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A SARCOID REACTION TO INJURY BY SEA URCHIN SPINES P. COOPER AND M. C. WAKEE~ELD Department of Pathology, University of Lee& PLATE XI11 A FEW cases of granulomata with a sarcoidal histology following injury by sea urchin spines have already been reported (Gate, 1936; Tkmime, 1953; Rocha and Fraga, 1962; Kinmont, 1965). Most authors stress the chronicity of these lesions, but we have been unable to find another case where the florid and des- tructive nature of the inflammatory process has necessitated amputation of the affected digit. CASE REPORT E. C., a 21-yr-old male student, while on holiday in Jugoslavia in 1971, dived into the Adriatic and hit a rock on which lay a sea urchin. The urchin spines penetrated both hands and subsequently several hard nodules developed. The patient stated that one of these spines had penetrated right into the proximal interphalangealjoint of the left index finger, and in October, some 2 mth after the injury, the left index finger started to swell. When first seen as an out-patient in January 1972, he was found to have numerous small white nodules on fingers, palms and wrists of both hands. The left index finger was painful and diffusely swollen. At operation 2 days later the left index finger was explored. Most of the swelling was found to be within the flexor tendon sheath and on opening this the surgeon found a large quantity of " rheumatoid-like " granulation tissue lying between the tendons and the bone. Further exploration showed that the articular cartilage of the proximal interphalangeal joint had already been eroded. No foreign body was located and as much as possible of the granula- tion tissue was excised and the wound closed. When seen again 2 wk later the incision was well healed, but the finger had already swollen again and as it was obvious that the degree of damage was such that the proximal joint would never move again, amputation was advised and duly performed. At the same operation several sea urchin spine fragments were removed from the fingers and palm of the right hand. The patient showed no evidence of any generalised disease process. Pathology Two separate specimens were received for pathological examination. The first was a biopsy consisting of a few small fragments of soft white tissue which had been excised from within the flexor tendon sheath of the left index finger. The tissue was all embedded and stained with haematoxylin and eosin. Histology. The biopsy consisted of fibro-adipose connective tissue containing several granulomata composed of epithelioid cells and multinucleate giant cells surrounded by small lymphocytes (fig. 1). There was no caseation and the appearances were reported as being consistent with a sarcoid reaction to foreign material. Received 5 Dec. 1972; accepted 25 Feb. 1973. 1. PATH.-VOL. 112 (1974) 33 C

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Page 1: A sarcoid reaction to injury by sea urchin spines

A SARCOID REACTION TO INJURY BY SEA U R C H I N SPINES

P. COOPER AND M. C. WAKEE~ELD Department of Pathology, University of Lee&

PLATE XI11

A FEW cases of granulomata with a sarcoidal histology following injury by sea urchin spines have already been reported (Gate, 1936; Tkmime, 1953; Rocha and Fraga, 1962; Kinmont, 1965). Most authors stress the chronicity of these lesions, but we have been unable to find another case where the florid and des- tructive nature of the inflammatory process has necessitated amputation of the affected digit.

CASE REPORT

E. C., a 21-yr-old male student, while on holiday in Jugoslavia in 1971, dived into the Adriatic and hit a rock on which lay a sea urchin. The urchin spines penetrated both hands and subsequently several hard nodules developed. The patient stated that one of these spines had penetrated right into the proximal interphalangeal joint of the left index finger, and in October, some 2 mth after the injury, the left index finger started to swell.

When first seen as an out-patient in January 1972, he was found to have numerous small white nodules on fingers, palms and wrists of both hands. The left index finger was painful and diffusely swollen.

At operation 2 days later the left index finger was explored. Most of the swelling was found to be within the flexor tendon sheath and on opening this the surgeon found a large quantity of " rheumatoid-like " granulation tissue lying between the tendons and the bone. Further exploration showed that the articular cartilage of the proximal interphalangeal joint had already been eroded. No foreign body was located and as much as possible of the granula- tion tissue was excised and the wound closed.

