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A Life-Threatening Multilocalized Hidradenitis Suppurativa Case

Identifieur interne : 008060 ( Main/Exploration ); précédent : 008059; suivant : 008061

A Life-Threatening Multilocalized Hidradenitis Suppurativa Case

Auteurs : Oktay Buyukasik ; C. Gokhan Osmanoglu ; Yilmaz Polat ; Hulagu Kargici ; Gulay Kaya

Source :

RBID : PMC:1681757

Abstract

Abstract and IntroductionAbstract

The patient was a 38-year-old man. He had been suffering from hidradenitis suppurativa (HS) for approximately 20 years. He had active lesions at both axillas, hip, scrotum, and perineum, and inactive lesions located behind the ears, lower abdomen, and posterior neck. He was monitored and treated at different branches; he continuously used antibiotics and was given steroids at times. Antibiotic resistance developed subsequently. His general situation was bad; vital signs were poor; and he was in a state of sepsis and preshock, so this case was regarded as life-threatening.

Total excision was performed first on the lesion at the right axilla, then on the lesion at the left axilla, and the parascapular fasciocutaneous flap was reversed. A skin graft was applied to the triangular defect on the scapula. No relapse occurred. Then the lesions at the hip were managed. Broad excision was used twice with the patient under general anesthesia; because the lesions spread to the retrococcygeal and gluteal muscles, coccyx resection and partial gluteal muscle resection were implemented. The defect was eliminated with a progressive flap. At the intergluteal sulcus, small lesions emerging at the median line were debrided with the patient under local anesthesia, and together with secondary recovery, the disease was completely managed. Lesions at the perineum and scrotum and at both inguinal areas were broadly excised and grafted. No lesion has relapsed so far. One year later, Hodgkin's lymphoma was diagnosed, and the patient was treated with chemoradiotherapy easily, because there was no infective focus. The disease is in remission now. The patient weighs 110 kg, is healthy, and is working again.

Introduction

HS is a disease that is characterized by intensive development of sclerosis due to abscess, sinus and fistula formation, and fibrosis as a result of a chronic infection of the apocrine sweat glands. The disease can be seen at the scrotum, perineum, perianal region, hip, axilla, periumbilical region, and breast areola – any area that has apocrine sweat glands. It has a typical appearance (Figure 1). The disease was first defined by Valpeau in 1839. The incidence and prevalence of the disease are not known; however, in some studies, a 1% incidence is declared.[13] This disease occurs more frequently in women.[4] Hereditary factors, obesity, and sports are regarded as predisposing factors. The disease is not seen before puberty, because the apocrine sweat glands become active with the effect of the sex hormones, which are inactive during prepuberty.[2,5] However, plantar HS or palmoplantar HS can be seen in children.[6,7] The facilitation of bacterial translocation due to the obstruction of sweat glands and abnormal excretion of sweat glands as a result of insufficient or inaccurate development is regarded as the cause of HS. Recent research indicates that these patients have neutrophil dysfunction as well.[8]

In some patients, the disease becomes complicated, and serious antibiotic resistance develops. Because of antibiotic resistance, infections cannot be controlled, and large abscesses may develop. Septic complications may develop that necessitate hospitalization of the patient and administration of broad-spectrum antibiotics. This period can last 10 to 15 years. In addition, in patients with HS lymphedema, complications such as contracture formation at the region of the lesion, squamous cell cancer development, restriction of movement, urethral or rectal fistula development, anemia, arthritis, amyloidosis, renal failure, and interstitial keratitis may develop.3,4,9] On the other hand, in some patients the disease may regress spontaneously.

Fatal complications of HS are rare; however, especially in multilocalized cases, serious morbidity occurs, affecting a patient's social and professional life.


