An Atypical Presentation of Erythema Induratum
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Letters to Editor Address for correspondence: Wg Cdr Sandeep Arora, Skin diagnosis of EN. Skin biopsy from a leg nodule revealed Department, 5 AFH, c/o 99 APO, India. lobular panniculitis with caseous necrosis, epithelioid cell E-mail: [email protected] granulomas, and Langhans giant cells [Figure 1]. Small REFERENCES vessel vasculitis with extravasation of RBCs, fibrinoid degeneration of vessel wall, and karyorrhexis were also 1. Saoji VA. Hand, foot and mouth disease in Nagpur. Indian J present. The histopathology was consistent with EI. Dermatol Venereol Leprol 2008;74:133-5. Findings from her hemogram, liver and renal function 2. Frydenberg A, Starr M. Hand, foot and mouth disease. Aust tests, urine and stool examination, anti-nuclear antibody, Fam Physician 2003;32:594-5. anti-streptolysin O titer were within normal limits. Chest 3. Chang LY, Lin TY, Hsu KH, Huang YC, Lin KL, Hsueh C, et x-ray was normal, but findings from Mantoux test was al. Clinical features and risk factors of pulmonary edema strongly positive (45 mm induration with necrosis). after enterovirus-71-related hand, foot, and mouth disease. Lancet 1999;354:1682-6. 4. Podin Y, Gias EL, Ong F, Leong YW, Yee SF, Yusof MA, et al. Based on these findings, we initiated four-drug antitubercular Sentinel surveillance for human enterovirus 71 in Sarawak, therapy (ATT). Within 3 weeks, the lesions resolved and no Malaysia: lessons from the first 7 years. BMC Public Health new lesions appeared. ATT was continued for 6 months. 2006;6:180. 5. Sasidharan CK, Sugathan P, Agarwal R, Khare S, Lal S, The clinical manifestation of recurrent tender subcutaneous Jayaram Paniker CK. Hand-foot-and-mouth disease in Calicut. erythematous nodules on extensors of limbs that Indian J Pediatr 2005;72:17-21. resolved with hyperpigmentation suggested EN. However, histopathology revealed granulomatous lobular panniculitis with vasculitis, which was consistent with EI. An underlying An atypical presentation of tubercular focus was suggested by a strongly positive Mantoux reaction and prompt clearance of lesions without erythema induratum any recurrence with ATT. EN and EI are facultative tuberculids in which M. tuberculosis/ Sir, bovis may be one of the several etiopathogenic factors. They Erythema induratum (EI) and erythema nodosum (EN) are are the most commonly encountered panniculitides in daily diverse entities in the group of panniculitides with distinct practice. In majority of the cases, clinical differentiation clinicopathological features. They have varying etiologies, between EN and EI is possible.[1,2] EN is usually bilateral, of which tuberculosis is common to both. In majority of with lesions developing on shins, knees, and ankles; the patients, by meticulous history-taking, cutaneous and and occasionally on the face, neck, trunk, arms, and systemic examination, a fairly accurate clinical diagnosis thighs; while EI occurs more commonly on calves on the can be made, which is further confirmed by histopathology. background of erythrocyanotic circulation. Lesions of EN We present an unusual case of EI that clinically mimicked EN. The diagnosis was confirmed histologically. A 40-year-old woman presented with recurrent crops of erythematous subcutaneous tender nodules on bilateral shins, arms, and forearms since 4 months, each crop subsiding within 2 to 4 weeks, leaving behind hyperpigmentation. It was not associated with constitutional features or systemic complaints. There was no personal or family history of tuberculosis. On examination, she had multiple, discrete, erythematous, tender subcutaneous nodules, 1 to 2 cm in size; and dark- to light-brown post-inflammatory hyperpigmented macules on bilateral shins, extensors of both arms, and forearms. The results of systemic examination were within normal Figure 1: Lobular granulomatous panniculitis with infiltrate limits. There was no lymphadenopathy, pedal edema, or of epithelioid cells, Langhan’s giant cells, lymphocytes and evidence of erythrocyanotic circulation. We made a clinical histiocytes (H & E, ×100) Indian J Dermatol Venereol Leprol | September-October | Vol 74 | Issue 5 505 Letters to Editor never ulcerate, and they subside with hyperpigmentation Address for correspondence: Dr. Sujay Khandpur, Department of within 3 to 6 weeks, followed by recurrences; while EI Dermatology and Venereology, All India Institute of Medical Sciences, New Delhi - 110 029, India. E-mail: [email protected] nodules usually ulcerate, resolve with atrophic scars, and have a more protracted course. The clinical diagnosis is REFERENCES further supplemented by a characteristic histopathology, which in fully developed EN is a septal panniculitis without 1. Requena L, Sanchez YE. Panniculitis: Part II. J Am Acad vasculitis with fibrosis of septa and mixed inflammatory Dermatol 2001;45:325-61. infiltrate of lymphocytes, histiocytes, multinucleate giant 2. Requena L, Yus ES. Panniculitis, Part I: Mostly septal cells, neutrophils and eosinophils, and Miescher’s radial panniculitis. J Am Acad Dermatol 2001;45:163-83. 