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Dorsal Extradural Melanotic Schwannoma AL-AZHAR ASSIUT MEDICAL JOURNAL AAMJ ,VOL 13 , NO 3 , JULY 2015 DORSAL EXTRADURAL MELANOTIC SCHWANNOMA: A CASE REPORT AND REVIEW OF LITERATURE Bokhary Mahmoud*, Ahmad Adel* and Hatem Elkhouly** Department of Neurosurgery, *Armed Force Hospital Southern Region, Saudi Arabia, and **Al Azhar University, Egypt. ـــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــــ ABSTRACT Melanotic schwannoma is a rare variants of schwannomas usually described as a case reports only with the largest series contain five cases. It is accounting for less than 1% of all nerve sheath tumors, with a predilection for spinal nerve involveSment. However, they can occur in other areas, such as cerebellum, acoustic nerve, sympathetic chain, heart, orbit, oral cavity, esophagus, stomach, bronchus, retroperitonium, and parotid gland. It is composed of Schwann cells and melanin pigment with 50% of the cases have psammomatous calcifications and this type of melanotic schwannoma is related to Carney complex with autosomal dominant inheritance, characterized by the association of cutaneous pigmentation, fibromyxoid tumors of the skin, myxoma of the heart, and endocrine overactivity. Most cases of melanotic schwannoma are benign, though 10% of them are malignant with metastatic potential. It is necessary to differentiate these tumors from metastatic malignant melanomas, which have a bad prognosis. There are a few theories about the etiopathogenesis of these tumors, including melanomatous transformation of Schwann neoplastic cells and phagocytosis of melanin by Schwann cells. Melanotic schwannoma has nonspecific clinical presentation and Magnetic resonance imaging is the most widely used test for the diagnosis, revealing hyperintense T1-weighted sequences and hypointense T2-weighted sequences. Total excision of this lesion is recommended and diagnostic confirmation is obtained by histological and immunohistochemical studies. KEYWORDS: Schwannoma, Melanotic Schwannoma, Spinal Tumors. INTRODUCTION arise sporadically or as part of Carney Melanotic schwannoma was first complex in association with other lesions described by Millar in 1932 as a (3). malignant melanotic tumor of ganglion There are a few theories about the cells, which was subsequently termed etiopathogenesis of these tumors, melanocytic schwannoma in 1975 by Fu including melanomatous transformation et al (1). Their occurrence is described in of Schwann neoplastic cells and case reports only, the largest series phagocytosis of melanin by Schwann consists of five cases (2). cells (4). It accounting for less than 1% of all nerve The melanocytic schwannoma occurs at sheath tumors, with a predilection for younger ages compared to other spinal nerve involvement, but they can schwannomas (2), and spinal melanotic occur in other locations, such as the schwannoma arises in the lumbosacral sympathetic chain, acoustic nerve, region (47.2%) and thoracic (30.5%) and cerebellum, orbit, choroid, soft tissues, cervical (22.2%) levels and rarely heart, oral cavity, esophageal wall, intramedullary types are seen (5). stomach, bronchus, retroperitonium, It usually, presents with features of uterine cervix, and parotid gland. They compression of spinal cord or exiting 366 | P a g e Bokhary Mahmoud et al AAMJ ,VOL 13 , NO 3 , JULY 2015 nerve roots and Magnetic resonance similar condition and he has no history of imaging (MRI) is the most commonly other symptoms suggesting Carney used image diagnosis test, revealing complex disease. hyperintense T1 and hypointense T2 By examination of this patient there was images, whereas in schwannomas of the spastic paraparesis with sensory level at non-melanocytic type, the image pattern D6 with extensor planter bilaterally and is the opposite (6). with scissoring gait. By review of literature, there are few The patient submitted for investigations cases of melanocytic type schwannoma in and Dorsal MRI with and without contrast the spine and due to its rarity; we report a done (Figure 1 and 2), which showed a case of melanocytic schwannoma of the well-circumscribed oval shaped extra dorsal spine with literature review. dural lesion at level of D5-D6 that extends through and expands the left CASE REPORT neural foramen with appearance of Our case is a male patient, 40 years old, dumbbell-shape, the lesion is slightly without relevant medical history hyperintense signal on T1 WIs, and iso presented with one year duration of intense signal on T2 WIs while the gradual onset of weakness of both lower capsule of the lesion exhibits low T2 limbs without sphincteric troubles. He signal. After contrast administration; the has no history of fever or trauma. Also lesion displays moderate homogeneous the patient had no family history of enhancement with central necrotic area. Fig 1. A, B images are sagittal pre-contrast T1 WI of the dorsal spine in different planes. C and D images are sagittal and coronal T2 WI of the same lesion respectively. They display a well-defined, well-circumscribed oval shape extra dural lesion at level of D5-D6 that extends through and expands the left neural foramen (B). It displays slightly hyperintense signal on T1 WIs, iso intense signal on T2 WIs while the capsule of the lesion exhibits low T2 signal. The lesion measures 3.3 x 2 cm in maximal craniocaudal and AP dimensions. 