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Actinomycotic bowel and ureteral obstruction in a patient with Hodgkin's disease
Actinomycotic Bowel and Ureteral Obstruction in a Patient With Hodgkin's Disease By David L. Tilford, Arnold G. Coran, and Ruth M. Heyn 9 This is a case report of a patient who presented with colonic and ureteral obstruction 4 yr following diagnosis and abdominal-pelvic radiation therapy of stage I I A Hodgkin's disease. Biopsy of a pelvic mass revealed actinomycosis. I N D E X W O R D S : Actinomycosis; Hodgkin's disease.
N V O L V E M E N T of the gastrointestinal tract by H o d g k i n s disease is r a r e . Kaplan r e p o r t e d a b s e n c e of i n v o l v e m e n t in 340 consecutive patients with previously untreated Hodgkin's disease and noted that in autopsied patients only 8% of all cases had GI involvement.~ Ureteral obstruction is likewise unusual. Persistent GI complications following radiation therapy for Hodgkin's disease are also uncommon. Kaplan in his series had only a single patient with radiation enteritis and stenosis following abdominal-pelvic radiotherapy.
I
CASE REPORT B. S., a 14-yr-old male, presented in April 1972 with a 5-mo history of an enlarging node in the left groin. The pathologic diagnosis of the excised node was Hodgkin's disease. On admission to Mott Children's Hospital on 5/17/72, physical exam revealed no palpable nodes in any location and no palpable liver, spleen, or abdominal masses. The chest x-ray was normal. A bipedal lymphangiogram revealed abnormal nodes in the left para-aortic area and left inguinal area. At l a p a r o t o m y on 5 / 2 4 / 7 2 , the s u r g e o n s observed enlargement of the left iliac and para-aortic nodes. Appropriate nodes were excised and splenectomy and liver biopsy were performed. The left iliac node was replaced by Hodgkin's lymphoma. The para-aortic node, liver, and spleen'were all negative. T h e patient was staged IIA. He received radiation therapy to the abdomen and pelvis in an inverted Y field. Between 6/6/72 and 8/2/72, he was given 3740 rad to a 24 x 24 cm pelvic field and 3600 rad to a 20 x i0 cm abdominal field. He had marked nausea during radiatibn therapy but no significant vomiting, diarrhea, or skin change. T h e patient remained well until the spring of 1976. On 4/23/76, he presented with a history of dysuria for 1 mo, progressive midabdominal cramping, lack of a bowel movem e n t for 1 wk, and vomiting for 1 day. Physical exam was unremarkable. A WBC was 18,000 with 64% segs and 7% bands. A chest x-ray was normal, and an abdominal fiat plate revealed a large a m o u n t of fecal material in the colon with no evidence of obstruction. Over the next 10 days, he began having bowel m o v e m e n t s but of unusually small size. His appetite was poor and he
Journal of Pediatric Surgery, Vol. 13, No, 2 (April), 1978
lost 9 lb prior to admission on 5/4/76. A temperature spike to 101 ~ occurred daily. On admission the physical exam was unchanged. The chest and abdominal x-rays were unchanged. A lymphangiogram and liver scan were normal. An IVP demonstrated a delayed nephrogram of the right kidney and delayed filling of a dilated right pyelocalyceal system (Fig. I A). T h e right ureter was dilated to the level of the pelvic inlet and was not seen below. A barium enema revealed filling of the rectum to the junction of the sigmoid colon with complete obstruction to barium above this juncture (Fig. 2A). T h e abdominal contents appeared normal at laparotomy on 5/14/76. The entire pelvis, however, was filled with a hard mass encasing the appendix, ureters, and sigmoid colon. Frozen sections of biopsies of the mass were read as chronic inflammation, free of malignancy. Bacterial cultures were taken. A loop sigmoid colostomy was done. Histology of the biopsy revealed fibroadipose tissue with marked fibrosis and chronic purulent inflammatory infiltrate. A single large colony of organisms resembling A ctinomyces was present within a microabscess (Fig. 3). With a presumptive diagnosis, he received i.v. penicillin, 12 million units a day, oral trimethoprin, 320 m g a day, and oral sulfamethoxazole, 1600 mg a day, for 21 days. At this time repeat IVP and b a r i u m e n e m a showed m a r k e d improvement (Fig. 1B and 2B). The patient was discharged on 2 g of penicillin a day to be continued for 6 mo. Four mo later the patient remained asymptomatic and regained lost weight. A repeat barium enema showed the rectal obstruction to be completely resolved. Closure of the colostomy was carried out 1 yr later.
