0
We're unable to sign you in at this time. Please try again in a few minutes.
Retry
We were able to sign you in, but your subscription(s) could not be found. Please try again in a few minutes.
Retry
There may be a problem with your account. Please contact the AMA Service Center to resolve this issue.
Contact the AMA Service Center:
Telephone: 1 (800) 262-2350 or 1 (312) 670-7827  *   Email: subscriptions@jamanetwork.com
Error Message ......
Case Report/Case Series |

Intractable Epistaxis Due to Isolated Primary Telangiectasias FREE

Timothy Stoddard, MD, MS1; Todd A. Loehrl, MD1; Bryan C. Hunt, MD2; David M. Poetker, MD, MA1
[+] Author Affiliations
1Department of Otolaryngology and Communication Sciences, Medical College of Wisconsin, Milwaukee
2Department of Pathology, Medical College of Wisconsin, Milwaukee
JAMA Otolaryngol Head Neck Surg. 2014;140(2):160-163. doi:10.1001/jamaoto.2013.5991.
Text Size: A A A
Published online

Importance  Epistaxis is the most common otolaryngologic emergency in the United States. Most cases are controlled with first-line measures, but intractable epistaxis can be a challenging clinical problem requiring posterior nasal packing and surgical or endovascular intervention. Bleeding from nasal telangiectasias is well known in hereditary hemorrhagic telangiectasia, but there are no reports in the literature of recurrent epistaxis due to isolated telangiectasias not associated with systemic disease. This report describes a series of cases in which intractable epistaxis due to isolated primary telangiectasias was effectively controlled with bipolar electrosurgery.

Observations  We describe a patient with intractable epistaxis that had failed management with posterior packing and embolization. We also report a series of 16 cases of epistaxis, 6 of which were intractable, in adults without hereditary hemorrhagic telangiectasia who received treatment between 2009 and 2012. These cases reveal a common pattern of bleeding from telangiectasias on the anterior septum (8 cases [42%]), nasal sidewall (3 [16%]), inferior meatus (2 [10%]), posterior septum (2 [10%]), nasal floor (2 [10%]), middle turbinate (1 [5%]), and inferior turbinate (1 [5%]).

Conclusions and Relevance  Telangiectasias not associated with systemic disease are a previously unreported source of significant nasal bleeding that, when identified endoscopically, can be treated successfully with bipolar electrosurgery rather than with more invasive and costly surgical and endovascular measures.

Figures in this Article

Epistaxis is the most common otolaryngologic emergency in the United States, accounting for 1 in 200 emergency department visits annually.1 In most cases, the bleeding is easily controlled with hemostatic agents, anterior nasal packs, or chemical cautery. In the few patients who continue to bleed, more aggressive treatment with posterior nasal packing, hospital admission, and surgical or endovascular intervention is frequently required. Contemporary surgical management of posterior epistaxis using transnasal endoscopic sphenopalatine artery ligation (TESPAL) and embolization of the internal maxillary artery have comparable success rates between 80% and 90%.2

Telangiectasias are dilated, thin-walled blood vessels in the dermis and mucosal surfaces that usually present as small pink or red macular lesions with a punctate, linear, stellate, or lacy appearance.3 To date, there have been no reports describing primary telangiectasias causing significant epistaxis in patients without hereditary hemorrhagic telangiectasia (HHT).

A 67-year-old man with a history of long-term warfarin therapy after aortic valve replacement presented with a 2-week history of recurrent left-sided epistaxis that began spontaneously at rest. The patient initially went to an outside emergency department, and an inflatable nasal pack was placed in the left nasal passage. He was discharged but returned several hours later with recurrent bleeding. A Foley catheter (Bard Medical) was then inflated behind the nasal pack with adequate hemostasis.

The patient was admitted for 7 days and underwent embolization of the left sphenopalatine artery system. The packing was removed 2 days after embolization and he was discharged. Several days later, the bleeding resumed and he returned to the outside hospital; a second inflatable nasal pack was placed and he was transferred to our emergency department. His international normalized ratio determined on presentation was 4.1, and there was no active bleeding.

The patient was then taken to the operating room for endoscopic evaluation under anesthesia. A pulsatile telangiectasia was found on the left anterior nasal septum just inferior to the cribriform plate. It bled profusely with minimal manipulation and was cauterized with bipolar electrosurgery, with no further hemorrhage. No other telangiectasias or bleeding sources were identified in either nasal cavity. The patient was discharged 2 days later once his international normalized ratio was stabilized within the therapeutic range of 2.0 to 3.0, and he had no subsequent epistaxis.

