp\u003eThe patient recovered well post-operatively and was discharged the following day.\u003c/p\u003e \u003cp\u003eInitially, during the routine obstetric scans, there were no radiological findings to suggest the existence of the haemangioma. The patient was asymptomatic throughout pregnancy.\u003c/p\u003e \u003cp\u003eThe histopathological report showed, macroscopically, a segment of fallopian tube measuring 14 x 5 mm. Upon slicing, no focal lesion was identified. Microscopically, the sections showed a circumscribed and encapsulated lesion (Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003e) composed of proliferating, variably sized, dilated, and thin-walled vessels (Fig.\u0026nbsp;\u003cspan refid=\"Fig2\" class=\"InternalRef\"\u003e2\u003c/span\u003e). These were lined by a single layer of flattened endothelial cells (Fig.\u0026nbsp;\u003cspan refid=\"Fig3\" class=\"InternalRef\"\u003e5\u003c/span\u003e). Focal fibrin thrombi could be seen in some of the vessels (Figs.\u0026nbsp;\u003cspan refid=\"Fig4\" class=\"InternalRef\"\u003e3\u003c/span\u003e and Fig.\u0026nbsp;4).\u003c/p\u003e \u003cp\u003eThe preservation of the specimen was not optimal, but there was no evidence of cytologic atypia or mitotic activity.\u003c/p\u003e \u003cp\u003eThese findings were consistent with a fallopian tubal cavernous haemangioma.\u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003e \u003c/p\u003e"},{"header":"Discussion","content":"\u003cp\u003eFallopian tube hemangiomas are rare benign vascular tumours, with only 16 documented cases in the literature. Most of these tumours, ranging from 2 to 10 mm in size, were asymptomatic and discovered incidentally during surgeries for other conditions. Therefore, it is likely that the actual incidence of fallopian tube hemangiomas is much higher [\u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e]. Most of these vascular lesions appear during the second and third decades of life, and parity does not seem to have a significant impact [\u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eThe aetiology of hemangiomas remains unclear. Although hemangiomas are generally considered to be of congenital origin, they can also develop in adults from developmental remnants that retain the capacity for later proliferation [\u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e]. The growth of hemangiomas in the genital tract is also believed to be influenced by hormonal effects, such as the growth-stimulating effects of ooestrogen on blood vessels, which have been confirmed through immunohistochemistry demonstrating ooestrogen receptors. Also, Bonetti et al. hypothesised that ooestrogen might play a role in the development of hemangiomas due to the presence of ooestrogen receptors in endothelial cells. In their study, they found that all three tumours were immunoreactive for CD31, CD34, and factor VIII-related antigens [\u003cspan citationid=\"CR14\" class=\"CitationRef\"\u003e14\u003c/span\u003e]. Additionally, ovarian hemangiomas can exert a mass effect on the ovary, causing stromal luteinization and the production of androgens. These androgens are then converted to oestrogen peripherally, potentially affecting the endometrium and myometrium. This mechanism could be one reason for adenomyosis in our case [\u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e]. Furthermore, pregnancy and contraceptive oral pills may promote the growth or exacerbate the symptoms of an existing lesion, suggesting a hormonal influence. There is a possibility that these changes can lead to actual blood vessel growth or angiogenesis in preexisting vascular anomalies [\u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e, \u003cspan citationid=\"CR15\" class=\"CitationRef\"\u003e15\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eThe patient in our case report had a history of adenomyosis, IBS, and endometriosis. Endometriosis is linked to nearly a three-fold increase in the risk of irritable bowel syndrome, with over 20% of women with endometriosis also having irritable bowel syndrome. Endometriosis is a chronic, oestrogen-dependent inflammatory condition characterised by the presence of endometrial tissue outside the uterine cavity. The relationship between endometriosis and irritable bowel syndrome remains unclear, with several theories having been proposed to explain it. One theory suggests an immunological link through the increased