8
CASE REPORTS J Oral Maxillofac Surg 69:520-527, 2011 Retrograde Hemorrhage (Hemolacria) From the Lacrimal Puncta After a Le Fort I Osteotomy: A Report of 2 Cases and a Review of the Literature Craig C. Humber, DMD, MSc,* Dennis T. Lanigan, DMD, MD,† and Frank I. Hohn, DDS‡ Acquired lacrimal obstruction caused by trauma, inflam- mation, and ablative or corrective surgeries can poten- tially occur in various anatomical regions of the nasolac- rimal system. 1-3 The incidence of a nasolacrimal apparatus injury in the nontrauma setting, however, is exceedingly low. 2 Occasionally, damage to the distal lacrimal duct apparatus has been associated with epi- phora, the continuous accumulation of tears in the lac- rimal lake, due to an impeded outflow. 1,2,4 Although the distal portion of the lacrimal apparatus is normally pro- tected within a bony framework, it has the potential to be obstructed secondarily after maxillectomies, maxil- lary and/or nasal osteotomies, midfacial fractures, and antrostomies. 4,5 The nasolacrimal obstruction that fol- lows these events normally occurs early during the post- operative phase and is typically transient and self-limit- ing. This obstruction is generally secondary to edema, which creates a temporary functional blockage to the passage of tears. Only rarely does surgical disruption of the nasolacrimal system lead to a permanent obstruc- tion, resulting in persistent epiphora and/or recurrent dacryocystitis. 2,4 Even fewer reports have discussed hemolacria, hem- orrhage from the lacrimal puncta, because of retrograde blood flow from the nasolacrimal system. 6 Although rare, hemolacria has been reported as a feature of mid- facial trauma, hereditary hemorrhagic telangiectasia, and tumors of the lacrimal apparatus. 3,7,8 While anecdotal evidence would suggest that hemolacria is commonly encountered coincident with epistaxis, only a few case reports exist in this context. 6,9,10 The anatomical basis for this phenomenon is based on the intimate connec- tion of the nasal cavity and the eye via the lacrimal apparatus. Increased intranasal pressure during epi- staxis, caused by pinching or blowing the nose, can cause a retrograde flow of blood through the nasolacri- mal apparatus and thus lead to hemolacria associated with the ipsilateral lacrimal lake. To date there have been no previously reported cases of hemolacria associated with maxillary orthog- nathic surgery. Report of Cases CASE 1 A 17-year-old male of Native American descent was seen for orthognathic surgical correction of his Class III maloc- clusion. He was an otherwise healthy individual, with no medical contraindications to surgical intervention. Clini- cal evaluation showed an obvious midfacial retrusion with associated malar deficiency and normal midfacial height. An intraoral evaluation showed a severe negative overjet and a deep overbite. The molar and canine rela- tionships displayed an Angle Class III malocclusion. On June 21, 2004, a Le Fort I osteotomy with advancement, combined with an anterior iliac crest bone graft for malar augmentation, was completed. The intraoral procedure was conducted using a routine Le Fort I osteotomy technique, with the pterygomaxillary disjunction achieved with a micro-oscil- lating saw. 11 The maxillary down fracture was somewhat dif- ficult because the patient’s bony architecture was quite dense and thick, particularly posteriorly. After redefinement of all the maxillary osteotomies, the maxilla was successfully down frac- tured and the posterior bony attachments were released. Both descending palatine arteries were noted to be intact at this point and throughout the surgical procedure. After appropri- ate occlusal splint stabilization and maxillomandibular fixation (MMF), the maxilla was rotated into position and stabilized *Chief Resident in Oral and Maxillofacial Surgery and Anesthesia, University of Toronto, Ontario, Canada. †Clinical Professor, College of Dentistry, University of Saskatche- wan, Saskatoon, Saskatchewan, Canada. ‡Department Head, Department of Dentistry/Oral and Maxillofa- cial Surgery, Saskatoon Regional Health Authority, Saskatoon, Saskatchewan, Canada. Address correspondence and reprints requests to Dr Hohn: 601 The Tower at Midtown, 201 1st Avenue South, Saskatoon, SK, Canada, S7K 1J5; e-mail: [email protected] © 2011 American Association of Oral and Maxillofacial Surgeons 0278-2391/11/6902-0030$36.00/0 doi:10.1016/j.joms.2009.12.031 520

Retrograde Hemorrhage (Hemolacria)

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Page 1: Retrograde Hemorrhage (Hemolacria)

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CASE REPORTS

Oral Maxillofac Surg9:520-527, 2011

Retrograde Hemorrhage (Hemolacria)From the Lacrimal Puncta After a

Le Fort I Osteotomy: A Report of 2Cases and a Review of the Literature

Craig C. Humber, DMD, MSc,* Dennis T. Lanigan, DMD, MD,†

and Frank I. Hohn, DDS‡

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cquired lacrimal obstruction caused by trauma, inflam-ation, and ablative or corrective surgeries can poten-

ially occur in various anatomical regions of the nasolac-imal system.1-3 The incidence of a nasolacrimalpparatus injury in the nontrauma setting, however, isxceedingly low.2 Occasionally, damage to the distalacrimal duct apparatus has been associated with epi-hora, the continuous accumulation of tears in the lac-imal lake, due to an impeded outflow.1,2,4 Although theistal portion of the lacrimal apparatus is normally pro-ected within a bony framework, it has the potential toe obstructed secondarily after maxillectomies, maxil-

ary and/or nasal osteotomies, midfacial fractures, andntrostomies.4,5 The nasolacrimal obstruction that fol-ows these events normally occurs early during the post-perative phase and is typically transient and self-limit-

ng. This obstruction is generally secondary to edema,hich creates a temporary functional blockage to theassage of tears. Only rarely does surgical disruption ofhe nasolacrimal system lead to a permanent obstruc-ion, resulting in persistent epiphora and/or recurrentacryocystitis.2,4

