The Journal of Craniofacial Surgery
Brief Clinical Studies
anatomy if an adequate overview of the surgical site for odontosection may not be obtained. Endoscopes were recently introduced into oral and maxillofacial surgery to provide a minimally invasive approach for the removal of ectopic teeth.7 Iwai et al8 used an endoscopic approach to maxillary third molars displaced into the maxillary sinus via the socket. Moreover, the use of an endoscope enabled easy removal of roots in the anterior maxillary region without a need for a flap elevation.9 The results of the current study reveals that the mandibular third molar roots, after exposure of the furcation area, can be mobilized and removed using endoscopes without open surgery. The endoscopically assisted approach has an additional advantage of a low bone marrow hemorrhage, which facilitates a clear identification of root fragments. Compared with the difficult lateral view during conventional osteoclastic surgery of lingually displaced molars, the view angle of the endoscope can be directed toward the intraalveolar site via 30- or 70-degree scopes from various directions. The improved view allows a precise localization of roots adjacent to the mandibular canal and facilitates the mobilization with microelevators to reduce the risk for nerve injury. Because of precise surgery, complete maintenance of the buccal and lingual bone plates can be demonstrated, which practically eliminates the risk for fractures. As reported previously, there is no increased risk for sensorineuronal disturbances after endoscopically assisted procedures.4,10 This may be caused by the use of rotating diamond burs that allows a careful reduction of bone under direct vision. However, piezoelectric tools, as proposed by other authors,3 may be helpful to mobilize isolated roots. Apart from the need for special training of the surgeon, the technique presented here seems to be a valuable complementation of flapless procedures.
& Volume 24, Number 6, November 2013
10. Beltra´n V, Fuentes R, Engelke W. Endoscopic visualization of anatomic structures as a support tool in oral surgery and implantology. J Oral Maxillofac Surg 2012;70:e1Ye6
Bilateral Parotid Swelling With Blindness Harun Ucuncu, MD,* Sezgin Kurt, MD,* Elif Demirci, MD,Þ Suat Eren, MD,þ Enver Altas, MD* Abstract: We report a case of a 32-year-old woman who presented with bilateral parotid gland enlargement. She had no systemic symptoms such as fever, cough, and weight loss. She had bilateral blindness for a long time. The results of a physical examination revealed a bilateral firm, painless mass in the parotid region. A computed tomographic scan showed no clear lymphadenopathy in the neck but showed localized infiltrates and multiple pulmonary nodules with enlargement of the mediastinal and axillary lymph nodes in the chest. The patient underwent a superficial parotidectomy. A histopathologic finding revealed an epithelioid noncaseating granuloma, which is consistent with sarcoidosis. In summary, this current study shows that sarcoidosis should be considered in the differential diagnosis of all painless swelling of the parotid gland, especially in women, which could be an earlier complaint. Key Words: Sarcoidosis, parotid gland, head and neck
CONCLUSIONS Endoscopically assisted removal of lingually displaced mandibular third molars seems to be beneficial for the maintenance of surrounding bone structures. The technique allows a reduction of the surgical trauma in complex anatomic situations.
