Severe Xerostomia Induced by Multiple Systemic Diseases in a Patient with Psoriasis Vulgaris: A Case Report and Literature Review

Introduction

Psoriasis is derived from the Greek words “psora”, meaning itch, and “iasis”, meaning action or condition.^1–3 Psoriasis is an autoimmune disease characterized by skin inflammation, epidermal hyperplasia, an increased risk of arthritis, cardiovascular morbidity, and psychosocial problems.^1–3 Psoriasis is a chronic, non-contagious skin disorder that presents as well-circumscribed erythematous plaques—rough, caked, silvery-white squamous plaques mainly on the scalp, elbows, knees, feet, body, and nails.^3–6 The etiology of psoriasis is unknown, but it is thought to be related to genetics, autoimmune diseases, and environmental factors.^3,5,7

The prevalence of psoriasis in various countries ranges from 0.09% to 11.4%.⁷ Psoriasis may occur at any age, especially between the ages of 15 and 30.³ There are five types of psoriasis: psoriasis vulgaris, intertriginous psoriasis, guttate psoriasis, pustular psoriasis, and erythrodermic psoriasis.^3,5,7,8 Psoriasis vulgaris is the most common type of psoriasis seen in 90% of patients.^3,9 A commonly seen manifestation of extracutaneous psoriasis is arthritis in 40% of patients.^3,10,11

Psoriasis may be associated with other diseases or comorbidities, including oral cavity involvement.¹² Oral psoriasis is a rare manifestation of psoriasis that may appear together with classic psoriasis, type vulgaris.¹³ Oral psoriasis findings, such as geographic tongue, fissure tongue, and localized psoriasis plaques.^4,5,13–19 Oral psoriasis is usually asymptomatic, but some patients complain of discomfort, dry mouth, stinging, and even burning sensations, which can affect the patient’s quality of life.¹⁴

Multiple factors may cause xerostomia, such as dehydration, aging, habits such as breathing through the mouth, smoking, alcohol, excessive caffeine consumption, systemic diseases, autoimmune diseases, xerogenic drugs, psychological conditions, head and neck radiotherapy, and malnutrition.^14,20–27 Autoimmune diseases can cause xerostomia, such as psoriasis in this case. Determining the etiology of xerostomia allows for an appropriate diagnosis and early intervention. Therefore, it is important to understand the factors that promote xerostomia in order to treat the underlying condition, alleviate symptoms, and prevent unwanted complications that can reduce the patient’s quality of life.

This report presents the condition of severe xerostomia in a patient with psoriasis vulgaris and other systemic diseases. This case report aims to describe the clinical mechanism of xerostomia in a patient with psoriasis vulgaris. The presence of multiple systemic diseases affecting the patient’s condition makes this report unique. This report also encompasses a review of 11 reported cases of xerostomia and summarizes the risk factors, systemic disease, and treatment options.

Case Report

A 51-year-old inpatient man was referred to the Oral Medicine Department of Hasan Sadikin Bandung Hospital with difficulty swallowing due to a sore throat and dry mouth last week. The patient could not taste the food. He also complained of fever, weakness accompanied by cold sweat, blurred vision, weak extremities, itchy skin, redness, and body swelling. He was diagnosed with chronic adrenal insufficiency, severe psoriasis vulgaris with psoriatic arthritis, acute circulatory collapse, anemia of inflammation (AI), acute kidney injury (AKI) stage III, dehydration, gastritis related to drugs (methotrexate and cyclosporine), urinary tract infections, and malnutrition by the Dermatovenereology Department and the Internal Medicine Department. He was given 0.9% NaCl, ceftriaxone, omeprazole, metoclopramide, methotrexate, cetirizine, loratadine, folic acid, calcium, albumin, 5% dextrose, chlorpheniramine maleate, cefadroxil, and paracetamol.

According to his medical history, he has complained of reddish, scaly, itchy patches on his skin with a burning sensation on the scalp, nape, and knees since 15 years ago. He was diagnosed with psoriasis vulgaris by the Dermatovenereology Department and was treated with methylprednisolone. After the complaints of itching and redness on the skin subsided, he bought it himself and took the drug for 15 years without doctor monitoring. Four months before his hospitalization, he stopped taking medication on his own. He reported no history of other systemic diseases, such as hypertension or diabetes mellitus. He has no family history of having the same complaint. He denied a history of allergies to food, drugs, or other allergens.