Even fewer reports have discussed hemolacria, hem-rrhage from the lacrimal puncta, because of retrogradelood flow from the nasolacrimal system.6 Although

*Chief Resident in Oral and Maxillofacial Surgery and Anesthesia,

niversity of Toronto, Ontario, Canada.

†Clinical Professor, College of Dentistry, University of Saskatche-

an, Saskatoon, Saskatchewan, Canada.

‡Department Head, Department of Dentistry/Oral and Maxillofa-

ial Surgery, Saskatoon Regional Health Authority, Saskatoon,

askatchewan, Canada.

Address correspondence and reprints requests to Dr Hohn: 601

he Tower at Midtown, 201 1st Avenue South, Saskatoon, SK,

anada, S7K 1J5; e-mail: [email protected]

2011 American Association of Oral and Maxillofacial Surgeons

278-2391/11/6902-0030$36.00/0

(oi:10.1016/j.joms.2009.12.031

520

are, hemolacria has been reported as a feature of mid-acial trauma, hereditary hemorrhagic telangiectasia, andumors of the lacrimal apparatus.3,7,8 While anecdotalvidence would suggest that hemolacria is commonlyncountered coincident with epistaxis, only a few caseeports exist in this context.6,9,10 The anatomical basisor this phenomenon is based on the intimate connec-ion of the nasal cavity and the eye via the lacrimalpparatus. Increased intranasal pressure during epi-taxis, caused by pinching or blowing the nose, canause a retrograde flow of blood through the nasolacri-al apparatus and thus lead to hemolacria associatedith the ipsilateral lacrimal lake.To date there have been no previously reported

ases of hemolacria associated with maxillary orthog-athic surgery.

eport of CasesCASE 1A 17-year-old male of Native American descent was seen

or orthognathic surgical correction of his Class III maloc-lusion. He was an otherwise healthy individual, with noedical contraindications to surgical intervention. Clini-

al evaluation showed an obvious midfacial retrusionith associated malar deficiency and normal midfacialeight. An intraoral evaluation showed a severe negativeverjet and a deep overbite. The molar and canine rela-ionships displayed an Angle Class III malocclusion.

On June 21, 2004, a Le Fort I osteotomy with advancement,ombined with an anterior iliac crest bone graft for malarugmentation, was completed. The intraoral procedure wasonducted using a routine Le Fort I osteotomy technique, withhe pterygomaxillary disjunction achieved with a micro-oscil-ating saw.11 The maxillary down fracture was somewhat dif-cult because the patient’s bony architecture was quite densend thick, particularly posteriorly. After redefinement of all theaxillary osteotomies, the maxilla was successfully down frac-

ured and the posterior bony attachments were released. Bothescending palatine arteries were noted to be intact at thisoint and throughout the surgical procedure. After appropri-te occlusal splint stabilization and maxillomandibular fixation

MMF), the maxilla was rotated into position and stabilized
Page 2: Retrograde Hemorrhage (Hemolacria)

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HUMBER, LANIGAN, AND HOHN 521

ith 4 miniplates and screws. The malar augmentation bonerafts were stabilized along the anterolateral wall of the maxillasing transantral 26-gauge wires. After release of MMF, thecclusion was deemed stable. There were no intraoperativeomplications.

The anesthesia record showed an atraumatic naso-endo-racheal intubation on the first attempt with a size 7.0 nasalAE endotracheal tube. The intubated naris was not speci-ed as right or left on the anesthetic record. The extubationas likewise uneventful. Controlled hypotension was useduring the general anesthetic. The estimated blood loss was00 mL for the entire procedure. Fluid resuscitation waspproximately 2,000 mL of crystalloid solutions.

The patient was initially transferred postoperatively to theostanesthesia care unit (PACU) and then to an observationnit, for monitoring on the day of his surgery. He was given 8g dexamethasone IV preoperatively, and a subsequent iden-

ical dose later that evening. Antibiotics were also given ategular intervals, initially intravenously and then orally. On therst postoperative day, the patient’s only complaint was par-sthesia associated with the second division of the tri-eminal nerve. Upon discharge, routine postoperativenstructions, Class III vector guiding elastics, feeding sy-inges, and nasal sprays were given to the patient. Dis-harge prescriptions included Amoxil suspensionGlaxoSmithKline, Research Triangle Park, NC) 500 mg 3imes a day for 7 days; Naprosyn suspension (Rocheaboratories, Mississauga, Ontario, Canada) 250 mg 4imes a day for 5 days, and Dilaudid liquid (Purdueharma LP, Stamford, CT) 2 mg every 4 hours as neededo total 30 mg.