REFERENCES 1. Ward TG. The split bone technique for removal of lower third molars. Br Dent J 1956;101:297Y304 2. Rud J. The split-bone technique for removal of impacted mandibular third molars. J Oral Surg 1970;28:416Y421 3. Pippi R, Alvaro R. Piezosurgery for the lingual split technique in mandibular third molar removal: a suggestion. J Craniofac Surg 2013;24:531Y533 4. Engelke W, Bierbaum J, Choi EJ. Microsurgical removal of inferior third molars using an occlusal approach. Dtsch Zahna¨rztl Z 2011;66:415Y423 5. Silva JL, Jardim EC, dos Santos PL, et al. Comparative analysis of 2-flap designs for extraction of mandibular third molar. J Craniofac Surg 2011;22:1003Y1007 6. Kim HR, Choi BH, Engelke W, et al. A comparative study on the extractions of partially impacted mandibular third molars with or without a buccal flap: a prospective study. J Oral Maxillofac Surg 2011;69:966Y970 7. Viterbo S, Griffa A, Boffano P. Endoscopic removal of an ectopic tooth in maxillary sinus. J Craniofac Surg 2013;24:e46Ye48 8. Iwai T, Matsui Y, Hirota M, et al. Endoscopic removal of a maxillary third molar displaced into the maxillary sinus via the socket. J Craniofac Surg 2012;23:e295Ye296 9. Engelke W, Beltra´n V, Fuentes R, et al. Endoscopically assisted root splitting (EARS): method and first results. Int J Odontostomat 2012;6:313Y316
2174
S
arcoidosis involves the lungs and the intrathoracic lymph nodes in more than 90% of cases. Histologically, it is characterized by the formation of immune granulomas consisting of typical noncaseating epitheloid cells.1 Its diagnosis is based on the presence of noncaseating granulomas, concordant clinicoradiologic findings, and exclusion of other granulomatous disorders.2 Sarcoidosis may involve virtually any organ in the body. Besides intrathoracic involvement, skin lesions and extrathoracic lymph node involvement are regarded the most common.3 Head and neck involvement includes mainly cervical adenopathy, the facial skin, the eye/lacrimal gland, the cranial nerves, the tonsils, the sinonasal tract, the middle ear, the larynx, and the salivary glands.2 The lungs are often affected, and in such cases, the patient may present with a dry cough, dyspnea, or chest pain. Other common symptoms include a flulike illness with fever, night sweats, fatigue, and arthralgia. Ocular discomfort and reduced visual acuity due to uveitis may also occur.4 Sarcoidosis of the parotid gland, on the other hand, accounts for only approximately 6% of the cases and is known to be associated with bilateral parotitis. It peaks between 20 and 40 years and occurs
From the Departments of *Otorhinolaryngology, †Pathology, and ‡Radiology, Medical School, Atatu¨rk University, Erzurum, Turkey. Received May 9, 2013. Accepted for publication May 27, 2013. Address correspondence and reprint requests to Harun Ucuncu, MD, Department of Otorhinolaryngology, Medical School, Atatu¨rk University, Erzurum 25240, Turkey; E-mail: [email protected] The authors report no conflicts of interest. Copyright * 2013 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0b013e3182a2d865
* 2013 Mutaz B. Habal, MD
Copyright © 2013 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery
& Volume 24, Number 6, November 2013
Brief Clinical Studies
predominantly in females.5 The involvement of the bilateral parotid gland in sarcoidosis is rare; it should be taken into account in the differential diagnosis of all painless swelling of the gland, particularly in female patients. It may occasionally be the presenting and only manifestation of the disease.
CLINICAL REPORT A 32-year-old woman presented with bilateral parotid swelling. The results of her physical examination revealed a quite firm, semimobile, and painless mass in the bilateral parotid region. The results of the rest of the ear, nose, and throat examination were normal. The size of the mass had gradually increased last 4 to 5 years. She had no pain, tenderness, fever, or chills. The patient had bilateral blindness. She had blindness of the left eye since she was 18 years old, which turned into blindness of the right eye 2 years later. The results of an ophthalmological assessment verified optic atrophy, which could be a consequence of uveitis. She had no complaints of fatigue, fever, cough, wheeze, or night sweats. There was no evidence of lymphadenopathy in the neck. The results of a complete blood count, liver function test, chemistry panel, and urinalysis were normal. The urine calcium level was normal. Antinuclear antibody, anti-Ro, anti-La, rheumatoid factor, and antineutrophilic cytoplasmic antibodies were negative. Her serum angiotensin-converting enzyme (ACE) was high (81.8 U/L; normal level, 8Y52 U/L). A pathologic report revealed a mild chronic inflammation in lower lip biopsy. There was a bilateral enlargement in the parotid gland, more notable on the left side in magnetic resonance imaging. A contrastenhanced image revealed heterogeneous high signal intensity (Fig. 1). However, no lymphadenopathy was noted in the neck. The chest computed tomographic scan showed localized infiltrates and multiple pulmonary nodules with enlargement of the mediastinal and axillary lymph nodes. The patient underwent a superficial parotidectomy. The histopathologic finding was typical for sarcoidosis, as reflected by epitheloid noncaseating granulomas (Figs. 2 & 3). Histiocytes aggregated into well-formed tight noncaseating granulomas. Multinucleated Langhans-type giant cells were present. The granulomas may become progressively hyalinized.