3. Thurber S, Kohler S. Histopathologic spectrum of erythema granulomas. Interestingly acute EN may show features nodosum. J Cutan Pathol 2006;33:18-26. of lobular panniculitis or even small- and medium-vessel 4. Sanz Vico MD, De Diego V, Sanchez Yus E. Erythema vasculitis.[3] EI reveals granulomatous lobular panniculitis nodosum versus nodular vasculitis. Int J Dermatol with or without vasculitis. 1993;32:108-12. 5. Chew GY, Henderson C, Ouin JW. Erythema induratum: A Atypical forms of EI have been described chiefly in men, case of mistaken identity. Med J Aust 2005;183:534. 6. Neimi KM, Forstrom M, Hannuksela M, Mustakallio KK, including unilateral lesions; lesions involving the shins, Salo OP. Nodules on the legs: A clinical, histological and thighs, buttocks, arms, and other sites; and those resolving immunohistological study of 82 patients representing [1] without ulceration. Sanz Vico et al, reported 9 cases of different types of nodular panniculitis. Acta Derm Venereol atypical nodular vasculitis in which the clinical presentation 1977;57:145-54. included 2 nodules or 1 plaque or lesions that resolved spontaneously without ulceration or scarring.[4] Chew GY et al, described erythematous painful nodules on the thighs and buttock of a 29-year-old woman, mimicking Pyoderma gangrenosum affecting EN clinically, but biopsy revealed lobular granulomatous the vulva panniculitis.[5] The lesions cleared within 2 months of ATT. Our patient represented one such atypical variant of EI. Sir, Inflammatory nodules occurring on the extremities should Nonvenereal vulvar ulcers often pose a diagnostic and also be differentiated from cutaneous polyarteritis nodosa, therapeutic challenge to the clinician. Behcet’s disease, superficial thrombophlebitis, subcutaneous sarcoidosis, and Crohn’s disease, pyoderma gangrenosum, premalignant lymphoma.[1] Niemi et al, described a group of non-definite or malignant ulcers, traumatic and factitial ulceration may panniculitis with large, diffuse, indurated subcutaneous involve the vulva. plaques without ulceration that healed with scars or pits, and some cases with small acral nodules with or without A 48-year-old married woman presented with multiple ulceration.[6] Histology revealed chiefly lobular panniculitis painful ulcers over the genitalia, of 3 months’ duration; with granulomatous infiltrate and caseation or palisaded and oral aphthae for the past 1 month. Lesions started granulomas with/without vasculitis and occasionally, only as erythematous papules rupturing to form painful ulcers septal panniculitis. Of those who received complete course with serosanguinous discharge. There was no history of of ATT, 67% showed no recurrence while lesions reappeared vesiculation, itching, vaginal discharge, joint pain or eye in the remaining cases. None of their EI cases on ATT complaints. Findings of general physical examination were developed recurrence. unremarkable except for pallor. The present case was an atypical presentation of EI that Genital examination showed large kissing ulcers involving both labia majora and labia minora. Ulcers were tender clinically mimicked EN. The purpose of this report is to re- with undermined edges and indurated base, which were emphasize the diverse clinical presentations of EI. covered with slough and hemorrhagic crusts [Figure 1]. A few superficial ulcers were present on both thighs, with size Sujay Khandpur, Pradeep K. Sethy, varying from 1 to 3 cm in diameter and with undermined Vinod K. Sharma, Prasenjit Das1 Departments of Dermatology and Venereology, and 1Pathology, All edges, and were covered with slough. Bilateral inguinal India Institute of Medical Sciences, New Delhi-110 029, India. lymph nodes were enlarged and non-tender. 506 Indian J Dermatol Venereol Leprol | September-October | Vol 74 | Issue 5.