On the eighth day postoperatively, the patient presentedor follow-up with complaints of intermittent nonbrisk ep-staxis from his right naris, retrograde hemorrhage from hisight nasolacrimal puncta, and pooling of serosanguineousecretions in his right lacrimal lake (Fig 1). There was aoderate amount of facial swelling present bilaterally, but

his was within the normal range for the postoperative timerame. The patient’s maxilla and occlusion remained stable.ue to the nonbrisk nature of his epistaxis and retrograde

acrimal hemorrhage, conservative measures including nasalrecautions (no nose blowing, air humidification, nasalprays, elevated head of the bed at 30°) and observationere used for management of these postoperative compli-

IGURE 1. Hemorrhage from the right lacrimal puncta resulting incollection of sanguineous tears associated with the right lacrimal

ake.

iumber, Lanigan, and Hohn. Retrograde Hemorrhage From theacrimal Puncta. J Oral Maxillofac Surg 2011.

ations. At 1 month postoperatively, most of the midfacialwelling had subsided and the patient did not report anyurther episodes of hemorrhage from his nares or theuncta of the nasolacrimal apparatus. There were no fur-her complications associated with this patient’s care andollow-up has remained uneventful for over 4 years.

CASE 2A 41-year-old female was admitted for orthognathic surgery

n May 20, 2008, for the correction of a mild Class III maloc-lusion secondary to a midfacial retrusion and maxillary trans-erse constriction. Clinically she exhibited a mild negativeverjet, zero overbite, and bilateral posterior crossbites. Shead no significant medical problems, except for depression forhich she was taking Celexa (Forest Pharmaceuticals, St

ouis, MO) 30 mg once a day. A 2-piece Le Fort I osteotomyith advancement, and transverse expansion via palatal splitsn both sides, was performed under general anesthesia withontrolled hypotension. The naso-endotracheal intubation wasn the right side and was straightforward and atraumatic. Thee Fort I osteotomy cuts were carried out in a standardashion. The pterygomaxillary disjunction was achievedith a curved osteotome. The maxillary down fractureas then easily accomplished. Both descending palatine

rteries were noted to be intact at this time and through-ut the procedure. Although no obvious major vesselsere severed, the patient was noted to ooze more thansual throughout the surgery. The patient was placed

nto MMF, and the maxilla was stabilized with 4iniplates and screws. The estimated blood loss during

he operation was 500 mL.The patient was transferred in stable condition immedi-

tely after surgery to the PACU for monitoring. Early onfter her transfer to the PACU, the surgeon was informedhat the patient had developed increased right-sided softissue swelling over the midface, especially around the rightye, in association with active, nonpulsatile bleeding fromhe puncta of the right eye and mild bleeding from the rightares. The surgeon placed an anterior nasal pack in the rightide of the nose and that appeared to arrest the hemorrhage.ater that evening a nurse from the observation unit calledhe surgeon to report that the patient had developed briskleeding from the right naris and a recurrence of the right-ided hemolacria. The surgeon then placed bilateral anteriorasal packs, as well as bilateral posterior nasal packs usingoley catheters. The patient was stable overnight and wasransferred to the ward the next day with the nasal packstill in situ. The nasal packs were removed later that sameay. The patient was discharged from the hospital on theecond day postoperatively. Discharge medications in-luded Keflex (Middlebrook Pharmaceuticals, Westlake,X) 250 mg 4 times a day for 7 days, dexamethasone 4 mgtimes a day for 4 days, and Tylenol (McNeil Consumerealthcare, Guelph, Ontario, Canada) with codeine elixir0 mL every 4 to 6 hours as required for pain. There wereo further episodes of epistaxis or hemolacria.

iscussion

ANATOMY OF THE NASOLACRIMALAPPARATUS AND PATHOPHYSIOLOGY OFNASOLACRIMAL OBSTRUCTION

The lacrimal drainage system is divided arbitrarily

nto proximal and distal segments. The proximal seg-
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Humber, Lanigan, and Hohn. Retrograde Hemorrhage From theLacrimal Puncta. J Oral Maxillofac Surg 2011.

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522 RETROGRADE HEMORRHAGE FROM THE LACRIMAL PUNCTA

ent includes the puncta and the canaliculi, whereashe distal segment includes the lacrimal sac and theasolacrimal duct (Fig 2). The upper punctum is 5m lateral to the medial canthus, with the lowerunctum located slightly further lateral. Both punctarain into the common canaliculus before entry intohe lacrimal sac on its posterolateral surface throughhe valve of Rosenmüeller. The sac is supportedithin a fossa of the lacrimal bone, shrouded byeriorbita, and the middle third is enveloped by the 2

imbs of the medial canthal tendon. The inferior onehird of the lacrimal sac lacks fibrous protection and isarticularly vulnerable to laceration.12 The nasolacri-al duct travels in a bony canal, in an inferior andosterior direction for 15 to 20 mm along the medialall of the antrum and empties through the valve ofasner into the inferior meatus (Fig 2). This valvesually lies 11 to 17 mm above the nasal floor and isrotected under the anterior curve of the inferiorurbinate.13 Schaeffer14,15 has noted considerableariation in the size, morphology, and location of thisrifice as it approaches the inferior meatus. Hasner’salve can differ significantly in size, and the morphol-gy of the duct orifice includes round, oval, andlit-like variants. These variants may predispose sus-eptible individuals to an increased incidence of anbstructive-type iatrogenic injury in the nasolacrimalystem and subsequent epiphora (Fig 3).16