When seen again 2 wk later the incision was well healed, but the finger had already swollen again and as it was obvious that the degree of damage was such that the proximal joint would never move again, amputation was advised and duly performed. At the same operation several sea urchin spine fragments were removed from the fingers and palm of the right hand.

The patient showed no evidence of any generalised disease process.

Pathology Two separate specimens were received for pathological examination. The first was a

biopsy consisting of a few small fragments of soft white tissue which had been excised from within the flexor tendon sheath of the left index finger. The tissue was all embedded and stained with haematoxylin and eosin.

Histology. The biopsy consisted of fibro-adipose connective tissue containing several granulomata composed of epithelioid cells and multinucleate giant cells surrounded by small lymphocytes (fig. 1). There was no caseation and the appearances were reported as being consistent with a sarcoid reaction to foreign material.

Received 5 Dec. 1972; accepted 25 Feb. 1973. 1. PATH.-VOL. 112 (1974) 33 C

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34 P. COOPER AND M. C. WAKEFIEL.D

The second specimen was a whole left index finger showing diffuse swelling and apparent fixation of the interphalangeal joints. A white nodule 1 cm in diameter was present on the dorsum of the proximal interphalangealjoint. In view of the diffuse character of the swelling and in the light of the previous biopsy, it was decided to bisect the specimen longitudinally with a bandsaw, decalcify and process the whole fhger in order to have complete longitudinal sections of the digit.

Macroscopically, the bisected specimen showed extensive destruction with subluxation of the proximal interphalangeal joint and the presence of a considerable quantity of soft pink tissue between tendons and bone.

Giant microtome sections were prepared and stained with haematoxylin and eosin, and Masson’s trichrome, and also by Ziehl-Neelsen’s method.

Himlogy. This method of preparation showed clearly the presence of a chronic follicular granulomatous process (fig. 2), which had commenced subcutaneously over the dorsum of the proximal interphalangeal joint and had spread through the joint causing total destruction of the articular surfaces. The inflammatory process had continued to spread to involve bone marrow and the distal interphalangeal joint.

The granulomas were identical to those seen in the previous biopsy and showed no evi- dence of caseation.

No acid-fast bacilli were detected in a giant section stained by Ziehl-Neelsen’s method. The sections were also viewed by polarised light, but no birefringent particles or spiny fragments were detected.

DISCUSSION Injuries inflicted by sea urchin spines will probably be seen in this country

with increasing frequency now that foreign holiday travel is so popular. The commonest species of Echinoidea found along the Mediterranean and Adriatic shores is Paracentrotus lividus, with a black body 4-5 cm in diameter and dark purple spines 2-3 cm long. The sharp spines are movable, being articulated with the body plates by a ball and socket joint. Each spine is composed of a central core of calcite covered by epithelium. The usual sequelae to injury by these spines are simple puncture wounds which heal quickly when the spiny fragments are removed. Occasionally sepsis, foreign-body granulomata or inclusion dermoids may supervene.

Granulomata showing typical sarcoidal histology appearing several weeks or months after the injury have been reported by several observers (Gate, 1936; Tkmime, 1953; Rocha and Fraga, 1962; O’Neal, 1964; Kinmont, 1965; Moynahan and Montgomery, 1968). In our case the history, clinical and his- tological findings are similar to the cases already reported, but the pathogenesis of the lesions remains uncertain.

No spine fragments were demonstrated on X-ray or on dissection of the amputated digit, although fragments were removed from nodules on the other hand. The possibility that grains of sand had been introduced at the time of injury was excluded by failure to demonstrate any birefringence when the giant sections were examined under polarised light.

Two authors (Kinmont, 1965; Moynahan, 1968) analysed sea urchin spines, but found only a minute quantity of silica and no beryllium, zirconium or traces of any other metal likely to produce a sarcoid reaction in soft tissues.

Some varieties of sea urchin, particularly those found in tropical waters, do have poison glands embedded in the epidermal covering of the spines. These

Page 3: A sarcoid reaction to injury by sea urchin spines

COOPER AND WAKEFIELD PLATE XI11

SARCOID REACTION TO SEA URCHINS

FIG. 1 .-Biopsy from left index finger showing several granulomata. Each consists of a central part formed by epithelioid cells and multinucleate giant cells surrounded by small lymphocytes. There is no caseation. Haematoxylin and eosin (HE). x 100.

