Letters to the Editor
125
Authors should devote considerable efforts in writing a good title for their scientific paper. P. Gjersvik,1,* M. Nylenna2 1 Department of Dermatology, Institute of Clinical Medicine, University of Oslo, 2Norwegian Knowledge Centre for Health Services, Oslo, Norway *Correspondence: P. Gjersvik. E-mail: [email protected]
Table 1 Laboratory tests N.Q Haemoglobin (g/dL) ASAT (IU/L)
1 Langdon-Neuner E. Titles in medical articles: what do we know about them? The Write Stuff 2007; 16: 158–160. 2 Gjersvik P. This is a good title. Tidsskr Nor Legeforen 2013; 133: 129. http://tidsskriftet.no/article/2964153. 3 Goodman NW. Survey of active verbs in the titles of clinical trial reports. BMJ 2000; 320: 914–915. 4 Ioannidis JPA. Why most published research findings are false. PLoS Medicine 2005; 2: e124. 5 Silver N. The signal and the noise. The art and science of prediction. Penguin Press, New York, NY, 2012.
9.8 78.2
8.6 77
ALAT (IU/L)
100
99
CPK (IU/L)
1075
290
>1000
>1000
FT3 (pmol/L)
0
0
FT4 (pmol/L)
0
0
99
553
Antitransglutaminase antibodies
Negative
Negative
Anti 21 hydroxylase antibodies
Negative
Negative
Anti-glutamic acid decarboxylase antibodies
Negative
Negative
TSHus (mIU/mL)
References
N.T
Antithyroperoxidase antibodies (IU/L)
ALAT, alanine aminotransferase; ASAT, aspartate aminotransferase; CPK, creatine phosphokinase; FT3, tree tri-iodothyronine; FT4, free tetra-iodothyronine.
DOI: 10.1111/jdv.12605
Autoimmune thyroiditis and isotretinoin: real association or coincidence? Editor Isotretinoin is widely used to treat severe acne and other skin diseases. The majority of its side-effects is well known and mainly includes dryness and disturbance of hepatic and lipid parameters. The association of this drug with autoimmune disease is rarely reported. N.Q and N.T are twins borne from non-consanguineous parents. Since the age of 1 year, they developed inflammatory acne conglobata initially treated with oral zinc (30 mg/day) and topical benzoyl peroxide without success. Systemic isotretinoin was then started at the age of 2 years at 0.5 mg/kg/day up to a cumulative dose of 140 mg/kg spread over 12 months. The treatment was remarkably efficient within the first month. Apart cheilitis, isotretinoin was well tolerated. Eighteen months after stopping treatment, the brothers complained of increased weakness, frequent falls, myalgia, constipation and lower reactivity in school. Clinical examination revealed a failure to thrive, skin pallor and hirsutism. Laboratory tests showed normocytic anaemia, rhabdomyolysis and deep peripheral autoimmune hypothyroidism (Table 1). Cervical ultrasound showed hypotrophy of the thyroid gland that indicated no fixation at the I-123 scintigraphy. No autoimmune disease was associated (Table 1). Hormone replacement with levothyroxine allowed a gradual amendment of the clinical
JEADV 2016, 30, 124–200
and biological disorders, notably with a recovery of normal growth and psychomotor development. Hypothyroidism is an unusual complication of isotretino€ıne. In 47 patients treated with isotretinoin for acne, Karadag et al.1 demonstrated a significant decrease in serum thyreostimulin hormone (TSH) and tree tri-iodothyronine after 3 months of treatment. However, no changes in serum levels of thyreoperoxydase antibodies were observed. Authors explained this central hypothyroidism by the retinoid 9 receptor mediated suppression of the TSHb gene expression. Hypothyroidism has also been reported with bexarotene, a derivative of vitamin A very similar to isotretinoin. The mechanism is both central and peripheral by increased degradation of thyroid hormones by a non-deiodinase-mediated pathway.2 Isotretinoin has an immunomodulatory effect through its role in apoptosis and activation of B and T lymphocytes.3 This effect was suspected to trigger some autoimmune diseases such as diabetes.4 Gursoy et al. reported the first case of autoimmune hypothyroidism associated with ocular myasthenia appeared after 6 months of treatment with isotretinoin in a 19-year-old patient with severe acne.5 Our twins had hereditary severe acne regarding to the simultaneous apparition of the disease, and to familial antecedents of acne. This treatment was particularly efficient with no clinical or biological abnormalities indicative of thyroid disorders during treatment. The mechanism by which isotretinoin may induce autoimmune thyroiditis remains uncertain. However, the occurrence of the autoimmune thyroiditis in the two brothers who received the same treatment, and the absence of a clear aetiological factor or autoimmune-associated disorder suggest that isotretinoin may be responsible through its immunomodulatory effect.3 Isotretinoin may have triggered an insidious autoimmune reaction that became symptomatic after treatment discontinuation.