Between 2009 and 2012, a total of 16 patients in our institution with no personal history, no family history, and no other features of HHT received treatment for epistaxis from a bleeding primary telangiectasia (Table 1). In 6 of these cases (38%), multiple attempts at hemorrhage control had been performed before endoscopic identification of the telangiectasia in the operating room or clinic. The patients were predominantly men (10 [62%]) aged 41 to 86 years, and 6 patients (38%) were receiving warfarin or clopidogrel for cardiac comorbidities. Only 2 patients (12%) had been using topical nasal corticosteroid sprays in the 12 months before the onset of epistaxis.

Table Graphic Jump LocationTable 1.  Case Series of Intractable Epistaxis Due to Isolated Primary Telangiectasias

The telangiectasias in this series were identified by their characteristic appearance (Figure 1). Two of the lesions were biopsied, revealing sinonasal mucosa with ectatic thin-walled vessels, confirming the diagnosis of telangiectasia (Figure 2). Telangiectasias were most commonly located on the anterior septum (8 cases [42%]), nasal sidewall (3 [16%]), inferior meatus (2 [10%]), posterior septum (2 [10%]), nasal floor (2 [10%]), middle turbinate (1 [5%]), and inferior turbinate (1 [5%]) (Table 2).

Place holder to copy figure label and caption
Figure 1.
Endoscopic Photograph of an Isolated Primary Telangiectasia on the Anterior Nasal Septum

Isolated primary telangiectasias were identified as raised red lesions containing a lacy network of blood vessels.

Graphic Jump Location
Place holder to copy figure label and caption
Figure 2.
Biopsy Section of Bleeding Lesion on Nasal Septum

Hematoxylin-eosin staining reveals reactive sinonasal epithelium with squamous metaplasia and an underlying proliferation of dilated, thin-walled vascular spaces consistent with primary telangiectasia. Original magnification ×10.

Graphic Jump Location
Table Graphic Jump LocationTable 2.  Distribution of Endonasal Telangiectasias

In 12 of the 16 patients (75%), bleeding from the telangiectasia was controlled with bipolar electrosurgery in the operating room (4 cases [25%]) or in the clinic (8 [50%]). The decision to proceed to the operating room was based on anticipated heavy bleeding in patients for whom general anesthesia was considered appropriate. Before cauterization of the telangiectasias, the nasal mucosa was anesthetized by placing cotton balls soaked in lidocaine, 4%, in the affected nasal cavity for 5 minutes. The cauterized telangiectasia was covered with an absorbable hemostatic agent, such as oxidized regenerated cellulose (Surgicel; Ethicon Inc) or gelatin granules and human thrombin (Floseal; Baxter Healthcare Corp). In one patient (6%), epistaxis had ceased on presentation to the clinic, and the identified telangiectasia was not treated; no subsequent bleeding occurred. Chemical cautery with silver nitrate was used in 4 patients (25%), but bleeding recurred 2 weeks later in one of these patients (6%) and bipolar electrosurgery was used successfully.

This case series demonstrates the challenges of managing intractable epistaxis. The case described involved 3 emergency department visits, 10 days of hospitalization, an embolization procedure, and endoscopic examination under anesthesia before the source of bleeding was identified and treated. In this case, bipolar electrosurgery was the simplest, most cost-effective therapy used, but it required precise identification of the bleeding point.

Contemporary management of intractable epistaxis usually involves TESPAL or arterial embolization. Both techniques have comparable success rates, and selecting a modality depends on patient comorbidity, institutional expertise, patient preference, use of anticoagulation, and health care costs.4 It is estimated that TESPAL costs between $6000 and $7500 compared with approximately $12 000 for arterial embolization in 2005 US dollars.5 The estimated cost of nasal endoscopy with bipolar electrosurgery in our clinic or operating room is $1900, not including associated facility and anesthesia fees.

There is disagreement in the literature about the most common locations for posterior bleeding sites in intractable epistaxis. In a prospective study6 of 50 consecutive adult cases of idiopathic posterior epistaxis, most (70%) of the bleeding sites were found on the septum, with 36% on the upper septum and 34% on the lower septum. Earlier work,7 however, reported that most posterior bleeding occurred on the lateral nasal wall, particularly in a groove of mucosa on the inferolateral aspect of the middle and inferior turbinates. The distribution and appearance of endonasal telangiectasias causing epistaxis have been described8 in patients with HHT; to our knowledge, however, no similar mapping of telangiectasias in patients without HHT who have epistaxis has been performed.