activation of mast cells observed in both conditions. Key features of this immunological connection include abnormal levels of inflammatory cytokines and immune cell activation in the peritoneal cavity. Retrograde menstruation, which involves the flow of menstrual blood containing endometrial cells into the pelvic cavity, is another plausible explanation, potentially triggering irritable bowel syndrome symptoms. Another theory for the increased association between these two disorders is a hormonal linkage. This connection involves gonadotropin-releasing hormone-containing neurons and receptors for luteinizing hormone within the pelvic organs and the enteric nervous system. It is hypothesised that the pain experienced by some women with irritable bowel syndrome may result from female sex hormones, as studies have shown that IBS symptoms often worsen during menstruation [\u003cspan citationid=\"CR16\" class=\"CitationRef\"\u003e16\u003c/span\u003e, \u003cspan citationid=\"CR17\" class=\"CitationRef\"\u003e17\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eDifferential diagnoses for fallopian tube hemangiomas include lymphangiomas, vascular leiomyomas, mesothelioma, endometriosis, and adenomatoid tumors. Histologically, distinguishing hemangiomas from these neoplasms and fallopian tube cancer is not challenging. However, an immunohistochemical study is recommended for reliable confirmation of the diagnosis. Immunohistochemically, hemangiomas test positive for CD31, CD34, and smooth muscle actin (SMA). Lymphangiomas show positivity for LYVE-1. Both adenomatoid tumours and mesotheliomas exhibit positivity for mesothelial markers, including calretinin, D2-40, and WT-1 protein. Vascular leiomyomas are positive for SMA, desmin, and caldesmon. [\u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e, \u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e, \u003cspan citationid=\"CR18\" class=\"CitationRef\"\u003e18\u003c/span\u003e, \u003cspan citationid=\"CR19\" class=\"CitationRef\"\u003e19\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eIn most instances, hemangiomas are small, asymptomatic, and discovered incidentally. Larger lesions can present with an ovarian torsion, acute abdomen, or rupture leading to hemoperitoneum. In our case, the hemangioma was found incidentally and did not produce any specific signs or symptoms due to its size and location.\u003c/p\u003e \u003cp\u003eMost fallopian tube hemangiomas have been incidentally discovered in the left fallopian tube. However, three cases of cavernous hemangiomas have presented with an acute abdomen due to hemoperitoneum. Histologically, cavernous hemangiomas are characterized by irregularly dilated, thick-walled vascular spaces lined with endothelium, whereas capillary hemangiomas consist of proliferating endothelial cells forming lumina with a single layer of endothelial lining and red blood cells [\u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eConsidering sparing surgery is an appropriate treatment for fallopian tube hemangioma, as no relapses have been documented in previous cases.\u003c/p\u003e \u003cp\u003eDiagnosing before surgery is nearly impossible, and macroscopic diagnosis during surgery is unlikely. Hence, conducting a frozen section examination or taking a biopsy, especially for small lesions, is necessary. Misinterpretations of the tumor\u0026rsquo;s origin can lead to inadequate surgeries. Misinterpreting a benign tumour in youth may result in unnecessary radical surgery, causing fertility loss, while overlooking a malignant process can endanger life [\u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e, \u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e, \u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e].\u003c/p\u003e"},{"header":"Conclusion","content":"\u003cp\u003eThis case report presents a fallopian tube cavernous hemangioma, a highly rare benign tumour, which was found during a caesarean section. Previous medical and gynaecology histories involved endometriosis, adenomyosis, and irritable bowel syndrome. The aetiology of hemangiomas is still unclear, but it is commonly believed that the growth of fallopian tube hemangiomas is stimulated by oestrogens, which promote blood vessel growth. Most hemangiomas, which are mainly asymptomatic, are discovered incidentally during surgeries for other conditions or complications. To the best of our knowledge, we report the first ever case of cavernous haemangioma of the fallopian tube identified in pregnancy.