DISCUSSION
Actinomycosis is a chronic granulomatous infection characterized by dense fibrous tissue that softens to form abscesses and fistulas. It is diagnosed on tissue section by the presence of mycelial masses known as sulfa granules. The two species t h a t can c a u s e this t y p e of p a t h o l o g y are Actinomyces israeli and Nocardia brasiliensis. 2 Actinomyces israeli is a saprophyte in the oral cavity, lungs, and GI tract. It is generally felt that Actinomyees does not p e n e t r a t e
From the Department o f Pediatrics and Communicable Diseases, Department o f Surgery, University o f Michigan Medical School, Ann Arbor, Mich. Address reprint requests to David L. Tilford, M.D., University Hospital, F6522, Ann Arbor, Mich. 48109. 9 1978 by Grune & Stratton, Inc. 0022 3468/78/1302-0020501.00/0
189
190
CASE REPORT
Cope into two clinical patterns: (1) those that start with acute perforating appendicitis and (2) those that present with an abdominal mass. 3 In P u t n a m ' s series, 88 of 122 cases were preceded by typical acute appendicitis? In contrast, of seven cases at the Manchester Royal Infirmary, four presented as a mass in the right iliac fossa and one as recurrent attacks of small bowel obstruction? Patients with cancer are at high risk for deep
Fig. 1. (A) Intravenous pyelography demonstrated a dilated right pyelocalyceal system, (B) The IVP 1 mo later showed marked improvement.
healthy m u c o s a and that underlying local disease or previous trauma or surgery is a requirement for infection. Nocardia brasiliensis is an ubiquitous saprophyte. Infection is usually in the form of a mycetoma, a localized granulomatous process, at the site of trauma. It is clinically important to attempt to differentiate b e t w e e n t h e s e two o r g a n i s m s b e c a u s e Actir~myces is m o s t sensitive to penicillin therapy and Nocardia to sulfa. Pericecal actinomycosis was categorized by
Fig. 2. (A) B a r i u m enema shortly after admission revealed obstruction at the junction of the sigmoid colon and rectum. (B) Barium e n e m a 1 m o later showed resolution of obstruction.
CASE REPORT
Fig. 3. (A) The sulfa granule is surrounded by inflammatory cells, H & E x 250. (B)The mycelium are better defined by methyl silver staining, x 250.
191
192
CASE REPORT
fungal infections. In a review of 202 cases of fungal infections seen in a cancer hospital, Hutter and Collins r e p o r t e d 13% due to Actinomyces, second only to 56% due to Candida. Sixteen of these 28 patients with actinomycosis had an underlying malignancy including two with Hodgkin's disease. ~ Our patient presented 4 yr after abdominalpelvic radiotherapy for stage IIA Hodgkin's disease with obstruction of the sigmoid colon and of the right ureter. The normal lymphangiogram was not suggestive of recurrent Hodgkin's
disease. Consideration was given to the possibility of radiation fibrosis but there were no increased subcutaneous changes to indicate excessive radiation damage. At surgery, a diagnosis could not be made. Actinomycosis was diagnosed on a single sulfa granule found in tissue sections. No predisposing local factor can be identified for the establishment of this infection in our patient. T h e r e was no history compatible with appendicitis and no surgery other than a staging laparotomy 4 yr earlier.