The patients in the present series demonstrated only 1 of the 4 Curaçao criteria for the diagnosis of HHT9: intractable epistaxis, multiple telangiectasias in characteristic sites (lips, oral cavity, fingers, and nose), visceral lesions, and family history. The HHT diagnosis is definite in a patient meeting 3 of the criteria but unlikely in patients with only 1 of them. Epistaxis due to endonasal telangiectasias has been reported10 in patients with the combination of calcinosis, Raynaud phenomenon, esophageal dysmotility, sclerodactyly, and telangiectasias (CREST). However, there have been no reports of epistaxis arising from endonasal telangiectasias in other systemic diseases associated with telangiectasias, such as dermatomyositis, systemic lupus erythematosus, cirrhosis, or ataxia-telangiectasia syndrome.

Topical corticosteroids have well-known local adverse effects when applied to the skin, including skin thinning, telangiectasias, striae, and bruising.11 However, the long-term administration of intranasal corticosteroids has not been associated with mucosal thinning or the formation of isolated telangiectasias.1214 It is unlikely that the 2 patients in our series who had been using nasal corticosteroids prior to the onset of epistaxis acquired telangiectasias from using the sprays. Epistaxis can be associated with the application of intranasal corticosteroids, but the mechanism for bleeding is thought to be mechanical trauma to the septal mucosa and not formation of telangiectasias.15

The successful management of epistaxis in this case series required identifying the precise bleeding sites endoscopically and applying bipolar electrosurgery to the telangiectasias. Electrosurgery appears to be more effective than silver nitrate at controlling bleeding telangiectasias, perhaps because it more effectively penetrates the mucosa to reach the numerous vessels within the telangiectasia. In addition, electrosurgery allows more precise treatment of the bleeding source with less risk of traumatizing adjacent mucosa with the inadvertent spread of silver nitrate. It is important to be vigilant for telangiectasias during nasal endoscopy, particularly those high on the anterior septum, when localizing a bleeding site in anterior or posterior epistaxis. Bipolar electrosurgery provides a relatively simple, cost-effective treatment option for the isolated bleeding telangiectasia.

Submitted for Publication: June 21, 2013; final revision received September 3, 2013; accepted September 12, 2013.

Corresponding Author: Timothy Stoddard, MD, MS, Department of Otolaryngology and Communication Sciences, Medical College of Wisconsin, 9200 W Wisconsin Ave, Milwaukee, WI 53226 (tstoddard@mcw.edu).

Published Online: December 12, 2013. doi:10.1001/jamaoto.2013.5991.

Author Contributions: Dr Stoddard had full access to all the data in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis.

Study concept and design: Loehrl, Poetker.

Acquisition of data: Stoddard, Loehrl.

Analysis and interpretation of data: Stoddard, Hunt.

Drafting of the manuscript: Stoddard.

Critical revision of the manuscript for important intellectual content: All authors.

Administrative, technical, or material support: Stoddard.

Study supervision: Loehrl, Poetker.

Conflict of Interest Disclosures: None reported.

