letter to the editor comment on facial atrophy in oral

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Letter to the Editor Comment on (Facial Atrophy in Oral Submucous Fibrosis: An Association or a Coincidence) Sonal Grover and George Koshy Department of Oral & Maxillofacial Pathology & Microbiology, Christian Dental College, CMC, Ludhiana, Punjab, India Correspondence should be addressed to Sonal Grover; [email protected] Received 18 July 2016; Accepted 8 September 2016 Academic Editor: Giuseppe Colella Copyright © 2016 S. Grover and G. Koshy. is is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. With reference to a case report titled “Facial Atrophy in Oral Submucous Fibrosis: An Association or a Coincidence” [1] published in your esteemed journal, we would like to emphasize the importance of considering systemic sclerosis (SS) as an important differential diagnosis in oral submucous fibrosis (OSMF). Systemic sclerosis is a clinically heterogeneous gener- alized disorder which affects the connective tissue of the skin and internal organs such as gastrointestinal tract, lungs, heart, and kidneys. It is characterized by enormous depo- sition of collagen, alterations of the microvasculature, and disturbances of the immune system [2]. Oral manifestations in SS are very common and these include limited ability to open the mouth, facial and tongue rigidity, thinning of lips, xerostomia, periodontal disease, increased periodontal ligament width, and osseous resorption of the mandible [2, 3]. A few decades back, plenty of cases have been reported with neurological manifestations as well [4, 5]. Oral submucous fibrosis (OSMF) is a chronic insidious collagen related disorder associated with betel quid chewing and characterized by progressive hyalinization of the sub- mucosa. e hallmark of the disease is submucosal fibrosis that affects most parts of the oral cavity, causing progressive trismus with rigid lips, cheeks, pharynx, and upper third of the esophagus leading to dysphagia [6]. Since extensive fibrosis plays a key role in the pathogenesis of OSMF also, it is not possible to distinguish OSMF from SS histologically. In the OSMF case reported [1], the patient had extensive atrophy of facial muscle along with restricted mouth opening. e striking finding in this case was cerebral and cerebellum atrophy. is finding should have prompted the authors to rule out the possibility of SS, as extensive facial atrophy with neurological manifestations is more likely to be seen in SS than in OSMF. Except for the epithelial dysplasia observed, all the remaining findings are common to SS as well. e epithelial dysplasia can easily be correlated with the long term consumption of areca nut by the patient. e diagnostic criteria for SS are as given below: Major Criterion (i) Proximal diffuse (truncal) sclerosis (skin tightness, thickening, and nonpitting induration) Minor Criteria (i) Sclerodactyly (only fingers and/or toes) (ii) Digital pitting scars or loss of substance of the digital finger pads (pulp loss) (iii) Bibasilar pulmonary fibrosis e patient should fulfill the major criterion or two of the three minor criteria. us, through this letter, the authors intend to highlight the differential diagnosis of SS in OSMF cases involving parts of the body other than the oral cavity and pharynx. Competing Interests ere were no competing interests related to this paper. Hindawi Publishing Corporation Case Reports in Dentistry Volume 2016, Article ID 5747458, 2 pages http://dx.doi.org/10.1155/2016/5747458

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Page 1: Letter to the Editor Comment on Facial Atrophy in Oral

Letter to the EditorComment on (Facial Atrophy in Oral Submucous Fibrosis:An Association or a Coincidence)

Sonal Grover and George Koshy

Department of Oral & Maxillofacial Pathology & Microbiology, Christian Dental College, CMC, Ludhiana, Punjab, India

Correspondence should be addressed to Sonal Grover; [email protected]

Received 18 July 2016; Accepted 8 September 2016

Academic Editor: Giuseppe Colella

Copyright © 2016 S. Grover and G. Koshy. This is an open access article distributed under the Creative Commons AttributionLicense, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properlycited.

With reference to a case report titled “Facial Atrophy inOral Submucous Fibrosis: An Association or a Coincidence”[1] published in your esteemed journal, we would like toemphasize the importance of considering systemic sclerosis(SS) as an important differential diagnosis in oral submucousfibrosis (OSMF).