REFERENCES
1. Kaplan HS: Hodgkin's Disease. Cambridge, Harvard University Press, 1972 2. Gonz~ilez-Ochoa A: Actinomycosis, in Robinson HM Jr (ed): The Diagnosis and Treatment of Fungal Infections. Springfield, Ill., Thomas, 1974 3. Cope Z: Actinomycosis. London, Oxford University Press, 1938
4. Putnam HC Jr, Dockerty MB, Waugh JM: Abdominal actinomycosis. Surgery 28:781, 1950 5. Davies M, Keddie NC: Abdominal actinomycosis. Br J Surg 60:18, 1973 6. Hutter RVP, Collins HS: The occurrence of opportunistic fungus infections in a cancer hospital. Lab Invest 11:1035, 1962
N V O L V E M E N T of the gastrointestinal tract by H o d g k i n s disease is r a r e . Kaplan r e p o r t e d a b s e n c e of i n v o l v e m e n t in 340 consecutive patients with previously untreated Hodgkin's disease and noted that in autopsied patients only 8% of all cases had GI involvement.~ Ureteral obstruction is likewise unusual. Persistent GI complications following radiation therapy for Hodgkin's disease are also uncommon. Kaplan in his series had only a single patient with radiation enteritis and stenosis following abdominal-pelvic radiotherapy.
I
CASE REPORT B. S., a 14-yr-old male, presented in April 1972 with a 5-mo history of an enlarging node in the left groin. The pathologic diagnosis of the excised node was Hodgkin's disease. On admission to Mott Children's Hospital on 5/17/72, physical exam revealed no palpable nodes in any location and no palpable liver, spleen, or abdominal masses. The chest x-ray was normal. A bipedal lymphangiogram revealed abnormal nodes in the left para-aortic area and left inguinal area. At l a p a r o t o m y on 5 / 2 4 / 7 2 , the s u r g e o n s observed enlargement of the left iliac and para-aortic nodes. Appropriate nodes were excised and splenectomy and liver biopsy were performed. The left iliac node was replaced by Hodgkin's lymphoma. The para-aortic node, liver, and spleen'were all negative. T h e patient was staged IIA. He received radiation therapy to the abdomen and pelvis in an inverted Y field. Between 6/6/72 and 8/2/72, he was given 3740 rad to a 24 x 24 cm pelvic field and 3600 rad to a 20 x i0 cm abdominal field. He had marked nausea during radiatibn therapy but no significant vomiting, diarrhea, or skin change. T h e patient remained well until the spring of 1976. On 4/23/76, he presented with a history of dysuria for 1 mo, progressive midabdominal cramping, lack of a bowel movem e n t for 1 wk, and vomiting for 1 day. Physical exam was unremarkable. A WBC was 18,000 with 64% segs and 7% bands. A chest x-ray was normal, and an abdominal fiat plate revealed a large a m o u n t of fecal material in the colon with no evidence of obstruction. Over the next 10 days, he began having bowel m o v e m e n t s but of unusually small size. His appetite was poor and he
Journal of Pediatric Surgery, Vol. 13, No, 2 (April), 1978
lost 9 lb prior to admission on 5/4/76. A temperature spike to 101 ~ occurred daily. On admission the physical exam was unchanged. The chest and abdominal x-rays were unchanged. A lymphangiogram and liver scan were normal. An IVP demonstrated a delayed nephrogram of the right kidney and delayed filling of a dilated right pyelocalyceal system (Fig. I A). T h e right ureter was dilated to the level of the pelvic inlet and was not seen below. A barium enema revealed filling of the rectum to the junction of the sigmoid colon with complete obstruction to barium above this juncture (Fig. 2A). T h e abdominal contents appeared normal at laparotomy on 5/14/76. The entire pelvis, however, was filled with a hard mass encasing the appendix, ureters, and sigmoid colon. Frozen sections of biopsies of the mass were read as chronic inflammation, free of malignancy. Bacterial cultures were taken. A loop sigmoid colostomy was done. Histology of the biopsy revealed fibroadipose tissue with marked fibrosis and chronic purulent inflammatory infiltrate. A single large colony of organisms resembling A ctinomyces was present within a microabscess (Fig. 3). With a presumptive diagnosis, he received i.v. penicillin, 12 million units a day, oral trimethoprin, 320 m g a day, and oral sulfamethoxazole, 1600 mg a day, for 21 days. At this time repeat IVP and b a r i u m e n e m a showed m a r k e d improvement (Fig. 1B and 2B). The patient was discharged on 2 g of penicillin a day to be continued for 6 mo. Four mo later the patient remained asymptomatic and regained lost weight. A repeat barium enema showed the rectal obstruction to be completely resolved. Closure of the colostomy was carried out 1 yr later.