Pallin  DJ, Chng  Y-M, McKay  MP, Emond  JA, Pelletier  AJ, Camargo  CA  Jr.  Epidemiology of epistaxis in US emergency departments, 1992 to 2001. Ann Emerg Med. 2005;46(1):77-81. doi:10.1016/j.annemergmed.2004.12.014.
PubMed   |  Link to Article
Christensen  NP, Smith  DS, Barnwell  SL, Wax  MK.  Arterial embolization in the management of posterior epistaxis. Otolaryngol Head Neck Surg. 2005;133(5):748-753. doi:10.1016/j.otohns.2005.07.041.
PubMed   |  Link to Article
Willey A, Swanson NA. Recognition and treatment of skin lesions. In: Flint P, Haughey BH, Lund VJ, eds. Cummings Otolaryngology Head and Neck Surgery. 5th ed. Philadelphia, PA: Mosby Inc; 2010:281-294.
Rudmik  L, Smith  TL.  Management of intractable spontaneous epistaxis. Am J Rhinol Allergy. 2012;26(1):55-60. doi:10.2500/ajra.2012.26.3696.
PubMed   |  Link to Article
Miller  TR, Stevens  ES, Orlandi  RR.  Economic analysis of the treatment of posterior epistaxis. Am J Rhinol. 2005;19(1):79-82.
PubMed
Chiu  TW, McGarry  GW.  Prospective clinical study of bleeding sites in idiopathic adult posterior epistaxis. Otolaryngol Head Neck Surg. 2007;137(3):390-393. doi:10.1016/j.otohns.2006.10.035.
PubMed   |  Link to Article
Thornton  MA, Mahesh  BN, Lang  J.  Posterior epistaxis: identification of common bleeding sites. Laryngoscope. 2005;115(4):588-590. doi:10.1097/01.mlg.0000161365.96685.6c.
PubMed   |  Link to Article
Folz  BJ, Wollstein  AC, Lippert  BM, Werner  JA.  Morphology and distribution of nasal telangiectasia in HHT-patients with epistaxis. Am J Rhinol. 2005;19(1):65-70.
PubMed
Shovlin  CL, Guttmacher  AE, Buscarini  E,  et al.  Diagnostic criteria for hereditary hemorrhagic telangiectasia (Rendu-Osler-Weber syndrome). Am J Med Genet. 2000;91(1):66-67. doi:10.1002/(SICI)1096-8628(20000306)91:1<66:AID-AJMG12>3.0.CO;2-P.
PubMed   |  Link to Article
Ueda  M, Abe  Y, Fujiwara  H,  et al.  Prominent telangiectasia associated with marked bleeding in CREST syndrome. J Dermatol. 1993;20(3):180-184.
PubMed
Coskey  RJ.  Adverse effects of corticosteroids, I: topical and intralesional. Clin Dermatol. 1986;4(1):155-160. doi:10.1016/0738-081X(86)90019-2.
PubMed   |  Link to Article
Minshall  E, Ghaffar  O, Cameron  L,  et al.  Assessment by nasal biopsy of long-term use of mometasone furoate aqueous nasal spray (Nasonex) in the treatment of perennial rhinitis. Otolaryngol Head Neck Surg. 1998;118(5):648-654.
PubMed
Baroody  FM, Cheng  CC, Moylan  B,  et al.  Absence of nasal mucosal atrophy with fluticasone aqueous nasal spray. Arch Otolaryngol Head Neck Surg. 2001;127(2):193-199.
PubMed   |  Link to Article
Klossek  JM, Laliberté  F, Laliberté  MF, Mounedji  N, Bousquet  J.  Local safety of intranasal triamcinolone acetonide: clinical and histological aspects of nasal mucosa in the long-term treatment of perennial allergic rhinitis. Rhinology. 2001;39(1):17-22.
PubMed
Benninger  MS, Hadley  JA, Osguthorpe  JD,  et al.  Techniques of intranasal steroid use. Otolaryngol Head Neck Surg. 2004;130(1):5-24. doi:10.1016/j.otohns.2003.10.007.
PubMed   |  Link to Article

Figures

Place holder to copy figure label and caption
Figure 1.
Endoscopic Photograph of an Isolated Primary Telangiectasia on the Anterior Nasal Septum

Isolated primary telangiectasias were identified as raised red lesions containing a lacy network of blood vessels.

Graphic Jump Location
Place holder to copy figure label and caption
Figure 2.
Biopsy Section of Bleeding Lesion on Nasal Septum

Hematoxylin-eosin staining reveals reactive sinonasal epithelium with squamous metaplasia and an underlying proliferation of dilated, thin-walled vascular spaces consistent with primary telangiectasia. Original magnification ×10.

Graphic Jump Location

Tables

Table Graphic Jump LocationTable 1.  Case Series of Intractable Epistaxis Due to Isolated Primary Telangiectasias
Table Graphic Jump LocationTable 2.  Distribution of Endonasal Telangiectasias

