Nasal Alar Necrosis. John D. Rachel, MD; Robert H. Mathog, MD

Transcription

1 The Laryngoscope Lippincott Williams & Wilkins, Inc., Philadelphia 2000 The American Laryngological, Rhinological and Otological Society, Inc. Nasal Alar Necrosis John D. Rachel, MD; Robert H. Mathog, MD Objective: To describe an unrecognized clinical entity, nasal alar necrosis, and propose recommendations regarding the diagnosis, pathophysiology, and management of these cases. Study Design: Retrospective review of four patients with this condition. Methods: Histories, treatments, and outcomes were evaluated using office and hospital chart data. Results: We noted that three patients had prior trauma or surgery, other than to the nose, in the head and neck region. All four patients had comorbidities such as diabetes, hypothyroidism, depression, or tobacco abuse. Three patients had sensory deficits over the distribution of the maxillary nerve, and three volunteered that they had a habit of picking the crusted wound. Two patients improved over several months with aggressive wound care. One patient refused treatment and another underwent successful reconstruction. Conclusions: After malignant and granulomatous diseases were ruled out, our evaluations suggested that the pathogenesis was multifactorial including several factors alone or in combination, such as, hypoesthesia, self-mutilation, and an inadequate blood supply. Deficits in vascularity and sensory innervation must be considered as potential obstacles in reconstruction. Psychological problems causing factitious wounding may complicate therapeutic interventions. Key Words: Nasal alar necrosis, pathophysiology, diagnosis, management. Laryngoscope, 110: , 2000 INTRODUCTION Nasal alar necrosis is a rare condition resulting in a cosmetic defect. Although necrosis can occur after prolonged nasotracheal intubation and rhinoplasty, a progressive destructive process can also occur with no other obvious causes. The literature contains several case reports suggesting a multifocal etiology including the loss of nerve function to the affected area. 1,2 This paucity of information prompts us to report on four patients presenting with this condition with reference to the pathophysiology, outcomes, and treatments available. Presented at the Meeting of the Middle Section of the American Laryngological, Rhinological and Otological Society, Inc., Cincinnati, Ohio, January 22, From the Department of Otolaryngology Head and Neck Surgery, Wayne State University, Detroit, Michigan. Editor s Note: This Manuscript was accepted for publication May 22, Send Correspondence to Robert H. Mathog, MD, Department of Otolaryngology Head and Neck Surgery, 5E-UHC, 540 East Canfield Avenue, Detroit, MI 48201, U.S.A. MATERIALS AND METHODS Histories, treatments, and outcomes were evaluated retrospectively using office and hospital chart data. A literature search of 30 years was completed to analyze previously reported causes and management of the condition. Inclusion criteria were alar necrosis of unknown cause. Any patients with direct injury to the nose or known destructive processes were excluded (i.e.,: gunshot wounds, knife wounds, animal bites, tumors, or granulomatous disorders). RESULTS The patients in this series had necrosis of one of the ala nasi. Table I describes the historical background of the four patients in this series. Three patients had either prior trauma or surgery, other than to the nose, in the head and neck region. All four patients had comorbidities such as diabetes, hypertension, hypothyroidism, depression, or a history of tobacco abuse. Three patients had a sensory loss over the distribution of the maxillary division of the trigeminal nerve and three patients volunteered that they had a habit of picking the crusted area. Case 1 This 39-year-old woman was involved in a motor vehicle accident in August 1992 and sustained multiple injuries (Fig. 1). She had a history of depression, hypothyroidism, and tobacco use, but denied a history of intravenous drug abuse. After injury she complained of left-sided facial pain. She had multiple nerve blocks, but when these failed she underwent a trigeminal rhizotomy. This procedure was successful in providing complete anesthesia over the distribution of the nerve and she experienced total remission of pain. Three months later a small lesion developed in the left nasal ala. Biopsy revealed focal ulceration, epidermal hyperplasia, and hyperkeratosis with no evidence of malignancy or granulomatous disease. She admitted to frequent manual manipulation of the site secondary to the constant irritation. The patient was treated with aggressive wound care and was asked to avoid picking at the ulcer, but the lesion progressed to necrosis of the entire ala. Recommendations for reconstruction of the defect were refused and the patient continued to suffer from a persistent ulcer at that site. Case 2 This 48-year-old man with a history of diabetes mellitus, hypertension, depression, and tobacco use developed mucormycosis of the sinuses in April 1993 (Fig. 2). He 1437

