Seleit I, Attia A, Maraee A
… +3 more, Samaka R, Bakry O, Eid E
J Dermatol Case Rep
· 2011 Jun · PMID 21894251
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BACKGROUND: Kaposi sarcoma (KS) is a neoplasm of the endothelial cells. It often manifests with multiple vascular nodules on the skin and other organs. It is a systemic, malignant and multifactorial disease and has a var...BACKGROUND: Kaposi sarcoma (KS) is a neoplasm of the endothelial cells. It often manifests with multiple vascular nodules on the skin and other organs. It is a systemic, malignant and multifactorial disease and has a variable course. There are four types: classic, endemic, iatrogenic and HIV-associated. The primary presentation on the penis and face is uncommon and is mainly observed in HIV-positive patients. Multiple treatment modalities are used including surgery, cryotherapy, electrosurgery, laser and radiation therapy. MAIN OBSERVATION: The authors present two cases of isolated Kaposi sarcoma in HIV negative, human herpes virus 8 (HHV-8) positive non immunocompromised patients. One case with facial KS and the other one with penile KS. Both were treated surgically with no recurrence in the following 6 months of the follow up period. CONCLUSIONS: Kaposi sarcoma is rare in HIV negative patients and is associated with HHV-8 infection. Lesions are usually solitary and can be treated surgically. It should be included in the differential diagnoses of penile and facial lesions that are clinically suspecious and resistent to therapy.
Theiler M, Kempf W, Kerl K
… +2 more, French LE, Hofbauer GF
J Dermatol Case Rep
· 2011 Jun · PMID 21894250
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BACKGROUND: Molluscum contagiosum (MC) is a frequent and usually benign cutaneous infection with molluscum contagiosum virus (MCV), affecting mainly children and young sexually active adults. With the emergence of HIV/AI...BACKGROUND: Molluscum contagiosum (MC) is a frequent and usually benign cutaneous infection with molluscum contagiosum virus (MCV), affecting mainly children and young sexually active adults. With the emergence of HIV/AIDS, a new patient group at risk has been identified with often extensive skin involvement and recalcitrant disease. MAIN OBSERVATIONS: We report a case of a girl with connatal HIV-infection, suffering from extensive, disseminated MC. Due to multi-resistance, an effective antiretroviral therapy could not be established for years, rendering an effective treatment of MC by established treatment options virtually impossible. An off-label use of imiquimod showed a marked improvement of lesion counts in this patient, whereas a complete clearance could only be achieved once effective antiretroviral therapy was introduced. CONCLUSIONS: We believe that imiquimod may represent a valuable treatment option for molluscum contagiosum especially in the context of marked immunosuppression, where sensitive areas like the face and neck are often involved and scarring must be avoided.
J Dermatol Case Rep
· 2011 Mar · PMID 21886761
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Cutaneous myxoid fibroblastoma is an extremely rare soft tissue tumor, diagnosed based on histopathology. The histopathological triad for the diagnosis of the disease includes 1) spindle-shaped and/or stellate cells with...Cutaneous myxoid fibroblastoma is an extremely rare soft tissue tumor, diagnosed based on histopathology. The histopathological triad for the diagnosis of the disease includes 1) spindle-shaped and/or stellate cells with elongated nuclei and eosinophilic cytoplasm 2) distribution of cell in amyxoid stroma containing very fewcollagen fibers 3) cellular atypiawithout marked nuclear polymorphism in some cells. To date, only single cases of this disease have been reported. We report a new case and discuss the disease.
J Dermatol Case Rep
· 2011 Mar · PMID 21886760
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IFAP is an acronym for a rare congenital ectodermal disorder characterized by ichthyosis follicularis, alopecia and photophobia. A recessive X-linked mode of inheritance was initially proposed but recent reports in girls...IFAP is an acronym for a rare congenital ectodermal disorder characterized by ichthyosis follicularis, alopecia and photophobia. A recessive X-linked mode of inheritance was initially proposed but recent reports in girls suggested genetic heterogeneity of this syndrome. We herein describe a 1-year-old boy with clinical features typical of IFAP syndrome plus psoriasis-like lesions and palmoplantar keratoderma (PPK).
