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Revista de Gastroenterología de México. 2014;79(1):58---66 REVISTA DE GASTROENTEROLOGIA DE MEXICO ´ ´ www.elsevier.es/rgmx SCIENTIFIC LETTERS Anorectal leiomyoma: A case report and literature review Leiomioma anorrectal. Presentación de un caso y revisión de la literatura Leiomyoma is a tumor strain that derives from the smooth muscle fibers and develops in the regions where these fibers are present. In the digestive tract, the anorectal location is uncommon and is usually diagnosed late due to intralumi- nal growth in over 50% of the cases. Given the exceptional location, we present the case of a 49-year-old woman who sought medical attention for a tumor of 7-month progres- sion in the perineal region that had progressively increased in size. She was asymptomatic during this period and did not present with tenesmus, rectorrhagia, or proctalgia. Physi- cal examination revealed a right perianal tumor of 3 cm in diameter that was mobile and had an elastic consistency to the touch (fig. 1). Nuclear magnetic resonance imag- ing showed a well-defined, nodular, oval-shaped lesion that measured about 5 cm in diameter, with heterogeneous con- trast medium uptake. Its location was intersphincteric and Figure 1 Right perianal tumor. Please cite this article as: García-Santos EP, Ruescas-García FJ, Estaire-Gómez M, Martín-Fernández J, González-López L. Leiomioma anorrectal. Presentación de un caso y revisión de la literatura. Revista de Gastroenterología de México. 2014;79:58---60. adjacent to the intergluteal fold. An ultrasound image iden- tified a hypoechogenic ovoid nodule with abundant vascular flow. A thick needle biopsy was performed and the preoper- ative anatomopathologic diagnosis was leiomyoma (fig. 2). The patient was operated on and an oval intersphincteric tumor with a smooth external surface and a conglomeratic aspect was removed; there was no solution of continuity with the superficial fascicule of the external anal sphincter and minimum continuity with residual fibers of the internal anal sphincter (fig. 3). Patient progression was favorable and she is presently asymptomatic, with no tumor recurrence and with complete sphincteric continence. Leiomyoma was first described by Virchow in 1854, defin- ing it as a benign tumor of mesenchymal origin dependent on smooth muscle fibers. It represents approximately 3.8% of all the benign, soft tissue tumors and its pathogeny con- tinues to be debated today. 1---3 It is classified into 2 variants: superficial and deep. One of the better known examples of the superficial variant is angioleiomyoma of the female genital tract. Leiomyoma of the deep variant, described by Kilpatrick and Billings, is in turn classified as somatic and as retroperitoneal. Unlike the somatic leiomyomas, 20% of the retroperitoneal type show low levels of mitotic activity. Leiomyomas of the superficial variant tend to more often affect the extremities and they affect both sexes, whereas the retroperitoneal variant involves the pelvic area in post- menopausal women. The organ most commonly affected in the gastrointesti- nal tract is the stomach, followed by the small bowel. The esophagus, colon, and rectum are less frequent locations. 4,5 The anorectal location presents in under 0.1% and is con- sidered an extraordinary entity with an overall rectal tumor incidence of 1:2000. Tumor growth is intraluminal in 50% of the cases, extrarectal in 30%, intra and extraluminal in 10%, and intramural in the remaining 10%. 6 The clinical course of these tumors varies widely, and it is the location and vascularization that determine patient symptomatology. 7 Due to their submucosal origin, they are often asymptomatic in their initial stages. Later, when they reach significant sizes, anal and perineal pain, bowel transit alterations, pruritus, rectorrhagia, or tenesmus are some of the most frequent symptoms for which the patient seeks medical attention. Our patient was asymptomatic during the progression of tumor growth, probably being in the initial stage of the disease. Tumors of the deep soft tissues tend to grow larger than the superficial variant due to the fact that they go by unde- tected and are diagnosed in the late stages. 2255-534X/$ see front matter © 2013 Asociación Mexicana de Gastroenterología. Published by Masson Doyma México S.A. All rights reserved.