FIGURE 1. A fat-suppressed T1 magnetic resonance image on axial plain. There is enlargement in the bilateral parotid gland, more notable on the left side and heterogeneous diffuse contrast.
FIGURE 2. The image shows numerous closely-packed, conglomerate nonnecrotizing granulomas effacing the architecture in a salivary gland affected by sarcoidosis (hematoxylin-eosin, original magnification 100).
At the most recent follow-up (1.5 y after the surgery), the patient neither had been under treatment and nor manifested systemic sarcoidosis.
DISCUSSION Sarcoidosis is a multisystem granulomatous disorder characterized pathologically by the presence of noncaseating granulomas in the involved organs, occurring in patients between 10 and 40 years in 70% to 90% of cases. It typically presents bilateral hilar adenopathy, pulmonary reticular opacities, and skin, joint, or eye lesions. The lung is the most common organ involved.6 Sarcoidosis is considered to be the consequence of chronic immunologic response associated with a genetic susceptibility and specific infectious or environmental factors. The granuloma formation is believed to be an inappropriate immunologic response initiated by an antigen presentation to a CD4-positive T cell. Activation of the T cells causes a rise in cytokines, which in turn activates macrophages and F-interferon production, thereby creating the granuloma.2,7 Symptoms and findings in sarcoidosis vary according to the organs affected. Sarcoidosis is a multiorgan disorder with greater than 90%
FIGURE 3. Presence of reticulin inside the granulomas (reticulin, original magnification 100).
* 2013 Mutaz B. Habal, MD
Copyright © 2013 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
2175
The Journal of Craniofacial Surgery
Brief Clinical Studies
of pulmonary involvement. The skin, the eyes, the lymph nodes, the liver, the spleen, the heart, the nervous system, and the salivary glands are often involved.2 The main targets in the head and neck region are the lymph nodes, the skin, the nose, the oral mucosa, the palatine tonsils, the larynx, the trachea, the cranial nerves, and the salivary glands.8 The diagnosis of sarcoidosis is based on the finding of noncaseating granulomas, concordant clinicoradiologic findings, and exclusion of other granulomatous disorders.2 There is no laboratory diagnostic test for sarcoidosis. Results of a laboratory evaluation may reveal an elevated erythrocyte sedimentation rate or other acute reactants. Anemia, leukopenia, and eosinophilia are commonly seen in blood counts. Hypercalcemia and/or hypercalciuria may be found.8 Serum ACE is commonly elevated in sarcoidosis. The diagnostic value of ACE is debated because its elevation is both nonspecific and insensitive. However, increased ACE is considered useful in monitoring the course of disease.1 Histologically, the nonnecrotizing inflammatory process with formation of epithelioid cell granulomas characterized of sarcoidosis can be differentiated from the histologic changes occurring in tuberculosis, Hodgkin disease, nonYHodgkin lymphoma, and especially in the Sjo¨gren syndrome.5 The differential diagnosis for bilateral parotid swelling includes masseteric hypertrophy, sialoadenosis, bulimia, acute suppurative parotitis, mumps, human immunodeficiency virus, recurrent parotitis, the Sjo¨gren syndrome, Wegener granulomatosis, sarcoidosis, the Kimura disease, polycystic parotid disease, pneumoparotid, papillary cystadenoma lymphomatosum, MALT lymphoma, and radioactive iodine.9 In the head and the neck, sarcoidosis is commonly involved with the cervical lymph nodes (40%), the globe (50%), the parotid (7%), and the larynx (6%).10 Salivary gland involvement is the most frequent in the parotids (macroscopically) and the accessory salivary glands (microscopically), in 5% and 50% of all cases, respectively.8 Enlarged parotid glands involved by sarcoidosis are bilateral, nontender, and nodular. The parotid glands are typically enlarged, with high signal intensity on T2-weighted images and enhancement on contrastenhanced images. Parotid disease may appear as multiple, benignappearing, noncavitating masses with a ‘‘foamy’’ appearance that is often associated with cervical adenopathy.10 The glands tend to be firm, only slightly painful, and they do not fluctuate in size when eating. Because the sarcoidal granulomas replace the glandular parenchyma, a moderate decrease in salivary production results.9 If a patient has bilateral parotid enlargement with uveitis and facial paralysis, the condition is referred to as the Heerfordt syndrome. This patient had bilateral parotid swelling; however, there was no facial palsy to suggest a Heerfordt syndrome. Treatment of sarcoidosis may range from observation in asymptomatic or mild disease to systemic corticosteroid therapy in more severe cases. In the treatment of parotid gland sarcoidosis, pharmacotherapy is mostly applied. Some alternative agents such azathioprine, chloroquine, hydroxychloroquine, methotrexate, chlorambucil, pentoxifylline, cyclophosphamide, tetracycline derivatives, and infliximab may be used in refractory disease or as corticosteroid-sparing agents. Some patients, especially those with isolated disease, do not require pharmacotherapy because the disease may remit spontaneously. Isolated changes in the parotid glands can also be surgically removed, and when there are no other signs of the disease, pharmacotherapy is not required.4,7,9 Our patient refused therapy; thus, routine check was suggested.