The lacrimal “pump” theory popularized by Jonesnd Wobig17,18 has been widely discussed, even

IGURE 2. The nasolacrimal apparatus illustrating the relationshipf the medial fornix of the eye to the lateral nasal wall. Delineationeparates the proximal and distal nasolacrimal apparatus.

umber, Lanigan, and Hohn. Retrograde Hemorrhage From theacrimal Puncta. J Oral Maxillofac Surg 2011.

hough it is probably not strictly accurate. This theory

IGURE 3. (A-C) The location of the nasolacrimal apparatus withespect to the inferior turbinate and the sphenopalatine artery.asner’s valve may vary in size and location and is particularly

usceptible to iatrogenic injury if it approximates the nasal floor.

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HUMBER, LANIGAN, AND HOHN 523

uggests that the flow of tears depends on the rhyth-ic contraction around the lacrimal diaphragm with

ach blink. This mechanical action of the eyelids,analiculi, and lacrimal sac evacuates tears from theacrimal lake adjacent to the puncta. With palpebrallosure, the canaliculi shorten and the fluid within isqueezed into the lacrimal sac and then from theacrimal sac into the duct. As the eyelids open, theanaliculi re-expand and negative pressure siphonsears through the puncta. This, in turn, depends onpposition of the puncta against the globe, which isaintained by the medial canthal tendon and aug-ented by the preseptal and pretarsal portions of the

rbicularis oculi muscles during each blink. The otherrbicularis fibers split into deep and superficial heads,using with the limbs of the canthal tendon aroundhe lacrimal sac, and causing alternating compressionnd dilation of the lacrimal sac with each blink. Flows generally in the inferior direction but is based onhe patency of the ducts, and the competency, size,nd morphology of the valves. The functions ofosenmüeller’s and Hasner’s valves are complex,ith their competency varying with age, and theiratency influenced by hydrostatic pressures and vol-mes.19

Clinical examination of nasolacrimal functionould include inspection for visible trauma to theedial wall of the orbit, an assessment of the patency

f the nasolacrimal system, and a localization of theevel of obstructions via Jones I and II testing.20 Ra-iographic examination of the lacrimal drainage sys-em provides documentation of any obstruction,19

ut dye tests and probing of the proximal system withowman probes are usually sufficient to determinehe nature and location of an obstruction.2 In mostatients with posttraumatic epiphora, there is a func-ional obstruction from soft tissue edema or a nasalack. Nasolacrimal flow commonly resumes sponta-eously as the transient periorbital and nasal abnor-alities resolve.3

REVIEW OF THE LITERATURE

Ophthalmic complications associated with maxil-ary orthognathic surgery have been well documentedn the recent literature.21 Lanigan et al21 in 1993eviewed the ophthalmic complications associatedith orthognathic surgery and included a case of late

piphora after a Le Fort I osteotomy advancement.asolacrimal injuries associated with the Le Fort Isteotomy have also been reported by Little et al4 in991, and Shoshani et al22 in 1994.Demas and Sotereanos13 discussed the possibility of

njury to the nasolacrimal ductal system after Le Fortosteotomies with superior repositioning, as well as

he risk to the canaliculi and lacrimal sac during naso-

rbital osteotomies associated with Le Fort II or Le

HL

ort III procedures. In maxillary surgery, significantuperior repositioning of the maxilla may require in-erior turbinectomy and partial removal of lateral na-al walls, thereby approaching the nasolacrimal os-ium. The reported distance between the inferior

IGURE 4. A, Illustration of the lateral nasal wall showing theistance from the anterior attachment of the inferior turbinate to theasolacrimal canal orifice. B, The level of a Le Fort I osteotomyelow the level of the infraorbital foramen and the level of soft

issue elevation of the lateral nasal mucoperiosteum in relation tohe nasolacrimal duct. IT, inferior turbinate; MT, medial turbinate;

L, high Le Fort I osteotomy line; OLD, ostium of nasolacrimal duct.

umber, Lanigan, and Hohn. Retrograde Hemorrhage From theacrimal Puncta. J Oral Maxillofac Surg 2011.

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524 RETROGRADE HEMORRHAGE FROM THE LACRIMAL PUNCTA

spect of the inferior turbinate and the nasal flooranged between 5 and 10 mm. The position of theasolacrimal opening ranged from 11 to 14 mm pos-erior of the piriform rim and 11 to 17 mm above theasal floor. Demas and Sotereanos,13 however, didot report any actual cases in their experience ofasolacrimal injury or obstruction after inferior tur-inectomy and Le Fort I superior repositioning.A cadaveric study by You et al23 indicated that a Le

ort I osteotomy made below the infraorbital foramenill usually spare the nasolacrimal duct. They re-orted on the location of the nasolacrimal canal inelation to a high-level Le Fort I osteotomy. Theirimulated high-level Le Fort I osteotomy was beneathhe level of the inferior orifice of the nasolacrimalanal by a mean distance of 5.2 mm (range, 0.5 to1.5 mm) in 100 dry skull specimens (Fig 4). Further-ore, the inferior orifice of the canal was on average