Masmoudi A, Chermi ZM, Marrekchi S
… +5 more, Raida BS, Boudaya S, Mseddi M, Jalel MT, Turki H
J Dermatol Case Rep
· 2011 Mar · PMID 21886759
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BACKGROUND: Cowden syndrome is a rare genodermatosis charactarized by presence of multiple hamartomas. The aim of the study was to specify the clinical, therapeutic and prognostic aspects of Cowden syndrome. CASES REPORT...BACKGROUND: Cowden syndrome is a rare genodermatosis charactarized by presence of multiple hamartomas. The aim of the study was to specify the clinical, therapeutic and prognostic aspects of Cowden syndrome. CASES REPORT: Our study included 4 patients with Cowden syndrome, 2 males and 2 females between 14 and 46 years old. Clinical examination of the skin revealed facials papules (4 cases), acral keratosis (1 case), translucent keratotic papules (2 cases). Oral examination revealed papules (4 cases), papillomatosis (4 cases), gingival hypertrophy (4 cases) and scrotal tongue (2 cases). Investigations revealed thyroid lesions (2 cases), fibrocystic disease and lipoma of the breast in 1 case, "glycogenic acanthosis" (1 case), macrocephaly (2 cases), dysmorphic face (1 case) and lichen nitidus (1 case). Oral etretinate and acitretine were temporary efficient in 2 patients. Topical treatment with tretinoin lotion resulted in some improvement in cutaneous, but not mucosal lesions in one patient. No cancer was revealed. CONCLUSION: The pathognomonic mucocutaneous lesions were found in all patients. However, no degenerative lesions have been revealed. A new association of Cowden syndrome with lichen nitidus was found. Treatment with oral retinoids was efficient on cutaneous lesions.
J Dermatol Case Rep
· 2011 Mar · PMID 21886758
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BACKGROUND: A review of scientific literature reveals a sparse documentation of periungual pyogenic granulomas as an adverse effect of isotretinoin therapy MAIN OBSERVATIONS: Periungual pyogenic granulomas appeared in fo...BACKGROUND: A review of scientific literature reveals a sparse documentation of periungual pyogenic granulomas as an adverse effect of isotretinoin therapy MAIN OBSERVATIONS: Periungual pyogenic granulomas appeared in four patients receiving isotretinoin therapy for severe acne. Oral and topical antibiotic treatments were ineffective and all cases spontaneously resolved once isotretinoin is discontinued. CONCLUSION: This report demonstrates the idiosyncratic nature of this side effect. We suggest that, although infection is possible due to the transformed nature of the normally protective skin barrier, oral and topical antibiotic treatments appear ineffective. Pyogenic granulomas spontaneously resolve once isotretinoin is discontinued.
Apalla Z, Chloros D, Argyropoulou P
… +4 more, Georgopoulos D, Patakas D, Spanos D, Chaidemenos G
J Dermatol Case Rep
· 2011 Mar · PMID 21886757
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BACKGROUND: Pemphigus vulgaris belongs to a group of rare, autoimmune, blistering disorders, requiring intensive immunosuppressive therapy. Lung infections are common in patients with pemphigus vulgaris. OBJECTIVE: To ev...BACKGROUND: Pemphigus vulgaris belongs to a group of rare, autoimmune, blistering disorders, requiring intensive immunosuppressive therapy. Lung infections are common in patients with pemphigus vulgaris. OBJECTIVE: To evaluate pulmonary function in patients with pemphigus vulgaris, in order to detect possible factors that might predispose PV patients to lung damage. METHODS: Ten patients first diagnosed with severe pemphigus vulgaris and 10 healthy individuals,were included in the study. Pulmonary function testing, blood gas analysis and quantification of α1-antitrypsin serum levels were performed. RESULTS: Mild reduction of CO diffusing capacity was recorded, compared to the mean predicted normal value. DLCOSB (single-breath carbon monoxide diffusing capacity test) values did not significantly differ between patients with pemphigus vulgaris and healthy controls, while differences regarding DLCO/VA (VA: Alveolar Volume) were statistically significant. Alpha 1-antitrypsin serum levels were decreased (<2.0g/L) in 60% (6/10) of patients with pemphigus vulgaris and were found normal in none of the healthy controls. CONCLUSION: A mild reduction of pulmonary diffusing capacity was observed in patients with pemphigus vulgaris.
