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CASE REPORT
Year : 2008  |  Volume : 29  |  Issue : 2  |  Page : 98-100
 

Bilateral nonhealing ulcers in groin: An interesting case of Metastatic Crohn's disease


Department of Skin & V. D. Medical College and S.S.G. Hospital, Vadodara, Gujarat, India

Correspondence Address:
Yogesh S Marfatia
OPD-1, Department of Skin and V.D., Medical College, Vadodara, Gujarat
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0253-7184.48735

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   Abstract 

Metastatic Crohn's disease is a rare granulomatous extraintestinal manifestation of Crohn's disease. We are reporting a case of a 22-year-old male with knife-cut ulcers in his groin, and a biopsy suggestive of Metastatic Crohn's disease.


Keywords: Knife-cut ulcers, Metastatic Crohn′s disease


How to cite this article:
Sarna J, Sharma A, Marfatia YS. Bilateral nonhealing ulcers in groin: An interesting case of Metastatic Crohn's disease. Indian J Sex Transm Dis 2008;29:98-100

How to cite this URL:
Sarna J, Sharma A, Marfatia YS. Bilateral nonhealing ulcers in groin: An interesting case of Metastatic Crohn's disease. Indian J Sex Transm Dis [serial online] 2008 [cited 2019 Nov 19];29:98-100. Available from: http://www.ijstd.org/text.asp?2008/29/2/98/48735



   Introduction Top


Crohn's disease is a granulomatous inflammatory disease, which affects the gastrointestinal (GI) tract, with the terminal ileum being the most commonly involved. Cutaneous involvement with Crohn's disease at a site separated from the GI tract by normal skin is termed as Metastatic Crohn's disease. It is a rare extraintestinal manifestation of Crohn's disease, with multiple clinical presentations. The lesions may parallel gastrointestinal disease activity or may occur with a totally separate temporal pattern. [1]


   Case report Top


A 22-year-old unmarried male presented with a history of progressively increasing swelling in the groin folds, followed by painful ulcers of two week's duration. There was no history of a similar episode previously.

There was no history of fever, nausea, vomiting, diarrhea, constipation, genital ulcer or discharge. On examination he had ulcers on both groin folds in a knife-cut pattern [Figure 1]. There was no evidence of skin tags, fissures, fistula or sinuses in the perineal area. The genital examination was normal. Both lymph nodes were enlarged and fine needle aspiration cytology (FNAC) was suggestive of reactive lymphadenitis.

The standard hematologic and biochemical workup was normal. The blood Venereal Disease Research Laboratory (VDRL) test and the Treponema pallidun hemagglutination assay (TPHA) and Human Immunodeficiency Virus (HIV) screening were also nonreactive. The chest X-ray was normal and there was no evidence of tuberculosis (TB)/sarcoidosis. The patient was given antibiotics such as, doxycycline and azithromycin after considering it as a sexually transmitted disease (STD). However, there was no response to the treatment. A biopsy was taken from the ulcer, which showed multinucleated giant cells and noncaseating granulomas [Figure 2]. On screening the literature diagnosis of Metastatic Crohn's disease was considered, which could also present with knife-cut ulcers. The diagnosis could be supported by endoscopic biopsy, but it was not feasible in our case. The present manifestation could be the forerunner of Crohn's disease.

We had put the patient on Prednisolone 30 mg/day, which was tapered over a period of six weeks and the ulcers healed completely [Figure 3] and [Figure 4]. In such patients it is necessary to screen them for future development of Intestinal Crohn's disease, but our patient did not turn up on long follow-up.


   Discussion Top


Cutaneous manifestations in Crohn's disease are present in 22 to 44% of the cases. [2] Three distinct patterns of cutaneous involvement are seen in Crohn's disease. The commonest cutaneous presentation occurs as a direct extension from the bowel, such as, perineal skin, stomal sites or lips. They manifest as fissures, fistula or anal tags. Oral Crohn's disease presents with a thickened corrugated appearance on the oral mucosa and lips. A second pattern includes dermatoses associated with Crohn's disease, such as, Pyoderma gangrenosum, erythema nodosum, erythema multiforme, epidermolysis bullosa acquisita, oral aphthae or skin changes secondary to malabsorption. [3] The third patten is Metastatic Crohn's disease defined as sterile granulomatous skin lesions arising at sites discontinued from the GI tract. The disease was first described by Parks et al , in 1965. [4] It can present as cutaneous ulcerations, plaques, papules or nodules over skin folds, the infra-mammary area or limbs. [5]