References

Pallin  DJ, Chng  Y-M, McKay  MP, Emond  JA, Pelletier  AJ, Camargo  CA  Jr.  Epidemiology of epistaxis in US emergency departments, 1992 to 2001. Ann Emerg Med. 2005;46(1):77-81. doi:10.1016/j.annemergmed.2004.12.014.
PubMed   |  Link to Article
Christensen  NP, Smith  DS, Barnwell  SL, Wax  MK.  Arterial embolization in the management of posterior epistaxis. Otolaryngol Head Neck Surg. 2005;133(5):748-753. doi:10.1016/j.otohns.2005.07.041.
PubMed   |  Link to Article
Willey A, Swanson NA. Recognition and treatment of skin lesions. In: Flint P, Haughey BH, Lund VJ, eds. Cummings Otolaryngology Head and Neck Surgery. 5th ed. Philadelphia, PA: Mosby Inc; 2010:281-294.
Rudmik  L, Smith  TL.  Management of intractable spontaneous epistaxis. Am J Rhinol Allergy. 2012;26(1):55-60. doi:10.2500/ajra.2012.26.3696.
PubMed   |  Link to Article
Miller  TR, Stevens  ES, Orlandi  RR.  Economic analysis of the treatment of posterior epistaxis. Am J Rhinol. 2005;19(1):79-82.
PubMed
Chiu  TW, McGarry  GW.  Prospective clinical study of bleeding sites in idiopathic adult posterior epistaxis. Otolaryngol Head Neck Surg. 2007;137(3):390-393. doi:10.1016/j.otohns.2006.10.035.
PubMed   |  Link to Article
Thornton  MA, Mahesh  BN, Lang  J.  Posterior epistaxis: identification of common bleeding sites. Laryngoscope. 2005;115(4):588-590. doi:10.1097/01.mlg.0000161365.96685.6c.
PubMed   |  Link to Article
Folz  BJ, Wollstein  AC, Lippert  BM, Werner  JA.  Morphology and distribution of nasal telangiectasia in HHT-patients with epistaxis. Am J Rhinol. 2005;19(1):65-70.
PubMed
Shovlin  CL, Guttmacher  AE, Buscarini  E,  et al.  Diagnostic criteria for hereditary hemorrhagic telangiectasia (Rendu-Osler-Weber syndrome). Am J Med Genet. 2000;91(1):66-67. doi:10.1002/(SICI)1096-8628(20000306)91:1<66:AID-AJMG12>3.0.CO;2-P.
PubMed   |  Link to Article
Ueda  M, Abe  Y, Fujiwara  H,  et al.  Prominent telangiectasia associated with marked bleeding in CREST syndrome. J Dermatol. 1993;20(3):180-184.
PubMed
Coskey  RJ.  Adverse effects of corticosteroids, I: topical and intralesional. Clin Dermatol. 1986;4(1):155-160. doi:10.1016/0738-081X(86)90019-2.
PubMed   |  Link to Article
Minshall  E, Ghaffar  O, Cameron  L,  et al.  Assessment by nasal biopsy of long-term use of mometasone furoate aqueous nasal spray (Nasonex) in the treatment of perennial rhinitis. Otolaryngol Head Neck Surg. 1998;118(5):648-654.
PubMed
Baroody  FM, Cheng  CC, Moylan  B,  et al.  Absence of nasal mucosal atrophy with fluticasone aqueous nasal spray. Arch Otolaryngol Head Neck Surg. 2001;127(2):193-199.
PubMed   |  Link to Article
Klossek  JM, Laliberté  F, Laliberté  MF, Mounedji  N, Bousquet  J.  Local safety of intranasal triamcinolone acetonide: clinical and histological aspects of nasal mucosa in the long-term treatment of perennial allergic rhinitis. Rhinology. 2001;39(1):17-22.
PubMed
Benninger  MS, Hadley  JA, Osguthorpe  JD,  et al.  Techniques of intranasal steroid use. Otolaryngol Head Neck Surg. 2004;130(1):5-24. doi:10.1016/j.otohns.2003.10.007.
PubMed   |  Link to Article

Correspondence

CME
Also Meets CME requirements for:
Browse CME for all U.S. States
Accreditation Information
The American Medical Association is accredited by the Accreditation Council for Continuing Medical Education to provide continuing medical education for physicians. The AMA designates this journal-based CME activity for a maximum of 1 AMA PRA Category 1 CreditTM per course. Physicians should claim only the credit commensurate with the extent of their participation in the activity. Physicians who complete the CME course and score at least 80% correct on the quiz are eligible for AMA PRA Category 1 CreditTM.
Note: You must get at least of the answers correct to pass this quiz.
Please click the checkbox indicating that you have read the full article in order to submit your answers.
Your answers have been saved for later.
You have not filled in all the answers to complete this quiz
The following questions were not answered:
Sorry, you have unsuccessfully completed this CME quiz with a score of
The following questions were not answered correctly:
Commitment to Change (optional):
Indicate what change(s) you will implement in your practice, if any, based on this CME course.
Your quiz results:
The filled radio buttons indicate your responses. The preferred responses are highlighted
For CME Course: A Proposed Model for Initial Assessment and Management of Acute Heart Failure Syndromes
Indicate what changes(s) you will implement in your practice, if any, based on this CME course.
Submit a Comment

Multimedia

Some tools below are only available to our subscribers or users with an online account.

Related Content

Customize your page view by dragging & repositioning the boxes below.

Articles Related By Topic
Related Collections
PubMed Articles