Samdani AJ, Azhar A, Shahid SM
… +6 more, Nawab SN, Shaikh R, Qader SA, Mansoor Q, Khoso BK, Ismail M
J Dermatol Case Rep
· 2010 Dec · PMID 21886756
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BACKGROUND: The extracellular matrix protein 1 (ECM1) is a glycoprotein, expressed in skin and other tissues. Loss-of-function mutation in ECM1 causes a rare autosomal recessive disorder called lipoid proteinosis. Lipoid...BACKGROUND: The extracellular matrix protein 1 (ECM1) is a glycoprotein, expressed in skin and other tissues. Loss-of-function mutation in ECM1 causes a rare autosomal recessive disorder called lipoid proteinosis. Lipoid proteinosis is presented by varying degrees of skin scars, beaded papules along the eyelid margins, variable signs of hoarseness of voice and respiratory disorders. More than 250 cases of this disorder have been described in the literature, but occurrence of lipoid proteinosis in siblings is very rare. This study was designed to investigate the possible mutation causing lipoid proteinosis in a Pakistani family and to elaborate the scope of possible genetic changes, causing the genodermatosis in Pakistan. MAIN OBSERVATIONS: In this study, two siblings (12 and 9-years sisters) were presented with scaly itchy lesions on whole body, hoarse voice and macroglossia. Their deceased father had similar clinical manifestations but mother and younger brother were unaffected. Blood samples from clinically affected and unaffected family members were collected with informed consent. The coding region of ECM1 gene containing 10 exons were amplified and sequenced. Both the affected siblings were shown to have homozygous frame shift mutation by deletion of the nucleotide T at 507, codon 169, exon 6. This resulted in a frame shift from codon 169 and appearance of a premature stop codon at 177, causing formation of a mutated protein (176 amino acids) instead of normal ECM1 protein (540 amino acids). CONCLUSION: A case of homozygous 62-bp insertion in ECM1 gene causing lipoid proteinosis has been reported in another Pakistani family. The current study presents a homozygous frame shift mutation supporting an unusual function of ECM1 protein and broadens the spectrum of disease-linked mutations in this rare case of genodermatosis in this region.
J Dermatol Case Rep
· 2010 Dec · PMID 21886755
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BACKGROUND: Molluscum contagiosum, is a common skin infection caused by a pox virus usually present as raised nodule with umbilicated centre. Cytologic diagnosis has been documented in very few case reports as the lesion...BACKGROUND: Molluscum contagiosum, is a common skin infection caused by a pox virus usually present as raised nodule with umbilicated centre. Cytologic diagnosis has been documented in very few case reports as the lesion is subjected to fine needle aspiration rarely and awareness of the cytological features of Molluscum contagiosum is limited. The cytological diagnosis is further challenging in clinically unsuspected lesions. MAIN OBSERVATIONS: A 45-year-old female patient presented with 10 months history of a nodular nipple lesion which ulcerated after local application of caustic pencil. There was no associated breast lump. Cheesy material was expressed from the ulcer on the nipple and crush smeared on slides for cytological evaluation. Cytology smears revealed nucleate and anucleate squames in an inflammatory background. Characteristic intracytopalsmic and extracytopalsmic molluscum bodies were seen. CONCLUSION: The nipple is an unusual site of presentation for Molluscum contagiosum. Ulcerated lesions are rarer. In the present case, the cytological examination facilitated the diagnosis in a clinically unsuspected case. Cytology can be a useful rapid diagnostic aid in planning the management of these patients and help in avoiding unnecessary biopsies.
Chularojanamontri L, Tuchinda P, Kulthanan K
… +1 more, Manuskiatti W
J Dermatol Case Rep
· 2010 Dec · PMID 21886754
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BACKGROUND: Diphencyprone is a universal contact immunotherapy. The mechanism of action is based on an induction of the delayed-type hypersensitivity. Diphencyprone has been used in various forms for treatments of recalc...BACKGROUND: Diphencyprone is a universal contact immunotherapy. The mechanism of action is based on an induction of the delayed-type hypersensitivity. Diphencyprone has been used in various forms for treatments of recalcitrant and facial warts, and alopecia areata. However, this treatment modality has not been generally used in immunocompromised patients. MAIN OBSERVATION: The present report demonstrated the efficacy of diphencyprone immunotherapy on the treatment of generalized molluscum contagiosum in a human immunodeficiency virus (HIV)-infected patient. Minimal and transient side effects including pruritus, postinflammatory hyperpigmentation and irritation were noted. CONCLUSION: Diphencyprone contact immunotherapy appears to be a possible alternative treatment of widespread molluscum contagiosum in immunocompromised patients.
