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Tumour that challenged diagnosis: mandibular myofibroma Mahesh Chinta, 1 A J Sai Sankar, 2 Sribala Naga Gantha, 1 Pratej Kiran Kanumuri 3 1 Department of Pedodontics and Preventive Dentistry, Panineeya Mahavidyalaya Institute of Dental Sciences and Research Centre, Hyderabad, Telangana, India 2 Department of Pedodontics and Preventive Dentistry, Sibar Institute of Dental Sciences, Guntur, Andhra Pradesh, India 3 Panineeya Institute of Dental Sciences and Hospital, Hyderabad, Telangana, India Correspondence to Dr Pratej Kiran Kanumuri, [email protected] Accepted 29 September 2016 To cite: Chinta M, Sankar AJS, Gantha SN, et al. BMJ Case Rep Published online: [ please include Day Month Year] doi:10.1136/bcr-2016- 217890 DESCRIPTION Myofibroma is a rare, benign, nodular tumour of soft tissues, bones or internal organs. It was first described by Stout in 1954 as congenital generalised fibromatosis and it was renamed by Chung et al as ‘infantile myofibromatosis’ as the onset of the disease is at a young age. 1 Later Smith et al introduced the term ‘myofibroma’ and suggested that these lesions may not be exclusively paediatric. 2 Myofibroma represents the most common fibrous tumour of infancy, predominantly in the head and neck region. However, its involvement in jaws is rare. This disease presents as a locally invasive, hard painless mass unless it is secondarily infected. The lesion is considered to be completely benign but there is a potential for it, being confused with more aggressive spindle cell tumours. 3 So it is very import- ant to make an accurate diagnosis of such lesions, because of their common clinical and radiographical features. Advanced diagnostic options such as immu- nohistochemistry should be explored, which may give definitive diagnosis. This is a case report of a 4-year-old boy who came to the outpatient department with a diagnosis of fibrosarcoma for a swelling in the lingual surface of mandibular anterior. History revealed that patient went to the dentist with symptom of swel- ling in the floor of the mouth since 15 days. The patient’s parents reported that swelling was gradual in onset and was not associated with pain or pus discharge. Medical and dental histories were unre- markable. Incisional biopsy was performed and a diagnosis of fibrosarcoma was made by the dentist. After a week the patient reported to the department of paediatric dentistry with this history. On extraoral examination, bilateral palpable submandibular lymph nodes which were 1×1 cm in size, soft in consistency and tender on palpation were observed. On intraoral examination a 2×3 cm sized lesion, which was oval in shape was observed on the lingual side of primary mandibular anterior in relation to 71,72,73,81 (ISO system) crossing the midline and extending over the lingual fraenum. The mucosa over the growth was erythematous. On palpation the growth was sessile and firm in consistency, non-compressible, reducible, fluctulent and tender on palpation ( figure 1). Orthopantomogram showed evidence of a well- defined unilocular radiolucency in the mental region of the mandible extending from the mesial aspect of permanent canine follicle to the other side indicating erosive changes ( figure 2). All the haematological findings were in normal limits. An incisional biopsy was performed and histological examination revealed the presence of spindle-shaped cells with prominent nucleus and the cells were arranged in a ‘lace-like pattern’ ( figure 3) Figure 1 Intraoral picture showing diffuse swelling in the floor of the mouth. Figure 2 Orthopantomogram showing well-defined unilocular radiolucency in the mental region of mandible. Figure 3 Histopathological picture showing a ‘lace-like’ cell pattern. Chinta M, et al. BMJ Case Rep 2016. doi:10.1136/bcr-2016-217890 1 Images in … on 27 September 2020 by guest. Protected by copyright. http://casereports.bmj.com/ BMJ Case Reports: first published as 10.1136/bcr-2016-217890 on 13 October 2016. Downloaded from
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Tumour that challenged diagnosis: mandibularmyofibromaMahesh Chinta,1 A J Sai Sankar,2 Sribala Naga Gantha,1 Pratej Kiran Kanumuri3
1Department of Pedodonticsand Preventive Dentistry,Panineeya MahavidyalayaInstitute of Dental Sciencesand Research Centre,Hyderabad, Telangana, India2Department of Pedodonticsand Preventive Dentistry, SibarInstitute of Dental Sciences,Guntur, Andhra Pradesh, India3Panineeya Institute of DentalSciences and Hospital,Hyderabad, Telangana, India
Correspondence toDr Pratej Kiran Kanumuri,[email protected]
Accepted 29 September 2016
To cite: Chinta M,Sankar AJS, Gantha SN,et al. BMJ Case RepPublished online: [pleaseinclude Day Month Year]doi:10.1136/bcr-2016-217890
DESCRIPTIONMyofibroma is a rare, benign, nodular tumour ofsoft tissues, bones or internal organs. It was firstdescribed by Stout in 1954 as congenital generalisedfibromatosis and it was renamed by Chung et al as‘infantile myofibromatosis’ as the onset of the diseaseis at a young age.1 Later Smith et al introduced theterm ‘myofibroma’ and suggested that these lesionsmay not be exclusively paediatric.2