REVISTA DE GASTROENTEROLOGIA DE MEXICO · Revista de Gastroenterología de México. 2014;79:58---60. adjacent to the intergluteal fold. An ultrasound image iden-a hypoechogenic ovoid

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    evista de Gastroenterología de México. 2014;79(1):58---66

    REVISTA DEGASTROENTEROLOGIA

    DE MEXICO´

    ´

    www.elsevier.es/rgmx

    CIENTIFIC LETTERS

    norectal leiomyoma: A caseeport and literature review�

    adjacent to the intergluteal fold. An ultrasound image iden-tified a hypoechogenic ovoid nodule with abundant vascular

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    eiomioma anorrectal. Presentación de unaso y revisión de la literatura

    eiomyoma is a tumor strain that derives from the smoothuscle fibers and develops in the regions where these fibers

    re present. In the digestive tract, the anorectal location isncommon and is usually diagnosed late due to intralumi-al growth in over 50% of the cases. Given the exceptionalocation, we present the case of a 49-year-old woman whoought medical attention for a tumor of 7-month progres-ion in the perineal region that had progressively increasedn size. She was asymptomatic during this period and did notresent with tenesmus, rectorrhagia, or proctalgia. Physi-al examination revealed a right perianal tumor of 3 cm iniameter that was mobile and had an elastic consistencyo the touch (fig. 1). Nuclear magnetic resonance imag-ng showed a well-defined, nodular, oval-shaped lesion thateasured about 5 cm in diameter, with heterogeneous con-

    rast medium uptake. Its location was intersphincteric and

    Figure 1 Right perianal tumor.

    � Please cite this article as: García-Santos EP, Ruescas-GarcíaJ, Estaire-Gómez M, Martín-Fernández J, González-López L.eiomioma anorrectal. Presentación de un caso y revisión de laiteratura. Revista de Gastroenterología de México. 2014;79:58---60.

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    255-534X/$ – see front matter © 2013 Asociación Mexicana de Gastroente

    ow. A thick needle biopsy was performed and the preoper-tive anatomopathologic diagnosis was leiomyoma (fig. 2).he patient was operated on and an oval intersphinctericumor with a smooth external surface and a conglomeraticspect was removed; there was no solution of continuityith the superficial fascicule of the external anal sphincternd minimum continuity with residual fibers of the internalnal sphincter (fig. 3). Patient progression was favorable andhe is presently asymptomatic, with no tumor recurrencend with complete sphincteric continence.

    Leiomyoma was first described by Virchow in 1854, defin-ng it as a benign tumor of mesenchymal origin dependentn smooth muscle fibers. It represents approximately 3.8%f all the benign, soft tissue tumors and its pathogeny con-inues to be debated today.1---3It is classified into 2 variants:uperficial and deep. One of the better known examplesf the superficial variant is angioleiomyoma of the femaleenital tract. Leiomyoma of the deep variant, described byilpatrick and Billings, is in turn classified as somatic ands retroperitoneal. Unlike the somatic leiomyomas, 20% ofhe retroperitoneal type show low levels of mitotic activity.eiomyomas of the superficial variant tend to more oftenffect the extremities and they affect both sexes, whereashe retroperitoneal variant involves the pelvic area in post-enopausal women.The organ most commonly affected in the gastrointesti-

    al tract is the stomach, followed by the small bowel. Thesophagus, colon, and rectum are less frequent locations.4,5

    he anorectal location presents in under 0.1% and is con-idered an extraordinary entity with an overall rectal tumorncidence of 1:2000. Tumor growth is intraluminal in 50% ofhe cases, extrarectal in 30%, intra and extraluminal in 10%,nd intramural in the remaining 10%.6

    The clinical course of these tumors varies widely, and its the location and vascularization that determine patientymptomatology.7 Due to their submucosal origin, they areften asymptomatic in their initial stages. Later, when theyeach significant sizes, anal and perineal pain, bowel transitlterations, pruritus, rectorrhagia, or tenesmus are somef the most frequent symptoms for which the patient seeksedical attention. Our patient was asymptomatic during therogression of tumor growth, probably being in the initial

    tage of the disease.