Ishibashi M, Tojo G, Watanabe M
… +3 more, Tamabuchi T, Masu T, Aiba S
J Dermatol Case Rep
· 2010 Dec · PMID 21886753
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BACKGROUND: Geographic tongue, or benign migratory glossitis, is usually an asymptomatic inflammatory disorder of the tongue mucosa of unknown etiology. It is characterized by circinate, erythematous, ulcer-like lesions...BACKGROUND: Geographic tongue, or benign migratory glossitis, is usually an asymptomatic inflammatory disorder of the tongue mucosa of unknown etiology. It is characterized by circinate, erythematous, ulcer-like lesions of the dorsum and lateral border of the tongue due to loss of filiform papillae of the tongue epithelium. Symptomatic treatments have not been evaluated rigorously. MAIN OBSERVATION: We describe herein two cases of adult patients with persistent and painful geographic tongue successfully treated with topical application of 0.1% tacrolimus ointment. CONCLUSION: To our knowledge, this is the first report of successful treatment with topical 0.1% tacrolimus for symptomatic geographic tongue. Clinical trials are needed to confirm the efficacy and the safety of topical tacrolimus in treating geographic tongue.
J Dermatol Case Rep
· 2010 Dec · PMID 21886752
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BACKGROUND: Various atypical variants of pityriasis rosea were published in the literature. There are very few reports abour pityriasis rosea being unilateral. MAIN OBSERVATIONS: We present a female child who presented w...BACKGROUND: Various atypical variants of pityriasis rosea were published in the literature. There are very few reports abour pityriasis rosea being unilateral. MAIN OBSERVATIONS: We present a female child who presented with acute onset of scaly eruptions clinically consistent with a diagnosis of pityriasis rosea, affecting only one side of the body involving waist, thigh, forearm and leg on right side. CONCLUSIONS: It is extremely unusual for pityriasis rosea to present the eruptions only on one side of the body. This case is presented here for its rarity in clinical practice.
J Dermatol Case Rep
· 2010 Dec · PMID 21886751
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BACKGROUND: Schnitzler syndrome encompasses monoclonal gammopathy, urticaria, inflammation, recurrent fever, bone pain and arthralgia, with occasional lymphadenopathy and/or hepatosplenomegaly. It is a rare condition wit...BACKGROUND: Schnitzler syndrome encompasses monoclonal gammopathy, urticaria, inflammation, recurrent fever, bone pain and arthralgia, with occasional lymphadenopathy and/or hepatosplenomegaly. It is a rare condition with approximately 100 cases reported in the literature. To our knowledge, this is the first reported case of cold-induced physical urticaria with Schnitzler syndrome. MAIN OBSERVATIONS: A 43-year-old woman presented to an allergy and immunology clinic with a 7 year history of chronic urticaria, angioedema with anaphylaxis, monoclonal gammopathy and bone pain. Her urticaria was triggered by cooler temperatures and an ice cube test for cold induced urticaria was positive. In spite of aggressive therapies this patient remains symptomatic. CONCLUSIONS: Schnitzler syndrome is under-recognized, particularly variants of the classical description of Schnitzler syndrome. Other diseases, especially those of hematologic origin, should be ruled out. This condition is largely refractory to conventional therapies, although novel treatments, such as interleukin-1 receptor inhibitor, may show promise.
