Short Reports Bloody tears and recurrent nasolacrimal duct obstruction due to a retained silicone stent Pavlina S. Kemp, MD and Richard C. Allen, MD, PhD A 6-year-old girl with a history of bilateral nasolacrimal duct obstruction presented with recurrent tearing, discharge, and bloody tears from the right eye. The patient had a history of multiple prior probing and stenting procedures on the right nasolacrimal system. Endoscopic dacryocystorhinotomy revealed a retained portion of a prior stent and accompanying pyogenic granuloma, removal of which resolved her symptoms.
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6-year-old girl with a history of bilateral nasolacrimal duct obstruction (NLDO) presented at the University of Iowa with recurrence of right-sided tearing and mucous discharge as well as intermittent bloody tears from the right eye. The patient has a history of bilateral nasolacrimal duct obstruction, with spontaneous resolution on the left side. On the right nasolacrimal system, the patient had undergone three interventions with probing and stenting with bicanalicular silicone stents; the most recent procedure was performed 7 months before the patient presented at our oculoplastics service. The prior stent removals were performed in the office; the stent was prolapsed forward between the eyelids, cut, and then pulled, with the assumption that the entire stent was removed. Concern for neoplasm of the nasolacrimal duct prompted magnetic resonance imaging (MRI) which was performed prior to presentation at our clinic (Figure 1A) and was negative for a mass in the right nasolacrimal area. The eye examination was normal. There was no palpable mass in right medial canthal area on examination. We performed a right endoscopic dacryocystorhinostomy, with placement of bicanalicular silicone stents and concurrent biopsy of the lacrimal sac tissue. Intraoperatively, gentle palpation of the area of the right nasolacrimal duct elicited bloody reflux from the right puncta (Figure 1B). Intraoperative video shows an endoscopic view above the middle turbinate of the right nares (Video 1, available at jaapos.org). After the lacrimal sac was exposed, an incision was made along the superior margin of the sac. A Bowman probe was used to explore the area via the puncta, resulting in expression of a Author affiliations: Department of Ophthalmology, University of Iowa, Iowa City Submitted September 24, 2013. Revision accepted December 8, 2013. Correspondence: Richard C. Allen, MD, PhD, Department of Ophthalmology and Visual Sciences, University of Iowa Hospitals and Clinics, 200 Hawkins Drive, Iowa City, Iowa (email: [email protected]). J AAPOS 2014;-:1-2. Copyright Ó 2014 by the American Association for Pediatric Ophthalmology and Strabismus. 1091-8531/$36.00 http://dx.doi.org/10.1016/j.jaapos.2013.12.011
Journal of AAPOS
FIG 1. A, Axial T1-weighted fat-saturated post-gadolinium magnetic resonance imaging, read as negative for mass in the right nasolacrimal duct area. B, Bloody tears occurring with gentle palpation of the right nasolacrimal sac. C, Intraoperative photograph of specimens showing retained knot from prior stent (top) and tissue confirmed on pathology to be consistent with pyogenic granuloma.
mass and a retained portion of a prior silicone stent knot (Figure 1C). These specimens were sent to pathology and the mass was determined to be consistent with pyogenic granuloma. Silicone lacrimal stents were then passed through the right upper and lower canalicular system and tied in place. Three months after dacryocystorhinostomy, the patient had no recurrence of tearing or bloody discharge, but she developed purulent discharge not improved with topical antibiotics. The stent was removed under general anesthesia via the nares 4 months after dacryocystorhinostomy due to suspicion of biofilm on the stent.