367 | P a g e AL-AZHAR ASSIUT MEDICAL JOURNAL AAMJ ,VOL 13 , NO 3 , JULY 2015 Fig 2. Following IV contrast administration; A, B sagittal and coronal T1 WI with IV contrast respectively. C and D are axial T1 WI with IV contrast. The lesion displays moderate contrast homogeneous contrast enhancement with central necrotic area. The extension through left D5-D6 neural foramen is re-demonstrated giving the appearance of Dumbbell-shape. The patient prepared for surgery through arises in relation and adherent to a nerve posterior spinal approach with root insinuated inside left D5-D6 laminectomy of D4 – D6, and with aid of foramina, removed totally in its capsule surgical microscope, the tumor found including the intraforaminal part, the purely extradural, well circumscribed, attached nerve root coagulated and cut encapsulated, lobulated, gray in color, (Figure 3). covered by a thin, fibrous membrane, Fig 3. Intraoperative photograph shows removal of the tumor (arrow) totally in its capsule The removed material sent to melanotic schwannoma (formed of histopathological examination which melanin pigment with schwann cells) showed fusiform morphology with (Figure 4). cytoplasmic pigmentation, suggestive of 368 | P a g e Bokhary Mahmoud et al AAMJ ,VOL 13 , NO 3 , JULY 2015 Fig 4. Histopathological examination showed fusiform morphology with cytoplasmic pigmentation The patient recovered well without had spastic paraparesis with sensory level intraoperative complications and on at D6 with extensor planter bilaterally and second day postoperative and on with scissoring gait. examination of the patient, we observed Clinical presentation of melanotic that the spasticity became less. schwannoma is related to the involvement During follow up in outpatient clinic, the of a nerve (pain or sensory abnormality) patient started to walk without support, or due to mass effect as in extramedullary and his power improved gradually and he spinal tumors (2), with mean duration of followed in outpatient clinic with serial symptoms is 15 months (11). neurological examination and serial Our patient had no familial history or any spinal MRI, which showed no recurrence component of Carney complex (spotty for 3 years follow up period. pigmentation, endocrine overactivity or myxomas). DISCUSSION In spinal cord lesions, MRI remains the Melanotic schwannoma is an uncommon investigation of choice. Typically, the neoplasm, and can be divided into melanotic schwannomas are hyperintense psammomatous and nonpsammomatous on T1 and hypointense on T2 due to types and about half of the cases with paramagnetic free radicals in melanin, psammomatous melanotic schwannoma while non‑melanotic schwannomas are are related to Carney complex. While hypointense on T1 and hyperintense on nonpsammomatous melanotic T2. Both enhance on contrast (3). These schwannoma is considered to be a findings are similar to our case where his sporadic type, encountered in fourth lesion on MRI showed a well- decades (7) (8). Our case is a male circumscribed, encapsulated, oval shape patient, 40 years old. extra dural soft tissue lesion at D5-D6 Antonescu et. al. (9) found that, slight level. The lesion exhibits slightly female predominance in his study, with hyperintense signal on T1, isointense mean age 50 years old. signal on T2 and moderate contrast Melanotic schwannoma is characterized enhancement and causes widening of the by slow growth and low aggressiveness left D5-D6 neural foramen giving the (10). Our patient presented with one year appearance of Dumbbell-shape. duration of gradual onset of weakness of both lower limbs and on examination, he 369 | P a g e AL-AZHAR ASSIUT MEDICAL JOURNAL AAMJ ,VOL 13 , NO 3 , JULY 2015 Surgery is the primary treatment for 2 - Welling LC, Guirado VM, Tessari M, melanotic schwannoma and total excision Felix AR, Zanellato C, Figueiredo with tumor-free margins is recommended EG, et al: Spinal melanotic and usually curative. For large tumors, schwannomas. Arq Neuropsiquiatr. radiation and chemotherapy may be used 2012; 70 (2):156-7. in conjunction with surgery, and in cases, 3 - Hoover JM, Kumar R,
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