Matthiesen C, Thompson S, Ahmad S
… +2 more, Herman T, Bogardus C
J Dermatol Case Rep
· 2010 Dec · PMID 21886750
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BACKGROUND: Patients with locally advanced skin cancer often present an uncommon and unique treatment challenge. Surgical resection and reconstruction with an acceptable cosmetic outcome is difficult for larger lesions w...BACKGROUND: Patients with locally advanced skin cancer often present an uncommon and unique treatment challenge. Surgical resection and reconstruction with an acceptable cosmetic outcome is difficult for larger lesions with deep infiltration into subcutaneous tissues. Radiation therapy has been shown to be an effective treatment modality for advanced non-melanoma skin cancers, with cure rates ranging 50-100%. In this case report, we discuss the efficacy and outcome of treatment using an advanced radiation therapy technique to a large T4 squamous cell carcinoma of the face. MAIN OBSERVATIONS: The patient responded favorably to the advanced radiation treatment course, and achieved a clinical complete response to therapy. No further intervention was required. Advanced radiation therapy techniques offered treatment advantages that resulted in greater tumor dose escalation and minimizing of patient morbidity. CONCLUSIONS: Patients with advanced skin cancers of the head and neck should be considered for definitive radiation therapy using advanced treatment techniques. The use of definitive RT only for tumors deemed unresectable, or for inoperable patients at presentation deserves reconsideration. Further investigation is warranted.
J Dermatol Case Rep
· 2010 Dec · PMID 21886749
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BACKGROUND: New targeted therapies have been developed for inflammatory and neoplastic diseases. MAIN OBSERVATION: We report on a 73-year-old woman who developed multiple eruptive periungual and subungual pyogenic granul...BACKGROUND: New targeted therapies have been developed for inflammatory and neoplastic diseases. MAIN OBSERVATION: We report on a 73-year-old woman who developed multiple eruptive periungual and subungual pyogenic granulomas. Because of severe rheumatoid arthritis the patient was treated with monoclonal anti-CD20 antibodies. Eruptive granuloma pyogenicum developed after the second antibody application and remained more than 8 weeks after targeted therapy was over. New lesions, however, did not appear. CONCLUSION: Eruptive granuloma pyogenicum of the nail apparatus is a possible new rare adverse effect of targeted therapies. To the best of our knowledge this is the first case in association with anti-CD20 antibody treatment.
Sherlin HJ, Ramalingam K, Natesan A
… +3 more, Ramani P, Premkumar P, Thiruvenkadam C
J Dermatol Case Rep
· 2010 Dec · PMID 21886748
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BACKGROUND: Lichen sclerosus is a chronic muco-cutaneous inflammatory disorder of uncertain etiology. The prevalence of lichen sclerosus affecting only the oral mucosa is extremely rare and only 26 cases with histologica...BACKGROUND: Lichen sclerosus is a chronic muco-cutaneous inflammatory disorder of uncertain etiology. The prevalence of lichen sclerosus affecting only the oral mucosa is extremely rare and only 26 cases with histologically verified oral lichen sclerosus have been reported. MAIN OBSERVATIONS: A 60-year-old man was referred to our hospital for complaints of white lesions in the oral cavity, which was otherwise asymptomatic and did not have any cutaneous or anogenital lesions. Clinical examination revealed white patches with reddish areas on the buccal mucosa bilaterally and on the maxillary posterior gingiva. Microscopic analysis of the lesion showed atrophied epithelium with basal cell degeneration, hyalinized connective tissue stroma with minimal chronic inflammatory cell infiltrate. Verhoeff's staining revealed scantiness of elastic fibers in the connective tissue stroma. On the basis of these histological findings, the final diagnosis was given as Lichen Sclerosus. LS is rare in the oral cavity, particularly in the absence of simultaneous cutaneous and anogenital lesions. CONCLUSIONS: Only 7 cases of oral LS have been reported involving the gingiva. To our knowledge, this is the eigth case to be reported with gingival involvement and the first case to be reported with bilateral involvement of buccal mucosa and gingiva.
