{"paper_id":"8afedb2f-b68d-4ace-b896-f299e9e39ef8","body_text":"394\nHemolacria or bloody tears in presumed lacrimal endometriosis:  \na case report\nHemolacria o lágrimas hemáticas en sospecha de endometriosis \nlacrimal: reporte de un caso\nMaría Muñoz-Olarte*, Alba González-Sevilla, Gloria P . Martínez-Ramón, Andrea Gutiérrez-Landaluce, and \nPilar Sáenz-Pascual\nGynecology and Obstetrics Service, Hospital San Pedro, Logroño, La Rioja, España\nCLINICAL CASE\nAbstract\nBackground: Hemolacria or the presence of blood in tears is a rare condition, and there are only a few cases reported in \nthe literature. Hemolacria is associated with multiple underlying diseases, including vicarious menstruation due to extrageni -\ntal endometriosis. Case report:  We present a 26-year-old woman with hemolacria and abdominal pain related to her mens -\ntrual cycle. The patient was diagnosed with bilateral ovarian endometriomas. After ruling out other possible causes of hemo -\nlacria, a progestin-only treatment was applied, with improvement of the abdominal pain and complete remission of \nhemolacria. Conclusions: When faced with hemolacria, a thorough anamnesis and physical examination must be performed, \nsometimes involving more than one specialist to reach a diagnosis. Considering hemolacria is a sign of a subjacent patho -\nlogy, its treatment should be specific one for the disease in each case.\nKeywords: Hemolacria. Blood tears. Menstruation.\nResumen\nAntecedentes: La hemolacria o presencia de sangre en las lágrimas es una afección poco frecuente y sólo hay unos pocos \ncasos descritos en la literatura. La hemolacria se asocia a múltiples enfermedades subyacentes, incluida la menstruación \nvicaria debida a endometriosis extragenital. Caso clínico: Presentamos a una mujer de 26 años con hemolacria y dolor \nabdominal relacionado con su ciclo menstrual. La paciente fue diagnosticada de endometriomas ováricos bilaterales. T ras \ndescartar otras posibles causas de hemolacria, se aplicó un tratamiento sólo con progestágenos, con mejoría del dolor \nabdominal y remisión completa de la hemolacria. Conclusiones: Ante una hemolacria se debe realizar una anamnesis y \nexploración física minuciosa, en la que a veces interviene más de un especialista para llegar al diagnóstico. T eniendo en \ncuenta que la hemolacria es signo de una patología subyacente, su tratamiento debe ser el específico para la enfermedad \nen cada caso.\nPalabras clave: Hemolacria. Lágrimas hemáticas. Menstruación.\n*Correspondence: \nMaría Muñoz-Olarte  \nE-mail: mariamunozolarte@gmail.com\n0048-766X / © 2023 Sociedad Chilena de Obstetricia y Ginecología. Publicado por Permanyer. Este es un artículo open access  bajo la licencia \nCC BY-NC-ND (https://creativecommons.org/licenses/by-nc-nd/4.0/).\nAvailable online: 15-12-2023\nRev Chil Obstet Ginecol. 2023;88(6):394-396\nwww.rechog.com \nDate of reception: 04-03-2023\nDate of acceptance: 25-08-2023\nDOI: 10.24875/RECHOG.23000021\nRevista Chilena de \nObstetricia yGinecología\n\nM. Muñoz-Olarte et al.  Hemolacria and endometriosis\n395\nIntroduction\nHemolacria or the presence of blood in tears is a rare \ncondition with few cases reported in the literature. This \nsign is associated with multiple underlying diseases, \nbut its prevalence and epidemiology remain uncertain. \nSince the 16 th  century, there have been reports of \nwomen who experienced bloody tears related to mens -\ntrual cycle. The source of bleeding can be classified \ninto local causes or related to systemic diseases, and \nthe anamnesis and physical examination are essential \nto establish the diagnosis and decide on which additio -\nnal studies to perform 1,2.\nCase report\nWe report the case of a 26-year-old female patient \nwho experienced abdominal pain and cyclic hemolacria \nup to four occasions from her right eye related to her \nmenstrual cycle in the past year. The patient was refe -\nrred to an ophthalmologist, and the examination did not \nreveal any ocular pathology. Meanwhile, the dysmeno -\nrrhea was studied by the gynecologist, and the echo -\ngraphy revealed a 27-mm endometrioma in her right \novary and a 32-mm on the left side, and treatment with \na progestin-only pill was initiated. A complete labora -\ntory test including coagulation, renal, and liver profiles \nwas performed, with no additional findings. A magnetic \nresource imaging (MRI) of the orbits and head was not \nperformed due to claustrophobia, and the patient did \nnot consent to the performance of a computed tomo -\ngraphy (CT).