YIJOM 2895 1–3
Int. J. Oral Maxillofac. Surg. 2014; xxx: xxx–xxx http://dx.doi.org/10.1016/j.ijom.2014.04.004, available online at http://www.sciencedirect.com
Case Report Head and Neck Oncology
Recurrent head and neck desmoplastic melanoma with perineural spread along the nervus mandibularis revealed 18 by F-FDG PET/CT
H. Balink1, J. G. A. M.de Visscher2, E. H. van der Meij2 1
Department of Nuclear Medicine, Medical Center Leeuwarden, Leeuwarden, The Netherlands; 2Department of Oral and Maxillofacial Surgery, Medical Center Leeuwarden, Leeuwarden, The Netherlands
H. Balink, J. G. A. M. de Visscher, E. H. van der Meij: Recurrent head and neck desmoplastic melanoma with perineural spread along the nervus mandibularis revealed by 18F-FDG PET/CT. Int. J. Oral Maxillofac. Surg. 2014; xxx: xxx–xxx. # 2014 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Abstract. This report describes a rare case of recurrent head and neck desmoplastic neurotropic melanoma with perineural spread along the nervus mandibularis. An 87-year-old male presented with a rapidly growing mass on the right side of the chin, 4 years after surgical excision of a desmoplastic non-melanotic melanoma of the tip of the chin, with lymphadenectomy of the right side submental and submandibular areas. A panoramic X-ray showed extensive widening of the mandibular canal compatible with perineural tumour growth. 18Ffluorodeoxyglucose (18F-FDG) positron emission tomography/computed tomography (PET/CT) images revealed intense pathological uptake in the recurrent tumour mass located in the right lower buccal fold, and linear pathological uptake in the mandibular canal. Although magnetic resonance imaging is considered the standard of reference, recognition of perineural spread on 18F-FDG PET/CT is important, as it usually leads to a change in patient management from cure to palliation and may avert further diagnostic procedures.
Introduction Growth away from the primary tumour site along the pathway of peripheral nerves is known as perineural spread. Perineural spread is a well-described complication of head and neck malignancies. Most cases occur in squamous cell carcinoma due to its high incidence among all 0901-5027/000001+03 $36.00/0
head and neck cancers.1 Perineural tumour spread is also particularly recognized in adenoid cystic carcinoma.2 The presence of perineural spread has a great clinical impact on patient management, regardless of the histological subtype, as it is associated with a poor prognosis and a higher risk of local recurrence and metastasis.
Keywords: perineural spread; nervus mandibularis; head and neck melanoma; 18 F-FDG PET/CT. Accepted for publication 3 April 2014
Therefore, treatment goals usually change from cure to palliation.3 Furthermore, it may not be recognized at the time of surgery and it may occur in the absence of hematogenous or lymphatic metastasis.4 Although 18F-fluorodeoxyglucose (18FFDG) positron emission tomography/
# 2014 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
Please cite this article in press as: Balink H, et al. Recurrent head and neck desmoplastic melanoma with perineural spread along the 18 nervus mandibularis revealed by F-FDG PET/CT, Int J Oral Maxillofac Surg (2014), http://dx.doi.org/10.1016/j.ijom.2014.04.004
YIJOM 2895 1–3
2
Balink et al.
computed tomography (PET/CT) is nowadays part of the initial work-up and follow-up of patients with head and neck cancer, a limited number of cases have been reported demonstrating the diagnosis of perineural spread using 18F-FDG PET/ CT.3,5,6 In this case report we describe a rare case of perineural spread of head and neck melanoma and its imaging features on 18F-FDG PET/CT. Case report
An 87-year-old male presented with a rapidly growing mass on the right side of the chin accompanied by increasing numbness of the right mental region. Four years earlier he had undergone surgical excision of a desmoplastic non-melanotic melanoma of the tip of the chin, with lymphadenectomy of the right side submental and submandibular areas. On clinical examination, a subcutaneous growing mass was palpated at the site of the previous surgery, with extension into the oral cavity. Intraoral examination revealed a multilobular mass in the lower buccal fold of the right premolar region, being an extension of this tumour. Histopathological examination of a biopsy specimen obtained intraorally confirmed the clinical suspicion of tumour recurrence. On an initially taken panoramic X-ray, extensive widening of the mandibular canal was observed, compatible with perineural tumour growth (Fig. 1). 18F-FDG PET/CT images revealed intense pathological uptake in the recurrent tumour mass located in the right lower buccal fold, and linear pathological uptake in the mandibular canal almost to the base of the skull, but without involvement of the trigeminal ganglion (Fig. 2). In this specific situation, surgical treatment was
Fig. 1. A panoramic X-ray showing extensive widening of the mandibular canal compatible with perineural tumour growth.