© 2014 European Academy of Dermatology and Venereology
Letters to the Editor
126
This case confirms the efficacy of isotretinoin in children and highlights the importance of a careful research of autoimmune diseases in patients undergoing this treatment before the start and during follow-up. N. Guerouaz, M. Saint Jean, L. Peuvrel, B. Dreno* ^tel Dieu Hospital, Nantes, France Department of Dermatology, Ho *Correspondence: B. Dreno. E-mail: [email protected]
Table 6 of the original article that score of five points is accepted as ‘highly likely BD’.1 L. Tanrikulu Zekai Tahir Burak Women’s Health Training and Research Hospital, Ankara,Turkey Correspondence: L. Tanrikulu. E-mail: [email protected]
References References 1 Karadag AS, Ertugrul DT, Tutal E, Akin KO. Isotretinoin influences pituitary hormone levels in acne patients. Acta Derm Venereol 2011; 91: 31–34. 2 Smit JW, Stokkel MP, Pereira AM, Romijn JA, Visser TJ. Bexarotene-induced hypothyroidism: bexarotene stimulates the peripheral metabolism of thyroid hormones. J Clin Endocrinol Metab 2007; 92: 2496–2499. 3 Shapiro PE, Edelson RA. Effects of retinoids on the immune system. In Saturat JH, ed. Retinoids. New Trends in Research and Therapy. Karger, Basel, Switzerland, 1985: 225–235. 4 Dicembrini I, Bardini G, Rotella CM. Association between oral isotretinoin therapy and unmasked latent immuno-mediated diabetes. Diabetes Care 2009; 38: e99. 5 Gursoy H, Cakmak I, Yildirim N, Basmak H. Presumed isotretinoininduced, concomitant autoimmune thyroid disease and ocular myasthenia gravis: a case report. Case Rep Dermatol 2012; 4: 256–260.
1 International Team for the Revision of the International Criteria for Behcßet’s Disease (ICBD). The International Criteria for Behcßet’s Disease (ICBD): a collaborative study of 27 countries on the sensitivity and specificity of the new criteria. JEADV 2014; 28: 338–347. 2 Kural-Seyahi E, Fresko I, Seyahi N et al. The long-term mortality and morbidity of Behcßet syndrome: a 2-decade outcome survey of 387 patients followed at a dedicated center. Medicine (Baltimore) 2003; 82: 60–76. 3 Chattopadhyay A, Shetty KV. Recurrent aphthous stomatitis. Otolaryngol Clin North Am 2011; 44: 79–88. 4 Sardy M, Wollenberg A, Niedermeier A, Flaig MJ. Genital ulcers associated with Epstein-Barr virus infection (ulcus vulvae acutum). Acta Derm Venereol 2011; 91: 55–59. DOI: 10.1111/jdv.12608
DOI: 10.1111/jdv.12577
Scoring of international criteria for Behc ß et’s disease Editor I would like to thank International Team for the Revision of the International Criteria for Behcßet’s Disease (ICBD).1 As the mortality of the disease was reported higher in patients with the vascular and neurologic involvement,2 the addition of neurological and vascular manifestations to the diagnostic criteria in ICBD will permit early referral of these patients to expert centres. On the other hand, I have doubts about the false diagnosing of the disease with scoring four points or above. Prevalence of recurrent aphthous stomatitis derived from hospital-based studies was ranging from 1% to 66% among adults and 1% to 40% among children.3 Ulcus vulvae acutum is one of the most genital aphtosis I see in my daily practice. In addition to Epstein-Barr virus, Cytomegalovirus, Mycoplasma pneumonia and Influenza A virus have also been implicated in the pathogenesis of ulcus vulvae acutum.4 Many of these patients also have recurrent aphthous stomatitis. According to ICBD diagnostic criteria, they could be accepted as falsely having Behcßet’s Disease. In my opinion, accepting points not equal but higher than four would be better in proper diagnosis of Behcßet’s disease as mentioned in