    Tumors of the deep soft tissues tend to grow larger thanhe superficial variant due to the fact that they go by unde-ected and are diagnosed in the late stages.

    rología. Published by Masson Doyma México S.A. All rights reserved.

    dx.doi.org/http://www.elsevier.es/rgmxhttp://crossmark.crossref.org/dialog/?doi=10.1016/j.rgmxen.2014.05.006&domain=pdf

  • SCIENTIFIC LETTERS 59

    Figure 2 A) Neoplastic proliferation made up of short fascicles of fusiform cells, without atypia (H&E x20). B) Immunohistochem-ical positivity for smooth muscle markers (x20). C) Positivity for desmin (x20). D) Positivity for H-caldesmon (x20).

    Figure 3 A) Surgical removal of the anorectal tumor that does not appear to have a solution of continuity with the sphincteric

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    fibers. B) Smooth surface oval-shaped tumor.

    The clinical symptoms of the patient, together with acomplete physical examination including endoscopy withbiopsy are usually essential for diagnosis. Imaging tech-niques are useful for preoperative staging because theyenable the relation with the sphincters and urogenital tractto be defined.8 In the case of our patient, the preoperativestudies performed allowed us to make the diagnosis and planthe surgical intervention.

    In relation to management, complete surgical resectionof the tumor is the treatment of choice. Easily accessi-ble intrarectal leiomyomas can be transanally resected.Low anterior resection or abdominoperineal amputationshould be reserved for those cases in which local resection

    cannot be performed due to tumor size or wall involve-ment. Another approach should be contemplated, whichcould be transperineal, pararectal, or transvaginal, whenthe tumor is extrarectal.9 Our patient developed a

    mrIp

    rogressive increase in tumor size with no other accompany-ng symptoms, and so the decision was made to extirpate theumor.

    These tumors recur in up to 40% of the cases andp to 10% recur as leiomyosarcomas; this appears to beelated to incomplete resection or a size greater than 5 cm.he fact that there is the possibility of recurrence, even in

    ow-grade tumors, makes follow-up essential.9,10

    In conclusion, leiomyoma of the anal sphincter is aare entity in the gastrointestinal tract, and because ofts location it is important to make the correct differ-ntial diagnosis with other entities that develop at theame site. When there is suspicion, biopsy is recom-

    ended, as well as an imaging study to determine the

    elation with the sphincteric complex and adjacent organs.n this manner, the adequate surgical approach can belanned.

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    0 SCIENTIFIC LETTERS

    inancial disclosure

    o financial support was received in relation to this article.

    onflict of interest

    he authors declare that there is no conflict of interest.

    eferences

    1. Kim HJ, Lee KY, Kim YW. Case report: Imaging features of peri-anal leiomyoma. Br J Radiol. 2010;83:88.

    2. Dasari BV, Khosraviani K, Irwin TS, et al. Perianal leiomyomainvolving the anal sphincter. Ulster Med J. 2007;76:173---4.

    3. Misumi S, Irie T, Fukuda K, et al. Un caso de leiomioma pro-fundidad del tejido blando. Hallazgos en TC y RM. Radiat Med.2000;18:253---6.

    4. Salvans S, Gimeno J, Parés D. Leiomioma perianal. Rev EspEnferm Dig. 2009;101:209---11.

    5. Campos FG, Leite AF, Araújo SE, et al. Anorectal leiomy-omas: Report of two cases with different anatomical patternsand literature review. Rev Hosp Clin Fac Med Sao Paulo.2004;59:296---301.

    6. Vorobyov GI, Odaryuk TS, Kapuller LL, et al. Surgical treat-ment of benign, myomatous rectal tumors. Dis Colon Rectum.1992;35:328---31.

    7. Hatch KF, Blanchard DK, Hatch 3rd GF, et al. Tumors of therectum and anal canal. World J Surg. 2000;24:437---43.

    8. Marca MI, Saclarides TJ. Linfoma, tumores de tejidos blandosneuroendocrinos y del recto. Clínicas de Colon Rectal Surg.2002;15:71---9.

