Hemolacria or bloody tears in presumed lacrimal endometriosis: a case report

Background: Hemolacria or the presence of blood in tears is a rare condition, and there are only a few cases reported in the literature. Hemolacria is associated with multiple underlying diseases, including vicarious menstruation due to extrageni - tal endometriosis. Case report: We present a 26-year-old woman with hemolacria and abdominal pain related to her mens - trual cycle. The patient was diagnosed with bilateral ovarian endometriomas. After ruling out other possible causes of hemo - lacria, a progestin-only treatment was applied, with improvement of the abdominal pain and complete remission of hemolacria. Conclusions: When faced with hemolacria, a thorough anamnesis and physical examination must be performed, sometimes involving more than one specialist to reach a diagnosis. Considering hemolacria is a sign of a subjacent patho - logy, its treatment should be specific one for the disease in each case.

Hemolacria. Blood tears. Menstruation. Resumen Antecedentes: La hemolacria o presencia de sangre en las lágrimas es una afección poco frecuente y sólo hay unos pocos casos descritos en la literatura. La hemolacria se asocia a múltiples enfermedades subyacentes, incluida la menstruación vicaria debida a endometriosis extragenital. Caso clínico: Presentamos a una mujer de 26 años con hemolacria y dolor abdominal relacionado con su ciclo menstrual. La paciente fue diagnosticada de endometriomas ováricos bilaterales. T ras descartar otras posibles causas de hemolacria, se aplicó un tratamiento sólo con progestágenos, con mejoría del dolor abdominal y remisión completa de la hemolacria. Conclusiones: Ante una hemolacria se debe realizar una anamnesis y exploración física minuciosa, en la que a veces interviene más de un especialista para llegar al diagnóstico. T eniendo en cuenta que la hemolacria es signo de una patología subyacente, su tratamiento debe ser el específico para la enfermedad en cada caso. Palabras clave: Hemolacria. Lágrimas hemáticas. Menstruación. *Correspondence: María Muñoz-Olarte E-mail: [email protected] 0048-766X / © 2023 Sociedad Chilena de Obstetricia y Ginecología. Publicado por Permanyer. Este es un artículo open access bajo la licencia CC BY-NC-ND (https://creativecommons.org/licenses/by-nc-nd/4.0/). Available online: 15-12-2023 Rev Chil Obstet Ginecol. 2023;88(6):394-396 www.rechog.com Date of reception: 04-03-2023 Date of acceptance: 25-08-2023 DOI: 10.24875/RECHOG.23000021 Revista Chilena de Obstetricia yGinecología M. Muñoz-Olarte et al. Hemolacria and endometriosis 395

Hemolacria or the presence of blood in tears is a rare condition with few cases reported in the literature. This sign is associated with multiple underlying diseases, but its prevalence and epidemiology remain uncertain. Since the 16 th  century, there have been reports of women who experienced bloody tears related to mens - trual cycle. The source of bleeding can be classified into local causes or related to systemic diseases, and the anamnesis and physical examination are essential to establish the diagnosis and decide on which additio - nal studies to perform 1,2. Case report We report the case of a 26-year-old female patient who experienced abdominal pain and cyclic hemolacria up to four occasions from her right eye related to her menstrual cycle in the past year. The patient was refe - rred to an ophthalmologist, and the examination did not reveal any ocular pathology. Meanwhile, the dysmeno - rrhea was studied by the gynecologist, and the echo - graphy revealed a 27-mm endometrioma in her right ovary and a 32-mm on the left side, and treatment with a progestin-only pill was initiated. A complete labora - tory test including coagulation, renal, and liver profiles was performed, with no additional findings. A magnetic resource imaging (MRI) of the orbits and head was not performed due to claustrophobia, and the patient did not consent to the performance of a computed tomo - graphy (CT). In two follow-up visits, 3 and 6 months after starting treatment with progestin, the patient referred improve - ment in the dysmenorrhea, and she had not suffered any other episode of hemolacria. The echography revealed similar findings, with slightly smaller bilateral endometriomas. Given the favorable progression, the patient refused to perform any additional studies, and the diagnosis remained uncertain, with a high suspicion of extragenital endometriosis located in the right lacri - mal gland.

