Grinspan syndrome with periodontitis: Coincidence or correlation?

Grinspan syndrome is a syndromic complex which comprises a triad of hypertension, diabetes, and oral lichen planus. It remains an enigmatic condition that whether it is separate entity or drug-induced lichenoid reaction emerging due to medications used to treat hypertension and diabetes. Diabetes is related to long-term hyperglycinemia leading to accelerated destruction of both nonmineralized connective tissue and bone leading to periodontitis. Moreover, there is also a possible link between cardiovascular disease and periodontitis. Here, we are presenting a case with the symptomatic triad fulfilling the diagnostic criteria of Grinspan syndrome. A 50-year-old female with type 2 diabetes mellitus and vascular hypertension came with the chief complaint of bleeding gums and severe burning sensation of the oral cavity. In addition, she was having generalized alveolar bone loss and clinical attachment loss. To the best of our knowledge, this coexistence has not been reported earlier. This needs further evaluation so that the preventive measures can be taken at early stage.

INTRODUCTION

Oral lichen planus is one of the common chronic inflammatory, noninfectious disease affecting mucosa of the oral cavity. It may be associated with several systemic disease. A association that exists between the most severe form of lichen planus- the erosive form, diabetes mellitus and arterial hypertension is called Grinspan syndrome.[1] It was reported by Grinspan and named by Grupper and Avil as “Grinspan Syndrome.”[2] The oral lichenoid lesion in Grinspan syndrome may be a reaction to the drugs used to treat diabetes mellitus and/or hypertension. However, the precise relationship is not clear.

It is suggested that prolonged hyperglycemia in diabetes can lead to exaggerated inflammatory and immune response, which can lead to more periodontal breakdown.[3] Evidence supports the formation of advanced glycation end products resulting in greater breakdown of collagen fibers and shows the accelerated destruction of bone leading to periodontitis.[4] Evidence also suggests poor blood pressure control in patients suffering from periodontitis.[5] Oral lichen planus with chronic periodontitis patients also show higher serum interleukin (IL)-17 expression suggesting its role in the immunopathogenesis of both diseases.[6] No published data has been presented regarding the association of Grinspan syndrome and periodontitis. This case report presents a rare case of Grinspan syndrome with generalized periodontitis.

CASE REPORT

A 50-year-old female came to Department of Periodontics with chief complaint of bleeding gums and severe burning sensation of the oral cavity for the past 5 years. She was on topical corticosteroids on and off for the past 5 years but never got complete relief. Her medical history revealed that she was having diabetes mellitus for the past 4 years and was under medication for the same for the past 2 years and then she discontinued. Her history further revealed hypertension, for which she is taking no medication but does practice general measures such as diet control and control of salt intake. There are no signs of skin involvement. Intraoral examination reveals patches and white lacy lesion crossing each other on the buccal mucosa and tongue. Gingiva was red, and there was bleeding on probing [Figure 1]. Orthopantomogram radiograph reveals alveolar bone loss around molars and upper incisors [Figure 2]. Routine hemogram was normal. Random blood sugar and blood pressure were elevated. Biopsy confirmed the diagnosis of lichen planus. The section showed saw-tooth rete pegs, lymphocytic infiltration, and degeneration of basal layer [Figure 3].

Figure 1

Preoperative view showing white lacy patterns on the buccal mucosa and tongue

Figure 2

Orthopantomogram showing severe bone loss

Figure 3

Histopathological examination showing lymphocytic infiltration and saw-tooth rete pegs

The patient was referred to a physician for diabetes and hypertension and reevaluated after 1 month. The patient was treated with 0.1% tacrolimus for topical application 2–3 times daily for 14 days, and thorough scaling and root planing was done along with 0.2% chlorhexidine mouthwash. The patient got complete symptomatic relief and tacrolimus was continued for another 15 days. Then, tacrolimus was discontinued, and for the past 9 months, she is symptom free [Figure 4].

Figure 4

Postoperative view complete relief

DISCUSSION

Grinspan syndrome is an association that exists between erosive lichen planus, diabetes mellitus, and arterial hypertension. Grinspan syndrome with periodontitis is rarely reported. It remains a rare entity as little research has been performed in this area.

In the present case, the patient was suffering from diabetes and hypertension in addition to chronic periodontitis. Literature also supports the association of periodontitis with altered blood sugar levels, obesity, and hypertension which are main components of metabolic syndrome.[7] Systemic oxidative stress and exaggerated inflammatory response are responsible for this kind of association. As the number of contributing metabolic components increases, odds of having periodontitis also increases.[7]

This patient was suffering from erosive lichen planus for the past 5 years which was not responding to corticosteroid therapy. Periodontitis and oral lichen planus share common immunopathogenesis through serum IL-17 expression.[6] Lichen planus is an autoimmune mucocutaneous disorder responsible for T-cell activation and CD54 expression responsible for the release of significant amount of cytokines and basement membrane damage.[6] In oral lichen planus, burning sensation in oral cavity, oral ulceration, and sensitivity to spices, etc., may lead to inadequate oral hygiene and poor nutrition, which is a predisposing factor for periodontitis. Studies also support that periodontal condition in oral lichen planus patients is worse than the control group.[8]

Thus, all the above-mentioned entities, that is, diabetes, hypertension, and lichen planus are related to periodontitis through systemic inflammation. Antimicrobial peptide could also be possible link between periodontitis and its established risk factors such as diabetes and hypertension.[9] A clinical interrelated pathway for correlation between all these four components can be understood from the mentioned [Figure 5].

Figure 5

Clinical pathways to show correlation between the four different clinical entities (lichen planus, diabetes, hypertension, and periodontitis) of this case report

Understanding the complex association between these entities can lead to better understanding of these diseases. It can also unravel the mystery and complexity concerning the treatment of these complex associations. Since multiple components are involved, only multidisciplinary treatment approach will be the key to successful treatment. In this patient diabetes and hypertension was successfully managed by pharamacological and lifestyle modification approach. Thorough scaling, root planning, and subgingival curettage were done to treat periodontitis. Studies support that periodontal therapy can improve the glycemic control.[10] In general, the treatment for oral lichen planus mainly revolves around topical and systemic corticosteroid, but the patients who do not respond to corticosteroid therapy are given tacrolimus as in the present scenario.[11] Tacrolimus is a macrolide antibiotic which interferes with the calcium-/calmodulin-dependent phosphatase calcineurin. It has been shown to produce less skin atrophy than corticosteroid, but local irritation at the site of application is a side effect in some cases.[12] This patient responded well with 0.1% tacrolimus without any adverse effect.

Although literature supports the common link between periodontitis, diabetes, oral lichen planus and hypertension individually, yet their syndromic nature with periodontitis is rarely reported. Apart from inflammation and immunity mediators, there is also possibility of association through unexplained confounders which are difficult to measure such as lifestyle, socioeconomic status, stress, health service utilization, and social network behavior.[11] Extensive study is needed to delineate the relationship among four entities. Until we make further progress, we should accept the fact that the association is worth investigation, and the patient suffering from any of the above entities should also be explored for periodontitis for better management of the condition.

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.