|LETTER TO THE EDITOR
|Ahead of print
Mental nerve paresthesia in non-Hodgkin lymphoma: A subtle sign of occult malignancy
Adarsh Kudva1, Ravindranath Vineetha2, Shruti Gunashekhar1, Ranjini Kudva3, Mathangi Kumar1
1 Department of Oral and Maxillofacial Surgery, Manipal College of Dental Sciences, Manipal Academy of Higher Education, Manipal, Karnataka, India
2 Department of Oral Medicine and Radiology, Manipal College of Dental Sciences, Manipal Academy of Higher Education, Manipal, Karnataka, India
3 Department of Pathology, Kasturba Medical College, Manipal Academy of Higher Education, Manipal, Karnataka, India
|Date of Submission||24-Mar-2020|
|Date of Decision||21-Apr-2020|
|Date of Acceptance||20-May-2020|
|Date of Web Publication||02-Jul-2021|
Department of Oral Medicine and Radiology, Manipal College of Dental Sciences, Manipal Academy of Higher Education, Manipal, Karnataka
Source of Support: None, Conflict of Interest: None
|How to cite this URL:|
Kudva A, Vineetha R, Gunashekhar S, Kudva R, Kumar M. Mental nerve paresthesia in non-Hodgkin lymphoma: A subtle sign of occult malignancy. Indian J Cancer [Epub ahead of print] [cited 2021 Jul 27]. Available from: https://www.indianjcancer.com/preprintarticle.asp?id=320437
Mental nerve paresthesia or numb chin is a common clinical manifestation seen in various conditions ranging from common odontogenic infections to systemic malignancies with neural involvement. The mental nerve is the branch of the inferior alveolar nerve of the mandibular division of trigeminal nerve supplies mucosa of the lower lip, skin of the chin, and the mandibular gingival around the incisors. Numb chin syndrome (NCS) described by Charles Bell, is widely described in the literature as a reflection of systemic malignancy. However, this sign is not pathognomonic and can be associated with common odontogenic causes such as osteomyelitis, local trauma, and so on due to which it is often an overlooked clinical sign of malignancy.
Herein, we report a case of non-Hodgkin lymphoma (NHL), which clinically resembled an odontogenic fascial space infection but also presented with mental nerve paresthesia.
A 56-year-old diabetic man presented with a painful swelling on the right side of the face of a 1-week duration. Although atraumatic extraction of infected upper tooth was performed under antibiotic coverage, non-localizable pain and diffuse swelling persisted. He also reported of altered sensation and numbness in the chin region. Swelling slowly progressed to cause limited mouth opening. Laboratory values showed uncontrolled diabetic status (Random Blood Sugar (RBS): 310 mg/dL), increased total WBC count (12.8 × 103/μL), and increased ESR (30 mm/h).
On examination, a large diffuse swelling was present involving the right lower midface. The mandibular first molar tooth was decayed, tender on percussion with associated vestibular swelling extending to ramus. The radiograph showed vague periapical changes in molar teeth suggestive of infection. Contrast-enhanced computed tomography revealed unusual minute lytic areas and cortical irregularity in buccal and lingual cortices in the mandibular ramus and angle region [Figure 1] and [Figure 2]. Considering the acute duration, altered sensation, decayed teeth, and radiographic features, we formulated a working diagnosis of jaw osteomyelitis with buccal and submandibular fascial space involvement.