JEADV 2016, 30, 124–200
Malignant form of atrophic papulosis with lethal abdominal involvement Editor Atrophic papulosis (K€ ohlmeier–Degos disease or Degos disease) is a rare occlusive arteriopathy involving small-calibre vessels of the dermis, gastrointestinal tract, central nervous system (CNS) and occasionally other organs.1,2 It would appear to be a vasculopathy or an endovasculitis, with a purely cutaneous benign variant and a systemic variant with cutaneous manifestations (malignant atrophic papulosis). Gastrointestinal involvement is the most frequent and lethal systemic complication.3 This is the most common cause of death, followed, less frequently, by CNS bleeding, and pleural or pericardial involvement.4 A 47-year-old woman was admitted to our Department because of a pruritic erythematous rash on the face, upper chest and arms. We noted over 100 erythematous dome-shaped papules of 5 10 mm in diameter that progressively developed central porcelain-like atrophic scars (Fig. 1c) surrounded by a telangiectatic rim involving the face, trunk, arms, lower limbs (Fig. 1a). The complete blood count, serum biochemistry analysis, kidney, liver and thyroid function tests and the erythrocyte sedimentation rate were all normal. ANA, ANCA, ENA, nDNA were all negative. Histopathological examination showed a widespread
© 2014 European Academy of Dermatology and Venereology
125
Authors should devote considerable efforts in writing a good title for their scientific paper. P. Gjersvik,1,* M. Nylenna2 1 Department of Dermatology, Institute of Clinical Medicine, University of Oslo, 2Norwegian Knowledge Centre for Health Services, Oslo, Norway *Correspondence: P. Gjersvik. E-mail: [email protected]
Table 1 Laboratory tests N.Q Haemoglobin (g/dL) ASAT (IU/L)
1 Langdon-Neuner E. Titles in medical articles: what do we know about them? The Write Stuff 2007; 16: 158–160. 2 Gjersvik P. This is a good title. Tidsskr Nor Legeforen 2013; 133: 129. http://tidsskriftet.no/article/2964153. 3 Goodman NW. Survey of active verbs in the titles of clinical trial reports. BMJ 2000; 320: 914–915. 4 Ioannidis JPA. Why most published research findings are false. PLoS Medicine 2005; 2: e124. 5 Silver N. The signal and the noise. The art and science of prediction. Penguin Press, New York, NY, 2012.
9.8 78.2
8.6 77
ALAT (IU/L)
100
99
CPK (IU/L)
1075
290
>1000
>1000
FT3 (pmol/L)
0
0
FT4 (pmol/L)
0
0
99
553
Antitransglutaminase antibodies
Negative
Negative
Anti 21 hydroxylase antibodies
Negative
Negative
Anti-glutamic acid decarboxylase antibodies
Negative
Negative
TSHus (mIU/mL)
References
N.T
Antithyroperoxidase antibodies (IU/L)
ALAT, alanine aminotransferase; ASAT, aspartate aminotransferase; CPK, creatine phosphokinase; FT3, tree tri-iodothyronine; FT4, free tetra-iodothyronine.
