Trunk muscles are an important source for pedicled and free flaps in reconstructive surgery. Unilateral deficiencies of trunk muscles are well known, either isolated or as part of Poland's syndrome. Bilateral muscular deficiencies and a "bilateral Poland anomaly” have also been sporadically reported, but this is rare. We report on an 82-year-old male cadaver with clinically obscure, asymmetric bilateral deficiencies of the majority of trunk muscles. There was a history of acute poliomyelitis in childhood. Histological examination of representative muscle samples of the trunk showed extensive muscle atrophy with fat and connective tissue replacement. This was compatible with the prior diagnosis of poliomyelitis. However, representative sections of the spinal cord failed to reveal the antecedent poliomyelitis. The possibility of subclinical bilateral deficiencies of trunk muscles has to be taken into account in patients with a history of poliomyelitis when planning reconstructions in cases of regional pedicled muscle transfers or free microvascular tissue transfers in reconstructive surger

Beer, Gertrude

Goebel, Hans-Hilmar

Groscurth, Peter

Manestar, Mirjana

Mihic-Probst, Daniela

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CASE REPORTMultiple bilateral asymmetrical deficiency of trunk musclesGertrude M. Beer & Hans-Hilmar Goebel &Daniela Mihic-Probst & Peter Groscurth &Mirjana ManestarReceived: 20 July 2008 /Accepted: 19 November 2008 / Published online: 16 December 2008# Springer-Verlag 2008Abstract Trunk muscles are an important source forpedicled and free flaps in reconstructive surgery. Unilateraldeficiencies of trunk muscles are well known, either isolatedor as part of Poland’s syndrome. Bilateral muscular deficien-cies and a “bilateral Poland anomaly” have also been spora-dically reported, but this is rare. We report on an 82-year-oldmale cadaver with clinically obscure, asymmetric bilateraldeficiencies of the majority of trunk muscles. There was ahistory of acute poliomyelitis in childhood. Histologicalexamination of representative muscle samples of the trunkshowed extensive muscle atrophy with fat and connectivetissue replacement. This was compatible with the priordiagnosis of poliomyelitis. However, representative sectionsof the spinal cord failed to reveal the antecedent poliomyeli-tis. The possibility of subclinical bilateral deficiencies oftrunk muscles has to be taken into account in patients with ahistory of poliomyelitis when planning reconstructions incases of regional pedicled muscle transfers or free micro-vascular tissue transfers in reconstructive surgery.Keywords Acquired muscle deficiency . Poliomyelitis .Reconstructive surgery . Trunk musclesIntroductionTrunk muscles are an important source for pedicled and freeflaps in reconstructive surgery. Deficiencies of the trunkmuscles are rather rare. Unilateral deficiencies occurcomparatively frequently, whereas bilateral deficienciesare virtually unknown. Poland in 1841 [1] described suchmuscular deformities including a completely absent pectoralisminor muscle, deficiency of the sternocostal portions of thepectoralis major muscle, absence of the inferior digitations ofthe serratus anterior muscle, and a tendinous appearance of theanterior and inferior parts of the obliquus externus abdominismuscle. He was not the first to appreciate this, but hisdescription was the most complete. Despite many reports onPoland’s syndrome, neither the etiology nor the spectrum ofthis complex syndrome has yet been clearly analyzed. Thesame lack of knowledge about the etiology has been noted inthe few reports on bilateral deficiencies of trunk muscles. In1926, Jones [2] reported the occurrence of bilateral deficiencyof the pectoralis major muscles en passant. In his clinicaldescription of a 13-year-old girl with a “congenital” deficiencyof the right pectoral muscles accompanied by a rudimentarynipple and a missing breast (apart from wasting of the leftthigh and calf), he mentioned that the girl had suffered from anattack of acute anterior poliomyelitis when 2 years of age. In1932, Sheehan [3] reported on the bilateral complete absenceof the trapezius muscle in a 70-year-old male cadaver, and inEur J Plast Surg (2009) 32:199–203DOI 10.1007/s00238-008-0308-1G. M. Beer : P. Groscurth :M. ManestarInstitute of Anatomy, University Zurich-Irchel,Winterthurerstrasse 190,8057 Zurich, SwitzerlandH.