.4 mm (0 to 8 mm) above, and 14.6 mm (10 to 18.5m) distal, to the anterior attachment of the inferior

urbinate (Fig 4). Their results indicate that a high-evel Le Fort I osteotomy made beneath the infraor-ital foramen and extending to the piriform rim at the

evel of the anterior attachment of the inferior turbi-ate will usually not jeopardize the distal aspect of theasolacrimal duct within its bony canal.Little et al4 reported on the potential etiologic

asis for transient epiphora after a Le Fort I osteot-my. Transient epiphora is likely related to surgicaldema creating a functional obstruction of Hasner’salve. The low incidence of permanent epiphora isnfluenced by the angulation of the bony nasolacri-

al canal and the ability of the distal lacrimal com-lex to undergo soft tissue deformation within itsony framework, while still remaining patent. Theiradaveric study concluded that the lowest point ofhe meatal opening was 16 mm above the nasaloor, and the average meatal opening ranged from.5 to 5.5 mm within the inferior meatus. Theeatal opening usually lies 30 to 35 mm behind the

ateral margin of the anterior nares, at the junctionf the anterior and middle third of the roof of the

nferior meatus. Thus, the standard Le Fort I levelsteotomy will usually be 8 to 10 mm below theeatal opening and any superior movement should

e limited by this distance. The transient nature ofpiphora after this procedure may relate to theverage interosseous width of the lacrimal fossa (4o 7 mm), the diameter of the valve of Hasner (3 to.5 mm), and the posterior-lateral inclination of theanal (15 to 25°), all of which allow a plastic de-ormation of the nasolacrimal system without nec-ssarily losing the patency of the duct.Calhoun et al5 reported on the proximity of the

asolacrimal duct to other relevant structures within

he lateral nasal wall. The nasolacrimal canal coursed s

nteriorly and superiorly from the inferior meatus, at0° to the nasal floor, into the nasolacrimal sac. Ofarticular note was the presence of thick corticalone around the duct, particularly in the proximalegion of the nasolacrimal apparatus, signifying thenherent bony protection afforded to the distal naso-acrimal apparatus as it courses through the lateralasal wall.Shoshani et al22 reported a case of a nasolacrimal

uct injury after a Le Fort I osteotomy for advance-ent and impaction of the maxilla to correct an

nterior open bite, in combination with a mandibularetback and genioplasty for treatment of severe man-ibular prognathism. Two weeks after the procedure,he patient presented with epiphora and signs ofacryocystitis. After a short unsuccessful course ofonservative measures, a subsequent dacryocystorhi-ostomy resulted in the patient becoming symptomree. The pathophysiology of the obstruction hereas likely mechanical in nature, possibly caused byucosal tears in the duct during the down fracture.natomic variations associated with Hasner’s valvend its proximity to the nasal floor were postulated toe contributing factors (Fig 3).

BLEEDING AND THE LE FORT I OSTEOTOMY

Bleeding after Le Fort I osteotomies primarily takeshe form of epistaxis, which can be anterior, poste-ior, or both.24,25 Isolated anterior epistaxis after a Leort I osteotomy may be the result of a traumaticntubation procedure or secondary to stripping theasal mucosa off the underlying nasal floor and septalreas. Brisk hemorrhage from both nares is apt to berom an injury to an artery posteriorly.24 Hemorrhagean be classified as primary, reactionary, or second-ry.25 Secondary hemorrhage generally occurs 7 to 14ays postoperatively.25 If brisk arterial bleeding oc-urs, either intraoperatively or postoperatively, it isost likely to be due to damage to the maxillary

rtery or one of its terminal branches, particularly theescending palatine or sphenopalatine arteries.24,26

he maxillary artery and its branches are the mostulnerable to damage in their course through theterygopalatine fossa during the pterygomaxillaryeparation and the maxillary down fracture.11

There has been no literature discussing potentialleeding complications related to the lateral nasalall osteotomy during a Le Fort I procedure. Theasal mucosa receives most of its blood supply fromranches of the ophthalmic artery, originating fromhe internal carotid arterial system, and the spheno-alatine artery, which is a terminal branch of theaxillary artery.27 The sphenopalatine artery arises in

he pterygopalatine fossa and passes though the sphe-opalatine foramen to supply the mucosa of the nasal

eptum and the superior, middle, and inferior turbi-
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HUMBER, LANIGAN, AND HOHN 525

ates via its 2 terminal branches, the septal artery andhe posterior lateral nasal artery (PLNA).28-30 TheLNA courses along the mucosa of the lateral nasalall in an anteroinferior direction on the perpendic-lar plate of the palatine bone and branches to supplyhe middle and inferior turbinates, and the area ap-roaching the orifice of the nasolacrimal duct. Theerminal branch of the PLNA enters the inferior tur-inate on the superior aspect of its lateral attachmentetween 10 and 15 mm from its posterior tip, where

t then branches into two. One branch remains highnd lateral, while the other runs in a lower and moreedial position. Both remain in bony canals or are in-

imate with the bone for much of the length of theurbinate. As the lower branch courses anteriorly, thealiber of the main artery narrows and forms a vascu-ar arcade at the midportion of the inferior turbinateFig 5). From the midportion, the artery progressivelyncreases in size as it passes anteriorly, suggesting thathere is a significant anterior vascular anastomosis.his may be anastomotic with the facial artery via theiriform aperture, or with other intranasal vessels. Asresult, during inferior turbinectomies, significant

rterial bleeding does not usually occur when remov-ng the anterior and middle portion of the inferiorurbinate as the caliber of the vasculature is minimalnd surgical access for hemorrhage control is great-st. Conversely, a potential for more significant arte-ial bleeding can be expected when removing theosterior and superior portion of the inferior turbi-ate, both due to poor surgical access and to a largeraliber arterial supply.30

