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Cervico-Auricular Fistulae a Review of Published Cases with a Report

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Cervico-Auricular Fistulae a Review of Published Cases with a Report Arch Dis Child: first published as 10.1136/adc.40.210.218 on 1 April 1965. Downloaded from Arch. Dis. Childh., 1965, 40, 218. CERVICO-AURICULAR FISTULAE A REVIEW OF PUBLISHED CASES WITH A REPORT BY J. C. R. LINCOLN* From the Department ofSurgery, Hammersmith Hospital, Postgraduate Medical School ofLondon (RECEIVED FOR PUBLICATION JUNE 4, 1964) Congenital anomalies of the first branchial cleft or arches with four intervening clefts, the internal are rare. Approximately 32 cases of cervico- clefts being lined by foregut endoderm and the auricular fistulae have been reported in the past external clefts by ectoderm, but divided by a thin hundred years: this includes the writer's own case in layer of mesoderm. Though there is confusion in this review. The majority of sinuses and fistulae the nomenclature, the general consensus of opinion derived from the lateral cervical vestiges are con- names the pharyngeal clefts pharyngeal pouches, and cerned with the second pharyngeal pouch and its the corresponding external clefts branchial clefts. corresponding cleft. Each arch has a central core of cartilage, a main Auricular and pre-auricular fistulae are not blood vessel, and nerve. excessively rare, but cervico-auricular fistulae are still infrequently recorded. In some cases reported MAXILLARt >=j as cervico-auricular fistulae, there is little doubt from PROCESS the description that these are auricular or pre- Ist ARCH CARTILAGE copyright. 1st PHARYNGEAL ARCH auricular fistulae. - MAN1IRULAN NERVE Harding (1890) and Konig (1896) both recorded - 7th NERVE cases, but it was not until 1908 that Flint published a 2nd PHARYNGEAL ARCH , AYI well-documented case report on this condition. 3rd PHARYNGEAL ARCH gth NERVE Frazer (1923) postulated defects of closure of the first NI- branchial cleft, though up to that time he thought &thPOARYREAL ARCH 74 SUOP ULARYNGEAL NERVE that none had been He stated 'If a TRACHEAL GROOVE reported. that, http://adc.bmj.com/ cyst of such a vestige were present it would lie below PERICARDIAL CAVITY the tube, behind (at any rate in part) the tensor palati (a) and in front of the carotid and stylopharyngeus and if by any chance it opened in to the pharynx it would do so through the sinus of Morgagni.' In the following review only those cases that are well documented have been considered. on September 28, 2021 by guest. Protected Embryology The branchial apparatus appears as but a transient stage in the development of the human embryo, appearing between the 3 mm. and 14 mm. stage. The embryology of these cases is concerned with the branchial or lateral cervical vestiges. (b) At the fifth week, when the human embryo is about Fio. la.-Horizontal section through a reconstructed 5-week human 5 mm. in length, five ridges are visible on the outer embryo to show the pharyngeal arches and their constituents. Externally the arches are covered by ectoderm, internally by foregut surface in the region of the primitive pharynx (Fig. endoderm with an intervening layer of mesoderm. The roman la). The walls of the pharynx are lined by five bars numerals (i-iv) indicate endodermal pouches. FiG. lb.-A diagram of the pharyngeal region of a human embryo at * Present address: Surgical Unit, University College Hospital approximately 6 weeks, demonstrating the relative overgrowth of the Medical School, London W.C. I. first and second arches. 218 Arch Dis Child: first published as 10.1136/adc.40.210.218 on 1 April 1965. Downloaded from CER VICO-A URICULAR FISTULAE 219 The muscular derivatives of each arch are supplied tubercles which develop on the first and second by the nerve of that arch. The nerve of the first arch arches on the dorsal end of the first groove which lies is the mandibular division of the trigeminal nerve between. By growth and fusion, the tubercles and and supplies the muscles of mastication, the tensor the immediately surrounding area give rise to the palati, the tensor tympani, the mylohyoid, and the primitive pinna. This is situated around the end of anterior belly of the digastric. The muscles of the the developing external auditory meatus. The second arch are supplied by the facial nerve, these tragus and its immediate area is derived from the first muscles being those concerned with facial expression, arch, and the greater contribution to the formation the posterior belly of the digastric, the stapedius, and of the pinna comes from the second arch (Wood- the stylohyoid. The third arch muscles make up Jones and I-Chuan, 1934; Streeter, 1922). The part of the soft palate and the stylopharyngeus external auditory meatus is formed from two ele- muscle, the nerve of supply being the glossopharyn- ments: the dorsal end of the first groove and an geal. The constrictors of the pharynx and