2 TABLE I. Patient Background. Patient No. Condition Sensory loss Y Y Y N Picking habit Y Y Y N Diabetes N Y N Y Hypertension N Y N Y Hypothyroidism Y N N N Depression Y Y Y Y Tobacco use Y Y N Y underwent debridement of the right maxillary and ethmoid sinuses with decompression of the optic nerve, followed by amphotericin-b. Sensation over the distribution of the maxillary nerve and vision from the right eye were lost. In October 1996 he returned to the operating room for treatment of a frontal sinus mucocele with a frontal sinus obliteration. In May 1997 the patient presented to the office after a 4-mm ulceration developed in the right ala. His wife stated that the patient was frequently picking at Fig. 2. Case 2. Anterior view of remaining right nasal alar defect. the site, but he denied the habit. He agreed to a wound care regimen and avoided further digital manipulation of the site. The ulceration healed, leaving a defect of the alar rim. Case 3 This 43-year-old female was assaulted with a gun shot wound to the face in March 1986 (Fig. 3). She suffered blindness bilaterally with numbness of the right face. The patient presented in October 1996 with ulceration of the nose that had persisted for several years. Examination revealed a lesion of the right ala with crusting along the caudal septum and scarring of the upper right lip. She also admitted to digital manipulation. The ulcerated area improved with application of bacitracin ointment and daily saline douches over several months. She was well healed with a depressed ala at the time of this writing. Fig. 1. Case 1. Lateral view of left nasal alar necrosis with focal ulceration and erythema Case 4 This 55-year-old man with a history of diabetes mellitus, hypertension, and depression presented in June 1993 with an 18-month history of a nonhealing left-sided nasal ulcer (Fig. 4). He denied any history of surgery or trauma in the head and neck region and denied a picking habit. A sensory examination of the face was within normal limits. Examination of the nose demonstrated neartotal erosion of the nasal ala with exophytic granulation

3 Fig. 3. Case 3. Ulcerated right nasal sill lesion. Fig. 5. Case 4. Intraoperative reconstructive photograph of stage 1. tissue along the borders of the lesion. Biopsy results were negative for malignancy or granulomatous disease. He underwent a two-stage reconstruction with a nasolabial flap and septal cartilage graft with subsequent debulking of the flap in August 1993 (Fig. 5). Fig. 4. Case 4. Oblique view. Preoperative photograph demonstrating a deep full-thickness defect. DISCUSSION Necrosis of the nasal ala is a rare phenomenon that occurs after invasive treatment for trigeminal neuralgia. 1,2 In a case reported by Nusem-Horowitz et al., 1 such a lesion occurred 3 weeks after retrogasserian alcohol injection for treatment of facial pain. The mechanism was thought to be a delayed avascular necrosis of the ala nasi as a result of either damage to the sympathetic plexus accompanying the feeding vessels or a distal microembolic effect during the injection. In a similar case, Srinivas et al. 2 noted erosion after this procedure and attributed it to a picking habit. Others reported a similar lesion of neuroparalytic keratitis after corneal anesthesia from interruption of trigeminal pathways and minor trauma. 3,4 Our series also included causal elements of external trauma, factitious self-induced destruction of tissue, and loss of sensation. In one of our patients (described in case 1) an alar lesion developed within 3 months of the onset of facial anesthesia. This then progressed to necrosis of the entire alar subunit. She admitted to frequent manipulation and refused treatment of the lesion, despite its nonhealing nature and cosmetic deformity. The patient described in case 2 denied any manipulation of the site, but his wife stated that he was constantly picking at the ulcer. The patient in case 3 admitted to occasionally touching the wound, which she stopped with the onset of treatment. The lesions described in cases 1, 2, and 3 were also related to numbness. In case 4, the patient had normal sensation of the face and no history of trauma or prior surgery. None of our patients had overt evidence of a reduced blood supply (avascular necrosis) to the side of the face. Factitious or self-induced injury was prevalent in our patients. It has been suggested that the skin is highly vulnerable to self-inflicted lesions because it is accessible and serves as an important medium of communication between the individual and the social environment. 5,6 Disorders of wound healing may be manifestations of psychoneuroses. Various personality disorders are associated with factitious skin lesions and can range from the sane but dishonest to the severely disturbed. Munchausen s syndrome is a psychiatric disturbance associated with factitious wounding in which the intrapsychic gains are a 1439