The physical examination revealed a general state: looks moderately ill; consciousness: compos mentis; weight: 63 kg; height: 167 cm; blood pressure: 110/70 mmHg; pulse: 80 times/minute; respiration: 20 times/minute; body temperature: 36.5°C. Extra-oral examination revealed dry, scaly skin all over the body. He had joint stiffness. His face looked symmetrical, and the skin was scally, dry, and reddish (Figure 1A). The conjunctiva was anemic, and the sclera was non-icteric. The lips were dry and peeling, and there were painful fissures at both corners of the lips (Figure 1B). The nails appeared thickened with a ragged texture (Figure 1C). The submandibular and submental lymph nodes showed no abnormalities.

Figure 1 Extraoral condition on the first visit; (A) Reddish and peeling skin on the face; (B) Dry and exfoliative lips; (C) Nail thickening and joints appearing stiff.

Intraoral examination revealed dryness and a glassy appearance of the oral mucosa, and the mouth mirror was adherent to the buccal mucosa (Figures 2A–D). The palate had a glassy appearance and was free of debris (Figure 2E). The tongue was erythematous, depapillated, and lobulated; two-thirds of the anterior dorsal tongue had shallow longitudinal fissures, and the mouth mirror stuck to the dorsal tongue (Figures 2F–H). The ventral tongue appeared glassy (Figure 2I). There was no saliva pooling on the floor of the mouth (Figure 2J). Plaque and calculus were present in all regions. A serological examination may be seen in Table 1. Based on anamnesis and clinical examinations, he was diagnosed with severe xerostomia (a score of 8 according to the Challacombe scale and a score of 17 according to the Indonesian Summated Xerostomia Inventory (SXI-ID) questionnaire), a fissured tongue, exfoliative cheilitis, angular cheilitis, and generalized chronic marginal gingivitis.

Table 1 Serological Examination Result

Figure 2 The clinical appearance of intraoral dryness: (A) Upper labial mucosa; (B) Lower labial mucosa; (C) Right buccal mucosa; (D) Left buccal mucosa; (E) Palatum; (F) Tongue dorsum; (G) Right lateral tongue; (H) Left lateral tongue; (I) Tongue ventral; (J) Floor of the mouth.

The oral management of the patient was to apply a thin layer of petroleum jelly on the lips three times a day for exfoliative cheilitis, gargle with chlorine dioxide mouthwash three times a day for xerostomia, apply 2% miconazole cream two times a day for angular cheilitis, and suck benzydamine HCl lozenges three times a day for painful swallows. On the fifth day of his hospitalization, the patient developed respiratory distress and was diagnosed with hospital-acquired pneumonia (HAP). On the sixth day of his hospitalization, he had decreased consciousness, and his condition deteriorated. The patient’s family refused to have him intubated and transferred him to the Intensive Care Unit (ICU), and the patient passed away.

Discussion

Xerostomia was first introduced by Bartley in 1968. It is derived from the Greek “xeros”, which means dry, and “stoma”, which means mouth. Synonyms for xerostomia are oligosalia or stomatitis sicca.^8,28,29 Xerostomia is a subjective feeling of oral dryness and may be determined by questioning the individual’s perception of oral dryness. Clinicians often use the terms xerostomia and hyposalivation. Hyposalivation is an objective condition that refers to a lower salivary flow rate than normal, which may or may not be accompanied by xerostomia.^5,14

Patients with xerostomia have difficulty speaking, chewing, swallowing, tasting, and digesting, leading to weight loss.¹⁴ Xerostomia is associated with an increased risk of dental caries and bacterial and fungal infections in the oral cavity, affecting inpatients’ healing processes. Xerostomia not only has an impact on general health but also has a significant impact on the quality of life, both socially and emotionally.^{14,20,30–33} The etiology of xerostomia is multifactorial, including local factors, psychogenic factors, and underlying disease.^4,14