\u003c/p\u003e"},{"header":"Declarations","content":"\u003cp\u003e\u003cu\u003eFunding\u003c/u\u003e\u003c/p\u003e\n\u003cp\u003eNo specific funding was sought for this publication.\u003c/p\u003e\n\u003cp\u003e\u003cu\u003eConsent for publication\u003c/u\u003e\u003c/p\u003e\n\u003cp\u003eEthics approval and consent to participate\u003c/p\u003e\n\u003cp\u003eWritten informed consent was obtained from the patient. A copy of the written consent is available for review by the editor of this journal.\u003c/p\u003e\n\u003cp\u003e\u003cu\u003eAvailability of data and materials\u003c/u\u003e\u003c/p\u003e\n\u003cp\u003eThe datasets used and/or analyzed during the current publication are available from the corresponding author upon reasonable request.\u003c/p\u003e\n\u003cp\u003e\u003cu\u003eCompeting interests\u003c/u\u003e\u003c/p\u003e\n\u003cp\u003eThe authors declare no competing interests.\u003c/p\u003e\u003ch2\u003eAuthor Contribution\u003c/h2\u003e\u003cp\u003eKMN - wrote the main manuscript GH - contributed in the writing of the case report from the obstetric point of viewHM - contributed in the writing of the case report from the histopathology point of view and prepared the figuresCAR- contributed in the writing of the literature reviewAN - reviewed the manuscript from the histopathology point of view and prepared the figuresSS - reviewed the manuscript from the obstetric point of viewAll authors reviewed the manuscript.\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\n\u003cli\u003eYoon G, \u0026amp; KHS. Characterization of clinicopathological features of tubal cavernous hemangioma. \u003cem\u003eJournal of Clinical and Experimental Pathology, International\u003c/em\u003e 2016;\u003cstrong\u003e9(7), 7476-7481.\u003c/strong\u003e\u003c/li\u003e\n\u003cli\u003eMerrow AC, Gupta A, Patel MN \u003cem\u003eet al.\u003c/em\u003e 2014 Revised Classification of Vascular Lesions from the International Society for the Study of Vascular Anomalies: Radiologic-Pathologic Update. \u003cem\u003eRadioGraphics\u003c/em\u003e 2016;\u003cstrong\u003e36\u003c/strong\u003e:1494\u0026ndash;516.\u003c/li\u003e\n\u003cli\u003eKarpathiou G, Chauleur C, Da Cruz V \u003cem\u003eet al.\u003c/em\u003e Vascular lesions of the female genital tract: Clinicopathologic findings and application of the ISSVA classification. \u003cem\u003ePathophysiology\u003c/em\u003e 2017;\u003cstrong\u003e24\u003c/strong\u003e:161\u0026ndash;7.\u003c/li\u003e\n\u003cli\u003eVilanova JC, Barcel\u0026oacute; J, Smirniotopoulos JG \u003cem\u003eet al.\u003c/em\u003e Hemangioma from Head to Toe: MR Imaging with Pathologic Correlation. \u003cem\u003eRadioGraphics\u003c/em\u003e 2004;\u003cstrong\u003e24\u003c/strong\u003e:367\u0026ndash;85.\u003c/li\u003e\n\u003cli\u003eKalaivanan C, Subbarayan D, Kanna NRR \u003cem\u003eet al.\u003c/em\u003e Capillary hemangioma: A concurrent presentation in ovary and fallopian tube. \u003cem\u003eJ Midlife Health\u003c/em\u003e 2019;\u003cstrong\u003e10\u003c/strong\u003e:93.\u003c/li\u003e\n\u003cli\u003eWu JS, Hochman MG. Soft-Tissue Tumors and Tumorlike Lesions: A Systematic Imaging Approach. \u003cem\u003eRadiology\u003c/em\u003e 2009;\u003cstrong\u003e253\u003c/strong\u003e:297\u0026ndash;316.\u003c/li\u003e\n\u003cli\u003eKransdorf MJ, Jelinek JS, Moser RP. Imaging of soft tissue tumors. \u003cem\u003eRadiol Clin North Am\u003c/em\u003e 1993;\u003cstrong\u003e31\u003c/strong\u003e:359\u0026ndash;72.\u003c/li\u003e\n\u003cli\u003eWenger DE, Wold LE. Benign vascular lesions of bone: radiologic and pathologic features. \u003cem\u003eSkeletal Radiol\u003c/em\u003e 2000;\u003cstrong\u003e29\u003c/strong\u003e:63\u0026ndash;74.\u003c/li\u003e\n\u003cli\u003eRagins AB, Crane RD. Cavernous Hemangioma of the Fallopian Tube. \u003cem\u003eAm J Obstet Gynecol\u003c/em\u003e 1947;\u003cstrong\u003e54\u003c/strong\u003e:883\u0026ndash;6.\u003c/li\u003e\n\u003cli\u003eKatiyar R. Capillary Hemangioma of the Fallopian Tube. \u003cem\u003eJOURNAL OF CLINICAL AND DIAGNOSTIC RESEARCH\u003c/em\u003e 2016, DOI: 10.7860/JCDR/2016/16774.7570.\u003c/li\u003e\n\u003cli\u003eSUMTSOV D, SUMTSOV G, HYRIAVENKO N \u003cem\u003eet al.\u003c/em\u003e Fallopian Tube Hemangioma Discovered on Follow-up for Uterine Leiomyoma. \u003cem\u003eMedeni Med J\u003c/em\u003e 2024:0\u0026ndash;0.