\nIn two follow-up visits, 3 and 6 months after starting \ntreatment with progestin, the patient referred improve -\nment in the dysmenorrhea, and she had not suffered \nany other episode of hemolacria. The echography \nrevealed similar findings, with slightly smaller bilateral \nendometriomas. Given the favorable progression, the \npatient refused to perform any additional studies, and \nthe diagnosis remained uncertain, with a high suspicion \nof extragenital endometriosis located in the right lacri -\nmal gland.\nDiscussion\nHemolacria has been reported in all age groups, and \nin most cases, it appears unilaterally, which does not \ndisprove a systemic cause for this sign 1-3.\nThe different causes of hemolacria can be classified \ninto local and general ones. Between local causes, a \nrelatively common one is retrograde epistaxis, caused \nby nasal packing or by pinching the nose in a patient \nwith congenital absence or incompetence of the valves \nin the nasolacrimal duct. Other local causes include \nophthalmic inflammation or infections such as pyogenic \ngranuloma, which is a highly vascularized tumor on the \nconjunctiva or the lacrimal sac, and its excision with \ndiathermy is advised to stop hemolacria. Vascular mal -\nformations (varicose veins and hemorrhagic telangiec -\ntasias), foreign bodies, or tumors at the lacrimal level, \nmainly hemangiomas and melanomas, should also be \nconsidered. The reported treatment for patients with a \nvascular malformation is the application of ophthalmic \ntimolol. Hemolacria can also be found after head and \nfacial surgical procedures or traumatisms (cranial and \nocular trauma, nasal bone fractures, and Le Fort frac -\ntures). In women of reproductive age, a hormonal cause \nhas been reported related to endometriosis on the lacri -\nmal gland 3-7.\nThe conjunctiva may cyclically bleed during mens -\ntruation or hormonal disturbances, especially around \nmenarche, and this phenomenon is known as vicarious \nmenstruation. The source of blood in hemolacria cau -\nsed by vicarious menstruation has been thought to be \nendometriosis of the lacrimal gland or conjunctiva, hor -\nmonal stimulation of the conjunctiva, or light hyperten -\nsion caused by estrogen. The ophthalmic examination \nmay be normal in some patients. Hemolacria in these \ncases is usually painless, may last for seconds to minu -\ntes, and may involve one or both eyes. The prevalence \nof extragenital endometriosis is unknown, and the \nnasolacrimal canal is an infrequent site for its appea -\nrance and unusual presentation. Histology is the tool \nfor definitive diagnosis, but for some locations, it is \nimpossible to obtain, making the cyclic character of the \nsymptoms the way to establish diagnosis 2,8.\nRegarding general causes, hemolacria can be a \nmanifestation of Henoch-Schönlein purpura due to the \nformation of IgA1 immune complexes in small-caliber \nvessels. Any scenario related to the presence or appea-\nrance of telangiectasias is a potential cause of hemo -\nlacria, such as Rendu-Osler-Weber disease, pregnancy, \nRaynaud’s syndrome, or various immune diseases \n(dermatomyositis, scleroderma, mastocytosis, and sys -\ntemic lupus erythematosus). Hemolacria can also be \nfound due to hypertension or the combination of diffe -\nrent treatments, such as anticoagulants and antiplatelet \naggregators, especially aspirin and warfarin combined. \nMore infrequent causes of this sign include Vitamin C \ndeficiency that leads to vascular fragility or Gardner-\nDiamond syndrome known for ecchymotic outbreaks. \nThere are also idiopathic cases of hemolacria, when all \n\nRev  Chil Obstet  GineCOl. 