no longer considered to be curative. Due to the threat of tumour growth through the skin, palliative treatment with 5 4 Gy irradiation was started. The nerve trunk of the trigeminal ganglion and the skull base were not within the field of irradiation to stop the perineural spread. Discussion
‘Perineural spread’ is defined as dissemination of tumour cells along a nerve and can be visualized with imaging techniques; the term ‘perineural invasion’ is reserved for the histological diagnosis and this cannot be diagnosed with macroscopic imaging techniques.7 The cranial nerves most commonly affected by perineural spread are the trigeminal and facial nerves. The risk of perineural spread increases with a midface location of the tumour, male gender, increasing tumour size, recurrence after treatment, and poor histological differentiation.4 Although melanoma is not typically included in the category of head and neck cancer, desmoplastic (non-pigmented) melanoma in particular may develop peri-
neural invasion, especially in cases of recurrent disease.8 Of the melanoma subtypes reported, desmoplastic neurotrophic melanoma (DNM) represents approximately 1% of cases. Survival in the case of DNM may be better than that with other subtypes of melanoma. The growth pattern of this subtype may be slower than that of the conventional melanoma subtypes. However, the higher prevalence of perineural invasion increases the risk of intracranial dissemination. DNM is a rare subtype of spindle cell melanoma in which the sheets and fascicles of the spindle cells show a tendency for neurotropism.9 The pathogenesis of perineural spread is not fully understood; the collagenous perineurium creates an almost impermeable boundary between the nerve and the surrounding tissues. Perineural spread and tumour growth is stimulated by chemotactic and chemokinetic agents such as glial cell line-derived neurotrophic factor, neurotrophins, and neuron growth factor.4 Perineural spread is strongly associated with tumour expression of the neural cell adhesion molecule CD56.3,10 Not all cases of pathological FDG uptake along a nerve path are caused by
Fig. 2. Hybrid 18F-FDG PET/CT images showing the corresponding sagittal PET, hybrid PET/CT, and CT slices, with the CT slice in the ‘skeletal’ setting. Pathological uptake is observed in a linear pattern in the widened right mandibular canal, without involvement of the trigeminal ganglion.
Please cite this article in press as: Balink H, et al. Recurrent head and neck desmoplastic melanoma with perineural spread along the 18 nervus mandibularis revealed by F-FDG PET/CT, Int J Oral Maxillofac Surg (2014), http://dx.doi.org/10.1016/j.ijom.2014.04.004
YIJOM 2895 1–3
Perineural spread of desmoplastic melanoma malignancy. Perineural spread associated with invasive fungal infections such as mucormycosis and aspergillosis has been reported, but mainly affects severely individuals.10 immune-compromised Furthermore paragangliomas, sinonasal sarcoidosis, neurofibromas, and schwannomas may show a similar linear appearance of FDG uptake, albeit with different anatomical spaces involved and no perineural location on the corresponding CT slices. Meningioma may protrude through the skull base foramina, mimicking perineural spread, and benign dermatological conditions are also associated with perineural spread.3 Magnetic resonance imaging (MRI) is considered the standard of reference for the imaging evaluation of perineural spread. However, metabolic changes precede the anatomical changes; it has been reported that occult perineural spread (proven by fine-needle aspiration cytology) can be detected by 18F-FDG PET/ CT before identification on MRI.6 In conclusion, desmoplastic neurotropic melanoma of the head and neck is rare, and although MRI is considered the standard of reference, the recognition of perineural spread on 18F-FDG PET/CT is important, as it usually leads to a change in patient management from cure to palliation.
Funding
None. 6.
Competing interests
None. 7.
Patient consent
Written informed consent was obtained.
8.
References
9.