part of ingrowth of epithelium from this groove, the ventral the soft palate are derived from the fourth arch and end becoming flattened then disappearing. The the nerve is the superior laryngeal. The fifth arch endodermal derivatives of the first pouch are nerve, which is the recurrent laryngeal, supplies the concerned with the epithelium of the internal part of intrinsic muscles of the larynx. the tympanic membrane, the tympanic cavity, The vascular derivatives of each arch are modified, mastoid air cells, the eustachian tube, and epithelium and there is a reduction in the number of arch vessels of much of the mouth and body of the tongue. to facilitate pulmonary respiration and suit the new By the end of the sixth week, the growth of the first conditions of life. The original arteries of the first and second arches has exceeded that of the third and and second arches persist as the mandibular and fourth and a sinus is formed, the pre-cervical sinus of stapedial arteries. The third arch arteries remain; His (Fig. I b). The third arch still contributes in the right third arch artery contributes to the develop- part to the skin over the anterior triangle of the neck; ment of the right common carotid artery and the the fourth arch is completely overgrown. Two deep commencement of the right internal carotid artery. recesses are formed, and these are the cervical The left third arch artery forms the left common vesicles: meanwhile, the first pouch fills up ventrally copyright. carotid artery and distally the proximal part of the to become part of the tongue, and the dorsal end, left internal carotid artery. The right fourth arch together with the dorsal end of the second pouch, artery becomes the right subclavian artery and part forms the tubo-tympanic recess, this recess forming of the arch of the aorta, and the left fourth arch the eustachian tube and the middle ear; the meso- artery forms the arch of the definitive aorta. The dermic membrane forms the tympanic membrane. fifth arch arteries are present for only a short time Ectodermal remains may persist from the ventral and the sixth arch arteries persist as the pulmonary part of the first groove, from any part of the second http://adc.bmj.com/ arteries. groove, and from the cervical vesicles. Endodermal The cartilaginous part of the first arch gives rise to remains may persist as cysts or sinuses and accessory the malleus, incus, the spleno-mandibular ligament, glands. and the central core of the body of the mandible. The following anomalies ofthe branchial apparatus The second arch cartilage forms the stapes, the lesser are recorded: branchial cysts, external sinuses, cornu of the hyoid bone, the styloid process, the internal sinuses, complete pharyngo-cutaneous stylohyoid ligament persisting as the intervening fistulae, cervico-aural fistulae, branchial cartilages, portion. The greater cornu and the lower part of and cervical auricles. on September 28, 2021 by guest. Protected the body of the hyoid bone is formed from third arch cartilage, and the cartilage of the fourth and fifth Case Report arches make up the skeleton of the larynx. A girl aged 2 years was admitted with a tender swelling The glandular derivatives of the pharyngeal arches in the left submandibular region. This had been present form parathyroid and thymic tissue, and the palatine for three days and the child had been pyrexial. Since tonsils. The palatine tonsils develop from the birth there had always been a 'dimple' in the region of the endodermal elements of the second pouch. The swelling. inferior Physical examination revealed a tender cystic swelling thymus, together with the parathyroid gland, approximately 5 x 2 cm. in the left submandibular region is developed from the third pouch, the parathyroid anterior to the sternocleidomastoid muscle, at thejunction gland in this case being drawn caudally by the thymic of the upper and middle third. No other abnormality rudiment. The superior parathyroid gland is was noted. Radiographs of the chest and mandible were derived from the fourth pouch. normal. The swelling was incised and brownish pultace- The external ear is developed from a series of six ous material was evacuated. This was sterile on culture. Arch Dis Child: first published as 10.1136/adc.40.210.218 on 1 April 1965. Downloaded from 220 LINCOLN diameter, lined by epithelium with hairs clearly visible and surrounded by its own wall ofcartilage, the cartilage being continuous with that of the external ear. The anterior 'tract' was 2 cm. long and fanned out in the sub-mandibular region as a series of tails that ran superficial to the sub-mandibular structures. There was some doubt as to the nature of this 'tract' and its exten- sions, as it was thought that it might be a branch of the facial nerve. Faradic stimulation of these structures provoked no response: the mandibular division of the facial nerve was positively identified using this technique (Fig.
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