2023;88(6)\n396\nother causes are excluded, which are estimated to be \naround 30% of all cases. Finally, there have been \nreports of psychological simulation of hemolacria, with \na difficult diagnosis in these cases 3,4,7.\nFor diagnosis, an accurate anamnesis and a systemic \nphysical, especially ocular examination are essential, per-\nformed preferably by an ophthalmologist. The initial study \nshould include complete laboratory tests with coagulation, \nrenal and liver profiles, and factor deficiencies (VIII and \nvon Willebrand factor antigen assays). A regurgitation test \nto rule out blockades as reflux of blood may suggest the \npresence of a lacrimal gland tumor as well as a capillary \nfragility test is also recommended. In recurrent cases, \nimaging studies may be performed such as a CT scan or \nan MRI of the orbits and head to rule out masses or an \norbital varix. If a mass of the lacrimal gland or a conjunc-\ntival lesion is found, a biopsy is indicated1,3,5.\nThe treatment of hemolacria depends on the subja -\ncent cause. Most cases of hemolacria are mild and \nprogress without complications after identification and \nmanagement of the triggering factor 2,4.\nConclusion\nAnamnesis and ophthalmological examination when \nconfronted with hemolacria are essential, and in most \ncases, determine the etiology. When no signs of a local \ncause are found, there are different studies that can be \nperformed according to the rest of the signs or symp -\ntoms referred to by the patient.\nExtragenital endometriosis is an infrequent disease, \nand the lacrimal glands are one of its rarest locations, \nwith the main symptom of its diagnosis being the appea-\nrance of hemolacria. Biopsy is the gold standard for \ndiagnosis, although it may be challenging, and someti -\nmes, the cyclic nature of hemolacria is the only tool to \nestablish diagnosis. Treatment for endometriosis of the \nlacrimal gland is not defined, and in our case, it presented \na favorable progression with progestin-only treatment.\nAcknowledgments\nThe authors thank the patient for her consent to the \npublication of this case report.\nFunding\nThe authors declare that they have not received \nfunding.\nConflicts of interest\nThe authors declare no conflicts of interest.\nStatement of ethics\nThe authors state that ethical approval is not required \nfor this study in accordance with national guidelines. \nWritten informed consent was obtained from the patient \nfor publication of the details of their medical case and \nany accompanying images.\nEthical disclosures\nProtection of human and animal subjects.  The \nauthors declare that no experiments were performed \non humans or animals for this study.\nConfidentiality of data.  The authors declare that \nthey have followed the protocols of their work center on \nthe publication of patient data.\nRight to privacy and informed consent.  The \nauthors have obtained the written informed consent of \nthe patients or subjects mentioned in the article. \nThe corresponding author is in possession of this \ndocument.\nReferences\n 1. AlGoraini Y, Şeyhibrahim A, Jawish M. A  healthy young girl crying out \nblood: a case report. Heliyon. 2021;7:e07143.\n 2. Tripathy K, Salini B. Hemolacria. In: StatPearls. Treasure Island, \nFL: StatPearls Publishing; 2022. Available from: https://www.ncbi.nlm.nih.\ngov/books/NBK539774 [Last accessed on 2023 Jan 14].\n 3. Dillivan KM. Hemolacria in a patient with severe systemic diseases. \nOptom Vis Sci. 2013;90:e161-6.\n 4. Billoir P, Feugray G, Chrétien MH, Fresel M, Le Cam Duchez V. Quel \nbilan devant une haemolacria  ? À propos d’un cas et revue de la littéra -\nture. Rev Méd Interne. 2020;41:339-42.\n 5. Audelan T, Best AL, Ameline V. Hémolacrie, à propos d’un cas pédiatri -\nque. J Français Ophtalmol. 2019;42:e15-7.\n 6. Drake AE, Packer CD. Epistaxis complicated by hemolacria: a case re -\nport. Clin Med Res. 2020;18:99-101.\n 7. Idowu OO, Kaidonis G, Husain S, Winn BJ. Case report: crying blood. \nOptom Vis Sci. 2021;98:217-21.\n 8. Türkçüo ğlu I, Türkçüo ğlu P, Kurt J, Yldrm H. Presumed nasolacrimal \nendometriosis. Ophthalmic Plast Reconstr Surg. 2008;24:47-8.","source_license":"CC0","license_restricted":false}