1. Carvalho AL, Nishimoto IN, Califano JA, Kowalski LP. Trends in incidence and prognosis for head and neck cancer in the United States: a site-specific analysis of the SEER database. Int J Cancer 2005;114:806–16. 2. Vrielinck LJ, Ostyn F, van Damme B, van den Bogaert W, Fossion E. The significance of perineural spread in adenoid cystic carcinoma of the major and minor salivary glands. Int J Oral Maxillofac Surg 1988;17:190–3. 3. Paes FM, Singer AD, Checkver AN, Palmquist RA, La Vega GD, Sidani C. Perineural spread in head and neck malignancies: clinical significance and evaluation with 18F-FDG PET/CT. Radiographics 2013;33:1717–36. 4. Liebig C, Ayala G, Wilks JA, Berger DH, Albo D. Perineural invasion in cancer: a review of the literature. Cancer 2009;115:3379–91. 5. Moulin-Romsee G, Benamor M, Neuenschwander S. Desmoplastic neurotropic mela-
10.
3
noma in a patient with trigeminal neuralgia: FDG PET/CT and MRI. Clin Nucl Med 2008;33:353–5. Liu SH, Kao PF, Chiu CH, Liu DW, Hsieh HJ, Chen PR. Perineural recurrence of buccal cancer demonstrated by F-18 FDG PET/ CT. Clin Nucl Med 2010;35:189–91. Ginsberg LE. Imaging of perineural tumor spread in head and neck cancer. Semin Ultrasound CT MR 1999;20:175–86. Batsakis JG, Raymond AK. Desmoplastic melanoma. Ann Otol Rhinol Laryngol 1994;103:77–9. Chang PC, Fischbein NJ, McCalmont TH, Kashani-Sabet M, Zettersten EM, Liu AY, et al. Perineural spread of malignant melanoma of the head and neck: clinical and imaging features. Am J Neuroradiol 2004;25:5–11. Gandhi D, Gujar S, Mukherji SK. Magnetic resonance imaging of perineural spread of head and neck malignancies. Top Magn Reson Imaging 2004;15:79–85.
Address: Medical Center Leeuwarden Borniastraat 34 8934 AD Leeuwarden The Netherlands Tel.: +31 58 2867488 fax: +31 58 2866139 E-mail: [email protected]
Please cite this article in press as: Balink H, et al. Recurrent head and neck desmoplastic melanoma with perineural spread along the 18 nervus mandibularis revealed by F-FDG PET/CT, Int J Oral Maxillofac Surg (2014), http://dx.doi.org/10.1016/j.ijom.2014.04.004
Int. J. Oral Maxillofac. Surg. 2014; xxx: xxx–xxx http://dx.doi.org/10.1016/j.ijom.2014.04.004, available online at http://www.sciencedirect.com
Case Report Head and Neck Oncology
Recurrent head and neck desmoplastic melanoma with perineural spread along the nervus mandibularis revealed 18 by F-FDG PET/CT
H. Balink1, J. G. A. M.de Visscher2, E. H. van der Meij2 1
Department of Nuclear Medicine, Medical Center Leeuwarden, Leeuwarden, The Netherlands; 2Department of Oral and Maxillofacial Surgery, Medical Center Leeuwarden, Leeuwarden, The Netherlands
H. Balink, J. G. A. M. de Visscher, E. H. van der Meij: Recurrent head and neck desmoplastic melanoma with perineural spread along the nervus mandibularis revealed by 18F-FDG PET/CT. Int. J. Oral Maxillofac. Surg. 2014; xxx: xxx–xxx. # 2014 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Abstract. This report describes a rare case of recurrent head and neck desmoplastic neurotropic melanoma with perineural spread along the nervus mandibularis. An 87-year-old male presented with a rapidly growing mass on the right side of the chin, 4 years after surgical excision of a desmoplastic non-melanotic melanoma of the tip of the chin, with lymphadenectomy of the right side submental and submandibular areas. A panoramic X-ray showed extensive widening of the mandibular canal compatible with perineural tumour growth. 18Ffluorodeoxyglucose (18F-FDG) positron emission tomography/computed tomography (PET/CT) images revealed intense pathological uptake in the recurrent tumour mass located in the right lower buccal fold, and linear pathological uptake in the mandibular canal. Although magnetic resonance imaging is considered the standard of reference, recognition of perineural spread on 18F-FDG PET/CT is important, as it usually leads to a change in patient management from cure to palliation and may avert further diagnostic procedures.
Introduction Growth away from the primary tumour site along the pathway of peripheral nerves is known as perineural spread. Perineural spread is a well-described complication of head and neck malignancies. Most cases occur in squamous cell carcinoma due to its high incidence among all 0901-5027/000001+03 $36.00/0
head and neck cancers.1 Perineural tumour spread is also particularly recognized in adenoid cystic carcinoma.2 The presence of perineural spread has a great clinical impact on patient management, regardless of the histological subtype, as it is associated with a poor prognosis and a higher risk of local recurrence and metastasis.