-H. GoebelInstitute of Neuropathology, Department of Pathology,University Hospital Zurich,Rämistrasse 100,8091 Zurich, SwitzerlandD. Mihic-ProbstInstitute of Surgical Pathology, Department of Pathology,University Hospital Zurich,Rämistrasse 100,8091 Zurich, SwitzerlandG. M. Beer (*)Division of Plastic and Aesthetic Surgery, Bodenseeklinik Swiss,Neuseeland 14,CH-9404 Rorschacherberg, Switzerlande-mail: gertrude.beer@access.uzh.ch1972, Horan and Bonafede [4] presented a case of a youngman with a bilateral absence of the trapezius and the sternalhead of the pectoralis major muscle. These authors did notcomment on a possible etiology.Recently, Karnak and Tanyel [5] reported on a younggirl with bilateral absence of the pectoralis major muscle,symmetrical chest wall deformities, and hand involvementas “the first case of a bilateral Poland anomaly.” Anotherreport in 2003 [6] addresses a bilateral asymmetricdeficiency of the pectoralis major muscle in a 72-year-oldfemale cadaver. There were no obvious abnormalities onthe outward appearance of the anterior thorax, includingwell-developed breasts. The authors speculated on theetiology being a congenital deficiency rather than aPoland’s syndrome or the sequel to acute poliomyelitis,although the patient was diagnosed with poliomyelitis at theage of 26 years and even had a reduced size of the left andright anterior horns of the cervical spine.A case of bilateral asymmetric deficiencies of themajority of trunk muscles with a known history of acutepoliomyelitis in childhood is presented.Case reportIn 2004 in the medical student gross anatomy dissectioncourse, an 82-year-old male cadaver had an external appea-rance which appeared to be normal in all anatomical areas.The cause of death was a metastasized rectal carcinomawhich had been diagnosed and operated on in 1997. A polio-myelitis infection had occurred in childhood. His relativesand internist revealed that he had survived the poliomyelitiswithout visible physical sequelae. As he became older, hedeveloped a humpback, and after a knee replacement, he useda walking stick. He slowly lost muscle strength until finally,he could not stand without help. How much this physicaldeterioration was the sequela of the carcinoma, the advancedage, or the expression of a possible postpolio syndrome [7, 8]remains unknown. In his final hospitalization in 1997, therewas a short note in the discharge letter, which stated thatthere was visible atrophy of the upper and lower extremitiesand the paravertebral musculature.During his anatomical dissection, a bilateral sternocostaldeficiency of the pectoralis major muscle was noted. Theright complete sternocostal portion was absent and replacedby a tough fascial membrane. On the left, the caudalsternocostal portion alone was replaced by connectivetissue and fat. In the anterior axillary fold, both muscleswere fleshy and inserted into the lateral lip of the bicipitalgroove by a thin broad tendon. On the left side, theposterior lamina of the tendon was smaller than that on thecontralateral right side (Fig. 1a). The pectoralis minormuscle showed similar deformities. On the right side, themuscle was smaller and arose from the third to the fourthrib, but part of its cranial and caudal origins were replacedby connective tissue. On the left side, a well-developed,normal pectoralis minor muscle was present arising fromthe second to the fourth rib (Fig. 1b). These findingsprompted us to look for further muscular deformities.On the ventral side of the trunk, the obliquus externusabdominis, the rectus abdominis, the pyramidalis muscles,and the transversus abdominis and thoracic muscles showeda similar picture of spotted fatty and fibrous degeneration(Fig. 1b).On the dorsal side of the trunk, the trapezius muscleswere grossly replaced by connective tissue and fat withFig. 1 Overview of the anterior trunk. a Superficial muscular layerwith view to the pectoralis major and serratus anterior muscles. bDeep layer showing deficiencies of the pectoralis minor, the obliquusexternus abdominis, the rectus muscles, and the pyramidalis muscle200 Eur J Plast Surg (2009) 32:199–203only few muscle fibers intermingled. The deficiency wasmore pronounced on the left than on the right side (Fig. 2a).In addition, the latissimus dorsi, the teres major, the serratusanterior and posterior, both rhomboids, and both serratusposterior muscles had deficiencies of a differing intensity(Fig. 2b). Additionally, the paraspinal muscles (iliocostalis,longissimus, and spinalis muscles) were significantlyaffected by the same deformities (Fig. 2c).The surface appearance of the muscles of the lowerextremities appeared to be normal. However, on cross-section, the adductor longus magnus and brevis, the tensorfasciae latae, the vastus intermedius, all flexor muscles ofthe thighs, and the soleus revealed spotted areas ofconnective tissue and fat within their muscle bellies. Themuscles of the upper extremities were