This patient complained of dry mouth, sore tongue, and difficulty eating. The patient was then asked to complete the SXI-ID questionnaire prior to the clinical examination. The SXI-ID is used by health services in Indonesia for the early detection of xerostomia. A score of over 11 on the SXI-ID questionnaire indicates xerostomia.³⁴ The SXI-ID score on this patient was 17, indicating xerostomia. The intraoral examination revealed that the mouth mirror was stuck to the buccal mucosa and dorsum of the tongue, there was no saliva pooling at the floor of the mouth, the tongue was depapillated, the gingiva was smooth, the oral mucosa and palate looked shiny, the tongue had fissures, and the patient had cervical caries. Based on these clinical findings, the patient was diagnosed with severe xerostomia with a Challacombe scale score of 8.

The patient was diagnosed with severe psoriasis vulgaris with psoriatic arthritis, chronic adrenal insufficiency, acute circulatory collapse, anemia of inflammation (AI), stage III acute kidney injury (AKI), dehydration, gastritis related to drugs (methotrexate and cyclosporine), urinary tract infections, and malnutrition by the Dermatovenereology Department and the Internal Medicine Department. Another possibility that could be explored during the anamnesis of dry mouth in this patient may be Sjögren’s syndrome, although the patient only complained of the oral region without genital and eye involvement. Further examination for Sjögren’s syndrome itself is needed but has not been done because the patient has passed away. The multiple systemic diseases in this patient may contribute to the xerostomia. Recognition of the multifactorial causes of xerostomia allows for an accurate diagnosis and more effective treatment.³⁵

Psoriasis vulgaris is the most common form of psoriasis. Clinically, the skin appears as thick, well-demarcated erythematous plaques of various sizes covered by silvery scales.^36,37 In addition to the skin, psoriasis manifestations can involve the ocular, nail, joint, and oral cavity.² Cutaneous psoriasis can appear before, during, or after an attack of psoriatic arthritis, with symptoms of joint stiffness, swelling, and pain.³⁶

Psoriasis with oral involvement is still controversial and cases are rarely reported. The main clinical features most commonly seen in psoriasis patients are a fissured tongue and a geographic tongue.^{10,12,13,16–19,36,38–40} The diagnosis of oral psoriasis is made when the clinical course of skin lesions coincides with oral lesions, which should preferably be confirmed histopathologically.^15,16,19,40 The manifestations of oral psoriasis in this patient were severe xerostomia, diffuse–type fissured tongue, exfoliative cheilitis, angular cheilitis, gingivitis, and periodontitis.

A recent study has shown salivary gland dysfunction and psoriasis vulgaris are directly related.⁴¹ Salivary gland dysfunction in psoriasis vulgaris patients is caused by inflammation and nitrosative stress. The inflammatory effect of psoriasis vulgaris may injure the salivary glands, affect saliva production, and increase the risk of xerostomia.⁴¹ The pathophysiology of psoriasis involves a chronic systemic inflammatory process that not only affects the skin but also the salivary glands. The salivary gland responds to the inflammation by increasing the amount of nitrogen oxide (NO) and peroxynitrate production as strong pro-apoptotic agents. Apoptosis of salivary gland structures can affect salivary gland function. The pro-apoptotic effect of NO on salivary glands leads to a reduction in salivary secretion. NO may inhibit the production of adenosine triphosphate (ATP), which is necessary to maintain anabolic processes in cells. As the amount of NO increases, an ATP deficiency occurs, which also disrupts the turnover mechanism of damaged cellular elements.^41–43

Psoriasis patients had chronic inflammation characterized by increased levels of C-reactive protein (CRP) and levels of inflammatory cytokines, including tumor necrosis factor-α (TNF-α), interferon-gamma (IFN-ᵞ), interleukin-2 (IL-2), interleukin-17 (IL-17), and interleukin-23 (IL-32).^2,44,45 TNF- α and IL-2 stimulate the production of metalloproteinases (MMPs), which can alter the structure of the salivary gland basement membrane. Excessive MMP production can damage the acinar basement cell membrane interactions and decrease the number of secretory units (acini and ducts). Salivary gland dysfunction occurs in psoriasis patients with a long duration of disease.⁴¹