Keywords: perineural spread; nervus mandibularis; head and neck melanoma; 18 F-FDG PET/CT. Accepted for publication 3 April 2014
Therefore, treatment goals usually change from cure to palliation.3 Furthermore, it may not be recognized at the time of surgery and it may occur in the absence of hematogenous or lymphatic metastasis.4 Although 18F-fluorodeoxyglucose (18FFDG) positron emission tomography/
# 2014 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
Please cite this article in press as: Balink H, et al. Recurrent head and neck desmoplastic melanoma with perineural spread along the 18 nervus mandibularis revealed by F-FDG PET/CT, Int J Oral Maxillofac Surg (2014), http://dx.doi.org/10.1016/j.ijom.2014.04.004
YIJOM 2895 1–3
2
Balink et al.
computed tomography (PET/CT) is nowadays part of the initial work-up and follow-up of patients with head and neck cancer, a limited number of cases have been reported demonstrating the diagnosis of perineural spread using 18F-FDG PET/ CT.3,5,6 In this case report we describe a rare case of perineural spread of head and neck melanoma and its imaging features on 18F-FDG PET/CT. Case report
An 87-year-old male presented with a rapidly growing mass on the right side of the chin accompanied by increasing numbness of the right mental region. Four years earlier he had undergone surgical excision of a desmoplastic non-melanotic melanoma of the tip of the chin, with lymphadenectomy of the right side submental and submandibular areas. On clinical examination, a subcutaneous growing mass was palpated at the site of the previous surgery, with extension into the oral cavity. Intraoral examination revealed a multilobular mass in the lower buccal fold of the right premolar region, being an extension of this tumour. Histopathological examination of a biopsy specimen obtained intraorally confirmed the clinical suspicion of tumour recurrence. On an initially taken panoramic X-ray, extensive widening of the mandibular canal was observed, compatible with perineural tumour growth (Fig. 1). 18F-FDG PET/CT images revealed intense pathological uptake in the recurrent tumour mass located in the right lower buccal fold, and linear pathological uptake in the mandibular canal almost to the base of the skull, but without involvement of the trigeminal ganglion (Fig. 2). In this specific situation, surgical treatment was
Fig. 1. A panoramic X-ray showing extensive widening of the mandibular canal compatible with perineural tumour growth.
no longer considered to be curative. Due to the threat of tumour growth through the skin, palliative treatment with 5 4 Gy irradiation was started. The nerve trunk of the trigeminal ganglion and the skull base were not within the field of irradiation to stop the perineural spread. Discussion
‘Perineural spread’ is defined as dissemination of tumour cells along a nerve and can be visualized with imaging techniques; the term ‘perineural invasion’ is reserved for the histological diagnosis and this cannot be diagnosed with macroscopic imaging techniques.7 The cranial nerves most commonly affected by perineural spread are the trigeminal and facial nerves. The risk of perineural spread increases with a midface location of the tumour, male gender, increasing tumour size, recurrence after treatment, and poor histological differentiation.4 Although melanoma is not typically included in the category of head and neck cancer, desmoplastic (non-pigmented) melanoma in particular may develop peri-
neural invasion, especially in cases of recurrent disease.8 Of the melanoma subtypes reported, desmoplastic neurotrophic melanoma (DNM) represents approximately 1% of cases. Survival in the case of DNM may be better than that with other subtypes of melanoma. The growth pattern of this subtype may be slower than that of the conventional melanoma subtypes. However, the higher prevalence of perineural invasion increases the risk of intracranial dissemination. DNM is a rare subtype of spindle cell melanoma in which the sheets and fascicles of the spindle cells show a tendency for neurotropism.9 The pathogenesis of perineural spread is not fully understood; the collagenous perineurium creates an almost impermeable boundary between the nerve and the surrounding tissues. Perineural spread and tumour growth is stimulated by chemotactic and chemokinetic agents such as glial cell line-derived neurotrophic factor, neurotrophins, and neuron growth factor.4 Perineural spread is strongly associated with tumour expression of the neural cell adhesion molecule CD56.3,10 Not all cases of pathological FDG uptake along a nerve path are caused by
Fig. 2. Hybrid 18F-FDG PET/CT images showing the corresponding sagittal PET, hybrid PET/CT, and CT slices, with the CT slice in the ‘skeletal’ setting. Pathological uptake is observed in a linear pattern in the widened right mandibular canal, without involvement of the trigeminal ganglion.