    9. Nappi C, Di Spiezio Sardo A, Mandato VD, et al. Leiomy-omatosis peritonealis disseminata in association with Currarinosyndrome? BMC Cancer. 2006;6:127.

    10. Núñez Martínez O, Salinas Moreno S, Manceñido Marcos N, et al.Leiomioma rectal: resección endoscópica. Gastroenterol Hepa-tol. 2012;35:373---5.

    E.P. García-Santosa,∗, F.J. Ruescas-Garcíaa,M. Estaire-Gómeza, J. Martín-Fernándeza,L. González-Lópezb

    a Servicio de Cirugía General y de Aparato Digestivo,Hospital General Universitario de Ciudad Real,Ciudad Real, Spainb Servicio de Anatomía Patológica, Hospital GeneralUniversitario de Ciudad Real, Ciudad Real, Spain

    ∗ Corresponding author: Hospital General Universitario deCiudad Real, c/Obispo Rafael Torija S/N; 13005, Ciudad Real,Spain. Phone: +926 27 80 00.E-mail address: esther garcia [email protected](E.P. García-Santos).

    anenteric melanosis secondaryo melanoma of the rectum:

    case report�

    elanosis panentérica secundaria a melanomaectal. Reporte de caso

    78-year-old woman, resident of Mexico City, had a pastistory of open cholecystectomy, scoliosis in occasionalreatment with anti-inflammatory agents for 10 years, andllergy to penicillin.

    Study motive. One year and 6 months with hyporexia,ccasional nausea with no vomiting, mild intermittent painn the epigastrium and hypogastrium, weight loss of 4 kg inne year, stools with reduced consistency, and hematocheziauring the past 4 months.

    The patient had a body mass index of 25, oval-shapedphelides measuring 2-3 mm on the face and neck, and 1-

    mm on the back of the hands, a mildly distended abdomen,nd hepatomegaly.

    Laboratory tests reported negative fecal occult blood,emoglobin 8.9 mg/dl, and hematocrit 28%.

    and fine needle biopsy of the lesions was performed.The histopathologic study reported malignant melanomametastasis. Cytology was also consistent with malignantmelanoma.

    Lower gastrointestinal endoscopy revealed a vegetative,lobulated, hyperchromic lesion 2 cm from the anal mar-gin consistent with malignant melanoma and diffuse flathyperchromic lesions measuring 2-3 mm in the descendingcolon and the sigmoid colon (fig. 1). The histopathologicreport confirmed malignant melanoma. Upper gastrointesti-nal endoscopy showed unaltered esophageal mucosa andmultiple non-confluent 2-3 mm flat, hyperchromic lesions inthe stomach. The first and second portion of the duodenumhad multiple flat, hyperchromic lesions of similar charac-teristics (fig. 2). The histopathologic study reported gastricand duodenal melanosis due to melanin deposits at themacrophage level of the lamina propria. For the differentialdiagnosis a Fontana-Masson stain was done and its result waspositive (fig. 3); a Pearls stain was negative in both biopsies.

    A distinction should be made between melanosis, as thepigmentation of mucosae from the deposit of melanin inthe macrophages-lysosomes at the level of the lamina pro-pria, and pseudomelanosis, described as a pigmentation of

    She had been studied by the Oncology Service 4 monthsarlier for suspicion of cancer. An abdominal tomographycan showed hepatic lesions consistent with metastasis

    � Please cite this article as: Armienta SR, Camacho NM, Hernán-ez GME, Plata PEJ. Melanosis panentérica secundaria a melanomaectal. Reporte de caso. Revista de Gastroenterología de México.014;79:60---62.

    thaaciilt

    he mucosae secondary to deposits of iron, ferrous sulfide,emosiderin, lipomelanin, lipofuscin, calcium, potassium,luminum, magnesium, and silver at the same level.1 After

    review of the medical literature, we found reports ofases with pseudomelanosis secondary to the deposit ofron mainly at the level of the duodenum, with a predom-

    nance in Afro-American women in the seventh decade ofife (range of 18 months -79 years).2---5 Important pigmenta-ion pathology references at the level of the digestive tract

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