Salivary gland dysfunction indicates reduced salivation (hyposalivation) or other quantitative changes in salivary secretion.¹⁴ Xerostomia is the perception of oral dryness caused by reduced or absent saliva flow.^4,46 Salivary gland dysfunction can be physiological or pathological. Salivary glands are involved due to multiple systemic diseases, with the resulting complication of xerostomia.^14,47 According to the literature, the underlying diseases that can cause xerostomia include endocrine diseases (diabetes mellitus, thyroid disease, adrenal gland disorders), viral infections (human immunodeficiency virus, Epstein-Barr virus, hepatitis C virus, cytomegalovirus, human T-lymphotropic virus type 1), bacterial infections (tuberculosis, actinomycosis), autoimmune diseases (rheumatoid arthritis, systemic lupus erythematosus, primary biliary cirrhosis, scleroderma, psoriasis), sarcoidosis, hemochromatosis, amyloidosis, kidney disease, ectodermal dysplasia, chronic graft-versus-host disease, and Parkinson’s disease.^{14,21,24,35,47–56}

The patient was diagnosed with chronic adrenal insufficiency, most commonly caused by the long-term use of exogenous corticosteroids. Adrenal insufficiency associated with a deficiency of all adrenocortical hormones (aldosterone, cortisol, and androgens) may increase the risk of xerostomia. Mechanisms of xerostomia associated with adrenal insufficiency include dehydration due to excessive sweating and salivary gland hypofunction due to adrenal conditions.^52,57 Endocrine disease and skin disease can lead to systemic collagen disease or metabolic disorders that may be a cause of salivary gland disease.⁵⁸

Anemia of inflammation (AI) in this patient may raise the risk of xerostomia due to changes in the epithelial tissue and oral mucous membranes.^59,60 Anemia reduces the oxygen-carrying capacity of the blood and leads to tissue hypoxia. AI, also known as anemia of chronic disease, is the most common type of anemia seen in hospitalized patients with chronic diseases. Common oral manifestations of anemia are angular cheilitis, depapilation of the tongue, pallor of the oral mucosa, and a burning sensation, which is found in this patient.⁶¹ These oral findings are due to impaired cellular immunity, poor bactericidal activity of polymorphonuclear leukocytes, an inadequate antibody response, and epithelial abnormalities.¹⁴ The laboratory test results in Table 1 show the patient’s anemia; the red blood cell count has decreased.

The condition of the oral cavity is directly related to kidney disease. One of the most common oral conditions in patients with kidney disease is xerostomia. The prevalence of xerostomia ranges from 33–56% in patients with kidney disease, compared to 0–29% in controls without kidney disease.¹⁴ The mechanisms of xerostomia in patients with kidney disease are decreased fluid intake, polyuria, stress, and salivary gland dysfunction. Many elderly patients with kidney disease tend to drink less, combined with a reduced ability of the kidneys to reabsorb sodium and polyuria, resulting in fluid loss and an increased risk of xerostomia. Stress and depression may reduce salivary gland activity in patients with kidney disease. Changes in the chemical composition of saliva, fibrosis, atrophy of minor salivary glands, and cell damage in the salivary glands contribute to an increased risk of xerostomia in patients with kidney disease.¹⁴

Cardiovascular disease (CVD) and kidney disease are closely related, and disease of one organ causes dysfunction of the other, ultimately leading to the failure of both organs.^14,62 Acute Kidney Injury (AKI) is strongly linked to the progression of CVD.⁶² The risk of xerostomia may be raised by the underlying disease, such as cardiovascular disease and renal disease, or by the drug intake for the disease.^{14,21,24,52–54}

The patient was diagnosed with moderate dehydration, which was exacerbated by low intake, increasing the risk of xerostomia. Dehydration is also an indirect causative factor for xerostomia.⁵⁸ Individual hydration levels may interfere with salivary secretion.⁶³ When the body is dehydrated, salivary flow decreases because the salivary glands reduce secretion to maintain the amount of water in the body.⁵⁸ When the body’s water content is reduced by 8%, salivary flow is almost zero. Even during dehydration, the salivary glands stop secreting saliva to conserve water.⁶³