Url:
PubMed: 16614641
PubMed Central: 1681757


Affiliations:


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<p>The patient was a 38-year-old man. He had been suffering from hidradenitis suppurativa (HS) for approximately 20 years. He had active lesions at both axillas, hip, scrotum, and perineum, and inactive lesions located behind the ears, lower abdomen, and posterior neck. He was monitored and treated at different branches; he continuously used antibiotics and was given steroids at times. Antibiotic resistance developed subsequently. His general situation was bad; vital signs were poor; and he was in a state of sepsis and preshock, so this case was regarded as life-threatening.</p>
<p>Total excision was performed first on the lesion at the right axilla, then on the lesion at the left axilla, and the parascapular fasciocutaneous flap was reversed. A skin graft was applied to the triangular defect on the scapula. No relapse occurred. Then the lesions at the hip were managed. Broad excision was used twice with the patient under general anesthesia; because the lesions spread to the retrococcygeal and gluteal muscles, coccyx resection and partial gluteal muscle resection were implemented. The defect was eliminated with a progressive flap. At the intergluteal sulcus, small lesions emerging at the median line were debrided with the patient under local anesthesia, and together with secondary recovery, the disease was completely managed. Lesions at the perineum and scrotum and at both inguinal areas were broadly excised and grafted. No lesion has relapsed so far. One year later, Hodgkin's lymphoma was diagnosed, and the patient was treated with chemoradiotherapy easily, because there was no infective focus. The disease is in remission now. The patient weighs 110 kg, is healthy, and is working again.</p>
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<title>Introduction</title>
<p>HS is a disease that is characterized by intensive development of sclerosis due to abscess, sinus and fistula formation, and fibrosis as a result of a chronic infection of the apocrine sweat glands. The disease can be seen at the scrotum, perineum, perianal region, hip, axilla, periumbilical region, and breast areola – any area that has apocrine sweat glands. It has a typical appearance (
<xref ref-type="fig" rid="F1">Figure 1</xref>
). The disease was first defined by Valpeau in 1839. The incidence and prevalence of the disease are not known; however, in some studies, a 1% incidence is declared.
<xref ref-type="bibr" rid="R1">[1</xref>
<xref ref-type="bibr" rid="R3">3]</xref>
This disease occurs more frequently in women.
<xref ref-type="bibr" rid="R4">[4]</xref>
Hereditary factors, obesity, and sports are regarded as predisposing factors. The disease is not seen before puberty, because the apocrine sweat glands become active with the effect of the sex hormones, which are inactive during prepuberty.
<xref ref-type="bibr" rid="R2">[2</xref>
,
<xref ref-type="bibr" rid="R5">5]</xref>
However, plantar HS or palmoplantar HS can be seen in children.
<xref ref-type="bibr" rid="R6">[6</xref>
,
<xref ref-type="bibr" rid="R7">7]</xref>
The facilitation of bacterial translocation due to the obstruction of sweat glands and abnormal excretion of sweat glands as a result of insufficient or inaccurate development is regarded as the cause of HS. Recent research indicates that these patients have neutrophil dysfunction as well.
<xref ref-type="bibr" rid="R8">[8]</xref>
</p>
<p>In some patients, the disease becomes complicated, and serious antibiotic resistance develops. Because of antibiotic resistance, infections cannot be controlled, and large abscesses may develop. Septic complications may develop that necessitate hospitalization of the patient and administration of broad-spectrum antibiotics. This period can last 10 to 15 years. In addition, in patients with HS lymphedema, complications such as contracture formation at the region of the lesion, squamous cell cancer development, restriction of movement, urethral or rectal fistula development, anemia, arthritis, amyloidosis, renal failure, and interstitial keratitis may develop.
<xref ref-type="bibr" rid="R3">3</xref>
,
<xref ref-type="bibr" rid="R4">4</xref>
,
<xref ref-type="bibr" rid="R9">9</xref>
] On the other hand, in some patients the disease may regress spontaneously.</p>
<p>Fatal complications of HS are rare; however, especially in multilocalized cases, serious morbidity occurs, affecting a patient's social and professional life.</p>
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