CONCLUSIONS In summary, as mentioned previously, sarcoidosis is a multisystemic disease, with head and neck involvement. In rare cases, salivary gland involvement may not be accompanied by systemic
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& Volume 24, Number 6, November 2013
symptoms. Bilateral parotid mass without systemic symptoms should be questioned for relevance of sarcoidosis by otorhinolaryngologists.
REFERENCES 1. Gulati S, Krossnes B, Olofsson J. Sinonasal involvement in sarcoidosis: a report of seven cases and review of literature. Eur Arch Otorhinolaryngol 2012;269:891Y896 2. Plaschke CC, Owen HH, Rasmussen N. Clinically isolated laryngeal sarcoidosis. Eur Arch Otorhinolaryngol 2011;268:575Y580 3. Aladesanmi OA. Sarcoidosis: an update for the primary care physician. MedGenMed 2004;6:7 4. Poate TW, Sharma R, Moutasim KA, et al. Orofacial presentations of sarcoidosisVa case series and review of the literature. Br Dent J 2008;205:437Y442 5. Fischer T, Filiminow S, Petersein J, et al. Diagnosis of Heerfordt’s syndrome by state-of-the-art ultrasound in combination with parotid biopsy: a case report. Eur Radiol 2002;12:134Y137 6. Ahmad Y, Shahril NS, Hussein H, et al. Case review of sarcoidosis resembling Sjo¨gren’s syndrome. J Clin Med Res 2010;2;284Y288 7. Kata KM, Kata D, Lange D, et al. Sarcoidosis and its otolaryngological implications. Eur Arch Otorhinolaryngol 2010;267:1507Y1514 8. Voruexakis Z, Dulguerov P, Bouayed S, et al. Sarcoidosis of the submandibular gland: a systematic review. Am J Otolaryngol 2010;31:422Y428 9. Mandel L, Surattanont F. Bilateral parotid swelling: a review. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002; 93:221Y237 10. Razek AA, Castillo M. Imaging appearance of granulomatous lesions of head and neck. Eur J Radiol 2010;76:52Y60
Long-Term Complication After Rhinoplasty Using Porous Polyethylene Implant: Cutaneous Fistula of the Forehead Nivaldo Alonso, MD, PhD,* Victor Diniz de Pochat, MD, PhD,Þ Aline Ribas Gondim de Barros,Þ Laı´s Santos TavaresÞ Abstract: Nasal dorsum augmentation is one of the most frequently performed procedures during rhinoplasty, especially in Asians and reconstructive cases. One can use autogenous cartilage grafts or alloplastic implants for this purpose. However, the potential for permanent damage to the skin and soft tissues as well as complications such as infection and extrusion of the implant make autogenous tissue augmentation preferable to alloplastic implantation. Furthermore, there is scant literature information about long-term outcomes and complications related to these implants. This brief report aimed to From the *University of Sa˜o Paulo SP, Brazil; and †Federal University of Bahia, Salvador BA, Brazil. Received June 2, 2013. Accepted for publication June 23, 2013. Address correspondence and reprint requests to Dr. Nivaldo Alonso Rua Afonso Braz, 473 cj 65 V. N. Conceic¸a˜o, Sa˜o Paulo, Brazil 04511-011; E-mail: [email protected] The authors report no conflicts of interest. Copyright * 2013 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0b013e3182a2de02
* 2013 Mutaz B. Habal, MD
Copyright © 2013 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
Brief Clinical Studies