DOI: 10.1111/jdv.12605
Autoimmune thyroiditis and isotretinoin: real association or coincidence? Editor Isotretinoin is widely used to treat severe acne and other skin diseases. The majority of its side-effects is well known and mainly includes dryness and disturbance of hepatic and lipid parameters. The association of this drug with autoimmune disease is rarely reported. N.Q and N.T are twins borne from non-consanguineous parents. Since the age of 1 year, they developed inflammatory acne conglobata initially treated with oral zinc (30 mg/day) and topical benzoyl peroxide without success. Systemic isotretinoin was then started at the age of 2 years at 0.5 mg/kg/day up to a cumulative dose of 140 mg/kg spread over 12 months. The treatment was remarkably efficient within the first month. Apart cheilitis, isotretinoin was well tolerated. Eighteen months after stopping treatment, the brothers complained of increased weakness, frequent falls, myalgia, constipation and lower reactivity in school. Clinical examination revealed a failure to thrive, skin pallor and hirsutism. Laboratory tests showed normocytic anaemia, rhabdomyolysis and deep peripheral autoimmune hypothyroidism (Table 1). Cervical ultrasound showed hypotrophy of the thyroid gland that indicated no fixation at the I-123 scintigraphy. No autoimmune disease was associated (Table 1). Hormone replacement with levothyroxine allowed a gradual amendment of the clinical
JEADV 2016, 30, 124–200
and biological disorders, notably with a recovery of normal growth and psychomotor development. Hypothyroidism is an unusual complication of isotretino€ıne. In 47 patients treated with isotretinoin for acne, Karadag et al.1 demonstrated a significant decrease in serum thyreostimulin hormone (TSH) and tree tri-iodothyronine after 3 months of treatment. However, no changes in serum levels of thyreoperoxydase antibodies were observed. Authors explained this central hypothyroidism by the retinoid 9 receptor mediated suppression of the TSHb gene expression. Hypothyroidism has also been reported with bexarotene, a derivative of vitamin A very similar to isotretinoin. The mechanism is both central and peripheral by increased degradation of thyroid hormones by a non-deiodinase-mediated pathway.2 Isotretinoin has an immunomodulatory effect through its role in apoptosis and activation of B and T lymphocytes.3 This effect was suspected to trigger some autoimmune diseases such as diabetes.4 Gursoy et al. reported the first case of autoimmune hypothyroidism associated with ocular myasthenia appeared after 6 months of treatment with isotretinoin in a 19-year-old patient with severe acne.5 Our twins had hereditary severe acne regarding to the simultaneous apparition of the disease, and to familial antecedents of acne. This treatment was particularly efficient with no clinical or biological abnormalities indicative of thyroid disorders during treatment. The mechanism by which isotretinoin may induce autoimmune thyroiditis remains uncertain. However, the occurrence of the autoimmune thyroiditis in the two brothers who received the same treatment, and the absence of a clear aetiological factor or autoimmune-associated disorder suggest that isotretinoin may be responsible through its immunomodulatory effect.3 Isotretinoin may have triggered an insidious autoimmune reaction that became symptomatic after treatment discontinuation.
© 2014 European Academy of Dermatology and Venereology
Letters to the Editor
126
This case confirms the efficacy of isotretinoin in children and highlights the importance of a careful research of autoimmune diseases in patients undergoing this treatment before the start and during follow-up. N. Guerouaz, M. Saint Jean, L. Peuvrel, B. Dreno* ^tel Dieu Hospital, Nantes, France Department of Dermatology, Ho *Correspondence: B. Dreno. E-mail: [email protected]
Table 6 of the original article that score of five points is accepted as ‘highly likely BD’.1 L. Tanrikulu Zekai Tahir Burak Women’s Health Training and Research Hospital, Ankara,Turkey Correspondence: L. Tanrikulu. E-mail: [email protected]
References References 1 Karadag AS, Ertugrul DT, Tutal E, Akin KO. Isotretinoin influences pituitary hormone levels in acne patients. Acta Derm Venereol 2011; 91: 31–34. 2 Smit JW, Stokkel MP, Pereira AM, Romijn JA, Visser TJ. Bexarotene-induced hypothyroidism: bexarotene stimulates the peripheral metabolism of thyroid hormones. J Clin Endocrinol Metab 2007; 92: 2496–2499. 3 Shapiro PE, Edelson RA. Effects of retinoids on the immune system. In Saturat JH, ed. Retinoids. New Trends in Research and Therapy. Karger, Basel, Switzerland, 1985: 225–235. 4 Dicembrini I, Bardini G, Rotella CM. Association between oral isotretinoin therapy and unmasked latent immuno-mediated diabetes. Diabetes Care 2009; 38: e99. 5 Gursoy H, Cakmak I, Yildirim N, Basmak H. Presumed isotretinoininduced, concomitant autoimmune thyroid disease and ocular myasthenia gravis: a case report. Case Rep Dermatol 2012; 4: 256–260.