normal, both at thesurface and on cross-sections.Immunohistochemistry for fast and slow muscle myosinin representative muscle biopsies demonstrated a diffuse(and not a checkered) appearance of atrophic and normalmuscle fibers and an overall extensive muscle atrophy withfat and connective tissue substitution compatible with thediagnosis of prior poliomyelitis (Fig. 3). The histologicalexamination of representative biopsies of the thoracic spinedemonstrated normal content and distribution of motorneurons. Polymerase chain reaction for detection of poliovirus in extracted DNA from paraffin-embedded biopsy ofthoracic spine was negative.DiscussionThe anatomical dissection of an 82-year-old man revealed abilateral asymmetrical deficiency of the majority of thetrunk muscles. In addition, the typical deficiencies ofmuscles encountered in the lower extremities in poliomyeli-tis was observed. Some muscles of the trunk (trapezius andteres major) were almost completely deficient; others weremacroscopically replaced by connective tissue and fat in amore or less pronounced manner. Histological examinationof trunk muscles confirmed these findings, which werecompatible with the diagnosis of very advanced remoteneurogenic atrophy following poliomyelitis. Although rep-resentative sections of the spinal cord failed to reveal theantecedent poliomyelitis, as in other reports [6], there is littledoubt that the etiology of the muscle deformities of thetrunk, in addition to the muscle defects of the lowerextremities, must be attributed to prior poliomyelitis.Poliomyelitis was a common disease until the 1960swhen mass immunization with oral poliovirus vaccine wasbegun in developed countries. The last natural outbreak ofpoliomyelitis in the USA occurred in 1972. There are nowmillions of adults who suffered from a more or lessaggressive poliomyelitis infection in childhood and are leftFig. 2 Overview of the posterior trunk. a Superficial muscular layerof the trapezius, the teres major, and both latissimus dorsi muscles.The end of the indicator line for the latissimus dorsi simultaneouslyindicates the sites of histological examination (bilaterally mirror-inverted). b Deep layer showing the rhomboids and the serratusposterior inferior muscles. c Intrinsic muscles of the back, includingthe iliocostalis, longissimus, and spinalis musclesEur J Plast Surg (2009) 32:199–203 201with a postpolio syndrome. In this, there is limitation in therange and power of movements and pain in the affectedareas [9, 10]. At the moment, poliomyelitis has been largelyeradicated in industrialized countries, but since immuniza-tion programs have failed to reach the global population, ithas not been possible to eradicate the disease worldwide.On the other hand, poliomyelitis is still endemic in somedeveloping countries [11, 12], and any decrease in intensityof immunization could lead to large outbreaks of poliomy-elitis spread by travelers in this present world of modernglobal mobility.Although poliomyelitis is predominantly associated withmuscle defects in the lower extremities, theoretically, allmuscles may be affected. When muscles of the trunk areaffected only partially, surprisingly little disability mayresult, and the defects may pass mainly unnoticed by thepatient. When examining such patients, denervation byelectromyographic evidence is often far more extensivethan the subjects realize [13].In the case of muscles, which are visibly deficient, as inthe case of the pectoralis major muscle, females especiallyoften seek correction of the disfiguring appearance. Thisinvolves reconstruction of the asymmetric chests or themissing anterior axillary fold. The most successful recon-struction of such unilateral deformities of the pectoralismajor muscle has been the use of the ipsilateral pedicledlatissimus dorsi muscle. This is transferred to the anteriorchest wall to simulate the pectoralis major muscle and tocreate an anterior axillary fold [14]. When the latissimusdorsi muscle is deficient [15, 16], or the reconstruction withthe latissimus muscle is unsatisfactory, another favoriteoption has been the use of a transverse rectus abdominisflap. The ipsilateral rectus abdominis muscle may bedeficient and thus not available for reconstruction [17]. Insuch cases, the safe microvascular transfer of contralateralmuscles has been recommended; unfortunately, the contra-lateral muscles can be deficient, as has been sporadicallyreported [6] and our case clearly revealed.For the clinicians, knowledge of trunk muscle deficiencies,especially in bilateral cases, is of major importance. Thisknowledge will promote a search for further unapparentabnormalities, both unilateral and