anatomy if an adequate overview of the surgical site for odontosection may not be obtained. Endoscopes were recently introduced into oral and maxillofacial surgery to provide a minimally invasive approach for the removal of ectopic teeth.7 Iwai et al8 used an endoscopic approach to maxillary third molars displaced into the maxillary sinus via the socket. Moreover, the use of an endoscope enabled easy removal of roots in the anterior maxillary region without a need for a flap elevation.9 The results of the current study reveals that the mandibular third molar roots, after exposure of the furcation area, can be mobilized and removed using endoscopes without open surgery. The endoscopically assisted approach has an additional advantage of a low bone marrow hemorrhage, which facilitates a clear identification of root fragments. Compared with the difficult lateral view during conventional osteoclastic surgery of lingually displaced molars, the view angle of the endoscope can be directed toward the intraalveolar site via 30- or 70-degree scopes from various directions. The improved view allows a precise localization of roots adjacent to the mandibular canal and facilitates the mobilization with microelevators to reduce the risk for nerve injury. Because of precise surgery, complete maintenance of the buccal and lingual bone plates can be demonstrated, which practically eliminates the risk for fractures. As reported previously, there is no increased risk for sensorineuronal disturbances after endoscopically assisted procedures.4,10 This may be caused by the use of rotating diamond burs that allows a careful reduction of bone under direct vision. However, piezoelectric tools, as proposed by other authors,3 may be helpful to mobilize isolated roots. Apart from the need for special training of the surgeon, the technique presented here seems to be a valuable complementation of flapless procedures.
& Volume 24, Number 6, November 2013
10. Beltra´n V, Fuentes R, Engelke W. Endoscopic visualization of anatomic structures as a support tool in oral surgery and implantology. J Oral Maxillofac Surg 2012;70:e1Ye6
Bilateral Parotid Swelling With Blindness Harun Ucuncu, MD,* Sezgin Kurt, MD,* Elif Demirci, MD,Þ Suat Eren, MD,þ Enver Altas, MD* Abstract: We report a case of a 32-year-old woman who presented with bilateral parotid gland enlargement. She had no systemic symptoms such as fever, cough, and weight loss. She had bilateral blindness for a long time. The results of a physical examination revealed a bilateral firm, painless mass in the parotid region. A computed tomographic scan showed no clear lymphadenopathy in the neck but showed localized infiltrates and multiple pulmonary nodules with enlargement of the mediastinal and axillary lymph nodes in the chest. The patient underwent a superficial parotidectomy. A histopathologic finding revealed an epithelioid noncaseating granuloma, which is consistent with sarcoidosis. In summary, this current study shows that sarcoidosis should be considered in the differential diagnosis of all painless swelling of the parotid gland, especially in women, which could be an earlier complaint. Key Words: Sarcoidosis, parotid gland, head and neck
CONCLUSIONS Endoscopically assisted removal of lingually displaced mandibular third molars seems to be beneficial for the maintenance of surrounding bone structures. The technique allows a reduction of the surgical trauma in complex anatomic situations.