Metastatic Crohn's disease may present during active or quiescent periods of intestinal disease. [6] In adults it usually appears after the initial diagnosis of Crohn's disease in 70% of the cases, whereas, in children it appears at the same time as Crohn's disease in half of the cases. [7] However in our case, the patient did not have any past history suggestive of Crohn's disease. Metastatic Crohn's disease is a great imitator, as some granulomatous diseases like sarcoidosis, tuberculosis, and syphilis may present in a similar way. Hence a biopsy is essential to support the diagnosis. Emanuel PO et al , described non suppurative granuloma with a slight cuff of lymphocytes in a nodular or diffuse pattern with an associated superficial and deep perivascular mixed inflammatory infiltrate as the most common pattern observed among the 12 histopathological specimens of Metastaic Crohn's disease. [8]

In immunocompromised patients, Herpes simplex virus (HSV) may present as linear intertriginous fissures similar to the 'knife-cut' ulcers associated with metastatic Crohn's disease. [9]

There is no definite treatment of choice for Metastatic Crohn's disease. It is usually palliative and not curative. Many treatment modalities have been tried, such as, topical, intralesional, systemic steroids, sulfasalazine, mesalamine, oral metronidazole, hyperbaric oxygen, [10] and anti TNF-α antibodies (Infliximab). [11] In our patient, treatment with systemic steroids was very effective.

 
   References Top

1.Graham RM, Cox NH. Systemic Diseases and the Skin. In: Burns T, Breathnach S, Cox N, Griffiths C, editors. Rook's textbook of dermatology, 7th ed. Blackwell Science Limited; 2004. p. 59.29.   Back to cited text no. 1    
2.Goyal A, Mansel RE, Young HL, Douglas-Jones A. Metastatic cutaneous Crohn's disease of the nipple: Report of a case. Dis Colon Rectum 2006;49:132-4.   Back to cited text no. 2  [PUBMED]  [FULLTEXT]
3.Burgdorf W. Cutaneous manifestations of Crohn's disease. J Am Acad Dermatol 1981;5:689-95.   Back to cited text no. 3  [PUBMED]  
4.Parks AG, Morson BC, Pegum JS. Crohn's disease with cutaneous involvement. Proc R Soc Med 1965;58:241-2.  Back to cited text no. 4  [PUBMED]  [FULLTEXT]
5.Sangueza OP, Davis LS, Gourdin FW. Metastatic Crohn's disease. South Med J 1997;90:897-900.  Back to cited text no. 5    
6.Billings JK, Ellis CN, Hilgraum SS. Cutaneous granuloma formation in Crohn's disease. JAMA 1986;25:2261.  Back to cited text no. 6    
7.Palamaras I, El-JabbourJ, Pietropaolo N, Thomson P, Mann S, Robles W, et al . Metastatic Crohn's disease: A review. J Eur Acad Dermatol Venereol 2008;22:1033-43.  Back to cited text no. 7    
8.Emanuel PO, Phelps RG. Metastatic Crohn's disease: A histopathologic study of 12 cases. J Cutan Pathol 2008;35:457-61.  Back to cited text no. 8  [PUBMED]  [FULLTEXT]
9.Lieb JA, Brisman S, Herman S, Macgregor J, Grossman ME. Linear erosive herpes simplex virus infection in immunocompromised patients: The "knife-cut sign". Clin Infect Dis 2008;47:1440-1.  Back to cited text no. 9  [PUBMED]  [FULLTEXT]
10.Brady CE 3rd, Cooley BJ, Davis JC. Healing of severe perineal and cutaneous Crohn's disease with hyperbaric oxygen. Gastroenterology 1989;97:756-60.  Back to cited text no. 10  [PUBMED]  
11.Van Dullemen HM, de Jong E, Slors F, Tytgat GN, van Deventer SJ. Treatment of therapy-resistant perineal metastatic Crohn's disease after proctectomy using anti-tumor necrosis factor chimeric monoclonal antibody, cA2: Report of two cases. Dis Colon Rectum 1998;41:98-102.  Back to cited text no. 11  [PUBMED]  


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]


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