Ramot Y, Maly A, Zlotogorski A
… +1 more, Nanova K
J Dermatol Case Rep
· 2010 Dec · PMID 21886747
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BACKGROUND: The allergic crease (nasal crease) is a transverse line, common among patients who suffer from allergic rhinitis. Although it is a very prevalent condition, the skin manifestations which accompany this condit...BACKGROUND: The allergic crease (nasal crease) is a transverse line, common among patients who suffer from allergic rhinitis. Although it is a very prevalent condition, the skin manifestations which accompany this condition have been only sparsely reported. MAIN OBSERVATION: We describe an 18-year-old man with allergic rhinitis, who developed cornified papules along an allergic crease. Dermoscopic examination revealed oval, slightly raised, gray, sharply demarcated papules. Histopathology revealed seborrheic keratosis like hyperplasia, accompanied by horn cysts. CONCLUSIONS: To our knowledge, cornified papules have not been reported previously along the allergic crease. Considering the fact that allergic rhinitis is a common chronic condition of childhood, increased awareness of the skin manifestations which accompany this condition is essential.
Nantel-Battista M, Al Dhaybi R, Hatami A
… +3 more, Marcoux D, Desroches A, Kokta V
J Dermatol Case Rep
· 2010 Dec · PMID 21886746
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BACKGROUND: Linear IgA bullous disease (LABD) is a rare mucocutaneous autoimmune subepidermal blistering disease that can affect children mostly of pre-school age. As many as two-thirds of LABD are related to drug ingest...BACKGROUND: Linear IgA bullous disease (LABD) is a rare mucocutaneous autoimmune subepidermal blistering disease that can affect children mostly of pre-school age. As many as two-thirds of LABD are related to drug ingestion, particularly certain antibiotics, non-steroidal anti-inflammatory drugs and diuretics. MAIN OBSERVATION: We describe a 3-year-old boy who presented a CMV infection followed by LABD induced by trimtheporim-sulfametoxazole. To our knowledge, this is the first reported case of trimethoprim-sulfamethoxazole that was confirmed by a rechallenge. CONCLUSIONS: Most cases of drug-induced LABD are patients being treated with multiple systemic drugs that could induce the LABD. In the lack of suitable alternative treatment, the identification of the causative drug can be achieved by a rechallenge under close medical surveillance.
Lazaridou E, Giannopoulou C, Apalla Z
… +3 more, Fotiadou C, Trigoni A, Ioannides D
J Dermatol Case Rep
· 2010 Nov · PMID 21886745
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BACKGROUND: Parry-Romberg syndrome (PRS) or idiopathic hemifacial atrophy is a rare neurocutaneous syndrome. It is characterized by slowly progressive atrophy, located on one side of the face, primarily involving the ski...BACKGROUND: Parry-Romberg syndrome (PRS) or idiopathic hemifacial atrophy is a rare neurocutaneous syndrome. It is characterized by slowly progressive atrophy, located on one side of the face, primarily involving the skin, fat and connective tissue. PRS seems to overlap with "en coupe de sabre" morphea. MAIN OBSERVATIONS: We present a case of hemifacial atrophy in a 14-year-old boy treated with topical calcipotriol-betamethasone ointment. The diagnosis of PRS was established mainly based on the clinical findings and histological picture. The time to diagnosis was almost 9 years, similar to the mean time reported in the literature. CONCLUSIONS: Understanding the pathogenesis and stopping disease progression is important as it can cause severe disfigurement and has neurological and psychiatric complications. Not much is known about the efficacy of agents used in the treatment of this syndrome making treatment decision very difficult. Possible complications, pathophysiology and therapeutic options are being discussed.
J Dermatol Case Rep
· 2010 Nov · PMID 21886744
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BACKGROUND: Cinnamon contact stomatitis (CCS) is a rare reaction to the use of products containing artificial cinnamon flavor ingredients. Such products are gums, toothpastes and mouthwashes. MAIN OBSERVATIONS: A 20-year...BACKGROUND: Cinnamon contact stomatitis (CCS) is a rare reaction to the use of products containing artificial cinnamon flavor ingredients. Such products are gums, toothpastes and mouthwashes. MAIN OBSERVATIONS: A 20-year-old female patient presented with white elevated mucosal patches in the right lateral board of her tongue. Based on anamnesis, the intitial diagnosis of allergy to cinnamon gum was established. Clinical differential diagnosis included hairy leukoplakia, leukoplakia and lichenoid reaction. The patient was advised to completely avoid the use of cinnamon flavoured chewing gums. On re-examination later she had a normal tongue appearance. CONCLUSIONS: Clinicians who treat patients with oral conditions should be aware of CCS in order to be able to correctly diagnose and manage this condition.