Discussion We believe that in our patient the bloody tears and recurrent nasolacrimal duct obstruction resulted from a pyogenic granuloma formed as a reaction to the retained lacrimal stent. Bloody tearing, or hemolacria, is typically a benign, unilateral, and self-limiting condition. The most common causes include disorders of the conjunctiva such as infections, lacerations, or inflammation. In her review of hemolacria, Dillivan1 noted lacrimal sac malignancies, pyogenic granuloma, orbital varix, hereditary hemorrhagic telangiectasia, anticoagulation, and coagulopathies as the most common etiologies. Neoplasms of the lacrimal drainage apparatus most commonly include epithelial carcinomas and papillomas, as well as lymphomas.2 In a retrospective chart review of 37 patients ranging in age from 21 to 90 years with tumors affecting the lacrimal drainage system, 2 presented with blood-stained tears or discharge.2 In the pediatric population, Ozcan and colleagues3 report the case of an 11-year-old girl with bilateral hemolacria accompanied by epistaxis that was
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2
Kemp and Allen
found to be secondary to hyperemia and increased vascularity of nasal cavity mucosa. Sen4 reported 4 cases of pyogenic granuloma of the palpebral conjunctiva, with 2 cases presenting with bloody tears. None of the patients had recurrence after complete surgical excision and fulgration of the base with diathermy. Yazici and colleagues5 reported 3 adults with lacrimal sac pyogenic granulomas, presenting with symptoms of hemolacria (2 cases), epiphora (all 3 cases), dacryocystitis (1 case), and lacrimal mass (1 case). All patients had nasolacrimal duct obstruction and were treated with dacryocystorhinostomy without recurrence at 11-23 months postoperatively. Generally, we believe that routine general anesthesia for bicanalicular stent removal through the nose is not necessary. If a bicanalicular stent is used, the knot should be rotated through the punctum prior to cutting the stent to ensure the entire stent is removed. Documentation of any difficulty in stent removal or uncertainty in complete removal of the stent is recommended. This case highlights the importance of tying the smallest knot possible so that it may be rotated through the punctum. Alternatively, a monocanalicular stent avoids problems with the knot altogether. Pyogenic granuloma is a benign, highly vascular tumor that characteristically bleeds with mild disturbance. The formation of these tumors may occur after trauma or minor surgery.5 Its frequency in the lacrimal sac is rare, occurring in about 0.3% of 741 lacrimal sac specimens obtained
Volume - Number - / - 2014 during dacryocystorhinostomy surgery in three separate studies.6-8
Literature Search PubMed was searched, without language or date restriction, using the following terms: bloody tears, hemolacria, nasolacrimal duct obstruction and pyogenic granuloma. References 1. Dillivan KM. Hemolacria in a patient with severe systemic diseases. Optom Vis Sci 2013;90:e161-6. 2. Valenzuela AA, McNab AA, Selva D, O’Donnell BA, Whitehead KJ, Sullivan TJ. Clinical features and management of tumors affecting the lacrimal drainage apparatus. Ophthal Plast Reconstr Surg 2006; 22:96-101. 3. Ozcan KM, Ozdas¸ T, Baran H, Ozdogan F, Dere H. Hemolacria: case report. Int J Pediatr Otorhinolaryngol 2013;77:137-8. 4. Sen DK. Granuloma pyogenicum of the palpebral conjunctiva. J Pediatr Ophthalmol Strabismus 1982;19:112-14. 5. Yazici B, Ayvaz AT, Aker S. Pyogenic granuloma of the lacrimal sac. Int Ophthalmol 2009;29:57-60. 6. Anderson NG, Wojno TH, Grossniklaus HE. Clinicopathologic findings from lacrimal sac biopsy specimens obtained during dacryocystorhinostomy. Ophthal Plast Reconstr Surg 2003;19:173-6. 7. Lee-Wing MW, Ashenhurst ME. Clinicopathologic analysis of 166 patients with primary acquired nasolacrimal duct obstruction. Ophthalmology 2001;108:2038-40. 8. Tucker N, Chow D, Stockl F, Codere F, Burnier M. Clinically suspected primary acquired nasolacrimal duct obstruction: clinicopathologic review of 150 patients. Ophthalmology 1997;104: 1882-8.
Journal of AAPOS
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6-year-old girl with a history of bilateral nasolacrimal duct obstruction (NLDO) presented at the University of Iowa with recurrence of right-sided tearing and mucous discharge as well as intermittent bloody tears from the right eye. The patient has a history of bilateral nasolacrimal duct obstruction, with spontaneous resolution on the left side. On the right nasolacrimal system, the patient had undergone three interventions with probing and stenting with bicanalicular silicone stents; the most recent procedure was performed 7 months before the patient presented at our oculoplastics service. The prior stent removals were performed in the office; the stent was prolapsed forward between the eyelids, cut, and then pulled, with the assumption that the entire stent was removed. Concern for neoplasm of the nasolacrimal duct prompted magnetic resonance imaging (MRI) which was performed prior to presentation at our clinic (Figure 1A) and was negative for a mass in the right nasolacrimal area. The eye examination was normal. There was no palpable mass in right medial canthal area on examination. We performed a right endoscopic dacryocystorhinostomy, with placement of bicanalicular silicone stents and concurrent biopsy of the lacrimal sac tissue. Intraoperatively, gentle palpation of the area of the right nasolacrimal duct elicited bloody reflux from the right puncta (Figure 1B). Intraoperative video shows an endoscopic view above the middle turbinate of the right nares (Video 1, available at jaapos.org). After the lacrimal sac was exposed, an incision was made along the superior margin of the sac. A Bowman probe was used to explore the area via the puncta, resulting in expression of a Author affiliations: Department of Ophthalmology, University of Iowa, Iowa City Submitted September 24, 2013. Revision accepted December 8, 2013. Correspondence: Richard C. Allen, MD, PhD, Department of Ophthalmology and Visual Sciences, University of Iowa Hospitals and Clinics, 200 Hawkins Drive, Iowa City, Iowa (email: [email protected]). J AAPOS 2014;-:1-2. Copyright Ó 2014 by the American Association for Pediatric Ophthalmology and Strabismus. 1091-8531/$36.00 http://dx.doi.org/10.1016/j.jaapos.2013.12.011
Journal of AAPOS
FIG 1. A, Axial T1-weighted fat-saturated post-gadolinium magnetic resonance imaging, read as negative for mass in the right nasolacrimal duct area. B, Bloody tears occurring with gentle palpation of the right nasolacrimal sac. C, Intraoperative photograph of specimens showing retained knot from prior stent (top) and tissue confirmed on pathology to be consistent with pyogenic granuloma.
mass and a retained portion of a prior silicone stent knot (Figure 1C). These specimens were sent to pathology and the mass was determined to be consistent with pyogenic granuloma. Silicone lacrimal stents were then passed through the right upper and lower canalicular system and tied in place. Three months after dacryocystorhinostomy, the patient had no recurrence of tearing or bloody discharge, but she developed purulent discharge not improved with topical antibiotics. The stent was removed under general anesthesia via the nares 4 months after dacryocystorhinostomy due to suspicion of biofilm on the stent.