Myofibroma represents the most common fibroustumour of infancy, predominantly in the head andneck region. However, its involvement in jaws israre. This disease presents as a locally invasive, hardpainless mass unless it is secondarily infected. Thelesion is considered to be completely benign butthere is a potential for it, being confused with moreaggressive spindle cell tumours.3 So it is very import-ant to make an accurate diagnosis of such lesions,because of their common clinical and radiographicalfeatures. Advanced diagnostic options such as immu-nohistochemistry should be explored, which maygive definitive diagnosis.This is a case report of a 4-year-old boy who
came to the outpatient department with a diagnosisof fibrosarcoma for a swelling in the lingual surfaceof mandibular anterior. History revealed thatpatient went to the dentist with symptom of swel-ling in the floor of the mouth since 15 days. Thepatient’s parents reported that swelling was gradualin onset and was not associated with pain or pusdischarge. Medical and dental histories were unre-markable. Incisional biopsy was performed and adiagnosis of fibrosarcoma was made by the dentist.After a week the patient reported to the departmentof paediatric dentistry with this history. On extraoralexamination, bilateral palpable submandibular lymphnodes which were 1×1 cm in size, soft in consistencyand tender on palpation were observed. On intraoralexamination a 2×3 cm sized lesion, which was ovalin shape was observed on the lingual side of primarymandibular anterior in relation to 71,72,73,81 (ISOsystem) crossing the midline and extending over thelingual fraenum. The mucosa over the growth waserythematous. On palpation the growth was sessileand firm in consistency, non-compressible, reducible,fluctulent and tender on palpation (figure 1).Orthopantomogram showed evidence of a well-
defined unilocular radiolucency in the mentalregion of the mandible extending from the mesialaspect of permanent canine follicle to the otherside indicating erosive changes (figure 2).All the haematological findings were in normal
limits. An incisional biopsy was performed andhistological examination revealed the presence ofspindle-shaped cells with prominent nucleus and thecells were arranged in a ‘lace-like pattern’ (figure 3)
Figure 1 Intraoral picture showing diffuse swelling inthe floor of the mouth.
Figure 2 Orthopantomogram showing well-definedunilocular radiolucency in the mental region of mandible.
Figure 3 Histopathological picture showing a ‘lace-like’cell pattern.
Chinta M, et al. BMJ Case Rep 2016. doi:10.1136/bcr-2016-217890 1
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suggesting the differential diagnosis of leiomyoma or myofibromawhich was contrary to the previous diagnosis. To make an accur-ate diagnosis immunohistochemical analysis was performed. Thetest was positive for smooth muscle actin and negative fordesmin, thus the final diagnosis of myofibroma was made.Complete surgical excision of the tumour en bloc along withwedge of the mandible (figure 4) and tooth buds of 31,32,41(ISO system) was performed under general anaesthesia (figure 5).Prosthetic rehabilitation of missing teeth with acrylic remov-able partial denture was carried out after 2 months (figure 6).
The patient was followed up for a year and no recurrence wasobserved.
Learning points
▸ Myofibroma is an uncommon spindle cell neoplasm rarelyfound in the oral cavity especially in the mandible.
▸ Immunohistochemical staining is an indispensable tool toidentify the nature of neoplastic cells and make an accuratediagnosis in cases of myofibroma.
▸ Myofibromas are unencapsulated tumours, treatment shouldinclude complete excision of the lesion along with a borderof the clinically normal bone.
▸ Early diagnosis of the lesion with advanced techniques ismandatory to eliminate bias in diagnosis and for propertreatment planning.
Contributors MC was involved in carrying out the case. AJSS was involved inguiding the case. SNG was involved in the preparation of the manuscript. PKK wasinvolved in the preparation of the manuscript.
Competing interests None declared.
Patient consent Obtained.
Provenance and peer review Not commissioned; externally peer reviewed.
REFERENCES1 Chung EB, Enzinger FM. Infantile myofi bromatosis. Cancer 1981;48:1807.2 Smith KJ, Skelton HG, Barrett TL, Lupton GP, Graham JH. Cutaneous myofibroma.
Mod Pathol 1989;2:603–9.3 Sabri A, Korban Z, Rizk SA, et al. Parapharyngeal space myofibroma: a case report
and review of the literature. SM J Surg 2015;1:1002.
Figure 4 Postoperative intraoral picture showing complete surgicalexcision of the tumour en bloc along with wedge of the mandible.
Figure 5 Postoperative orthopantomogram showing the resectedanterior segment of the mandible along with tooth buds.
Figure 6 Postoperative intraoral picture with acrylic removable partialdenture.
2 Chinta M, et al. BMJ Case Rep 2016. doi:10.1136/bcr-2016-217890
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