Hemolacria has been reported in all age groups, and in most cases, it appears unilaterally, which does not disprove a systemic cause for this sign 1-3. The different causes of hemolacria can be classified into local and general ones. Between local causes, a relatively common one is retrograde epistaxis, caused by nasal packing or by pinching the nose in a patient with congenital absence or incompetence of the valves in the nasolacrimal duct. Other local causes include ophthalmic inflammation or infections such as pyogenic granuloma, which is a highly vascularized tumor on the conjunctiva or the lacrimal sac, and its excision with diathermy is advised to stop hemolacria. Vascular mal - formations (varicose veins and hemorrhagic telangiec - tasias), foreign bodies, or tumors at the lacrimal level, mainly hemangiomas and melanomas, should also be considered. The reported treatment for patients with a vascular malformation is the application of ophthalmic timolol. Hemolacria can also be found after head and facial surgical procedures or traumatisms (cranial and ocular trauma, nasal bone fractures, and Le Fort frac - tures). In women of reproductive age, a hormonal cause has been reported related to endometriosis on the lacri - mal gland 3-7. The conjunctiva may cyclically bleed during mens - truation or hormonal disturbances, especially around menarche, and this phenomenon is known as vicarious menstruation. The source of blood in hemolacria cau - sed by vicarious menstruation has been thought to be endometriosis of the lacrimal gland or conjunctiva, hor - monal stimulation of the conjunctiva, or light hyperten - sion caused by estrogen. The ophthalmic examination may be normal in some patients. Hemolacria in these cases is usually painless, may last for seconds to minu - tes, and may involve one or both eyes. The prevalence of extragenital endometriosis is unknown, and the nasolacrimal canal is an infrequent site for its appea - rance and unusual presentation. Histology is the tool for definitive diagnosis, but for some locations, it is impossible to obtain, making the cyclic character of the symptoms the way to establish diagnosis 2,8. Regarding general causes, hemolacria can be a manifestation of Henoch-Schönlein purpura due to the formation of IgA1 immune complexes in small-caliber vessels. Any scenario related to the presence or appea- rance of telangiectasias is a potential cause of hemo - lacria, such as Rendu-Osler-Weber disease, pregnancy, Raynaud’s syndrome, or various immune diseases (dermatomyositis, scleroderma, mastocytosis, and sys - temic lupus erythematosus). Hemolacria can also be found due to hypertension or the combination of diffe - rent treatments, such as anticoagulants and antiplatelet aggregators, especially aspirin and warfarin combined. More infrequent causes of this sign include Vitamin C deficiency that leads to vascular fragility or Gardner- Diamond syndrome known for ecchymotic outbreaks. There are also idiopathic cases of hemolacria, when all Rev Chil Obstet GineCOl. 2023;88(6) 396 other causes are excluded, which are estimated to be around 30% of all cases. Finally, there have been reports of psychological simulation of hemolacria, with a difficult diagnosis in these cases 3,4,7. For diagnosis, an accurate anamnesis and a systemic physical, especially ocular examination are essential, per- formed preferably by an ophthalmologist. The initial study should include complete laboratory tests with coagulation, renal and liver profiles, and factor deficiencies (VIII and von Willebrand factor antigen assays). A regurgitation test to rule out blockades as reflux of blood may suggest the presence of a lacrimal gland tumor as well as a capillary fragility test is also recommended. In recurrent cases, imaging studies may be performed such as a CT scan or an MRI of the orbits and head to rule out masses or an orbital varix. If a mass of the lacrimal gland or a conjunc- tival lesion is found, a biopsy is indicated1,3,5. The treatment of hemolacria depends on the subja - cent cause. Most cases of hemolacria are mild and progress without complications after identification and management of the triggering factor 2,4.

Anamnesis and ophthalmological examination when confronted with hemolacria are essential, and in most cases, determine the etiology. When no signs of a local cause are found, there are different studies that can be performed according to the rest of the signs or symp - toms referred to by the patient. Extragenital endometriosis is an infrequent disease, and the lacrimal glands are one of its rarest locations, with the main symptom of its diagnosis being the appea- rance of hemolacria. Biopsy is the gold standard for diagnosis, although it may be challenging, and someti - mes, the cyclic nature of hemolacria is the only tool to establish diagnosis. Treatment for endometriosis of the lacrimal gland is not defined, and in our case, it presented a favorable progression with progestin-only treatment. Acknowledgments The authors thank the patient for her consent to the publication of this case report. Funding The authors declare that they have not received funding. Conflicts of interest The authors declare no conflicts of interest. Statement of ethics The authors state that ethical approval is not required for this study in accordance with national guidelines. Written informed consent was obtained from the patient for publication of the details of their medical case and any accompanying images. Ethical disclosures Protection of human and animal subjects. The authors declare that no experiments were performed on humans or animals for this study. Confidentiality of data. The authors declare that they have followed the protocols of their work center on the publication of patient data. Right to privacy and informed consent. The authors have obtained the written informed consent of the patients or subjects mentioned in the article. The corresponding author is in possession of this document.

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