REFERENCES 1. Ward TG. The split bone technique for removal of lower third molars. Br Dent J 1956;101:297Y304 2. Rud J. The split-bone technique for removal of impacted mandibular third molars. J Oral Surg 1970;28:416Y421 3. Pippi R, Alvaro R. Piezosurgery for the lingual split technique in mandibular third molar removal: a suggestion. J Craniofac Surg 2013;24:531Y533 4. Engelke W, Bierbaum J, Choi EJ. Microsurgical removal of inferior third molars using an occlusal approach. Dtsch Zahna¨rztl Z 2011;66:415Y423 5. Silva JL, Jardim EC, dos Santos PL, et al. Comparative analysis of 2-flap designs for extraction of mandibular third molar. J Craniofac Surg 2011;22:1003Y1007 6. Kim HR, Choi BH, Engelke W, et al. A comparative study on the extractions of partially impacted mandibular third molars with or without a buccal flap: a prospective study. J Oral Maxillofac Surg 2011;69:966Y970 7. Viterbo S, Griffa A, Boffano P. Endoscopic removal of an ectopic tooth in maxillary sinus. J Craniofac Surg 2013;24:e46Ye48 8. Iwai T, Matsui Y, Hirota M, et al. Endoscopic removal of a maxillary third molar displaced into the maxillary sinus via the socket. J Craniofac Surg 2012;23:e295Ye296 9. Engelke W, Beltra´n V, Fuentes R, et al. Endoscopically assisted root splitting (EARS): method and first results. Int J Odontostomat 2012;6:313Y316
2174
S
arcoidosis involves the lungs and the intrathoracic lymph nodes in more than 90% of cases. Histologically, it is characterized by the formation of immune granulomas consisting of typical noncaseating epitheloid cells.1 Its diagnosis is based on the presence of noncaseating granulomas, concordant clinicoradiologic findings, and exclusion of other granulomatous disorders.2 Sarcoidosis may involve virtually any organ in the body. Besides intrathoracic involvement, skin lesions and extrathoracic lymph node involvement are regarded the most common.3 Head and neck involvement includes mainly cervical adenopathy, the facial skin, the eye/lacrimal gland, the cranial nerves, the tonsils, the sinonasal tract, the middle ear, the larynx, and the salivary glands.2 The lungs are often affected, and in such cases, the patient may present with a dry cough, dyspnea, or chest pain. Other common symptoms include a flulike illness with fever, night sweats, fatigue, and arthralgia. Ocular discomfort and reduced visual acuity due to uveitis may also occur.4 Sarcoidosis of the parotid gland, on the other hand, accounts for only approximately 6% of the cases and is known to be associated with bilateral parotitis. It peaks between 20 and 40 years and occurs
From the Departments of *Otorhinolaryngology, †Pathology, and ‡Radiology, Medical School, Atatu¨rk University, Erzurum, Turkey. Received May 9, 2013. Accepted for publication May 27, 2013. Address correspondence and reprint requests to Harun Ucuncu, MD, Department of Otorhinolaryngology, Medical School, Atatu¨rk University, Erzurum 25240, Turkey; E-mail: [email protected] The authors report no conflicts of interest. Copyright * 2013 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0b013e3182a2d865
* 2013 Mutaz B. Habal, MD
Copyright © 2013 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery
& Volume 24, Number 6, November 2013
Brief Clinical Studies
predominantly in females.5 The involvement of the bilateral parotid gland in sarcoidosis is rare; it should be taken into account in the differential diagnosis of all painless swelling of the gland, particularly in female patients. It may occasionally be the presenting and only manifestation of the disease.
CLINICAL REPORT A 32-year-old woman presented with bilateral parotid swelling. The results of her physical examination revealed a quite firm, semimobile, and painless mass in the bilateral parotid region. The results of the rest of the ear, nose, and throat examination were normal. The size of the mass had gradually increased last 4 to 5 years. She had no pain, tenderness, fever, or chills. The patient had bilateral blindness. She had blindness of the left eye since she was 18 years old, which turned into blindness of the right eye 2 years later. The results of an ophthalmological assessment verified optic atrophy, which could be a consequence of uveitis. She had no complaints of fatigue, fever, cough, wheeze, or night sweats. There was no evidence of lymphadenopathy in the neck. The results of a complete blood count, liver function test, chemistry panel, and urinalysis were normal. The urine calcium level was normal. Antinuclear antibody, anti-Ro, anti-La, rheumatoid factor, and antineutrophilic cytoplasmic antibodies were negative. Her serum angiotensin-converting enzyme (ACE) was high (81.8 U/L; normal level, 8Y52 U/L). A pathologic report revealed a mild chronic inflammation in lower lip biopsy. There was a bilateral enlargement in the parotid gland, more notable on the left side in magnetic resonance imaging. A contrastenhanced image revealed heterogeneous high signal intensity (Fig. 1). However, no lymphadenopathy was noted in the neck. The chest computed tomographic scan showed localized infiltrates and multiple pulmonary nodules with enlargement of the mediastinal and axillary lymph nodes. The patient underwent a superficial parotidectomy. The histopathologic finding was typical for sarcoidosis, as reflected by epitheloid noncaseating granulomas (Figs. 2 & 3). Histiocytes aggregated into well-formed tight noncaseating granulomas. Multinucleated Langhans-type giant cells were present. The granulomas may become progressively hyalinized.