bilateral.This knowledge of unilateral muscular deficiencies, as inthe classical “Poland’s syndrome,” is important where therelevant ipsilateral muscles for reconstruction are absent,and a contralateral muscle has to be considered for a freetissue transfer. This knowledge is even more importantwhen bilateral muscular deficiencies are suspected, as inbilateral “Poland’s syndrome” or in cases with remotepoliomyelitis.This knowledge is mainly important in the era of advancedmicrosurgery; nearly all muscles can be used as donors.In cases of clinically unrecognized trunk muscle defi-ciencies, such as in patients with a history of poliomyelitis,to prevent an unfavorable surprise intraoperatively, at anyhint of muscle defects, the severity of the defect should beestablished and the surgery well planned before undertakingthe operative procedure. Although poliomyelitis is seldomencountered in its acute form currently in our industrializedcountries and the general knowledge of this infection fades,Fig. 3 Histological appearanceof the deficient muscles showingparts of the latissimus dorsimuscle on both sides. Thespecimens were taken from bothsides within the latissimus dorsimuscles as indicated by theindicator line in Fig. 2a. On theright side (a), the latissimus partis largely replaced by fat tissue,and only few muscle fibers areleft (arrows, H & E, ×100); onthe left side (b), the musclefibers are microscopicallypresent, though very small(H & E, ×50). Immunohisto-chemistry with diffuse evidenceof fast (c, ×50) and slow musclemyosin (d, ×50) in atrophicand normal muscle fibers(arrows)202 Eur J Plast Surg (2009) 32:199–203it should not be forgotten that many adults with a priorpoliomyelitis infection in childhood and with possiblemuscular trunk defects are still alive.Acknowledgment We thank Professor Kristian Borg from theKarolinska University in Stockholm for his helpful advice concerningthe histological assessment. Furthermore, we are grateful to PD Dr. C.T. Bock from the University in Tübingen for polio PCR analysis andto N. Wey from the University Zürich for photographic reproductions.References1. Poland A (1841) Deficiency of the pectoral muscles. Guys HospRep 6:191–1922. Jones H (1926) Congenital absence of the pectoral muscles. BrMed J 6:59–603. Sheehan D (1932) Bilateral absence of trapezius. J Anat 67:180–1814. Horan F, Bonafede R (1972) Bilateral absence of the trapezius andsternal head of the pectoralis major muscles. J Bone Joint Surg59:1335. Karnak I, Tanyel FC (2003) Reply to correspondence fromShipkov and Anastassov—“bilateral Poland anomaly: does itexist?”. Am J Med Genet A 117(3):310–3116. Mosconi T, Kamath S (2003) Bilateral asymmetric deficiency ofthe pectoralis major muscle. Clin Anat 16(4):346–3497. Chang CW, Huang SF (2001) Varied clinical patterns, physicalactivities, muscle enzymes, electromyographic and histologicfindings in patients with post-polio syndrome in Taiwan. SpinalCord 39(10):526–5318. Luciano CA, Sivakumar K, Spector SA et al (1996) Electrophys-iologic and histologic studies in clinically unaffected muscles ofpatients with prior paralytic poliomyelitis. Muscle Nerve 19(11):1413–14209. Bartels MN, Omura A (2005) Aging in polio. Phys Med RehabilClin N Am 16(1):197–21810. Nathanson N (1982) Eradication of poliomyelitis in the UnitedStates. Rev Infect Dis 4(5):940–95011. Sabin AB (1991) Perspectives on rapid elimination and ultimateglobal eradication of paralytic poliomyelitis caused by polio-viruses. Eur J Epidemiol 7(2):95–12012. Thompson KM, Tebbens RJ (2007) Eradication versus control forpoliomyelitis: an economic analysis. Lancet 369(9570):1363–137113. Hayward M, Seaton D (1979) Late sequelae of paralyticpoliomyelitis: a clinical and electromyographic study. J NeurolNeurosurg Psychiatry 42(2):117–12214. Hester TR Jr., Bostwick J 3rd (1982) Poland’s syndrome:correction with latissimus muscle transposition. Plast ReconstrSurg 69(2):226–23315. Cochran JH Jr., Pauly TJ, Edstrom LE et al (1981) Hypoplasia ofthe latissimus dorsi muscle complicating breast reconstruction inPoland’s syndrome. Ann Plast Surg 6(5):402–40416. Beer GM, Kompatscher P, Hergan K (1996) Poland’s syndromeand vascular malformations. Br J Plast Surg 49(7):482–48417. Drever JM, Zavala J (2002) Unilateral absence of the externaloblique muscle with hypoplasia of the rectus abdominis muscle ina patient with Poland syndrome. Plast Reconstr Surg 110(7):1802–1803Eur J Plast Surg (2009) 32:199–203 203

Multiple bilateral asymmetrical deficiency of trunk muscles

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Multiple bilateral asymmetrical deficiency of trunk muscles

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