During the Le Fort I osteotomy, the anterior lateral

IGURE 5. Arterial distribution of the inferior turbinate demonstrat-ng the distal branch of the posterior lateral nasal artery (PLNA) andhe narrow midturbinate vascular arcade.

umber, Lanigan, and Hohn. Retrograde Hemorrhage From theacrimal Puncta. J Oral Maxillofac Surg 2011.

asal wall is sectioned transantrally, ideally 3 to 4 mm n

r more above the apices of the maxillary teeth.31

ith a periosteal elevator or small malleable retractorassed subperiosteally medial to the lateral nasal wallo protect the nasal mucoperiosteum (Fig 4), thenterior aspect of the lateral nasal wall is osteoto-ized, either with a fissure bur or with a reciprocat-

ng saw. Damage to the inferior turbinate and theasal mucoperiosteum may be caused during the os-eotomy if this area is not well protected. The poste-ior portion of the lateral wall is sectioned with a thineaded osteotome until the pyramidal process of thealatine bone is encountered. Careful attention tourgical technique is paramount to ensure that thesteotome is not directed medially or superiorly, pos-ibly damaging the inferior turbinate and the nasalucoperiosteum.32 Furthermore, excessive posterior

xtension of the lateral nasal wall osteotomy mayesult in an injury to the descending palatine artery.24

ny damage to the bony framework in the superiorspect of the inferior meatus, or the inferior turbinatend the surrounding mucoperiosteum, may lead toisruption of the distal aspect of the nasolacrimalanal.23 Consequently, disruptions of the bony frame-ork of the lateral nasal wall may lead to mechanicalbstructions of the distal nasolacrimal apparatus andubsequent epiphora. If the disruptions at the lateralasal wall include damage to the distal branches ofhe PLNA as it travels through the midportion of thenferior turbinate, a low flow arterial bleed maynsue.In the first case reported in this article, the nature

f the unilateral epistaxis and hemorrhage from theacrimal puncta was fortunately self-limited and easily

anaged with conservative measures. The phenome-on accompanied a low flow epistaxis, 10 days afterLe Fort I osteotomy, which was consistent with a

econdary hemorrhage profile. The nature of theleed suggested it was likely the result of an injury tomall caliber arterial or venous vessels associated withhe nasal or turbinate mucosa, the medullary bone ofhe lateral nasal wall, or, less likely, from pterygoiduscle-related hemorrhage. The nasoendotracheal in-

ubation was atraumatic and accomplished on the firstttempt and was unlikely the cause of damage to theasal or inferior turbinate mucosa. A low-grade hem-rrhage combined with an aberrant fracture pattern

n the lateral nasal wall, and mucosal tears in theuctal system, may form a path of least resistanceesulting in retrograde hemorrhage into the nasolac-imal system, accounting for the clinical signs of he-olacria. In this case, the path of least resistance

ikely originated within the bony framework of theanal proximal to Hasner’s valve and allowed retro-rade flow into the nasolacrimal apparatus in theow-pressure “pump” system. The distal aspect of the

asolacrimal apparatus was not likely transected here
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526 RETROGRADE HEMORRHAGE FROM THE LACRIMAL PUNCTA

s the Le Fort I osteotomy was placed appropriatelyelow the infraorbital foramen, and the symptoms ofemolacria were self-limiting in nature. The fact thathe first case was a difficult down fracture because ofense thick bone posteriorly may have contributed tohe problem and increased the risk of vascular dam-ge. Despite the difficult down fracture, it is unlikelyhe main branch of the sphenopalatine artery or theescending palatine artery were severed or partiallyisrupted because the bleeding would then haveended to be more extensive and persistent. In theecond case, the epistaxis occurred in the immediateostoperative period and was brisk and recurrent inature. Although the maxillary down fracture wasasily accomplished in this patient, the pterygomax-llary disjunction had been achieved with a curvedsteotome, so aberrant fractures could have resultedhat extended to the pterygopalatine fossa area. Inhis case it is possible that the sphenopalatine artery,r a major branch of it, could have been injured basedn the nature of the bleeding. Fortunately the epi-taxis responded to anterior and posterior nasal pack-ng, and angiography and embolization were not re-uired to bring it under control.The literature associated with injuries to the naso-