FIGURE 1. A fat-suppressed T1 magnetic resonance image on axial plain. There is enlargement in the bilateral parotid gland, more notable on the left side and heterogeneous diffuse contrast.
FIGURE 2. The image shows numerous closely-packed, conglomerate nonnecrotizing granulomas effacing the architecture in a salivary gland affected by sarcoidosis (hematoxylin-eosin, original magnification 100).
At the most recent follow-up (1.5 y after the surgery), the patient neither had been under treatment and nor manifested systemic sarcoidosis.
DISCUSSION Sarcoidosis is a multisystem granulomatous disorder characterized pathologically by the presence of noncaseating granulomas in the involved organs, occurring in patients between 10 and 40 years in 70% to 90% of cases. It typically presents bilateral hilar adenopathy, pulmonary reticular opacities, and skin, joint, or eye lesions. The lung is the most common organ involved.6 Sarcoidosis is considered to be the consequence of chronic immunologic response associated with a genetic susceptibility and specific infectious or environmental factors. The granuloma formation is believed to be an inappropriate immunologic response initiated by an antigen presentation to a CD4-positive T cell. Activation of the T cells causes a rise in cytokines, which in turn activates macrophages and F-interferon production, thereby creating the granuloma.2,7 Symptoms and findings in sarcoidosis vary according to the organs affected. Sarcoidosis is a multiorgan disorder with greater than 90%
FIGURE 3. Presence of reticulin inside the granulomas (reticulin, original magnification 100).
* 2013 Mutaz B. Habal, MD
Copyright © 2013 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
2175
The Journal of Craniofacial Surgery
Brief Clinical Studies
of pulmonary involvement. The skin, the eyes, the lymph nodes, the liver, the spleen, the heart, the nervous system, and the salivary glands are often involved.2 The main targets in the head and neck region are the lymph nodes, the skin, the nose, the oral mucosa, the palatine tonsils, the larynx, the trachea, the cranial nerves, and the salivary glands.8 The diagnosis of sarcoidosis is based on the finding of noncaseating granulomas, concordant clinicoradiologic findings, and exclusion of other granulomatous disorders.2 There is no laboratory diagnostic test for sarcoidosis. Results of a laboratory evaluation may reveal an elevated erythrocyte sedimentation rate or other acute reactants. Anemia, leukopenia, and eosinophilia are commonly seen in blood counts. Hypercalcemia and/or hypercalciuria may be found.8 Serum ACE is commonly elevated in sarcoidosis. The diagnostic value of ACE is debated because its elevation is both nonspecific and insensitive. However, increased ACE is considered useful in monitoring the course of disease.1 Histologically, the nonnecrotizing inflammatory process with formation of epithelioid cell granulomas characterized of sarcoidosis can be differentiated from the histologic changes occurring in tuberculosis, Hodgkin disease, nonYHodgkin lymphoma, and especially in the Sjo¨gren syndrome.5 The differential diagnosis for bilateral parotid swelling includes masseteric hypertrophy, sialoadenosis, bulimia, acute suppurative parotitis, mumps, human immunodeficiency virus, recurrent parotitis, the Sjo¨gren syndrome, Wegener granulomatosis, sarcoidosis, the Kimura disease, polycystic parotid disease, pneumoparotid, papillary cystadenoma lymphomatosum, MALT lymphoma, and radioactive iodine.9 In the head and the neck, sarcoidosis is commonly involved with the cervical lymph nodes (40%), the globe (50%), the parotid (7%), and the larynx (6%).10 Salivary gland involvement is the most frequent in the parotids (macroscopically) and the accessory salivary glands (microscopically), in 5% and 50% of all cases, respectively.8 Enlarged parotid glands involved by sarcoidosis are bilateral, nontender, and nodular. The parotid glands are typically enlarged, with high signal intensity on T2-weighted images and enhancement on contrastenhanced images. Parotid disease may appear as multiple, benignappearing, noncavitating masses with a ‘‘foamy’’ appearance that is often associated with cervical adenopathy.10 The glands tend to be firm, only slightly painful, and they do not fluctuate in size when eating. Because the sarcoidal granulomas replace the glandular parenchyma, a moderate decrease in salivary production results.9 If a patient has bilateral parotid enlargement with uveitis and facial paralysis, the condition is referred to as the Heerfordt syndrome. This patient had bilateral parotid swelling; however, there was no facial palsy to suggest a Heerfordt syndrome. Treatment of sarcoidosis may range from observation in asymptomatic or mild disease to systemic corticosteroid therapy in more severe cases. In the treatment of parotid gland sarcoidosis, pharmacotherapy is mostly applied. Some alternative agents such azathioprine, chloroquine, hydroxychloroquine, methotrexate, chlorambucil, pentoxifylline, cyclophosphamide, tetracycline derivatives, and infliximab may be used in refractory disease or as corticosteroid-sparing agents. Some patients, especially those with isolated disease, do not require pharmacotherapy because the disease may remit spontaneously. Isolated changes in the parotid glands can also be surgically removed, and when there are no other signs of the disease, pharmacotherapy is not required.4,7,9 Our patient refused therapy; thus, routine check was suggested.