1 International Team for the Revision of the International Criteria for Behcßet’s Disease (ICBD). The International Criteria for Behcßet’s Disease (ICBD): a collaborative study of 27 countries on the sensitivity and specificity of the new criteria. JEADV 2014; 28: 338–347. 2 Kural-Seyahi E, Fresko I, Seyahi N et al. The long-term mortality and morbidity of Behcßet syndrome: a 2-decade outcome survey of 387 patients followed at a dedicated center. Medicine (Baltimore) 2003; 82: 60–76. 3 Chattopadhyay A, Shetty KV. Recurrent aphthous stomatitis. Otolaryngol Clin North Am 2011; 44: 79–88. 4 Sardy M, Wollenberg A, Niedermeier A, Flaig MJ. Genital ulcers associated with Epstein-Barr virus infection (ulcus vulvae acutum). Acta Derm Venereol 2011; 91: 55–59. DOI: 10.1111/jdv.12608
DOI: 10.1111/jdv.12577
Scoring of international criteria for Behc ß et’s disease Editor I would like to thank International Team for the Revision of the International Criteria for Behcßet’s Disease (ICBD).1 As the mortality of the disease was reported higher in patients with the vascular and neurologic involvement,2 the addition of neurological and vascular manifestations to the diagnostic criteria in ICBD will permit early referral of these patients to expert centres. On the other hand, I have doubts about the false diagnosing of the disease with scoring four points or above. Prevalence of recurrent aphthous stomatitis derived from hospital-based studies was ranging from 1% to 66% among adults and 1% to 40% among children.3 Ulcus vulvae acutum is one of the most genital aphtosis I see in my daily practice. In addition to Epstein-Barr virus, Cytomegalovirus, Mycoplasma pneumonia and Influenza A virus have also been implicated in the pathogenesis of ulcus vulvae acutum.4 Many of these patients also have recurrent aphthous stomatitis. According to ICBD diagnostic criteria, they could be accepted as falsely having Behcßet’s Disease. In my opinion, accepting points not equal but higher than four would be better in proper diagnosis of Behcßet’s disease as mentioned in
JEADV 2016, 30, 124–200
Malignant form of atrophic papulosis with lethal abdominal involvement Editor Atrophic papulosis (K€ ohlmeier–Degos disease or Degos disease) is a rare occlusive arteriopathy involving small-calibre vessels of the dermis, gastrointestinal tract, central nervous system (CNS) and occasionally other organs.1,2 It would appear to be a vasculopathy or an endovasculitis, with a purely cutaneous benign variant and a systemic variant with cutaneous manifestations (malignant atrophic papulosis). Gastrointestinal involvement is the most frequent and lethal systemic complication.3 This is the most common cause of death, followed, less frequently, by CNS bleeding, and pleural or pericardial involvement.4 A 47-year-old woman was admitted to our Department because of a pruritic erythematous rash on the face, upper chest and arms. We noted over 100 erythematous dome-shaped papules of 5 10 mm in diameter that progressively developed central porcelain-like atrophic scars (Fig. 1c) surrounded by a telangiectatic rim involving the face, trunk, arms, lower limbs (Fig. 1a). The complete blood count, serum biochemistry analysis, kidney, liver and thyroid function tests and the erythrocyte sedimentation rate were all normal. ANA, ANCA, ENA, nDNA were all negative. Histopathological examination showed a widespread
© 2014 European Academy of Dermatology and Venereology