acrimal apparatus during the Le Fort I osteotomyacks consistency with respect to the size, location,nd morphology of the ostium. In the literature, theres no standardization of bony landmarks from whichhe relationship of the nasolacrimal ostium is mea-ured.13-15,22,23 Functional blockages of the distal na-olacrimal canal are typically the result of localizeddema at the orifice and are usually transient andelf-limited in nature. Mechanical blockages of theistal nasolacrimal canal can be associated with aber-ant fractures within the bony framework around theanal and compounded by nasolacrimal mucosal tear-ng. Mechanical blockages may also be transient andelf-limited in nature, depending on the extent of theractures and the plasticity of the duct and surround-ng tissues. If there is a bony fracture pattern thaturpasses the threshold of self-recanalization of theuct, the nasolacrimal injury will persist, leading toossible permanent epiphora and associated compli-ations. Retrograde hemorrhage through the nasolac-imal system under low flow conditions may be seenn the event of a mechanical nasolacrimal obstruction

ith accompanied mucosal tearing of the duct prox-mal to Hasner’s valve. This is likely to represent aimilar clinical presentation as illustrated in case 1.owever, if the source of the bleed is distal to Has-er’s valve, a significant arterial hemorrhage is likely,nd the valve may become incompetent and over-helmed, resulting in a retrograde nasolacrimal hem-rrhage. Such a clinical presentation would be similar

o that illustrated in the second case and other reports

2

f hemolacria in association with severe epista-is.6,9,10 Nasolacrimal injuries after Le Fort I osteoto-ies are rare and one cannot easily predict the sus-

eptibility of an individual to such complications.ortunately, the typically transient and self-limitingature of nasolacrimal injuries and hemolacria after ae Fort I osteotomy makes its management oftenmendable to conservative management during theostoperative phase.

eferences1. Harris GJ, Fuerste FH: Lacrimal intubation in the primary repair

of midfacial fractures. Ophthalmology 94:242, 19872. Osguthorpe JD, Hoang G: Nasolacrimal injuries. Evaluation and

management. Otolaryngol Clin North Am 24:59, 19913. Becelli R, Renzi G, Mannino G, et al: Posttraumatic obstruction

of lacrimal pathways: A retrospective analysis of 58 consecu-tive naso-orbitoethmoid fractures. J Craniofac Surg 15:29, 2004

4. Little C, Mintz S, Ettinger A: The distal lacrimal duct system andtraumatic Epiphora. Int J Oral Maxillofac Surg 20:31, 1991

5. Calhoun KH, Rotzler WH, Stiernberg CM: Surgical anatomy ofthe lateral nasal wall. Otolarygnol Head Neck Surg 102:156,1990

6. Abel S: Haemorrhage from the lacrimal punctum. Br J Ophthal-mol 34:754, 1950

7. Soong HK, Pollock DA: Hereditary hemorrhagic telangiectasiadiagnosed by the ophthalmologist. Cornea 19:849, 2000

8. Sendra-Tello J, Campillo N, Rodriguez-Peralto J, et al: Malignantmelanoma of the lacrimal sac. Otolaryngol Head Neck Surg131:334, 2004

9. Wiese WF: Bloody tears, and more! An unusual case of epi-staxis. Br J Ophthalmol 87:1043, 2003

0. Banta RG, Seltzer JL: Bloody tears from epistaxis through thenasolacrimal duct. Am J Ophthalmol 75:726, 1973

1. Lanigan DT, Guest P: Alternative approaches to pterygomaxil-lary separation. Int J Oral Maxillofac Surg 22:131, 1993

2. Stranc MF: The pattern of lacrimal injuries in naso-ethmoidfractures. Br J Plast Surg 23:339, 1970

3. Demas PN, Sotereanos GC: Incidence of nasolacrimal injuryand turbinectomy associated atrophic rhinitis with Le Fort Iosteotomies. J Craniomaxillofac Surg 17:116, 1989

4. Schaeffer JP: Types of ostia nasolacramalia in man and theirgenetic significance. Am J Anat 13:183, 1912

5. Schaeffer JP: Variations in the anatomy of the nasolachrymalpassages. Ann Surg 148:148, 1911

6. Carter SR, Gausas RE: Gender and racial variations of thelacrimal system, in Cohen AJ, Mercandetti M, Brazzo BG (eds):The Lacrimal System—Diagnosis, Management and Surgery.New York, NY, Springer, 2006, p 20

7. Jones LT, Wobig JL: Congenital anomalies of the lacrimal sys-tem, in Jones LT, Wobig JL (eds): Surgery of the Eyelids andLacrimal System. Birmingham, AL, Aesculapius Publishing Co,1976, p 157

8. Jones LT: An anatomical approach to problems of the eyelidsand lacrimal apparatus. Arch Ophthalmol 66:111, 1961

9. Dutton JJ, White JJ: Imaging and clinical evaluation of thelacrimal drainage system, in Cohen AJ, Mercandetti M, BrazzoBG (eds): The Lacrimal System—Diagnosis, Management andSurgery. New York, NY, Springer, 2006, p 74

0. Amato J, Harstein ME: Evaluation of the tearing patient, inCohen AJ, Mercandetti M, Brazzo BG (eds): The Lacrimal Sys-tem—Diagnosis, Management and Surgery. New York, NY,Springer, 2006, p 66

1. Lanigan DT, Romanchuk K, Olson CK: Ophthalmic complica-tions associated with orthognathic surgery. J Oral MaxillofacSurg 51:480, 1993