CONCLUSIONS In summary, as mentioned previously, sarcoidosis is a multisystemic disease, with head and neck involvement. In rare cases, salivary gland involvement may not be accompanied by systemic
2176
& Volume 24, Number 6, November 2013
symptoms. Bilateral parotid mass without systemic symptoms should be questioned for relevance of sarcoidosis by otorhinolaryngologists.
REFERENCES 1. Gulati S, Krossnes B, Olofsson J. Sinonasal involvement in sarcoidosis: a report of seven cases and review of literature. Eur Arch Otorhinolaryngol 2012;269:891Y896 2. Plaschke CC, Owen HH, Rasmussen N. Clinically isolated laryngeal sarcoidosis. Eur Arch Otorhinolaryngol 2011;268:575Y580 3. Aladesanmi OA. Sarcoidosis: an update for the primary care physician. MedGenMed 2004;6:7 4. Poate TW, Sharma R, Moutasim KA, et al. Orofacial presentations of sarcoidosisVa case series and review of the literature. Br Dent J 2008;205:437Y442 5. Fischer T, Filiminow S, Petersein J, et al. Diagnosis of Heerfordt’s syndrome by state-of-the-art ultrasound in combination with parotid biopsy: a case report. Eur Radiol 2002;12:134Y137 6. Ahmad Y, Shahril NS, Hussein H, et al. Case review of sarcoidosis resembling Sjo¨gren’s syndrome. J Clin Med Res 2010;2;284Y288 7. Kata KM, Kata D, Lange D, et al. Sarcoidosis and its otolaryngological implications. Eur Arch Otorhinolaryngol 2010;267:1507Y1514 8. Voruexakis Z, Dulguerov P, Bouayed S, et al. Sarcoidosis of the submandibular gland: a systematic review. Am J Otolaryngol 2010;31:422Y428 9. Mandel L, Surattanont F. Bilateral parotid swelling: a review. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002; 93:221Y237 10. Razek AA, Castillo M. Imaging appearance of granulomatous lesions of head and neck. Eur J Radiol 2010;76:52Y60
Long-Term Complication After Rhinoplasty Using Porous Polyethylene Implant: Cutaneous Fistula of the Forehead Nivaldo Alonso, MD, PhD,* Victor Diniz de Pochat, MD, PhD,Þ Aline Ribas Gondim de Barros,Þ Laı´s Santos TavaresÞ Abstract: Nasal dorsum augmentation is one of the most frequently performed procedures during rhinoplasty, especially in Asians and reconstructive cases. One can use autogenous cartilage grafts or alloplastic implants for this purpose. However, the potential for permanent damage to the skin and soft tissues as well as complications such as infection and extrusion of the implant make autogenous tissue augmentation preferable to alloplastic implantation. Furthermore, there is scant literature information about long-term outcomes and complications related to these implants. This brief report aimed to From the *University of Sa˜o Paulo SP, Brazil; and †Federal University of Bahia, Salvador BA, Brazil. Received June 2, 2013. Accepted for publication June 23, 2013. Address correspondence and reprint requests to Dr. Nivaldo Alonso Rua Afonso Braz, 473 cj 65 V. N. Conceic¸a˜o, Sa˜o Paulo, Brazil 04511-011; E-mail: [email protected] The authors report no conflicts of interest. Copyright * 2013 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0b013e3182a2de02
* 2013 Mutaz B. Habal, MD
Copyright © 2013 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