Systemic sclerosis is a clinically heterogeneous gener-alized disorder which affects the connective tissue of theskin and internal organs such as gastrointestinal tract, lungs,heart, and kidneys. It is characterized by enormous depo-sition of collagen, alterations of the microvasculature, anddisturbances of the immune system [2]. Oral manifestationsin SS are very common and these include limited abilityto open the mouth, facial and tongue rigidity, thinning oflips, xerostomia, periodontal disease, increased periodontalligamentwidth, and osseous resorption of themandible [2, 3].A few decades back, plenty of cases have been reported withneurological manifestations as well [4, 5].

Oral submucous fibrosis (OSMF) is a chronic insidiouscollagen related disorder associated with betel quid chewingand characterized by progressive hyalinization of the sub-mucosa. The hallmark of the disease is submucosal fibrosisthat affects most parts of the oral cavity, causing progressivetrismus with rigid lips, cheeks, pharynx, and upper thirdof the esophagus leading to dysphagia [6]. Since extensivefibrosis plays a key role in the pathogenesis of OSMF also, itis not possible to distinguish OSMF from SS histologically.

In the OSMF case reported [1], the patient had extensiveatrophy of facialmuscle alongwith restrictedmouth opening.

The striking finding in this case was cerebral and cerebellumatrophy. This finding should have prompted the authors torule out the possibility of SS, as extensive facial atrophy withneurological manifestations is more likely to be seen in SSthan in OSMF. Except for the epithelial dysplasia observed,all the remaining findings are common to SS as well. Theepithelial dysplasia can easily be correlated with the longterm consumption of areca nut by the patient.The diagnosticcriteria for SS are as given below:

Major Criterion(i) Proximal diffuse (truncal) sclerosis (skin tightness,

thickening, and nonpitting induration)

Minor Criteria(i) Sclerodactyly (only fingers and/or toes)(ii) Digital pitting scars or loss of substance of the digital

finger pads (pulp loss)(iii) Bibasilar pulmonary fibrosis

The patient should fulfill the major criterion or two of thethree minor criteria.

Thus, through this letter, the authors intend to highlightthe differential diagnosis of SS in OSMF cases involving partsof the body other than the oral cavity and pharynx.

Competing Interests

There were no competing interests related to this paper.

Hindawi Publishing CorporationCase Reports in DentistryVolume 2016, Article ID 5747458, 2 pageshttp://dx.doi.org/10.1155/2016/5747458

Page 2: Letter to the Editor Comment on Facial Atrophy in Oral

2 Case Reports in Dentistry

References

[1] S. S. Shetty, P. Shetty, and A. R. Chougule, “Facial atrophy inoral submucous fibrosis: an association or a coincidence,” CaseReports inDentistry, vol. 2016, Article ID 2080467, 3 pages, 2016.

[2] R. Srivastava, B. Jyoti, M. Bihari, and S. Pradhan, “Progressivesystemic sclerosis with intraoral manifestations: a case reportand review,” Indian Journal of Dentistry, vol. 7, no. 2, pp. 99–104,2016.

[3] I. H. Bajraktari, A. Kryeziu, F. Sherifi, H. Bajraktari, A. Lahu,and G. Bajraktari, “Oral manifestations of systemic sclerosisand correlationwith anti-topoisomerase I antibodies (SCL-70),”Medical Archives, vol. 69, no. 3, pp. 153–156, 2015.

[4] L. Pinheiro, J. Freitas, M. Lucas, and R. M. M. Victorino, “Cer-ebellar atrophy in systemic sclerosis,” Journal of the Royal Societyof Medicine, vol. 97, no. 11, pp. 537–538, 2004.

[5] P. Lee, J. Bruni, and S. Sukenik, “Neurological manifestations insystemic sclerosis (scleroderma),” Journal of Rheumatology, vol.11, no. 4, pp. 480–483, 1984.

[6] F. M. Ali, A. Patil, K. Patil, and M. Prasant, “Oral submucousfibrosis and its dermatological relation,” Indian DermatologyOnline Journal, vol. 5, no. 3, pp. 260–265, 2014.

Page 3: Letter to the Editor Comment on Facial Atrophy in Oral

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