Discussion We believe that in our patient the bloody tears and recurrent nasolacrimal duct obstruction resulted from a pyogenic granuloma formed as a reaction to the retained lacrimal stent. Bloody tearing, or hemolacria, is typically a benign, unilateral, and self-limiting condition. The most common causes include disorders of the conjunctiva such as infections, lacerations, or inflammation. In her review of hemolacria, Dillivan1 noted lacrimal sac malignancies, pyogenic granuloma, orbital varix, hereditary hemorrhagic telangiectasia, anticoagulation, and coagulopathies as the most common etiologies. Neoplasms of the lacrimal drainage apparatus most commonly include epithelial carcinomas and papillomas, as well as lymphomas.2 In a retrospective chart review of 37 patients ranging in age from 21 to 90 years with tumors affecting the lacrimal drainage system, 2 presented with blood-stained tears or discharge.2 In the pediatric population, Ozcan and colleagues3 report the case of an 11-year-old girl with bilateral hemolacria accompanied by epistaxis that was
1
2
Kemp and Allen
found to be secondary to hyperemia and increased vascularity of nasal cavity mucosa. Sen4 reported 4 cases of pyogenic granuloma of the palpebral conjunctiva, with 2 cases presenting with bloody tears. None of the patients had recurrence after complete surgical excision and fulgration of the base with diathermy. Yazici and colleagues5 reported 3 adults with lacrimal sac pyogenic granulomas, presenting with symptoms of hemolacria (2 cases), epiphora (all 3 cases), dacryocystitis (1 case), and lacrimal mass (1 case). All patients had nasolacrimal duct obstruction and were treated with dacryocystorhinostomy without recurrence at 11-23 months postoperatively. Generally, we believe that routine general anesthesia for bicanalicular stent removal through the nose is not necessary. If a bicanalicular stent is used, the knot should be rotated through the punctum prior to cutting the stent to ensure the entire stent is removed. Documentation of any difficulty in stent removal or uncertainty in complete removal of the stent is recommended. This case highlights the importance of tying the smallest knot possible so that it may be rotated through the punctum. Alternatively, a monocanalicular stent avoids problems with the knot altogether. Pyogenic granuloma is a benign, highly vascular tumor that characteristically bleeds with mild disturbance. The formation of these tumors may occur after trauma or minor surgery.5 Its frequency in the lacrimal sac is rare, occurring in about 0.3% of 741 lacrimal sac specimens obtained
Volume - Number - / - 2014 during dacryocystorhinostomy surgery in three separate studies.6-8
Literature Search PubMed was searched, without language or date restriction, using the following terms: bloody tears, hemolacria, nasolacrimal duct obstruction and pyogenic granuloma. References 1. Dillivan KM. Hemolacria in a patient with severe systemic diseases. Optom Vis Sci 2013;90:e161-6. 2. Valenzuela AA, McNab AA, Selva D, O’Donnell BA, Whitehead KJ, Sullivan TJ. Clinical features and management of tumors affecting the lacrimal drainage apparatus. Ophthal Plast Reconstr Surg 2006; 22:96-101. 3. Ozcan KM, Ozdas¸ T, Baran H, Ozdogan F, Dere H. Hemolacria: case report. Int J Pediatr Otorhinolaryngol 2013;77:137-8. 4. Sen DK. Granuloma pyogenicum of the palpebral conjunctiva. J Pediatr Ophthalmol Strabismus 1982;19:112-14. 5. Yazici B, Ayvaz AT, Aker S. Pyogenic granuloma of the lacrimal sac. Int Ophthalmol 2009;29:57-60. 6. Anderson NG, Wojno TH, Grossniklaus HE. Clinicopathologic findings from lacrimal sac biopsy specimens obtained during dacryocystorhinostomy. Ophthal Plast Reconstr Surg 2003;19:173-6. 7. Lee-Wing MW, Ashenhurst ME. Clinicopathologic analysis of 166 patients with primary acquired nasolacrimal duct obstruction. Ophthalmology 2001;108:2038-40. 8. Tucker N, Chow D, Stockl F, Codere F, Burnier M. Clinically suspected primary acquired nasolacrimal duct obstruction: clinicopathologic review of 150 patients. Ophthalmology 1997;104: 1882-8.
Journal of AAPOS