Decreased fluid intake, impaired thirst mechanisms, and xerogenic drugs or beverages (alcohol and caffeine) can lead to dehydration.⁵⁸ Clinical signs of dehydration include oral and nasal dryness and longitudinal fissures on the tongue dorsum. If systemic dehydration is suspected, patients should be asked about their daily fluid intake and urine output (taking dehydrating beverages such as coffee and alcohol), their daily sodium intake, and xerogenic medications such as diuretics, laxatives, and antihistamines.¹⁴

Malnutrition may be a consequence of this patient’s underlying disease, gastritis. Malnutrition may also increase the risk of xerostomia. The changes in nutrition and deficiencies may affect salivary function. An increase in salivary protein could result from a modest decrease in daily calorie intake, whereas severe caloric restrictions reduce salivary flow, cell numbers, and salivary composition.⁶⁴ Another study described that individuals with xerostomia were found to have poor nutrition.⁵¹

Other possible factors that influence the xerostomia in this patient include drugs. Side effects of medication can cause xerostomia.¹⁴ There is evidence that taking numerous drugs may increase the risk of xerostomia.⁴⁷ Xerostomia in this patient may be caused by using methotrexate, cetirizine, loratadine, chlorpheniramine maleate, and omeprazole.^{4,5,8,14,65,66}

The multiple systemic diseases may contribute to the severity of xerostomia in this patient. The complications of systemic diseases in these patients may affect psychological conditions, cause emotional stress, and increase the risk of xerostomia. The systemic diseases may affect the patient’s ability to clean the oral cavity. The systemic diseases may worsen the patient’s condition, impede healing, and increase comorbidities.^41,67

After taking the prescribed medications for three days, the patient’s son claimed that his father was able to swallow food. Unfortunately, the patient developed septic shock due to hospital-acquired pneumonia (HAP) after five days in the hospital and eventually passed away. The patient’s son agreed and signed an informed consent form before this case report’s publication. The institution also consented to the publication of this case report.

Literature Review

For a better understanding of the factors that promote xerostomia, this case report also provides a review of other xerostomia cases that have been published between 2019 and 2023. The knowledge of the possible risk factors related to xerostomia is greatly enhanced by this review. Eleven cases of xerostomia were found and summarized in Table 2.

Table 2 Review of Xerostomia Case Reports

The results of the literature search yielded 3 articles from Indonesia,^68,71,77 2 articles from India,^69,75 2 articles from Brazil,⁷² 1 article from Iran,⁷⁰ 1 article from Australia,⁷³ 1 article from China,⁷⁴ and 1 article from the USA.⁷⁶ The age range of the reported xerostomia patients was the youngest at 7 years and the oldest at 85 years. The most reported patients in the literature review were 12 females and 9 males.

Based on the results of the literature review, factors that can affect xerostomia conditions include psychological stress, menopause, radiotherapy, chemotherapy, medication, COVID-19 vaccination, smoking, drinking alcohol, polyuria, and underlying diseases (carcinoma, COVID-19, gastroesophageal reflux disease, peptic ulcer disease, allergies, valvular heart disease, systemic scleroderma, hypercholesterolemia, Ewing’s sarcoma, Sjögren’s syndrome, hyponatremia, lacrimo-auriculo-dento-digital syndrome, human immunodeficiency virus, schizoaffective disorder, and hepatitis C). Patients with xerostomia who have systemic disease require collaboration with other disciplines besides oral medicine for successful treatment. Nine of the eleven case report articles in the review reported successful treatment.

Conclusion

Multiple systemic diseases may increase the risk and severity of xerostomia in this psoriasis patient. Multiple systemic diseases could affect xerostomia conditions such as, carcinoma, COVID-19, gastroesophageal reflux disease, peptic ulcer disease, allergies, valvular heart disease, systemic scleroderma, hypercholesterolemia, Ewing’s sarcoma, Sjögren’s syndrome, hyponatremia, lacrimo-auriculo-dento-digital syndrome, human immunodeficiency virus, schizoaffective disorder, and hepatitis C.