Rare case of red tears: ocular vicarious menstruation

Abstract

Bloody tears or haemolacria is a rare clinical entity. It is caused by various ocular and systemic conditions. Haemolacria due to vicarious menstruation is even rarer. In this article, we presented a case of cyclical episodes of bloody tears coinciding with menstrual cycle in a 25-year-old married female patient. Extensive physical, ophthalmological and radiological evaluation failed to reveal other potential causes of her complaint. A diagnosis of ocular vicarious menstruation was made and she was treated with oral contraceptive pills. No such episode recurred during 3 months follow-up period.

Background

Haemolacria is an extremely rare condition and extensive evaluation is needed to exactly identify the cause of blood in tears. In some cases, a multidisciplinary discussion is needed to establish a confident diagnosis. Various aetiologies like trauma, orbital varix, bleeding disorders, infections, ocular endometriosis, medications and lastly psychiatric disorders should be considered in the differential diagnosis. Relevant clinical history, examination and investigations should be done to establish the correct diagnosis. Cyclical bleeding in extragenital organs during the normal menstrual period is referred to as vicarious menstruation.¹ In this article, we presented an interesting case of ‘bloody tears’, which used to occur periodically during menstruation in a young female. This is a rare and unusual clinical case. There is no such recent case reported in the literature. In order to understand its pathophysiology and management, more research is needed.

Case Presentation

A 25-year-old married woman came to emergency medicine department at Post Graduate Institute of Medical Education and Research (PGIMER), Chandigarh with crying red tears. That was her second such episode. First episode occurred 1 month prior to current episode, which was associated with mild nasal bleeding but she did not seek medical attention. On further enquiry, she revealed that during both the episodes, she was menstruating. Drops of blood were seen to be coming out from both the eyes (figure 1A, B). There was no injury to the eye. It was not accompanied by headache or giddiness. She did not use any local or systemic medication. There was no family history of such condition. She used to live happily with her family according to her husband. At presentation, she was haemodynamically stable. On examination by ophthalmologist, her visual acuity was 6/6 in both the eyes, her eyelids, sclera, conjunctiva, lacrimal sac area, intraocular pressure and fundus were normal. There was no ocular swelling or congested vessels in conjunctiva. Anterior and posterior segments were normal. There was no bleeding from any other site. There was no vascular lesion in the body. Systemic examination was normal. Gynaecological and Ear, Nose, Throat (ENT) evaluation were normal. Psychiatry evaluation revealed no mental illness.

Figure 1.

(A) Figure depicting bloody tears in both the eyes. (B) Figure depicting bloody tears in close outlook.

Investigations

On investigations, complete haemogram, bleeding time, clotting time, prothrombin time index and platelet count were within normal limits. Capillary fragility test was negative. von Willebrand factor antigen assay and factor VIII levels were normal. Smear from the bloody tear film revealed only red blood cells and no abnormal cell. Blood group of peripheral blood and blood in tears was found to be same (B+). Conjunctival swab was sterile. Dacrocystorhinogram was normal. CT scan of the orbits and sinus did not reveal any mass lesion or any other abnormality.

Treatment

Thus, excluding all common causes of haemolacria, ocular vicarious menstruation was diagnosed. She was started treatment with combination oestrogen–progesterone oral contraceptive pills.

Outcome and follow-up

During subsequent 3 months follow-up, no such episode recurred.

Discussion

Bleeding tears is an alarming clinical entity. It can be from various structures of the eye like conjunctiva, lacrimal gland and vascular structures. It may be uniocular or biocular. For biocular haemolacria, systemic causes should always be sought. It is known to occur in various clinical conditions. Most common among them is trauma to the eye.² Purulent conjunctivitis, acute Epstein-Barr virus infection,³ idiopathic thrombocytopenic purpura,⁴ malignant melanoma of conjunctiva,⁴ orbital varix,⁵ giant papillary conjunctivitis,⁶ hereditary haemorrhagic telangiectasis (Osler-Weber-Rendu syndrome),⁷ retrograde epistaxis,⁸ some topical medications,⁹ blood coagulation disorders,¹⁰ lacrimal gland pathology like dacryoadenitis,² lacrimal sac pathology like dacryocystitis,² various vascular lid or conjunctival lesions like cavernous haemangioma,¹¹ are important causes of it. Some cases have been reported in hysteric women.¹² Almost 30% cases are idiopathic.¹³ Another rare cause of bilateral haemolacria is vicarious menstrual bleeding.¹⁴ Vicarious menstruation is defined as cyclical bleeding in extrauterine organs during menstruation. It usually occurs between the third and fourth decades of life.¹ It is known to occur from many sources like nose, conjunctiva, eyelid, retina, ears, intestine, lungs, nipples and even skin.¹ In our patient, the fact that ocular bleeding was periodical, used to occur only during menstruation from both the eyes raised the suspicion of vicarious menstrual bleeding. Also, after hormonal treatment, there was no recurrence of such episode. Despite extensive clinical, laboratory and radiological evaluation, no other alternative cause was found.

There are very few case reports of vicarious menstruation in ocular and periocular structures in the literature. Abboud and Hanna,¹⁴ Barat and Kwedar,¹⁵ Tarasova and Briukhina¹⁶ and Sobol and Barmettler¹⁷ reported four separate cases of ocular vicarious menstruation, where exact anatomical site of bleeding could not be identified. Türkçüoğlu et al presented a case of nasolacrimal endometriosis as a cause of haemolacria.¹⁸ Dua et al reported a case of orbital endometriosis as a cause of haemolacria.¹⁹ Lee et al and Guaba et al presented two cases of subconjuctival amyloid which produced red tears periodically during menstruation.^{20 21} Even occult haemolacria in the absence of frank blood in tears has been reported. Ottovay and Norn studied 125 healthy fertile women for occult haemolacria and found blood in tears in 18% female in general and 39% female during the menstruation.²² Vicarious menstruation is considered to be due to response of vasculature to the hormones in the presence or absence of endometrial tissue at extrauterine sites, although its exact pathophysiology is not very clear. Oestrogen and progesterone can increase permeability of capillaries resulting in hyperaemia, congestion and secondary bleeding from extrauterine tissue.²³ Certain ocular and orbital tissues are seen to be hormonally responsive. Recent studies have shown that during the menstruation and menopause, cyclical variation is seen in conjunctival epithelium. Also, variation in corneal thickness and curvature have been noted during different phases of menstrual period, pregnancy and lactation.²³ Some authors believe that the cause of ocular vicarious menstruation is instability of the nervous system and not hormonal disturbances.⁴ In order to evaluate a patient for ocular vicarious menstruation, extensive workup is needed to rule out other common aetiological causes. Once the diagnosis is made, its management includes hormonal therapy as it leads to ovulatory suppression. Bleeding due to ocular or orbital endometriosis is managed by surgical resection of aberrant endometrial tissue if possible.¹⁴ Our patient also responded to the hormonal therapy. No fresh bleeding episode occurred in 3 months follow-up.

Learning points.

• Haemolacria is a rare medical condition. A multispecialty approach is needed for its evaluation.

• Ocular vicarious menstruation causing haemolacria is rarer. In a menstruating female, it should always be considered as a differential diagnosis.

• Haemolacria due to vicarious menstruation responds well to the medical management and rarely requires surgery.