|Figure 1: Coronal section of contrast-enhanced computed tomography depicting bulkiness of the right masticator space with focal lytic areas in the mandible|
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|Figure 2: 3D reconstructed image of right mandible showing multiple punched out osteolytic lesions in the mandibular body and ramus region|
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Suspected lower molar teeth were extracted under general anesthesia, and the infected bone was curetted. Histopathology demonstrated dense infiltration of fibrous tissue and skeletal muscle by monotonous medium-sized cells with round nuclei. Immunohistochemistry confirmed the presence of lymphoid cells, which were positive for CD20, CD10, BCL6, BCL2 [Figure 3]a, [Figure 3]b, [Figure 3]c, [Figure 3]d, [Figure 3]e, [Figure 3]f, and was negative for c-MYC and CD-3. [Figure 3]g and [Figure 3]h. Thus, the final diagnosis of this case was NHL large B-cell germinal center subtype. Bone marrow biopsy was done and it revealed no abnormal cells. The patient was advised treatment with chemotherapy and he underwent three cycles of chemotherapy according to the standard R-CHOP protocol with the following drugs: Intravenous injection of rituximab 375 mg/m2, cyclophosphamide 750 mg/m2, doxorubicin 50 mg/m2, and vincristine 1.4 mg/m2 along with oral prednisolone 100 mg per day for 5 days. The patient tolerated the chemotherapy well and is on regular follow-up. Full body PET-CT was done as a part of metastatic workup which showed no evidence of metastasis. Since the disease was isolated to right masticatory space, the patient was in stage I as per Ann Arbor staging of NHL. The serum lactate dehydrogenase (LDH) level was 180 IU/L (Normal range: 125–220 IU/L). Besides, our patient did not have any B symptoms (absence of fever >38°C, night sweats, and/or weight loss >10% of body weight in the 6 months preceding the time of presentation). Considering these factors, the International Prognostic Index (IPI) for this patient was 1 which suggests a good prognosis.
|Figure 3: (a) Photomicrograph (hematoxylin and eosin staining, original magnification ×100) showing sheets of medium-sized neoplastic lymphoid cells in a sclerosed stroma; (b-f) Immunohistochemicalstaining positive for CD10, CD20, CD45, BCL2, BCL6, and; (g and h) negative for c-MYC and CD3 (Original magnification ×200)|
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In the present case, mental nerve paresthesia could be attributed to NCS, resulting from malignant infiltration of inferior alveolar nerve in metastatic NHL.,, As the features of NCS overlap with common odontogenic conditions, this clinical diagnostic clue should be considered with caution and diagnosed by exclusion.
Though the exact pathophysiology is unclear, the suggested mechanisms include direct tumoral infiltration into the nerve or compression of the mental nerve by tumor mass. Another possible hypothesis is the microscopic seeding of the tumor cells into the nerve, which may not be visible in radiologic imaging. Till date, metastatic involvement of jawbones by breast cancer is reported to be the most common non-hematologic neoplastic cause of NCS while NHL is the most common myeloproliferative disorder causing NCS. Other malignancies presenting with NCS include leukemia, multiple myeloma, Burkitt's lymphoma, and metastasis from lung, kidney, thyroid, and prostate cancers.
Extranodal involvement of lymphoma may occur mainly via hematogenous spread to the central nervous system (CNS), direct invasion from contiguous structures, or extension along nerves. Diffuse large B-cell lymphoma (DLBCL) is the most common lymphoma found in the CNS. Secondary central nervous system lymphoma (SCNSL) or metastatic lymphoma is defined as secondary CNS involvement in patients with systemic lymphoma. SCNSL is a rare but fatal complication of systemic lymphoma occurring in 1% of cases., CSF (cerebrospinal fluid) flow cytometry is a test that can accurately diagnose CNS involvement by lymphoma. This technique involves the detection of clonal B-cells as low as 0.9%. National Comprehensive Cancer Network recommends the submission of CSF for both cytology and flow cytometry analysis (FCA) for patients with high clinical suspicion for CNS lymphoma.
Although panoramic radiographs can be initial screening imaging modality, computed tomography or magnetic resonance imaging is essential for better visualization in suspected cases. Identification and appropriate treatment of the underlying primary etiology will result in complete resolution of the symptom.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
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Conflicts of interest
There are no conflicts of interest.
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Please see the related commentary [Jandial A, Sahu KK. Not just the tip of the iceberg: Commentary on Numb Chin syndrome. Indian J Cancer (this issue)]
[Figure 1], [Figure 2], [Figure 3]