4 result of a poorly adapted response to stress in an unbalanced personality. Zohar et al. 7 describe cases in which damage to the external auditory canal, nose, and cheeks was self-inflicted or inflicted by parents (Munchausen s syndrome by proxy) with the goal of obtaining medical attention. Once the diagnosis of a self-inflicted wound is entertained, it is difficult to confirm unless the patient is observed traumatizing the wound or acknowledges involvement. None of our patients appeared to have this type of personality or evidence of self-mutilation, although the underlying disorder is difficult to diagnose and treat. Another not-so-obvious cause that must be considered is avascular necrosis as a result of disrupted blood flow. The vascular anatomy has been well described in the literature In cadaver dissections Toriumi et al. 8 demonstrated that most specimens have an independent blood supply to each side of the nose that varies from one side to the other. The superficial arterial supply to the external structures of the nose is derived from both the internal and the external carotid arteries. An external branch of the ophthalmic artery, the dorsal nasal artery, runs downward along the lateral surface of the nose and is supplemented by branches of the infraorbital artery. The external nasal branch of the anterior ethmoid artery courses over the dorsal surface of the nasal bones and proceeds to the nasal tip, where it contributes to the arterial arcade of the nasal tip. The lateral nasal artery branches off the angular artery and passes medially along the cephalic margin of the of the lateral crura, giving off branches that course in a caudal direction over the lateral crura toward the nostril rim. The artery then passes medially over the domes and continues down the columella to the base of the nose. The columellar arteries branch off the facial artery or the superior labial artery and meet the lateral nasal artery over the dome of the nose to form an alar arcade that runs along the cephalic margin of the lateral crura. In most cases the lateral nasal artery feeds the alar arcade from above and the columellar artery from below. A subdermal plexus is found superficial to the alar arcade and receives contributions from the lateral nasal, anterior ethmoid, and columellar arteries. The sympathetic control to these blood vessels of the external nose originates as preganglionic fibers in the thoracolumbar region of the spinal cord, which pass into the vagosympathetic trunk to relay in the superior cervical ganglion. The postganglionic sympathetic fibers follow the internal carotid artery and reach the nose via the ethmoidal and infraorbital branches of the trigeminal nerve as well as the inferior lateral nasal branch of the greater palatine nerve. 11 With the loss of blood flow, tissue is susceptible to ischemia and necrosis. In our series we believe the patients had an intact vascular anatomy or would have adequate collateral contributions from the feeding vessels in this region to supply the nasal ala and surrounding tissues. Three patients did not receive injections in or near the vessels, which rules out the possibility of a distal microembolic effect. Avascular necrosis as a result of impaired blood flow autoregulation by the sympathetic system must be considered because there was trauma to these nerves in three of our patients The loss of sensation also appears contributory. Sensory innervation to the external nose is derived from the ophthalmic and maxillary division of the trigeminal nerve. The ophthalmic nerve gives rise to the nasociliary nerve, which in turn branches into the anterior and posterior ethmoidal nerves. These fibers follow their respective arteries and also the infratrochlear nerve, providing distribution to the eyelids and the skin of the upper part of the side of the nose. The infraorbital nerve, a branch of the maxillary division of the fifth cranial nerve, supplies the lateral nasal wall. The loss of innervation to the ala would render the region anesthetic and even minor trauma could go unrealized and cause ulceration with tissue loss. The potential for a diminished blood supply, coupled with persistent irritation, may contribute to the development and chronicity of these lesions. Reconstructive strategies for the ala vary with the depth and location of the defect. 12,13 The complexity and challenge of the repair generally escalates with the loss of additional tissue layers that provide support and lining. Nasal alar defects that extend into deep soft tissue or approach the alar rim can lead to collapse of the alar rim, producing a functional as well as a cosmetic deficit. Various techniques have been developed for reconstruction of a defect involving the nasal ala In two of the four patients in this series, a superficial or partialthickness defect was evident. In these defects, healing by secondary intention provided an acceptable result. This treatment requires meticulous wound care that maintains a moist wound environment free of infection. The other two patients presented with deep fullthickness defects requiring replacement of mucosa, supporting cartilage, and overlying skin. In case 4, the inner lining was created using a turnover cutaneous flap; support was provided with septal cartilage and coverage by a nasolabial flap. The patient underwent revision of the flap at a later date. In case 1 the patient had a large fullthickness defect. She was presented with a reconstruction plan using a nasolabial flap for internal lining and a forehead flap for external lining. The patient, however, refused surgery and the defect remains under some control with local wound care. CONCLUSION Nasal alar necrosis developed in the patients in this series as a complicating condition of an underlying disease or an inciting event. Patients with nasal alar necrosis can provide a diagnostic and reconstructive challenge. Defects in vascularity and sensory innervation must be considered as causes of the condition and obstacles in reconstruction. Moreover, psychological problems causing factitious wounding may complicate preventative and therapeutic interventions. BIBLIOGRAPHY 1. 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