2. Shoshani Y, Samet N, Ardekian L, et al: Nasolacrimal duct injuryafter Le Fort I osteotomy. J Oral Maxillofac Surg 52:406, 1994

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SIMON, DOMINIC, AND VARGHESE 527

3. You Z-H, Bell WH, Finn RA: Location of the nasolacrimal canalin relation to the high Le Fort 1 osteotomy. J Oral MaxillofacSurg 50:1075, 1992

4. Lanigan DT, West RA: Management of postoperative hemor-rhage following the Le Fort I maxillary osteotomy. J OralMaxillofac Surg 42:367, 1984

5. Lanigan DT: Hemorrhage associated with orthognathic surgery.Oral Maxillofac Surg Clin North Am 2:887, 1990

6. Lanigan D, Hay J, West R: Major vascular complications oforthognathic surgery: Hemorrhage associated with Le Fort Iosteotomies. J Oral Maxillofac Surg 48:561, 1990

7. Pothier DD, MacKeith S, Youngs R: Sphenopalatine artery liga-

tion: Technical note. J Laryngol Otol 119:810, 2005

K. George Varghese, BDS,

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8. Padgham N, Vaughan-Jones R: Cadaver studies of the anat-omy of arterial supply to the inferior turbinates. J R Soc Med84:728, 1991

9. Prades JM, Asanau A, Timoshenko AP, et al: Surgical anatomy ofthe sphenopalatine foramen and its arterial content. Surg Ra-diol Anat 30:583, 2008

0. Lee HYL, Kim H-U, Kim S-S, et al: Surgical anatomy of the spheno-palatine artery in lateral nasal wall. Laryngoscope 112:1813, 2002

1. Bell WH, Mannai C, Luhr HG: Art and science of the Le Fort Idown fracture. Int J Adult Orthodon Orthognath Surg 3:23, 1988

2. Menendez LF, Biedlingmaier JF, Tilghman D: Osteomeatal com-plex obstruction and sinusitis following Le Fort I osteotomy.

J Oral Maxillofac Surg 54:103, 1996

J Oral Maxillofac Surg69:527-531, 2011

Juxtacortical Osteogenic Sarcoma ofthe Jaws: Case Report and Review of

the LiteratureDeepti Simon, BDS, MDS, DNB,* Shiney Dominic, BDS, MDS,†

MDS, DSS (Vienna)‡

steogenic sarcoma is the most common malignantumor of bone cells, occurring in 1 of every 100,000eople.1 Approximately 7% of osteogenic sarcomasccur in the head and neck, and patients present inhe third and fourth decades of age.1 Lesions of theandible and maxilla are usually noted as bony, hard

wellings of the buccal and lingual cortices and areften associated with separation of the teeth.1 Some

esions are exophytic hard nodules on the attached gin-iva, appearing as soft tissue epulides.1 Such lesions arencommon and have been termed “juxtacortical osteo-enic sarcomas.”1

Juxtacortical osteosarcomas account for approxi-ately 5% of all osteosarcomas; they are rarely seen in

*Postdoctoral Fellow, Sree Chithra Thirunal Institute of Medical

ciences and Technology, and Senior Lecturer, Department of Oral

nd Maxillofacial Surgery, Government Dental College, Kottayam,

erala, India.

†Senior Lecturer, Department of Oral and Maxillofacial Surgery,

overnment Dental College, Kottayam, Kerala, India.

‡Professor and Head, Department of Oral and Maxillofacial Sur-

ery, Government Dental College, Kottayam, Kerala, India.

Address correspondence and reprint requests to Dr Simon: Depart-

ent of Oral and Maxillofacial Surgery, Government Dental College,

angalam, Sree Nagar A-33, Venchavode, Sreekariyam, Thiruvanantha-

uram Kerela, 695017 India; e-mail: [email protected]

2011 American Association of Oral and Maxillofacial Surgeons

278-2391/11/6902-0031$36.00/0

he jaws.2 Most are of the biologically low-gradearosteal subtype or, rarely, the high-grade periostealubtype.2 Parosteal osteosarcoma has a peak occur-ence at 39 years and commonly affects the distalemoral metaphysis. Women have a greater risk thanen (3:2) when the long bones are involved. In con-

rast, men are more often affected in the jaws. Theumor has well-defined margins and presents as aadiodense or homogenous opacity without medul-ary invasion. Atypical osteoblasts with osteoid tissueormation and foci of cartilaginous tissue are charac-eristic. Metastasis is rare. Periosteal osteosarcomaccurs at a peak age of 20 years, with a male predi-

ection (2:1). The margins are ill-defined, and theesion is radiolucent. Malignant cartilaginous tissue

ith foci of osteoid will be seen. This subtype has areater capacity for local recurrence and distant me-astasis than the parosteal subtype.3,4

We report on a case of parosteal juxtacortical os-eosarcoma of mandible in a 54-year-old woman thatresented as an epulis in the mandibular canine-pre-olar region.

ase Report

A 54-year-old woman presented to the oral and maxillofacialurgery department at the Government Dental College (Kot-ayam, Kerala, India) complaining of a 3-week history of a firmainless, gradually growing mass in the buccal aspect of theight mandibular canine-premolar region. She did not havearesthesia or mobility of her mandibular dentition. Her med-

cal history was otherwise unremarkable.