FIG. 2.-Longitudinal section of left index finger after decalcification. This clearly shows an extensive granulomatous process causing destruction and subluxation of the proximal interphalangeal joint. HE. x3.

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SARCOID REACTION TO SEA URCHIN SPINES 35

are not usually a feature of Paracentrotus Zividus, but Kinmont (1965) reported one case where puncture of some cystic lesions, produced by this variety, resulted in a severe burning pain exactly like that of the original injury. He suggested that the pain was due to the release of venom and speculated that the cysts might contain some venom-secreting epithelium.

The destructive properties of the granulomatous process in our case and the tendency to spread to involve other joints suggested the possibility of a myco- bacterial infection. However, prolonged search of a giant section stained by Ziehl-Neelsen’s method failed to demonstrate any acid-fast bacilli in the tissues. Moynahan and Montgomery (1968) did demonstrate the presence of scanty acid and alcohol fast bacili in ground-up biopsy tissue, but they failed to culture the organism.

Tkmime in 1936 suggested that as few people develop this delayed granulo- matous reaction it might reflect some form of individual predisposition. Others, noting the similarity of the microscopical changes to those of sarcoidosis (Boeck’s disease), have suggested that the reaction is the result of trauma in a patient with a sarcoid diathesis. Refvem (1954) presented considerable experi- mental evidence to refute this suggestion, and in particular pointed out that a variety of agents produce a sarcoid reaction in the subcutis, but when the lesions are limited to those parts containing adipose tissue, no conclusions can be drawn about the relationship of the reaction to systemic sarcoidosis. All the reported cases presenting with sea urchin sarcoidal granulomata had been investigated for sarcoidosis, with negative results.

The final point to consider is whether any form of treatment might have halted this destructive process. Kinmont (1965) noted that lesions in one of his cases subsided after local steroid injections and it is possible that similar treatment given here at an early stage might have lessened the degree of damage to the involved joint. This particular patient, however, did not present until the articu- lar surfaces had already been eroded.

SUMMARY A case of sarcoid reaction to injury by sea urchin spines is reported. No

evidence of spine fragments, birefringent particles, or acid-fast bacilli were seen in the sections and there was nothing to suggest that the patient had systemic sarcoidosis. Theories as to possible pathogenesis are discussed.

We wish to thank Professor C. E. Lumsden and Mr D. H. Wilson for their encouragement and permission to publish this case, and Dr S. Aparicio and Mr J. Cousins for photographic assistance.

REFERENCES GATB, J., CUILLERET, P., BOUQUIN, C., AND H. 1936. Usions papulo-nhotiques reaction

histologique tuberculoide dues a I’inclusion d‘epines d‘oursins. Bull. SOC. FranF. Derm. Syph., 43, 937.

KINMONT, P. D. C. 1965. Sea-Urchin sarcoidal granuloma. Brit. J. Derm., 77, 335. MOYNAHAN, E. J., AND MONTGOMERY, P. R. 1968. Echinoderm granuloma. Brit. J . Clin.

Pract., 22, 265. O’NEAL, R. L. 1964. Injury to human tissues from sea urchin spines. Calif. Med., 101, 199.

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36 P. COOPER AND M. C, WAKEFIELD

NICHOLS, D. 1962. Echinoderms, Hutchinson, London. PIOVANO, P. B. 1968. Multiple granulomas of the upper extremities caused by sea urchins.

Minerva derm., 43, 538. REFVEM, 0. 1954. The pathogenesis of Boeck’s disease. Acta. med. sand., Suppl. 294. ROCHA, G., AND FRAGA, S. 1962. Sea urchin granuloma of the skin. Arch. Derm., 85,406. TBMIME, P. 1953. Les lCsions cutanQs causkes par les piquants d’oursins. Presse mkd., 61,

1509.