Please cite this article in press as: Balink H, et al. Recurrent head and neck desmoplastic melanoma with perineural spread along the 18 nervus mandibularis revealed by F-FDG PET/CT, Int J Oral Maxillofac Surg (2014), http://dx.doi.org/10.1016/j.ijom.2014.04.004
YIJOM 2895 1–3
Perineural spread of desmoplastic melanoma malignancy. Perineural spread associated with invasive fungal infections such as mucormycosis and aspergillosis has been reported, but mainly affects severely individuals.10 immune-compromised Furthermore paragangliomas, sinonasal sarcoidosis, neurofibromas, and schwannomas may show a similar linear appearance of FDG uptake, albeit with different anatomical spaces involved and no perineural location on the corresponding CT slices. Meningioma may protrude through the skull base foramina, mimicking perineural spread, and benign dermatological conditions are also associated with perineural spread.3 Magnetic resonance imaging (MRI) is considered the standard of reference for the imaging evaluation of perineural spread. However, metabolic changes precede the anatomical changes; it has been reported that occult perineural spread (proven by fine-needle aspiration cytology) can be detected by 18F-FDG PET/ CT before identification on MRI.6 In conclusion, desmoplastic neurotropic melanoma of the head and neck is rare, and although MRI is considered the standard of reference, the recognition of perineural spread on 18F-FDG PET/CT is important, as it usually leads to a change in patient management from cure to palliation.
Funding
None. 6.
Competing interests
None. 7.
Patient consent
Written informed consent was obtained.
8.
References
9.
1. Carvalho AL, Nishimoto IN, Califano JA, Kowalski LP. Trends in incidence and prognosis for head and neck cancer in the United States: a site-specific analysis of the SEER database. Int J Cancer 2005;114:806–16. 2. Vrielinck LJ, Ostyn F, van Damme B, van den Bogaert W, Fossion E. The significance of perineural spread in adenoid cystic carcinoma of the major and minor salivary glands. Int J Oral Maxillofac Surg 1988;17:190–3. 3. Paes FM, Singer AD, Checkver AN, Palmquist RA, La Vega GD, Sidani C. Perineural spread in head and neck malignancies: clinical significance and evaluation with 18F-FDG PET/CT. Radiographics 2013;33:1717–36. 4. Liebig C, Ayala G, Wilks JA, Berger DH, Albo D. Perineural invasion in cancer: a review of the literature. Cancer 2009;115:3379–91. 5. Moulin-Romsee G, Benamor M, Neuenschwander S. Desmoplastic neurotropic mela-
10.
3
noma in a patient with trigeminal neuralgia: FDG PET/CT and MRI. Clin Nucl Med 2008;33:353–5. Liu SH, Kao PF, Chiu CH, Liu DW, Hsieh HJ, Chen PR. Perineural recurrence of buccal cancer demonstrated by F-18 FDG PET/ CT. Clin Nucl Med 2010;35:189–91. Ginsberg LE. Imaging of perineural tumor spread in head and neck cancer. Semin Ultrasound CT MR 1999;20:175–86. Batsakis JG, Raymond AK. Desmoplastic melanoma. Ann Otol Rhinol Laryngol 1994;103:77–9. Chang PC, Fischbein NJ, McCalmont TH, Kashani-Sabet M, Zettersten EM, Liu AY, et al. Perineural spread of malignant melanoma of the head and neck: clinical and imaging features. Am J Neuroradiol 2004;25:5–11. Gandhi D, Gujar S, Mukherji SK. Magnetic resonance imaging of perineural spread of head and neck malignancies. Top Magn Reson Imaging 2004;15:79–85.
Address: Medical Center Leeuwarden Borniastraat 34 8934 AD Leeuwarden The Netherlands Tel.: +31 58 2867488 fax: +31 58 2866139 E-mail: [email protected]
Please cite this article in press as: Balink H, et al. Recurrent head and neck desmoplastic melanoma with perineural spread along the 18 nervus mandibularis revealed by F-FDG PET/CT, Int J Oral Maxillofac Surg (2014), http://dx.doi.org/10.1016/j.ijom.2014.04.004