DISCUSSION
Actinomycosis is a chronic granulomatous infection characterized by dense fibrous tissue that softens to form abscesses and fistulas. It is diagnosed on tissue section by the presence of mycelial masses known as sulfa granules. The two species t h a t can c a u s e this t y p e of p a t h o l o g y are Actinomyces israeli and Nocardia brasiliensis. 2 Actinomyces israeli is a saprophyte in the oral cavity, lungs, and GI tract. It is generally felt that Actinomyees does not p e n e t r a t e
From the Department o f Pediatrics and Communicable Diseases, Department o f Surgery, University o f Michigan Medical School, Ann Arbor, Mich. Address reprint requests to David L. Tilford, M.D., University Hospital, F6522, Ann Arbor, Mich. 48109. 9 1978 by Grune & Stratton, Inc. 0022 3468/78/1302-0020501.00/0
189
190
CASE REPORT
Cope into two clinical patterns: (1) those that start with acute perforating appendicitis and (2) those that present with an abdominal mass. 3 In P u t n a m ' s series, 88 of 122 cases were preceded by typical acute appendicitis? In contrast, of seven cases at the Manchester Royal Infirmary, four presented as a mass in the right iliac fossa and one as recurrent attacks of small bowel obstruction? Patients with cancer are at high risk for deep
Fig. 1. (A) Intravenous pyelography demonstrated a dilated right pyelocalyceal system, (B) The IVP 1 mo later showed marked improvement.
healthy m u c o s a and that underlying local disease or previous trauma or surgery is a requirement for infection. Nocardia brasiliensis is an ubiquitous saprophyte. Infection is usually in the form of a mycetoma, a localized granulomatous process, at the site of trauma. It is clinically important to attempt to differentiate b e t w e e n t h e s e two o r g a n i s m s b e c a u s e Actir~myces is m o s t sensitive to penicillin therapy and Nocardia to sulfa. Pericecal actinomycosis was categorized by
Fig. 2. (A) B a r i u m enema shortly after admission revealed obstruction at the junction of the sigmoid colon and rectum. (B) Barium e n e m a 1 m o later showed resolution of obstruction.
CASE REPORT
Fig. 3. (A) The sulfa granule is surrounded by inflammatory cells, H & E x 250. (B)The mycelium are better defined by methyl silver staining, x 250.
191
192
CASE REPORT
fungal infections. In a review of 202 cases of fungal infections seen in a cancer hospital, Hutter and Collins r e p o r t e d 13% due to Actinomyces, second only to 56% due to Candida. Sixteen of these 28 patients with actinomycosis had an underlying malignancy including two with Hodgkin's disease. ~ Our patient presented 4 yr after abdominalpelvic radiotherapy for stage IIA Hodgkin's disease with obstruction of the sigmoid colon and of the right ureter. The normal lymphangiogram was not suggestive of recurrent Hodgkin's
disease. Consideration was given to the possibility of radiation fibrosis but there were no increased subcutaneous changes to indicate excessive radiation damage. At surgery, a diagnosis could not be made. Actinomycosis was diagnosed on a single sulfa granule found in tissue sections. No predisposing local factor can be identified for the establishment of this infection in our patient. T h e r e was no history compatible with appendicitis and no surgery other than a staging laparotomy 4 yr earlier.
REFERENCES
1. Kaplan HS: Hodgkin's Disease. Cambridge, Harvard University Press, 1972 2. Gonz~ilez-Ochoa A: Actinomycosis, in Robinson HM Jr (ed): The Diagnosis and Treatment of Fungal Infections. Springfield, Ill., Thomas, 1974 3. Cope Z: Actinomycosis. London, Oxford University Press, 1938
4. Putnam HC Jr, Dockerty MB, Waugh JM: Abdominal actinomycosis. Surgery 28:781, 1950 5. Davies M, Keddie NC: Abdominal actinomycosis. Br J Surg 60:18, 1973 6. Hutter RVP, Collins HS: The occurrence of opportunistic fungus infections in a cancer hospital. Lab Invest 11:1035, 1962