\u003c/li\u003e\n\u003cli\u003eKondi-Pafiti A, Kairi-Vassilatou E, Spanidou-Carvouni H \u003cem\u003eet al.\u003c/em\u003e Vascular tumors of the female genital tract: a clinicopathological study of nine cases. \u003cem\u003eEur J Gynaecol Oncol\u003c/em\u003e 2003;\u003cstrong\u003e24\u003c/strong\u003e:48\u0026ndash;50.\u003c/li\u003e\n\u003cli\u003eKouach J. Haemangioma of the Cervix Micking Cervical Endometriosis: A Case Report. \u003cem\u003eJournal of Gynecology and Obstetrics\u003c/em\u003e 2015;\u003cstrong\u003e3\u003c/strong\u003e:66.\u003c/li\u003e\n\u003cli\u003eReggiani Bonetti L, Boselli F, Lupi M \u003cem\u003eet al.\u003c/em\u003e Expression of estrogen receptor in hemangioma of the uterine cervix: reports of three cases and review of the literature. \u003cem\u003eArch Gynecol Obstet\u003c/em\u003e 2009;\u003cstrong\u003e280\u003c/strong\u003e:469\u0026ndash;72.\u003c/li\u003e\n\u003cli\u003eCherkis RC, Kamath CP. Hemangioma of the uterine cervix and pregnancy. A case report. \u003cem\u003eJ Reprod Med\u003c/em\u003e 1988;\u003cstrong\u003e33\u003c/strong\u003e:393\u0026ndash;5.\u003c/li\u003e\n\u003cli\u003eNabi MY, Nauhria S, Reel M \u003cem\u003eet al.\u003c/em\u003e Endometriosis and irritable bowel syndrome: A systematic review and meta-analyses. \u003cem\u003eFront Med (Lausanne)\u003c/em\u003e 2022;\u003cstrong\u003e9\u003c/strong\u003e, DOI: 10.3389/fmed.2022.914356.\u003c/li\u003e\n\u003cli\u003eResta C, Moustogiannis A, Chatzinikita E \u003cem\u003eet al.\u003c/em\u003e Gonadotropin-Releasing Hormone (GnRH)/GnRH Receptors and Their Role in the Treatment of Endometriosis. \u003cem\u003eCureus\u003c/em\u003e 2023, DOI: 10.7759/cureus.38136.\u003c/li\u003e\n\u003cli\u003eWojnar A, Drożdż K, Dzięgiel P. Cavernous haemangioma of the oviduct. \u003cem\u003ePol J Pathol\u003c/em\u003e 2010;\u003cstrong\u003e61\u003c/strong\u003e:103\u0026ndash;4.\u003c/li\u003e\n\u003cli\u003eHourani R, Sarkis R, Atallah D \u003cem\u003eet al.\u003c/em\u003e Rectal hemangioma mimicking uterosacral endometriosis on transvaginal ultrasound examination. \u003cem\u003eUltrasound in Obstetrics \u0026amp; Gynecology\u003c/em\u003e 2008;\u003cstrong\u003e31\u003c/strong\u003e:363\u0026ndash;5.\u003c/li\u003e\n\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":true,"highlight":"","institution":"","isAcceptedByJournal":false,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true},"keywords":"","lastPublishedDoi":"10.21203/rs.3.rs-4521730/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-4521730/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003eThis case report presents a fallopian tube cavernous hemangioma, a highly rare benign tumour, which was found during a caesarean section. Previous medical and gynaecology histories involved endometriosis, adenomyosis, and irritable bowel syndrome. Fallopian tube hemangiomas are sporadic benign vascular tumours, with only 16 cases documented in the literature. These tumours are typically small, ranging from 5 mm to 30 mm in size. The aetiology of hemangiomas is still unclear, but it is commonly believed that the growth of fallopian tube hemangiomas is stimulated by oestrogens, which promote blood vessel growth. Most hemangiomas, which are mainly asymptomatic, are discovered incidentally during surgeries for other conditions or complications. To the best of our knowledge, we report the first ever case of cavernous haemangioma of the fallopian tube identified in pregnancy.\u003c/p\u003e","manuscriptTitle":"Unveiling the Uncommon: Fallopian Tube Cavernous Hemangioma in pregnancy with endometriosis background – a case report and literature review","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2024-07-03 19:04:38","doi":"10.21203/rs.3.rs-4521730/v1","editorialEvents":[{"type":"communityComments","content":0}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"9f79c47e-9b2d-4588-9acb-e445e8d06cbb","owner":[],"postedDate":"July 3rd, 2024","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"posted","subjectAreas":[],"tags":[],"updatedAt":"2024-07-08T12:01:48+00:00","versionOfRecord":[],"versionCreatedAt":"2024-07-03 19:04:38","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-4521730","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-4521730","identity":"rs-4521730","version":["v1"]},"buildId":"WvIrzKhiLBfengagbw6Ux","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}

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